ArticleLiterature Review

Looking in the Mouth for Crohn's Disease

Authors:
To read the full-text of this research, you can request a copy directly from the authors.

Abstract

It is widely acknowledged among gastroenterologists that the oral cavity may be involved in Crohn's disease (CD). However, the specific manifestations are poorly appreciated. Although oral aphthous ulceration is probably not diagnostically useful in patients with suspected CD, disease-specific manifestations do occur and are particularly common in children presenting with CD. These manifestations can be subtle, often are subclinical, yet commonly harbor diagnostically useful material (granulomas). Orofacial granulomatosis (OFG) is conventionally used to describe patients with overt oral disease without obvious involvement of the gastrointestinal tract. However, many patients with OFG have subclinical intestinal CD or will progress to develop overt intestinal CD with time. The management of severe oral disease is challenging and lacks a clear evidence base.

No full-text available

Request Full-text Paper PDF

To read the full-text of this research,
you can request a copy directly from the authors.

... Inflammation in CD most frequently affects the distal ileum and colon but may occur in any part of the gastrointestinal tract including the mouth (47). The term oral Crohn's disease (OCD) is used to describe patients with intestinal CD who exhibit involvement of the oral cavity with a wide variety of disease-specific oral lesions (48). Many oral lesions have been described in CD patients, including swelling of the lips, buccal mucosal swelling or "cobble-stoning, " mucogingivitis, deep linear ulceration, particularly along the buccal gutters, and mucosal tags. ...
... Many oral lesions have been described in CD patients, including swelling of the lips, buccal mucosal swelling or "cobble-stoning, " mucogingivitis, deep linear ulceration, particularly along the buccal gutters, and mucosal tags. Submental lymphadenopathy, perioral erythema with scaling, recurrent buccal abscesses, and angular cheilitis are often observed in patients with CD (43,48). The oral manifestations of IBD are diverse and based on their relationship with CD activity can be classified as specific (e.g., mucosal swelling) and non-specific oral lesions (e.g., angular cheilitis) (49). ...
... The oral manifestations of IBD are diverse and based on their relationship with CD activity can be classified as specific (e.g., mucosal swelling) and non-specific oral lesions (e.g., angular cheilitis) (49). Similar oral manifestations have been recorded in different patient cohorts throughout the world (47)(48)(49)(50)(51)(52). Although patients with CD can exhibit oral manifestations, pediatric CD has no specific clinical manifestations. ...
Article
Full-text available
The oral cavity is continuous with the gastrointestinal tract and in children, oral health may be closely linked with the overall health of the GI tract. In the case of pediatric Crohn's disease (CD), oral manifestations are an important clinical indicator of intestinal disease. Recent studies of the microbiome in IBD suggest that translocation of oral microbes to the gut may be a common feature of the microbial dysbiosis which is a signature of both CD and ulcerative colitis (UC). Murine studies suggest that translocation of oral bacteria and yeasts to the lower GI tract may trigger inflammation in susceptible hosts, providing a mechanistic link to the development of IBD. Conversely, some studies have shown that dysbiosis of the oral microbiome may occur, possibly as a result of inflammatory responses and could represent a useful source of biomarkers of GI health. This review summarizes our current knowledge of the oral microbiome in IBD and presents current hypotheses on the potential role of this community in the pathogenesis of these diseases.
... Oral EIM are usually asymptomatic [41] and resolve without specific oral treatment, but with severe clinical presentations, the treatment given to control the intestinal disease is highly efficient and thus is considered the first-line treatment [42]. However, not infrequently especially when symptomatic, they may need specific treatment [43] as will be discussed later. ...
... Treatment of the underlying disease is usually effective [6]; however, they can be treated with topical tacrolimus [54] and intra-lesional steroid injection with or without mandibular blockade [55]. In severe nonresponsive to topical therapy, treatment with immunosuppressive agents is required [43] (Fig. 1c). ...
... In the majority of patients with oral lesions related to IBD, treatment of the underlying intestinal activity is the foremost and the cornerstone for resolving the oral lesions [42], as these lesions will resolve over time in association with the treatment of gastrointestinal disease using anti-inflammatory-immunomodulatory and biological drugs whichever are indicated [43,49,94]. The treatment armamentarium includes topical and systemic steroids, 5-ASA compounds, immunosuppressive agents, biologic treatments and even antibiotics such as tetracycline [95,96]. ...
Article
Inflammatory bowel disease (IBD) is a chronic relapsing remitting autoimmune disease including Crohn's disease and ulcerative colitis. IBD is associated with various extra-intestinal manifestations including oral manifestation. To date, only limited studies addressing the characteristics of the oral manifestations are available. The aim of the present review is to report the oral manifestations and their characteristics in IBD. A Medline/PubMed and Embase databases search were conducted and all relevant studies were extracted and analyzed. Overall, the oral manifestations in IBD were mostly associated with Crohn's disease rather than Ulcerative colitis where their prevalence ranged from 8 to 50%. Specific lesions for Crohn's disease include mucosal tags, cobblestoning and deep linear ulcerations with vertical fissures, while for ulcerative colitis, pyostomatisis vegetans was more disease specific. Notably, most of the oral manifestations were unrelated to disease activity, however more data are needed to accurately assess this correlation. Oral manifestations among IBD patients are not uncommon as Crohn's disease account for most of them. More data are warranted to precisely characterize their prevalence and association to intestinal activity.
... The recurrent facial swelling, with or without intraoral manifestations, usually remain the single most common presentation at onset [Al Johani, 2009;Scully & Eveson, 1991;Gagoh et al., 1999;Eveson, 1996;Wiesen, et al., 2007]. The differential diagnosis is usually challenging as OFG may precede gastrointestinal disease, such as CD, and in addiction may remain the only obvious focus of the disease [Al Johani 2009;Wiesen et al., 2007;Halme et al., 1993;Field & Allan 2001;Rowland et al., 2010].Traditionally, CD is described as a disorder affecting 'the mouth to the anus'; oral CD or isolated CD of the upper gastrointestinal tract remain a relatively uncommon findings. Different oral manifestations in patients affected by CD have been reported; many oral lesions are somewhat non specific, as the differential diagnosis includes several forms of nutritional glossitis [Field & Allan, 2001;Reamy et al., 2010]. ...
... A similar question exists for oral lesions associated to CD, as patients may suffer from intestinal disease, also with detectable endoscopic and histologic abnormalities, but without intestinal symptoms [Al Johani, 2009;Sanderson et al., 2005;Scully et al., 1982;Eckel et al., 2017;Harbord et al., 2016]. The prevalence of oral manifestations of CD is extremely variable, ranging from 10% to 80% [Scully et al., 1982;Rowland, Fleming & Bourke 2010;. Clinical signs can be specific, such as diffuse lip and buccal swelling, tags, cobblestones and notspecific, such as aphthous ulcers, pyostomatitis vegetans, and gingivitis [Triantafillidis, 2008;Al Johani, 2009;Gagoh et al., 1999;Wiesen et al., 2007;Field & Allan, 2001]. ...
... The main problem which can delay the diagnosis is the fact that oral signs of CD might precede or coincide with intestinal inflammation [Scully & Eveson, 1991;Sanderson et al., 2005;. For such reason and in addition for the increasing incidence in the paediatric population, clinicians should always consider CD in the differential diagnosis when one or more oral manifestations are detectable [Al Johani, 2009;Wiesen et al. 2007;Rowland et al., 2010;Eckel et al., 2017;Harbord et al., 2016;. In fact, according to the most recent consensus papers of the European Crohn's and Colitis Organisation "The First European Evidence-based Consensus on Extra-intestinal Manifestations in Inflammatory Bowel Disease" published in 2016, and according to the following suggestions proposed by the "3rd European Evidence-based Consensus on the Diagnosis and Management of Crohn's Disease 2016: Part 1: Diagnosis and Medical Management" published in 2017, a single gold standard for the diagnosis of CD is not available for a precise diagnosis and the correct management of CD [Harbord et al., 2016;Gomollón et al., 2017;. ...
Article
Full-text available
Aim: Oro-facial granulomatosis is a descriptive term commonly encompassing a variety of conditions that exhibit similar clinical and microscopic features. It is generally used to describe persistent enlargement of the soft tissues of the oral and maxillofacial region. Materials and methods: We report on the salient clinical features of 8 cases of Crohn's disease in paediatric patients (age range from 9 to 13 years old), with oral lesions as first clinical manifestations. Results: The clinical presentation of oro-facial granulomatosis is highly variable but usually recurrent facial swelling, mainly in the lips with or without intraoral manifestations, is the single most common clinical sign at onset. The association with systemic conditions such as sarcoidosis and Crohn's disease has been widely reported in literature. In paediatric age, oro-facial granulomatosis may frequently represent an extra-intestinal manifestation of Crohn's disease and oral lesions can be the first sign of an unknown intestinal disease. The diagnosis in paediatric patients is challenging as oro-facial granulomatosis may precede Crohn's disease by several years, frequently remaining the only evident active focus of the disease. Conclusion: The detection of specific oral manifestations often preceded by painless gingival enlargement (diffuse lip and buccal mucosal swelling, oral cobblestoning, buccal sulcus ulceration and mucosal tags) and/or unspecific or ancillary ones (cheilitis, scaly perioral erythematous rashes and frank intraoral abscess formation, labial and tongue fissuring, glossitis and aphthous stomatitis) is mandatory for the early diagnosis of intestinal Crohn's disease.
... The reported prevalence of oral manifestations in CD varies widely and ranges from 0.5% to 80%. [1][2][3] These manifestations may coincide with the intestinal symptoms of CD, or precede them, but there are very few systematic studies at different stages of the disease. Furthermore, we are not aware of reports on the otorhinolaryngological health of patients with CD. ...
... In the Laranjeira et al 21 study, patients in the clinically active phase of inflammatory bowel disease were the most affected, but this may not have been the case in children. 1 Our study of patients with pediatric onset CD who had reached adulthood showed no correlation between the oral findings and the subjective evaluation of the severity of gut symptoms in CD or with fecal calprotectin levels, a surrogate marker for intestinal inflammation. In our previous study of 29 patients with pediatric onset OFG, including 72% who had CD, there was no association between fecal calprotectin and orofacial findings. ...
... Specific lesions result from the same disease process as CD in the gut, namely granulomas that can be identified by histology. These specific lesions include swelling of the lips, cheeks and gingiva, cobblestoning of the mucosa, deep linear ulcers and mucosal tags, 1,20 which are also typical lesions in OFG. Nonspecific lesions are reactive lesions without granulomas. ...
Article
Full-text available
Background: Up to 50% of pediatric patients with Crohn's disease (CD) report oral manifestations, but less is known about their oral health when they become adults. Goals: Our aim was to provide detailed descriptions of the presence of oral and otorhinolaryngological manifestations in patients with pediatric onset CD once they reached adulthood, to look for predisposing factors and to compare the findings to matched controls. Study: Adult patients diagnosed with CD in childhood at the Children's Hospital, University of Helsinki, Finland, after 2000 were invited for a follow-up appointment in 2016 and 24 were examined by a dentist and otorhinolaryngologist. They were compared with 22 matched controls from the Population Register Centre. The participants completed questionnaires about their general health, any special diets, and their health-related quality of life. Their nutrition was evaluated from food records. Results: Patients with CD had minor oral manifestations at a median of 9 years after their childhood diagnosis and the most common was angular cheilitis, which affected 6 patients and 1 control, but was not statistically significant (P=0.0984). CD with perianal abscessing disease correlated to orofacial findings (P=0.0312). Most of the patients had normal otorhinolaryngological findings. Subjects with oral lesions had lower mean health-related quality of life scores than subjects without oral findings and the differences were clinically but not statistically significant. Oral manifestations were not associated with differences in energy intake. Conclusions: Oral manifestations in adult patients with pediatric onset CD were mild and were not associated with otorhinolaryngological pathology.
... Oral involvement in patients with intestinal CD is typically termed oral Crohn's disease (OCD). [42] Although well-described and classified, they are often unrecognized or overlooked by gastroenterologists [4]. The reported prevalence of OCD is highly variable, depending on the ethnicity and age of the population studied and involvement (or not) of an oral medicine or dentistry specialist. ...
... dedicated specialist multidisciplinary oral and gastrointestinal diseases clinic. [42] The authors reported a prevalence of OCD in up to 41.7% of children, at a mean age of 12.4 months. In adults, the prevalence of OCD appears to be lower at approximately 20%, but has been reported as high as 50%. ...
... In adults, the prevalence of OCD appears to be lower at approximately 20%, but has been reported as high as 50%. [7,42,44] Comparatively, OCD patients have a lower age of presentation of than the average CD population [45]. There is also a greater degree of perianal involvement in patients with OCD than those with intestinal CD. ...
Article
Crohn's disease is a heterogeneous, inflammatory condition that can affect any location of the gastrointestinal tract. Proximal gastrointestinal involvement occurs in 0.5-16% of patients, and it is usually diagnosed after recognition of intestinal disease. Symptoms are often mild and non-specific, however upper gastrointestinal disease predicts a more severe Crohn's phenotype with a greater frequency of complications such as obstruction and perforation. Gastroscopy and biopsy is the most sensitive diagnostic investigation. There is a paucity of data examining the treatment of this condition. Management principles are similar to those for intestinal disease, commencing with topical therapy where appropriate, progressing to systemic therapy such as glucocorticoids, 5-aminosalicylic acid, immunomodulators and biologics. Acid suppression therapy has symptomatic but no anti-inflammatory benefit for gastroduodenal and esophageal involvement. Surgical intervention with bypass, strictureplasty or, less frequently, endoscopic balloon dilation, may be required for complications or failed medical therapy.
... Inflammation in CD most frequently affects the distal ileum and colon but may occur in any part of the gastrointestinal tract including the mouth 10 . Oral Crohn"s disease (OCD) is a phenomenon that can occur in patients with intestinal CD and is characterised by a wide variety of disease-specific oral lesions 11 . The most frequently described oral lesions in CD patients include swelling of the lips, buccal mucosal swelling or "cobble-stoning", mucogingivitis, deep linear ulceration along the buccal gutters and mucosal tags 11,12 . ...
... Oral Crohn"s disease (OCD) is a phenomenon that can occur in patients with intestinal CD and is characterised by a wide variety of disease-specific oral lesions 11 . The most frequently described oral lesions in CD patients include swelling of the lips, buccal mucosal swelling or "cobble-stoning", mucogingivitis, deep linear ulceration along the buccal gutters and mucosal tags 11,12 . The oral manifestations of IBD are diverse and based on their relationship with CD activity can be classified as specific (e.g. ...
Article
Full-text available
Background: There is a limited literature describing the oral microbiome and its diagnostic potential in paediatric inflammatory bowel disease (IBD). Methods: We examined the dorsum tongue microbiome by V1-V2 sequencing in a cohort of 156 treatment naïve children diagnosed with IBD compared to 102 healthy control children. Microbiome changes over time following treatment were examined in a subset of patients and associations between IBD diagnosis and dysbiosis were explored. Results: Analysis of community structure of the microbiome in tongue samples revealed that IBD samples significantly diverged from healthy control samples (PERMANOVA P=0.0009) and exhibited a reduced abundance of Clostridia in addition to several major oral genera (Veillonella, Prevotella, Fusobacterium species) with an increased abundance of streptococci. This dysbiosis was more marked in patients with severe disease. Higher levels of the potential pathobionts Klebsiella and Pseudomonas spp. were also associated with IBD. In terms of predicted functions, the IBD oral microbiome was potentially more acidogenic and exhibited reduced capacity for B vitamin biosynthesis. We used a machine learning approach to develop a predictive model of IBD which exhibited a mean-prediction AUC: 0.762. Finally, we examined a subset of 53 patients following 12 months of therapy and could show resolution of oral dysbiosis demonstrated by a shift towards a healthy community structure and a significant reduction in oral dysbiosis. Conclusion: Oral dysbiosis found in children with IBD is disease severity related and resolves over time following successful IBD treatment.
... The most common mucosal findings related to celiac disease are mucosal ulcers (28). Around 0.5 -32 % of patients with Crohn´s disease get oral manifestations during the disease process (37). Oral symptoms of Crohn´s disease are similar to those with orofacial granulomatosis, including lip swelling, cobblestone lesions (Fig.7), mucosal ulcers with indurated borders, and gingival swelling and erythema. ...
... Patients with multiple recurrent oral ulcers, gingival swellings, and erythema and/or mucosal cobblestone lesions should be carefully examined to find out possible systemic disease behind oral lesions. Orofacial granulomatosis is confirmed by a tissue biopsy (37). Histologically, granuloma formation with lymphocytes and epithelioid histiocytes with or without multinucleated giant cells are seen. ...
... In all, 0.5%-80% of adult patients with CD manifest oral pathology. 156,157 In children, 42% of new diagnoses of CD had oral manifestations. 158 Docktor et al. ...
... 50 The loss of Fusobacteria and Firmicutes in children with CD were resonated in studies examining the intestinal microbiome. 156,158,159 Docktor et al. 50 commented that with the prevalence of oral pathology in CD and the ease of oral mucosal sampling, further study could explore the potential of using the oral microbiome as a diagnostic and prognostic tool for pediatric IBD. ...
Article
Full-text available
The human microbiome functions as an intricate and coordinated microbial network, residing throughout the mucosal surfaces of the skin, oral cavity, gastrointestinal tract, respiratory tract, and reproductive system. The oral microbiome encompasses a highly diverse microbiota, consisting of over 700 microorganisms, including bacteria, fungi, and viruses. As our understanding of the relationship between the oral microbiome and human health has evolved, we have identified a diverse array of oral and systemic diseases associated with this microbial community, including but not limited to caries, periodontal diseases, oral cancer, colorectal cancer, pancreatic cancer, and inflammatory bowel syndrome. The potential predictive relationship between the oral microbiota and these human diseases suggests that the oral cavity is an ideal site for disease diagnosis and development of rapid point-of-care tests. The oral cavity is easily accessible with a non-invasive collection of biological samples. We can envision a future where early life salivary diagnostic tools will be used to predict and prevent future disease via analyzing and shaping the infant’s oral microbiome. In this review, we present evidence for the establishment of the oral microbiome during early childhood, the capability of using childhood oral microbiome to predict future oral and systemic diseases, and the limitations of the current evidence.
... Oral manifestations have been reported to occur in up to 80% of Crohn's disease patients, in particular, children. 30,31 The symptoms are not in any form of tonsillitis but, e.g., as mucogingivitis, mucosal tags, labial swelling, deep ulceration, or cobblestoning. 30,31 As argued in the introduction, it is unlikely that severe tonsillitis or tonsillectomy itself may trigger inflammatory bowel disease, because neither lymphoid tissues nor bacterial colonies in the oropharynx and intestine are directly connected. ...
... 30,31 The symptoms are not in any form of tonsillitis but, e.g., as mucogingivitis, mucosal tags, labial swelling, deep ulceration, or cobblestoning. 30,31 As argued in the introduction, it is unlikely that severe tonsillitis or tonsillectomy itself may trigger inflammatory bowel disease, because neither lymphoid tissues nor bacterial colonies in the oropharynx and intestine are directly connected. In addition, oral symptoms of Crohn's disease rarely precede systemic symptoms and often co-occur with perianal symptoms. ...
Article
Background: The possible etiologic link between tonsillectomy and inflammatory bowel diseases remains unclear. To investigate the hereditary component, we assessed the risk of inflammatory bowel disease after own tonsillectomy as well as after tonsillectomy among family members. Methods: A nationwide Danish cohort of 7,045,288 individuals was established and linked to comprehensive national registers with data on kinship, tonsillectomy surgery, and diagnosis of inflammatory bowel disease from all health sectors. We used Poisson regression models to estimate hospital contact rate ratios (RR) for Crohn's disease and ulcerative colitis, with 95% confidence intervals (CI), between individuals with or without tonsillectomy, as well as between individuals with or without tonsillectomized relatives. Results: During 189 million person-years of follow-up between 1977 and 2014, 276,673 individuals were tonsillectomized, 22,015 developed Crohn's disease, and 49,550 developed ulcerative colitis. Rates of inflammatory bowel disease were elevated up to 20 years after own tonsillectomy (Crohn's disease: RR 1.52 (95% CI, 1.43-1.61); ulcerative colitis: RR 1.24 (95% CI, 1.18-1.29)). RRs for Crohn's disease was 1.22 (95% CI, 1.17-1.27) after 1 degree relatives' tonsillectomy, 1.14 (95% CI, 1.08-1.19) after 2 degree relatives' tonsillectomy, and 1.08 (95% CI, 1.01-1.15 after 3 degree relatives' tonsillectomy. Corresponding RRs for ulcerative colitis were 1.10 (95% CI, 1.07-1.13), 1.05 (95% CI, 1.01-1.08), and 1.03 (95% CI, 0.98-1.09). Conclusions: Even individuals with tonsillectomized family members were at increased risk of inflammatory bowel disease. These findings call into question a direct influence of tonsillectomy on gastrointestinal inflammation and point instead towards shared hereditary or environmental factors.
... Anti-TNF agents have been reported to improve the condition. While control of intestinal disease and local treatments may be sufficient for managing nonspecific oral lesions, current treatment may need to be escalated for specific oral lesions [27][28][29]. Therapeutic options include topical and systemic steroids, immunosuppressive agents, and biologic treatments [21,30]. Philips et al. [13] reported that in a multicenter study of twentyeight patients, orofacial granulomatosis improved in twenty-three patients, with the use of anti-TNFs in nine patients, vedolizumab in one, ustekinumab in one, and thalidomide in two. ...
Article
Full-text available
Background: Oral manifestations of Crohn’s disease (CD) include non-specific lesions and specific lesions directly related to intestinal inflammation. Oral lesions that can be overlooked in CD are sometimes challenging to treat. Methods: In this retrospective single-center study, patients with CD aged over 18 years who complied with follow-up and treatment were included. Clinical definitions of specific oral lesions included pyostomatitis vegetans, glossitis with fissuring, lip swelling with fissuring, cobblestoning, and orofacial granulomatosis. Experienced dentists confirmed the specific lesions in each case. Three groups of patients were identified: those without oral lesions, those with non-specific oral lesions, and those with specific oral lesions. The groups were compared based on demographics, disease extent and behavior (based on the Montreal classification), extraintestinal involvement, biologic and steroid treatment, and the requirement of resective surgery. Results: A total of 96 patients (14.2%) with oral lesions were found among the 676 patients with CD (59.7% male, median age 38 years) who were followed for 6.83 years (IQR 0.5–29.87 years). Eight patients (1.2%, 9 lesions) had specific oral lesions, while eighty-eight patients (13%) had non-specific lesions. Orofacial granulomatosis (n = 3), cobblestoning (n = 2), glossitis with fissuring (n = 2), and lip swelling with fissuring (n = 2) were among the specific lesions. The majority of patients (75%) with specific lesions were male, and their median age was 46.5 years (range: 23–68 years). Disease localization was commonly ileocolonic (50%), and perianal disease was present in 25% of patients. Three patients were active smokers. Extraintestinal manifestations were peripheral arthritis/arthralgia (n = 7) and sacroiliitis (n = 1). All specific lesions were associated with moderate-to-severe disease. Five patients improved with biologic therapy, and two patients with immunomodulatory therapy. Conclusions: Specific oral lesions in CD were associated with active disease and improved with immunomodulators or biologic therapy. Close cooperation between gastroenterologists and dentists is essential for early diagnosis and optimal management of CD.
... 15 The oral lesions secondary to IBD will often resolve after the primary gastrointestinal disease is treated with medications which may include drugs targeting inflammation, heightened immune system, as well as some biological drugs. [16][17][18] ...
Article
Full-text available
Extra-intestinal signs of inflammatory bowel disease (IBD) can reach a site as far as oral cavity. The oral cavity manifestations of IBDs are many and more likely missed by the general practitioners and gastro- enterologists.
... These oral manifestations may be either asymptomatic or symptomatic with pain and impairment of oral function. 4,13 Caries and periodontitis are also common oral diseases in CD. 3 Over 50 systemic diseases or conditions are associated with periodontal diseases, including inflammatory bowel diseases. 14 In a meta-analysis by Nijakowski et al, the risk of periodontal disease in IBD patients was almost two and a half times more than that of controls. ...
Article
Full-text available
Oral mucosal lesions may persist years before symptoms or diagnosis of inflammatory bowel disease (IBD) and subsequent primary sclerosing cholangitis (PSC). Since a dental practitioner may be the first clinician to suspect IBD with extraintestinal manifestations (EIMs), early referral, and close collaboration with a gastroenterologist are recommended. image
... Топические ГКС, пероральные антисептики не имеют доказанной эффективности. В единичных случаях описаны ксеростомия, синдром Шегрена, халитоз, расширение мелких слюнных желез, изменение цвета слизистой оболочки ротовой полости, красный плоский лишай, неспецифический гингивит [39]. ...
... These oral manifestations may be either asymptomatic or symptomatic with pain and impairment of oral function. 4,13 Caries and periodontitis are also common oral diseases in CD. 3 Over 50 systemic diseases or conditions are associated with periodontal diseases, including inflammatory bowel diseases. 14 In a meta-analysis by Nijakowski et al, the risk of periodontal disease in IBD patients was almost two and a half times more than that of controls. ...
Preprint
Full-text available
Oral mucosal lesions may persist years before diagnosis of inflammatory bowel disease (IBD) and subsequent primary sclerosing cholangitis (PSC). It may be that a dental practitioner may be the first clinician to suspect IBD. Suspect IBD; early referral and close collaboration with a gastroenterologist are recommended.
... Selv om fle re stu di er an ty der at OFG og CD er to for skjel li ge til stan der (5,6,9,10), vi ser and re stu di er at så man ge som 40-50 % av unge pa si en ter med OFG en ten får stilt en CD dia gno se på sam me tids punkt som OFGdia gno sen el ler at de ut vik ler CD på et se ne re tids punkt (11)(12)(13). Et ter som både OFG og CD ofte blir opp da get i ung al der, kan OFG vaere et tid lig tegn på CD el ler også en un der grup pe av CD (11,13,14). Der for er det vik tig å vaere klar over at de ora le ma ni fes ta sjo ner ved OFG kan vaere et tid lig tegn på CD. ...
Article
Full-text available
Hovedbudskap Orofacial granulomatose kan være forløper eller første tegn på Crohns sykdom Klinikere må kjenne til ulike karakteristika og symptomer ved disse tilstandene. De kan variere fra pasient til pasient og ligne mer banale oralmedisinske forandringer Tannleger og tannpleiere har en viktig rolle ved å sikre tidlig diagnostikk av sykdommer som initialt kan manifestere seg i munnhulen
... This general inflammatory state associated with the IBD condition could affect the oral mucosa and lead to an oral dysbiosis that could contribute to the worsening of the inflammatory state and play a crucial role in the oral manifestations of these patients [5]. However, only a few studies have focused on analyzing changes in the oral microbiome of IBD patientsmost of them performed in the CD phenotype [9][10][11][12][13][14][15]. Regarding UC, most of the data come from pediatric populations or animal models [2,3,[16][17][18], and only one study has focused on the analysis of the oral microbiome of a specific UC cohort, performed in a murine model of colitis [16]. ...
Article
Full-text available
Ulcerative colitis (UC) is a recurrent pathology of complex etiology that has been occasionally associated with oral lesions, but the overall composition of the oral microbiome in UC patients and its role in the pathogenesis of the disease are still poorly understood. In this study, the oral microbiome of UC patients and healthy individuals was compared to ascertain the possible changes in the oral microbial communities associated with UC. For this, the salivary microbiota of 10 patients diagnosed with an active phase of UC and 11 healthy controls was analyzed by 16S rRNA gene sequencing (trial ref. ISRCTN39987). Metataxonomic analysis revealed a decrease in the alpha diversity and an imbalance in the relative proportions of some key members of the oral core microbiome in UC patients. Additionally, Staphylococcus members and four differential species or phylotypes were only present in UC patients, not being detected in healthy subjects. This study provides a global snapshot of the existence of oral dysbiosis associated with UC, and the possible presence of potential oral biomarkers.
... Oral manifestations may sometimes precede intestinal disease 6 and approximately 30% of the patients continue to manifest oral lesions despite control of their intestinal disease activity. 7,8 Previous studies have shown that dental professionals were knowledgeable about oral-systemic health associations, but had mixed feelings about translating this information into the dental practice. 9,10 On the other hand, recent surveys among general practitioners concluded that their knowledge about the relation between periodontal diseases and systemic disorders needed to be improved. ...
Article
Full-text available
Background Gastrointestinal diseases can have oral manifestations. The aim of this study was to investigate the knowledge of gastroenterologists and dentists about gastrointestinal diseases with oral manifestations and to assess the frequency, extent and content of communication between gastroenterologists and oral healthcare professionals. Methods Separate questionnaires were developed and sent to all 523 gastroenterologists and a random selection of 500 dentists in the Netherlands. Both questionnaires contained questions about demographic characteristics of the participants, 10 statements about gastrointestinal diseases with possible oral manifestations and questions about the communication between gastroenterologists and oral healthcare professionals. Additionally, the questionnaire for gastroenterologists contained 9 statements about general dentistry and the questionnaire for dentist had 9 questions about gastrointestinal diseases. Results Gastroenterologists answered 47.6% ± 31.9% of the questions correct about gastrointestinal diseases with possible oral manifestations and 57.5% ± 27.9% of the questions correct about general dentistry. Dentists answered 26.6% ± 20.5% of the questions correct about possible oral manifestations of gastrointestinal diseases and 50.3% ± 18.7% of the questions correct about gastrointestinal diseases. Gastroenterologists and dentists valued interdisciplinary consultation as very useful with scores of 4.07 ± 0.70 and 4.67 ± 0.49 on a 5-point Likert scale, respectively, but the frequency of consultation was considered insufficiently with a mean score of 2.88 ± 1.01 and 2.24 ± 1.05 on a 5-point Likert scale, respectively. Conclusions This study suggests that the knowledge of gastroenterologists and dentists about gastrointestinal diseases with oral manifestations could be improved. Interdisciplinary consultation was considered valuable for the optimal treatment of their patients but was assessed as insufficient.
... O diagnóstico pode ser muito difícil, uma vez que a CrD orofacial é indistinguível da granulomatose orofacial, que pode ser detectada em várias condições (como sarcoidose, queilite granulomatosa de Miescher, síndrome de Melkersson-Rosenthal, granuloma de corpo estranho, rosácea e várias doenças infecciosas granulomatosas) [Wiesenfeld , 1985;Bogenrieder, 2003]. Até 40-50% dos pacientes jovens com granulomatose orofacial podem desenvolver CrD e ela pode ser notada mesmo anos após o primeiro aparecimento dos sintomas orais [Rowland, 2010]. ...
Article
Full-text available
A cavidade oral faz parte do sistema gastrointestinal e, como tal, a presença de alterações nesta região pode ser o primeiro sinal de doenças sistémicas e gastrointestinais. Como essas alterações são muito comuns, principalmente em crianças, é importante que o dentista saiba quando elas são expressão de um quadro gastrointestinal. O objetivo desta revisão é fornecer ao dentista dados úteis para o diagnóstico, tratamento e manejo das condições mais comuns, como doença de Crohn, colite ulcerativa, doença do refluxo gastroesofágico e doença celíaca. Uma das alterações orais mais comuns é a erosão dentária, com perda de esmalte e aumento do risco de cárie dentária, que tem sido relatada em crianças e adolescentes com refluxo gastroesofágico. Hipoplasia do esmalte dentário e úlceras aftosas são mais comuns em crianças com doença celíaca do que na população em geral. Outra alteração oral muito comum é a gengivite, que afeta 9-95% das crianças na Europa e na América do Norte e mais de 60% dos adolescentes. Pioestomatite vegetante pode ser um sinal de colite ulcerosa e doença de Crohn, esta última também foi relacionada a edema difuso da mucosa, mucosa de paralelepípedo, muco-gengivite localizada, ulceração linear profunda, marcas de tecido fibroso, pólipos, nódulos e úlceras aftosas. O reconhecimento imediato de doenças sistêmicas e gastrointestinais por meio de um exame cuidadoso da cavidade oral pode ser o primeiro passo para novas investigações que podem levar a um diagnóstico precoce e tratamento oportuno.
... In regard oral manifestation, 57% did not have any oral manifestation, 17% had angular cheilitis, 16% had aphthous ulcer, and 11% had diffuse labial swelling. This finding is in good agreement with those reported by the previously conducted studies (33,34). The observed oral symptoms were halitosis, dry mouth, and acid taste. ...
... They can appear before the clinically apparent onset of the abdominal disease (5-10% of affected patients), be present during the disease process, or persistent even after the abdominal disease is resolved [120][121][122]. The prevalence rate of oral lesions in IBD is estimated between 20% and 50% in most publications [121,123,124]. Recurrent deep granulomatous aphthous-like ulcers are the most common oral manifestation of Crohn's Disease [120,125]. ...
Article
Full-text available
Most prevalent food allergies during early childhood are caused by foods with a high allergenic protein content, such as milk, egg, nuts, or fish. In older subjects, some respiratory allergies progressively lead to food-induced allergic reactions, which can be severe, such as urticaria or asthma. Oral mucosa remodeling has been recently proven to be a feature of severe allergic phenotypes and autoimmune diseases. This remodeling process includes epithelial barrier disruption and the release of inflammatory signals. Although little is known about the immune processes taking place in the oral mucosa, there are a few reports describing the oral mucosa-associated immune system. In this review, we will provide an overview of the recent knowledge about the role of the oral mucosa in food-induced allergic reactions, as well as in severe respiratory allergies or food-induced autoimmune diseases, such as celiac disease.
... Crohn's disease (CD) is a chronic, systemic, immune-mediated inflammatory bowel disease that frequently exhibit extra-intestinal manifestations, including oro-facial signs in both adults and children. Clinical oral signs of CD are usually distinguished in specific (diffuse lip and buccal swelling, tags, cobblestones) and not-specific (aphthous ulcers, pyostomatitis vegetans, and gingivitis) (Figure 13a,b) [116,117], In patient showing one or more of the lesions, the biopsy is mandatory to achieve final diagnosis and for the differential diagnosis mostly with foreign-body reactions, sarcoidosis, mycobacterial infection and fungal sepsis. Overall, oral lesions may be variably identified in up to 60% of patients, while in 5-10% of cases they represent the first manifestation of a still unknown CD [101,102,118]. ...
Article
Full-text available
Many systemic (infective, genetic, autoimmune, neoplastic) diseases may involve the oral cavity and, more generally, the soft and hard tissues of the head and neck as primary or secondary localization. Primary onset in the oral cavity of both pediatric and adult diseases usually represents a true challenge for clinicians; their precocious detection is often difficult and requires a wide knowledge but surely results in the early diagnosis and therapy onset with an overall better prognosis and clinical outcomes. In the current paper, as for the topic of the current Special Issue, the authors present an overview on the most frequent clinical manifestations at the oral and maxillo-facial district of systemic disease.
... Though UC is generally limited to the colon, CD can occur anywhere in the gastrointestinal tract, and can include involvement of the oral cavity, with approximately 40% of a cohort of Irish paediatric CD patients (over a 3 year period) having oral involvement [81]. A significant decrease in diversity was seen in tongue samples of paediatric CD patients when compared to healthy and though non-significant, a decrease was also seen in the buccal samples of these paediatric CD patients [82]. ...
Article
Full-text available
Paediatric inflammatory bowel disease (IBD) is a chronic inflammatory disorder of the digestive tract, comprising of Crohn’s disease (CD), ulcerative colitis (UC), and, where classification is undetermined, inflammatory bowel disease unclassified (IBDU). Paediatric IBD incidence is increasing globally, with prevalence highest in the developed world. Though no specific causative agent has been identified for paediatric IBD, it is believed that a number of factors may contribute to the development of the disease, including genetics and the environment. Another potential component in the development of IBD is the microbiota in the digestive tract, particularly the gut. While the exact role that the microbiome plays in IBD is unclear, many studies acknowledge the complex relationship between the gut bacteria and pathogenesis of IBD. In this review, we look at the increasing number of studies investigating the role the microbiome and other biomes play in paediatric patients with IBD, particularly changes associated with IBD, varying disease states, and therapeutics. The paediatric IBD microbiome is significantly different to that of healthy children, with decreased diversity and differences in bacterial composition (such as a decrease in Firmicutes). Changes in the microbiome relating to various treatments of IBD and disease severity have also been observed in multiple studies. Changes in diversity and composition may also extend to other biomes in paediatric IBD, such as the virome and the mycobiome. Research into biome differences in IBD paediatric patients may help progress our understanding of the aetiology of the disease.
... In regard oral manifestation, 57% did not have any oral manifestation, 17% had angular cheilitis, 16% had aphthous ulcer, and 11% had diffuse labial swelling. This finding is in good agreement with those reported by the previously conducted studies (33,34). The observed oral symptoms were halitosis, dry mouth, and acid taste. ...
Article
The aim of this study is to evaluate the association between IBD and oral symptom and mucosal lesions in patients with Crohn’s disease and ulcerative colitis Methods: This is a cross-sectional study that has been done in (Kurdistan center for Gastroenterology and hepatology) of Teaching Hospital in Sulaymaniyah-Iraq, which included 101 patients previously diagnosed with Inflammatory Bowel Disease who were interviewed regarding manifestations of inflammatory bowel disease especially oral manifestations. Required data were collected through a specially designed questionnaire, Results: The patients’ mean age was 45.74±12.58 years. Patients with ulcerative colitis and Crohn’s disease were not significantly different in terms of age, sex, smoking, and drinking alcohol (p>0.05), and it was seen that age groups 41-50 and 51-60, males, smokers, and drinkers were more affected by these two diseases. They were not significantly different in terms of chief complaint, drug history, oral hygiene, and disease duration (p>0.05). Most of the patients (78 out of 101) had fair or poor oral hygiene. The two groups of the patients were not significantly different in terms of oral manifestations and symptoms (p>0.05). The most common oral manifestations were found to be respectively angular cheilitis, aphthous ulcer, and diffuse lip swelling in both diseases. The most common symptoms were respectively dry mouth and halitosis. Conclusion: This study revealed that dentists and oral and maxillofacial Medicine could involve in a diagnosis of IBD, by at least in the referral process, because patients in clinical setting may give a history of orofacial complaints giving a hint on possible systemic background of IBD
... The most acceptable theory of CD is that it brings about when an inappropriate mucosal inflammation response to an intestinal bacteria in a genetically subjected host. 3,4 The study of Falodia et al reported that female (60%) were more affected by CD as compared to male patients (40%) and the oral manifestations were more common in female (22) as compared to male patients (11).1Similar to this in our study found that female patients (66.6%) were mostly affected by CD than male patients (33.3%) and oral manifestations were commonly seen in female (36) than male (18) patients. ...
... For example, miR-2909 in urinary exosomes of patients with prostate cancer has been recognized as a noninvasive and specific biomarker for prostate cancer (64). Reports also highlight the involvement of the oral cavity in IBD, such as the co-occurrence of oral lesions with IBD-associated intestinal damage (49 example, salivary exosomal Proteasome 20S Subunit Alpha 7 (PSMA7) has been reported as the most promising IBD biomarker, which exhibits an increased expression in the diseased state (7,75). In addition, salivary exosomal PMSA7 has higher expression in UC than in CD patients and therefore could be used in differential diagnosis between the two forms of IBD. ...
Article
Full-text available
Exosomes represent secretory membranous vesicles used for the information exchange between cells and organ-to-organ communication. Exosome crosstalk mechanisms are involved in the regulation of several IBD-associated pathophysiologic intestinal processes such as barrier function, immune responses and intestinal flora. Functional biomolecules, mainly non-coding RNAs (ncRNAs), are believed to be transmitted between the mammalian cells via exosomes which likely play important roles in cell-to-cell communication, both locally and systemically. MicroRNAs (miRNAs) encapsulated in exosomes have generated substantial interest because of their critical roles in multiple pathophysiological processes. In addition, exosomal miRNAs are implicated in the gut health. MiRNAs are selectively and actively loaded into the exosomes and then transferred to the target recipient cell where they manipulate cell function through post-transcriptional silencing of target genes. Intriguingly, miRNA profile of exosomes differ from their cellular counterparts suggesting an active sorting and packaging mechanism of exosomal miRNAs. Even more exciting is the involvement of posttranscriptional modifications in the specific loading of miRNAs into exosomes, but the underlying mechanisms of how these modifications direct ncRNA sorting have not been established. This review gives a brief overview of the status of exosomes and exosomal miRNAs in IBD and also discusses potential mechanisms of exosomal miRNA sorting and delivering.
... Oral mucosal inflammation is well-studied in patients with IBD with a reported prevalence of 0.5-80% in CD patients (Rowland et al., 2010). Symptoms could vary from mild and non-specific inflammation such as minor aphthous lesions, mucogingivitis, and angular cheilitis to more specific findings, such as cobblestoning, deep linear ulcerations, and more severe orofacial granulomatosis (OFG) (Katz et al., 2003;Ojha et al., 2007). ...
Article
Full-text available
Crohn's disease is a chronic disorder that typically affects the gastrointestinal tract. The increased incidence in the recent years, especially in Asian countries, prompts for performing studies and gain newer insights into the etiology and pathogenesis of the disease. Among other causative factors, gut microbiome and its cross-talk with the salivary microbiome is a known factor that has a plausible role in the pathogenesis of Crohn's disease. The gut microbiome has been extensively studied, however, the salivary microbiome and its dynamics during different phases of this disease remain understudied. In this study, we obtained saliva samples from the patients during active and remission phases of the disease and compared them with control samples and highlighted the differences in taxonomic as well as predicted functional pathways among them. Our results indicated that the α and β diversities were significantly lower during the active phase in contrast with remission phase and healthy samples. In general, Firmicutes were most abundant among the three sample groups, followed by Bacteroidetes and Proteobacteria. Genus level distribution highlighted Streptococcus, Neisseria, Prevotella, Haemophilus, and Veillonella as the five most abundant taxa. Differential abundance analysis of the three sample groups identified significant enrichment of 30 bacterial taxa in the active phase that included g_Prevotella, f_Prevotellaceae, and p_Bacteroidetes. Furthermore, remission phase and control also exhibited significant enrichment of 24 and 22 bacterial taxa, respectively. Eleven differentially abundant pathways were also identified, four were significantly enriched in healthy controls whereas other seven were significantly enriched in active phase of the disease. Several important pathways, such as ribosome biogenesis and Energy metabolism were depleted in the active phase. Our study has highlighted several taxa and functional categories that could be implicated with the onset of Crohn's disease and thus have the potential to serve as biomarkers of the active disease. However, these findings require further validation through functional studies in the future.
... Other findings include aphthous ulcerations, pyostomatitis vegetans, dental caries, gingivitis and periodontitis, angular cheilitis, glossitis, gingival hyperplasia, altered taste perception and candidiasis (Johannsen et al., 2015;Tan et al., 2016). To relieve the pain of aphthous ulcerations, topical agents such as lidocaine and/or topical steroids such as triamcinolone 0.1% can be used, though appropriate treatment of the underlying IBD is crucial (Lankarani et al., 2013;Rowland et al., 2010). If orthodontic treatment is being provided, it is essential to minimise trauma. ...
Article
A wide variety of patients with medical co-morbidities may present to general orthodontic practice. It is important for the treating clinician to have a general understanding of key medical conditions that may impact upon the treatment and management options. This clinical supplement provides a treatment-focused summative update for the orthodontist regarding significant medical co-morbidities, their general prevalence and an exploration of potential impacts upon orthodontic treatment. This review also discusses the significance of key medications and provides suggestions for the safe provision of orthodontic treatment.
... Other non-specific oral manifestations of IBD include stomatitis, glossitis, odynophagia and dysphagia, perioral dermatitis, diffuse pustules and non-specific gingivitis, lichenoid reactions, candidiasis, gingival hyperplasia, papillomatosis of the oral mucosa, pemphigus vegetans, persistent submandibular lymphadenopathy, recurrent buccal abscesses, and metallic dysgeusia [47]. ...
Article
Full-text available
Inflammatory bowel disease (IBD), which includes Crohn’s disease (CD) and ulcerative colitis (UC), can be associated with several extra-intestinal manifestations requiring a multidisciplinary management both in terms of work-up and therapy. Oral lesions are common in patients with IBD, with a prevalence ranging from 5% to 50%. These can represent an oral location of IBD as well as a side-effect of drugs used to treat the intestinal disease. Oral manifestations, occurring in patients with IBD, can be divided in nonmalignant, specific, and non-specific ones, and malignant lesions. While there is undoubtedly a need to search for an IBD in patients with oral lesions associated with intestinal symptoms, the work-up of those with an exclusive oral lesion should be personalized. Fecal calprotectin is a non-invasive marker of intestinal inflammation and may be used to select which patients need to undergo endoscopic examination, thereby avoiding unnecessary investigations. The pharmacological armamentarium to treat oral lesions associated with IBD includes topical or systemic corticosteroids, immunosuppressive agents, and biologic drugs.
... Although several studies indicate that OFG and CD are two distinct disorders (Challacombe 1997;Sanderson et al. 2005;Grave et al. 2009;Zbar et al. 2012), approximately 40-50% of young patients with OFG will either subsequently develop CD (Leao et al. 2004;Saalman et al. 2009;Rowland et al. 2010;Campbell et al. 2011a) or receive a concomitant CD diagnosis (Lazzerini et al. 2014). Since both OFG and CD often are discovered at young age, OFG may denote an initial presentation of CD or could be a subtype of CD (Saalman et al. 2009;Lazzerini et al. 2014Lazzerini et al. , 2015Gale et al. 2016). ...
Article
Full-text available
Aims To evaluate awareness on orofacial granulomatosis (OFG) and oral Crohn disease (oral CD) among Norwegian dental clinicians. Methods A precoded questionnaire (QuestBack) was sent electronically to dentists and dental hygienists treating children and adolescents in the public dental service (PDS) in Norway. Data on the clinicians’ knowledge of OFG and oral CD related to working experience were analysed by Chi square tests and bivariate logistic regression analyses. Results A total of 1097 clinicians were invited to participate, 778 dentists and 319 dental hygienists; 48.2% returned the completed form. Fifty-four percent of the participants had ≥ 10 year experience of clinical practice. Two-thirds (68.4%) of the dentists and all but one of the dental hygienists graduated in Norway. Approximately half of the respondents were aware of OFG (41.2%) and oral CD (57.8%). One-quarter (24.6%) reported that they most likely had seen a patient with OFG and 20.6% with oral CD. Recently graduated dentists (≤ 10 years ago) were more aware of OFG and oral CD than those who graduated > 10 years ago (p ≤ 0.001). Regarding dental hygienists, this difference was observed for OFG only (p < 0.05). Country of education did not affect the clinicians’ reported knowledge. Approximately 90% would refer a patient suspected of having OFG or oral CD either to a dental specialist or to a physician. Conclusion The high prevalence of clinicians observing OFG and oral CD in this study may indicate that OFG and/or oral CD are under-reported and that OFG in particular is more common than hitherto believed. The high frequency of awareness was promising for the benefit of the patients.
... Many studies have shown that medical conditions affect the sense of taste, for example, endocrine, neurological, and nutritional factors (deficiency of niacin and vitamin B12), and viral infections (Boyce & Shone, 2006;Cermak, Curtin, & Bandini, 2010;Kato & Roth, 2012;Santos, Santos Marreiros, Soares de Oliveira, Torres de Freitas, & Clímaco Cruz, 2019;Schiffman & Graham, 2000). The inflammatory process in Crohn's disease is usually located in the distal part of the small intestine but it may also affect the function of the oral cavity (Padmavathi, Sharma, Astekar, Rajan, & Sowmya, 2014;Rowland, Fleming, & Bourke, 2010;Steinbach et al., 2013;Swora, Ślebioda, Grzymisławski, & Szponar, 2008). Decreased salivary flow results in a significant oral discomfort, which may be manifested by altered sensitivity to taste. ...
Article
Full-text available
The aim of the study was to compare differences between healthy young people and young people with Crohn's disease in their sensory sensitivity to basic tastes. The investigations were conducted on a group of 185 people with Crohn's disease and 288 healthy people aged 20–31. The recognition thresholds of sweet, salty, sour, and bitter tastes were determined. The results showed that the young people with Crohn's disease were more sensitive to sour taste and less sensitive to bitter taste than the healthy subjects. The results also showed a relationship between the young Crohn's disease patients' sensitivity to bitter and sour tastes and the folic acid supplementation of their diet. However, there were no differences between the groups in their sensory sensitivity to salty and sweet tastes. Practical applications The sensory properties and perception of foods are very important aspects of food selection and consumption, as they might regulate the nutritional status. Increased or reduced taste sensitivity usually suppresses appetite and results in weight loss, malnutrition, impaired immunity, and deterioration of medical conditions. The knowledge of the taste sensitivity of people with Crohn's disease will help researchers design food products of the right taste intensity. This may improve the nutritional status and quality of life of people suffering from Crohn's disease.
... A wide variety of disease-specific oral lesions have been described in patients with intestinal Crohn's disease. These include swelling of the lips and buccal mucosa, cobblestoning, mucogingivitis, deep linear ulceration or mucosal tags (3). ...
Article
Full-text available
Crohn’s disease (CD) is a chronic disease of the digestive system. It is characterized by lesions predominantly located in the small intestine and colon, although they may also occur in any segment of the gut, including the oral cavity. The involvement of oral mucosa in CD may be underreported, as up to 42% of pediatric patients with CD were found to have oral lesions after undergoing a thorough oral examination. Here, we present a case of CD in which the patient was referred to a dentist due to non-healing aphthous ulcers in the mouth. Our patient, a 16-year-old boy, was admitted to the dentistry clinic with swelling of the oral mucosa and the lips which had been ongoing for 3 months. The patient was referred to our department due to the non-response of the mucosal lesions to repeated cycles of medical treatment. Colonoscopy revealed a cobblestone appearance especially in the left colon, partly normal mucosa, and exudative ulcers. Biopsy samples showed increased inflammatory cell infiltration in the lamina propria and cryptitis in some of the crypts. A close collaboration between gastroenterologists and dentists is useful when addressing the diagnosis and appropriate management of these patients.
... Other features include gingival erythema, 5 mucosal tags, and "cobblestoning" caused by buccal oedema. 6 Being facially disfiguring, the disease carries a significant psychological burden for affected individuals. 7 The underlying cause of OFG remains unknown, but it is likely to have a multifactorial aetiology. ...
Article
Full-text available
Background Orofacial granulomatosis (OFG) is a rare disease characterised by chronic, noncaseating, granulomatous inflammation primarily affecting the oral cavity. Histologically, it is similar to Crohn’s disease (CD), and a proportion of patients have both OFG and CD. The cause of OFG remains elusive, but it has been suggested that microbial interactions may be involved. The aim of this study was to compare the salivary microbial composition of subjects with OFG and/or CD and healthy controls. Methods Two hundred sixty-one subjects were recruited, of whom 78 had OFG only, 40 had both OFG and CD, 97 had CD only with no oral symptoms, and 46 were healthy controls. Bacterial community profiles were obtained by sequencing the V1-V3 region of the 16S rRNA gene. Results There were no differences in richness or diversity of the salivary bacterial communities between patient groups and controls. The relative abundance of the Streptococcus salivarius group was raised in patients with OFG or CD only compared with controls, whereas that of the Streptococcus mitis group was lower in CD compared with both OFG and controls. One S. salivarius oligotype made the major contribution to the increased proportions seen in patients with OFG and CD. Conclusions The salivary microbiome of individuals with OFG and CD was similar to that found in health, although the proportions of S. salivarius, a common oral Streptococcus, were raised. One specific strain-level oligotype was found to be primarily responsible for the increased levels seen.
... Richa Wadhawan E-mail: richawadhawan@gmail.com favour either CD or UC (Rowland et al., 2010). ...
Article
Diagnosis of the disease by dentists and other clinicians through the evaluation of oral clinical findings is a rare incident. Mucocutaneous and granulomatous lesions of the oral cavity alert the clinician to investigate the gastrointestinal tract. This review highlights oral manifestations of gastrointestinal disorders, how various diseases and their presentations are integrated and intertwined. Oral manifestations include stomatitis, glossitis, cheilitis, aphthous ulceration, pyostomatitis vegetans, macrocheilia, cobblestoning of oral mucosa, deep linear ulcers of buccal vestibule and polypoid mucosal tags.
... Persistent inflammation can result in disfiguring fibrotic disease which in some cases causes permanent lip swelling refractory to medical therapy which requires debulking surgery. Other features include gingival erythema [5], mucosal tags and 'cobblestoning' caused by buccal oedema [6]. ...
Preprint
Full-text available
Objective Orofacial granulomatosis (OFG) is a rare disease characterised by chronic, non-caseating, granulomatous inflammation primarily affecting the oral cavity. Histologically, it is similar to Crohn’s disease (CD) and a proportion of patients have both OFG and CD. The cause of OFG remains elusive but it has been suggested that microbial interactions may be involved. The aim of this study was to compare the salivary microbial composition of subjects with OFG and/or CD and healthy controls. Design 261 subjects were recruited, of whom 78 had OFG only, 40 had both OFG and CD, 97 had CD only with no oral symptoms and 46 were healthy controls. Bacterial community profiles were obtained by sequencing the V1-V3 region of the 16S rRNA gene. Results There were no differences in richness or diversity of the salivary bacterial communities between patient groups and controls. The relative abundance of the Streptococcus salivarius -group were raised in patients with OFG or CD only compared to controls while that of the Streptococcus mitis -group was lower in CD compared to both OFG and controls. One S. salivarius oligotype made the major contribution to the increased proportions seen in patients with OFG and CD. Conclusion The salivary microbiome of individuals with OFG and CD was similar to that found in health although the proportions of S. salivarius , a common oral Streptococcus were raised. One specific strain-level oligotype was found to be primarily responsible for the increased levels seen.
... 2 Specific oral lesions resemble the characteristics of intestinal changes observed in CD, and upon histological inspection, these lesions often contain granulomas. 3,4 All tissues of the oral cavity can be affected, including teeth and salivary glands. 3,5 Both reduced saliva secretion rate and xerostomia have been reported as non-specific oral symptoms. ...
Article
Background In Crohn’s disease (CD) patients, many oral complaints have been reported. The aim of this study was to determine whether salivary function is contributing to reduced oral health in CD. Oral and dental complaints in patients were explored. The prevalence of xerostomia in conjunction with salivary flow rates and biochemical saliva composition was studied. Methods The Xerostomia Inventory score (XI-score), the salivary flow rates, the concentrations of salivary amylase and mucin 5B, and the type of oral and dental complaints were evaluated. These outcomes were stratified by disease activity, using the Harvey Bradshaw Index (HBI) and the Inflammatory Bowel Disease Questionnaire (IBDQ-9). Results Fifty-three CD patients in a Dutch tertiary referral hospital were included. Of the patients evaluated, 9.4% had hyposalivation under resting conditions, and 28.3% had hyposalivation under chewing stimulated conditions. Saliva secretion rates were not correlated to XI-scores. Median XI-score was 25 (11–45). XI-scores were correlated to the IBDQ scores (r = −0.352, P = 0.010). Salivary mucin 5B was correlated to disease activity (r = 0.295, P = 0.04). Regarding the number of oral complaints, a correlation with disease activity (HBI r = 0.349, P = 0.011) and experienced xerostomia (r = −0.554, P = 0.000) was observed. Oral and dental problems like oral ulcers (37.7%) and cavities (46%) occurred more frequently in CD patients, especially when compared with a non-IBD population. Conclusions Oral and dental complaints are common in CD patients. Xerostomia is correlated with disease activity–associated quality of life and with the number of oral and dental complaints. Changes in salivary function may contribute to reduced oral health in CD patients. Close
... Patients may also experience mouth and teeth disorders. In CD, oral lesions usually are more IBD-specific (such as cobblestoning, lip and tongue fissures, mucogingivitis, and cheilitis granulomatosa) but also a-specific lesions can be observed (e.g., aphthous stomatitis) [5][6][7][8]. Conversely, in UC, only nonspecific lesions are seen and, broadly speaking, these kinds of lesions are more common than specific lesions [9]. ...
Article
Full-text available
Background Patients with inflammatory bowel diseases could experience mouth and teeth disorders and alterations in psychological mood. Vice versa, the psychological status may influence the presence of oral diseases. Aim To evaluate in inflammatory bowel disease patients the prevalence of sleep bruxism and its correlation with the presence of oral diseases, quality of sleep, and psychological disturbances. Methods Patients were consecutively recruited in our clinic and examined for temporomandibular disorders, dental enamel disorders, sleep bruxism, and recurrent aphthous stomatitis by two dentists. Patients also underwent Pittsburgh Sleep Quality Index and Beck Depression Inventory Scale questionnaires. Results 47 patients and 46 controls were included. Sleep bruxism and enamel wear disorders were more frequent in Crohn's disease patients when compared with ulcerative colitis patients and controls (p = 0.03 and p = 0.02, resp.). Among groups, no differences were noted for enamel hypoplasia, temporomandibular disorders, recurrent aphthous stomatitis, depression, and quality of sleep. We found a positive correlation between bruxism and temporomandibular disorders (Spearman 0.6, p < 0.001) and between bruxism and pathological sleep (Pittsburgh Sleep Quality Index > 5) (Spearman 0.3, p < 0.005). Conclusion Bruxism and enamel wear disorders should be routinely searched in Crohn's disease patients. Moreover, the attention of healthcare givers to sleep disturbances should be addressed to all inflammatory bowel disease patients.
... Oral findings complicating inflammatory bowel disease are found more often in patients with CD rather than UC, more prevalent in children compared to adults, and are found more commonly in men than women [57][58][59]. In the CD population, initial presentation complicated by oral complaints ranges from as low as 0.5% to as high as 60% in the literature [60,61]. ...
Article
Full-text available
Inflammatory bowel disease (IBD) is primarily a disease of the gastrointestinal tract, though it can often affect other organ systems. These extraintestinal manifestations occur in a quarter to one-third of patients with Crohn’s disease and ulcerative colitis. While musculoskeletal and dermatologic manifestations are the most common, it is also important to be cognizant of head, eye, ear, nose, and throat (HEENT) manifestations and educate IBD patients about them. Here we review the ocular manifestations in conjunction with the lesser-known but increasingly recognized ENT manifestations. Considering the lack of randomized controlled trials in treating HEENT manifestations of IBD, this review is primarily based on case reports, case series, and expert opinion with a particular focus on the newer literature supporting use of anti-TNF agents.
... Many studies reported an increased rate of granuloma detection in oral biopsies, reaching up to 77% (14); granulomas were found in 14/16 patients biopsied in two different cohorts (12). In a prospective cohort study, the presence of oral granulomas was the most important diagnostic criterion used for classifying the patient as having CD as opposed to UC (14). ...
Article
Inflammatory bowel diseases (IBD), including Crohn’s disease and ulcerative colitis, have important extraintestinal manifestations, notably in the oral cavity. These oral manifestations can constitute important clinical clues in the diagnosis and management of IBD, and include changes at the immune and bacterial levels. Aphthous ulcers, pyostomatitis vegetans, cobblestoning and gingivitis are important oral findings frequently observed in IBD patients. Their presentations vary considerably and might be well diagnosed and distinguished from other oral lesions. Infections, drug side effects, deficiencies in some nutrients and many other diseases involved with oral manifestations should also be taken into account. This article discusses the most recent findings on the oral manifestations of IBD with a focus on bacterial modulations and immune changes. It also includes an overview on options for management of the oral lesions of IBD.
Article
Full-text available
Idiopathic gingival fibromatosis (GF), also known as gingivomatosis, is a rare condition in childhood, with an unknown aetiology. the oral manifestations of the condition are varied and depend on the severity and age of involvement. this paper describe the case of a 5-year-old male child with extensive gingival enlargement covering almost all the maxillary and mandibular teeth resulted in difficulty with speech, mastication and poor aesthetics. Clinical and radiographic examination along with haematological investigations ruled out any systemic association. the case was managed with conventional scalpel blade surgery along with electrocautery under general anaesthesia yielding good results without any recurrence after a 12-month follow-up. the results revealed that the oral manifestations of GF depend on its severity and the age of onset. timely intervention can help to prevent associated complications in a growing child.
Article
Full-text available
Objective The relationship between oral health and Crohn’s disease is uncertain. Previous studies have yielded contradictory results, reflecting perhaps the different phenotypes of the disease. The aim of the present study was to describe and analyse the dental status of a group of patients with Crohn’s disease (CD), considering the positions of the inflammatory loci and disease phenotype. Methods In total, 47 patients with Crohn’s disease (18 males and 30 females; mean age. 48.7 years; range, 23–61 years) were consecutively recruited to this study. Interviews and clinical examinations were performed to assess dental status, medication, smoking history, heredity of inflammatory bowel disease (IBD), duration of disease, oral mucosal manifestations of Crohn’s disease. Furthermore, data on subjective health assessments and family status, along with medical histories from the patients were obtained through questionnaires. The disease phenotypes were assessed and classified according to the Montreal classification. The data on oral health status were first correlated with the Montreal classifications of IBD, and, thereafter, all the collected data were included in a multivariate generalised linear model. Results The dental status of the patients was comparable to that of the Swedish average. No statistically significant associations were found between oral status and the different CD phenotypes. However, within the Montreal classification, there were significantly fewer teeth in those patients with perianal lesions than in those without such lesions, and there was a significant correlation between deeper pocket depth and problems with strictures and penetrations. No significant differences (p = .074) between the patients with CD (N = 47) and controls (N = 38) were found regarding the presence of oral mucosal lesions. Conclusion Dental health may be adversely affected in severe cases of CD whereas most of the remaining patients with CD appear to have a level of dental health that is comparable to that in the general population.
Article
Full-text available
Crohn's disease (CD) is a multifactorial inflammatory disorder that can affect all segments of the gastrointestinal (GI) tract but typically involves the ileum and/or colon. To assess patient prognosis and choose appropriate treatment, it is necessary to accurately evaluate the factors influencing poor outcomes, including disease phenotype. Pediatric CD involving the upper GI (UGI) tract has become increasingly recognized with the introduction of routine upper endoscopy with biopsies for all patients and the increased availability of accurate small bowel evaluations. Most clinical manifestations are mild and nonspecific; however, UGI involvement should not be overlooked since it can cause serious complications. Although controversy persists about the definition of upper gastrointestinal involvement, aphthoid ulcers, longitudinal ulcers, a bamboo joint-like appearance, stenosis, and fistula are endoscopic findings suggestive of CD. In addition, the primary histological findings, such as focally enhanced gastritis and non-caseating granulomas, are highly suggestive of CD. The association between UGI involvement and poor prognosis of CD remains controversial. However, the unstandardized definition and absence of a validated tool for evaluating disease severity complicate the objective assessment of UGI involvement in CD. Therefore, more prospective studies are needed to provide further insight into the standardized assessment of UGI involvement in and long-term prognosis of CD. Our review summarizes the findings to date in the literature as well as UGI involvement in CD and its clinical implications.
Article
Oral Crohn's disease (OCD) refers to the spectrum of oral manifestations seen in Crohn`s disease (CD) patients and could precede other signs of the disease elsewhere, which requires dental practitioners to be vigilant for oral mucosal changes that may accompany such systemic disorders. Classical treatment modalities are based on the use of corticosteroids, immune‐modulators and more recently, biologics. Ustekinumab is a novel agent from this last group that has shown efficacy in a limited number of case reports. A case of a debilitating CD presenting initially with oral manifestations and ultimately managed with ustekinumab is reported.
Chapter
This chapter focuses upon the clinical presentation, diagnostic approach, and advanced treatment strategies for children diagnosed with Crohn disease. The pediatric IBD community continues to make great progress in the evaluation and management of this complex patient group. New therapies are being developed and therapy optimization continues to improve. Optimal management requires care delivered by experienced, multidisciplinary teams focused upon comprehensive care, quality improvement, and care standardization. Advancements in research/clinical trials are ongoing and promise to continue to improve outcomes for children with Crohn disease.
Chapter
Local and systemic conditions frequently have oral and dental manifestations. This chapter reviews some common and not so common pediatric disorders and describes their genetic patterns and disease characteristics. In addition, clinical findings are described as well as contemporary medical treatments. Oral and dental manifestations are described as well as precautions that the dental team must consider because of drugs the patient may be taking. Pediatric cancer, a common cause of fatalities in children, is reviewed and how the multimodal treatment approach of surgery, radiotherapy, and chemotherapy is used to eradicate the disease. The acute and long-term oral and dental sequelae of medical therapies are described, and the importance of routine oral hygiene through the entire oncology treatment is stressed. Finally, hematopoietic stem cell transplantation is discussed as well as the importance of a healthy mouth to reduce the severe morbidity associated with the treatment.
Article
Full-text available
Background and Objectives: Inflammatory bowel disease (IBD) is a term that refers to crohn's disease (CD) and ulcerative colitis (UC). Oral manifestations in this disease category precedes the onset of gastrointestinal symptoms. In many patients, intestinal symptoms may be minimal or remain undiagnosed. In this paper, two cases of Pyostomatitis vegetans have been investigated. Case Report: The first case was a 29 year old man who was referred with complaints of diffuse oral lesions. According to the clinical diagnosis and oral lesions of the patient, he was referred to a gastroenterologist with possible diagnosis of ulcerative colitis. The second case was a 28 year old woman with painful gingival lesions who stated that she has been diagnosed with ulcerative colitis since three years ago and has not received appropriate treatment.
Article
Orofacial granulomatosis (OFG) is an uncommon chronic inflammatory disorder that can present in childhood. It has a range of clinical manifestations with the common features being lip swelling and oral ulceration. It can be idiopathic or associated with systemic granulomatous conditions such as Crohn's disease and sarcoidosis. Patients presenting with features suggestive of OFG, with or without gastrointestinal symptoms, should be referred promptly to secondary care. The management of OFG is often challenging and includes dietary restrictions; topical, intralesional and systemic corticosteroids; and other systemic immunomodulatory drugs. This condition can lead to significant psychological morbidity for the child if left untreated. CPD/Clinical Relevance: Orofacial granulomatosis can present in childhood and its features may be first noted on routine check-up with a general dental practitioner (GDP).
Thesis
La maladie de Crohn, maladie inflammatoire chronique de l'intestin, est marquée par l'alternance depoussées et de rémissions. Cela pose des difficultés dans sa prise en charge. Son incidence est globalementen augmentation, notamment dans les pays d'Europe de l'Ouest, d'où notre intérêt pour cette affection.Des manifestations extra-intestinales touchant la cavité buccale peuvent y être associées. Celles-ci, peuconnues des chirurgiens-dentistes, ont fait l'objet de nombreuses études. Aujourd'hui, les conséquences decette maladie sur la sphère bucco-dentaire sont clairement identifiées.L'objectif de notre travail est de présenter cette maladie, son impact sur les tissus de la cavité buccale ainsique sa prise en charge dans le cadre d'un cabinet dentaire.
Article
Crohn's disease is a chronic granulomatous inflammatory disorder that is often accompanied by oral symptoms. We herein report a case of Crohn's disease that initially occurred as gingival swelling. A 63-year-old woman complained of gingival swelling and was referred to our clinic. During the disease course, Behcet's disease was denied despite the development aphthous ulcers and erythema nodosum. Thereafter, abdominal pain, diarrhea and weight loss were observed, and Crohn's disease was subsequently diagnosed by endoscopy. Because oral symptoms may precede Crohn's disease, it is important to include this possibility in the differential diagnosis when encountering patients presenting with oral swelling and ulcerative lesions.
Article
Full-text available
Nineteen patients with clinical evidence of oral Crohn's disease but no intestinal symptoms were studied. Oral lesions in all patients were shown histologically to have lymphoedema with or without chronic granulomas consistent with Crohn's disease. Seven patients (37%) had demonstrable intestinal disease on rectal biopsy and four of these had abnormal bowel radiology. All seven had evidence of nutritional deficiency. Patients with clinical features suggesting oral Crohn's disease may have evidence of Crohn's disease in the intestine, although this may not be clinically apparent.
Article
Full-text available
Crohn's disease of the mouth or perineum is more common in young people, and notably resistant to treatment. However, there is increasing evidence that topical therapy with tacrolimus (FK506) may be effective in skin diseases resistant to cyclosporin because of its high uptake in inflamed skin and subsequent reduction in keratinocyte chemokine production. Tacrolimus ointment was made up inhouse from the intravenous or oral formulation and suspended in appropriate vehicles for perioral or perianal administration at an initial concentration of 0.5 mg/g. This was administered open label to eight children (aged 5-18 years) with treatment resistant oral (three patients) and/or ulcerating perineal (six patients) Crohn's disease. Marked improvement was seen in 7/8 patients within six weeks and healing within 1-6 months. One child with gross perineal and colonic disease showed little response. Two of the responders showed rebound worsening when tacrolimus was stopped or the dosage reduced rapidly, and one of these eventually required proctectomy. Slower weaning of drug concentration has been successful in 6/8 patients, with four receiving intermittent treatment and two on regular reduced dosage (0.1-0.3 mg/g) with follow up times of six months to 3.5 years. Serum concentrations of tacrolimus were undetectable in all patients. Topical tacrolimus at low concentrations (0.5 mg/g) shows promise in the management of childhood perineal and oral Crohn's disease, with no evidence of significant systemic absorption. However, rapid weaning or abrupt cessation of therapy may cause rebound worsening of disease. Further controlled studies are required to assess the efficacy and safety of this treatment.
Article
Full-text available
Granulomatous cheilitis is characterized by recurrent swelling of the labial tissues and granulomatous histology. Granulomatous cheilitis has been recognized as an early manifestation of Crohn's disease. It may follow, coincide with or precede the onset of Crohn's disease. The first case presented involved an extraintestinal manifestation of Crohn's disease, and the second case presented is of development of granulomatous cheilitis a year before the onset of symptomatic Crohn's disease. Although chronic granulomatous cheilitis is a very rare disorder, once it is diagnosed, the patient should be followed up carefully. These patients should be investigated for asymptomatic Crohn's disease either when the diagnosis of granulomatous cheilitis is confirmed or when gastrointestinal symptoms develop.
Article
Full-text available
No previous correlation between phenotype at diagnosis of Crohn's disease (CD) and mortality has been performed. We assessed the predictive value of phenotype at diagnosis on overall and disease related mortality in a European cohort of CD patients. Overall and disease related mortality were recorded 10 years after diagnosis in a prospectively assembled, uniformly diagnosed European population based inception cohort of 380 CD patients diagnosed between 1991 and 1993. Standardised mortality ratios (SMRs) were calculated for geographic and phenotypic subgroups at diagnosis. Thirty seven deaths were observed in the entire cohort whereas 21.5 deaths were expected (SMR 1.85 (95% CI 1.30-2.55)). Mortality risk was significantly increased in both females (SMR 1.93 (95% CI 1.10-3.14)) and males (SMR 1.79 (95% CI 1.11-2.73)). Patients from northern European centres had a significant overall increased mortality risk (SMR 2.04 (95% CI 1.32-3.01)) whereas a tendency towards increased overall mortality risk was also observed in the south (SMR 1.55 (95% CI 0.80-2.70)). Mortality risk was increased in patients with colonic disease location and with inflammatory disease behaviour at diagnosis. Mortality risk was also increased in the age group above 40 years at diagnosis for both total and CD related causes. Excess mortality was mainly due to gastrointestinal causes that were related to CD. This European multinational population based study revealed an increased overall mortality risk in CD patients 10 years after diagnosis, and age above 40 years at diagnosis was found to be the sole factor associated with increased mortality risk.
Article
We report four new cases of oral manifestations in Crohn's disease (CD) and evaluate 75 reported cases for morphology and site of oral and intestinal manifestations of CD, clinical manifestation, and treatment. Oral CD was the presenting symptom in 43 of 72 (60%) patients and relapsed in 34 of 60 (57%). Median age at presentation was 22 (range 6-57) years, and males were affected more often (1.85:1, male:female ratio). From a total of 228 oral lesions in 79 patients, lips (57 lesions), gingiva (40 lesions), vestibular sulci (31 lesions), and buccal mucosa (25 lesions) were the sites most frequently affected. Edema (62 lesions), ulcers (57 lesions), and polypoid papulous hyperplastic mucosa (45 lesions) were the most common type of lesions. The rate of granuloma detection was high in oral (67-77%) and intestinal lesions (45-71%). A total of 66 courses of drug therapy in 51 patients were analyzed. Complete remission of oral symptoms was achieved by systemic steroids and/or azathioprine in 13 of 26 (50%) patients, whereas strictly topical treatment with steroids resulted in complete remission of oral symptoms in 7 of 12 (58%). We conclude that oral CD exhibits a characteristic morphologic appearance, as often as not preceding intestinal symptoms in adolescents and young adults. Thus, patients with orofacial granulomatosis CD should be vigorously searched for by complete gastrointestinal endoscopic investigation. Oral CD may cause disabling pain and facial distortion, and results of treatment remain unrewarding. In the absence of data from controlled therapeutic trials, systemic steroids and/or azathioprine are recommended if topical treatment has failed to control symptoms.
Article
Orofacial granulomatosis is a term generally used to describe lip swelling secondary to an underlying granulomatous inflammatory process. Granulomatous cheilitis is the histopathological description of such inflammation occurring in the lips and surrounding tissues. Melkersson‐Rosenthal syndrome (a triad of orofacial swelling, facial paralysis and a fissured tongue) is one manifestation of orofacial granulomatosis, which more commonly presents as granulomatous cheilitis alone. Oral Crohn's disease also belongs to the entity of orofacial granulomatosis. Most reported cases of orofacial granulomatosis have been in adults and some in adolescents. We present six children presenting with orofacial granulomatosis at an early age (range 5–8 y) whose course points towards the development of Crohn's disease. Conclusion: Orofacial granulomatosis in the paediatric population may be an initial manifestation of Crohn's disease and so careful surveillance is recommended.
Article
Orofacial granulomatosis (OFG) is a chronic inflammatory disorder presenting characteristically with lip swelling but also affecting gingivae, buccal mucosa, floor of mouth, and a number of other sites in the oral cavity. Although the cause remains unknown, there is evidence for involvement of a dietary allergen. Patch testing has related responses to cinnamon and benzoate to the symptoms of OFG, with improvement obtained through exclusion diets. However, an objective assessment of the effect of a cinnamon- and benzoate-free diet (CB-free diet) as primary treatment for OFG has not previously been performed. Thus, this study was undertaken to investigate the benefits of a CB-free diet as first-line treatment of patients with OFG. Thirty-two patients with a confirmed diagnosis of OFG were identified from a combined oral medicine/gastroenterology clinic. All had received a CB-free diet as primary treatment for a period of 8 weeks. Each patient underwent a standardized assessment of the oral cavity to characterize the number of sites affected and the type of inflammation involved before and after diet. There was a significant improvement in oral inflammation in patients on the diet after 8 weeks. Both global oral and lip inflammatory scores improved (P<0.001), and there was significant improvement in both lip and oral site and activity involvement. However, improvement in lip activity was less marked than oral activity. Response to a CB-free diet did not appear to be site specific. A history of OFG-associated gut involvement did not predict a response to the diet. The impact of dietary manipulation in patients with OFG can be significant, particularly with regard to oral inflammation. With the disease most prevalent in the younger population, a CB-free diet can be recommended as primary treatment. Subsequent topical or systemic immunomodulatory therapy may then be avoided or used as second line.
Article
It has become widely recognised that oral lesions may occur in patients with Crohn's disease of the lower gastro-intestinal tract. Patients have also been described with oral lesions of this kind unassociated with gut lesions. The purpose of this paper is to describe the clinical presentation of seven such patients. The resemblance of their lesions to those of the condition known as chronic granulomatous cheilitis is pointed out. The association between the oral lesions and those of the lower gut is discussed and it is reported that in one patient acute toxic dilation of the colon due to Crohn's disease followed a period in which oral lesions only were recognised.
Article
The records of a series of 700 patients with inflammatory bowel disease, 498 with Crohn's disease and 202 with ulcerative colitis, have been analyzed to determine the relative incidence and characteristic features of their extra-intestinal manifestations. The group with Crohn's disease included 62 with colitis, 223 with ileocolitis, and 213 with regional enteritis. A consideration of the clinical patterns and an understanding of their pathophysiology suggested a subdivision into two main groups: one "colitis related" and one related to the pathophysiology of the small nonspecific third group. Group A, colitis related, comprises joint, skin, mouth, and eye disease. The complications might be immunologically determined, were closely associated with active inflammation, and often responded to medical or surgical treatment of the underlying bowel disease. They occurred in 36% of the entire series of patients: joints were involved in 23%, skin in 15%, and mouth and eye each in 4%. Pyoderma gangrenosum was observed most often in ulcerative colitis and erythema nodosum most often in granulomatous colitis. The incidence of Group A complications was higher in disease involving the colon (42%) than in disease restricted exclusively to the small bowel (23%). There were interrelationships among the various members of Group A, with multiple manifestations occurring in a third of affected patients. Group B, related to small bowel pathophysiology, includes malabsorption, gallstones, kidney stones, and non-calculous hydronephrosis and hydroureter. Disorders in this group were generally related to the severity of the disease in the small bowel and tended to persist even in the absence of active inflammation. In contrast to Group A, this group occurred most frequently in small bowel disease, and least in colonic disease. Malabsorption was virtually confined to the patients with small bowel disease (10% incidence), while gallstones and renal stones were also both more frequent in Crohn's disease (11% and 9% respectively), the latter usually in association with small bowel resection or ileostomy. Group C, found in a small percentage of patients, consists of nonspecific complications, including osteoporosis (3%), liver disease (5%), peptic ulcer (10%), and amyloidosis (1%).
Article
In a systematic study of 100 patients with Crohn's disease, 100 with ulcerative colitis, and of 100 normal subjects matched for age, sex, and denture status, nine patients with Crohn's disease, two with ulcerative colitis, and one normal control were found to have oral lesions. In Crohn's disease, the macroscopic and histological appearances resembled those encountered elsewhere in the gastrointestinal tract and their incidence was related to the activity of the disorder. The lesions in the other two groups were different macroscopically and histologically. Production of salivary IgA was found to be reduced in Crohn's patients with active bowel disease. It is suggested that the occurrence of oral lesions in patients with Crohn's disease might represent a local immunological reaction to oral antigens.
Article
Oral Crohn's disease has been reported frequently in the last two decades with and without intestinal manifestations. In the latter case it is considered one of the orofacial granulomatoses. This reference is claimed to eliminate the patient's distress caused by the term Crohn's disease. There has been much doubt whether intestinal Crohn's disease will eventually develop in these patients. A case is reported in a 10-year-old West Indian girl in whom oral Crohn's disease developed at the age of 10 years and was followed by rectal Crohn's disease 9 years later. In this article the term Crohn's disease is used to describe those patients with intestinal manifestations with or without oral disease whereas oral Crohn's disease is used for those who have only oral manifestations.
Article
A patient is described with generalized gastrointestinal involvement by Crohn's disease. Symptoms of recurrent ulceration and mucosal tags are well-described oral manifestations of Crohn's disease; however, in our patient recurrent facial abscesses, which required extraoral drainage, also developed. This complication has not previously been reported.
Article
The clinical features, treatment and outcome of 29 patients with oral Crohn's disease seen over a 6-year period have been reviewed. Findings on clinical examination included labial swelling (19 patients), buccal mucosal cobblestoning (11), linear ulceration (11), lumps (five), and mucosal tags (two). Eleven patients had multiple features. Eight patients developed symptoms within the first decade of life and nine patients had symptoms for more than 4 years before diagnosis; the mean age at diagnosis was 30 (range 6-78) years. Fourteen of these patients (48 per cent) have Crohn's disease elsewhere in the alimentary tract, and in nine patients the oral disease predated the development or detection of Crohn's disease at other sites. Eight patients (25 per cent) have required no specific therapy for their oral disease and 12 have been treated with systemic corticosteroids of whom three are steroid-dependent. No other pharmacological approach to treatment has been successful and elimination diets, tried by five patients, had no effect. Oral Crohn's disease has a characteristic naked-eye appearance, may be the first or only manifestation of Crohn's disease and usually improves with oral corticosteroid treatment.
Article
The clinical presentation of seven patients with oral lesions of Crohn's disease, but with no evidence of other gastrointestinal involvement was described by Tyldesley in 1979. The clinical progress of five of these patients available for review is reported after a minimum period of 10 years. The gross oedema of the lips and painful buccal lesions present in all five patients have shown gradual resolution from the time of initial diagnosis. The nomenclature for oral Crohn's disease and related conditions is discussed with specific reference to the term 'orofacial granulomatosis'.
Article
Pyostomatitis vegetans is a rare and unusual disorder of the oral cavity, characterized by erythema and edema of the mucosa and numerous small, superficial yellow pustules. Its significance lies in its association with inflammatory disease of the bowel, either ulcerative colitis or Crohn's disease. We report here a patient with pyostomatitis vegetans who, upon medical workup, was discovered to have asymptomatic Crohn's disease. The clinical features, histopathology, and treatment of pyostomatitis vegetans, and its purported relationship to pemphigus vegetans of Neumann are discussed.
Article
Crohn disease, a devastating chronic illness, often develops in the childhood years. The disease is a segmental transmural intestinal disease that may involve one or more segments of the gut from the mouth to the anus. Crohn disease is frequently confused with ulcerative colitis. Frequent oral manifestations include persistent oral ulcerations, diffuse gingival swelling, mucosal hyperplasia and fissuring, cheilitis, and pyostomatitis vegetans. Frequently, oral biopsy is essential in establishing a correct diagnosis of the disease. Although no cure is currently available for the Crohn disease, the proper diagnosis of the problem is essential for providing symptomatic relief. A case report is presented documenting the value of the proper diagnosis of the disease. The literature is reviewed and updated.
Article
A study of 60 patients with oro-facial granulomatosis has been conducted and the clinical presentation of this disorder defined. It encompasses the previously recognised clinical entities of Melkersson-Rosenthal syndrome and cheilitis granulomatosa. The pathological features of the disease are lymphoedema and the presence of multiple non-caseating giant cell granulomata. These granulomata are histologically indistinguishable from those found in both gastrointestinal Crohn's disease and systemic sarcoidosis. Within this series of patients, nine had evidence suggestive of gastrointestinal Crohn's disease, and in six this was confirmed. A diagnosis of sarcoidosis was made in a further two patients. The relationship of oro-facial granulomatosis to these systemic granulomatous diseases is not yet clear. Patients with oro-facial granulomatosis who have gastrointestinal symptoms should be investigated for the presence of gastrointestinal Crohn's disease. Those without symptoms should be investigated for evidence of malabsorption or serological evidence of Crohn's disease. Within the present study, the erythrocyte sedimentation rate, full blood count, corrected whole blood folate, serum albumin and calcium were the most sensitive markers of gastrointestinal involvement. Sarcoidosis should be considered in all patients with oro-facial granulomatosis. The absence of clinical signs suggestive of sarcoidosis, a normal chest radiograph and normal levels of serum angiotensin-converting enzyme makes sarcoidosis unlikely.
Article
Oral lesions, both symptomatic and asymptomatic, occur in a significant number of patients with IBD. The majority occur in patients with active intestinal disease and their presence frequently correlates with results of laboratory tests of activity. However, a minority occur either before there is any evidence of intestinal disease or even following panproctocolectomy. Of more practical importance is the additional finding of abnormal biopsies from a macroscopically normal mouth mucosa in the majority of patients with CD and in some with UC. Abnormal immunofluorescent staining has also been described in biopsies of normal lips in IBD. However, the disease specificity of this finding remains to be clarified. It is concluded that more attention should be given to the mouth in IBD. It is accessible and it is frequently involved in the disease process. Such studies could help in understanding the pathogenic mechanisms underlying IBD.
Article
The frequency and type of oral mucosal lesions, dental infections, and salivary constituents were evaluated in 53 patients with Crohn's disease, who were divided into inactive, mildly active, and severely active groups on the basis of clinical and endoscopic criteria. Buccal biopsies from nine patients with active disease showed morphologic changes that suggested Crohn's disease-related lesions. Panoramic radiographs revealed more infectious foci in the teeth of patients with active Crohn's disease than in patients with inactive disease. Salivary flow rate, buffering capacity, total protein, amylase, and IgA and IgG concentrations did not differ with respect to the activity of Crohn's disease. The observed mucosal inflammation in patients with active Crohn's disease, although high in frequency, was mild and did not need therapy, but the great number of dental infections in association with the activity of Crohn's disease should be taken into account in the treatment of these patients.
Article
Oral features of Crohn's disease include ulcers, lip fissuring, cobblestone plaques, angular cheilitis, polypoid lesions, and perioral erythema. Pyostomatitis vegetans is a rare eruption of the oral mucosa characterized by tiny yellow pustules. It is considered a marker for inflammatory bowel disease. We describe a 45-year-old woman with a 6-month history of painful sores in her mouth, diarrhea, weight loss, and cutaneous lesions. Oral examination revealed cobblestone plaques and indentation on the tongue and friable vegetating pustules on the labial commissures. Staphylococcus simulans was isolated from the pustules. Laboratory studies revealed leucocytosis, eosinophilia, and low hemoglobin and zinc levels. Histologic study of the labial lesions revealed hyperplastic epithelium with intraepithelial clefts that contain eosinophils and neutrophils. Tongue lesions showed chronic inflammation with noncaseating granulomas. Later, colonoscopy and biopsy demonstrated Crohn's disease of the anorectal region. Pyostomatitis vegetans lesions regressed after oral zinc supplementation. Prednisone treatment resulted in healing of the tongue lesions. In our patient, pyostomatitis vegetans appeared to be related to zinc deficiency that may have been caused by malabsorption. The pathogenetic interrelationship between pyostomatitis vegetans and Crohn's disease is discussed.
Article
To obtain precise data on the prevalence of oral lesions in inflammatory bowel disease (IBD). Oral lesions were carefully sought in a consecutive series of 198 Italian IBD outpatients, 77 with Crohn's disease (CD) and 121 with ulcerative colitis (UC); 89 subjects with functional intestinal motility disorders served as controls. The oral lesions detected were angular cheilitis (in 7.8% of CD patients, 5% of UC patients, and 0% of controls (p < 0.05, patients vs controls), lichen (6.5, 5.8, and 3.3%, respectively, p = not significant), aphthous ulcers (5.2, 5.8, and 5.6%, respectively, p = not significant), candidiasis (5.2, 0.8, and 0%, respectively, p < 0.05, CD patients vs controls), benign tumors (5.2, 0, and 7.8%, respectively, p < 0.05, patients vs controls), leukoplakia (5.2, 11, and 3.3%, respectively, p = not significant), and, less frequently, glossitis and herpes labialis. No specific CD oral lesions were observed in this series. No correlation was found between clinical disease activity and frequency of oral lesions. Aphthous ulcers are not common in IBD patients. Oral candidiasis is more frequent in CD than UC patients and controls.
Article
Crohn's disease is an inflammatory disease of unknown etiology. Oral manifestations appear most frequently on the lips, gingival tissue and buccal mucosa. The case presented here shows how a patient with oral lesions resulting from Crohn's disease can be treated by laser therapy and obtain optimal esthetic results.
Article
Oral localization of Crohn disease is uncommon and must be differentiated from nonspecific lesions. Its natural course and its long-term prognosis are unknown. We studied 9 patients (8 male, 1 female; age range, 7-52 years; median age, 16 years) with Crohn disease and specific oral lesions, including deep linear ulcers, pseudopolyps, and/or labial or buccal swelling and induration. The prevalence of such lesions was 0.5%. The median follow-up was 11 years. Oral localization developed before (n = 2), at the same time as (n = 2), or after (n = 5) the onset of the digestive disease. Noticeable associated localizations were observed in the anoperineum (n = 8) and the esophagus (n = 3). The median duration of the oral lesions was 4 years (range, 1-13 years), without necessary parallelism with the digestive localization. Five patients had complete healing after a median delay of 2 years. Oral localization of Crohn disease is characterized by a marked male predominance, a young age at onset of Crohn disease, and a very protracted course. The high prevalence of associated anal and esophageal involvement suggests that Crohn lesions have a particular trophicity for squamous cell epithelium.
Article
Orofacial granulomatosis is a granulomatous inflammatory disorder, affecting the soft tissues of the face and mouth. The predominant feature is disfiguring lip swelling. Patients with this condition may be exhibiting a Type IV hypersensitivity reaction to dietary or environmental allergens, or these may be the orofacial manifestations of underlying gastrointestinal Crohn's disease. The results of 99Tcm-HMPAO leucocyte labelling of the gastrointestinal tract in 14 patients with orofacial granulomatosis and 15 patients with known gastrointestinal Crohn's disease are presented, indicating that this is a useful and non-invasive screening test for the identification of gastrointestinal Crohn's disease in paediatric and young adult patients presenting with orofacial granulomatosis.
Article
Figure 1. An eight-year-old girl had a four-month history of painful mouth lesions, resulting in decreased oral intake and weight loss, and a one-year history of intermittent abdominal pain and irregular bowel movements. On examination, she had cheilitis, redness and swelling of the gingiva, and oral ulcerations (Panels A and B), as well as tenderness of the right lower quadrant and a perianal fissure. Colonoscopy with multiple biopsies revealed findings consistent with the presence of Crohn's disease. Five weeks after the initiation of treatment with 40 mg of oral prednisone daily (2 mg per kilogram of body weight), the patient's . . .
Article
Orofacial manifestations of Crohn's disease can be difficult to diagnose and treat. We report a case in which the orofacial lesions occurred 7 years prior to the diagnosis of underlying inflammatory bowel disease. The patient was refractory to mesalamine and systemic corticosteroids but responded to infliximab, the chimeric monoclonal antibody to tumor necrosis factor (TNF-alpha). A review of the literature of the orofacial granulomatoses is presented as well.
Article
We assessed the utility of expert oral examination as a part of the diagnostic evaluation of patients with suspected Crohn's disease. Of 45 patients with newly diagnosed CD, 25 had been examined by a dentist. Twelve (48%) of these had oral CD lesions. Mucosal tags constituted the most frequent form of oral lesion (8/12). Of 8 oral biopsy specimens, 6 (75%) contained non-caseating granulomas. Patients with oral CD had more oral symptoms, presented for diagnosis sooner, and were more likely to have other upper gastrointestinal inflammation than those without oral lesions. Oral manifestations of CD are common in children; therefore, expert oral examination may be useful during diagnostic evaluation of children with suspected inflammatory bowel disease.
Article
Patients with Crohn's disease generally present with chronic diarrhoea and/or abdominal pain. However, it may be the extraintestinal manifestations as orofacial granulomatosis (OFG)--a rare syndrome with chronic swelling of the lips and the lower half of the face combined with oral ulcerations and hyperplastic gingivitis--that urge patients to seek medical advice. We report two rare cases in which swelling of the lips and cheeks were the initial symptoms that finally led to the diagnosis of Crohn's disease.
Article
A 12 year old boy presented via his orthodontist with a three month history of facial swelling and a one year history of intermittent diarrhoea. Examination revealed a pale boy with asymmetrical thickening of his cheeks, a buccal mucosal fold on the left, and friable mucosa on the right. Investigations revealed a mild hypochromic microcytic anaemia and raised inflammatory markers. An oral biopsy showed occasional granulomata and a mixed inflammatory infiltrate with negative fungal stains consistent with a diagnosis of orofacial granulomatosis. 99Tcm-HMPAO leucocyte scintigraphy showed grade 3 white cell activity in the terminal ileum …
Article
Dermatologic extraintestinal manifestations of Crohn disease may be refractory to treatment with corticosteroids and immunomodulators. The authors describe four children with Crohn disease with dermatologic manifestations: pyoderma gangrenosum, orofacial involvement, erythema nodosum, and idiopathic lymphedema. These dermatologic conditions were unresponsive to conventional therapy but had rapid and sustained response to the anti-TNF-alpha antibody infliximab. No adverse reactions occurred. Infliximab should be considered for treating the extraintestinal dermatologic manifestations of Crohn disease in children.
Article
A 35-year-old woman with severe fistulizing Crohn's disease presented with pyostomatitis vegetans affecting both the mouth and the vulva. The coalescing pustules transformed within several days into vegetating lesions on areas of inflammation. Microbial assessments revealed no pathogenic agent. Histology showed neutrophilic microabscesses, but no granulomas. Three injections of infliximab and maintenance therapy with methotrexate resulted in rapid and complete regression of both the pyostomatitis vegetans and the Crohn's disease. Infliximab and methotrexate may be a promising treatment for the rare cases of pyostomatitis vegetans associated with Crohn's disease.