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Pilonidal sinus of atypical areas: presentation and management

Authors:

Abstract

ABSTRACT Introduction: Pilonidal sinus (PNS) is an inflammatory disorder due to hair penetrating the epidermis. It occurs usually in sacrococcygeal region. Clinical course and management of PNS in atypical areas has never been addressed in a systematic review. Methods: Google scholar was used to identify articles describing pilonidal sinus in any place other than the sacro-coccygeal area. Results: From 37 papers, there were reports of 302 cases of pilonidal sinus occurring in various sites other than sacro-coccygeal area. 253 cases (85%) were male. The age of presentation ranged from 16 to 70 years with average of 24.3 years. The most common atypical sites of pilonidal sinus was umbilicus (91%), followed by hand (4%). Conclusion: Umbilical PNS is the most common one. Pain, swelling and discharge is the usual presentation. Apart from umbilical PNS which is mostly managed conservatively, PNS of atypical sites is managed by excision and primary repair.
Pilonidal sinus of atypical areas: presentation and
management
Salih AM1,
Kakamad FH2,3,
Essa RA3,4 ,
Aziz MS3,5,
Salih RQ3,6 ,
Mohammed SH3,6,
Saeed YA4,
Habibullah IJ4,
Muhialdeen AS4,
Othman S3,
Mohameed HA3
REVIEW ARTICLE
Corresponding author: 2F. Hussein
Kakamad, Faculty of Medical Sciences,
School of Medicine, Department
Cardiothoracic and Vascular Surgery,
University of Sulaimani,
François Mitterrand Street,
Sulaymaniyah, Iraq; Email:
fahmi.hussein@univsul.edu.iq
1,3,4,5,6 see pages 12, 13
Reviewers: DS, AW
Date of submission: 28/12/2016
INTRODUCTION
Pilonidal sinus (PNS) is an inflammatory disorder caused by hair penetrating the epidermis.
Granulation tissue lines a blind tract, which ends in a cavity containing pus (1). It is clinically
manifested by a classical inflammatory pattern: local pain, heat, tenderness and erythema (2). It
may present with purulent discharge (3). It is commonly identified in drivers involving the sacro-
coccygeal region (4).
Several methods have been proposed and practiced for management of PNS including
removing the hair and cotton-like dirt in an outpatient fashion and excision with or without
ABSTRACT
Introduction: Pilonidal sinus (PNS) is an inflammatory disorder due
to hair penetrating the epidermis. It occurs usually in sacro-
coccygeal region. Clinical course and management of PNS in atypical
areas has never been addressed in a systematic review.
Methods: Google scholar was used to identify articles describing
pilonidal sinus in any place other than the sacro-coccygeal area.
Results: From 37 papers, there were reports of 302 cases of pilonidal
sinus occurring in various sites other than sacro-coccygeal area. 253
cases (85%) were male. The age of presentation ranged from 16 to
70 years with average of 24.3 years. The most common atypical sites
of pilonidal sinus was umbilicus (91%), followed by hand (4%).
Conclusion: Umbilical PNS is the most common one. Pain, swelling
and discharge is the usual presentation. Apart from umbilical PNS
which is mostly managed conservatively, PNS of atypical sites is
managed by excision and primary repair.
Keywords: pilonidal sinus, unusual area, atypical area, umbilical,
scalp.
DOI:
Pilonidal Sinus Journal (2017) 3(1), 8 - 15
Cite this Article as: Salih A, Kakamad F, Essa R. et al. "Pilonidal sinus of atypical areas: presentation and management".
PSJ (2017) 3(1): 8-15.
9
reconstruction. Both primary closure and allowing for secondary healing with daily dressing are
reported (5-7). General, regional or local anesthesia can be used (7). PNS of atypical areas
started to be increasingly reported in literature in form of case reports and small case series (5-
8).
Atypical areas include groin, interdigital web, umbilicus, nose, inter mammary areas, supra-pubic
area, clitoris, prepuce, penis, occiput, and on the feet. Clinical course and management of PNS in
atypical area has never been addressed in a systematic review. The current study highlights the
presentation and management of pilonidal sinus in atypical areas.
METHODS
Google Scholar engine was searched for the key words (pilonidal sinus of unusual area, pilonidal
sinus of atypical area and pilonidal sinus case report). The search found 155 items. From which,
40 papers were excluded by title because either they were duplicate or unrelated to the subject.
From the remaining 115, 75 articles excluded because they did not fulfill the inclusion criteria.
The inclusion criteria were any article which mentioned presentation and management provided
that the pilonidal sinus occurred in any place other than the sacro-coccygeal area. The data were
re-analyzed and presented in tables for some variables and others are presented without re-
analysis.
RESULTS
From 40 papers, 302 cases of pilonidal sinus were reported occurring in sites other than sacro-
coccygeal area. From which, 255 cases (84%) were male. The age of presentation ranged from 16
to 70 years with an average of 24.3 years. The most common atypical site of pilonidal sinus was
umbilicus (272 cases; 90%) followed by hand (12 cases; 4%). Table 1 shows the reported atypical
sites of pilonidal sinus with their frequency.
Pilonidal Sinus Journal (2017) 3(1), 8 - 15
Cite this Article as: Salih A, Kakamad F, Essa R. et al. "Pilonidal sinus of atypical areas: presentation and management".
PSJ (2017) 3(1): 8-15.
10
References
%
No. of cases
Site
3-5,9-17
90.0
272
umblicus
6,7,18-24
3.9
12
hand
2,25,26
1.7
5
scalp
27-29
1.0
3
intermammary
1,30-32
1.3
4
perianal
33,34
0.7
2
face
35
0.3
1
periareola
36
0.3
1
penis
37
0.3
1
clitoris
38
0.3
1
prepuce
100
302
Total
Tale 1: Atypical sites of pilonidal sinus.
The most common presentation of umbilical pilonidal sinus was discharge, swelling and pain
(85%) followed by only discharge (11.8%) associated with bleeding (2.3%). Removal of hair with
daily dressing was the most frequent treatment modality (73.5%) which was performed without
anesthesia. Otherwise umbilectomy under general anesthesia was associated with a recurrence
rate of 2%.
Discharging sinus was the most common presentation of the interdigital and hand pilonidal
sinuses (90%) which were most commonly treated by surgical excision under general anesthesia
(80%). Scalp pilonidal sinus presented with subcutaneous nodules in 4 cases (80%). All were
treated by excision but one patient needed craniotomy.
Intermammary PNS presented with pain and discharge typically treated by excision and primary
repair. Perineal PNS presented with pain on defecation with discharge, treated with drainage
and left for secondary intention. Penile PNS presented with abscess formation treated by
excision and primary closure. Clitoris PNS presented with swelling and pain treated with excision
and primary closure. Prepuce PNS presented with swelling and discharge treated with excision
and primary closure.
DISCUSSION
Eryilmaz et al. identified the following risk factors for umbilical PNS: young age, male sex,
hairiness, and poor personal hygiene. The same team highlighted the most common presenting
symptoms as purulent discharge, umbilical mass, bloody discharge, and pain with frequency of
23%, 26% 69%, 100%, respectively (11). The current review found that discharge, swelling and
Pilonidal Sinus Journal (2017) 3(1), 8 - 15
Cite this Article as: Salih A, Kakamad F, Essa R. et al. "Pilonidal sinus of atypical areas: presentation and management".
PSJ (2017) 3(1): 8-15.
11
pain occur in 85%, only discharge in 11%, and bloody discharge in 2.3% of cases with umbilical
PNS. The key difference between umbilical PNS and sacro-coccygeal PNS is the method of
management.
In sacro-coccygeal PNS, conservative management has minimal role and the condition requires
excision (drainage if there is collection) with either off midline primary closure or daily dressing
having left the wound for the secondary healing (1). However, umbilical PNS can be successfully
treated by conservative measures provided that the hair is removed and daily dressings applied
(3-5,9-17). Removal of hair in umbilical PNS does not even need local anesthesia (3-5,8-17).
Sacro-coccygeal PNS is a recurring condition with recurrence rates of 15% being often reported,
while PNS in atypical areas (including umbilical) has lower recurrence rate as low as 2%
(11,21,33).
Six cases of umbilical PNS were reported to be recurrent. 4 cases were treated conservatively
(2% recurrence) and 2 cases (3% recurrence) underwent surgery (10,30). This suggests that both
conservative and surgical modalities may have the same rate of recurrence although this
conclusion is strongly rejected by some (11). A randomized controlled trial with high statistical
power is necessary to overcome this controversy.
While dealing with umbilical PNS, other pathologies such as Sister Joseph nodule, umbilical
hernia, endometriosis, urachal cyst, epidermoid cyst and pyogenic granuloma should not be
neglected as the differential diagnosis. Preoperative imaging may help in this regard (9).
Barber's disease is another name for PNS of the hand (6). While sacro-coccygeal PNS occurs
often in drivers, Barber’s disease occurs in hair dressers, sheep shearers, dog groomers and cow
milkers (6). It is reported more commonly among male hair dressers (6). Higher incidence of
Barber's disease among male gender might be attributed to more frequent female obsessive
personality regarding cleansing of interdigital webs (6) or that most barbers are male.
Scalp PNS is reported in teenagers and children ranging between 1 to 20 years with average 10
years of age. This differentiates scalp PNS from other types of PNS which tend to occur in older
adults (average 23 years old). Excision under local anesthesia is the most common modality of
treatment with no report of recurrences (2,25,26). Borges et al reported a scalp pilonidal sinus in
a 20-year-old male with intracranial extension which needed craniotomy under general
anesthesia. This may alert doctors to investigate scalp PNS before intervention searching for
intracranial extension. Plain antero-posterior skull x-ray may be the first investigation, followed
by brain Computed Tomography (2).
Perianal PNS occurs in adults older than typical PNS (average 34 years old) (31). Excision under
regional anesthesia is the usual therapeutic approach (1,29,30). Doll et al reported a 61-year-old
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PSJ (2017) 3(1): 8-15.
12
lady presenting with intermittent anal pain for 10 years due to endoanal PNS treated by opening
the cavity and leaving for healing by secondary intension (32).
Intermammary PNS presented with pain and discharge treated by excision and primary repair.
Perineal PNS presented with pain on defecation with discharge, treated with drainage and was
left for healing by secondary intention. Penile PNS presented with abscess formation treated by
excision and primary closure. Clitoris PNS presented with swelling and pain - treated with
excision and primary closure. Prepuce PNS presented with swelling and discharge - treated with
excision and primary closure (29,36-38).
As expected, PNS of atypical areas is a rare presentation. Umbilical PNS is the most common -
pain, swelling and discharge is the usual presentation. Apart from umbilical PNS which is mostly
managed conservatively, PNS of atypical sites is managed by excision and primary repair.
ACKNOWLEDGMENTS
N/A
MULTIMEDIA
N/A
CONTRIBUTION AND AFFILIATION
Salih AM1,
Kakamad FH2,3,
Essa RA3,4 ,
Aziz MS3,5,
Salih RQ3,6 ,
Mohammed SH3,6,
Saeed YA4,
Habibullah IJ4,
Muhialdeen AS4,
Othman S3,
Mohameed HA3
1. Faculty of Medical Sciences, School of Medicine, Department Surgery, University of Sulaimani,
François Mitterrand Street, Sulaimani, Kurdistan Region, Iraq
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Cite this Article as: Salih A, Kakamad F, Essa R. et al. "Pilonidal sinus of atypical areas: presentation and management".
PSJ (2017) 3(1): 8-15.
13
2. Faculty of Medical Sciences, School of Medicine, Department Cardiothoracic and Vascular
Surgery, University of Sulaimani, François Mitterrand Street, Sulaimani, Kurdistan Region, Iraq
3. Bioscience Center, Goran Street, Sulaimani, Kurdistan region, Iraq
4. Sulaimani Teaching Hospital, Sulaimani, Kurdistan region, Iraq
5. Daik laboratory, Goran Street, Sulaimani, Kurdistan region, Iraq
6. University of Sulaimani, Faculty of Science & Science Education, School of Science, Biology
Department, Raperin Street, Sulaimani, Kurdistan region, Iraq
Salih, Kakamad and Essa: substantial contribution to the concept and design, drafting the
manuscript and final revision and approval of the manuscript.
Aziz, Salih, Mohammed and Saeed: literature review, drafting the manuscript and final revision
and approval of the manuscript.
Habibullah, Muhialdeen, Othman and Mohameed: extraction of data, analysis of data, drafting
the manuscript and final revision and approval of the manuscript.
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... The fourth cause is pressure on atypical areas, such as intermammary area by pressure effect of breasts [8,16]. Reports of atypical PNS have increased in the last few decades [16]. ...
... The fourth cause is pressure on atypical areas, such as intermammary area by pressure effect of breasts [8,16]. Reports of atypical PNS have increased in the last few decades [16]. A systemic review by Salih et al. found that in more than 300 patients, there are 10 sites for atypical region for PNS to occur other than sacrococcygeal region [16]. ...
... Reports of atypical PNS have increased in the last few decades [16]. A systemic review by Salih et al. found that in more than 300 patients, there are 10 sites for atypical region for PNS to occur other than sacrococcygeal region [16]. ...
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Introduction: Pilonidal sinus (PNS) is a chronic inflammatory perianal disorder that rarely occurs outside sacrococcygeal region. The aim of this study is to report an extremely rare case of bilateral inframammary PNS with brief literature review. Case report: A 25-year-old female presented with a discharging sinuses in both inframammary region for two years. Examination showed multiple discharging sinuses with several centimeters of induration and tenderness. Under general anesthesia, complete excision of the sinuses with primary closure done. Histopathological examinations showed chronic foreign body granuloma surrounding hair shaft pictures consistent with PNS. Discussion: Inframammary PNS has never been reported in the literature. As with intermammary PNS, in this case also it is associated with obesity and large breasts with tight brassieres. Diagnosis is usually clinical. In contrast to sacrococcygeal PNS, operation under general anesthesia is main treatment modality. Conclusion: Pilonidal sinus of inframammary region is an extremely rare condition. High index of suspicion is required for diagnosis. Excision with primary closure is the definitive therapy.
... PSD occurs mostly in the intergluteal cleft or sacrococcygeal while rarely in axilla, umbilicus, submental area, nose, ear, finger and toe web spaces, groin, suprapubic area, clitoris, prepuce, penis, occiput, or female's mammary glands breasts. [1][2][3][4][5][6][7][8][9] To our knowledge, there is no reported case of PSD occurs in male mammary gland. In order to improve the cognition of PSD and make the correct diagnosis and treatment, We analyze the clinical data, related images, and some pathological features of a male breast PSD patient and reviews pertinent literature of PSD as well. ...
... Researcher also found that recurrent hair removal could cause initiating trauma that could trigger the development of PS. [21] PSD is now understood to be an acquired condition, It usually presents as a cyst, abscess, sinus tract, singly or in clusters. PSD is a common, perianal, recurring, inflammatory condition caused by hair penetrating to the outer layer of skin, [6] the incidence of PSD is 0.07% and account of 15% of perianal diseases, [22] it rarely occurs in other parts of the body. Shareef observed 12 patients with intermammary PSD, but all cases were female. ...
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Rationale: Pilonidal sinus disease (PSD) involving the breast is extremely rare and has not been described in man. Patient concerns: This current case report presents a case of a pilonidal cyst in a 46-year-old man which was surgically treated. He had intermittent pain in his left breast for 2 months and came for local rupture and discharge for 1 week. Diagnosis: The initial diagnosis is male mastitis, on the basis of the histological features of H&E-stained specimens and immunohistochemistry of the resected lump, this case was diagnosed as PSD. Interventions: The patient underwent "enlarged resection of the left breast lesion" under local anesthesia. Outcomes: The patient's surgical area healed well, without any signs of recurrence. Conclusion: PSD involving the breast is extremely rare in man, with no typically clinical manifestations, and could be easily ignored. This disease requires great attentions from clinicians.
... The usual area for pilonidal sinus disease is the sacrococcygeal region [3]. Although other rare areas have been reported like the umbilicus, axilla, groin, nose, interdigital web, neck, preauricular, postauricular prepuce, clitoris, suprapubic area, endoanal, penis, or occiput [4,5,6,7,8,9,10,11]. Intermammary PSD (iPSD) is an extremely rare variant of the condition with only 18 reported cases currently present in the English literature [1,12,13,14,15,16,17]. ...
... Nonoperative management has been proposed and practiced in the form of laser therapy, fibrin glue and paste injection [18]. Conservative management has not been advocated for atypical PSD (PSD occurring in areas other than the sacrococcygeal region) [4]. Local excision and primary closure is the standard reported therapy for iPSD. ...
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Introduction: Intermammary pilonidal sinus disease (iPSD) is a rare variant of a common condition. A case report is presented. Methods: A 22-year-old woman presented with recurrent iPSD 15 months after excision and open management. She was lost to follow up. Intermammary PSD literature is reviewed. Results: iPSD is rare. It affects obese women. Large breasts and a tight bra may be predisposing factors. Conclusion: iPSD should be managed along the same surgical principles as classical pilonidal disease.
... Barber is the most prevalent job among those patients complaining from PNS of areas other than Sacrococcygeal region [12]. Classically being a driver is one of the risk factors for developing PNS due to prolonged sitting [13]. ...
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Background Pilonidal sinus disease (PNS) is a disorder of the young population. The aim of this study is to analyze the risk factors associated with development of PNS in the secondary school students. Methods This case control study enrolled 189 participants, (Control group: 95 cases, case group: 94 cases). The inclusion criterion for the trial group was those secondary school students with PNS and age ranged between 16 and 20 years without the known risk factors of PNS. Result About 80 (42.3%) patients were male and 109 (57.7%) were female. Each group included 95 patients. There was no significant difference in both groups regarding basic features. Among the control group 36 (37.9%) participants were used to study while sitting on a hard place whereas among the case group 62 (66%) cases were used to study while sitting on a hard place, the difference was statistically significant (<0.001). Conclusion Sitting on the hard places could be regarded as a risk factor of developing PNS among secondary school students.
... In the case of intermammary pilonidal sinus, the presenting age of the female along with breast architecture can aid in supporting the diagnosis of pilonidal sinus. Pilonidal sinus is a disease of young patients with age around 15-30 years [13,16]. Regarding the age of affection, intermammary pilonidal sinus is consistent with other types of pilonidal sinus. ...
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There are controversies regarding the management of umbilical pilonidal sinuses. The current study aims to report on the efficacy of a non-operative, umbilical conserving procedure in the treatment of umbilical pilonidal sinus. This is a prospective, single cohort study. The cases were managed in a single private practice center. Patients were assessed and managed throughout the previous nine years (from January 2013 to June 2022). The required information was obtained from the center's medical database. The current study included 114 patients. There were 82 (71.9%) male patients. The patients' ages varied from 14 to 56 years (mean = 23.24). The umbilicus was retracted under local anesthesia. The sinuses were cleaned with povidone-iodine. Following the drying of the cavity, the mixture was put in the umbilicus, and the area was dressed. The amount of mixture was determined by the size of the cavity. Following treatment, the patients were directly discharged home with instructions to remove all hair from the chest and abdomen and keep the dressing dry for three days. After three days, the patients were advised to use a clean cotton swab to remove the injected mixture. Recurrence was reported in 5 cases (4.4%). The current technique might be used effectively in the treatment of umbilical pilonidal sinus. It is an umbilical preserving technique with a minimal recurrence rate.
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Introduction Pilonidal sinus (PNS) is an inflammatory disease arising from the sacrococcygeal region. The current study aims to report a case of pilonidal sinus developed in the posterior aspect of the auricle. Case report A 27-year-old male presented with a painless post auricular discharge for 2 weeks. it was diagnosed clinically as a case of pilonidal sinus. The opening sinus was anesthetized locally. The wound opened, the sinus excised through a small opening to remove the hair. The wound was injected with a prepared mixture and covered with the Kurdish gum. Discussion Although the etiology of pilonidal sinus is unknown, in the literature many factors have been described as risk factors. Positive family history particularly first-degree relative is an important risk factor with the incidence of 52.4%. Conclusion Although it is rare and atypical, PNS could occur in the post auricular region. It could be managed as sacrococcygeal PNS.
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Introduction Breast pilonidal sinus is an extremely rare variant of the condition, the report aims to present a case of pilonidal sinus occurring in the breast. Case presentation A 35-year-old female presented with a left breast discharging lesion for a 5-month duration. There was a local redness with multiple discharging sinuses on her left breast at 3-5 o`clock, 3-4 cm away from the nipple. The discharge was serosanguinous in appearance. The lesion was excised totally with primary closure under general anesthesia. Histopathological examination was consistent with pilonidal sinus. Discussion For atypical pilonidal sinus disease, surgery is the modality of choice but regarding sacrococcygeal pilonidal sinus, non-operative treatment begins to grow. The cardinal risk factors for recurrence and complications are male gender, family history, tobacco smoking, obesity, size of sinus, poor personal hygiene and surgical methods. Conclusion Breast pilonidal sinus is an extremely rare variant of the condition. It should be suspected on clinical examination. Surgical excision is the definitive treatment strategy.
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International Journal of Case Reports and Images (IJCRI) is an international, peer reviewed, monthly, open access, online journal, publishing high-quality, articles in all areas of basic medical sciences and clinical specialties. Aim of IJCRI is to encourage the publication of new information by providing a platform for reporting of unique, unusual and rare cases which enhance understanding of disease process, its diagnosis, management and clinico-pathologic correlations. ABSTRACT Introduction: Pyoderma gangrenosum is a rare neutrophilic dermatosis in childhood, of unknown etiology, characterized by ulcerations. Case Report: We report a case of a boy of 5 years, hospitalized for extensive phagedenic ulcers with a characteristic centrifugal extension for 30 months. The parents had consulted several health institutions without success. The lesions were localized on the left lower limb, to the medial aspect of the right leg and the right clavicular hollow. There was ankyloses of the right knee. The hemogram showed severe anemia. Bacteriological, mycological, viral samples were negative. Histology showed a rich infiltrate of polymorphonuclear neutrophils. No other underlying pathology was identified. The child was treated with prednisone 1 mg/kg/day in digressive manner over a period of two months with a full recovery at the end of this period. He received a blood transfusion and orthopedic surgery of his right knee, followed by rehabilitation sessions. The evolution was good leaving unsightly scars from the ulcers and a recovery of walking. Conclusion: The peculiarity of this case lies in the extensiveness of the lesions, the intensity of the anemia and the diagnostic difficulties due to the large and chronic cutaneous ulcerations of the child in Africa. This results in delay of diagnosis and threatens functionality in life.
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