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British Journal of Dermatology 02/2013; · 3.67 Impact Factor
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British Journal of Haematology 09/2012; · 4.94 Impact Factor
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ABSTRACT: IL-1 antagonists are used in the treatment of patients with rheumatoid arthritis and cryopyrinopathies. As yet anecdotal observations suggest that they may allow effective treatment of patients with different types of inflammatory skin disease. This review focuses on our current knowledge of the use of IL-1 antagonists in dermatology.
A Medline search was performed combining the keywords: "anakinra; canakinumab; rilonacept" AND "skin; neutrophilic dermatoses; Sweet syndrome; pyoderma gangrenosum; hidradenitis suppurativa; Schnitzler syndrome; Still disease". The precise dermatological phenotype of patients with IL-1 antagonist-responsive auto-inflammatory disorders was analysed in order to compare it to related complex disorders.
Double-blind randomized controlled trials have demonstrated the efficacy of these treatments in cryopyrinopathies with dermatological involvement including chronic infantile neurological cutaneous and articular (CINCA) syndrome, Muckle-Wells syndrome and familial cold urticaria. Anakinra is the only treatment for Schnitzler syndrome that is almost constantly efficacious, even in refractory disease, as attested by numerous case reports. It is also efficacious in the treatment of patients with adult-onset Still disease and systemic juvenile arthritis. Neutrophilic dermatoses constitute the cutaneous hallmark of IL-1-responsive auto-inflammatory disorders, and neutrophilic dermatoses could thus form an indication for this treatment. However, to date, only 9 reports have been published showing efficiency in patients with Sweet syndrome, in one case of neutrophilic panniculitis, and in two cases of pustular psoriasis. Anakinra appears less efficacious in patients with pyoderma gangrenosum.
IL-1 antagonists are a first-line treatment in patients with Schnitzler syndrome and cryopyrinopathies. They could become important alternatives in patients with acute and febrile neutrophilic dermatoses either unresponsive to or with contraindications to conventional treatments, but this requires confirmation by further clinical trials.
Annales de Dermatologie et de Vénéréologie 06/2012; 139(6-7):459-67. · 0.72 Impact Factor
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E-P Meyer,
D Heranney,
J Foeglé,
V Chamouard,
C Hernandez,
S Mechkour,
R Passemard,
M Berthel,
G Kaltenbach, D Lipsker,
D Christmann,
T Lavigne
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ABSTRACT: An outbreak of scabies occurred in the geriatric department of the Strasbourg University Hospital in September 2005. The index case presented with hyperkeratosic scabies, an extremely contagious form. The epidemic spread to several wards and pavilions and also contaminated healthcare staff and patient's families.
Our objective was to describe the outbreak, its progression, and the measures taken to eradicate it.
All healthcare workers, patients, and families affected in the outbreak were retrospectively studied, using medical prescriptions recorded by the hospital pharmacy, listings established by the occupational health department, and patient files.
Two epidemic waves were recorded, between August 31 and December 16, 2005, affecting 51 patients and staff members in the geriatric department, with a total of 58 episodes of scabies, seven of which were recurrences. Three main measures were taken to eradicate the epidemic: setting up of "contact" isolation precautions, information for the affected individuals, and treatment of the infected patients associated to mass treatment of contact cases. The mass treatment was widely applied, involving 490 patients and 592 caregivers. All of these measures successfully curtailed the outbreak in 3 months.
Rapid and radical action is essential to prevent extension of scabies within a community.
Médecine et Maladies Infectieuses 02/2011; 41(2):92-6. · 0.72 Impact Factor
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ABSTRACT: Since the year 2000, measles epidemics have occurred throughout Europe, with a number of severe cases in adults leading to hospitalisation. Herein, we discuss the case of a young adult male with measles presenting clinical and histological features of rash having a follicular nature.
An 18-year-old male consulted for diffuse, febrile morbilliform exanthem consisting chiefly of follicular papules surrounded by erythematosus macules. In the absence of any features suggesting possible contagion or implicated medication, biopsy was performed on one of these elements and showed strictly follicular involvement. Perifollicular lymphocytic inflammation was noted, with necrotic keratinocytes having monster nuclear cells, and above all, multinucleated cells within the outer root sheath and the sebaceous gland. Positive IgM measles serology was noted.
Follicular involvement is a very common feature in cases of measles that we have seen in recent years. In the present case, histological examination showed a follicular and sebaceous cytopathogenic effect, which while far less well known, has occasionally been seen with measles in the sweat glands or the epidermis, in which viral presence was detected. Thus, the skin rash was accounted for at least in part by the presence of the virus itself within keratinocytes and adnexal cells. This follicular involvement of measles, histologically confirmed in the present case, is frequently seen and can be helpful in differential clinical diagnosis with regard to other viral rashes. It could be caused by special affinity of the virus for body hairs.
Annales de Dermatologie et de Vénéréologie 02/2011; 138(2):111-5. · 0.72 Impact Factor
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British Journal of Dermatology 09/2010; 163(3):645-6. · 3.67 Impact Factor
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ABSTRACT: In this review we address the main cutaneous manifestations and diseases associated with deficiencies in components of the complement system. The first part is devoted to hereditary angioedema, in which acute, sometimes life-threatening recurrent attacks of acute swelling, usually associated with gastrointestinal symptoms, occur. It is related to a structural or functional deficiency of C1 esterase inhibitor. Patients usually have lowered C4 levels, and diagnosis relies on determination of antigenic and/or functional C1 inhibitor level. The second part focuses on lupus erythematosus, as deficiencies in early components of the complement system, such as C1q, C1r, C1s, C2 or C4, are the strongest known disease susceptibility genes for the development of human systemic lupus erythematosus. Severe infections early in life and marked photosensitivity in a patient with lupus erythematosus are clues to an underlying complement deficiency. The genetic background and the clinical associations of the different components of the complement system will be detailed.
Lupus 08/2010; 19(9):1096-106. · 2.34 Impact Factor
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ABSTRACT: Ehlers-Danlos syndrome (EDS) comprises a heterogeneous group of diseases involving genetic collagen fibre impairment. We describe a case of a patient presenting the rare type VIII, in which dermatitis ocre was associated with parodontal disease, and which was diagnosed late.
A 29-year-old man consulted for a pretibial ulcer present for seven years, resulting from a post-traumatic haematoma that had failed to heal. In view of the longiliner morphology, it had previously been diagnosed as Marfan syndrome. Subsequently, edentation was observed as well as "alveolar bone fragility". Examination revealed a marfanoid morphotype, a pretibial ulcer set within long-standing bilateral dermatitis ocre and papyraceous scars, but no joint hyperlaxity or cutaneous hyperelasticity. The diagnosis was consequently corrected to EDS type VIII.
Type VIII is a rare form of EDS, and the molecular mechanism is poorly understood. The involvement of parodontal connective tissue suggests impairment of collagen I and III proteins. It is important to identify this type of the disease since it involves parodontal disease for which early treatment is required in order to try to prevent edentation. The present case demonstrates the importance of diagnosis, which may be based upon appearance of bilateral dermatitis ocre from the age of 15 years associated with skin fragility. This sign is not part of the classical picture of Marfan syndrome, with which EDS type VIII is often confounded.
Annales de Dermatologie et de Vénéréologie 03/2010; 137(3):194-7. · 0.72 Impact Factor
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ABSTRACT: Photopheresis is a leucopheresis procedure in which cells are photoactivated by psoralen and then irradiated by ultraviolet A. We report four cases of women with refractory cutaneous lupus erythematosus (LE) who responded to this treatment.
We treated one patient with subacute LE having a contraindication to antimalarials and to thalidomide and three patients with chronic LE (lupus panniculitis, lupus tumidus and disseminated discoid LE) refractory to treatment with hydroxychloroquine, chloroquine, thalidomide and dapsone, and also, in some cases, to oral and intravenous corticosteroids, methotrexate, colchicine, acitretine, sulfasalazine, mycophenolate mofetil and intravenous immunoglobulin. Treatment consisted of two 4-hour sessions fortnightly. Only antimalarials were continued during photopheresis.
Photopheresis had a positive effect on all four patients. We noticed complete remission in two patients and interruption of progression followed by partial remission in the other two after a mean delay of two to three months of treatment. All treatments other than antimalarials were stopped.
Photopheresis appears to be an effective treatment option in patients with cutaneous LE. Due to its high cost, it should nevertheless remain an exceptional therapeutic option restricted to patients with cutaneous LE resistant to standard therapy.
Annales de Dermatologie et de Vénéréologie 12/2009; 136(12):861-7. · 0.72 Impact Factor
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Clinical and Experimental Dermatology 06/2009; 34(7):e295-6. · 1.20 Impact Factor
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ABSTRACT: Only about 30 cases of borrelial lymphocytoma (BL) with identification of the causative species of Borrelia have been published to date, mainly from Eastern or Central European countries.
To identify the species of B. burgdorferi complex responsible for BL in France.
Nine patients with BL acquired in France and for whom skin samples were sent to the national reference centre laboratory between 1994 and 2007 were included in this retrospective study. Direct detection of Borrelia in skin samples was made by polymerase chain reaction targeting the fla gene. Culture was performed when technically possible, and identification of each species was made by hybridization of a fragment of the fla gene with a panel of species-specific oligonucleotides.
Borrelia afzelii was identified in three cases, B. garinii and B. burgdorferi sensu stricto in one case each. Culture was positive in only one case (B. garinii).
Borrelia afzelii seems to be the predominant species of Borrelia responsible for BL in France, as already reported in other European countries.
British Journal of Dermatology 04/2009; 161(1):174-6. · 3.67 Impact Factor
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ABSTRACT: The term Facial Afro-Caribbean childhood eruption (FACE) was coined in 1990 to describe a perioral granulomatous eruption in black-skinned children. We present a typical case of this condition, together with a histological and immunohistochemical study.
A seven-year-old boy with black skin was referred for numerous nonpruritic papules that appeared on his face several months prior to the consultation. He had a past history of atopic dermatitis, but his mother denied any use of steroids or other topical treatment on his face. Examination showed numerous small lupoid and flesh-colored papules localised in the perioral, perinasal and periocular areas. Biopsy revealed a diffuse granulomatous infiltrate of the dermis comprising histiocytes, multinucleated giant cells and a heavy lymphocytic component. Histiocytes were CD68+ and CD1a-, while the majority of lymphocytes were CD3+ (70% CD8+ and 30% CD4+).
This particular type of perioral dermatitis is seen chiefly in male children with black skin. Our histologic study revealed a diffuse and massive granulomatous infiltrate of the dermis, in contrast with the small size of the papules. Expression of CD68, absence of necrosis and dense CD3+ infiltrate in this disease resembles findings in the granulomatous variant of rosacea. This entity must be differentiated from lupus miliaris disseminatus faciei and from sarcoidosis.
Annales de Dermatologie et de Vénéréologie 11/2008; 135(10):663-7. · 0.72 Impact Factor
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Clinical and Experimental Dermatology 08/2008; 33(4):518-9. · 1.20 Impact Factor
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ABSTRACT: Vulvar vestibulitis syndrome (VVS) is one of the most frequent causes of superficial dyspareunia in young women. VVS has a pronounced psychological impact. The results of pathological studies published thus far are controversial.
Fourteen women with VVS were included in this study and underwent vestibular biopsy. Vulvar biopsies were taken from the orifice of Bartholin's gland. The biopsy samples were stained with a standard stain and PAS and 25 serial sections were prepared for each specimen.
The mean patient age was 26 years and VVS had been present for a mean 30 months. Extensive inflammation of mononuclear cells was observed in the vulvar chorionic epithelium. This inflammation was seen mainly around the minor vestibular glands. Mild exocytosis of lymphocytes was noted in the vestibular glands and ducts.
Most studies concerning this disease report chronic inflammation of the vulvar vestibular mucosa. This inflammation is seen mainly around the minor vestibular glands. We report the same pattern in our study. Moreover, we observed some exocytosis into the epithelium of minor vestibular glands and the excretory duct. This aspect has not been reported to date, further supporting the individual nature of this entity.
Annales de Dermatologie et de Vénéréologie 06/2008; 135(5):367-72. · 0.72 Impact Factor
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ABSTRACT: Community-acquired cutaneous infections caused by methicillin-resistant Staphylococcus aureus (MRSA) are a growing concern. These bacteria may produce Panton-Valentine leucocidin potentially leading to necrotizing pneumonia. We studied the prevalence of MRSA and Panton-Valentine leucocidin in dermatology clinic outpatients in order to adapt therapy where possible.
This was a prospective study including all patients seen at a dermatology outpatient clinic between 1st March 2005 and 31st December 2006 and presenting mucocutaneous bacteriological samples. The main MRSA risk factors studied were frequent hospital consultations, hospitalization, antibiotic therapy within the last three months and community life. The following risk factors were also analysed, although less routinely: substance abuse, immunosuppression, diabetes mellitus, recent travel abroad and a history of similar lesions.
One hundred and twenty-two patients were included in the study and 235 samples (143 lesion samples and 92 nasal swabs) were carried out and S. aureus was isolated in 68 patients (56%). Twelve patients had MRSA (17.6%); seven of these were normal outpatients but five attended frequent hospital consultations (7.3%). MRSA resistance rates were as follows: 64% to ofloxacin, 36% to amikacin and erythromycin, 27% to fusidic acid, 9.1% to sulfamethoxazole-trimethoprim and 0% to pristinamycin. Community life was the only significant risk factor for MRSA in this study (p=0.045). Four of the 11 MRSA strains tested produced Panton-Valentine leucocidin.
Dermatologists are increasingly faced with cutaneous infections caused by community-acquired MRSA. Bacterial samples should be taken routinely and probabilistic antibiotic therapy for MRSA instituted in severe infections.
Annales de Dermatologie et de Vénéréologie 05/2008; 135(4):263-70. · 0.72 Impact Factor
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La Revue de Médecine Interne 03/2008; 29(10):825-6. · 0.61 Impact Factor
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ABSTRACT: Contiguous cutaneous inflammation is a circumscribed inflammatory reaction of the skin overlying a pathological process affecting an adjacent deep-seated anatomical structure. Contiguous cutaneous inflammation can sometimes reveal serious internal diseases. Through a case report and a literature review, we characterize the concept of contiguous cutaneous inflammation.
A 77-year-old woman with a past medical history of rhinorrhoea and chronic headache was admitted for a verrucous and erythematous lesion on her right cheek, evolving for two months with incomplete remission after numerous antibiotics. Histopathological examination was inconclusive. Facial CT revealed pansinusitis. Diagnosis of contiguous cutaneous inflammation to sinusitis was retained. Complete healing of cutaneous lesions was observed after surgical treatment of the sinusitis.
Contiguous cutaneous inflammation is characterized by localization over the causal disease, parallel evolution with the latter and the absence of clinicopathological specificity. The underlying pathological process is variable and may be tumoral or infectious, or simply the presence of an internal device. Migration of inflammatory cells and/or diffusion of mediators in a localized skin area could be involved in pathogenesis.
Annales de Dermatologie et de Vénéréologie 03/2008; 135(2):127-30. · 0.72 Impact Factor
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ABSTRACT: It has been suggested that the incidence of thin melanomas but not of thick tumours is rising in fair-skinned populations, although the reason for this discrepancy is not understood.
To describe temporal trends in melanoma epidemiology in a limited part of France in order to confirm this observation and to provide an explanation.
This is a retrospective population- and academic centre-based study in which all melanomas diagnosed in the department of the Bas-Rhin, France between January 1980 and December 2004 were included.
The study included 2094 melanomas diagnosed in 2020 patients. There was a steady increase in incidence of thin (< 1 mm) melanomas, mainly located on the trunk, and to a lesser extent in the head and neck region, in both sexes, and of intermediate (1-2 mm) melanomas in men. The incidence of intermediate melanomas in women and of thick (> 2 mm) melanomas, as well as mortality related to melanoma, remained stable. There was a steady decline of mean and median Breslow thickness. The 12 months median delay to diagnosis of thick tumours was significantly shorter than the 24 months delay to diagnosis of thin tumours.
Temporal trends suggest the existence of three unrelated types of melanoma: type I, thick melanomas, with stable incidence; type II, thin melanoma with a steady and important increase in incidence, mainly located on the trunk; and type III, melanoma with a slower increase in incidence, mainly located on the head and neck region.
British Journal of Dermatology 08/2007; 157(2):338-43. · 3.67 Impact Factor
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ABSTRACT: Leukocytoclastic vasculitis following Parvovirus B19 primary infection has occasionally been reported in children but it occurs rarely in adults. We present an original case report with severe renal complications.
A 33-year-old man presented with fever and eruption of the abdomen and members. Papules and vesiculopustules were associated with oral and genital ulcerations. These lesions subsequently became purpuric and necrotic. Histological analysis confirmed the diagnosis of pustulous leukocytoclastic vasculitis with IgA deposits. Laboratory investigations showed elevated sedimentation rate, hepatic cytolysis and renal impairment (hematuria, leucocyturia and proteinuria 1.5 g/24 hours). Anti-parvovirus B19 IgM were positive. Three months after the eruption resolved, IgM were undetectable while anti-parvovirus B19 IgG appeared. Renal injury progressively worsened: elevation of proteinuria (5 g/24 hours) and diminution of creatinine clearance (51 ml/min). Renal biopsy showed glomerulonephritis with mesangial IgA deposits. Major proteinuria persisted one year after the disappearance of dermatological lesions in spite of ACE inhibitor treatment.
The role of Parvovirus B19 has been suspected as an aetiological agent in many kinds of vasculitis, e.g. polyarteritis nodosa, Wegener's disease and leucocytoclastic vasculitis. In this case report, the detection of specific IgM and the absence of other factors associated with vasculitis are consistent with a causal role of Parvovirus B19. In previously published cases, the prognosis of parvovirus B19-associated vasculitis does not seem to differ from that of idiopathic vasculitis. To our knowledge, this is the first case exhibiting concomitant and persistent severe renal involvement.
Annales de Dermatologie et de Vénéréologie 03/2007; 134(2):160-3. · 0.72 Impact Factor
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ABSTRACT: Deficiencies in components of the classical pathway of complement activation are strong risk factors for lupus erythematosus (LE).Yet, it has not been addressed whether the conventional measurements of the serum hemolytic CH50 activity and antigenic concentrations of C3 and C4 are sufficient to asses a deficiency in C4A, C4B or C2 components, the most common deficiencies associated with LE.
In a retrospective series, we performed complement analyses in 35 patients with LE who were systematically screened for a complement deficiency. The majority of patients had cutaneous LE with mild systemic involvement and no complement consumption. Of 25 patients (72%) with complement deficiency we found 13 with a partial C4A deficiency, 2 with a complete C4A deficiency, 6 with a partial C4B deficiency, 2 with a complete C4B deficiency and 2 with a combined partial C2 and C4A deficiency.
The total complement activity (CH50) was decreased in only one out of two patients with complete C4B deficiency. CH50 level was found to be low-normal (35-38 U/ml(-1)) in one patient with partial C4B deficiency, one patient with complete C4B deficiency and both patients with combined partial C4A and C2 deficiency. Total C4 levels were normal in 9 out of 13 the patients with a partial C4A deficiency and in 2 out of 6 patients with a complete C4B deficiency. The antigenic concentration of C3 was low in only 1 patients with a complete C4B deficiency and within the normal range in all the others patients. Overall, 50% of the patients had normal or elevated C3, C4, and CH50 levels.
This study emphasizes that the usual measurements of CH50, C3 and C4 levels are not adequate to detect a C4 and/or C2 deficiency in patients with LE. In epidemiologic or investigative studies addressing the prevalence of complement deficiency, more elaborated diagnostic tests, such as C4 protein allotyping, C2 level measurement and genetic screening for type I C2 deficiency should also be performed.
Clinical Immunology 12/2006; 121(2):198-202. · 4.05 Impact Factor
