[Subcutaneous phaeohyphomycosis due to Exophiala spinifera in a renal transplant recipient]. / Phaéohyphomycose sous-cutanée à Exophiala spinifera chez une malade greffée rénale.

INTRODUCTION: Among the dematiaceous fungi responsible for human or animal phaeohyphomycosis, the Exophiala genus is a well-known etiologic agent and presently includes nine species considered as opportunist pathogens. To our knowledge, Exophiala spinifera has been reported as causative agent of only thirteen cases of cutaneous or systemic phaeohyphomycosis. We describe some typical phaeohyphomycotic cysts. CASE-REPORT: A 59 year-old female renal transplant recipient, treated with ciclosporine and prednisone, presented with two painless nodular and suppurative lesions of the leg, extending slowly. Histological and microbiological examinations identified Exophiala spinifera. The patient's condition improved with voriconazole treatment. DISCUSSION: Phaeohyphomycosis is a rare but cosmopolitan mycosis found throughout the world. Immunocompromised hosts are more vulnerable to these infections and more likely to develop severe and disseminated forms of uncertain outcome. Mycological and histological findings are important to confirm the diagnosis. The prognosis is benign and complete cure is common in cutaneous and superficial forms. Treatment is not well defined, often empirical and usually relies on antifungals and/or complete surgical resection.

[Lichenoid cutaneous reaction to imatinib]. / Toxidermie lichénoïde à l'imatinib (Glivec).

INTRODUCTION: Imatinib (Glivec) is a new therapeutic molecule used for the treatment of chronic myeloid leukemia. Cutaneous side effects with this treatment are common but lichenoid drug eruption is exceptional. We report the first case with profuse cutaneous lichen. CASE REPORT: A 52 year-old woman presented with a 5-year history of chronic myeloid leukemia. The different chemotherapies had failed (persisting polyadenopathies and splenomegalia). She was treated with imatinib (400 mg/day). Two months after the beginning of this treatment a disseminated cutaneous eruption appeared on the trunk, legs, arms and face without mucosal involvement and composed of dark purple, prurigenous, papules suggestive of lichen planus. The cutaneous biopsy confirmed the diagnosis of lichen planus. Suspension of the drug led to the complete regression of the eruption, without any other local treatment, within 2 months. Reintroduction of the drug led to the recurrence of the lesions. DISCUSSION: Cutaneous reactions to imatinib are common and occur in 11 to 67 p. 100 of patients depending on the series. More severe cutaneous reactions have been described: exfoliating dermatites, generalized pustulosis, epidermal necrolysis. The aspect of profuse lichenoid eruption of the skin and the correlation with cutaneous lesions clinically and histologically evocative of lichen planus has not been described other than an isolated buccal involvement in a 72 year-old woman. In our patient, study of the imputability criteria is in favor of imatinib's responsibility.

[Nail disease due to Scytalidium in Martinique (French West Indies)]. / Onychomycoses à Scytalidium en Martinique.

INTRODUCTION: Scytalidium is an endemic mold in tropical and subtropial areas. Our purpose was to study the prevalence and clinical and epidemiological features of onychomycoses due to Scytalidium in Martinique (French West Indies). PATIENTS AND METHODS: We performed a prospective study on 106 patients (46 men and 60 women) with clinical onychomycosis, in the dermatological department of the Centre Hospitalier Universitaire of Fort-de-France. All patients underwent mycological sampling and were divided into two groups depending on the presence or not of Scytalidium. Age, sex, localization, clinical aspects, time of duration and environmental factors (place of residence, garden, animals, bare foot walk, immunodepression) were compared between the two groups using chi2, Fisher and Student's t test. Ten control volonteers without clinical onycomycosis underwent mycological sampling. RESULTS: Onychomycosis due to scytalidium represented 42 p. 100 of patients (Scytalidium hyalinum in 91 p. 100 of cases) and 56 p. 100 after elimination of patients with negative results. Medium age was significantly higher in Scytalidium group (62 versus 54 years; p<0.02). Toe nail was involved in 95 p. 100 of patients (big toe nail in 77 p. 100). Sole involvement was more frequent in Scytalidium group (47 p. 100 versus 14 p. 100; p<0.001). Sampling of controls showed scytalidium in one case. DISCUSSION: Our study confirmed the endemicity of Scytalidium hyalinum in Martinique and the frequence of sole involvement. Presence of Scytalidium without clinical features in one control is of epidemiological interest, and may explain the frequence of the disease.

[Kimura's disease on the hard palate in a patient from Martinique]. / Maladie de Kimura à localisation palatine chez un Antillais.

INTRODUCTION: We report an observation of Kimura's disease (angiolymphoid hyperplasia with eosinophilia) in a Black patient from West Indies, in a rare localisation on hard palate. EXEGESIS: A 46-year-old male patient from French West Indies (Martinique) presented with a tumor of hard palate evolving from 2 years with locoregional lymph nodes. He had a history of chronic urticaria, prurigo, rhinitis and alopecia areata. Pathological examination of the tumor was consistent with the diagnosis of Kimura's disease. Blood tests showed hypereosinophilia, elevation of IgG and IgE. The serology of Toxocara canis with Western blott was positive. Corticosteroid therapy (0.7 mg/kg/d) and albendazole (800 mg/d) allowed regression of the tumor, lymph nodes, and urticaria. The remaining tumor was surgically excised. CONCLUSION: Kimura's disease was first described in Japanese young men, between 20 and 40 years old, with subcutaneous nodules on head and neck area. Involvement of oral mucosa is rare, and the occurence in West Indian patients has been rarely reported.

[Obesity persistent scleredema: study of 49 cases]. / Scléroedème persistant de l'obèse.

BACKGROUND: Buschke sclerodema is a very rare disease. Our objective was to show that persistent scleredema is frequent in certain group of patients at risk. PATIENTS AND METHODS: We studied 49 patients, diagnosed between 1995 and 1999 in dermatology, pneumology and endocrinology departments in Martinique. Diagnosis was performed on classical clinical and histopathological aspects of sclerodema. Data studied were age, sex, mode of occurrence, clinical and histopathological aspects and associated diseases. RESULTS: The 49 patients presented with cutaneous infiltration of the upper part of the trunk, with thick dermis and large collagen bundles on histopathological examination. Forty-two had mucoid substance deposition, stained with Alcian Blue (this criteria was considered as inconstant by most authors in the literature). Sex ratio H/F was 0.06 (93 p. 100 females). Mediam age at onset was 50 years ranging from 20 to 79 years. The occurrence was insidious in 97 p. 100 of cases. All patients had neck and nuchae involvement. The disease involved the back in 93 p. 100, upper limbs in 50 p. 100 and lower limbs and face in 43 p. 100 of patients. Fifty-six percent of patients had limitation of shoulder movements, 16 p. 100 limitation of mouth opening, 20 p. 100 limitation of eyelid opening, 36 p. 100 had myalgia, 73 p. 100 had pruritus and 66 p. 100 had dyspnea. Obesity was present in 95 p. 100, diabetes in 79.5 p. 100, elevated blood pressure in 81.5 p. 100 and monoclonal dysglobulinemia in 46 p. 100 of patients. Twenty-five patients had a polysomnography showing severe obstructive sleep apnea syndrome. DISCUSSION: The large number of patients in our study can be explained by the search for sclerodema in patients with obesity, diabetes and high blood pressure. The disease is usually unknown by patients and physicians unless a systematic examination is performed. Association with obstructive sleep apnea syndrome was not previously reported and a larger study is ongoing.

Familial aggregation of vitiligo in the French West Indies (Isle of Martinique).

Environmental factors are thought to influence the genetic transmission of vitiligo, and may change in families of different extraction. Most studies on vitiligo have been performed in occidental countries or in India, therefore, our purpose was to study the familial vitiligo aggregation in Martinique (French West Indies). Data on 16 families were collected from 1995 to 1999. Information was compared to 36 controls affected with sporadic vitiligo, using the chi(2) test. The prevalence among relatives of patients was 7%, as compared to 0.34% in the general population (p < 0.001). The age of onset of vitiligo was 31 in family cases and 33 in controls. Vitiligo occurred before the age of 20 in 19% of family cases and in 36% of controls. Most families (75%) have no more than 2 affected members. No difference was observed in triggering and environmental factors in family cases and controls. Our observations are in agreement with the literature data, leading to the assumption that the environmental factors involved in the expression of vitiligo in our island do not differ from other areas.

[Panniculitis induced by MINE chemotherapy]. / Hypodermite secondaire à une chimiothérapie de type MINE.

BACKGROUND: Drug-induced panniculitis are uncommon. We report the second case of panniculitis induced by MINE chemotherapy. CASE REPORT: A 31-year-old woman with relapsed Hodgkin disease was treated with MINE cytostatic regimen. Multiple erythematous and painful nodules of panniculitis developed on her chest, abdomen and thighs fifteen days after the beginning of drug administration with a second flare up after second administration of the same drugs. The eruption cleared slowly after treatment withdrawal. DISCUSSION: To our knowledge, our case is the second reported case of panniculitis induced by MINE chemotherapy. Drug-induced panniculitis is uncommon and usually induced by steroid treatment. Some cases of panniculitis induced by atenolol, potassium bromide, apomorphine, interferon alpha and interleukin 2 have been described. Few cutaneous adverse effects are reported with MINE chemotherapy: rash, erythema and swelling of extremities. A case of inflammatory swelling of thighs with hemorrhagic panniculitis due to this treatment has been described recently.

High prevalence of vitiligo in lepromatous leprosy.

BACKGROUND: Leprosy and vitiligo are common affections in the West Indies. Vitiligo frequently occurs in lepromatous patients, an observation rarely reported in the literature. METHODS: We studied the prevalence of vitiligo in patients affected by leprosy by performing a retrospective study between 1978 and 1999 in the French West Indies (Martinique). RESULTS: Eleven patients presented with vitiligo among 101 with lepromatous (multibacillary) leprosy. None presented with vitiligo among the 364 with the tuberculoid (paucibacillary) form. The mean age of the vitiligo patients was 55. 4 years at vitiligo onset. The sex ratio was 0.8. Vitiligo occurred 19 years after the diagnosis of leprosy, with a range from 3 to 42 years. The prevalence of vitiligo in lepromatous patients was 10.9%, compared to 0% in tuberculoid patients. Such an increase in prevalence compared with that in the general population (0.34%) was shown to be highly significant (P< 0.0001). CONCLUSIONS: Our data confirmed that the association of vitiligo and leprosy was not fortuitous. The physiopathology leading to this high rate of vitiligo in lepromatous leprosy is unclear, despite the fact that autoimmunity plays a major role in both diseases.

[Microsporum canis mycetoma of the scalp]. / Mycétome à Microsporum canis du cuir chevelu.

BACKGROUND: Mycetoma is a chronic subcutaneous tumefaction with presence of grains or granules. Etiological agents include bacteria or filamentous fungi. Mycetoma due to dermatophytes is uncommon, mainly occurring in Africa. To our knowledge, no case has been reported in the West Indies. Only two observations of Micosporum canis mycetoma in humans have been reported in the literature. We report a third case of mycetoma of the scalp caused by this fungus. CASE REPORT: A 22-year-old woman from Martinique, French West Indies, presented with an indolent tumefaction of the scalp evolving over five years. She had mental retardation due to congenital adrenal hyperplasia with 21-hydroxylase deficiency. The lesion was extracted surgically. Pathology and mycology examinations showed features of Microsporum canis mycetoma. Two months later, the scalp lesion recurred and the patient was treated with griseofulvin after surgical extraction. DISCUSSION: Mycetoma due to dermatophytes is very uncommon, mainly observed on the scalp and nape of the neck. A history of a skin lesion is frequent, leading to transcutaneous penetration of the fungus and mycetoma formation. Several dermatophyte species have been identified as causal agents (Microsporum ferrugineum, Trichophyton rubrum, Trichophyton verrucosum, Trichophyton mentagrophytes, Microsporum audouinii, Microsporum langeronii). Microsporum canis is rarely demonstrated in humans: two cases in children in Africa and Australia. Our observation was similar to the two cases in the literature: indolent and mobile tumefaction of the scalp, in a child or young adult, suggestive of lipoma or epidermal cyst, with excision leading to diagnosis. Association with tinea capitis and skin or nail involvement can also be observed.

Epidemiology of vitiligo in the French West Indies (Isle of Martinique).

BACKGROUND: The frequency of vitiligo in white populations has been generally estimated to be about 0.5-1%. The same prevalence is expected in black populations, despite the few investigations reported. No studies have been performed in black populations living in the Caribbean Islands. Therefore, our purpose was to report an epidemiologic study of vitiligo in the French West Indies (Isle of Martinique). METHODS: We performed a prospective study between October 1995 and March 1996; 2077 outpatients of the Department of Dermatology at the Fort de France University Hospital were examined to detect vitiligo. Concurrently, 32 patients (23 women and nine men), presenting with vitiligo, were questioned about their family history, personal diseases, age, and circumstances of vitiligo occurrence. RESULTS: Vitiligo was found in seven patients (five women and two men) out of 2077. The prevalence in the studied population was 0.34%. Of the 32 patients with vitiligo who were investigated, 11 (34%) had a family history of vitiligo, two (6%) suffered from thyroid disease, two (6%) from psoriasis, and one (3%) from atopic dermatitis. The median age at vitiligo onset was 29 years. CONCLUSIONS: Despite the bias due to the recruitment of patients in the Dermatology Department, this study demonstrates a prevalence in a black population comparable, or slightly inferior, to the currently accepted data in white people. Our results concerning the age of onset and pathologic associations showed no difference with the literature data related to white populations.

Epidemiology of histoplasmosis in the French West Indies (Martinique).

The Caribbean islands are presumed to be an endemic zone for Histoplasma capsulatum infection, but no epidemiological studies have been done in this area. Our purpose was to report the epidemiology of histoplasmosis from 1991 to 1997 in the French West Indies (Martinique). Cases identified from the register of the mycology laboratory were analysed retrospectively. Ten cases (9 male and 1 female) were identified; 8 of the patients were infected with HIV (average T4 lymphocyte count in these 8 patients was 32/mm3). Eight patients had cutaneous involvement. The incidence in AIDS patients was 1.7%. The annual incidence in the general population was 0.34/100,000. Our data showed that histoplasmosis is endemic in Martinique, with an incidence in AIDS patients slightly inferior to that in endemic areas of the USA. The high rate of cutaneous forms (80%) is uncommon.

[Cutaneous sensitivity to histoplasmin in a Martinique hospitalized population]. / Sensibilité cutanée à l'histoplasmine dans une population hospitalière martiniquaise.

OBJECTIVE: Histoplasma capsulatum infection is considered to be endemic in the West Indies. Nevertheless, few epidemiologic studies have been conducted in this area. The histoplasmin skin test reflects the frequency of asymptomatic forms of histoplasmosis. We studied the prevalence of positive skin tests in a population of the French West Indies (Martinique). MATERIALS AND METHODS: Forty one patients (24 females and 17 males), age range 29 to 90 years, were tested for histoplasmin skin sensitivity between August and October 1997, in the department of dermatology of Fort de France (French West Indies). Patients with immunosuppression or personal history of histoplasmosis were excluded. RESULTS: Five patients had a positive skin test (12 p. 100). No significative association was found between a positive skin test and diabetes, rural occupations or exposure to bats. DISCUSSION: Despite the small number of cases, related with difficulties in obtaining histoplasmin, our study showed a sensitivity level similar to medium endemic areas of the USA. The positive skin test rate is much higher than the rate reported before in West Indies, in a sample of the population under 25 years of age.

Epidemiology of cutaneous melanoma in the French West Indies (Martinique).

The epidemiology of melanoma in populations of African-European descent has rarely been reported. The authors studied melanoma in the French West Indies (Martinique), where black Caribbeans and whites represented 96% and 4% of the population, respectively. Among the 85 cases of melanoma collected from 1976 to 1995, blacks represented 75% and whites, 25%. The average incidence rates were 1.48 and 0.9 per 100,000 per year in females and males, respectively. The sole of the foot represented 72% of the primary sites in blacks. Breslow's tumor thickness was > 1.5 mm in 68% of the cases. The 5-year survival was 44%.

EGF receptor expression and growth of psoriatic and normal human keratinocytes are modulated by 1.25 (OH)2-vitamin D3 ex vivo.

Calcitriol or 1.25 (OH)2-vitamin D3 is used in the treatment of psoriasis as an inhibitor of cell proliferation. We studied the action of calcitriol ex vivo on the growth of psoriatic and normal human keratinocytes, and on the expression of the EGF receptor. Third passaged normal and psoriatic keratinocytes were seeded (10(4)/cm2) in 24-well dishes in serum-free medium (MCDB supplemented with amino acids, with either 0.1 or 1.1 mM of calcium) and 10(-9) M calcitriol. When subconfluence was reached, cell counts and 125I-EGF binding studies were performed. Cell counts showed at least a 50% decrease in growth under all conditions studied (normal or psoriatic keratinocytes; 0.1 or 1.1 mM calcium) when calcitriol was added. 125I-EGF binding studies showed a decrease in total receptor numbers in the presence of calcitriol with acceleration of binding at low concentrations of 125I-EGF. Scatchard plot analysis showed only one type of high affinity receptor. Receptor sites were decreased (30% to 40% of controls) in the presence of calcitriol together with a decrease in the dissociation constant. In conclusion, at almost physiological concentrations ex vivo, calcitriol strongly decreased normal and psoriatic keratinocyte growth. This potent antiproliferative effect could in part be explained by the capacity of calcitriol to downregulate EGF receptor expression.

Histiocytic sarcoma that mimics benign histiocytosis.

A 28-year-old man presented with a histiocytic sarcoma of a very uncommon origin, as it had developed for several years like a benign cutaneous histiocytosis resembling generalized eruptive histiocytoma before becoming acute, with nodal and massive pulmonary involvement. Despite various chemotherapies, the patient died within 8 months. Skin biopsies showed histiocytic proliferation in the dermis and node biopsies showed histiocytic proliferation with a sinusoidal pattern. Immunohistochemical analysis, performed on paraffin-embedded sections, demonstrated strong labeling of tumoral cells for CD68 and moderate labeling for CD3 and CD4. CD30 labeling was negative. S-100 protein was positive on a Langerhans' cell reactive subpopulation. Electron microscopy confirmed the histiocytic nature of malignant cells and showed cytoplasmic inclusions such as regularly laminated bodies, dense bodies and pleomorphic inclusions. No Birbeck granules were seen. A gene rearrangement study of T-cell receptor gamma and immunoglobulin heavy chain genes showed a germline configuration. Histiocytic sarcoma is an extremely rare true histiocytic malignancy, the existence of which has been recently debated since it has often been mistaken in the past for large cell lymphomas. Such a deceptive onset as benign cutaneous histiocytosis has not been described in the literature to our knowledge.

Psoriatic onycho-pachydermo-periostitis. A variant of psoriatic distal interphalangeal arthritis?

BACKGROUND: Joint and nail involvement in psoriasis is relatively common. In contrast, bony involvement of the terminal phalanx under a psoriatic nail is rare. We report on this psoriatic onycho-pachydermo-periostitis and suggest pathophysiologic mechanisms. OBSERVATIONS: Two new cases are reported and the data are compared with the data from eight similar cases reported in the literature. All subjects presented with similar changes--onychopathy, soft-tissue thickening, and radiologic features consisting of bone erosions and a periosteal reaction of the terminal phalanx. Of the 10 patients, two had no history of psoriasis before a diagnosis of psoriatic onycho-pachydermoperiostitis was made. The inflammation is likely transmitted from the psoriatic nail to the adjacent underlying bone by the same mechanism as in enthesopathies. CONCLUSION: Psoriatic onycho-pachydermo-periostitis should be recognized as a specific entity in the spectrum of psoriatic disease.