Phaeohyphomycosis Caused by a Novel Species, Pseudochaetosphaeronema martinelli.

Among the opportunistic mycoses that are emerging in patients with immunosuppression or severe underlying illness, many isolates lack of characteristic sporulation and until recently could not be identified. Clinical signs are mostly nonspecific and therefore such infections have often been disregarded. In the present paper we describe a novel, nonsporulating fungal species causing subcutaneous phaeohyphomycosis in two patients of different origin. One is a 73-year-old female from Martinique who suffered from rheumatoid arthritis, while the other case concerns a 72-year-old male from Mexico who had a history of type 2 diabetes mellitus. Sequencing of the partial ribosomal operon revealed that in both cases a member of the order Pleosporales was concerned which could not be affiliated to any family within this order. Multilocus analysis revealed that the fungus was related to another, unaffiliated agent of human mycetoma, Pseudochaetosphaeronema larense, and therefore the name Pseudochaetosphaeronema martinelli was introduced.

[Pyoderma gangrenosum and neutrophilic aortitis preceding the development of rheumatoid arthritis]. / Pyoderma gangrenosum et aortite neutrophilique précédant l'apparition d'une polyarthrite rhumatoïde.

BACKGROUND: We report a case of pyoderma gangrenosum (PG) associated with a complication comprising ascending aortic dissection (neutrophilic aortitis) in a setting of rheumatoid arthritis (RA). PATIENTS AND METHODS: A 79-year-old female patient was hospitalized in late 2009 for vegetating PG. Treatment with general steroids followed by colchicine and topical steroids resulted in complete healing of skin lesions. During hospitalization, the patient presented dissection of the ascending part of the aorta, for which emergency surgery proved effective. Histological examination of the excised tissue revealed diffuse neutrophilic aortitis. Diagnoses of Takayashu's disease and of lupus were ruled out. A chest CT scan showed interstitial lung disease with mild lymphocytosis in the bronchoalveolar fluid, but with no isolated pathogenic organisms. Relapse of skin lesions occurred 3 and 4 years later, associated with the development of RA, and worsening of the interstitial lung disease was noted in a scan carried out it in 2013, following which stabilization was observed in April 2014. There was no recurrence of the PG lesions. CONCLUSION: To our knowledge, no other cases involving association of neutrophilic aortitis with PG and RA has been published to date. The literature describes the emergence of the concept of systemic neutrophilic dermatoses, and this notion is reinforced by the presence of a cutaneous and aortic site of the neutrophilic disease in a single patient.

[Incidence and characteristics of calciphylaxis in Martinique (2006-2012)]. / Incidence et caractéristiques de la calciphylaxie en Martinique (2006-2012).

BACKGROUND: Calciphylaxis is a rare and severe disease with an annual incidence of around 1 % in dialysis patients. The main study aim was to determine its incidence in Martinique, where there is a significant population of patients on dialysis. PATIENTS AND METHODS: All patients diagnosed with calciphylaxis between 2006 and 2012 and living in Martinique were included, retrospectively. Social, demographic, biological, anatomic, pathological, histological and outcome data were analysed. RESULTS: Fifteen patients were included (8 women, 7 men). The incidence of calciphylaxis in this population was about 4.62/1,000,000 inhabitants per year. All patients presented very painful skin ulcerations and necrosis, chiefly on the lower extremities in 53.3 % of cases. All patients were on haemodialysis and two had undergone renal transplantation. Fourteen of the 15 patients were presenting secondary hyperparathyroidism, 12 had hypertension, 9 peripheral arterial disease, 8 obesity and 8 diabetes mellitus. Raised calcium and phosphorus were noted in 8 patients, with hypoalbuminaemia in 9 patients. Treatment with sodium thiosulfate was given for 8 patients, and was beneficial for all after a mean duration of 3.4 months. After 6 months of follow-up, 8 of the 15 patients were cured, 1 showed improvement and 6 had died. CONCLUSION: To our knowledge, this is the first study to examine the incidence of calciphylaxis in the general population. The relatively large number of patients could be accounted for by the high number of comorbidities in end-stage renal disease patients in Martinique, including obesity, diabetes, hypertension and arteritis. Treatment with sodium thiosulfate was beneficial for 8 patients.

[Diagnosis of skin cancer by dermatologists in the French West Indies: a prospective study]. / Étude prospective de l'incidence des cancers cutanés dépistés en pratique dermatologique aux Antilles françaises.

BACKGROUND: The incidence of skin cancer is not well established in the French West Indies, aside from old data concerning cutaneous melanoma. METHOD: A prospective study was performed over a 3-month period in late 2007 in the French West Indies. The number of new cases of histologically confirmed skin cancer was determined using a questionnaire. RESULTS: The rate of participation of dermatologists was 82 %. During the study period, 166 skin cancers were diagnosed in 134 patients (66 women and 68 men: mean age=63.3 years, SD=2.5), including 137 basal cell carcinomas, 12 melanomas, seven squamous cell carcinomas, six Bowen's disease, one B lymphoma and one Paget's disease. The raw incidence of skin cancers detected by dermatologists was calculated as 64.6 cases/100 000 inhabitants/year for basal cell carcinoma, 5.7 cases/100 000 inhabitants/year for melanoma, 3.3 cases/100 000 inhabitants/year for squamous cell carcinoma, and 2.8 cases/100 000 inhabitants/year for Bowen's disease. Seven melanomas (almost exclusively of plantar topography) and seven basal cell carcinomas were diagnosed in patients of phototype V or VI. DISCUSSION: Although lower than in metropolitan France, the number of skin cancers diagnosed by dermatologists in the French West Indies is fairly high. In addition, this study indicates the significant incidence of basal cell carcinomas and melanomas in subjects with phototype V or VI, underreported in the literature. These findings highlight the need to begin longer-term studies and to include skin carcinomas in the cancer registry of the French West Indies in order to better identify their characteristics among the Caribbean population.

[Occupational exposure to syphilis during venous ulcer curettage in Martinique]. / Accident professionnet d'exposition à a syphilis a près curetage d'ulcere veineux en Martinique.

The purpose of this report is to describe the first case of occupational exposure to syphilis. This accident occurred during curettage of a venous ulcer in a 68-year-old woman in Martinique Island. The patient presented primary syphilis without HIV. This case points out the need for enhanced syphilis screening in epidemic areas.

[Publication rate for oral presentations made at the Journées Dermatologiques de Paris meeting]. / Publication des communications orales présentées aux << Journées dermatologiques de Paris >>

BACKGROUND: The fate of oral presentations presented at a dermatological meeting has not been assessed to date. Our aim was to determine the publication rate for oral presentations presented at the "Journées dermatologiques de Paris" (JDP) meeting in peer-reviewed journals. Our secondary goals were to identify factors associated with publication and to examine the consistency of reporting of research findings presented in the conference abstract and subsequent full publication. METHODS: Abstracts presented orally at the JDP 1999-2004 were identified in the book of congress abstracts. Two independent operatives performed a Medline search cross-referencing lead and last authors and keywords. RESULTS: The publication rate was 57.6% with mean time to publication of 20.3months. The median impact factor was 2.8. Factors associated with subsequent publication were study topic (p=0.04 for oncology) and study type (p=0.03 for fundamental research and p=0.005 for randomized controlled trials). The congress abstracts and full-text publication differed primarily in terms of sample size and data given in the "Results" section. DISCUSSION: More than half of all abstracts presented orally at the JDP congress are subsequently published in journals with a median impact factor comparable to those seen for other scientific congresses for which similar analysis has been conducted. These results confirm the scientific quality of this particular congress, in addition to its vocation of continuous medical training.

[Ulcerated nodular lesions on extremities of patient undergoing treatment for glioblastoma on French Caribbean island of Martinique]. / Lésions nodulaires et ulcérées des membres chez un Martiniquais traité pour un glioblastome. Phaéohyphomycose à Exophiala spinifera.

Phaeohyphomycosis comprises a disparate group of cutaneous and systemic mycotic infections caused by dematiaceous fungi. The Exophiala genus includes more than 20 species. The purpose of this report is to describe a case of phaeohyphomycosis due to Exophiala spinifera in a patient under treatment for cerebral glioblastoma on the French Caribbean island of Martinique. A 73-year-old man who was undergoing treatment for a cerebral glioblastoma using radiotherapy, chemotherapy and systemic corticosteroids presented with ulcerated nodular lesions on all four extremities for several months. Histological and mycological examinations led to diagnosis of cutaneous phaeohyphomycoseis due to Exophiala spinifera. Treatment using itraconazole was initiated but the patient died from complications of glioblastoma. Phaeohyphomycosis is a rare and cosmopolitan mycosis. Infection due to Exophiala spinifera is uncommon in humans. It usually but not always occurs in immunocompromised hosts. Although cutaneous involvement is usually localized and has a favorable prognosis, disseminated forms may occur and can be life-threatening. Histological and mycological examinations are useful to confirm the diagnosis. Systematic antifungals with or without surgical resection are often effective. To our knowledge, only 15 reported cases of phaeohyphomycosis due to Exophiala spinifera have been reported up to now.

[Clustered cases of histoplasmosis due to Histoplasma capsulatum var. capsulatum in Martinique: Case reports and environmental investigation]. / Cas groupés d'histoplasmoses à Histoplasma capsulatum var. capsulatum à la Martinique : description des cas et enquête environnementale.

Between the 24th of June and the 6th of July 2005, nine men came to Fort-de-France emergency department (Martinique, French West Indies) with more or less pronounced pulmonary symptoms associated in two cases with skin lesions. Three weeks before these nine men performed work in a deserted house. The diagnosis of histoplasmosis was based on pulmonary sample mycological analysis (direct examination and culture), molecular biology and serological tests. Interrogatory and environmental investigations on the presumed place of exposition to H. capsulatum var. capsulatum spores allowed confirming how and where contamination took place.

[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.

Diffuse cutaneous infection caused by a presumed monoxenous trypanosomatid in a patient infected with HIV.

A patient infected with human immunodeficiency virus developed a diffuse cutaneous nodular syndrome. The parasite isolated from a skin nodule was studied by isoenzymatic characterization and transmission electron microscopy of both culture forms and those in the patient's skin biopsy. The parasite's ultrastructure was that of a typical member of the family Trypanosomatidae, but it differed isoenzymatically from all 'new and 'old World' species of Leishmania, Trypanosoma and Sauroleishmania. We believe that it was a (presumably) monoxenous 'lower trypanosomatid.

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.

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.

[Epidemiological characteristics of HIV infection in Martinique]. / Caractéristiques épidémiologiques de l'infection à VIH en Martinique.

The epidemiologic study of HIV infected patients in Martinique from 1985 to 1992 allowed to point out a stability of new cases by year, and confirm the heterosexual transmission in this area. The sexual comportment of Martinicans seems to be different of the continental French people and different between men and women. This constation is important to know for the preventive strategy of the infection.

[Acute disseminated encephalomyelitis preceding cutaneous lupus]. / Encéphalomyélite aiguë disséminée précédant un lupus cutané.

STUDY DESIGN: A case of acute disseminated encephalomyelitis (ADEM) preceding the features of cutaneous lupus is reported. OBJECTIVES: To suggest that ADEM and cutaneous lupus are pathophysiologically related. SUMMARY OF THE BACKGROUND DATA: Neurological complications of systemic lupus erythematosus are common. However, demyelinative central nervous system manifestations are rare, and restricted to neuromyelitis optica (NMO). NMO is thought to be a partial form of ADEM. METHODS: A clinical, neuroradiological and immunological study was performed in this case. RESULTS: ADEM was diagnosed in a young black female. Three years later, she developed a cutaneous lupus without hypocomplementemia. CONCLUSION: A relationship between ADEM and cutaneous lupus is suggested. The normal serum complement level in our patient might be a predisposing factor for the development of ADEM.

[Leprosy in young children. 2 cases at 3 years of age]. / La lèpre chez le jeune enfant. Deux observations à l'âge de 3 ans.

Two children (age 3 years) presented with leprosy in endemic area: one was type T (tuberculoid paucibacillary); the other was type BL (borderline lepromatous) considering a very fast evolution, and was treated as a multibacillary leprosy. The family of the second child reported that the onset of the disease was consecutive to an insect bite at 5 1/2 months of age. Such observations remind us that leprosy must be suspected or thought about very early since the incubation period is likely to be shorter than previously proclaimed.

[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.

[Progressive macular confluent hypomelanosis in mixed ethnic melanodermic subjects: an epidemiologic study of 511 patients]. / Hypomélanose maculeuse confluente et progressive du métis mélanoderme: étude épidémiologique sur 511 sujets.

INTRODUCTION: The pathogenesis of the recently described (1985) entity macular confluent progressive hypomelanosis in black subjects of mixed ethnic origin, also called creole dyschromia is unknown. Patients are generally black adults of mixed ethnic origin and present with hypopigmented maculae located asymmetrically in unexposed areas. The mechanism appears to be a phenotypic modification of produced melanosomes. The cases published to date do not provide clear epidemiological data. MATERIALS AND METHODS: We took histories and examined 511 patients in the French West Indies (Martinique) during systematic screening for leprosy. Observations included presence or absence of creole dyschromia, the intensity of the depigmentation. History reports included chronology of the lesions and factors affecting disease course. RESULTS: One-hundred twenty-one cases of dyschromia were identified, often with few clinical signs. Creole dyschromia was found in one-third of the examined subjects between the age of 17 and 48 years and appeared to be more exception outside this age range. More men than women were found to have the disease and the duration of the clinical course was about 25 years. Clearer skin appeared to be more sensitive and only responded to intermittent exposure to sun. DISCUSSION: Due to the fact that the examination was mandatory, it was possible to identify a large number of cases unknown to dermatologists and sometimes to the subject himself. The clinical description corresponded to those given in the literature, but the higher frequency in males, the duration of the clinical course and the sensitivity of clearer skin appear to have been unreported to date.