RESUMO
BACKGROUND: Kimura's disease is a rare benign chronic inflammatory disease of unknown aetiology. CASE REPORT: An 18-year-old atopic Brazilian patient consulted for recurrent facial eczema that improved slightly under topical corticosteroids. He presented a large infiltrated and eczematous subcutaneous plaque on the right supraorbital area, together with eyelid oedema. This was associated with complete right eyebrow alopecia, oedema of the upper lip and infiltrated facial papules. The tests showed hypereosinophilia (4000/mm(3)), a high IgE level (3786 kIU/L) and proteinuria (0.3g/24h). Histological examination revealed a lymphocytic eosinophil-rich inflammatory infiltrate in the superficial and deep dermis, with some lymphoid follicles in depth and proliferation of post-capillary venules. All of these elements led to the diagnosis of Kimura's disease. DISCUSSION: We report an unusual clinical form of Kimura's disease in a Brazilian patient. This disease has been classically described in young Asian men. In our case, a particularly large infiltrated and oedematous subcutaneous plaque was noted. In fact, the more common forms appear as subcutaneous nodules on the head and neck, which may be associated with locoregional adenopathy, involvement of the salivary glands, hypereosinophilia and raised serum IgE. In our case, the skin lesions, characterized by a large infiltrated and oedematous subcutaneous plaque, were unusual and could evoke IgG4-related disease. CONCLUSION: We report a case of Kimura's disease with an atypical presentation. The diagnosis was established by comparing certain histopathological features, and the possibility of IgG4-related disease was discussed.
Assuntos
Hiperplasia Angiolinfoide com Eosinofilia/diagnóstico , Adolescente , Corticosteroides/uso terapêutico , Hiperplasia Angiolinfoide com Eosinofilia/tratamento farmacológico , Hiperplasia Angiolinfoide com Eosinofilia/imunologia , Hiperplasia Angiolinfoide com Eosinofilia/patologia , Brasil/etnologia , Ciclosporina/uso terapêutico , Derme/patologia , Humanos , Imunoglobulina E/sangue , Imunoglobulina G/sangue , Imunofenotipagem , Imunossupressores/uso terapêutico , Masculino , Proteinúria/etiologia , Subpopulações de Linfócitos T/patologiaRESUMO
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.
Assuntos
Dermatomicoses/epidemiologia , Histoplasmose/epidemiologia , Síndrome da Imunodeficiência Adquirida/epidemiologia , Adulto , Dermatomicoses/microbiologia , Feminino , Histoplasmose/patologia , Humanos , Incidência , Masculino , Martinica/epidemiologia , Pessoa de Meia-Idade , Estudos RetrospectivosRESUMO
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.