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  • Title: Congenital solitary histiocytoma: a variant of Hashimoto-Pritzker histiocytosis. A retrospective study of 8 cases.
    Author: Zunino-Goutorbe C, Eschard C, Durlach A, Bernard P.
    Journal: Dermatology; 2008; 216(2):118-24. PubMed ID: 18216473.
    Abstract:
    BACKGROUND: Self-healing solitary-lesion Hashimoto-Pritzker histiocytosis (HPH), a rare, congenital, purely cutaneous Langerhans histiocytosis (only 30 cases reported), carries a good prognosis. OBJECTIVE: To describe the clinical and histopathological characteristics of solitary HPH. METHODS: To conduct a retrospective, observational study on 8 affected newborns. RESULTS: For these infants, with otherwise normal physical examinations, the unique nodule or papule (5-15-mm diameter) was congenital. Systematic routine histological examination of the lesions found dermal infiltrates constituted predominantly of histiocytes with lymphocytes and eosinophils. Protein S100 and CD1a immunolabelings, done for 7 patients, were positive. Electron microscopy (n = 4) observed Birbeck granules. No visceral involvement or recurrence has ever been observed after 2-12 years of follow-up. CONCLUSIONS: Because of its self-healing nature, congenital solitary HPH frequency has probably been underestimated. In the absence of systemic involvement, regular physical examination for at least 2 years seems a valid approach.
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