Biomedical subjects
Publications and source records attributed to F Cambazard.
Three cases of generalized skin reactions occurred in patients undergoing oral therapy with Fenbufen (Cinopal), a new anti-inflammatory agent. The lesions spontaneously improved when the drug was discontinued. The role of Fenbufen in the pathogenesis of the lesions remains unclear. In the three cases, the detection of circulating immune complexes would suggest a sensibilization mechanism.
Three clinically and histopathologically typical cases of histiocytosis X have been studied by indirect immunofluorescence using monoclonal antibodies on skin lesions. An ultrastructural and immunoperoxidase technique has also been performed. The results confirm that the proliferative skin population of histiocytosis X is composed of Langerhans cells and that histochemical immunology is an effective process which can replace electron microscopy to confirm the diagnosis.
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The authors reviewed the literature of cases of mastocytosis with either anetoderma or pseudo-pelade. They studied the different substances in the secretions of mastocytes which could have an effect on collagen and elastic fibres. They supported the theory that there is a susceptible connective tissue. They stressed the underestimation of these associated features of mastocytosis.
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The authors report the observation of pyoderma gangrenosum (P. G.) leading to the discovery of an underlying biclonal gammapathy, which despite the absence of bony lesions, almost certainly represents a malignant myeloma. The authors have reviewed the different known associations with pyoderma gangrenosum, with special reference to benign and malignant gammapathies. Rare cases of biclonal gammapathies with pyoderma gangrenosum have been published but none of a malignant nature. This therefore appears to be the first reported cases. The authors are aware of the reported tendency of pyoderma gangrenosum lesions to appear in areas of previous trauma, as may be the case in this patient. Finally systemic corticosteroid therapy produced a rapid remission of the skin lesions, but with the reactivation of known treated pulmonary tuberculosis.
BACKGROUND: Solitary histiocytoma is an uncommon form of Hashimoto-Pritzker syndrome and an exceptional type of histiocytosis with cells of undetermined origin. A solitary often ulcerated congenital nodule is generally observed. We report two cases, one of each form. CASE REPORTS: Both cases presented an ulcerative budding congenital tumefaction of the plantar aspect of the right foot for the first child and the parieto-axillary region in the second. Histology disclosed a granulomatous infiltrate of histiocytes positive for specific immunolabels (protein S100 and CD1a). In the first case, electron microscopy revealed histiocytes devoid of Birbeck granules and myelinoid bodies leading to the diagnosis of Langerhans histiocytosis with cells of unknown origin. In the second case, 18 p. 100 of the cells contained Birbeck granules. There has been no recurrence after a 5-year follow-up in a case. DISCUSSION: These cases recall the congenital nature of some types of solitary histiocytomas. Indeed, congenital Langerhans histiocytoma can occur as a unique nodule. The tumefaction may lie in any localization. Histological diagnosis is required. The benign nature of these lesions is confirmed by the absence of distant lesions and the lack of recurrence after complete excision. About a dozen cases have been reported. Most have been Hashimoto-Pritzker syndromes. Only one case has been reported with cells of undetermined origin. The diagnosis of histiocytosis with cells of undetermined origin is made when the ultrastructure study demonstrates the vacuity of the histiocyte cytoplasm. This condition is similar to Hashimoto-Pritzker syndrome by the absence of recurrence and systemic diffusion. It can however be observed in adults. The undetermined cell types would correspond different phases of Langerhans cell maturation or involution.
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