Cutaneous syndromes of non-X histiocytosis. A review of the macrophage-histiocyte diseases of the skin

Winkelmann, R. K.

doi:10.1001/archderm.117.10.667

Cited by 66 publications

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“…In both of our case, the patients' age was around 1 year which is the commonest age for the occurrence of cutaneous lesions. Winkelmann reported no any sexual or racial predilection of JXG [12]. However, the male to female ratio of 1.5:1 cutaneous JXG in children has been reported in the literature.…”

Section: Discussionmentioning

confidence: 97%

Case Reports on Juvenile Xanthogranuloma and Brief Review of Literature

Pradhan¹

2017

SJCM

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Abstract:Juvenile xanthogranuloma is an unusual, self-limiting dermatological disorder occurring especially in infants, late childhood and rarely in adults. It belongs to the broad group of non-Langerhans cell histiocytosis. It usually appears as solitary or multiple papules, macules or nodules several millimeters in diameter with the head and neck being the most common site of involvement and vulva being the rare site. We report two cases of juvenile xanthogranuloma in an 18 months old female and 18 months old male. The female presented with three yellow papules measuring 2-3 mm in diameter around vulva region since 4 months. Similarly, the male presented with multiple tan-orange color macules on head and forehead measuring 3-4mm in diameter since 6 months. In both the cases, a biopsy without total excision was performed. The clinical and histopathological evaluation confirmed the diagnosis of juvenile xanthogranuloma. In childhood, juvenile xanthogranuloma is necessary to differentiate from another probable differential diagnosis by biopsy.

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Section: Discussionmentioning

confidence: 97%

Case Reports on Juvenile Xanthogranuloma and Brief Review of Literature

Pradhan¹

2017

SJCM

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“…Já a histiocitose de células não-Langerhans é representada por uma série de doenças cutâneas caracterizadas por xantomatose cutânea localizada e não agressiva, com tendência à regressão espontânea, e ausência dos grânulos de Birbeck (2).…”

Section: Discussionunclassified

“…Contudo, o xantoma disseminado manifesta-se, freqüentemente, por numerosas pápulas coalescentes em áreas de flexão cutânea (11,12). Dois casos anteriormente descritos na literatura como exemplos da associação do xantoma disseminado ao DIC foram posteriormente questionados, pois, segundo a opinião de alguns autores, poderiam se tratar do xantoma papular, uma vez que não havia o infiltrado inflamatório e o quadro clínico característico do xantoma disseminado (2).…”

Section: Discussionunclassified

“…Acomete principalmente crianças no primeiro ano de vida, podendo, contudo, ocorrer em cerca de 15% dos casos em adultos (2,13). O quadro clínico clássico é constituído por lesões cutâneas solitárias.…”

Section: Discussionunclassified

“…São geralmente classificadas em histiocitose de células de Langerhans e histiocitose de células não-Langerhans, de acordo com as características morfológicas e os marcadores fenotípicos das células envolvidas (2).…”

Section: Introductionunclassified

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Histiocitose cutânea não-Langerhans como causa de diabetes insípido central

Miranda

Bittencourt

et al. 2007

Arq Bras Endocrinol Metab

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RESUMOAs histiocitoses são doenças raras, resultantes de alterações na linhagem monocítica-histiocítica, com manifestações clínicas diversas. Entre as síndromes cutâneas de células não-Langerhans, o xantoma disseminado é a única entidade desse grupo classicamente associada ao diabetes insípido central (DIC). O caso clínico relatado refere-se a um paciente de 30 anos de idade que, dois anos após o diagnóstico de DIC, evoluiu com lesões cutâneas papulosas, eritêmato-acastanhadas, difusas, discretas e não confluentes. Os achados histológicos, imuno-histoquímicos e a microscopia eletrônica mostraram resultados compatíveis com a histiocitose de células não-Langerhans e sugestivos do xantogranuloma juvenil. A avaliação endócrino-metabólica não mostrou alterações durante o seguimento por 10 anos, com exceção do DIC. A ressonância magnética da hipófise demonstrou ausência do sinal hiperintenso (mancha brilhante) correspondente à neuro-hipófise. As radiografias e a cintilografia dos ossos não mostraram lesões osteolíticas. Este caso desperta a atenção para a importância do exame da pele nos casos de DIC e de sua associação com a histiocitose de células não-Langerhans de maneira mais ampla, e não restrita aos casos de xantoma disseminado. ABSTRACT Cutaneous Non-Langerhans Cells Histiocytoses as Cause of Central Diabetes Inspidus.The histiocytoses are rare diseases caused by alterations in the monocytehistiocytic series with several clinical findings. Among the cutaneous syndromes of non-Langerhans cells, xanthoma disseminatum is the only disease of this group that has been classically associated to the central diabetes insipidus (CDI). The case reported describes a 30-year-old man that two years after presenting with CDI developed non confluent disseminated cutaneous brown papular lesions throughout the body. The histopathology, immunohistochemistry, and electronic microscopy were compatible with the diagnosis of non-Langerhans histiocytoses, suggesting the diagnosis of juvenile xanthogranuloma. The endocrine-metabolic evaluation did not show other alterations besides CDI in a 10-year follow up. The magnetic resonance of hypophysis showed absence of the pituitary hyperintense sign (bright spot). The radiologic and scinthigraphic evaluation of the bones did not show the presence of osteolytic lesions. This case prints out the importance of skin examination in cases of CDI and its association with cutaneous non-Langerhans histiocytoses in a broader spectrum, rather then restricted to the cases of xanthoma disseminatum.

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Indeterminate‐cell histiocytosis: Immunophenotypic and cytogenetic findings in an infant

Flores-Stadler

González-Crussí

Greene

et al. 1999

Med. Pediatr. Oncol.

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Background The authors report the immunohistochemical, ultrastructural, and cytogenetic findings in a case of malignant histiocytic proliferation in an infant. Procedure The patient presented initially with bone lesions without skin or systemic involvement. Multiple biopsies were studied extensively by immunohistochemistry and electron microscopy. Cytogenetic studies of cell cultures supplemented with granulocyte‐monocyte colony stimulating factor (GM‐CSF) were also performed. Results Morphologically, the cells resembled Langerhans cells, although with greater pleomorphism, as evinced by cells with usual polylobated nuclei. These cells expressed markers for macrophages and antigen presenting cells and were CD1a– and S‐100–positive, but lacked Birbeck granules. The cells grown in culture supplemented with GM‐CSF showed a unique combination of numerical and structural abnormalities affecting chromosomes 1, 6, 8, and 10. The disease followed a malignant course leading to the patient's demise despite aggressive chemotherapy and bone marrow transplant. Conclusions The findings suggest a malignant hematopoietic stem‐cell neoplasm with a capacity for macrophage or dendritic‐cell differentiation. Morphology and immunophenotypic features place this neoplasm within the group recently conceptualized as indeterminate‐cell histiocytosis. Med. Pediatr. Oncol. 32:250–254, 1999. © 1999 Wiley‐Liss, Inc.

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Cutaneous syndromes of non-X histiocytosis. A review of the macrophage-histiocyte diseases of the skin

Cited by 66 publications

References 0 publications

Case Reports on Juvenile Xanthogranuloma and Brief Review of Literature

Case Reports on Juvenile Xanthogranuloma and Brief Review of Literature

Histiocitose cutânea não-Langerhans como causa de diabetes insípido central

Indeterminate‐cell histiocytosis: Immunophenotypic and cytogenetic findings in an infant

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