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Isolating Human Peripheral Blood Mononuclear Cells and CD4+ T cells from Sézary Syndrome Patients for Transcriptomic Profiling
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[Xanthoma disseminatum with asymptomatic multisystem involvement].

M Zinoun1, F Hali1, F Marnissi2

  • 1Service de dermatologie-vénéréologie, CHU Ibn Rochd, 1, quartier des Hôpitaux, Casablanca, Maroc.

Annales De Dermatologie Et De Venereologie
|January 29, 2015
PubMed
Summary

Xanthoma disseminatum, a rare non-Langerhans histiocytosis, can present with localized skin lesions masking multisystem involvement. Early screening for visceral disease is crucial for prognosis and management of this condition.

Keywords:
Cellule de ToutonHistiocytose non langerhansienneMultisystem involvementNon-Langerhans cell histiocytosisTouton giant cellXanthogranulomaXanthogranulomeXanthoma disseminatum

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Area of Science:

  • Dermatology
  • Histiocytosis
  • Oncology

Background:

  • Xanthogranulomas are rare non-Langerhans histiocytoses, with Xanthoma disseminatum (XD) being a specific subtype.
  • XD typically involves skin and mucous membranes, but can present with asymptomatic multisystemic involvement.

Observation:

  • A 28-year-old man presented with facial xanthomatous lesions, later found to have gastric, lung, and potential bone involvement.
  • Histopathology confirmed a histiocytic infiltrate positive for CD68 and negative for CD1a, consistent with non-Langerhans histiocytosis.
  • The patient had no ophthalmic complications, diabetes insipidus, or monoclonal gammopathy.

Findings:

  • Localized facial and mucosal lesions in this XD case revealed asymptomatic multisystemic non-Langerhans histiocytosis.
  • Diagnosis was confirmed via clinical, histological, and immunohistochemical findings.
  • Treatment with corticosteroids and thalidomide led to significant regression of cutaneous lesions.

Implications:

  • This case highlights the importance of screening for visceral involvement in all xanthogranulomas, even with seemingly localized cutaneous manifestations.
  • Accurate diagnosis of XD relies on a combination of clinical and histopathological assessments.
  • Effective management of XD is challenging due to its rarity and variable presentation.