Endolymphatic non-langerhans cell histiocytosis of the larynx: report of an uncommon disease manifestation

J Pediatr Hematol Oncol. 2010 Jul;32(5):e173-6. doi: 10.1097/MPH.0b013e3181d746eb.

Abstract

We report on an uncommon laryngeal non-Langerhans cell histiocytosis. An 11-year-old boy presented with a 6 months history of progressive breath inhibition. Magnetic resonance imaging showed diffuse laryngeal and local lymph node swelling. Histology first resembled sarcoidosis, however, corticosteroids were ineffective. Lymphoma, infection, immunodeficiency, and autoimmune disease were excluded. Six months later, biopsies were repeated, now showing numerous ectatic lymph vessels with clusters of histiocytes bearing stellate extensions and emperipolesis. S100 protein and CD1a were negative. Indomethacin treatment led to a gradual improvement. In conclusion, we observed a nonmalignant non-Langerhans cell endolymphatic reticulohistiocytosis, not fitting into any of the described categories.

Publication types

  • Case Reports

MeSH terms

  • Antigens, CD1 / metabolism
  • Child
  • Endolymph*
  • Histiocytosis, Non-Langerhans-Cell / diagnosis*
  • Histiocytosis, Non-Langerhans-Cell / metabolism
  • Histiocytosis, Non-Langerhans-Cell / therapy
  • Humans
  • Laryngeal Diseases / diagnosis*
  • Laryngeal Diseases / metabolism
  • Laryngeal Diseases / therapy
  • Magnetic Resonance Imaging
  • Male
  • Prognosis
  • S100 Proteins / metabolism

Substances

  • Antigens, CD1
  • S100 Proteins