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Rosai-Dorfman disease.

G Santra1, B K Das, B Mandal

  • 1Department of Medicine, Medical College, 88 College Street, Kolkata 700073, India. g.santra@yahoo.com

Singapore Medical Journal
|November 25, 2010
PubMed
Summary

Sinus histiocytosis with massive lymphadenopathy (Rosai-Dorfman disease) is a rare disorder. This case highlights its rare thyroid involvement and multiple extranodal sites in a young patient, successfully treated with steroids.

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Area of Science:

  • Rare Histiocytic Disorders
  • Pediatric Endocrinology
  • Head and Neck Pathology

Background:

  • Sinus histiocytosis with massive lymphadenopathy, or Rosai-Dorfman disease (RDD), is a rare, benign histiocytic proliferative disorder of unknown cause.
  • Typically presents as painless cervical lymphadenopathy, but extranodal involvement, particularly in the head and neck, is frequent.

Observation:

  • A 13-year-old girl presented with a rare manifestation of RDD.
  • She exhibited cervical and mediastinal lymphadenopathy, scalp soft tissue swellings, bilateral proptosis, and goiter due to thyroid gland involvement.
  • Thyroid gland involvement in RDD is exceptionally uncommon in reported literature.

Findings:

  • The patient's extensive RDD involved both nodal and multiple extranodal sites, including the thyroid.
  • The clinical presentation was characterized by significant lymphadenopathy and prominent head and neck extranodal manifestations.
  • Steroid therapy led to remission of swellings and symptoms, indicating a positive treatment response.

Implications:

  • This case underscores the diverse and rare extranodal manifestations of Rosai-Dorfman disease.
  • It highlights the importance of considering RDD in pediatric patients with unexplained lymphadenopathy and head/neck masses, including thyroid involvement.
  • The successful steroid treatment suggests its potential efficacy in managing complex RDD cases with extranodal disease.