Document Detail

Otolaryngologic manifestations of Rosai-Dorfman Disease.
MedLine Citation:
PMID:  11397505     Owner:  NLM     Status:  MEDLINE    
PURPOSE: To describe an unusual head and neck occurrence of Rosai-Dorfman Disease (RDD) and to review the otolaryngologic manifestations of this rare entity.
METHODS: A case presentation with review of the literature describing Rosai-Dorfman Disease and its head and neck involvement.
SETTING: A tertiary care, urban children's hospital.
RESULTS: This is the first description, to the best of our knowledge, of RDD [Sinus Histiocytosis with Massive Lymphadenopathy (SHML)] involving bilateral external auditory canals and middle ear in a 12 year old patient previously diagnosed with 'asthma' and hearing loss. This patient also had extensive involvement of the tracheobronchial tree. Representative clinical, radiographic and histological findings are presented. Its etiology, diagnosis and management are also reviewed.
CONCLUSION: This is the first reported case of middle ear and external auditory canal involvement of RDD in a patient with hearing loss and associated tracheobronchial lesions. RDD, although rare, may be considered in the differential diagnosis of unusual histiocytic lesions of the external auditory canal, especially with findings of similar or comparable lesions involving the respiratory tract. Confirmation is with identification of emperipolesis and appropriate immunohistochemical staining (S-100 positive, CD-68 positive and CD-1a negative). Intervention is recommended in cases where there is increased risk of mortality, as in severe obstruction of the tracheobronchial tree. Otherwise, since these lesions are self-limiting, the patients can be observed closely.
S F Ahsan; D N Madgy; J Poulik
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  International journal of pediatric otorhinolaryngology     Volume:  59     ISSN:  0165-5876     ISO Abbreviation:  Int. J. Pediatr. Otorhinolaryngol.     Publication Date:  2001 Jul 
Date Detail:
Created Date:  2001-06-08     Completed Date:  2001-08-02     Revised Date:  2014-07-28    
Medline Journal Info:
Nlm Unique ID:  8003603     Medline TA:  Int J Pediatr Otorhinolaryngol     Country:  Ireland    
Other Details:
Languages:  eng     Pagination:  221-7     Citation Subset:  IM    
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MeSH Terms
Bronchial Diseases / diagnosis*,  pathology
Ear Canal / pathology
Ear Diseases / diagnosis*,  pathology
Ear, Middle / pathology
Histiocytosis, Sinus / complications,  diagnosis*,  pathology
Tracheal Diseases / diagnosis*,  pathology

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