Abstract | OBJECTIVE AND IMPORTANCE:
Rosai-Dorfman disease (RDD) is a benign and rare non-Langerhans cell histiocytic proliferative disorder. Laryngeal involvement is an unusual site of extranodal involvement of RDD. Laryngeal RDD can cause life-threatening airway obstruction that requires effective control of the disease. In this study, we report three cases of laryngeal RDD with excellent and durable responses to thalidomide. CLINICAL PRESENTATION: Patient 1 was a 39-year-old male who presented with a two-year history of nasal obstruction. Patient 2 was a 26-year-old woman who presented complaining of a hoarse voice for one year. Patient 3 was a 24-year-old man who presented with complaints of a hoarse voice and progressing dyspnea for five months. Electronic laryngoscopy revealed submucous nodular lesions in the nasal cavity, nasopharynx, and larynx of the three patients. Biopsy of the lesions showed large histiocytes with abundant pale cytoplasm which were S-100 and CD68 positive consistent with RDD. INTERVENTION: CONCLUSION:
Thalidomide may induce long-term remission in laryngeal RDD.
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Authors | Chong Wei, Dao-Bin Zhou |
Journal | Hematology (Amsterdam, Netherlands)
(Hematology)
Vol. 26
Issue 1
Pg. 552-555
(Dec 2021)
ISSN: 1607-8454 [Electronic] England |
PMID | 34348601
(Publication Type: Case Reports, Journal Article, Video-Audio Media)
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Chemical References |
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Topics |
- Adult
- Female
- Histiocytosis, Sinus
(drug therapy, metabolism, pathology)
- Humans
- Laryngeal Diseases
(drug therapy, metabolism, pathology)
- Larynx
(metabolism, pathology)
- Male
- Remission Induction
- Thalidomide
(administration & dosage)
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