Management of intracranial Rosai-Dorfman disease: An institutional experience

J Clin Neurosci. 2024 Sep:127:110758. doi: 10.1016/j.jocn.2024.110758. Epub 2024 Jul 24.

Abstract

Objective: Rosai-Dorfman disease (RDD) is a rare benign proliferative disorder of histiocytes. The study discusses the intracranial RDD approach, its management, and its outcome.

Methods: It is a retrospective study performed in a tertiary center, the National Institute of Mental Health and Neuroscience, Bangalore, from January 2010 to December 2022. The biopsy-proven patients of RDD were recruited in the present cohort. Demographic and surgical details were collected from the record section, and radiology was collected from the internal storage system. Follow-up assessments were done clinically and telephonically.

Results: A total of 25 patients matched the criteria. The mean age was 32 ± 13.4 years, with male predominance. We have included only cranial cases (N=25). Among the intracranial lesions, 5/25 (20 %) patients had multicentric lesions. All the lesions were avidly enhancing on contrast, and 16 (64 %) lesions were hypointense on T2. Perilesional edema (T2/Flair hyperintensities in the surrounding white matter) was seen in 12 (48 %) patients. Gross total resection (GTR) was carried out in six (24 %) cases. Sub-total resection was in 14 (56 %), and biopsy was in five cases (20 %). Nineteen patients received adjuvant therapy, either only steroid (40 %), only low-dose radiotherapy (16 %), only Chemotherapy (4 %), or a combination of both. At follow-up,44 % of patients had stable disease,28 % had primary disease or recurrence growth, and regression in 12 % of cases.

Conclusion: We demonstrate that surgical resection is an effective therapy for treating isolated intracranial RDD. Adjuvant therapy is an add-on treatment for skull base locations in multicentric locations or surgically inaccessible locations.

Keywords: Adjuvant therapy; Outcome; Rosai-Dorfman disease; Surgery.

MeSH terms

  • Adolescent
  • Adult
  • Brain Diseases / pathology
  • Brain Diseases / surgery
  • Brain Diseases / therapy
  • Disease Management
  • Female
  • Histiocytosis, Sinus* / diagnosis
  • Histiocytosis, Sinus* / pathology
  • Histiocytosis, Sinus* / surgery
  • Histiocytosis, Sinus* / therapy
  • Humans
  • Magnetic Resonance Imaging
  • Male
  • Middle Aged
  • Neurosurgical Procedures / methods
  • Retrospective Studies
  • Treatment Outcome
  • Young Adult