• Saleh S Baeesa, Hani Mahboob, Yazid Maghrabi, Mohammad Binmahfoodh, and Jaudah Almaghrabi.
• Division of Neurosurgery, Faculty of Medicine, King Abdulaziz University, Jeddah, Saudi Arabia; Department of Neurosciences, King Faisal Specialist Hospital and Research Center, Jeddah, Saudi Arabia. Electronic address: sbaeesa@kau.edu.sa.
• World Neurosurg. 2020 Dec 1; 144: 1-14.

BackgroundRosai-Dorfman disease (RDD) is a rare pathologic entity caused by sinus histiocytosis with massive cervical lymphadenopathy. Isolated spinal involvement is an infrequent presentation of extranodal RDD. The clinical and radiologic appearance of RDD represents a diagnostic challenge. We report 2 patients with paraparesis caused by RDD of the thoracic spine and a PRISMA-style systematic review.Case DescriptionThere were 2 patients with isolated extranodal thoracic spinal RDD without cervical lymphadenopathy. One patient presented with anterior thoracic RDD and a subtotal resection. The small residual disease completely responded to the postoperative course of steroids. The second patient had extradural thoracic spine RDD, which was resected completely. A 6-month postoperative follow-up magnetic resonance imaging (MRI) scan showed local recurrence, which responded to radiation therapy. Five years follow-up of both patients showed normal neurologic functions and no recurrence on MRI scan surveillance.ConclusionsRDD is a rare occurrence and should be considered in the differential diagnosis of extradural or intradural spinal lesions. Gross total resection is recommended, and long-term clinical follow-up with MRI is advised. Residual or recurrent RDD requires steroids or radiation therapy.Copyright © 2020 Elsevier Inc. All rights reserved.

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