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Isolated extradural Rosai-Dorfman disease causing the spinal cord compression: A case report.

ABSTRACT: RATIONALE:Rosai-Dorfman disease (RDD) is a rare benign histiocytic disease that is commonly characterized by massive painless cervical lymphadenopathy and systemic manifestations. Isolated extranodal involvement, especially spinal involvement, is extremely rare. PATIENT CONCERNS:A 28-year-old man presented with intermittent dorsodynia and bilateral lower-limb weakness and numbness. A magnetic resonance scan (MRI) showed an extradural lesion of the T6-T9 thoracic spine that lead to cord compression. DIAGNOSES:Histopathological findings showed distinctive emperipolesis and immunohistochemistry results that were positive for cluster of differentiation CD68 and S100. Therefore, we diagnosed the Rosai-Dorfman disease. INTERVENTIONS:we performed a nearly total surgical resection and a limited T6-T9 laminectomy. OUTCOMES:Postoperatively, the patient's symptoms were partially relieved and experienced no recurrence during the 6-month follow-up. The preoperative diagnosis of isolated spinal RDD still remains challenging. LESSONS:Thus, we should consider RDD in the differential diagnosis of the central nervous system. Besides surgical resection, the treatment also included radiation, chemotherapy or monoclonal antibodies. However, the optimal treatment remains controversial. Therefore, we should exert all our energies on the exploration of etiology and adjuvant therapy for this disease.

SUBMITTER: Li Y

PROVIDER: S-EPMC6200482 | biostudies-other | 2018 Oct

REPOSITORIES: biostudies-other

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Isolated extradural Rosai-Dorfman disease causing the spinal cord compression: A case report.

Li Yongning Y Wang Xin X Gao Jun J Yu Shuangni S Li Zhimin Z

Medicine 20181001 40

<h4>Rationale</h4>Rosai-Dorfman disease (RDD) is a rare benign histiocytic disease that is commonly characterized by massive painless cervical lymphadenopathy and systemic manifestations. Isolated extranodal involvement, especially spinal involvement, is extremely rare.<h4>Patient concerns</h4>A 28-year-old man presented with intermittent dorsodynia and bilateral lower-limb weakness and numbness. A magnetic resonance scan (MRI) showed an extradural lesion of the T6-T9 thoracic spine that lead to ...[more]

PMID: 30290681

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Project description:BackgroundSpontaneous spinal subarachnoid hemorrhage (SSH) is an infrequent yet critical condition, accounting for less than 1% of all spinal hemorrhages and presenting substantial diagnostic difficulties. It predominantly arises due to factors such as tumors, trauma, arteriovenous malformations (AVM), and certain medications including anticoagulants. The complexity of its presentation necessitates vigilance from healthcare providers to avert irreversible neurological impairments.Case descriptionThis study delineates the unusual case of an 82-year-old woman undergoing direct oral anticoagulant (DOAC) therapy who reported sudden, severe chest and back pain, rapidly escalating to lower limb motor and sensory deficits, coupled with urinary and fecal incontinence. An initial cranial computed tomography (CT) indicated a peri-mesencephalic subarachnoid hemorrhage (SAH). Subsequent spinal CT angiography (CTA) and magnetic resonance imaging (MRI) affirmed the diagnosis of a spontaneous SSH, displaying significant cord compression, particularly in the thoracic regions between the T6 and T9 vertebrae. Emergency laminectomy was done to evacuate the subarachnoid hematoma that was causing cord compression. Postoperatively, the patient recovered partial neurological function.ConclusionsSSH, though rare, embodies a significant diagnostic hurdle and potential for lasting neurological deficits if not identified and treated promptly. Particularly in patients on anticoagulant therapy presenting with acute onset back pain and neurological dysfunction, SSH ought to be a crucial consideration. It is imperative to integrate prompt and advanced neuroimaging techniques and consider spinal angiography as a golden standard for a comprehensive diagnosis. Treatment strategies depend largely on the presence of neurological deficits, emphasizing the necessity of quick identification and emergency intervention in severe cases. This study elucidates the critical role of timely intervention in optimizing recovery outcomes, highlighting the necessity of including anticoagulant-induced spinal hemorrhage in differential diagnoses for patients manifesting signs of acute spinal cord compression.

Dataset Information

Isolated extradural Rosai-Dorfman disease causing the spinal cord compression: A case report.

Publications

Isolated extradural Rosai-Dorfman disease causing the spinal cord compression: A case report.

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