Abstract Details

Title Infliximab-induced Demyelinating Polyneuropathy in Neurosarcoidosis

Topic Autoimmune Neurology

Presentation(s) P1 - Poster Session 1 (5:30 PM-6:30 PM)

Poster/Presentation
Number 15-034

Objective Tumor Necrosis Factor-α inhibitors (TNF-α) such as infliximab are increasingly being utilized for the management of neurosarcoidosis. Although there is growing evidence of efficacy for CNS involvement, adverse consequences for this patient population are not well-described. We report the first case of infliximab-induced demyelinating polyneuropathy (D-PN) in a patient with neurosarcoidosis.

Background A 55-year-old African-American woman with multi-systemic sarcoidosis including a history of diffuse CNS leptomeningeal involvement presented with a three-month history of progressive glove-stocking numbness and bilateral leg weakness. Six months prior, patient initiated infliximab and methotrexate with improvement of known CNS lesions that had manifested solely as gait unsteadiness. Neurologic examination now demonstrated length-dependent sensory changes, bilateral lower extremity weakness, and areflexia. NCS revealed several areas of conduction block, slowed conduction velocities and prolonged motor latencies consistent with a D-PN. MRI demonstrated worsening of the CNS disease with increased leptomeningeal enhancement. CSF cytology was negative on two separate samples although pleocytosis and hypoglycorrhachia were seen. Infliximab levels were normal (18μg/mL; normal >3μg/mL) with mildly elevated infliximab antibodies (79μg/mL; 22-200μg/mL indicates low titer). Infliximab was discontinued, and the patient was treated with a short course of methylprednisone resulting in gradual resolution of the D-PN. However, the CNS disease continued to progress until administration of cyclophosphamide.

Design/Methods *

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Conclusions While D-PN is a rare manifestation of neurosarcoidosis, it has been associated with TNF-α use in patients with inflammatory arthritis and ulcerative colitis, and typically occurs within eight months of drug initiation. The exact pathophysiology is unknown although a drug-induced dysimmunity hypothesis has been proposed. Given the temporal association, exclusion of other alternative etiologies and improvement on discontinuation of the infliximab despite worsening CNS disease, we believe the D-PN in this case is infliximab-induced. As infliximab may soon be considered first-line treatment for severe neurosarcoidosis, clinicians should be aware of this potential complication.

Authors/Disclosures
Christina Lineback, MD (McGaw Medical Center of Northwestern University) PRESENTER	Dr. Lineback has nothing to disclose.
Shailee S. Shah, MD	Dr. Shah has received personal compensation in the range of $500-$4,999 for serving on a Scientific Advisory or Data Safety Monitoring board for Horizon Therapeutics. Dr. Shah has received personal compensation in the range of $500-$4,999 for serving on a Scientific Advisory or Data Safety Monitoring board for Genentech. Dr. Shah has received personal compensation in the range of $500-$4,999 for serving on a Scientific Advisory or Data Safety Monitoring board for Genentech. Dr. Shah has received personal compensation in the range of $500-$4,999 for serving on a Scientific Advisory or Data Safety Monitoring board for Alexion. Dr. Shah has received publishing royalties from a publication relating to health care.
Jinny O. Tavee, MD (National Jewish Health)	Dr. Tavee has received personal compensation in the range of $10,000-$49,999 for serving as a Consultant for CSL Behring. Dr. Tavee has received personal compensation in the range of $500-$4,999 for serving as a Consultant for Johnson and Johnson. Dr. Tavee has received personal compensation in the range of $0-$499 for serving as an Editor, Associate Editor, or Editorial Advisory Board Member for Dynamed. The institution of Dr. Tavee has received research support from Woolsey. The institution of Dr. Tavee has received research support from Milken Foundation. The institution of Dr. Tavee has received research support from CSL Behring. Dr. Tavee has received personal compensation in the range of $0-$499 for serving as a Article author with Medlink.

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