Abstract Details

Title Acute NMOSD Attack in the Setting of Pembrolizumab Therapy: A Case Report

Topic Autoimmune Neurology

Presentation(s) P13 - Poster Session 13 (8:00 AM-9:00 AM)

Poster/Presentation
Number 6-009

Objective

To report a case of a 66-year-old woman with a history of aquaporin-4-IgG seropositive (AQP4-IgG+) Neuromyelitis Optica Spectrum Disorder (NMOSD) and concurrent metastatic oral cavity squamous cell carcinoma (SCC) who presented with acute left-sided hemiplegia in the setting of pembrolizumab therapy.

Background

Pembrolizumab is an immune checkpoint inhibitor (ICI) approved for the treatment of several malignancies. ICIs are known to cause a wide variety of immune-related adverse events. While there have been no studies on the safety of ICIs in NMOSD patients, there was a case reported of newly-diagnosed NMOSD following nivolumab treatment. This is a novel case of an NMOSD attack following pembrolizumab treatment in an AQP4-IgG+ NMOSD patient on long-term anti-CD19 B cell depleting therapy.

Design/Methods Case report

Results A 66-year-old woman with AQP4-IgG+ NMOSD, diagnosed in 2015 and treated with inebilizumab, was diagnosed with metastatic SCC of the tongue in February 2022. She had received inebilizumab every six months from February 2016 to October 2021; infusion was cancelled in April 2022 due to cytotoxic chemotherapy (CD19+ 2 cells/ml; CD27+ 0 cells/ml). Despite resective surgery of the tongue and adjuvant concurrent chemotherapy and radiation, she was found to have widely metastatic SCC in early August 2022 (CD19+ 29 cells/ml; CD27+ 17 cells/ml; CD38+IgM- 12 cells/ml). She was administered inebilizumab and then started on pembrolizumab twelve days later. Ten days after receiving her first pembrolizumab therapy, the patient developed acute onset left-sided weakness (CD19+ 4 cells/ml; CD27+ 0 cells/ml; CD38+IgM- 0 cells/ml). She was found to have a large, active cervical spine lesion consistent with a new NMOSD attack; serum AQP4-IgG had increased from 1:10,000 to 1:100,000.

Conclusions An NMOSD attack occurred rapidly following pembrolizumab in a B-cell depleted patient, suggesting a role for T cell activation in relapse. ICIs should be used with caution in NMOSD patients.

Authors/Disclosures
Margaret E. Upchurch, MD PRESENTER	The institution of Dr. Upchurch has received research support from National MS Society.
Nicole Caunt, DO	Dr. Caunt has nothing to disclose.
	No disclosure on file
Jeffrey L. Bennett, MD, PhD, FAAN (University of Colorado School of Medicine)	Dr. Bennett has received personal compensation in the range of $10,000-$49,999 for serving as a Consultant for Amgen. Dr. Bennett has received personal compensation in the range of $10,000-$49,999 for serving as a Consultant for Alexion. Dr. Bennett has received personal compensation in the range of $500-$4,999 for serving as a Consultant for Genentech. Dr. Bennett has received personal compensation in the range of $10,000-$49,999 for serving as a Consultant for Roche. Dr. Bennett has received personal compensation in the range of $5,000-$9,999 for serving as a Consultant for Mitsubishi Tanabe. Dr. Bennett has received personal compensation in the range of $10,000-$49,999 for serving as a Consultant for Novartis. Dr. Bennett has received personal compensation in the range of $500-$4,999 for serving as a Consultant for Immpact Bio. Dr. Bennett has received personal compensation in the range of $5,000-$9,999 for serving as a Consultant for Chugai. Dr. Bennett has received personal compensation in the range of $500-$4,999 for serving as a Consultant for Beigene. Dr. Bennett has received personal compensation in the range of $500-$4,999 for serving as a Consultant for Imcyse. Dr. Bennett has received personal compensation in the range of $5,000-$9,999 for serving on a Scientific Advisory or Data Safety Monitoring board for Roche. Dr. Bennett has received personal compensation in the range of $500-$4,999 for serving on a Scientific Advisory or Data Safety Monitoring board for MIAC. Dr. Bennett has received personal compensation in the range of $5,000-$9,999 for serving on a Scientific Advisory or Data Safety Monitoring board for CorEvitas. Dr. Bennett has received personal compensation in the range of $50,000-$99,999 for serving on a Speakers Bureau for Alexion. Dr. Bennett has received personal compensation in the range of $500-$4,999 for serving on a Speakers Bureau for Vindico. Dr. Bennett has received personal compensation in the range of $500-$4,999 for serving on a Speakers Bureau for Touch IME. Dr. Bennett has received personal compensation in the range of $500-$4,999 for serving on a Speakers Bureau for Efficient LLC. Dr. Bennett has received personal compensation in the range of $10,000-$49,999 for serving as an Expert Witness for Pavich. Dr. Bennett has received personal compensation in the range of $500-$4,999 for serving as an Expert Witness for Marie Bush. Dr. Bennett has received personal compensation in the range of $10,000-$49,999 for serving as an Expert Witness for Marks Gray. Dr. Bennett has received personal compensation in the range of $500-$4,999 for serving as an Expert Witness for Knight, Nicastro, MacKay. The institution of Dr. Bennett has received research support from Alexion. The institution of Dr. Bennett has received research support from Genentech. Dr. Bennett has received intellectual property interests from a discovery or technology relating to health care. Dr. Bennett has received publishing royalties from a publication relating to health care.
Amanda L. Piquet, MD, FAAN (University of Colorado)	The institution of Dr. Piquet has received personal compensation in the range of $10,000-$49,999 for serving as a Consultant for Genentech/Roche. The institution of Dr. Piquet has received personal compensation in the range of $5,000-$9,999 for serving as a Consultant for Alexion. The institution of Dr. Piquet has received personal compensation in the range of $5,000-$9,999 for serving as a Consultant for Kyverna . The institution of Dr. Piquet has received personal compensation in the range of $5,000-$9,999 for serving on a Scientific Advisory or Data Safety Monitoring board for Genentech/Roche. The institution of Dr. Piquet has received personal compensation in the range of $5,000-$9,999 for serving on a Scientific Advisory or Data Safety Monitoring board for Kyverna. The institution of Dr. Piquet has received personal compensation in the range of $10,000-$49,999 for serving on a Scientific Advisory or Data Safety Monitoring board for Alexion. Dr. Piquet has received personal compensation in the range of $10,000-$49,999 for serving as an Expert Witness for Sands Anderson PC. Dr. Piquet has received personal compensation in the range of $5,000-$9,999 for serving as an Expert Witness for Joe Jones Law Firm. Dr. Piquet has received personal compensation in the range of $500-$4,999 for serving as an Expert Witness for Cortez & Associates. Dr. Piquet has received personal compensation in the range of $500-$4,999 for serving as an Expert Witness for Falk Waas. The institution of Dr. Piquet has received research support from Rocky Mountain MS Center. The institution of Dr. Piquet has received research support from Roche/Genentech. The institution of Dr. Piquet has received research support from NYU. The institution of Dr. Piquet has received research support from Anokion. The institution of Dr. Piquet has received research support from UCB . The institution of Dr. Piquet has received research support from Foundation for Sarcoidosis. The institution of Dr. Piquet has received research support from Kyverna . Dr. Piquet has received publishing royalties from a publication relating to health care. Dr. Piquet has received publishing royalties from a publication relating to health care. Dr. Piquet has received personal compensation in the range of $10,000-$49,999 for serving as a Litigative Consultant with US-Dept HHS/DICP. Dr. Piquet has a non-compensated relationship as a Medical Advisory Board Member with Autoimmune Encephalitis Alliance (AEA) that is relevant to AAN interests or activities. Dr. Piquet has a non-compensated relationship as a Medical Advisory Board Member with Stiff Person Syndrome Research Foundation (SPSRF) that is relevant to AAN interests or activities.

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