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Case Reports
. 2018 Jan 30;6(1):11.
doi: 10.1186/s40425-018-0318-x.

Inflammatory demyelinating polyneuropathy versus leptomeningeal disease following Ipilimumab

Lorraine Cafuir  1 , David Lawson  2 , Nilesh Desai  3 , Vita Kesner  4 , Alfredo Voloschin  2
Affiliations
Case Reports

Inflammatory demyelinating polyneuropathy versus leptomeningeal disease following Ipilimumab

Lorraine Cafuir et al. J Immunother Cancer. .

Abstract

Background: Ipilimumab is an FDA-approved anti-CTLA-4 monoclonal antibody used in treatment of metastatic melanoma. We present an unusual neurological complication of Ipilimumab therapy and the diagnostic dilemma it caused.

Case presentation: A 42 year old male with Stage IV metastatic melanoma developed lower extremity weakness and sensory neuropathy following three doses of Ipilimumab. MRI of the lumbar spine was initially interpreted as diffuse leptomeningeal disease, and patient began Dexamethasone and radiation with improvement in symptoms. However, subsequent completion imaging revealed smooth nerve root involvement with sparing of the spinal cord, findings more compatible with inflammatory demyelinating polyneuropathy. The absence of malignant cells in the cerebrospinal fluid (CSF) and nerve conduction study (NCS) showing lumbar polyradiculoneuropathy with axonal involvement and demyelinating features supported the diagnosis of inflammatory demyelinating polyneuropathy. Later in the course of his disease, the patient developed frank leptomeningeal melanoma.

Conclusion: Ipilimumab immune-related toxicity presented as inflammatory demyelinating polyneuropathy, which was difficult to distinguish from leptomeningeal disease, a common complication of melanoma.

Keywords: Autoimmune; Immunotherapy; Inflammatory demyelinating polyneuropathy; Ipilimumab; Leptomeningeal carcinomatosis; Melanoma; Paraneoplastic autoimmune disease.

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Conflict of interest statement

Ethics approval and consent to participate

Not applicable, not a clinical trial.

Consent for publication

Not required by our local IRB if patient is deceased.

Competing interests

DL participates in pharmaceutical trials sponsored by Bristol-Myers Squibb, the maker of Ipilimumab. VK is the Principal Investigator for the NN103 Myasthenia Gravis Study funded by the NIH. LC, ND, and AV declare that they have no competing interests.

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Figures

Fig. 1
Fig. 1
Initial MRI lumbar spine following Ipilimumab. Sagittal and axial T1w fat (left and top right) saturated post contrast and axial T2w FSE (bottom right) images demonstrating smooth, non-nodular avid enhancement of cauda equina without significant nerve root thickening
Fig. 2
Fig. 2
Follow up post contrast cervical spine MRI. Parasagittal and axial T1w fat saturated post contrast images demonstrating smooth avid enhancement of all anterior and posterior cervical nerve roots (arrows) but no involvement of the cord surface. Both factors strongly mitigate against leptomeningeal metastatic disease
See this image and copyright information in PMC

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