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Case Reports
. 2018 Jul 31;6(1):77.
doi: 10.1186/s40425-018-0390-2.

Delayed onset of neurosarcoidosis after concurrent ipilimumab/nivolumab therapy

Irena Tan  1 , Michael Malinzak  2 , April K S Salama  3
Affiliations
Case Reports

Delayed onset of neurosarcoidosis after concurrent ipilimumab/nivolumab therapy

Irena Tan et al. J Immunother Cancer. .

Abstract

Background: Immune checkpoint inhibitors have transformed the treatment landscape for many cancers, including metastatic melanoma, but have also opened the door for a diverse variety of immune-related adverse effects.

Case presentation: We describe the first reported case of presumed neurosarcoidosis as an immune-related adverse effect that developed nearly a year after discontinuation of treatment with combination ipilimumab and nivolumab for recurrent metastatic melanoma. The patient was noted to develop clinical signs consistent with systemic sarcoidosis shortly after the initiation of treatment and underwent a biopsy of hilar lymphadenopathy that confirmed sarcoidosis and after which immunotherapy was discontinued. His melanoma remained stable on surveillance imaging for the next year after which time he developed neurological symptoms and was found to have MRI brain abnormalities without evidence of intracranial metastatic disease, consistent with probable neurosarcoidosis given biopsy-proven systemic sarcoidosis and lack of evidence of CNS infection or malignancy. He underwent treatment with high dose steroids, followed by infliximab, and then methotrexate with both clinical and radiographic improvement within 4 months of starting treatment.

Conclusions: Immune-related adverse effects often occur within 3-6 months of receiving immune checkpoint inhibitor therapy, with some reports of late toxicity. This report highlights a case of probable neurosarcoidosis nearly a year after discontinuation of immune checkpoint therapy. The potential for durable responses after discontinuation of therapy also likely underscores a potential for late toxicity. In patients presenting with new or unexplained symptoms after checkpoint inhibitor therapy, the index of suspicion for an immune-related adverse effect should remain high, irrespective of timing.

Keywords: Immune-related adverse events; Ipilimumab; Neurosarcoidosis; Nivolumab.

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Conflict of interest statement

Ethics approval and consent to participate

No formal ethics approval was needed since we were reporting an observation case report. Consent was obtained from the patient.

Consent for publication

Consent was obtained from the patient. He signed a generic consent for this journal.

Competing interests

The authors declare that they have no competing interests.

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Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Figures

Fig. 1
Fig. 1
Brain MRI obtained at onset of neurological symptoms. T1-weighted contrast-enhanced axial image shows leptomeningeal enhancement involving the left occipital and parietal lobes (arrows)
Fig. 2
Fig. 2
Brain MRI obtained after treatment with steroids in setting of continued neurological complaints. T1-weighted contrast-enhanced axial images at a the level of the internal capsule and b the level of the corona radiata. Note the increased leptomeningeal enhancement (arrows) and new patchy areas of adjacent parenchymal enhancement (arrowheads)
Fig. 3
Fig. 3
Brain MRI obtained 4 months after starting methotrexate in setting of neurological improvement. T1-weighted contrast-enhanced axial image through the parieto-occipital region shows resolution of leptomeningeal enhancement
See this image and copyright information in PMC

References

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