Ipilimumab-induced Adenohypophysitis and Orbital Apex Syndrome: Importance of Early Diagnosis and Management

ABSTRACT

Ipilimumab is a novel anti-melanoma agent known to infrequently cause multi-organ autoimmunity. We report a case of pituitary hypophysitis and orbital inflammation followed by an orbital apex syndrome.

A 64-year-old woman with a history of skin melanoma, receiving ipilimumab treatment, was seen for near total loss of vision in the right eye and proptosis. Headache of 3-month duration preceded the onset of diplopia followed by severe loss of vision in the right eye. Neuro-ophthalmologic examination was consistent with an orbital apex syndrome. Extensive blood work and magnetic resonance imaging of the brain and orbit suggested an inflammatory process, rather than a metastatic lesion. Accordingly, the patient received high-dose methylprednisolone followed by tapering oral prednisone. At the 6-month follow-up visit, visual acuity on the right eye had significantly improved but diplopia remained, associated with large amplitude esotropia that improved incompletely though while on prednisone. The favourable outcome supported a final diagnosis of ipilimumab-induced inflammatory orbital apex syndrome and clinically silent pituitary adenohypophysitis.

The case presented herein highlights unexpected ipilimumab-associated adverse effects and proposes the possibility of and interaction between inflammatory and immune mechanisms.

KEYWORDS:

• Adenohypophysitis
• ipilimumab
• orbital apex syndrome

Informed consent

Informed consent was obtained in writing from the patient for disclosure of clinical information and external photograph, for the purpose of this report.

Acknowledgements

The authors would like to thank Lynn Bannon, Ronda Boggs, Cecilia Myers, and Whitney Miller at the “OSF HealthCare Illinois Neurological Institute” Eye Center, for all their care and support for patients in the Midwest. In addition, we would like to thank Wyatt Verplaetse, medical student at UICOMP, for helping us with editing the article.

Declaration of interest

The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the article.