Immune Checkpoint Inhibitor-Related Sjögren's Syndrome: An Ocular Immune-Related Adverse Event

Hideki Fukuoka1, Akifumi Matsumoto1, Chie Sotozono1

  • 1Department of Ophthalmology, Kyoto Prefectural University of Medicine, Kyoto 602-8566, Japan.

Insights

Pembrolizumab therapy for HPV-positive oropharyngeal cancer can cause Sjögren's syndrome, an immune-related adverse event. Early diagnosis and treatment of ocular symptoms are crucial for symptom resolution and continued cancer immunotherapy.

Area of Science:

  • Oncology
  • Immunology
  • Ophthalmology

Background:

  • Metastatic human papillomavirus (HPV)-positive oropharyngeal cancer is a significant clinical challenge.
  • Immune checkpoint inhibitors, such as pembrolizumab (anti-programmed cell death protein 1 [PD-1]), are increasingly used for cancer immunotherapy.
  • Immune-related adverse events (irAEs) are known complications of PD-1 inhibitor therapy.

Purpose of the Study:

  • To report a case of Sjögren's syndrome presenting as an isolated ocular irAE in a patient receiving pembrolizumab.
  • To highlight the importance of recognizing and managing ocular irAEs in patients undergoing cancer immunotherapy.
  • To demonstrate the efficacy of topical treatment for Sjögren's syndrome in this context.

Main Methods:

  • A case study of a 67-year-old male with metastatic HPV-positive oropharyngeal cancer treated with pembrolizumab.
  • Ophthalmologic evaluation including fluorescein staining and Schirmer testing.
  • Serological testing for autoimmune antibodies (antinuclear and anti-SSb/La).
  • Treatment with topical fluorometholone 0.1% and diquafosol 3%.

Main Results:

  • The patient developed bilateral ocular dryness (keratoconjunctivitis sicca) eight months after initiating pembrolizumab.
  • Diagnosis of Sjögren's syndrome was confirmed by positive serological markers.
  • Complete resolution of ocular symptoms was achieved within one year with topical treatment.
  • Sustained ocular improvement and favorable oncologic response observed over 3.5 years without systemic autoimmune complications.

Conclusions:

  • Sjögren's syndrome can manifest as an isolated ocular irAE during pembrolizumab therapy for HPV-positive oropharyngeal cancer.
  • Prompt ophthalmologic evaluation and appropriate treatment are essential for managing ocular irAEs.
  • Recognizing Sjögren's syndrome as a potential irAE can prevent diagnostic delays and ensure continued effective cancer immunotherapy.

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