{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,31]],"date-time":"2025-10-31T14:35:38Z","timestamp":1761921338417,"version":"build-2065373602"},"reference-count":7,"publisher":"MDPI AG","issue":"9","license":[{"start":{"date-parts":[[2025,5,4]],"date-time":"2025-05-04T00:00:00Z","timestamp":1746316800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Diagnostics"],"abstract":"<jats:p>A 67-year-old male with metastatic human papillomavirus (HPV)-positive oropharyngeal cancer receiving pembrolizumab (anti-programmed cell death protein 1 [PD-1] immune checkpoint inhibitor) presented with bilateral ocular dryness. It is important to note that these symptoms appeared eight months after the initiation of the pembrolizumab therapy. Ophthalmologic evaluation revealed keratoconjunctivitis sicca with characteristic bulbar fluorescein staining and the Schirmer test showed 0 mm bilaterally. Serological testing demonstrated positive antinuclear and anti-SSb\/La antibodies, consistent with Sj\u00f6gren\u2019s syndrome as an immune-related adverse event (irAE). Treatment with topical fluorometholone 0.1% and diquafosol 3% led to complete symptom resolution within one year while maintaining cancer immunotherapy. Long-term follow-up over 3.5 years demonstrated sustained ocular improvement and a favorable oncologic response without development of systemic autoimmune manifestations. This case highlights that Sj\u00f6gren\u2019s syndrome as an irAE may present with isolated ocular manifestations, which could be overlooked in clinical practice.<\/jats:p>","DOI":"10.3390\/diagnostics15091168","type":"journal-article","created":{"date-parts":[[2025,5,4]],"date-time":"2025-05-04T20:09:14Z","timestamp":1746389354000},"page":"1168","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Immune Checkpoint Inhibitor-Related Sj\u00f6gren\u2019s Syndrome: An Ocular Immune-Related Adverse Event"],"prefix":"10.3390","volume":"15","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4893-7607","authenticated-orcid":false,"given":"Hideki","family":"Fukuoka","sequence":"first","affiliation":[{"name":"Department of Ophthalmology, Kyoto Prefectural University of Medicine, Kyoto 602-8566, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4672-785X","authenticated-orcid":false,"given":"Akifumi","family":"Matsumoto","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, Kyoto Prefectural University of Medicine, Kyoto 602-8566, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Chie","family":"Sotozono","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, Kyoto Prefectural University of Medicine, Kyoto 602-8566, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2025,5,4]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"348","DOI":"10.1016\/j.ajo.2024.08.044","article-title":"Ophthalmic Immune-Related Adverse Events and Association with Survival: Results From a Real-World Database","volume":"268","author":"Quiruz","year":"2024","journal-title":"Am. J. Ophthalmol."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"e007","DOI":"10.1097\/coa.0000000000000007","article-title":"A Case of Bilateral Keratitis and Bilateral Anterior Uveitis Induced by Pembrolizumab","volume":"2","author":"Fukuoka","year":"2023","journal-title":"Cornea Open"},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"2441","DOI":"10.1007\/s00262-020-02635-3","article-title":"Factors Associated with Ocular Adverse Event after Immune Checkpoint Inhibitor Treatment","volume":"69","author":"Kim","year":"2020","journal-title":"Cancer Immunol. Immunother."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"288","DOI":"10.1038\/s41584-023-00932-6","article-title":"Genetics and Epigenetics of Primary Sj\u00f6gren Syndrome: Implications for Future Therapies","volume":"19","author":"Thorlacius","year":"2023","journal-title":"Nat. Rev. Rheumatol."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"387","DOI":"10.1080\/14737140.2017.1296765","article-title":"Immune-Related Ocular Toxicities in Solid Tumor Patients Treated with Immune Checkpoint Inhibitors: A Systematic Review","volume":"17","author":"Oweira","year":"2017","journal-title":"Expert Rev. Anticancer Ther."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"34","DOI":"10.1016\/j.ejca.2017.05.032","article-title":"Prevalence of Immune-Related Systemic Adverse Events in Patients Treated with Anti-Programmed Cell Death 1\/Anti-Programmed Cell Death-Ligand 1 Agents: A Single-Centre Pharmacovigilance Database Analysis","volume":"82","author":"Champiat","year":"2017","journal-title":"Eur. J. Cancer"},{"key":"ref_7","first-page":"114","article-title":"Sicca\/Sj\u00f6gren\u2019s Syndrome Triggered by PD-1\/PD-L1 Checkpoint Inhibitors. Data from the International Immunocancer Registry (ICIR)","volume":"37","author":"Maria","year":"2019","journal-title":"Clin. Exp. Rheumatol."}],"container-title":["Diagnostics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2075-4418\/15\/9\/1168\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,9]],"date-time":"2025-10-09T17:26:57Z","timestamp":1760030817000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2075-4418\/15\/9\/1168"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,5,4]]},"references-count":7,"journal-issue":{"issue":"9","published-online":{"date-parts":[[2025,5]]}},"alternative-id":["diagnostics15091168"],"URL":"https:\/\/doi.org\/10.3390\/diagnostics15091168","relation":{},"ISSN":["2075-4418"],"issn-type":[{"type":"electronic","value":"2075-4418"}],"subject":[],"published":{"date-parts":[[2025,5,4]]}}}