Cytomegalovirus retinitis after golidocitinib therapy in a patient with cutaneous T-cell lymphoma: a case report

The expanding use of biologic and targeted immunomodulatory therapies has been accompanied by an increasing number of reports of opportunistic ocular infections. Cytomegalovirus (CMV) retinitis (CMVR), in particular, has recently been reported after exposure to several Janus kinase (JAK) inhibitors, raising concern about a potential class-related ocular safety signal. We report a case of CMVR following exposure to golidocitinib, a selective JAK1 inhibitor, in an immunocompromised patient without human immunodeficiency virus (HIV) infection, in whom undetectable plasma CMV DNA contributed to delayed diagnosis. A 62-year-old HIV-negative man with relapsed primary cutaneous T-cell lymphoma (CTCL) and diabetes developed CMV viremia after four cycles of a golidocitinib-containing regimen. Golidocitinib was discontinued, and intravenous ganciclovir rendered plasma CMV DNA undetectable. During maintenance antiviral therapy, he developed progressive bilateral visual loss while weekly plasma testing remained negative. Early fundus photographs showed the classic “ketchup-and-cheese” appearance of fulminant CMVR, but diabetic retinopathy was diagnosed because of his diabetes and negative plasma CMV DNA result. This misdiagnosis delayed treatment for one month. At referral, visual acuity was hand motion in the right eye and 20/133 in the left eye, with severe vitritis, retinal vascular whitening, and disruption of the retinal layers. Subsequent intraocular fluid testing showed a vitreous CMV DNA level of 4.25 × 10 4 copies/mL, and targeted next-generation sequencing detected CMV. Subsequent systemic and intravitreal ganciclovir controlled the retinitis, but vision did not recover. To the best of our knowledge, this is the first published case report describing CMVR occurring after golidocitinib therapy. Undetectable plasma CMV DNA does not exclude compartmentalized ocular infection and should not delay intraocular fluid testing when necrotizing retinitis is suspected.