Belzutifan-induced regression of retinal capillary hemangioblastoma: A case-series.
| Autor: | Ercanbrack CW; University of Arkansas for Medical Sciences, College of Medicine, Little Rock, AR, USA., Elhusseiny AM; Department of Ophthalmology, Harvey and Bernice Jones Eye Institute, University of Arkansas for Medical Sciences, Little Rock, AR, USA., Sanders RN; Department of Ophthalmology, Harvey and Bernice Jones Eye Institute, University of Arkansas for Medical Sciences, Little Rock, AR, USA., Santos Horta E; Department of Medical Oncology, University of Arkansas for Medical Sciences, Little Rock, AR, USA., Uwaydat SH; Department of Ophthalmology, Harvey and Bernice Jones Eye Institute, University of Arkansas for Medical Sciences, Little Rock, AR, USA. |
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| Jazyk: | angličtina |
| Zdroj: | American journal of ophthalmology case reports [Am J Ophthalmol Case Rep] 2024 Feb 10; Vol. 33, pp. 102011. Date of Electronic Publication: 2024 Feb 10 (Print Publication: 2024). |
| DOI: | 10.1016/j.ajoc.2024.102011 |
| Abstrakt: | Purpose: To report a series of three patients with von Hippel-Lindau (VHL) disease who demonstrated regression of their retinal hemangioblastomas (RH) using belzutifan in conjunction with photocoagulation therapy. Observations: Patient 1, a 23-year-old female, presented with multiple RHs in her right eye (OD) that were lasered. Her left eye (OS) revealed a large inferotemporal RH that measured approximately 2.1 mm 2 . Systemic belzutifan was administered. Four months after initiation of treatment, the lesion regressed to 1.4 mm 2 , but belzutifan was not well-tolerated and was discontinued due to side effects. At the date of belzutifan discontinuation, the lesion measured about 1.1 mm 2 . Focal laser photocoagulation was applied. The lesion regressed to around 0.6 mm 2 . Two additional laser treatments were applied one month later. On the most recent follow-up, the lesion was completely fibrosed.Patient 2, a 32-year-old male, presented with one RH OD and two RHs OS. Belzutifan was administered for one month before the patient began experiencing side effects of the medication. Consequently, the dose of belzutifan was decreased. After one month with the lowered dose, laser coagulation was applied to OS. In the most recent follow-up, five months after the initial presentation, the lesions remain less vascularized and reduced in size.Patient 3, is a 44-year-old male with a large RH OD. Following seven months of belzutifan daily, there was a significant reduction in the RH size. Conclusions: Belzutifan, a hypoxia-inducible factor inhibitor, is an FDA-approved medication for VHL disease associated with renal cell carcinoma, central nervous system hemangioblastomas, or pancreatic neuroendocrine tumors that do not require immediate surgical resection. Because of the high incidence of VHL-associated RHs, adjuvant laser photocoagulation therapy when belzutifan is suspended or withheld can allow for the regression of large lesions. In this case series, we also propose a reproducible and technically simple method to measure RH lesions size, using Optos fundus imaging. Competing Interests: The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper. (© 2024 The Authors.) |
| Databáze: | MEDLINE |
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