Intravitreal Injection of Propranolol for the Treatment of Retinal Capillary Hemangioma in a Case of Von Hippel–lindau

Karimi, Saeed; Nikkhah, Homayoun; Ahmadieh, Hamid; Safi, Sare

doi:10.1097/icb.0000000000000718

Cited by 16 publications

(19 citation statements)

References 9 publications

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“…These results provide us with the basis to explore higher doses of propranolol—up to 3 mg/kg body weight/day, adjusting dose by body weight—and even consider the use of a more selective β-blocker that could be more effective. It may be interesting as well to explore the effect of intravitreal injection of propranolol in clinical trials, as the case report recently published21 and based on the previous work by Nourinia et al , to determine the safe dose of intravitreal propranolol 22. Using intravitreal administration, the drug would achieve more easily the retina.…”

Section: Discussionmentioning

confidence: 99%

Evaluation of the safety and effectiveness of oral propranolol in patients with von Hippel-Lindau disease and retinal hemangioblastomas: phase III clinical trial

et al. 2019

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Backgroundvon Hippel-Lindau disease (VHL) is a multisystem cancer syndrome caused by mutations in the VHL gene. Retinal hemangioblastoma is one of the most common tumours, and when it appears near the optic nerve, its treatment is challenging and risky. To date, no treatment has proven effective in changing the course of the disease. This study was designed to evaluate the safety and effectiveness of propranolol in controlling these tumours.MethodsSeven patients were included. All patients took a daily dose of 120 mg of propranolol for 1 year. Clinical variables were assessed at baseline, and at 1, 3, 6, 9 and 12 months. The primary endpoint of the study was the number and size of retinal hemangioblastomas. On every visit, retinal outcomes and blood biomarkers (such as vascular endothelial growth factor (VEGF) and miR210) were analysed.ResultsNumber and size of retinal hemangioblastomas remained stable in all patients. All of them had initially increased levels of VEGF and miR210. There was a gradual reabsorption of retinal exudation in two patients, correlating with a progressive decrease of both biomarkers. The only adverse effect reported was hypotension in one patient.ConclusionsPropranolol could be used to treat retinal hemangioblastomas in VHL patients, although more studies are needed to determine the ideal dose and long-term effect. VEGF and miR210 should be explored as biomarkers of disease activity. As far as we know, these are the first biomarkers proposed to monitor the VHL disease activity.Trial registration number2014-003671-30

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Section: Discussionmentioning

confidence: 99%

Evaluation of the safety and effectiveness of oral propranolol in patients with von Hippel-Lindau disease and retinal hemangioblastomas: phase III clinical trial

et al. 2019

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“…It was demonstrated in vitro on hemangioblastoma cells from CNS (that are histopathologically identical to retinal hemangioblastomas) 12,23,24 treated with different concentrations of propranolol. 13,24,25 Propranolol also decreased VEGF in peripheral blood samples and reduced retinal exudation. 12 Our main objective is to assess long-term effect oral propranolol may have on the course of ophthalmic disease involvement.…”

Section: Introductionmentioning

confidence: 85%

Oral Propranolol in Von Hippel Lindau Ocular Affection

González-Rodríguez¹,

González-Rodríguez²,

Ramírez³

et al. 2021

Preprint

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Purpose. von Hippel Lindau (VHL) disease is a familiar syndrome associated with benign and malignant tumors. These tumors appear in the retina, cerebellum, spinal cord, and kidney. Retinal hemangioblastomas are one of the earliest and most frequent manifestations of this entity, and they can lead to blindness at a young age. Propranolol could be a promising treatment for retinal hemangioblastomas in von Hippel Lindau disease. Methods. Prospective cohort study. Seven patients with VHL disease and ocular affection that had rejected conventional treatment were included. Prospective analysis of seven patients was performed. We evaluated them for three years, with a complete ophthalmic evaluation that included: visual acuity, intraocular pressure, an examination of the anterior segment of the eye, fundoscopy, retinography, and optical coherence tomography (OCT). Heart rate and blood pressure on each patient were also measured. During the follow-up evaluation, two patients discontinued the treatment with propranolol after the first year and rejected any further treatment for their ocular affection; the rest continued therapy for the three years. Results: Visual acuity and tumor areas remained stable in 4 patients. Increased and new retinal exudation area was found in the two patients that discontinued the treatment with oral propranolol. Conclusions: Oral propranolol has shown a role in the reabsorption of retinal exudates in patients with von Hippel Lindau affection. It could delay or stabilize the ocular disease, maintaining visual acuity, and avoiding further complications in these patients. It is a well-known and available drug, without so many secondary effects, that could also have a role in other ocular diseases that course with exudation.

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“…No short-term adverse effects except a mild transient inflammatory response were observed in this case report. [104] Fluorescein leakage was decreased from the RHs located on the optic nerve head and in the inferonasal retinal periphery one month after the second intravitreal injection of propranolol. Decrease of the hemangioma vascularity and augmentation of its fibrosis associated with the attenuation of the feeder vessel were observed.…”

Section: Intravitreal Propranololmentioning

confidence: 93%

Von Hippel-Lindau Disease and the Eye

Karimi

Arabi

Shahraki

et al. 2020

JOVR

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Retinal hemangioblastoma (also referred to as retinal capillary hemangioma) is a benign lesion originating from the endothelial and glial components of the neurosensory retina and optic nerve head. Historically known as a manifestation of the von Hippel- Lindau (VHL) disease, it can be seen as an isolated finding or in association with some rare ocular conditions. In addition to characteristic ophthalmoscopic features, results of numerous ancillary tests including angiography, ultrasound, optical coherence tomography, and genetic tests may support the diagnosis and differentiate it from similar conditions. Because of serious life-threatening complications of VHL disease, every ocular approach to retinal hemangioblastomas should be in relationship with additional multidisciplinary diagnostic and therapeutic efforts. In addition, any patient with actual or probable diagnosis of VHL disease should be screened for ocular involvement. Unfavorable visual loss can occur early, and ocular complications of VHL range from exudative retinopathy to tractional retinal detachment, neovascular glaucoma, and phthisis bulbi. Accordingly, various treatment methods have been tested with overall acceptable responses, including photocoagulation, cryotherapy, photodynamic therapy, plaque radiotherapy, vitrectomy, and more novel intra-vitreal injections of anti-vascular endothelial growth factors and propranolol.

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Intravitreal Injection of Propranolol for the Treatment of Retinal Capillary Hemangioma in a Case of Von Hippel–lindau

Cited by 16 publications

References 9 publications

Evaluation of the safety and effectiveness of oral propranolol in patients with von Hippel-Lindau disease and retinal hemangioblastomas: phase III clinical trial

Evaluation of the safety and effectiveness of oral propranolol in patients with von Hippel-Lindau disease and retinal hemangioblastomas: phase III clinical trial

Oral Propranolol in Von Hippel Lindau Ocular Affection

Von Hippel-Lindau Disease and the Eye

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