Repurposing propranolol as a drug for the treatment of retinal haemangioblastomas in von Hippel-Lindau disease View Full Text


Ontology type: schema:ScholarlyArticle      Open Access: True


Article Info

DATE

2017-06-29

AUTHORS

Virginia Albiñana, Rosa María Jiménez Escribano, Isabel Soler, Luis Rodríguez Padial, Lucia Recio-Poveda, Karina Villar Gómez de las Heras, Luisa María Botella

ABSTRACT

BACKGROUND: Von Hippel-Lindau (VHL) disease is a rare oncological disease with an incidence of 1:36,000, and is characterized by the growth of different types of tumours. Haemangioblastomas in the central nervous system (CNS) and retina, renal carcinoma and pheochromocytomas are the most common tumours. The absence of treatment for VHL leads to the need of repeated surgeries as the only option for these patients. Targeting VHL-derived tumours with drugs with reduced side effects is urgent to avoid repeated CNS surgeries. Recent reports have demonstrated that propranolol, a β-blocker used for the treatment of hypertension and other cardiac and neurological diseases, is the best option for infantile hemangioma (IH). Propranolol could be an efficient treatment to control haemangioblastoma growth in VHL disease given its antiangiogenic effects that were recently demonstrated by us. The main objective of the present study was the assessment of the efficacy and safety of propranolol on retinal haemangioblastoma in von Hippel-Lindau disease (VHL). METHODS: 7 VHL patients, from different regions of Spain, affected from juxtapapillary or peripheral haemangioblastomas were administered 120 mg propranolol daily. Patients were evaluated every 3 months for 12 months, at Virgen de la Salud Hospital (Toledo). The patients had juxtapapillary or peripheral haemangioblastomas but had refused standard treatments. RESULTS: Propranolol was initiated with a progressive increase up to a final dose of 120 mg daily. All tumours remained stable, and no new tumours appeared. The reabsorption of retinal exudation was noted in the two patients having exudates. No adverse effects were recorded. VEGF and miRNA 210 levels were monitored in the plasma of patients as possible biomarkers of VHL. These levels decreased in all cases from the first month of treatment. CONCLUSIONS: Although more studies are necessary, the results of this work suggest that propranolol is a drug to be considered in the treatment of VHL patients with retinal haemangioblastomas. VEGF and miRNA 210 could be used as biomarkers of the VHL disease activity. TRIAL REGISTRATION: The study has a clinical trial design and was registered at EU Clinical Trials Register and Spanish Clinical Studies Registry, EudraCT Number: 2014-003671-30 . Registered 2 September 2014. More... »

PAGES

122

Identifiers

URI

http://scigraph.springernature.com/pub.10.1186/s13023-017-0664-7

DOI

http://dx.doi.org/10.1186/s13023-017-0664-7

DIMENSIONS

https://app.dimensions.ai/details/publication/pub.1090300842

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/28662711


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92 la Salud Hospital
93 levels
94 main objective
95 miRNA 210 levels
96 miRNA-210
97 months
98 more studies
99 need
100 nervous system
101 neurological diseases
102 new tumors
103 number
104 objective
105 oncological diseases
106 only option
107 options
108 patients
109 peripheral haemangioblastomas
110 pheochromocytoma
111 plasma
112 plasma of patients
113 possible biomarkers
114 present study
115 progressive increase
116 propranolol
117 rare oncological disease
118 reabsorption
119 recent reports
120 region
121 registry
122 renal carcinoma
123 report
124 results
125 retina
126 retinal exudation
127 retinal haemangioblastoma
128 safety
129 safety of propranolol
130 side effects
131 standard treatment
132 study
133 surgery
134 system
135 treatment
136 treatment of hypertension
137 trial design
138 tumors
139 types
140 von Hippel-Lindau disease
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