Abstract
Unlike adult neurofibromatosis type 2 (NF2), which presents with symptoms related to bilateral vestibular schwannomas, children with NF2 most frequently present with ocular, dermatological, and neurological symptoms. Arteriopathy, a well-established feature in neurofibromatosis type 1, is not a widely recognized feature of NF2. Here we report three children with NF2 with cerebral arteriopathy and/or arterial ischaemic stroke. Bevacizumab, a vascular endothethial growth factor inhibitor, is an established treatment for rapidly growing vestibular schwannomas; however, it carries a risk of both ischaemic and haemorrhagic stroke. Thus, the role of screening and risk to benefit ratio of bevacizumab in NF2 merit further consideration. WHAT THIS PAPER ADDS: Children with neurofibromatosis type 2 (NF2) may be at increased risk of cerebral vasculopathy and arterial ischaemic stroke. Targeted magnetic resonance angiography should be performed in children with NF2 who are being considered for bevacizumab therapy.
References
Oct 2, 1995·Neuroreport·J O ClaudioG A Rouleau
May 15, 2001·American Journal of Physiology. Cell Physiology·I Zachary
Jul 13, 2005·Neurology·Aisling M RyanJoan T Moroney
Dec 20, 2005·Developmental Medicine and Child Neurology·Nuno J V CordeiroMichael G Pike
Apr 15, 2008·European Journal of Paediatric Neurology : EJPN : Official Journal of the European Paediatric Neurology Society·Joanne NgPeter Baxter
Jun 4, 2011·American Journal of Medical Genetics. Part a·Rachael E HornigoldRosalie E Ferner
Jun 28, 2011·Journal of Neuroimaging : Official Journal of the American Society of Neuroimaging·Gayathri SreedherGiulio Zuccoli
Mar 21, 2012·Neoplasia : an International Journal for Oncology Research·Hon-Kit WongRakesh K Jain
Jul 19, 2012·Otology & Neurotology : Official Publication of the American Otological Society, American Neurotology Society [and] European Academy of Otology and Neurotology·Scott R PlotkinFred G Barker
Aug 13, 2013·Handbook of Clinical Neurology·Simon K W Lloyd, D Gareth R Evans
Jul 16, 2014·PloS One·Pei-Yuan ZuoCheng-Yun Liu
Oct 28, 2015·Developmental Medicine and Child Neurology·Nadine McCreaVijeya Ganesan
Apr 22, 2017·American Journal of Medical Genetics. Part a·Shazia K AfridiRosalie E Ferner
Dec 1, 2016·Neuro-oncology Practice·Katrina A MorrisAllyson Parry
Citations
Jul 3, 2018·Developmental Medicine and Child Neurology·Fenella J KirkhamDawn E Saunders
Jan 23, 2019·Neurological Sciences : Official Journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology·Claudia SantoroSilverio Perrotta
Aug 20, 2019·Current Opinion in Oncology·Dorothy HallidayD Gareth Evans
Apr 18, 2019·Clinical Genetics·Dorothy HallidayUNKNOWN English NF2 research group
Jul 9, 2020·Neurology. Genetics·John W HensonConnor McCormick
Aug 7, 2020·AJR. American Journal of Roentgenology·David DoigKshitij Mankad