Lloyd Simon K W, Evans D Gareth R
Salford Royal NHS Foundation Trust, Salford, and Department of Otolaryngology, Manchester Academic Health Science Centre, Central Manchester NHS Foundation Trust, Manchester Royal Infirmary, Manchester, UK.
Handb Clin Neurol. 2013;115:957-67. doi: 10.1016/B978-0-444-52902-2.00054-0.
Neurofibromatosis type 2 (NF2) is an autosomal dominant inherited tumor predisposition syndrome caused by mutations in the NF2 gene on chromosome 22. Affected individuals develop schwannomas typically involving both vestibular nerves leading to hearing loss and eventual deafness. Rehabilitation with brainstem implants and in some cases cochlear implants is improving this outcome. Schwannomas also occur on other cranial nerves, on spinal nerve roots and peripheral nerves, and intracutaneously as plaques. Cranial and spinal meningiomas and spinal ependymomas are other common tumors. Fifty to sixty percent of patients represent de novo mutations and as many as 33% of these are mosaic for the underlying disease causing mutation. Truncating mutations (nonsense, frameshift insertions/deletions) are the most frequent germline events and cause the most severe disease, whilst single and multiple exon deletions are common and are usually associated with milder NF2. Neurological deficits are a major feature of the condition and neurologists have a pivotal role in assigning symptoms to lesions and in managing neuropathies. NF2 represents a difficult management problem with most patients facing substantial morbidity and reduced life expectancy. Surgery remains the focus of current management although watchful waiting and occasionally radiation treatment have a role. We are seeing the advent of tailored drug therapies aimed at the genetic level and these are likely to provide huge improvements for this devastating, life-limiting condition.
2型神经纤维瘤病(NF2)是一种常染色体显性遗传的肿瘤易感综合征,由22号染色体上的NF2基因突变引起。受影响的个体通常会发展为神经鞘瘤,累及双侧前庭神经,导致听力丧失并最终失聪。脑干植入物以及在某些情况下的人工耳蜗植入物康复治疗正在改善这种情况。神经鞘瘤也会出现在其他颅神经、脊神经根和周围神经上,并以斑块形式出现在皮肤内。颅和脊髓的脑膜瘤以及脊髓室管膜瘤是其他常见肿瘤。50%至60%的患者存在新发突变,其中多达33%的患者存在导致潜在疾病的镶嵌突变。截短突变(无义突变、移码插入/缺失)是最常见的种系事件,会导致最严重的疾病,而单个和多个外显子缺失则较为常见,通常与较轻的NF2相关。神经功能缺损是该病的主要特征,神经科医生在将症状归因于病变以及管理神经病变方面起着关键作用。NF2是一个难以管理的问题,大多数患者面临严重的发病率和预期寿命缩短的问题。手术仍然是当前治疗的重点,尽管观察等待以及偶尔的放射治疗也有一定作用。我们正在见证针对基因水平的定制药物疗法的出现,这些疗法可能会为这种毁灭性的、限制生命的疾病带来巨大改善。