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神经纤维瘤病2型:一个大家族的临床及DNA连锁研究

Neurofibromatosis 2: clinical and DNA linkage studies of a large kindred.

作者信息

Wertelecki W, Rouleau G A, Superneau D W, Forehand L W, Williams J P, Haines J L, Gusella J F

机构信息

Department of Medical Genetics, University of South Alabama, Mobile 36688.

出版信息

N Engl J Med. 1988 Aug 4;319(5):278-83. doi: 10.1056/NEJM198808043190505.

Abstract

At least eight provisional categories of neurofibromatosis have been proposed. Among these, neurofibromatosis 1 (von Recklinghausen's disease or peripheral neurofibromatosis) and neurofibromatosis 2 (central or bilateral acoustic neurofibromatosis) have been established as distinct disorders. We studied 15 affected male and 8 affected female members of one large kindred with neurofibromatosis 2. None of the patients met the diagnostic criteria for neurofibromatosis 1. Between the ages of 15 and 53 years, the patients had multiple central nervous system tumors of various types--mainly, bilateral acoustic neuromas. Two or more tumors eventually developed in 20 of the patients; 9 had evidence of only bilateral acoustic neuromas. Meningiomas and ependymomas were more common among the young patients; those who initially presented with acoustic neuromas were nearly a decade older. Intracranial nontumoral calcifications were present in most patients and were also found in symptom-free children. The presence of such lesions is probably a prodromic feature of neurofibromatosis 2. Simultaneous analysis of D22S1 and IGLV DNA markers for coinheritance with neurofibromatosis 2 indicates that the locus for the disease is near the center of the long arm of chromosome 22 (22q11.1----22q13.1). The eventual isolation of this disease gene may reveal a cause of the most common intracranial tumors in humans.

摘要

至少已提出了八种类别的神经纤维瘤病暂定分类。其中,神经纤维瘤病1型(冯雷克林霍增氏病或外周神经纤维瘤病)和神经纤维瘤病2型(中枢性或双侧听神经纤维瘤病)已被确认为不同的疾病。我们研究了一个患有神经纤维瘤病2型的大家族中的15名患病男性成员和8名患病女性成员。所有患者均不符合神经纤维瘤病1型的诊断标准。在15至53岁之间,这些患者患有多种类型的中枢神经系统肿瘤——主要是双侧听神经瘤。20名患者最终出现了两个或更多肿瘤;9名患者仅有双侧听神经瘤的证据。脑膜瘤和室管膜瘤在年轻患者中更为常见;最初表现为听神经瘤的患者年龄要大近十岁。大多数患者存在颅内非肿瘤性钙化,在无症状儿童中也发现了这种钙化。这些病变的存在可能是神经纤维瘤病2型的前驱特征。对D22S1和IGLV DNA标记与神经纤维瘤病2型的共遗传进行同步分析表明,该疾病的基因座位于22号染色体长臂的中心附近(22q11.1----22q13.1)。最终分离出这种疾病基因可能会揭示人类最常见颅内肿瘤的病因。

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