Wimmer Katharina, Mühlbauer Manfred, Eckart Markus, Callens Tom, Rehder Helga, Birkner Thomas, Leroy Jules G, Fonatsch Christa, Messiaen Ludwine
Institut für Medizinische Biologie, University of Vienna, Austria.
Eur J Hum Genet. 2002 May;10(5):334-8. doi: 10.1038/sj.ejhg.5200807.
Spinal neurofibromas are found in up to 38% of NF1 patients. However, they cause clinical implications only in about 5% of the patients. In contrast, multiple symptomatic spinal neurofibromas are the main clinical finding in patients with familial spinal neurofibromatosis. Familial spinal neurofibromatosis has been considered to be a distinct clinical form of neurofibromatosis. Linkage analysis in two families and identification of a NF1 gene mutation in a third family strongly associate spinal neurofibromatosis with the NF1 gene. We describe a NF1 patient who satisfies the NIH diagnostic criteria and has severe spinal involvement with bilateral spinal root neurofibromas at every level. A recurrent splice site mutation (IVS19b-3C>G) was identified in the NF1 gene in the patient. We discuss the possibility that the clinical picture of this patient represents an additional example of spinal neurofibromatosis. By comparison of the clinical expression of NF1 in this patient and that in another patient with the identical mutation the hypothesis that spinal neurofibromatosis is associated with a particular mutation is highly unlikely. The involvement of other genes linked to the NF1 gene or modifying genes is currently the most likely explanation for the clinical phenotype of spinal neurofibromatosis.
高达38%的1型神经纤维瘤病(NF1)患者存在脊髓神经纤维瘤。然而,它们仅在约5%的患者中引起临床症状。相比之下,多发性有症状脊髓神经纤维瘤是家族性脊髓神经纤维瘤病患者的主要临床发现。家族性脊髓神经纤维瘤病被认为是神经纤维瘤病的一种独特临床类型。对两个家族进行连锁分析,并在第三个家族中鉴定出NF1基因突变,这有力地证明了脊髓神经纤维瘤病与NF1基因相关。我们描述了一名符合美国国立卫生研究院(NIH)诊断标准且脊髓严重受累、各节段均有双侧脊神经根神经纤维瘤的NF1患者。在该患者的NF1基因中鉴定出一个反复出现的剪接位点突变(IVS19b-3C>G)。我们讨论了该患者的临床表现可能代表脊髓神经纤维瘤病另一个实例的可能性。通过比较该患者与另一名具有相同突变的患者中NF1的临床表型,脊髓神经纤维瘤病与特定突变相关的假设极不可能成立。与NF1基因连锁的其他基因或修饰基因的参与目前是脊髓神经纤维瘤病临床表型最可能的解释。