Eng C, Murday V, Seal S, Mohammed S, Hodgson S V, Chaudary M A, Fentiman I S, Ponder B A, Eeles R A
Department of Pathology, University of Cambridge, UK.
J Med Genet. 1994 Jun;31(6):458-61. doi: 10.1136/jmg.31.6.458.
Cowden syndrome is an autosomal dominant condition of multiple hamartomas. Patients with this phakomatosis have an increased risk of breast cancer and thyroid tumours. Lhermitte-Duclos disease is usually a sporadic condition of cerebellar ganglion cell hypertrophy, ataxia, mental retardation, and self-limited seizure disorder. We describe a three generation family with Cowden syndrome and Lhermitte-Duclos disease. Karyotyping performed on the peripheral lymphocytes of the proband and her affected mother showed a 46,XX complement. Single strand conformational polymorphism analysis failed to show any germline p53 mutations as a cause of the syndrome in this family.
考登综合征是一种常染色体显性遗传的多发性错构瘤病症。患有这种 phakomatosis 的患者患乳腺癌和甲状腺肿瘤的风险增加。Lhermitte-Duclos 病通常是一种散发的小脑神经节细胞肥大、共济失调、智力迟钝和自限性癫痫症病症。我们描述了一个患有考登综合征和 Lhermitte-Duclos 病的三代家族。对先证者及其患病母亲的外周淋巴细胞进行核型分析显示为 46,XX 核型。单链构象多态性分析未能显示该家族中作为该综合征病因的任何种系 p53 突变。