Matsumoto Tadashi, Tsuru Akira, Amamoto Nagisa, Shimizu Takashi, Kondoh Tatsuro, Saitoh Nakamichi, Tsujii Tomomi, Tamagawa Kimiko
Department of Nursing, Nagasaki University School of Medicine, 1-7-1 Sakamoto, Nagasaki 852-8520, Japan.
J Hum Genet. 2003;48(3):125-9. doi: 10.1007/s100380300019.
The childhood cerebral form of X-linked adrenoleukodystrophy (X-ALD) is a severe congenital metabolic disease without a definite effective therapy except for hematopoietic stem cell transplantation in the appropriate disease stage. Seven Japanese families with X-ALD were analyzed for mutations in the ALD gene ( ALD). Of the seven families, three were referred to us for prenatal diagnosis, four for carrier detection, and three for confirmation diagnosis of patients. By nucleotide sequencing and/or restriction analysis, all the subjects to be examined were successfully diagnosed. Six different missense mutations in ALD were identified. There was a G-->A substitution (G512S) in two unrelated families, and a G-->A (R617H), a C-->T (R660W), a G-->C (R163P), a C-->T (S606L), or a G-->A (G116E) substitution in each of the other five families. Among the six substitutions, five were those reported previously and the other was a novel mutation. In three families, prenatal diagnosis was carried out after genetic counseling.
X连锁肾上腺脑白质营养不良(X-ALD)的儿童脑型是一种严重的先天性代谢疾病,除了在适当的疾病阶段进行造血干细胞移植外,没有明确有效的治疗方法。对7个患有X-ALD的日本家庭进行了ALD基因(ALD)突变分析。在这7个家庭中,3个家庭前来进行产前诊断,4个家庭进行携带者检测,3个家庭进行患者确诊。通过核苷酸测序和/或限制性分析,所有待检测对象均成功确诊。在ALD基因中鉴定出6种不同的错义突变。在两个无亲缘关系的家庭中存在G→A替换(G512S),在其他5个家庭中分别存在G→A(R617H)、C→T(R660W)、G→C(R163P)、C→T(S606L)或G→A(G116E)替换。在这6种替换中,5种是先前报道过的,另一种是新突变。在3个家庭中,经过遗传咨询后进行了产前诊断。