Robinson R, Curran J L, Hall W J, Halsall P J, Hopkins P M, Markham A F, Stewart A D, West S P, Ellis F R
Department of Genetics, The University, Leeds, UK.
J Med Genet. 1998 Mar;35(3):196-201. doi: 10.1136/jmg.35.3.196.
Malignant hyperthermia (MH) is an autosomal dominant genetic condition that presents in susceptible people undergoing general anaesthesia. The clinical disorder is a major cause of anaesthetic morbidity and mortality. The UK Malignant Hyperthermia Group has performed genetic linkage analysis on 20 large, well defined malignant hyperthermia families, using hypervariable markers on chromosome 19q13.1, including the candidate MH gene RYR1, the gene coding for the skeletal muscle ryanodine receptor protein. The results were analysed using LINKAGE to perform two point and multipoint lod scores, then HOMOG to calculate levels of heterogeneity. The results clearly showed genetic heterogeneity between MH families; nine of the families gave results entirely consistent with linkage to the region around RYR1 while the same region was clearly excluded in three families. In the remaining eight MHS families there were single recombinant events between RYR1 and MH susceptibility. HOMOG analysis was of little added benefit in determining the likelihood of linkage to RYR1 in these families. This confirmation of the presence of heterogeneity in the UK MH population, along with the possibility of the presence of two MH genes in some pedigrees, indicates that it would be premature and potentially dangerous to offer diagnosis of MH by DNA based methods at this time.
恶性高热(MH)是一种常染色体显性遗传疾病,在接受全身麻醉的易感人群中出现。这种临床病症是麻醉并发症和死亡的主要原因。英国恶性高热研究小组对20个大型、明确界定的恶性高热家族进行了基因连锁分析,使用位于19号染色体q13.1上的高变标记,包括候选MH基因RYR1,即编码骨骼肌兰尼碱受体蛋白的基因。使用LINKAGE软件分析结果以进行两点和多点对数优势分数计算,然后用HOMOG软件计算异质性水平。结果清楚地显示了MH家族之间的基因异质性;其中9个家族的结果与RYR1周围区域的连锁完全一致,而在3个家族中该区域被明确排除。在其余8个MH易感家族中,RYR1与MH易感性之间存在单个重组事件。HOMOG分析在确定这些家族与RYR1连锁的可能性方面几乎没有额外的益处。英国MH人群中异质性的这一证实,以及某些家系中可能存在两个MH基因表明,此时通过基于DNA的方法进行MH诊断还为时过早且可能存在危险。