Briggs M D, Rasmussen I M, Weber J L, Yuen J, Reinker K, Garber A P, Rimoin D L, Cohn D H
Ahmanson Department of Pediatrics, Cedars-Sinai Medical Center, Los Angeles, California 90048.
Genomics. 1993 Dec;18(3):656-60. doi: 10.1016/s0888-7543(05)80369-6.
Pseudoachondroplasia (PSACH) is a dominantly inherited form of short-limb dwarfism characterized by dysplastic changes in the spine, epiphyses, and metaphyses and early onset osteoarthropathy. Chondrocytes from affected individuals accumulate an unusual appearing material in the rough endoplasmic reticulum, which has led to the hypothesis that a structural abnormality in a cartilage-specific protein produces the phenotype. We recently identified a large family with a mild form of pseudoachondroplasia. By genetic linkage to a dinucleotide repeat polymorphic marker (D19S199), we have localized the disease gene to chromosome 19 (maximum lod score of 7.09 at a recombination fraction of 0.03). Analysis of additional markers and recombinants between the linked markers and the phenotype suggests that the disease gene resides within a 6.3-cM interval in the immediate pericentromeric region of the chromosome.
假性软骨发育不全(PSACH)是一种常染色体显性遗传的短肢侏儒症,其特征为脊柱、骨骺和干骺端发育异常以及早期骨关节炎。患病个体的软骨细胞在内质网中积累一种外观异常的物质,这导致了一种假说,即软骨特异性蛋白的结构异常产生了该表型。我们最近发现了一个患有轻度假性软骨发育不全的大家族。通过与二核苷酸重复多态性标记(D19S199)进行基因连锁分析,我们已将该疾病基因定位到19号染色体上(在重组率为0.03时,最大对数优势得分为7.09)。对其他标记以及连锁标记与表型之间的重组体进行分析表明,该疾病基因位于染色体着丝粒周围紧邻区域的一个6.3厘摩区间内。