Department of Sports Medicine and Adult Reconstructive Surgery, Drum Tower Hospital, School of Medicine, Nanjing University, Nanjing, Jiangsu 210008, P.R. China.
Laboratory for Bone and Joint Disease, Model Animal Research Center (MARC), Nanjing University, Nanjing, Jiangsu 210093, P.R. China.
Mol Med Rep. 2018 Aug;18(2):1761-1765. doi: 10.3892/mmr.2018.9143. Epub 2018 Jun 6.
Schwartz-Jampel syndrome type 1 (SJS1) is a rare autosomal recessive disease caused by mutations in the gene heparan sulfate proteoglycan 2 (HSPG2; also known as basement membrane‑specific heparin sulfate). In the present study, a 10‑year‑old female SJS1 proband from a Chinese family, who was diagnosed by X‑ray and physical examination, was recruited. The key clinical features of the patient with SJS1 included short stature, joint contractures, pigeon breast, and myotonia that led to progressive stiffness of the face and limbs; barely discernible kyphosis was also noted. Genetic testing using whole exome sequencing and Sanger sequencing was performed for the proband and family members. A total of 2 novel mutations (c.8788G>A; p.Glu2930Lys and c.11671+5G>A) in the HSPG2 gene were identified in the proband. The family members harboring 1 heterozygous mutation in HSPG2 did not exhibit any skeletal abnormalities. The results of the present study suggested that the compound heterozygous mutations in HSPG2 may be responsible the induction of SJS1, and demonstrated the genotype‑phenotype associations between mutations in the HSPG2 gene and clinical characteristics of SJS1.
施瓦茨-詹佩尔综合征 1 型(SJS1)是一种罕见的常染色体隐性遗传病,由硫酸乙酰肝素蛋白聚糖 2 基因(HSPG2;也称为基底膜特异性肝素硫酸)突变引起。本研究招募了一名来自中国家庭的 10 岁女性 SJS1 先证者,其通过 X 射线和体格检查进行诊断。SJS1 患者的主要临床特征包括身材矮小、关节挛缩、鸽胸和肌强直,导致面部和四肢逐渐僵硬;还注意到几乎不可察觉的脊柱后凸。对先证者及其家庭成员进行了全外显子组测序和 Sanger 测序的基因检测。在 HSPG2 基因中发现了共 2 种新的突变(c.8788G>A;p.Glu2930Lys 和 c.11671+5G>A)。携带 HSPG2 中 1 种杂合突变的家庭成员未表现出任何骨骼异常。本研究结果表明,HSPG2 的复合杂合突变可能导致 SJS1 的发生,并证实了 HSPG2 基因中的突变与 SJS1 的临床特征之间的基因型-表型相关性。