Literature DB >> 29901129

Novel HSPG2 mutations causing Schwartz‑Jampel syndrome type 1 in a Chinese family: A case report.

Wenjin Yan1, Jin Dai1, Dongquan Shi1, Xingquan Xu1, Xiao Han1, Zhihong Xu1, Dongyang Chen1, Huajiang Teng2, Qing Jiang1.   

Abstract

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.

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Year:  2018        PMID: 29901129     DOI: 10.3892/mmr.2018.9143

Source DB:  PubMed          Journal:  Mol Med Rep        ISSN: 1791-2997            Impact factor:   2.952


  1 in total

1.  Whole exome sequencing of fetal structural anomalies detected by ultrasonography.

Authors:  Hiromi Aoi; Takeshi Mizuguchi; Toshifumi Suzuki; Shintaro Makino; Yuka Yamamoto; Jun Takeda; Yojiro Maruyama; Rie Seyama; Shiori Takeuchi; Yuri Uchiyama; Yoshiteru Azuma; Kohei Hamanaka; Atsushi Fujita; Eriko Koshimizu; Satoko Miyatake; Satomi Mitsuhashi; Atsushi Takata; Noriko Miyake; Satoru Takeda; Atsuo Itakura; Naomichi Matsumoto
Journal:  J Hum Genet       Date:  2020-11-03       Impact factor: 3.172

  1 in total

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