Hirshoren Nir, Gross Menachem, Banin Eyal, Sosna Jacob, Bargal Ruth, Raas-Rothschild Annick
Department of Otolaryngology/Head and Neck Surgery, Hadassah Hebrew University Medical Center, 91120 Jerusalem, Israel.
Eur J Med Genet. 2008 Jul-Aug;51(4):351-7. doi: 10.1016/j.ejmg.2008.02.008. Epub 2008 Mar 20.
We report on a new family with Teunissen-Cremers syndrome. The proband presented with congenital conductive hearing loss due to stapes ankylosis and incus short process fixation with skeletal anomalies including symphalangism, broad thumbs and broad first toes, syndactyly, brachydactyly, contractures of the elbows and knees, hyperopia and lens opacities. This constellation of symptoms is compatible with the diagnosis of one of the joint-fusion syndromes namely the Teunissen-Cremers syndrome (TCS), which was first reported in 1990. Mutations in the NOG gene which encodes the noggin protein, a bone morphogenetic protein antagonist, have been identified in TCS as well as in four other autosomal dominant disorders including proximal symphalangism (SYM1), multiple synostosis (SYNS1), Tarsal-Carpal coalition syndrome and brachydactyly type B (BDB). Interestingly, we found that the mutation P35S described in this family has already been reported in patients affected with SYM1 as well as with BDB syndromes.
我们报告了一个患有特尼斯森 - 克雷默斯综合征的新家族。先证者因镫骨强直和砧骨短突固定而出现先天性传导性听力损失,并伴有骨骼异常,包括关节粘连、拇指宽大、第一趾宽大、并指、短指、肘和膝关节挛缩、远视和晶状体混浊。这一系列症状与其中一种关节融合综合征即特尼斯森 - 克雷默斯综合征(TCS)的诊断相符,该综合征于1990年首次报道。在TCS以及其他四种常染色体显性疾病中已鉴定出编码骨形态发生蛋白拮抗剂头蛋白的NOG基因突变,这四种疾病包括近端关节粘连(SYM1)、多发性骨连接(SYNS1)、跗腕骨联合综合征和B型短指症(BDB)。有趣的是,我们发现这个家族中描述的P35S突变已在患有SYM1以及BDB综合征的患者中报道过。