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伴有 Kohlschütter-Tönz 综合征的软组织综合征。

SOFT syndrome with kohlschutter-Tonz syndrome.

机构信息

Growth and Pediatric Endocrinology Unit, Hirabai Cowasji Jehangir Medical Research Institute, Jehangir Hospital, Pune, Maharashtra, India.

Department of Neurology, CNS Hospital, Solapur, Maharashtra, India.

出版信息

J Postgrad Med. 2024 Jan-Mar;70(1):56-59. doi: 10.4103/jpgm.jpgm_1001_22.

Abstract

We report a 2.2 year-old-boy, born of consanguineous marriage, referred for short stature, with history of neonatal death and skeletal deformities in his older sibling. Rhizo-mesomelic dwarfism was detected antenatally. Within 24 hours of birth, he developed multiple seizures. Examination revealed severe short stature, dolichocephaly, broad forehead, deep set eyes, low set ears, bulbous nose, small, irregular teeth, pointed chin, and triangular facies. He had rhizomelic shortening, stubby fingers, pes planus, and scanty hair. Neurological evaluation revealed ataxia, hypotonia, and global developmental delay. Skeletal survey radiograph revealed shallow acetabuli, short femurs and humerus, short, broad metacarpals and short cone-shaped phalanges with cupping of phalangeal bases. Clinical exome analysis revealed homozygous mutations involving the POC1A gene and the SLC13A5 gene responsible for SOFT syndrome and Kohlschutter-Tonz syndrome respectively, which were inherited from the parents. Both these syndromes are extremely rare, and their co-occurrence is being reported for the first time.

摘要

我们报告了一例 2.2 岁男孩,其父母为近亲结婚,因身材矮小就诊,其兄姊有新生儿死亡和骨骼畸形病史。产前发现短肢-中胚层发育不良。出生后 24 小时内,他出现多次癫痫发作。检查发现严重身材矮小、长头畸形、宽额、深凹眼、低位耳、球状鼻、小而不规则的牙齿、尖下巴和三角面。他有短肢性缩短、短指、扁平足和稀疏的头发。神经学评估显示共济失调、张力减退和全面发育迟缓。骨骼显像显示髋臼浅、股骨和肱骨短、掌骨短而宽、锥形短指、指骨基底杯状凹陷。临床外显子组分析显示 POC1A 基因和 SLC13A5 基因的纯合突变,分别导致 SOFT 综合征和 Kohlschutter-Tonz 综合征,这些突变均由父母遗传而来。这两种综合征都极为罕见,它们的同时发生尚属首次报道。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a358/10947733/20a08134400c/JPGM-70-56-g001.jpg

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