Literature DB >> 9557886

Three new cases of spondylocarpotarsal synostosis syndrome: clinical and radiographic studies.

K E Coêlho1, E S Ramos, T M Felix, L Martelli, J M de Pina-Neto, N Niikawa.   

Abstract

Spondylocarpotarsal synostosis syndrome (SSS) or congenital synspondylism is a recently delineated clinical entity. At least 15 patients have been reported. We present 3 new patients, 2 of whom were sibs born to first-cousin parents. All of our patients had multiple synostoses involving cervical, thoracic and/or lumbar vertebral bodies and carpal/tarsal bones, scoliosis/lordosis, and short stature. Sensorineural deafness was found in 2 of the 3 patients. Analysis of clinical manifestations suggests clinical variability and genetic heterogeneity in SSS. Of a total of 18 SSS patients, 10 were five pairs of sibs from five families, with first-cousin consanguinity of parents in 3, indicating that at least one type of SS is an autosomal-recessive disorder.

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Year:  1998        PMID: 9557886     DOI: 10.1002/(sici)1096-8628(19980428)77:1<12::aid-ajmg3>3.0.co;2-n

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  7 in total

1.  Recessive Spondylocarpotarsal Synostosis Syndrome Due to Compound Heterozygosity for Variants in MYH3.

Authors:  Sophia R Cameron-Christie; Constance F Wells; Marleen Simon; Marja Wessels; Candy Z N Tang; Wenhua Wei; Riku Takei; Coranne Aarts-Tesselaar; Sarah Sandaradura; David O Sillence; Marie-Pierre Cordier; Hermine E Veenstra-Knol; Matteo Cassina; Kathrin Ludwig; Eva Trevisson; Melanie Bahlo; David M Markie; Zandra A Jenkins; Stephen P Robertson
Journal:  Am J Hum Genet       Date:  2018-05-24       Impact factor: 11.025

2.  Spondylocarpotarsal synostosis syndrome: MRI evaluation of vertebral and disk malformation.

Authors:  Magnus Breitling; Edmond G Lemire; Michael Rabin
Journal:  Pediatr Radiol       Date:  2006-06-08

3.  Spondylocarpotarsal synostosis syndrome (with a posterior midline unsegmented bar).

Authors:  A Al Kaissi; M Ben Ghachem; N Nassib; F Ben Chehida; K Kozlowski
Journal:  Skeletal Radiol       Date:  2004-11-16       Impact factor: 2.199

4.  Disruption of the Flnb gene in mice phenocopies the human disease spondylocarpotarsal synostosis syndrome.

Authors:  Claire Farrington-Rock; Veneta Kirilova; Lisa Dillard-Telm; Alexander D Borowsky; Sara Chalk; Matthew J Rock; Daniel H Cohn; Deborah Krakow
Journal:  Hum Mol Genet       Date:  2007-07-17       Impact factor: 6.150

5.  Spondylocarpotarsal synostosis: long-term follow-up of a case due to FLNB mutations.

Authors:  Nicola Brunetti-Pierri; Valentina Esposito; Daniele De Brasi; Dario Maria Mattiacci; Deborah Krakow; Brendan Lee; Mariacarolina Salerno
Journal:  Am J Med Genet A       Date:  2008-05-01       Impact factor: 2.802

6.  Atlanto-axial rotatory fixation in a girl with Spondylocarpotarsal synostosis syndrome.

Authors:  Ali Al Kaissi; Farid Ben Chehida; Hassan Gharbi; Maher Ben Ghachem; Franz Grill; Klaus Klaushofer
Journal:  Scoliosis       Date:  2006-10-16

7.  Spondylocarpotarsal synostosis syndrome due to a novel loss of function FLNB variant: a case report.

Authors:  Samina Yasin; Outi Makitie; Sadaf Naz
Journal:  BMC Musculoskelet Disord       Date:  2021-01-06       Impact factor: 2.362

  7 in total

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