Literature DB >> 31903681

Three new patients with Steel syndrome and a Puerto Rican specific COL27A1 mutation.

Louise Amlie-Wolf1, Sue Moyer-Harasink1, Ann-Marie Carr2, Philip Giampietro3, Adele Schneider4, Mitchell Simon5.   

Abstract

Steel syndrome was initially described by H. H. Steel in 1993 in Puerto Rico, at which time he described the clinical findings required for diagnosis. The responsible gene, COL27A1, was identified in 2015 (Gonzaga-Jauregui et al., European Journal of Human Genetics, 2015;23:342-346). Eleven patients have previously been described with Steel syndrome and homozygous COL27A1 mutations, with eight having an apparent founder mutation, p.Gly697Arg. We describe three more patients identified at Einstein Medical Center Philadelphia and St. Christopher's Hospital for Children (Philadelphia, PA) diagnosed with Steel syndrome. All three are of Puerto Rican ancestry with the previously described founder mutation and had either hip dislocations or hip dysplasia. Radial head dislocation was only identified in one patient while short stature and scoliosis were noted in two of these patients. There are now 51 patients in the literature with Steel syndrome, including the 3 patients in this article, and 14 patients with a genetically confirmed Steel syndrome diagnosis.
© 2020 Wiley Periodicals, Inc.

Entities:  

Keywords:  COL27A1; congenital hip dislocation; radial head dislocation

Year:  2020        PMID: 31903681     DOI: 10.1002/ajmg.a.61465

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  3 in total

1.  Histopathology of recurrent Steel syndrome in fetuses caused by novel variants of COL27A1 gene.

Authors:  Gerard Frigola; Olga Gómez Del Rincón; Virginia Borobio Florián; Anna Vallmajó Fita; Berta Campos; Montse Pauta; Maria Segura Puimedon; Rafael Oliva; Antoni Borrell; Alfons Nadal
Journal:  Virchows Arch       Date:  2021-01-07       Impact factor: 4.064

2.  Mutations in COL1A1 and COL27A1 Associated with a Pectus Excavatum Phenotype in 2 Siblings with Osteogenesis Imperfecta.

Authors:  Nelimar Cruz-Centeno; Jean F Saenz-Maisonet; Paola M López-Dones; Alberto Santiago-Cornier; Victor N Ortiz-Justiniano
Journal:  Am J Case Rep       Date:  2022-05-18

3.  Biallelic novel mutations of the COL27A1 gene in a patient with Steel syndrome.

Authors:  Jong Seop Kim; Hyoungseok Jeon; Hyeran Lee; Jung Min Ko; Yonghwan Kim; Murim Choi; Gen Nishimura; Ok-Hwa Kim; Tae-Joon Cho
Journal:  Hum Genome Var       Date:  2021-05-07
  3 in total

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