Literature DB >> 25835322

Sclerosteosis (craniotubular hyperostosis-syndactyly) with complex hyperphalangy of the index finger.

Hiroko Yagi1, Masaki Takagi, Yukihiro Hasegawa, Hülya Kayserili, Gen Nishimura.   

Abstract

We report a 4-year-old boy with sclerosteosis associated with severe digital dysostosis. The initial medical consultation was prompted by bilateral, asymmetrical syndactyly of the index and middle fingers. The left index finger had complicated phalangeal anomalies: hyperphalangy (supernumerary phalanx distal to the middle phalanx) and hypoplasia with bracket epiphyses of the proximal and middle phalanges. Development of facial nerve palsy, hearing impairment and generalized osteosclerosis had occurred between 3 years and 4 years of age, with the subsequent identification of a homozygous SOST mutation. Bilateral second and third fingers syndactyly associated with abnormal patterning of the same fingers should be considered prodromal signs of sclerosteosis.

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Mesh:

Year:  2015        PMID: 25835322     DOI: 10.1007/s00247-015-3292-1

Source DB:  PubMed          Journal:  Pediatr Radiol        ISSN: 0301-0449


  7 in total

1.  The male phenotype in osteopathia striata congenita with cranial sclerosis.

Authors:  Sarah K Holman; Phil Daniel; Zandra A Jenkins; Rachel L Herron; Tim Morgan; Ravi Savarirayan; C W Chow; Axel Bohring; Annette Mosel; Didier Lacombe; Bernhard Steiner; Thomas Schmitt-Mechelke; Barbara Schroter; Annick Raas-Rothschild; Sixto Garcia Miñaur; Mary Porteous; Michael Parker; Oliver Quarrell; Dagmar Tapon; Valérie Cormier-Daire; Sahar Mansour; Ruth Nash; Laurence A Bindoff; Torunn Fiskerstrand; Stephen P Robertson
Journal:  Am J Med Genet A       Date:  2011-10       Impact factor: 2.802

2.  Increased bone density in sclerosteosis is due to the deficiency of a novel secreted protein (SOST).

Authors:  W Balemans; M Ebeling; N Patel; E Van Hul; P Olson; M Dioszegi; C Lacza; W Wuyts; J Van Den Ende; P Willems; A F Paes-Alves; S Hill; M Bueno; F J Ramos; P Tacconi; F G Dikkers; C Stratakis; K Lindpaintner; B Vickery; D Foernzler; W Van Hul
Journal:  Hum Mol Genet       Date:  2001-03-01       Impact factor: 6.150

Review 3.  Two novel WTX mutations underscore the unpredictability of male survival in osteopathia striata with cranial sclerosis.

Authors:  B Perdu; P Lakeman; G Mortier; R Koenig; A M A Lachmeijer; W Van Hul
Journal:  Clin Genet       Date:  2010-10-18       Impact factor: 4.438

4.  Bone dysplasia sclerosteosis results from loss of the SOST gene product, a novel cystine knot-containing protein.

Authors:  M E Brunkow; J C Gardner; J Van Ness; B W Paeper; B R Kovacevich; S Proll; J E Skonier; L Zhao; P J Sabo; Y Fu; R S Alisch; L Gillett; T Colbert; P Tacconi; D Galas; H Hamersma; P Beighton; J Mulligan
Journal:  Am J Hum Genet       Date:  2001-02-09       Impact factor: 11.025

5.  Bone overgrowth-associated mutations in the LRP4 gene impair sclerostin facilitator function.

Authors:  Olivier Leupin; Elke Piters; Christine Halleux; Shouih Hu; Ina Kramer; Frederic Morvan; Tewis Bouwmeester; Markus Schirle; Manuel Bueno-Lozano; Feliciano J Ramos Fuentes; Peter H Itin; Eveline Boudin; Fenna de Freitas; Karen Jennes; Barbara Brannetti; Nadine Charara; Hilmar Ebersbach; Sabine Geisse; Chris X Lu; Andreas Bauer; Wim Van Hul; Michaela Kneissel
Journal:  J Biol Chem       Date:  2011-04-06       Impact factor: 5.157

6.  Osteopathia striata with cranial sclerosis and developmental delay in a male with a mosaic deletion in chromosome region Xq11.2.

Authors:  Sébastien Chénier; Abdul Noor; Lucie Dupuis; Dimitri J Stavropoulos; Roberto Mendoza-Londono
Journal:  Am J Med Genet A       Date:  2012-09-17       Impact factor: 2.802

7.  Sclerosteosis in children.

Authors:  B J Cremin
Journal:  Pediatr Radiol       Date:  1979-07-24
  7 in total
  2 in total

1.  Craniometaphyseal dysplasia in a 14-month old: a case report and review of imaging differential diagnosis.

Authors:  Sumit Singh; Curtis Qin; Srikanth Medarametla; Shilpa V Hegde
Journal:  Radiol Case Rep       Date:  2016-05-20

2.  A Novel Mutation in a Gene Causes Sclerosteosis in a Family of Mediterranean Origin.

Authors:  Aishah A Ekhzaimy; Ebtihal Y Alyusuf; Meshael Alswailem; Ali S Alzahrani
Journal:  Medicina (Kaunas)       Date:  2022-01-28       Impact factor: 2.430

  2 in total

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