Literature DB >> 16715218

Terminal phalangeal accessory ossification center of the thumb: an additional radiographic finding in Larsen syndrome.

Yasemin Alanay1, Gulen Eda Utine, Ralph S Lachman, Deborah Krakow, Ergul Tuncbilek.   

Abstract

Larsen syndrome is an autosomal-dominant disorder characterized by multiple joint dislocations, vertebral anomalies and dysmorphic facies. Both autosomal-dominant and autosomal-recessive forms of the disorder have been proposed. Individuals with autosomal-dominant Larsen syndrome have characteristic "cylindrical-shape" thumbs caused by broad, shortened phalanges. Autosomal-dominant Larsen syndrome results from heterozygosity for mutations in filamin B, a cytoskeletal protein involved in multicellular processes. We report here a patient with a duplicated or accessory distal thumb phalanx and multiple large joint dislocations who was shown to be heterozygous for a filamin B mutation predicting the amino acid substitution G1691S. This adds a new radiographic finding, duplicated or accessory distal phalanx, to the radiographic abnormalities seen in this rare dominant disorder.

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Mesh:

Year:  2006        PMID: 16715218     DOI: 10.1007/s00247-006-0217-z

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


  9 in total

1.  Multiple congenital dislocations associated with characteristic facial abnormality.

Authors:  L J LARSEN; E R SCHOTTSTAEDT; F C BOST
Journal:  J Pediatr       Date:  1950-10       Impact factor: 4.406

2.  Radiographic findings in Larsen's syndrome.

Authors:  K Kozlowski; F Robertson; R Middleton
Journal:  Australas Radiol       Date:  1974-09

3.  [Larsen's syndrome: congenital dislocation of the knees and other joints, distinctive facies, and, frequently, cleft palate].

Authors:  F N Silverman
Journal:  Ann Radiol (Paris)       Date:  1972 Mar-Apr

4.  Larsen's syndrome: a skeletal dysplasia with multiple joint dislocations and unusual facies.

Authors:  R J Latta; C B Graham; J Aase; S M Scham; D W Smith
Journal:  J Pediatr       Date:  1971-02       Impact factor: 4.406

5.  Cervical kyphosis associated with anteroposterior dissociation and quadriparesis in Larsen's syndrome.

Authors:  Danielle A Katz; John E Hall; John B Emans
Journal:  J Pediatr Orthop       Date:  2005 Jul-Aug       Impact factor: 2.324

6.  The Larsen syndrome. The diagnostic contribution of the analysis of the metacarpophalangeal pattern profile.

Authors:  L De Smet; E Legius; G Fabry; J P Fryns
Journal:  Genet Couns       Date:  1993

7.  Multiple congenital dislocations associated with other skeletal anomalies (Larsen's syndrome) in three siblings.

Authors:  H H Steel; E J Kohl
Journal:  J Bone Joint Surg Am       Date:  1972-01       Impact factor: 5.284

8.  Mutations in the gene encoding filamin B disrupt vertebral segmentation, joint formation and skeletogenesis.

Authors:  Deborah Krakow; Stephen P Robertson; Lily M King; Timothy Morgan; Eiman T Sebald; Cristina Bertolotto; Sebastian Wachsmann-Hogiu; Dora Acuna; Sandor S Shapiro; Toshiro Takafuta; Salim Aftimos; Chong Ae Kim; Helen Firth; Carlos E Steiner; Valerie Cormier-Daire; Andrea Superti-Furga; Luisa Bonafe; John M Graham; Arthur Grix; Carlos A Bacino; Judith Allanson; Martin G Bialer; Ralph S Lachman; David L Rimoin; Daniel H Cohn
Journal:  Nat Genet       Date:  2004-02-29       Impact factor: 38.330

Review 9.  Larsen's syndrome: review of the literature and analysis of thirty-eight cases.

Authors:  J M Laville; P Lakermance; F Limouzy
Journal:  J Pediatr Orthop       Date:  1994 Jan-Feb       Impact factor: 2.324

  9 in total
  2 in total

1.  A molecular and clinical study of Larsen syndrome caused by mutations in FLNB.

Authors:  Louise S Bicknell; Claire Farrington-Rock; Yousef Shafeghati; Patrick Rump; Yasemin Alanay; Yves Alembik; Navid Al-Madani; Helen Firth; Mohammad Hassan Karimi-Nejad; Chong Ae Kim; Kathryn Leask; Melissa Maisenbacher; Ellen Moran; John G Pappas; Paolo Prontera; Thomy de Ravel; Jean-Pierre Fryns; Elizabeth Sweeney; Alan Fryer; Sheila Unger; L C Wilson; Ralph S Lachman; David L Rimoin; Daniel H Cohn; Deborah Krakow; Stephen P Robertson
Journal:  J Med Genet       Date:  2006-06-26       Impact factor: 6.318

2.  Case report: Congenital knee dislocation in a patient with larsen syndrome and a novel filamin B mutation.

Authors:  Matthew B Dobbs; Stephanie Boehm; Dorothy K Grange; Christina A Gurnett
Journal:  Clin Orthop Relat Res       Date:  2008-03-06       Impact factor: 4.176

  2 in total

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