Literature DB >> 9799296

Phalangeal cone-shaped epiphyses of the hand: their natural history, diagnostic sensitivity, and specificity in cartilage hair hypoplasia and the trichorhinophalangeal syndromes I and II.

A Giedion1.   

Abstract

Phalangeal cone-shaped epiphyses are an ideal object for the radiologist to study with temporal reasoning, to examine their shape, diagnostic usefulness, natural history and effect on pathophysiology. Radiographs of the hands of 60 patients with cartilage hair hypoplasia (CHH), 69 with trichorhinophalangeal syndrome I (TRP I) and 11 with TRP II were examined, including 26 longitudinal observations. The early phases of cone development were recorded. In CHH a characteristic mesophalangeal type of cone in 42/47 patients of the "Age Suitable for Classification" (ASC) was detected. All 46 TRP I and 9 TRP II patients in the ASC had the previously described mesophalangeal cone type 12. However, 1/4 of these TRP I and all TRP II cases presented a milder variant of type 12: type 12 A. A complex sequence of secondary changes in the proximal interphalangeal joints in TRP I leading eventually to subluxation was recorded. It can be concluded that in the ASC the cones described are highly sensitive but not specific diagnostic indicators. In infancy and early childhood, other phalangeal changes were found, which may be of diagnostic help.

Entities:  

Mesh:

Year:  1998        PMID: 9799296     DOI: 10.1007/s002470050460

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


  10 in total

Review 1.  Development of the long bones in the hands and feet of children: radiographic and MR imaging correlation.

Authors:  Tal Laor; Jeffrey P Clarke; Hong Yin
Journal:  Pediatr Radiol       Date:  2016-01-21

2.  Langer-Giedion syndrome: the evolving imaging features in hands and beyond.

Authors:  Wai Kan Tsang; Kwok Wai Michael Yang; Chi Ming Fong
Journal:  Skeletal Radiol       Date:  2013-09-27       Impact factor: 2.199

3.  Genotypic and phenotypic spectrum in tricho-rhino-phalangeal syndrome types I and III.

Authors:  H J Lüdecke; J Schaper; P Meinecke; P Momeni; S Gross; H Hirche; M J Abramowicz; B Albrecht; C Apacik; H J Christen; U Claussen; K Devriendt; E Fastnacht; A Forderer; U Friedrich; T H Goodship; M Greiwe; H Hamm; R C Hennekam; G K Hinkel; M Hoeltzenbein; H Kayserili; F Majewski; M Mathieu; R McLeod; A T Midro; U Moog; T Nagai; N Niikawa; K H Orstavik; E Plöchl; C Seitz; J Schmidtke; L Tranebjaerg; M Tsukahara; B Wittwer; B Zabel; G Gillessen-Kaesbach; B Horsthemke
Journal:  Am J Hum Genet       Date:  2000-12-07       Impact factor: 11.025

4.  Clinical and genetic distinction of Schimke immuno-osseous dysplasia and cartilage-hair hypoplasia.

Authors:  Alireza Baradaran-Heravi; Christian Thiel; Anita Rauch; Martin Zenker; Cornelius F Boerkoel; Ilkka Kaitila
Journal:  Am J Med Genet A       Date:  2008-08-01       Impact factor: 2.802

5.  Deletion of the GATA domain of TRPS1 causes an absence of facial hair and provides new insights into the bone disorder in inherited tricho-rhino-phalangeal syndromes.

Authors:  Talat H Malik; Dietrich Von Stechow; Roderick T Bronson; Ramesh A Shivdasani
Journal:  Mol Cell Biol       Date:  2002-12       Impact factor: 4.272

6.  Homozygous mutations in IHH cause acrocapitofemoral dysplasia, an autosomal recessive disorder with cone-shaped epiphyses in hands and hips.

Authors:  Jan Hellemans; Paul J Coucke; Andres Giedion; Anne De Paepe; Peter Kramer; Frits Beemer; Geert R Mortier
Journal:  Am J Hum Genet       Date:  2003-03-11       Impact factor: 11.025

Review 7.  Brachydactyly E: isolated or as a feature of a syndrome.

Authors:  Arrate Pereda; Intza Garin; Maria Garcia-Barcina; Blanca Gener; Elena Beristain; Ane Miren Ibañez; Guiomar Perez de Nanclares
Journal:  Orphanet J Rare Dis       Date:  2013-09-12       Impact factor: 4.123

8.  Clinical and molecular diagnosis of a cartilage-hair hypoplasia with IGF-1 deficiency.

Authors:  Inma Castilla-Cortázar; Julieta Rodríguez De Ita; Irene Martín-Estal; Fabiola Castorena; Gabriel A Aguirre; Rocío García de la Garza; Martha I Elizondo
Journal:  Am J Med Genet A       Date:  2016-11-14       Impact factor: 2.802

9.  Trichorhinophalangeal Syndrome Type I: A Patient with Two Novel and Different Mutations in the TRPS1 Gene.

Authors:  Catarina Dias; Lara Isidoro; Mafalda Santos; Helena Santos; Jorge Sales Marques
Journal:  Case Rep Genet       Date:  2013-04-17

10.  Cartilage hair hypoplasia: characteristics and orthopaedic manifestations.

Authors:  Patrick Riley; Dennis S Weiner; Bonnie Leighley; David Jonah; D Holmes Morton; Kevin A Strauss; Michael B Bober; Martin S Dicintio
Journal:  J Child Orthop       Date:  2015-03-13       Impact factor: 1.548

  10 in total

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