Literature DB >> 24791932

Clinicoradiological correlation of scoliosis in children with Jarcho-Levin and Escobar syndromes: associated "flat bone or wing-like" imaging findings.

Thangamadhan Bosemani1, Andrea Poretti, Jane E Benson, Andreas Meyer-Heim, Thierry A G M Huisman.   

Abstract

UNLABELLED: Congenital or early onset scoliosis may be the lead clinical feature in several rare syndromes. In this paper, we present the imaging findings in two children with early onset scoliosis related to the Jarcho-Levin and Escobar syndromes and an osseous plate or wing-like bar extending along the posterior elements of the spine on computed tomography. The clinical phenotypes in these syndromes are variable. A thorough clinical evaluation with imaging correlation is essential. The recognition of underlying spinal anomalies is essential in planning treatment and estimating prognosis. In young children with progressive scoliosis, cross-sectional imaging plays a major role in the diagnostic work-up.
CONCLUSION: Congenital scoliosis requires a comprehensive clinical evaluation and imaging work-up. The presence of an osseous plate or wing-like fusion of posterior elements of the spine may suggest the diagnosis of Jarcho-Levin and Escobar syndromes.

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Year:  2014        PMID: 24791932     DOI: 10.1007/s00431-014-2324-4

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  11 in total

1.  Mutations in the embryonal subunit of the acetylcholine receptor (CHRNG) cause lethal and Escobar variants of multiple pterygium syndrome.

Authors:  Neil V Morgan; Louise A Brueton; Phillip Cox; Marie T Greally; John Tolmie; Shanaz Pasha; Irene A Aligianis; Hans van Bokhoven; Tamas Marton; Lihadh Al-Gazali; Jenny E V Morton; Christine Oley; Colin A Johnson; Richard C Trembath; Han G Brunner; Eamonn R Maher
Journal:  Am J Hum Genet       Date:  2006-06-20       Impact factor: 11.025

2.  Patterns and progression in congenital scoliosis.

Authors:  G H Shahcheraghi; M H Hobbi
Journal:  J Pediatr Orthop       Date:  1999 Nov-Dec       Impact factor: 2.324

3.  Multiple pterygium syndrome.

Authors:  V Escobar; D Bixler; S Gleiser; D D Weaver; T Gibbs
Journal:  Am J Dis Child       Date:  1978-06

4.  Clinical and radiological distinction between spondylothoracic dysostosis (Lavy-Moseley syndrome) and spondylocostal dysostosis (Jarcho-Levin syndrome).

Authors:  Walter E Berdon; Brooke S Lampl; Alberto S Cornier; Norman Ramirez; Peter D Turnpenny; Michael G Vitale; Leonard P Seimon; Robert A Cowles
Journal:  Pediatr Radiol       Date:  2010-12-22

Review 5.  Segmental costovertebral malformations: association with neural tube defects. Report of 3 cases and review of the literature.

Authors:  S Duru; S Ceylan; B H Güvenç
Journal:  Pediatr Neurosurg       Date:  1999-05       Impact factor: 1.162

6.  Prevalence and patterns of scoliosis in children with multiple pterygium syndrome.

Authors:  Sunyoung Joo; Kenneth J Rogers; Maureen Donohoe; Marilyn Marnie King; Shanmuga Jay Kumar
Journal:  J Pediatr Orthop       Date:  2012-03       Impact factor: 2.324

7.  Health and function of patients with untreated idiopathic scoliosis: a 50-year natural history study.

Authors:  Stuart L Weinstein; Lori A Dolan; Kevin F Spratt; Kirk K Peterson; Mark J Spoonamore; Ignacio V Ponseti
Journal:  JAMA       Date:  2003-02-05       Impact factor: 56.272

Review 8.  Jarcho-Levin syndrome: four new cases and classification of subtypes.

Authors:  P S Karnes; D Day; S A Berry; M E Pierpont
Journal:  Am J Med Genet       Date:  1991-09-01

9.  Jarcho-Levin syndrome presenting as neural tube defect: report of four cases and pitfalls of diagnosis.

Authors:  Banu Dane; Cem Dane; Figen Aksoy; Ahmet Cetin; Murat Yayla
Journal:  Fetal Diagn Ther       Date:  2007-07-24       Impact factor: 2.587

10.  Limb pterygium syndromes: a review and report of eleven patients.

Authors:  J G Hall; S D Reed; K N Rosenbaum; J Gershanik; H Chen; K M Wilson
Journal:  Am J Med Genet       Date:  1982-08
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