Literature DB >> 23053755

Dysmorphic facies and diffuse posterior spine ankylosis in a patient with unusual form of spondyloenchondrodysplasia (Spranger type IV).

Ali Al Kaissi1, Farid Ben Chehida, Maher Ben Ghachem, Klaus Klaushofer, Franz Grill.   

Abstract

We describe a male patient, who was seen for the first time at the age of 8 years because of short trunk dwarfism. Spine radiographs showed platyspondyly with irregular areas of increased and decreased mineralization (irregular spotted appearance within lytic lesions located along the posterior vertebral bodies of the entire spine). Skeletal survey showed no enchondromatous lesions of the short/long tubular bones. At the age of 17, progressive spine stiffness associated with stooping posture developed. 3DCT scanning showed pathological transformation of the spinal enchondromas into generalized ossification and thickening of the posterior vertebral elements (vertebral laminae, supraspinal, and interspinal ligaments, respectively) causing effectively the development of a diffuse posterior spinal ankylosis. We report what might be a unique subtype of spondyloenchondrodysplasia (Spranger type IV).

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Year:  2012        PMID: 23053755      PMCID: PMC3641239          DOI: 10.1007/s00586-012-2518-2

Source DB:  PubMed          Journal:  Eur Spine J        ISSN: 0940-6719            Impact factor:   3.134


  12 in total

1.  D-2-hydroxyglutaric aciduria in association with spondyloenchondromatosis.

Authors:  I S Talkhani; J Saklatvala; J Dwyer
Journal:  Skeletal Radiol       Date:  2000-05       Impact factor: 2.199

2.  A new case of spondyloenchondrodysplasia with immune dysregulation confirms the pleiotropic nature of the disorder: comment on "A syndrome of immunodeficiency, autoimmunity, and spondylometaphyseal dysplasia" by M.L. Kulkarni, K. Baskar, and P.M. Kulkarni [2006].

Authors:  Raffaele Renella; Andrea Superti-Furga
Journal:  Am J Med Genet A       Date:  2007-06-15       Impact factor: 2.802

3.  Possible heterogeneity in spondyloenchondrodysplasia: quadriparesis, basal ganglia calcifications, and chondrocyte inclusions.

Authors:  M Frydman; J Bar-Ziv; R Preminger-Shapiro; A Brezner; N Brand; T Ben-Ami; R S Lachman; H E Gruber; D L Rimoin
Journal:  Am J Med Genet       Date:  1990-07

4.  Maffucci's syndrome. Report of a case with a review of the literature.

Authors:  I F Anderson
Journal:  S Afr Med J       Date:  1965-11-20

5.  Spondyloenchondrodysplasia.

Authors:  H Menger; K Kruse; J Spranger
Journal:  J Med Genet       Date:  1989-02       Impact factor: 6.318

6.  Generalized enchondromatosis in a boy with only platyspondyly in the father.

Authors:  F Halal; E M Azouz
Journal:  Am J Med Genet       Date:  1991-03-15

7.  Spondyloenchondrodysplasia. Enchondromatomosis with severe platyspondyly in two brothers.

Authors:  S Schorr; C Legum; M Ochshorn
Journal:  Radiology       Date:  1976-01       Impact factor: 11.105

8.  Dysspondylochondromatosis.

Authors:  P Freisinger; G Finidori; P Maroteaux
Journal:  Am J Med Genet       Date:  1993-02-15

9.  Genochondromatosis.

Authors:  M Le Merrer; P Fressinger; P Maroteaux
Journal:  J Med Genet       Date:  1991-07       Impact factor: 6.318

10.  Spondyloenchondrodysplasia: clinical variability in three cases.

Authors:  Beyhan Tüysüz; Müjde Arapoglu; Savaş Ungür
Journal:  Am J Med Genet A       Date:  2004-07-15       Impact factor: 2.802

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