Literature DB >> 19002453

The time of onset of abnormal calcification in spondylometaepiphyseal dysplasia, short limb-abnormal calcification type.

Beyhan Tüysüz1, Nurperi Gazioğlu, Savaş Ungür, Dolly Yafet Aji, Seval Türkmen.   

Abstract

A 1-month-old boy with shortness of extremities on prenatal US was referred to our department with a provisional diagnosis of achondroplasia. His height was normal but he had short extremities and platyspondyly, premature carpal epiphyses on both hands, and short tubular bones with irregular metaphyses on radiographs. Re-evaluation of the patient at the age of 1 year revealed very short height and premature calcification of the costal cartilages and epiphyses. Spondylometaepiphyseal dysplasia (SMED), short limb-abnormal calcification type was diagnosed. This condition is a very rare autosomal recessively inherited disorder, and most of the patients die in early childhood due to neurological involvement. At the age of 2 years and 5 months, a CT scan showed narrowing of the cervical spinal canal. One month later he died suddenly because of spinal cord injury. In conclusion early diagnosis is very important because the recurrence risk is high and patients may die due to early neurological complications. The time of onset of abnormal calcifications, a diagnostic finding of the disease, is at the age of around 1 year in most patients. When abnormal calcifications are not yet present, but radiological changes associated with SMED are present, this rare disease must be considered.

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Year:  2008        PMID: 19002453     DOI: 10.1007/s00247-008-1036-1

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


  4 in total

1.  Spondylo-meta-epiphyseal dysplasia, short limbs, abnormal calcification type: a new case with severe neurological involvement.

Authors:  V Fano; H Lejarraga; C Barreiro
Journal:  Pediatr Radiol       Date:  2001-01

2.  Further delineation of spondylo-meta-epiphyseal dysplasia, short limb-abnormal calcification type, with emphasis on diagnostic features.

Authors:  L O Langer; B J Wolfson; C I Scott; C S Reid; D V Schidlow; E A Millar; P F Borns; J P Lubicky; B L Carpenter
Journal:  Am J Med Genet       Date:  1993-02-15

3.  Spondylo-meta-epiphyseal dysplasia, short limb, abnormal calcification type.

Authors:  L I al-Gazali; D Bakalinova; L Sztriha
Journal:  Clin Dysmorphol       Date:  1996-07       Impact factor: 0.816

4.  Spondylo-meta-epiphyseal dysplasia (SMED), short limb-hand type: a congenital familial skeletal dysplasia with distinctive features and histopathology.

Authors:  Z Borochowitz; L O Langer; H E Gruber; R Lachman; M B Katznelson; D L Rimoin
Journal:  Am J Med Genet       Date:  1993-02-01
  4 in total
  6 in total

1.  Spondyloepimetaphyseal dysplasia, short limb-abnormal calcifications type: progressive radiological findings from fetal age to adolescence.

Authors:  Katya Rozovsky; Jacob Sosna; Martine Le Merrer; Natalia Simanovsky; Benjamin Z Koplewitz; Jacob Bar-Ziv; Valerie Cormier-Daire; Annick Raas-Rothschild
Journal:  Pediatr Radiol       Date:  2011-08-05

2.  Further expansion of the mutational spectrum of spondylo-meta-epiphyseal dysplasia with abnormal calcification.

Authors:  Gizem Ürel-Demir; Pelin Ozlem Simsek-Kiper; Özlem Akgün-Doğan; Rahşan Göçmen; Zheng Wang; Naomichi Matsumoto; Noriko Miyake; Gülen Eda Utine; Gen Nishimura; Shiro Ikegawa; Koray Boduroglu
Journal:  J Hum Genet       Date:  2018-06-08       Impact factor: 3.172

3.  Report of a Novel Homozygous Nonsense DDR2 Mutation in an Indian Adult Male with Spondylo-meta-epiphyseal Dysplasia, Short Limb-Abnormal Calcification Type.

Authors:  Neerja Gupta; Alec Reginald Errol Correa; Manisha Jana; Madhulika Kabra
Journal:  J Pediatr Genet       Date:  2019-03-12

4.  Spondylometaepiphyseal Dysplasia Short Limb-Abnormal Calcification Type in Turkish Patients Reveals a Novel Mutation and New Features.

Authors:  Elif Yilmaz Gulec; Bassam R Ali; Anne John; Beyhan Tuysuz
Journal:  Mol Syndromol       Date:  2021-09-28

5.  Trafficking defects and loss of ligand binding are the underlying causes of all reported DDR2 missense mutations found in SMED-SL patients.

Authors:  Bassam R Ali; Huifang Xu; Nadia A Akawi; Anne John; Noushad S Karuvantevida; Ruth Langer; Lihadh Al-Gazali; Birgit Leitinger
Journal:  Hum Mol Genet       Date:  2010-03-10       Impact factor: 6.150

6.  Mutations in DDR2 gene cause SMED with short limbs and abnormal calcifications.

Authors:  Ruth Bargal; Valerie Cormier-Daire; Ziva Ben-Neriah; Martine Le Merrer; Jacob Sosna; Judith Melki; David H Zangen; Sarah F Smithson; Zvi Borochowitz; Ruth Belostotsky; Annick Raas-Rothschild
Journal:  Am J Hum Genet       Date:  2008-12-24       Impact factor: 11.025

  6 in total

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