Literature DB >> 19050402

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

Sarah F Smithson1, David Grier, Christine M Hall.   

Abstract

We describe the clinical and radiological features of spondylo-meta-epiphyseal dysplasia, short limb-abnormal calcification type (OMIM 271665) in a 7-year-old boy from Pakistan. The key clinical features of this condition include disproportionate short stature with short limbs, a narrow chest with pectus excavatum, brachydactyly in the hands and feet, a characteristic craniofacial appearance and developmental delay in some but not all patients. The radiological findings are distinctive and comprise short long bones throughout the skeleton with striking epiphyses that are stippled, flattened and fragmented and flared, irregular metaphyses. Platyspondyly in the spine with wide intervertebral spaces is observed and some vertebral bodies are pear-shaped with central humps, anterior protrusions and posterior scalloping. In the patient, we describe that the pelvic sacroiliac notch was narrow, several acetabular spurs were seen and there were several ossification defects in the pelvic bones. This form of spondylo-meta-epiphyseal dysplasia has been reported in 14 patients previously. The occurrence of affected siblings with normal parents in three families and consanguinity on three occasions indicates autosomal recessive inheritance.

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Year:  2009        PMID: 19050402     DOI: 10.1097/MCD.0b013e3283189762

Source DB:  PubMed          Journal:  Clin Dysmorphol        ISSN: 0962-8827            Impact factor:   0.816


  7 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.  Identification of a novel homozygous mutation in the DDR2 gene from a patient with spondylo-meta-epiphyseal dysplasia by whole exome sequencing.

Authors:  Masoud Heidari; Morteza Soleyman-Nejad; Alireza Isazadeh; Mohammad Hossein Taskiri; Manzar Bolhassani; Nahid Sadighi; Zahra Shiri; Zahra Karimi; Mansour Heidari
Journal:  Iran J Basic Med Sci       Date:  2021-02       Impact factor: 2.699

7.  A novel mutation in DDR2 causing spondylo-meta-epiphyseal dysplasia with short limbs and abnormal calcifications (SMED-SL) results in defective intra-cellular trafficking.

Authors:  Adila Al-Kindi; Praseetha Kizhakkedath; Huifang Xu; Anne John; Abeer Al Sayegh; Anuradha Ganesh; Maha Al-Awadi; Lamya Al-Anbouri; Lihadh Al-Gazali; Birgit Leitinger; Bassam R Ali
Journal:  BMC Med Genet       Date:  2014-04-11       Impact factor: 2.103

  7 in total

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