Literature DB >> 29884795

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

Gizem Ürel-Demir1, Pelin Ozlem Simsek-Kiper2, Özlem Akgün-Doğan2, Rahşan Göçmen3, Zheng Wang4,5, Naomichi Matsumoto6, Noriko Miyake6, Gülen Eda Utine2, Gen Nishimura7, Shiro Ikegawa4, Koray Boduroglu2.   

Abstract

Spondylo-meta-epiphyseal dysplasia, short limb-abnormal calcification type, is a rare autosomal recessive disorder of the skeleton characterized by disproportionate short stature with narrow chest and dysmorphic facial features. The skeletal manifestations include platyspondyly, short flared ribs, short tubular bones with abnormal metaphyses and epiphyses, severe brachydactyly, and premature stippled calcifications in the cartilage. The abnormal calcifications are so distinctive as to point to the definitive diagnosis. However, they may be too subtle to attract diagnostic attention in infancy. Homozygous variants in DDR2 cause this disorder. We report on a 5-year-old girl with the classic phenotype of SMED, SL-AC in whom a novel homozygous nonsense mutation in DDR2 was detected using exome sequencing.

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Year:  2018        PMID: 29884795     DOI: 10.1038/s10038-018-0473-4

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  16 in total

1.  The discoidin domain receptor DDR2 is a receptor for type X collagen.

Authors:  Birgit Leitinger; Alvin P L Kwan
Journal:  Matrix Biol       Date:  2006-05-26       Impact factor: 11.583

2.  Novel DDR2 mutation identified by whole exome sequencing in a Moroccan patient with spondylo-meta-epiphyseal dysplasia, short limb-abnormal calcification type.

Authors:  Maria Mansouri; Hülya Kayserili; Siham Chafai Elalaoui; Gen Nishimura; Aritoshi Iida; Jaber Lyahyai; Noriko Miyake; Naomichi Matsumoto; Abdelaziz Sefiani; Shiro Ikegawa
Journal:  Am J Med Genet A       Date:  2015-10-13       Impact factor: 2.802

3.  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

4.  The collagen receptor DDR2 regulates proliferation and its elimination leads to dwarfism.

Authors:  J P Labrador; V Azcoitia; J Tuckermann; C Lin; E Olaso; S Mañes; K Brückner; J L Goergen; G Lemke; G Yancopoulos; P Angel; C Martínez; R Klein
Journal:  EMBO Rep       Date:  2001-05       Impact factor: 8.807

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

Authors:  Sarah F Smithson; David Grier; Christine M Hall
Journal:  Clin Dysmorphol       Date:  2009-01       Impact factor: 0.816

6.  Sudden death in spondylo-meta-epiphyseal dysplasia, short limb-abnormal calcification type.

Authors:  Cristina Dias; Robyn Cairns; Millan S Patel
Journal:  Clin Dysmorphol       Date:  2009-01       Impact factor: 0.816

7.  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

8.  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

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

Authors:  Beyhan Tüysüz; Nurperi Gazioğlu; Savaş Ungür; Dolly Yafet Aji; Seval Türkmen
Journal:  Pediatr Radiol       Date:  2008-11-11

10.  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

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  4 in total

1.  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

2.  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

3.  The collagen receptor, discoidin domain receptor 2, functions in Gli1-positive skeletal progenitors and chondrocytes to control bone development.

Authors:  Fatma F Mohamed; Chunxi Ge; Randy T Cowling; Daniel Lucas; Shawn A Hallett; Noriaki Ono; Abdul-Aziz Binrayes; Barry Greenberg; Renny T Franceschi
Journal:  Bone Res       Date:  2022-02-09       Impact factor: 13.362

Review 4.  Cranial Base Synchondrosis: Chondrocytes at the Hub.

Authors:  Shawn A Hallett; Wanida Ono; Renny T Franceschi; Noriaki Ono
Journal:  Int J Mol Sci       Date:  2022-07-15       Impact factor: 6.208

  4 in total

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