Literature DB >> 8255649

Osteosclerotic metaphyseal dysplasia.

G Nishimura1, K Kozlowski.   

Abstract

A new sclerosing bone disease in two Japanese siblings born to first-degree cousin parents is reported. Clinically the disease is characterized by early developmental delay, hypotonia and later spastic paraplegia. The unique radiographic changes consist of peripheral osteosclerosis affecting predominantly metaphyses of the long bones and to a lesser degree ends of the ribs and clavicles, iliac crests, acetabulae, ischio-pubic synchondroses and vertebrae. The epiphyses are sclerotic in early life. The round bones, short tubular bones and the skull are little affected. The shafts of the tubular bones are osteopenic. Increased serum alkaline phosphatase was the only laboratory abnormality detected. We suggest the name "osteosclerotic metaphyseal dysplasia" for this disorder.

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Year:  1993        PMID: 8255649     DOI: 10.1007/bf02012448

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


  7 in total

1.  Identification of biallelic LRRK1 mutations in osteosclerotic metaphyseal dysplasia and evidence for locus heterogeneity.

Authors:  Aritoshi Iida; Weirong Xing; Martine K F Docx; Tomoki Nakashima; Zheng Wang; Mamori Kimizuka; Wim Van Hul; Dietz Rating; Jürgen Spranger; Hirohumi Ohashi; Noriko Miyake; Naomichi Matsumoto; Subburaman Mohan; Gen Nishimura; Geert Mortier; Shiro Ikegawa
Journal:  J Med Genet       Date:  2016-04-07       Impact factor: 6.318

Review 2.  Human Genetics of Sclerosing Bone Disorders.

Authors:  Raphaël De Ridder; Eveline Boudin; Geert Mortier; Wim Van Hul
Journal:  Curr Osteoporos Rep       Date:  2018-06       Impact factor: 5.096

Review 3.  Osteosclerotic metaphyseal dysplasia with extensive interstitial pulmonary lesions: a case report and literature review.

Authors:  Helin Zheng; Jinhua Cai; Longlun Wang; Xiaoya He
Journal:  Skeletal Radiol       Date:  2015-06-18       Impact factor: 2.199

4.  Identification of a novel LRRK1 mutation in a family with osteosclerotic metaphyseal dysplasia.

Authors:  Long Guo; Katta M Girisha; Aritoshi Iida; Malavika Hebbar; Anju Shukla; Hitesh Shah; Gen Nishimura; Naomichi Matsumoto; Shifa Nismath; Noriko Miyake; Shiro Ikegawa
Journal:  J Hum Genet       Date:  2016-11-10       Impact factor: 3.172

5.  Deciphering the LRRK code: LRRK1 and LRRK2 phosphorylate distinct Rab proteins and are regulated by diverse mechanisms.

Authors:  Asad U Malik; Athanasios Karapetsas; Raja S Nirujogi; Sebastian Mathea; Deep Chatterjee; Prosenjit Pal; Pawel Lis; Matthew Taylor; Elena Purlyte; Robert Gourlay; Mark Dorward; Simone Weidlich; Rachel Toth; Nicole K Polinski; Stefan Knapp; Francesca Tonelli; Dario R Alessi
Journal:  Biochem J       Date:  2021-02-12       Impact factor: 3.857

6.  SCP2 variant is associated with alterations in lipid metabolism, brainstem neurodegeneration, and testicular defects.

Authors:  Melanie Galano; Shereen Ezzat; Vassilios Papadopoulos
Journal:  Hum Genomics       Date:  2022-08-22       Impact factor: 6.481

7.  A case report of dysosteosclerosis observed from the prenatal period.

Authors:  Kisho Kobayashi; Yusuke Goto; Hiroaki Kise; Hiroaki Kanai; Koji Kodera; Gen Nishimura; Kenji Ohyama; Kanji Sugita; Takayuki Komai
Journal:  Clin Pediatr Endocrinol       Date:  2010-08-31
  7 in total

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