Literature DB >> 21475513

Ciliopathy with special emphasis on kartageners syndrome.

Ashfaq Ul Hassan1, Ghulam Hassan, Sajad Hamid Khan, Zahida Rasool, Afeera Abida.   

Abstract

Cilia are hair-like structures extending from the cell membrane, perform diverse biological functions. Primary defects in the structure and function of sensory and motile cilia result in multiple ciliopathies. The most prominent genetic abnormality involving motile cilia is primary ciliary dyskinesia (PCD) or Kartageners syndrome. PCD is a rare, usually autosomal recessive, genetically heterogeneous disorder characterized by sino-pulmonary disease, laterality defects and male infertility. One of the important components of cilia is the Dynein. Ciliary ultrastructural defects are identified in approximately 90% of PCD patients and involve the outer dynein arms, inner dynein arms, or both. Diagnosing PCD is challenging and requires a compatible clinical phenotype together with tests such as ciliary ultrastructural analysis, immunofluorescent staining, ciliary beat assessment, and/or nasal nitric oxide measurements. Increased understanding of the pathogenesis will aid in better diagnosis and treatment of PCD. The aim of the article is to present the basic defect involved in the etiology of this interesting syndrome.

Entities:  

Keywords:  Axoneme; Ciliopathy; Laterality defect; Primary ciliary dyskinesia; Situs Inversus

Year:  2009        PMID: 21475513      PMCID: PMC3068795     

Source DB:  PubMed          Journal:  Int J Health Sci (Qassim)        ISSN: 1658-3639


  17 in total

Review 1.  Asymmetry of cilia and of mice and men.

Authors:  B A Afzelius
Journal:  Int J Dev Biol       Date:  1999-07       Impact factor: 2.203

Review 2.  Cilia and disease.

Authors:  Lorraine Eley; Laura M Yates; Judith A Goodship
Journal:  Curr Opin Genet Dev       Date:  2005-06       Impact factor: 5.578

3.  Sequence analysis of 21 genes located in the Kartagener syndrome linkage region on chromosome 15q.

Authors:  Maciej Geremek; Frederieke Schoenmaker; Ewa Zietkiewicz; Andrzej Pogorzelski; Scott Diehl; Cisca Wijmenga; Michal Witt
Journal:  Eur J Hum Genet       Date:  2008-02-13       Impact factor: 4.246

4.  Relationship between asymmetric nodal expression and the direction of embryonic turning.

Authors:  J Collignon; I Varlet; E J Robertson
Journal:  Nature       Date:  1996-05-09       Impact factor: 49.962

5.  Prolonged airway retention of insoluble particles in cystic fibrosis versus primary ciliary dyskinesia.

Authors:  J A Regnis; K L Zeman; P G Noone; M R Knowles; W D Bennett
Journal:  Exp Lung Res       Date:  2000 Apr-May       Impact factor: 2.459

6.  Ultrastructural abnormalities of bronchial cilia in children with recurrent airway infections and bronchiectasis.

Authors:  L Corbeel; F Cornillie; J Lauweryns; M Boel; G van den Berghe
Journal:  Arch Dis Child       Date:  1981-12       Impact factor: 3.791

7.  Kartagener's syndrome. A blinded, controlled study of cilia ultrastructure.

Authors:  R D Eavey; J B Nadol; L B Holmes; N M Laird; A Lapey; M P Joseph; M Strome
Journal:  Arch Otolaryngol Head Neck Surg       Date:  1986-06

8.  Genetic aspects of immotile cilia syndrome.

Authors:  J M Sturgess; M W Thompson; E Czegledy-Nagy; J A Turner
Journal:  Am J Med Genet       Date:  1986-09

9.  A molecular pathway determining left-right asymmetry in chick embryogenesis.

Authors:  M Levin; R L Johnson; C D Stern; M Kuehn; C Tabin
Journal:  Cell       Date:  1995-09-08       Impact factor: 41.582

10.  Conserved left-right asymmetry of nodal expression and alterations in murine situs inversus.

Authors:  L A Lowe; D M Supp; K Sampath; T Yokoyama; C V Wright; S S Potter; P Overbeek; M R Kuehn
Journal:  Nature       Date:  1996-05-09       Impact factor: 49.962

View more
  2 in total

1.  A human ciliopathy with polycystic ovarian syndrome and multiple subcutaneous cysts: A rare case report.

Authors:  Kangan Tan; Peng Liu; Lili Pang; Wanna Yang; Fengqin Hou
Journal:  Medicine (Baltimore)       Date:  2018-12       Impact factor: 1.817

2.  Basal body multipotency and axonemal remodelling are two pathways to a 9+0 flagellum.

Authors:  R J Wheeler; E Gluenz; K Gull
Journal:  Nat Commun       Date:  2015-12-15       Impact factor: 14.919

  2 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.