Literature DB >> 16205875

[Chediak-Higashi syndrome].

J Wolf1, C Jacobi, H Breer, A Grau.   

Abstract

Chediak-Higashi syndrome (CHS) is a rare autosomal recessive lysosomal disorder characterized by frequent infections, oculocutaneous albinism, high bleeding tendency, and various neurological symptoms. Onset in early childhood mostly leads to lymphohistiocytic infiltration into multiple organs, which is usually lethal without bone marrow transplantation. The adult form of CHS has a milder course, no lymphohistiocytic infiltration, and is characterized by neurological manifestations such as polyneuropathy, parkinsonism, dementia, and ataxia. In young adults, a combination of these defects with oculocutaneous albinism or recurrent infections should bring CHS into consideration. Diagnosis is established by the presence of characteristic eosinophilic peroxidase-positive giant granules in leukocytes. This article summarizes current knowledge about the pathogenesis, clinical course, and therapy of CHS and reports on experience with two adult CHS patients.

Entities:  

Mesh:

Year:  2006        PMID: 16205875     DOI: 10.1007/s00115-005-1989-3

Source DB:  PubMed          Journal:  Nervenarzt        ISSN: 0028-2804            Impact factor:   1.214


  30 in total

1.  Protein truncation test of LYST reveals heterogenous mutations in patients with Chediak-Higashi syndrome.

Authors:  S Certain; F Barrat; E Pastural; F Le Deist; J Goyo-Rivas; N Jabado; M Benkerrou; R Seger; E Vilmer; G Beullier; K Schwarz; A Fischer; G de Saint Basile
Journal:  Blood       Date:  2000-02-01       Impact factor: 22.113

Review 2.  Analysis of the lysosomal storage disease Chediak-Higashi syndrome.

Authors:  D M Ward; G M Griffiths; J C Stinchcombe; J Kaplan
Journal:  Traffic       Date:  2000-11       Impact factor: 6.215

3.  Peripheral neuropathy in the Chediak-Higashi syndrome.

Authors:  V P Misra; R H King; A E Harding; J R Muddle; P K Thomas
Journal:  Acta Neuropathol       Date:  1991       Impact factor: 17.088

4.  Dictyostelium LvsB mutants model the lysosomal defects associated with Chediak-Higashi syndrome.

Authors:  Edward Harris; Ning Wang; Wei-l Wu Wl; Alisha Weatherford; Arturo De Lozanne; James Cardelli
Journal:  Mol Biol Cell       Date:  2002-02       Impact factor: 4.138

5.  Chédiak-Higashi-Steinbrinck syndrome (CHS) in a 27-year-old woman--effects of G-CSF treatment.

Authors:  M Baldus; V Zunftmeister; G Geibel-Werle; B Claus; D Mewes; M Uppenkamp; T Nebe
Journal:  Ann Hematol       Date:  1999-07       Impact factor: 3.673

6.  Defective lysosomal exocytosis and plasma membrane repair in Chediak-Higashi/beige cells.

Authors:  Chau Huynh; Doris Roth; Diane M Ward; Jerry Kaplan; Norma W Andrews
Journal:  Proc Natl Acad Sci U S A       Date:  2004-11-22       Impact factor: 11.205

7.  Chediak-Higashi syndrome: four cases from Northern Finland.

Authors:  M Möttönen; M Lanning; P Baumann; U M Saarinen-Pihkala
Journal:  Acta Paediatr       Date:  2003-09       Impact factor: 2.299

Review 8.  Chediak-Higashi Syndrome: a rare disorder of lysosomes and lysosome related organelles.

Authors:  Shelly L Shiflett; Jerry Kaplan; Diane McVey Ward
Journal:  Pigment Cell Res       Date:  2002-08

9.  Evidence that Griscelli syndrome with neurological involvement is caused by mutations in RAB27A, not MYO5A.

Authors:  Yair Anikster; Marjan Huizing; Paul D Anderson; Diana L Fitzpatrick; Aharon Klar; Eva Gross-Kieselstein; Yackov Berkun; Gila Shazberg; William A Gahl; Haggit Hurvitz
Journal:  Am J Hum Genet       Date:  2002-06-07       Impact factor: 11.025

10.  Chédiak-Higashi syndrome. Neurologic appearance.

Authors:  R E Pettit; K G Berdal
Journal:  Arch Neurol       Date:  1984-09
View more
  2 in total

Review 1.  Periodontal and other oral manifestations of immunodeficiency diseases.

Authors:  M E Peacock; R M Arce; C W Cutler
Journal:  Oral Dis       Date:  2016-10-10       Impact factor: 3.511

2.  Parkinsonism and Other Movement Disorders Associated with Chediak-Higashi Syndrome: Case Report and Systematic Literature Review.

Authors:  Bettina Balint; Kailash P Bhatia
Journal:  Mov Disord Clin Pract       Date:  2015-01-14
  2 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.