Literature DB >> 17301379

Report of six cases of chediak-higashi syndrome with regard to clinical and laboratory findings.

Abolhasan Farhoudi1, Zahra Chavoshzadeh, Zahra Pourpak, Mina Izadyar, Mohammad Gharagozlou, Masoud Movahedi, Asghar Aghamohamadi, Bahram Mirsaeid Ghazi, Mostafa Moin, Nima Rezaei.   

Abstract

Chediak - Higashi Syndrome (CHS) is a rare, primary Immunodeficiency disorder with an autosomal recessive (AR) inheritance and characterized by recurrent infection, partial occulocutaneous albinism and an accelerated phase.In this report we describe clinical and laboratory findings from 6 CHS patients. Clinical and laboratory information of six patients who were referred to our center during the last 20 years (from 1983 - 2003) were reviewed.Onset age of disease was between 3 months to 10 years. All patients had history of consanguineous parents and two patients were siblings. All patients had oculocutaneous albinism, nystagmus, recurrent infections which included upper and lower respiratory tract (U and LRT) infections, stomatitis, thrush, and skin abscesses and hepatitis. In laboratory findings, all patients had neutropenia and normal immunoglobulins and normal CD3, CD4, CD8 and CD19 lymphocyte by flowcytometry and three of the four patients had chemotatic defect. Five patients certainly had giant granule in bone marrow neutrophil and in one patient it was equiovocal. Three patients had an accelerated phase, and for one patient bone marrow transplantation was done that was tolerated well and had been well after 7 years.We emphasize the need for early diagnosis on basis of characteristic facies and diagnostic laboratory examinations and early bone marrow transplantation (BMT) in patients.

Entities:  

Year:  2003        PMID: 17301379     DOI: 02.04/ijaai.189192

Source DB:  PubMed          Journal:  Iran J Allergy Asthma Immunol        ISSN: 1735-1502            Impact factor:   1.464


  6 in total

1.  Chediak-higashi syndrome in accelerated phase: a rare case report with review of literature.

Authors:  Shivani Sood; Biswajit Biswas; Vijay Kaushal; Tanish Mandal
Journal:  Indian J Hematol Blood Transfus       Date:  2014-01-22       Impact factor: 0.900

2.  Chediak-higashi syndrome presented as accelerated phase: case report and review of the literature.

Authors:  Amina Bouatay; Sondes Hizem; Amel Tej; Wided Moatamri; Lamia Boughamoura; Mondher Kortas
Journal:  Indian J Hematol Blood Transfus       Date:  2014-01-31       Impact factor: 0.900

3.  History of primary immunodeficiency diseases in iran.

Authors:  Asghar Aghamohammadi; Mostafa Moin; Nima Rezaei
Journal:  Iran J Pediatr       Date:  2010-03       Impact factor: 0.364

4.  The neuropsychological phenotype of Chediak-Higashi disease.

Authors:  Talia N Shirazi; Joseph Snow; Lillian Ham; Greta B Raglan; Edythe A Wiggs; Angela C Summers; Camilo Toro; Wendy J Introne
Journal:  Orphanet J Rare Dis       Date:  2019-05-06       Impact factor: 4.123

5.  Deleterious mutations in LRBA are associated with a syndrome of immune deficiency and autoimmunity.

Authors:  Gabriela Lopez-Herrera; Giacomo Tampella; Qiang Pan-Hammarström; Peer Herholz; Claudia M Trujillo-Vargas; Kanchan Phadwal; Anna Katharina Simon; Michel Moutschen; Amos Etzioni; Adi Mory; Izhak Srugo; Doron Melamed; Kjell Hultenby; Chonghai Liu; Manuela Baronio; Massimiliano Vitali; Pierre Philippet; Vinciane Dideberg; Asghar Aghamohammadi; Nima Rezaei; Victoria Enright; Likun Du; Ulrich Salzer; Hermann Eibel; Dietmar Pfeifer; Hendrik Veelken; Hans Stauss; Vassilios Lougaris; Alessandro Plebani; E Michael Gertz; Alejandro A Schäffer; Lennart Hammarström; Bodo Grimbacher
Journal:  Am J Hum Genet       Date:  2012-05-17       Impact factor: 11.025

6.  Chediak-Higashi Syndrome: A Case Series from Karnataka, India.

Authors:  Pradeep Rudramurthy; Hemalata Lokanatha
Journal:  Indian J Dermatol       Date:  2015 Sep-Oct       Impact factor: 1.494

  6 in total

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