Literature DB >> 26254864

Chediak-Higashi syndrome presenting in accelerated phase: A case report and literature review.

I Maaloul1, J Talmoudi2, I Chabchoub2, L Ayadi3, T H Kamoun2, T Boudawara4, C H Kallel5, M Hachicha2.   

Abstract

Chediak-Higashi syndrome (CHS) is a rare autosomal recessive lysosomal disorder characterized by frequent infections, oculocutaneous albinism, bleeding diathesis, and progressive neurologic deterioration. In 85% of cases, CHS patients develop the accelerated phase characterized by pancytopenia, high fever, and lymphohistiocytic infiltration of liver, spleen, and lymph nodes. Treatment of accelerated-phase CHS is difficult and the prognosis is poor. Here, we report a case of CHS in a 2-year-old boy who presented in the accelerated phase of the disease. CHS diagnosis was made on the basis of clinical characteristics, hair analysis, and identification of pathognomonic giant azurophilic granules in peripheral blood and bone marrow.
Copyright © 2015 King Faisal Specialist Hospital & Research Centre. Published by Elsevier Ltd. All rights reserved.

Entities:  

Keywords:  Accelerated phase; Chediak–Higashi syndrome; Hematology; Treatment

Mesh:

Year:  2015        PMID: 26254864     DOI: 10.1016/j.hemonc.2015.07.002

Source DB:  PubMed          Journal:  Hematol Oncol Stem Cell Ther


  5 in total

1.  The lysosomal trafficking regulator is necessary for normal wound healing.

Authors:  Jacob C Zbinden; Gabriel J M Mirhaidari; Kevin M Blum; Andrew J Musgrave; James W Reinhardt; Christopher K Breuer; Jenny C Barker
Journal:  Wound Repair Regen       Date:  2021-11-27       Impact factor: 3.617

2.  Accelerated Phase of Chediak-Higashi Syndrome at Initial Presentation: A Case Report of an Uncommon Occurrence in a Rare Disorder.

Authors:  Pooja Jaiswal; Yogesh Kumar Yadav; Nilam Bhasker; Rashmi Kushwaha
Journal:  J Clin Diagn Res       Date:  2015-12-01

3.  Novel MRI Finding of Bilateral Globus Pallidal Involvement in Accelerated Phase of Chédiak -Higashi Syndrome.

Authors:  Minal V Kekatpure; Venkatraman Bhat
Journal:  Ann Indian Acad Neurol       Date:  2020 Jan-Feb       Impact factor: 1.383

4.  Case series on Silvery Hair Syndromes: Single Center Experience.

Authors:  Sirisharani Siddiahgari; Santosh Kumar Soma; Chandravathi Penmetcha; Sandhya Vaddadi; Varshini Bandi; Lokesh Lingappa
Journal:  Indian J Dermatol       Date:  2022 Mar-Apr       Impact factor: 1.757

Review 5.  Making the Most of the Host; Targeting the Autophagy Pathway Facilitates Staphylococcus aureus Intracellular Survival in Neutrophils.

Authors:  Emilio G Vozza; Michelle E Mulcahy; Rachel M McLoughlin
Journal:  Front Immunol       Date:  2021-06-16       Impact factor: 7.561

  5 in total

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