| Literature DB >> 26254864 |
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.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