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Severe congenital neutropenia mimicking chronic idiopathic neutropenia: a case report.
Kim, Juhyung; Hwang, Soyoon; Hwang, Narae; Lee, Yeonji; Cho, Hee Jeong; Moon, Joon Ho; Sohn, Sang Kyun; Baek, Dong Won.
Afiliación
  • Kim J; Department of Hematology/Oncology, Kyungpook National University Hospital, School of Medicine, Kyungpook National University, Daegu, Korea.
  • Hwang S; Department of Infectious Diseases, Kyungpook National University Chilgok Hospital, School of Medicine, Kyungpook National University, Daegu, Korea.
  • Hwang N; Department of Clinical Pathology, School of Medicine, Kyungpook National University, Daegu, Korea.
  • Lee Y; Department of Laboratory Medicine, Kyungpook National University Chilgok Hospital, Daegu, Korea.
  • Cho HJ; Department of Internal Medicine, Kyungpook National University Hospital, Daegu, Korea.
  • Moon JH; Department of Hematology/Oncology, Kyungpook National University Hospital, School of Medicine, Kyungpook National University, Daegu, Korea.
  • Sohn SK; Department of Hematology/Oncology, Kyungpook National University Chilgok Hospital, School of Medicine, Kyungpook National University, Daegu, Korea.
  • Baek DW; Department of Hematology/Oncology, Kyungpook National University Hospital, School of Medicine, Kyungpook National University, Daegu, Korea.
J Yeungnam Med Sci ; 40(3): 283-288, 2023 Jul.
Article en En | MEDLINE | ID: mdl-35899327
Severe chronic neutropenia is classified as severe congenital, cyclic, autoimmune, or idiopathic. However, there is a lot of uncertainty regarding the diagnosis of severe congenital neutropenia (SCN) and chronic idiopathic neutropenia, and this uncertainty affects further evaluations and treatments. A 20-year-old man presented with fever and knee abrasions after a bicycle accident. On admission, his initial absolute neutrophil count (ANC) was 30/µL. He had no medical history of persistent severe neutropenia with periodic oscillation of ANC. Although his fever resolved after appropriate antibiotic therapy, ANC remained at 80/µL. Bone marrow (BM) aspiration and biopsy were performed, and a BM smear showed myeloid maturation arrest. Moreover, genetic mutation test results showed a heterozygous missense variant in exon 4 of the neutrophil elastase ELANE: c597+1G>C (pV190-F199del). The patient was diagnosed with SCN. After discharge, we routinely checked his ANC level and monitored any signs of infection with minimum use of granulocyte colony-stimulating factor (G-CSF), considering its potential risk of leukemic transformation. Considering that SCN can be fatal, timely diagnosis and appropriate management with G-CSF are essential. We report the case of a patient with SCN caused by ELANE mutation who had atypical clinical manifestations. For a more accurate diagnosis and treatment of severe chronic neutropenia, further studies are needed to elucidate the various clinical features of ELANE.
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Texto completo: 1 Base de datos: MEDLINE Idioma: En Revista: J Yeungnam Med Sci Año: 2023 Tipo del documento: Article

Texto completo: 1 Base de datos: MEDLINE Idioma: En Revista: J Yeungnam Med Sci Año: 2023 Tipo del documento: Article