严重先天性中性粒细胞减少模仿慢性特发性中性粒细胞减少:1例报告。

IF 1 Q3 MEDICINE, GENERAL & INTERNAL
Juhyung Kim, Soyoon Hwang, Narae Hwang, Yeonji Lee, Hee Jeong Cho, Joon Ho Moon, Sang Kyun Sohn, Dong Won Baek
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引用次数: 0

摘要

严重慢性中性粒细胞减少症分为严重先天性、周期性、自身免疫性或特发性。然而,关于重度先天性中性粒细胞减少症(SCN)和慢性特发性中性粒细胞减少症的诊断存在很多不确定性,这种不确定性影响了进一步的评估和治疗。一名20岁男子在自行车事故后出现发烧和膝盖擦伤。入院时,患者初始绝对中性粒细胞计数(ANC)为30/µL。患者无持续性严重中性粒细胞减少及ANC周期性振荡病史。虽然在适当的抗生素治疗后发烧消退,但ANC仍保持在80/µL。行骨髓穿刺和活检,骨髓涂片显示骨髓成熟停止。此外,基因突变检测结果显示中性粒细胞弹性酶ELANE外显子4杂合错义变异:c597+1G>C (pV190-F199del)。患者被诊断为SCN。出院后,考虑到白血病转化的潜在风险,我们常规检查他的ANC水平并监测任何感染迹象,同时尽量减少使用粒细胞集落刺激因子(G-CSF)。考虑到SCN可能是致命的,及时诊断和适当的G-CSF治疗是必不可少的。我们报告了一例由ELANE突变引起的SCN患者,其临床表现不典型。为了更准确地诊断和治疗重度慢性中性粒细胞减少症,需要进一步研究阐明ELANE的各种临床特征。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

Severe congenital neutropenia mimicking chronic idiopathic neutropenia: a case report.

Severe congenital neutropenia mimicking chronic idiopathic neutropenia: a case report.

Severe congenital neutropenia mimicking chronic idiopathic neutropenia: a case report.

Severe congenital neutropenia mimicking chronic idiopathic neutropenia: a case report.

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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