高嗜酸性粒细胞综合征伴心肌炎:JAK2-V617F突变的下一代测序鉴定。

IF 1.3 Q4 RHEUMATOLOGY
Ruta Tesfamicael, Thanda Aung, Thomas Domin Lee, Ernest Brahn
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引用次数: 1

摘要

嗜酸性粒细胞增多综合征要求外周嗜酸性粒绝对计数≥1.5×109/L,临床表现可归因于外周或组织嗜酸性粒增多。临床表现可能差异很大,大多数患者相对无症状,嗜酸性粒细胞增多是偶然发现的。然而,在少数嗜酸性粒细胞增多的病例中,他们可能会出现严重的危及生命的器官功能障碍,影响皮肤、肺、心脏、胃肠道和神经系统。讨论了一例嗜酸性粒细胞增多并可能危及生命的心血管疾病。初步实验室调查显示白细胞计数升高,嗜酸性粒细胞占60%。心内膜心肌活检显示嗜酸性心内膜心肌炎伴肉芽肿、罕见巨细胞,无血管炎、微生物或恶性肿瘤。她的表现符合嗜酸性粒细胞增多综合征或嗜酸性肉芽肿伴多发性脑膜炎的标准。骨髓增生异常综合征面板/血小板衍生生长因子受体β(PDGFRB)(5q32)/成纤维细胞生长因子受体1(FGFR1)荧光原位杂交(FISH)、Feline McDonough肉瘤相关酪氨酸激酶3(FLT3)内部串联重复(ITD)突变、钙调节蛋白(CALR)外显子9突变、,以及T细胞基因重排/聚合酶链式反应。骨髓活检显示轻度低细胞骨髓伴多系造血、+巨核细胞发育不良和局灶性嗜酸性粒细胞增多。没有发现过量的成纤维细胞,没有发现单型B细胞群,也没有发现离散的泛T细胞变异。骨髓细胞遗传学研究显示骨髓增生性肿瘤面板/Sec1家族结构域含2(SCFD2)-Numb蛋白-X配体(LNX)-血小板衍生生长因子受体α(PDGFRA)荧光原位杂交的正常信号模式,正常核型为46XX。然而,下一代测序对JAK2-V617F突变呈阳性,这是嗜酸性粒细胞增多综合征中一种罕见的分子异常。患病率约为0%至4%。JAK2点突变导致酪氨酸磷酸化异常和细胞因子活化增加。该病例证明了嗜酸性粒细胞增多症高级诊断机会的复杂性和挑战性,以及酪氨酸激酶抑制剂等靶向治疗在选定亚群中的潜在用途。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Hypereosinophilic Syndrome with Endomyocarditis: Identification by Next-Generation Sequencing of the JAK2-V617F Mutation.

Hypereosinophilic syndrome requires a peripheral absolute eosinophil count of ≥1.5 × 109 /L with clinical manifestations attributable to peripheral or tissue hypereosinophilia. Clinical manifestations can vary greatly, with the majority of patients relatively asymptomatic and the eosinophilia detected incidentally. However, in a minority of hypereosinophilia cases, they may present with severe lifethreatening organ dysfunction affecting skin, lung, heart, gastrointestinal tract, and nervous system. A case of hypereosinophilia with potentially life-threatening cardiovascular involvement is discussed. Initial laboratory investigations showed an elevated white blood cell count with 60% eosinophils. An endomyocardial biopsy revealed eosinophilic endomyocarditis with granuloma, rare giant cells, and no vasculitis, microorganisms, or malignancy. Her presentation met the criteria for either hypereosinophilic syndrome or eosinophilic granulomatosis with polyangitis. Molecular genetic analysis was negative for myelodysplastic syndrome panel/ Platelet Derived Growth Factor Receptor Beta (PDGFRB) (5q32)/Fibroblast Growth Factor Receptor 1 (FGFR1) Fluorescence In Situ Hybridization (FISH), Feline McDonough Sarcoma-related Tyrosine Kinase 3 (FLT3) Internal Tandem Duplication (ITD) mutation, Calregulin (CALR) exon 9 mutation, and T-cell gene rearrangement/polymerase chain reaction. Bone marrow biopsy revealed a mildly hypocellular marrow with multilineage hematopoiesis,+ megakaryocyte dysplasia, and focal eosinophilia. No excess blasts, no monotypic B-cell population, and no discrete pan T-cell aberrancies were found. Bone marrow cytogenetic studies showed a normal signal pattern for myeloproliferative neoplasms panel/Sec1 Family Domain Containing 2 (SCFD2)-Ligand of Numb Protein-X (LNX)-Platelet-derived Growth Factor Receptor Alpha (PDGFRA) fluorescence in situ hybridization with a normal karyotype of 46 XX. Next-generation sequencing, however, was positive for the JAK2-V617F mutation, a rare molecular abnormality in hypereosinophilic syndrome. The prevalence ranges from approximately 0% to 4%. The JAK2 point mutation leads to aberrant tyrosine phosphorylation and increased cytokine activation. The case demonstrates the complexity and challenging nature of advanced diagnostic opportunities in hypereosinophilia and the potential use, in select subsets, of targeted treatments such as tyrosine kinase inhibitors.

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