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伴有心内膜炎的嗜酸性粒细胞增多综合征:通过JAK2-V617F突变的下一代测序进行鉴定

Hypereosinophilic Syndrome with Endomyocarditis: Identification by Next-Generation Sequencing of the JAK2-V617F Mutation.

作者信息

Tesfamicael Ruta, Aung Thanda, Domin Lee Thomas, Brahn Ernest

机构信息

Department of Rheumatology, UCLA School of Medicine, Los Angeles, California, United States.

Department of Pathology and Laboratory Medicine, UCLA School of Medicine, Los Angeles, California, United States.

出版信息

Eur J Rheumatol. 2023 Apr;10(2):67-70. doi: 10.5152/eurjrheum.2023.22123.

Abstract

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.

摘要

高嗜酸性粒细胞综合征要求外周血绝对嗜酸性粒细胞计数≥1.5×10⁹ /L,且有可归因于外周或组织嗜酸性粒细胞增多的临床表现。临床表现差异很大,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病例中,患者可能出现严重的、危及生命的器官功能障碍,累及皮肤、肺、心脏、胃肠道和神经系统。本文讨论了一例伴有潜在危及生命的心血管受累的嗜酸性粒细胞增多病例。初步实验室检查显示白细胞计数升高,嗜酸性粒细胞占60%。心内膜活检显示嗜酸性粒细胞性心内膜炎伴肉芽肿、罕见巨细胞,无血管炎、微生物或恶性肿瘤。她的表现符合高嗜酸性粒细胞综合征或嗜酸性肉芽肿性多血管炎的标准。分子遗传学分析显示骨髓增生异常综合征检测板/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)、猫麦克多诺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)第9外显子突变以及T细胞基因重排/聚合酶链反应均为阴性。骨髓活检显示骨髓轻度细胞减少,多系造血,巨核细胞发育异常,局灶性嗜酸性粒细胞增多。未发现原始细胞增多、单克隆B细胞群或离散的全T细胞异常。骨髓细胞遗传学研究显示骨髓增殖性肿瘤检测板/含Sec1家族结构域2(SCFD2)-Numb蛋白X配体(LNX)-血小板衍生生长因子受体α(PDGFRA)荧光原位杂交信号模式正常,核型为46 XX正常。然而,二代测序显示JAK2-V617F突变呈阳性,这在高嗜酸性粒细胞综合征中是一种罕见的分子异常。其患病率约为0%至4%。JAK2点突变导致异常的酪氨酸磷酸化和细胞因子激活增加。该病例显示了高嗜酸性粒细胞增多症中先进诊断方法的复杂性和挑战性,以及在特定亚组中使用酪氨酸激酶抑制剂等靶向治疗的可能性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a5d2/10544436/866ff921dfb9/ejr-10-2-67_f001.jpg

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