Hassankrishnamurthy Shruthimurthy, Mody Mayur D, Kota Vamsi K
Department of Hematology/Oncology, JSS Medical College, Mysore, Karnataka, India.
Department of Internal Medicine, Atlanta Veterans Affairs Medical Center, Atlanta, GA, USA.
Am J Case Rep. 2019 Jan 3;20:10-14. doi: 10.12659/AJCR.911854.
BACKGROUND Essential thrombocythemia (ET) is one of the BCR-ABL gene fusion negative chronic myeloproliferative disorders (MPDs), which also include polycythemia vera (PV), and myelofibrosis. Few clinical cases have reported the progression of ET to chronic myelogenous leukemia (CML) with the expression of the BCR-ABL gene. This report describes such a case and includes a review of other reported cases of CML co-occurring with BCR-ABL-negative chronic MPDs. CASE REPORT A 49-year-old woman was diagnosed with ET in 2007. Cytogenetic testing was negative for expression of the JAK2 or BCR-ABL1 genes. Eight years later, in January 2015, she presented with excessive fatigue, poor appetite, unintentional weight loss, a white blood cell (WBC) count of 24,700 per mL, hemoglobin of 9.9 g/dl, and a platelet count of 557,000 per mL, with blasts and basophils in the blood film. Cytogenetic analysis with fluorescent in situ hybridization (FISH) confirmed a 9: 22 chromosomal translocation (Philadelphia chromosome), and quantitative reverse transcription polymerase chain reaction (qRT-PCR) detected the expression of the BCR-ABL gene, confirming a diagnosis of CML. In February 2015, first-line therapy commenced with nilotinib, which was changed to imatinib after three months. During the following nine months, qRT-PCR confirmed a trend to deep molecular remission (MR5). However, she developed early myelofibrosis, and myelosuppressive therapy was resumed. CONCLUSIONS This rare case highlights the importance of cytogenetic testing in cases of CMPD that transform to CML, not only to confirm the diagnosis but to plan treatment, as Philadelphia chromosome-positive and -negative cases differ in their management.
原发性血小板增多症(ET)是一种BCR-ABL基因融合阴性的慢性骨髓增殖性疾病(MPD),其他还包括真性红细胞增多症(PV)和骨髓纤维化。鲜有临床病例报道ET进展为伴有BCR-ABL基因表达的慢性粒细胞白血病(CML)。本报告描述了这样一例病例,并对其他报道的与BCR-ABL阴性慢性MPD同时发生的CML病例进行了综述。病例报告:一名49岁女性于2007年被诊断为ET。细胞遗传学检测JAK2或BCR-ABL1基因表达为阴性。八年后,即2015年1月,她出现极度疲劳、食欲不佳、体重无故减轻,白细胞(WBC)计数为每毫升24,700,血红蛋白为9.9 g/dl,血小板计数为每毫升557,000,血涂片中有原始细胞和嗜碱性粒细胞。荧光原位杂交(FISH)细胞遗传学分析证实存在9:22染色体易位(费城染色体),定量逆转录聚合酶链反应(qRT-PCR)检测到BCR-ABL基因表达,确诊为CML。2015年2月,一线治疗开始使用尼洛替尼,三个月后改为伊马替尼。在接下来的九个月里,qRT-PCR证实有深度分子缓解(MR5)的趋势。然而,她出现了早期骨髓纤维化,于是恢复了骨髓抑制治疗。结论:这个罕见病例凸显了在CMPD转化为CML的病例中进行细胞遗传学检测的重要性,这不仅有助于确诊,还能用于规划治疗,因为费城染色体阳性和阴性病例的治疗管理有所不同。