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一例伴有种系MSH6突变的孤立性儿童髓系肉瘤——病例报告

An isolated childhood myeloid sarcoma with germline MSH6 mutation-a case report.

作者信息

Liu Yu, GuLiBaHa MaiMaiTi, Yue Ying-Bin, Li Ming-Wei, Cao Shan-Bo, Yan Mei

机构信息

Pediatric Department, The First Affiliated Hospital of Xinjiang Medical University, Urumqi, China.

Lab of Gene, Acornmed Biotechnology Co., Ltd. Beijing Economic and Technological Development Zone, Beijing, China.

出版信息

Transl Pediatr. 2021 Aug;10(8):2136-2143. doi: 10.21037/tp-21-326.

DOI:10.21037/tp-21-326
PMID:34584885
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8429872/
Abstract

Myeloid sarcoma (MS) is a type of malignant tumor that originates in the bone marrow. This study reports on the treatment of an 11-year-old Uygur girl with a 15-day history of fever and paroxysmal cough, accompanied by right hip pain. During treatment, fatigue and anemia developed, physical strength decreased, and a few petechiae were seen in the lower extremities. Multiple enlarged lymph nodes were palpable in the neck, with slight congestion in the pharynx. Routine blood screening showed three major myeloid lineage abnormalities. Pathological examination revealed the presence of CD10 (-), CD99 (+), CD20 (+), CD3 (-), CD117 (weak+), CD34 (unclear location), TdT (-), Pax5 (-), Ki-67 (50%+), MPO (-), and CD43 (+). The patient was eventually diagnosed with isolated MS. After chemotherapy, no small particles were observed in bone marrow morphology. Complete remission was confirmed by flow cytometric detection of minimal residual disease. Genomic DNA was subjected to targeted sequencing of 236 gene panels to detect somatic mutations and the MSH6 c.3953_3954insAA p.R1318fs germline mutation. Unfortunately, the patient was subsequently lost to follow-up. To our knowledge, an MSH6 germline mutation had not previously been reported in children with MS, and we speculated that an MSH6 germline mutation led to genomic instability, triggering a somatic mutation in multiple genes and ultimately led to the development of MS in this patient. It is suggested that rare base abnormalities may be involved in the development of isolated myeloid sarcomas (IMS).

摘要

髓系肉瘤(MS)是一种起源于骨髓的恶性肿瘤。本研究报告了一名11岁维吾尔族女孩的治疗情况,该女孩有15天的发热和阵发性咳嗽病史,伴有右髋部疼痛。治疗期间,出现疲劳和贫血,体力下降,下肢可见少量瘀点。颈部可触及多个肿大淋巴结,咽部轻度充血。血常规筛查显示三大髓系谱系异常。病理检查显示CD10(-)、CD99(+)、CD20(+)、CD3(-)、CD117(弱阳性+)、CD34(定位不清)、TdT(-)、Pax5(-)、Ki-67(50%+)、MPO(-)和CD43(+)。该患者最终被诊断为孤立性MS。化疗后,骨髓形态未见小颗粒。通过流式细胞术检测微小残留病证实完全缓解。对基因组DNA进行236个基因panel的靶向测序,以检测体细胞突变和MSH6基因c.3953_3954insAA p.R1318fs胚系突变。不幸的是,该患者随后失访。据我们所知,此前尚未在MS患儿中报道过MSH6胚系突变,我们推测MSH6胚系突变导致基因组不稳定,引发多个基因的体细胞突变,最终导致该患者发生MS。提示罕见碱基异常可能参与孤立性髓系肉瘤(IMS)的发生发展。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3f13/8429872/57d18f4c954f/tp-10-08-2136-f3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3f13/8429872/6dba98fab7d4/tp-10-08-2136-f1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3f13/8429872/2bf766b6b4b2/tp-10-08-2136-f2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3f13/8429872/57d18f4c954f/tp-10-08-2136-f3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3f13/8429872/6dba98fab7d4/tp-10-08-2136-f1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3f13/8429872/2bf766b6b4b2/tp-10-08-2136-f2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3f13/8429872/57d18f4c954f/tp-10-08-2136-f3.jpg

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