Pandey Deepali, Shepro David Scott
Internal Medicine, Saint Vincent Hospital, Worcester, MA, USA
Hematology/Oncology, Saint Vincent Hospital, Worcester, MA, USA.
BMJ Case Rep. 2020 Apr 8;13(4):e230241. doi: 10.1136/bcr-2019-230241.
Lynch syndrome has been associated with predominantly colorectal, endometrial, and ovarian cancer. We report hereby an unusual case of thymic carcinoma in a patient with Lynch syndrome. A 45-year-old Caucasian woman with a personal history of Lynch syndrome (MLH1 heterozygous mutation) presented with dyspnea, chest pain, and dysphagia. CT chest showed a bulky anterior mediastinal mass, pulmonary nodules, and pericardial effusion. Lung biopsy demonstrated a poorly differentiated carcinoma with squamous features with extensive necrosis, favouring thymic origin. Genomic studies on the tumour revealed deficient mismatch repair status with a two-copy deletion of MLH1 at 3p22.2 and c-Kit mutation. She received carboplatin and paclitaxel, with initial clinical improvement, but then died within 3 months after diagnosis. This case highlights that thymic cancer may be one of the malignancies associated with Lynch syndrome, and MLH1 gene mutation may have a role in the pathogenesis of thymic cancer.
林奇综合征主要与结直肠癌、子宫内膜癌和卵巢癌相关。我们在此报告一例林奇综合征患者发生胸腺癌的罕见病例。一名45岁有林奇综合征个人史(MLH1杂合突变)的白种女性,出现呼吸困难、胸痛和吞咽困难。胸部CT显示前纵隔有一个巨大肿块、肺结节和心包积液。肺活检显示为具有鳞状特征的低分化癌,伴有广泛坏死,倾向于胸腺来源。对肿瘤的基因组研究显示错配修复缺陷状态,在3p22.2处有MLH1的双拷贝缺失以及c-Kit突变。她接受了卡铂和紫杉醇治疗,最初临床症状有所改善,但在诊断后3个月内死亡。该病例突出表明胸腺癌可能是与林奇综合征相关的恶性肿瘤之一,且MLH1基因突变可能在胸腺癌的发病机制中起作用。