Leng Qingqing, Lv Wanrui, Yang Heqi, Li Xiaofen, Wang Weiya, Cheng Ke, Chang Chen, Cao Dan
Division of Abdominal Tumor Multimodality Treatment, Cancer Center, West China Hospital, Sichuan University, Chengdu, Sichuan, China.
Department of Oncology, Meishan Municipal People's Hospital, Meishan, Sichuan, China.
Front Pharmacol. 2024 Jun 14;15:1361628. doi: 10.3389/fphar.2024.1361628. eCollection 2024.
Pancreatoblastoma (PB), a neoplasm derived from pancreatic follicular cells, primarily affects the pediatric population. Although infrequent in adults, it is associated with a considerably worse prognosis. Approximately one-third of patients are diagnosed with metastatic disease, with liver metastases being the most prevalent. Diagnosis relies on histopathological alterations including squamous vesicles, positive staining for CK8/CK18/CK19, and nuclear displacement of β-catenin. Additionally, liver metastases demonstrate substantial enhancement during the arterial phase of a contrast-enhanced computed tomography (CT) scan. Surgical resection serves as the principal therapeutic approach for addressing primary lesions and liver metastatic PB. In instances where surgical intervention is not viable, patients may derive benefits from systemic therapy and radiotherapy. This particular case report presents the clinical details of a 27-year-old female patient diagnosed with PB, who subsequently developed multiple liver metastases following a pancreaticoduodenectomy. Genomic examinations revealed the presence of ERBB2 amplification, RAD54L deletion, low TMB-L, and MSS in the patient. Despite the patient undergoing chemotherapy and Her-2 targeted therapy in conjunction with immunotherapy, no reduction in lesion size was observed until the administration of surufatinib. Subsequently, a notable outcome ensued, where the metastatic lesions were effectively excised via surgical intervention. Surufatinib has demonstrated a progression-free survival (PFS) of no less than 14 months, and the patient's survival has endured for a duration of 33 months. This indicates the potential efficacy of surufatinib as a viable therapeutic alternative for adult patients afflicted with PB.
胰腺母细胞瘤(PB)是一种起源于胰腺滤泡细胞的肿瘤,主要影响儿童群体。虽然在成人中不常见,但它与预后相当差有关。约三分之一的患者被诊断为转移性疾病,其中肝转移最为常见。诊断依赖于组织病理学改变,包括鳞状小泡、CK8/CK18/CK19阳性染色以及β-连环蛋白的核移位。此外,在增强计算机断层扫描(CT)扫描的动脉期,肝转移灶显示出明显强化。手术切除是治疗原发性病变和肝转移性PB的主要治疗方法。在无法进行手术干预的情况下,患者可能从全身治疗和放疗中获益。本病例报告介绍了一名27岁女性患者诊断为PB的临床细节,该患者在胰十二指肠切除术后出现了多处肝转移。基因组检查显示患者存在ERBB2扩增、RAD54L缺失、低肿瘤突变负荷-低(TMB-L)和微卫星稳定(MSS)。尽管患者接受了化疗、Her-2靶向治疗以及免疫治疗,但在使用索凡替尼之前未观察到病灶缩小。随后,出现了显著的结果,通过手术干预有效地切除了转移病灶。索凡替尼已显示出无进展生存期(PFS)不少于14个月,患者生存期长达33个月。这表明索凡替尼作为成年PB患者一种可行的治疗选择具有潜在疗效。