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骶尾部畸胎瘤的长期功能结局——对探索“真实世界”结局的已发表研究的系统评价

Long-term functional outcomes of sacrococcygeal teratoma - A systematic review of published studies exploring 'real world' outcomes.

作者信息

Salim Adeline, Raitio Arimatias, Losty Paul D

机构信息

Department of Paediatric Surgery, Alder Hey Children's NHS Foundation Trust, Liverpool, United Kingdom.

Department of Paediatric Surgery, University of Turku and Turku University Hospital, Turku, Finland.

出版信息

Eur J Surg Oncol. 2023 Jan;49(1):16-20. doi: 10.1016/j.ejso.2022.09.002. Epub 2022 Sep 7.

DOI:10.1016/j.ejso.2022.09.002
PMID:36127201
Abstract

Sacrococcygeal teratoma (SCT) is a rare neoplasm affecting 1:35,000 newborns. Long-term follow-up from small observational studies report impaired bladder/bowel function. This current study comprehensively analyses all published studies to better define true long-term functional sequelae. Medline/Embase databases were searched with PRISMA guidelines. Final analysis yielded 37 studies involving 1116 patients (854 female; 77%). Individual datasets were available in 14 studies (222 patients). According to Altman classification - 298/845 (35%) were Type I, 252/845 (30%) Type II, 133/845 (16%) Type III, and 128/845 (15%) Type IV tumours. Most neoplasms were benign (640/858; 75%), 77/858 (9%) immature and 141/858 (16%) malignant. Abnormal bladder function was reported in 7/39 (18%) Altman Type I, 23/61 (37.7%) Type II, 11/34 (32.4%) Type III, and 15/25 (60%) Type IV cases (p = 0.007). Adverse urological outcomes were notably common in immature/malignant neoplasms vs benign tumours and in patients requiring reoperation(s); p = 0.002 and p = 0.01. Bowel dysfunction was evident in 19% index cases and constipation in 26% with no significant association(s) with tumour characteristics. Higher Altman stage, unfavourable tumour biology and reoperation are associated with poor functional outcome(s). Multidisciplinary management from primary diagnosis of SCT is crucially important for all patients to best optimise functional outcomes across surgical specialities.

摘要

骶尾部畸胎瘤(SCT)是一种罕见的肿瘤,发病率为1:35000新生儿。小型观察性研究的长期随访报告了膀胱/肠道功能受损。本研究全面分析了所有已发表的研究,以更好地确定真正的长期功能后遗症。按照PRISMA指南检索了Medline/Embase数据库。最终分析纳入37项研究,涉及1116例患者(854例女性;77%)。14项研究(222例患者)有个体数据集。根据奥特曼分类法,845例中有298例(35%)为I型,252例(30%)为II型,133例(16%)为III型,128例(15%)为IV型肿瘤。大多数肿瘤为良性(640/858;75%),77例(9%)为未成熟型,141例(16%)为恶性。奥特曼I型39例中有7例(18%)报告膀胱功能异常,II型61例中有23例(37.7%),III型34例中有11例(32.4%),IV型25例中有15例(60%)(p = 0.007)。与良性肿瘤相比,未成熟/恶性肿瘤以及需要再次手术的患者不良泌尿系统结局尤为常见;p = 0.002和p = 0.01。19%的索引病例存在肠道功能障碍,26%的患者存在便秘,与肿瘤特征无显著关联。较高的奥特曼分期、不良的肿瘤生物学行为和再次手术与功能结局不良相关。从骶尾部畸胎瘤的初步诊断开始进行多学科管理对于所有患者至关重要,以便在各个外科专业中最佳地优化功能结局。

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