Abstract：
BACKGROUND: Dysplasia, carcinoma in situ, and other malignant transformation or premalignant/malignant histopathology (PMMH) seem uncommon in pediatric choledochal cyst (CC). A literature review and the authors\' experience are presented.
METHODS: All reports about PMMH in CC patients 15 years old or younger published in English and all cases of PMMH in specimens excised from CC patients 15 years old or younger by the authors were reviewed.
RESULTS: Of 20 published reports, PMMH was adenocarcinoma (n = 4), sarcoma (n = 4), and dysplasia (n = 12). Treatment for malignancies was primary pancreaticoduodenectomy (PD; n = 2) or cyst excision/hepaticojejunostomy (Ex/HJ; n = 6). Outcomes at the time of writing for malignancies: 2 deaths, 4 survivors after follow-up of 2 years, and 2 lost to follow-up. No dysplasia case has undergone malignant transformation. The authors have experienced 7 cases of PMMH; adenocarcinoma in situ (AIS; n = 1) and dysplasia (n = 6).
CONCLUSIONS: The present study identified the youngest cases of AIS and dysplasia from specimens excised when they were 3 years old and 4 months old, respectively. Both are published for the first time as evidence that PMMH can complicate CC in young patients. Long-term protocolized postoperative follow-up is mandatory when PMMH is diagnosed in pediatric CC.
METHODS: All reports about PMMH in CC patients 15 years old or younger published in English and all cases of PMMH in specimens excised from CC patients 15 years old or younger by the authors were reviewed.
RESULTS: Of 20 published reports, PMMH was adenocarcinoma (n = 4), sarcoma (n = 4), and dysplasia (n = 12). Treatment for malignancies was primary pancreaticoduodenectomy (PD; n = 2) or cyst excision/hepaticojejunostomy (Ex/HJ; n = 6). Outcomes at the time of writing for malignancies: 2 deaths, 4 survivors after follow-up of 2 years, and 2 lost to follow-up. No dysplasia case has undergone malignant transformation. The authors have experienced 7 cases of PMMH; adenocarcinoma in situ (AIS; n = 1) and dysplasia (n = 6).
CONCLUSIONS: The present study identified the youngest cases of AIS and dysplasia from specimens excised when they were 3 years old and 4 months old, respectively. Both are published for the first time as evidence that PMMH can complicate CC in young patients. Long-term protocolized postoperative follow-up is mandatory when PMMH is diagnosed in pediatric CC.
摘要：
背景：发育不良，原位癌，和其他恶性转化或癌前/恶性组织病理学（PMMH）在小儿胆总管囊肿（CC）中似乎并不常见。介绍了文献综述和作者的经验。
方法：回顾了作者发表的所有有关15岁或15岁以下CC患者的PMMH的英文报道以及从15岁或15岁以下CC患者切除的所有PMMH病例。
结果：在已发布的20份报告中，PMMH为腺癌（n=4），肉瘤（n=4），和发育不良（n=12）。恶性肿瘤的治疗方法是原发性胰十二指肠切除术（PD；n=2）或囊肿切除术/肝空肠吻合术（Ex/HJ；n=6）。在撰写恶性肿瘤时的结果：2例死亡，4名幸存者经过2年的随访，和2失去了后续。无异型增生病例发生恶变。作者经历了7例PMMH；原位腺癌（AIS；n=1）和异型增生（n=6）。
结论：本研究从3岁和4个月大切除的标本中确定了最年轻的AIS和发育不良病例，分别。两者均首次发表，作为PMMH可以使年轻患者的CC复杂化的证据。当在儿科CC中诊断为PMMH时，必须进行长期的protocoliled术后随访。
方法：回顾了作者发表的所有有关15岁或15岁以下CC患者的PMMH的英文报道以及从15岁或15岁以下CC患者切除的所有PMMH病例。
结果：在已发布的20份报告中，PMMH为腺癌（n=4），肉瘤（n=4），和发育不良（n=12）。恶性肿瘤的治疗方法是原发性胰十二指肠切除术（PD；n=2）或囊肿切除术/肝空肠吻合术（Ex/HJ；n=6）。在撰写恶性肿瘤时的结果：2例死亡，4名幸存者经过2年的随访，和2失去了后续。无异型增生病例发生恶变。作者经历了7例PMMH；原位腺癌（AIS；n=1）和异型增生（n=6）。
结论：本研究从3岁和4个月大切除的标本中确定了最年轻的AIS和发育不良病例，分别。两者均首次发表，作为PMMH可以使年轻患者的CC复杂化的证据。当在儿科CC中诊断为PMMH时，必须进行长期的protocoliled术后随访。
