十二指肠畸胎瘤1例报道并文献复习

Duodenal teratoma:a case report and literature review

目的探讨十二指肠畸胎瘤的临床、影像及病理学特征,提高临床医生对该肿瘤罕见发病部位的认识。方法回顾性分析2021年3月山东省烟台市烟台山医院收治的1例十二指肠实性成熟性畸胎瘤患者的临床、影像及病理资料。在中国知网、万方数据库、维普数据库及PubMed中以"十二指肠/duodenal"和"畸胎瘤/teratoma"为关键词,检索2020年12月之前有关十二指肠畸胎瘤的文献,共检出英文文献报道3篇3例和中文文献报道2篇2例;结合本文报道的1例,总结其临床影像及病理学特征。结果本例为40岁男性,临床表现为上腹部胀痛不适半年,影像学表现为十二指肠降段不规则混杂密度影,内见不规则钙化样高密度影及脂肪密度影;临床诊断十二指肠降部占位,行十二指肠肿物切除术,术后病理诊断为十二指肠实性成熟性畸胎瘤,恢复良好。结合文献报道的5例,共计6例十二指肠畸胎瘤,其中男4例、女2例,年龄7 d~40岁。临床均表现为肠梗阻或肠梗阻症状,其中2例伴有贫血、2例有腹部包块。发病部位均位于十二指肠降部及水平部。影像学检查:5例行CT和/或B超检查,均提示肿瘤或占位,显示囊性肿瘤1例、囊实性肿块1例、实性肿瘤3例,其中2例伴有特征性脂肪组织及钙化;另1例行X线检查,显示"腹部空气液位"特征性X线表现。6例均行手术治疗,切除肿瘤最大径为3~12 cm;术中4例发现肿瘤与十二指肠肠壁间有管道通连。术后病理诊断十二指肠成熟性畸胎瘤5例、恶性畸胎瘤1例,均为原发。5例成熟性畸胎瘤中的4例预后良好、随访无复发,另1例未报告;1例恶性畸胎术后半年、化疗后3个月复发转移。结论十二指肠畸胎瘤罕见,多见于十二指肠降部及水平部。多数为成熟性畸胎瘤,外科手术完整切除预后良好;少数为恶性畸胎瘤,术后易复发、转移。临床多表现为肠梗阻,病灶内出现脂肪和钙化征象是其最具诊断价值的影像学特征,病理组织学改变同其他部位畸胎瘤一致。

Objective This study aimed to investigate the clinical,imaging,and pathological features of duodenal teratoma,as well as to improve clinicians'understanding of the special site of the tumor.Methods The clinical and pathological data of a patient with duodenal solid mature teratoma treated in Yantai Mountain Hospital of Yantai City,from March 2021 were analyzed retrospectively.Using"十二指肠/duodenal"and"畸胎瘤/teratoma"as keywords,the literature on duodenal teratoma before December 2020 was searched in CNKI,Wanfang database,Weipu database,and PubMed.Three English literature reports and two Chinese literature reports were found.Combined with a case reported in this paper,the clinical,imaging,and pathological features were summarized.Results This patient was a 40-year-old male.The clinical manifestation was upper abdominal pain and discomfort for half a year.The imaging manifestations were irregular mixed density shadow in the descending segment of the duodenum and irregular calcification like high density shadow and fat density shadow.The clinical diagnosis was descending duodenal space-occupying lesion,and duodenal mass resection was performed.The postoperative pathological diagnosis was solid mature teratoma of the duodenum,which recovered well.Combined with the five patients reported in the literature,there were 6 patients of duodenal teratoma(including 4 males and 2 females)aged from 7 days to 40 years.The clinical manifestations were intestinal obstruction or symptoms of intestinal obstruction,including 2 patients with anemia and 2 patients with abdominal mass.The diseased sites were located in the descending and horizontal part of the duodenum.Imaging examination:Five patients underwent CT and/or B-ultrasound examination,and all showed tumor or space-occupying lesions,including 1 patient of cystic tumor,1 patient of cystic solid mass,and 3 patients of solid tumor;2 patients were accompanied with characteristic adipose tissue and calcification.Only X-ray examination was performed in 1 patient without CT and B-ultrasound;the characteristic X-ray manifestation of"abdominal air level"was displayed.All 6 patients underwent surgery,and the maximum meridian of the tumor was 3–12 cm.During the operation,4 patients presented a pipeline connection between the tumor and the duodenal intestinal wall.Five patients of duodenal mature teratoma and one case of malignant teratoma were diagnosed by postoperative pathology.Among the 5 patients of mature teratoma,1 patient without report,the rest had a good prognosis and no recurrence during follow-up.One patient of malignant teratoma recurred and metastasized half a year after the operation and 3 months after chemotherapy.Conclusion Duodenal teratomas are rare,mostly in the descending and horizontal parts of the duodenum.The most of duodenal teratomas are mature teratomas,and complete surgical resection has a good prognosis.A few are malignant teratomas,which are easy to relapse and metastasize after operation.The most of the clinical manifestations are intestinal obstruction.The imaging features of fat and calcification in the lesion are the most valuable diagnostic features.Pathological diagnosis is the gold standard,and the histopathological changes are consistent with teratoma in other sites.