巨大十二指肠错构瘤致慢性贫血1例报告并文献复习
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西安交通大学附属咸阳市中心医院 肝胆外科,陕西 咸阳 712000

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吴东东,西安交通大学附属咸阳市中心医院主治医师,主要从事肝胆胰疾病方面的研究。

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Giant Brunner's gland hamartoma with associated chronic anemia: a case report and literature review
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Department of Hepatobiliary Surgery, Xianyang Central Hospital Affiliated to Xi'an Jiaotong University, Xianyang, Shaanxi 712000, China

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    摘要:

    背景与目的 十二指肠错构瘤(BGH)是十二指肠良性肿瘤中比较少见的类型,因其临床表现不典型、缺乏特异性,导致术前诊断困难,往往易被漏诊或误诊,主要依靠术后病理确诊。巨大BGH的病例临床更是罕见。本文报告1例巨大BGH致慢性贫血的诊治过程,并结合既往文献对本病特点进行复习,以期为临床工作提供经验借鉴。方法 回顾性分析西安交通大学附属咸阳市中心医院肝胆外科收治的1例BGH患者的临床资料,结合前期文献资料进行复习总结。结果 患者,女性,49岁;因腹痛伴恶心、呕吐就诊西安交通大学附属咸阳市中心医院肝胆外科。患者既往有慢性贫血病史,月经规律,偶有柏油样便。入院上腹部B超检查见胰头内侧十二指肠部包块;上腹部CT检查见十二指肠降部及水平部肠壁弥漫性增厚,结构不清;上腹部MRI+MRCP检查见十二指肠降部及水平部与邻近空肠分界不清,肠管明显扩张扭曲,肠壁分界不清,肠壁水肿明显,可见同心圆样改变。胃镜检查考虑十二指肠占位,并镜下活检提示符合胃黏膜异位。上消化道造影提示十二指肠降段与水平段移行区近圆形充盈缺损,考虑良性占位可能性大。最终考虑十二指肠占位性病变并引起梗阻,剖腹探查术后行胰十二指肠切除术,切除标本送病理学检查,最终诊断为BGH,免疫组化MUC5AC(+),术后恢复顺利出院。术后监测血常规,红细胞及血红蛋白逐渐恢复正常,随访12个月,患者一般状况良好,未诉不适,复查各项指标正常,继续随访。结论 巨大BGH致慢性贫血临床罕见,术前影像学检查往往很难提供有价值的信息,胃镜下活检有助于诊断,但阳性率低,最后确诊需依靠手术切除标本的组织病理学检查,病变较大时外科手术切除是治疗该病的有效手段。

    Abstract:

    Background and Aims Brunner's gland hamartoma (BGH) is a rare type of duodenal benign tumor. It is difficult to diagnose before operation and often easily to be missed or misdiagnosed because of atypical clinical manifestations and lack of specificity. The diagnosis mainly depends on postoperative pathology. The cases of giant BGH are rare. This paper reports the diagnosis and treatment of a case of giant BGH with associated chronic anemia, and reviews the characteristics of this disease in combination with the previous literature, so as to provide experience for clinical management of this condition.Methods The clinical data of a patient with BGH admitted to the Department of Hepatobiliary Surgery of Xianyang Central Hospital Affiliated to Xi'an Jiaotong University were analyzed retrospectively in combination with a review of the previous literature.Results The patient was a 49-year-old female, and was admitted to the Department of Hepatobiliary Surgery of Xianyang Central Hospital Affiliated to Xi'an Jiaotong University because of abdominal pain accompanied by nausea and vomiting. The patient had a history of chronic anemia, regular menstruation, and occasional black stool. On admission, the B-ultrasound examination of the upper abdomen showed the mass of the duodenum overlapped by the head of the pancreas; CT examination of the upper abdomen showed diffuse thickening of the intestinal wall in the descending and horizontal parts of the duodenum, with unclear structure; MRI+MRCP examination of the upper abdomen showed that the descending and horizontal parts of the duodenum and the adjacent jejunum were indistinct, the intestinal tube was obviously distended and twisted, the intestinal wall was indistinct, with obvious intestinal wall edema and presence of concentric circles change. Duodenal space occupying was considered in gastroscopy, and biopsy under endoscopy suggested that it was consistent with the ectopic gastric mucosa. The upper gastrointestinal angiography showed a nearly round filling defect at the junction of the descending and horizontal segments of the duodenum, considered to be a benign occupying lesion. Finally, the obstruction caused by the duodenal space-occupying lesion was considered, and pancreaticoduodenectomy was performed after exploratory laparotomy. The final diagnosis was BGH and immunohistochemical staining for MUC5AC positive after the resected specimen was sent to pathological examination. The patient recovered uneventfully after surgery and was discharged from the hospital. After the operation, the blood routine was monitored, and the red blood cells and hemoglobin gradually returned to normal. The patients were followed up for 12 months. The patients were generally in good condition without complaints of discomfort. All indexes were normal in the reexamination, and the follow-up was continued.Conclusion Giant BGH with associated chronic anemia is rare in clinical practice. Preoperative imaging examination is often difficult to provide valuable information. Biopsy under gastroscope is helpful for diagnosis, but the positive rate is low. The final diagnosis depends on histopathological examination of the surgical specimens. Surgical resection is an effective means to treat the disease when the lesion is large.

    图1 入院影像学资料 A:CT示十二指肠降部及水平部肠壁弥漫性增厚,结构不清;B:轴位MRI示十二指肠降部及水平部与邻近空场分界不清,肠管明显扩张扭曲,肠壁分界不清,肠壁水肿明显,可见同心圆样改变;C:冠状位MRI示低位胆道梗阻并胆囊积液,扩张胆总管下段受牵拉向左移位Fig.1 Imaging data on admission A: CT showing diffuse thickening of the intestinal wall in the descending and horizontal parts of the duodenum, with unclear structure; B: Axial MRI imaging showing that indistinct sight of the descending and horizontal parts of the duodenum and the adjacent jejunum, with obviously distended and twisted intestinal tube, indistinct intestinal wall, obvious intestinal wall edema and presence of concentric circles change; C: Coronal CT imaging showing lower biliary obstruction and gallbladder collections, with a left dislocation of the lower common bile duct due to traction
    图2 胃镜资料 A:十二指肠降部;B:十二指肠水平部;C:胃镜活检胃型黏膜(HE ×100)Fig.2 Gastroscopic data A: View of the descending part of the duodenum; B: View of the horizontal part of the duodenum; C: Biopsy showing gastric mucosa (HE ×100)
    图3 术后资料 A:大体标本;B:组织病理示增生的导管及腺体形成分叶状结构(HE×100);C:术后复查CTFig.3 Postoperative data A: Gross specimen; B: Histopathological examination showing the lobulated structures of the hyperplastic ducts and glands (HE×100); C: Postoperative CT review
    图1 入院影像学资料 A:CT示十二指肠降部及水平部肠壁弥漫性增厚,结构不清;B:轴位MRI示十二指肠降部及水平部与邻近空场分界不清,肠管明显扩张扭曲,肠壁分界不清,肠壁水肿明显,可见同心圆样改变;C:冠状位MRI示低位胆道梗阻并胆囊积液,扩张胆总管下段受牵拉向左移位Fig.1 Imaging data on admission A: CT showing diffuse thickening of the intestinal wall in the descending and horizontal parts of the duodenum, with unclear structure; B: Axial MRI imaging showing that indistinct sight of the descending and horizontal parts of the duodenum and the adjacent jejunum, with obviously distended and twisted intestinal tube, indistinct intestinal wall, obvious intestinal wall edema and presence of concentric circles change; C: Coronal CT imaging showing lower biliary obstruction and gallbladder collections, with a left dislocation of the lower common bile duct due to traction
    图2 胃镜资料 A:十二指肠降部;B:十二指肠水平部;C:胃镜活检胃型黏膜(HE ×100)Fig.2 Gastroscopic data A: View of the descending part of the duodenum; B: View of the horizontal part of the duodenum; C: Biopsy showing gastric mucosa (HE ×100)
    图3 术后资料 A:大体标本;B:组织病理示增生的导管及腺体形成分叶状结构(HE×100);C:术后复查CTFig.3 Postoperative data A: Gross specimen; B: Histopathological examination showing the lobulated structures of the hyperplastic ducts and glands (HE×100); C: Postoperative CT review
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吴东东,董浩,马富平.巨大十二指肠错构瘤致慢性贫血1例报告并文献复习[J].中国普通外科杂志,2022,31(9):1237-1242.
DOI:10.7659/j. issn.1005-6947.2022.09.013

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  • 收稿日期:2022-05-12
  • 最后修改日期:2022-08-24
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  • 在线发布日期: 2022-09-30