Suzuki Keiichi, Abe Kodai, Ohbu Makoto
Department of Surgery, National Hospital Organization Tochigi Medical Center, Tochigi.
Department of Surgery, Keio University School of Medicine.
Surg Laparosc Endosc Percutan Tech. 2019 Aug;29(4):290-296. doi: 10.1097/SLE.0000000000000617.
A 59-year-old asymptomatic man underwent ultrasonography, which revealed gallstones and thickened gallbladder wall. Abdominal computed tomography (CT) showed a slightly swollen bilocular gallbladder and a soft tissue mass in the fundus site. Segmental adenomyomatosis (ADM) was suspected because numerous fundic cystic lesions were seen on magnetic resonance imaging. Endoscopic ultrasonography revealed numerous Rokitansky-Aschoff sinuses (RAS) and a papillary soft tissue shadow surrounded with irregular and remarkably thickened fundic gallbladder wall. Fluoro-2-deoxy-D-glucose-positron emission tomography/CT demonstrated slightly increased fluoro-2-deoxy-D-glucose uptake in the corresponding lesion. Surgery was performed under a diagnosis of gallbladder carcinoma (GBC) with concomitant ADM, and histopathology revealed a 30-mm papillotubular adenocarcinoma extending from the gallbladder body to fundus with invasion into the subserosa. Numerous RAS were present throughout the gallbladder showing various degrees of dysplasia. Ki67 and p53-labeling index (LI) was significantly higher in the dysplastic epithelium compared with normal fundic epithelium. p53-LI was also markedly increased (72.1%) in tissue in front of tumor invasion. Interestingly, these hyperproliferation indicators were extremely high (Ki67-LI: 28.8%; p53-LI: 91.9%) in RAS with low-grade dysplasia even in the gallbladder neck. Although, generally, tumors do not develop in the gallbladder neck with segmental ADM, our results suggest that a gallbladder with ADM has potential for carcinogenesis regardless of location, with segmental ADM. On the basis of histopathology, our patient was diagnosed with GBC arising from RAS with multicentric and multistep growth. A relationship between GBC and ADM, especially segmental ADM, has been suggested but remains controversial. Our experience is very suggestive of carcinogenesis developing from ADM.
一名59岁无症状男性接受了超声检查，结果显示有胆结石和胆囊壁增厚。腹部计算机断层扫描（CT）显示胆囊呈双房且略有肿大，底部有一软组织肿块。磁共振成像显示有许多底部囊性病变，怀疑为节段性腺肌增生症（ADM）。内镜超声检查发现大量罗-阿窦（RAS）以及一个乳头状软组织阴影，其周围是不规则且明显增厚的胆囊底部壁。氟代脱氧葡萄糖正电子发射断层扫描/CT显示相应病变处氟代脱氧葡萄糖摄取略有增加。在诊断为胆囊癌（GBC）合并ADM的情况下进行了手术，组织病理学显示一个30毫米的乳头管状腺癌，从胆囊体延伸至底部并侵犯至浆膜下层。整个胆囊都存在大量RAS，显示出不同程度的发育异常。与正常胆囊底部上皮相比，发育异常上皮中的Ki67和p53标记指数（LI）显著更高。在肿瘤侵犯前方的组织中，p53-LI也明显升高（72.1%）。有趣的是，即使在胆囊颈部，低度发育异常的RAS中这些增殖指标也极高（Ki67-LI：28.8%；p53-LI：91.9%）。虽然一般来说，节段性ADM的胆囊颈部不会发生肿瘤，但我们的结果表明，无论位置如何，合并节段性ADM的胆囊都有癌变的可能。根据组织病理学，我们的患者被诊断为起源于RAS的GBC，具有多中心和多步骤生长。GBC与ADM之间的关系，尤其是节段性ADM，已有研究提出，但仍存在争议。我们的经验强烈提示癌变是由ADM发展而来。
