Gastrointestinal stromal tumor in Carney's triad with laparoscopic total gastrectomy: a case report.

IF 0.7 Q4 SURGERY Surgical Case Reports Pub Date : 2024-10-23 DOI:10.1186/s40792-024-02041-2
Hajime Midoritani, Hironori Kawada, Kosuke Kaneda, Shuichiro Toda, Kento Awane, Keisuke Tanino, Kaichiro Harada, Keigo Tachibana, Masahiko Honjo, Koji Kitamura, Mami Yoshitomi, Yoshiharu Shirakata, Ryuta Nishitai
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Abstract

Introduction: Carney's triad is a rare syndrome characterized by the co-occurrence of gastric gastrointestinal stromal tumor (GIST), pulmonary chondroma, and extra-adrenal paraganglioma. We present a case of a young woman with GISTs associated with this triad.

Case presentation: A 28-year-old woman was identified with multiple gastric tumors and a right lung nodule during a routine health check-up. CT scans and upper gastrointestinal endoscopy revealed a 50 mm mass on the lesser curvature of the stomach, along with two additional gastric lesions and a 20 mm nodule in the right lung. The patient had a history of right middle lobectomy at the age of 19 for pulmonary chondroma. During surgery, enlarged lymph nodes were observed, indicating metastasis, which necessitated a total gastrectomy with radical (D2) lymph node dissection. Pathological examination confirmed seven GISTs, with immunohistochemical staining positive for KIT (+), DOG1 (+), and negative for SDHB (-). The postoperative course was uneventful, and the patient was discharged on the seventh postoperative day. Despite opting out of adjuvant imatinib therapy, she remains disease-free 2 years postoperatively.

Conclusions: This case underscores the necessity of total gastrectomy with lymph node dissection due to the high incidence of metastasis in GISTs associated with Carney's triad. Further research is required to determine the optimal extent of lymph node dissection in such cases.

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卡尼氏三联症胃肠道间质瘤腹腔镜全胃切除术:病例报告。
导言:卡尼三联征是一种罕见的综合征,其特点是同时存在胃肠道间质瘤(GIST)、肺软骨瘤和肾上腺外副神经节瘤。我们报告了一例年轻女性胃肠道间质瘤患者的病例:病例介绍:一名 28 岁的女性在例行体检中被查出患有多发性胃肿瘤和右肺结节。CT 扫描和上消化道内窥镜检查发现,胃小弯处有一个 50 毫米的肿块,另外还有两个胃部病变和右肺一个 20 毫米的结节。患者19岁时曾因肺软骨瘤接受过右中叶切除术。手术中观察到淋巴结肿大,表明已发生转移,因此必须进行全胃切除术,并进行根治性(D2)淋巴结清扫术。病理检查证实有7个GIST,免疫组化染色KIT(+)和DOG1(+)阳性,SDHB(-)阴性。术后过程顺利,患者于术后第七天出院。尽管她选择了伊马替尼辅助治疗,但术后2年仍未发病:本病例强调,由于与卡尼三联症相关的 GIST 转移发生率较高,有必要进行全胃切除术并进行淋巴结清扫。需要进一步研究确定此类病例淋巴结清扫的最佳范围。
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