ABSTRACT
Ovarian growing teratoma syndrome (GTS) is a rare disease characterized by growth of a benign tumor during or after chemotherapy, following the removal of germ cell gonadal cancers. Although benign, GTS tumors grow gradually and may compress surrounding organs. In addition, up to 3% of GTS cases can undergo malignant transformation. It is, therefore, needed to treat GTS. No standardized management protocol has been established to treat GTS; however, surgical resection is likely the only effective treatment because tumors in GTS are resistant to chemotherapy and radiation therapy. However, complete resection with conventional procedures is sometimes difficult when peritoneal metastasis is widespread. We report a rare case of ovarian GTS with widespread peritoneal metastases, which was totally resected by peritonectomy procedures. A 45-year-old Japanese woman was initially diagnosed with an immature teratoma grade 3, which was treated by hysterectomy and bilateral salpingo-oophorectomy. Adjuvant chemotherapy was performed after surgery with bleomycin, etoposide, cisplatin, and other chemotherapies. Due to recurrence of a chemoresistant tumor and normalization of tumor markers, GTS was suspected. She was referred to our institute, and complete cytoreductive surgery was performed using peritonectomy procedures, including parietal peritoneal resection, greater omentectomy, lesser omentectomy, rectosigmoid colectomy, diaphragm dissection, and cholecystectomy. A complete cytoreduction with no visible residual tumor tissue was achieved.
ABSTRACT
Cases of adenocarcinomas developed in Brunner gland hyperplasia (BGH) have been sporadically reported. Herein, we report the morphologic spectrum of hyperplastic changes culminating into dysplasia and carcinoma in 722 cases of BGH listed in our files. Fifteen of these cases showed dysplastic changes, with 8 graded as low-grade dysplasia, 5 as high-grade dysplasia, 11 as atypical hyperplasia, and 2 as invasive carcinoma, although each frequently coexisted in the same tumor. In two carcinomas, one had high-grade dysplasia in the mucosa, and another had only atypical hyperplasia. Interestingly, hyperplastic glands around dysplastic foci were associated with gastric foveolar metaplasia and papillary configuration in 13 cases, 11 of which showed a gradual increase in nuclear atypism in the transition from metaplastic to dysplastic glands. All of the metaplastic gastric glands showed diffuse and strong immunopositivity for gastric foveolar mucin (MUC5AC). Immunohistochemical profiles also supported the concept of a continuous spectrum in carcinogenesis from gastric foveolar hyperplasia through atypical hyperplasia or dysplasia and eventually to frank adenocarcinoma. The results of our study suggest, therefore, that dysplastic and/or carcinomatous change does occur in BGH, that they form the continuous morphologic spectrum, and that papillary foveolar metaplasia may be a precursor lesion in the process of carcinogenesis with a background of BGH.
