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Benign struma ovarii (SO) has a probability of metastasis named “peritoneal strumosis”, which is extremely rare, such that the specific clinical characteristics, treatment options, and survival outcomes remain unclear. We screened three cases of peritoneal strumosis among 229 cases of SO treated in our hospital. Case 1 was a 36-year-old woman with extensive peritoneal seedings at initial presentation. The second one was a 49-year-old with trocar site implant 11 years after laparoscopic adnexectomy. Case 3 was a 45-year-old woman who had an isolated lesion at the anterior surface of the rectum after laparoscopic ovarian cystectomy for SO 14 years ago. These three patients underwent surgery without any adjuvant treatment and remained disease-free after 30 to 68 months. A systematic review was then conducted and another 16 cases were identified. More than half (10/19, 52.6%) of the patients had previous SO-related ovarian surgery. The median interval between prior SO-related surgery and the initial presentation of peritoneal strumosis was 10.0 years; both regional and distant metastasis, even in the liver, lung, and heart, could also be affected. Surgery was the mainstay therapy (18/19, 94.7%), in which six patients (6/19, 31.7%) were treated with total thyroidectomy (TT) followed by radioiodine (RAI) therapy. Postoperative chemotherapy was only applied in one patient, and the last one only received a diagnostic biopsy without further treatment. Recurrence was noted in two patients with a median recurrence-free survival of 12 years, where surgical excision and RAI were then performed. No death occurred after a mean follow-up of 53 months, where 12 patients achieved no evidence of disease and five were alive with the disease. Peritoneal strumosis has unpredictable biological behaviors and the crude incidence is approximately 1.3% in SO. Patients with peritoneal strumosis have excellent survival outcomes, irrespective of different treatment strategies employed. Surgery with personalized RAI should be preferred and long-term close monitoring is recommended. Full article

Struma ovarii (SO) is a monodermal teratoma predominantly composed of thyroid tissue (TT) showing benign, “proliferative”, or malignant histology. By imaging, a 38-year-old patient with lower backache revealed a 6.2-cm vertebral lesion (L5). Core biopsy showed well-differentiated TT without features of papillary carcinoma. A 3.5-cm left ovarian mature teratoma (lacking TT) and peritoneal nodules (showing well-differentiated TT) were also identified and surgically removed. Thyroid ultrasound and cytological examination resulted negative. Four years before, left ovarian cystectomy was performed for a histologically “proliferative” SO. According to the malignant clinical course and WHO classification, this case was overall reassessed as a recurring well-differentiated follicular carcinoma arising in SO (WD-FC-SO), despite lacking malignant histological features in any specimens. Immunophenotype: TTF-1+/PAX-8+/thyroglobulin+/CK7+/chromogranin-/synaptophysin-/inhibin-/calretinin-/HNF1B-; Ki-67 index < 5%. Polymerase chain reaction analysis resulted negative for BRAF^V600E mutation. The patient refused further treatments, without recurrence after 17 months. The clinical behavior of SO may be unpredictable. Histologically benign or proliferative strumas extraordinarily metastasize, while SO with malignant features may not recur. The exceptional evidence of peritoneal implants of well-differentiated TT (peritoneal strumosis) in patients with histologically benign SO represents a metastasis of WD-FC-SO (like in our case). A multidisciplinary approach including clinical, laboratory, radiologic, and histopathological data is required. Full article