Extrauterine placental site trophoblastic tumour in association with a lithopedion

Abstract

A high-grade malignant placental site trophoblastic tumour with aggressive clinical course occurred at an extrauterine site. It complicated calcified abdominal pregnancy and resulted in repeated rectal perforation and peritonitis.

Extrauterine Lesions of Intermediate Trophoblast

“…Chronic salpingitis and endometriosis were noted in two of our patients. The previously reported case of a PSTT in the pouch of Douglas was associated with a lithopedion (11) and in another case there was a synchronous intrauterine pregnancy (9). The lesions had the typical microscopic appearance of previously described uterine PSTTs (14).…”

“…PSNs (4-8) and 3 PSTTs (9)(10)(11). Additionally, there are two reports of extrauterine ETTs occurring in the lung and colon (12,13) without adnexal involvement.…”

Developmental and Clinical Overview of Lithopaidion

“…In 2000, Passini et al [20] found fewer than 300 reported cases in 400 years of world medical literature; however, from 1968 to 1999 at least 47 new cases of lithopaidion were published. Moreover, it has been confirmed that in the last 10 years, as prenatal care and surveillance have increased, the presentation of lithopaidions is rare [28], even though at least 17 cases of lithopaidions were demonstrated in the last century [1,20,23,25,28,29,30,31,32,33,34,35,36,37,38,39,64]; only in this case, we have not considered the 4 cases of lithopaidions reported by Sunday-Adeoye et al [40] because it was not possible to determine the exact century of their occurrence. However, a complete analysis of the English literature has revealed more than 330 case of lithopaidion so far.…”

“…Lithopaidion is generally a single obstetric phenomenon; the literature includes 1 report of twins with lithopaidion [34] and a case of lithopaidion in twins [41], 1 case of lithopaidion with a coexistent live abdominal ectopic pregnancy [35], a case of lithopaidion formation after a cervical pregnancy [42], a case of lithopaidion evolving from hepatic pregnancy [43], 1 case with a coexistent placental site trophoblastic tumor that was discovered upon the removal of the lithopaidion (after a colostomy for rectal perforation was performed) [33], and 2 cases with coexistent cervical neoplasm [1,44] (table 3). …”

“…Overall, 21 articles were cited twice, 1 article was cited 3 times, and 86 papers were excluded because the abstract was not available (n = 40), the study was not published in English (n = 30) or the subject was not related to the topic of our review (n = 17). Thus, 52 references were selected [1,3,4,5,6,7,8,9,10,11,12,13,14,15,16,17,18,19,20,21,22,23,24,25,26,27,28,29,30,31,32,33,34,35,36,37,38,39,40,41,42,43,44,45,46,47,48,49,50,51,52,53]. The full paper was available for 30 articles, and 22 abstracts provided sufficient information.…”