Adenocarcinoma arising in a tailgut cyst with prominent meningothelial proliferation and thyroid tissue: case report and review of the literature

Abstract

Tailgut cysts (retrorectal cystic hamartomas) are rare lesions occurring in the retrorectal/presacral space of young women, considered to be of developmental origin. In this study, we report the clinicopathological characteristics of an adenocarcinoma developing in an unusual tailgut cyst. A 47-year-old female presented with a 3-month history of an enlarging presacral mass, extending into the gluteal area. The patient underwent surgical resection. Microscopic examination revealed a mucinous adenocarcinoma with intestinal features arising in a tailgut cyst. In addition to the usual components previously described in this lesion (cuboidal, columnar, ciliated or squamous epithelial lining with scattered smooth muscle fibers in the wall), our case was unique in that it was associated with a meningothelial proliferation and benign thyroid tissue with oncocytic changes. These additional features bring into question the proposed hindgut origin of tailgut cysts.

A Tailgut Cyst—Cystic Mass Diagnosed by Prenatal Ultrasonography

“…Tailgut cyst as well as teratoma can rarely become malignant transformations such as adenocarcinoma and carcinoid. 18 For this reason, this case highlights the need to consider tailgut cyst in the differential diagnosis when a presacral cyst is encountered. Fig.…”

A large cystic gastrointestinal stromal tumor of the rectum in the retrorectal space

“…It is likely that gastric or enteric GISTs may easily grow in a pedunculated, exophytic pattern without any spatial barriers. Central necrosis can then be caused by recurrent congestion, hemorrhage, and/or edema when the tumors are twisted or grow faster than the capacity of blood supply or vein drainage [6]. In contrast, according to the report by Miettinen et al [14] describing the clinicopathological features of 133 anorectal GISTs, macroscopic cystic spaces were found in only a few larger tumors, although the extent and incidence of these features was not mentioned.…”

“…Although TGC is a benign developmental cyst, complete surgical resection of the lesion is recommended to prevent the rare complication of malignant transformation, including adenocarcinoma and carcinoid [6][7][8][9][10]. Therefore, we proceeded with surgical extirpation by partial Fig.…”

Extramammary Paget Disease in a Retrorectal Dermoid Cyst

“…Carcinoid tumor, adenocarcinoma, and adenosquamous carcinoma have been reported to arise within these. 1,15,16,20 Rectal enteric duplication cysts are another potential mimic. Duplication cysts may occur at any point along the gastrointestinal tract and are congenital malformations consisting of hollow, epithelium-lined, cystic or tubular structures with a muscular wall, intimately attached to the portion of the tract from which they are derived and sharing a common blood supply.…”