Intest Res.  2014 Oct;12(4):328-332. 10.5217/ir.2014.12.4.328.

Adenocarcinoma Originating From a Completely Isolated Duplication Cyst of the Mesentery in an Adult

• Shin SY
• Cho MY
• Ryu H
• Kim JW
• Kim HS
• Kim JM
• Ko AN
• Yu TS
• Park HJ

• KMID: 2174411
• DOI: http://doi.org/10.5217/ir.2014.12.4.328

Abstract

Alimentary tract duplications are uncommon congenital abnormalities that usually have an anatomical connection with some part of the gastrointestinal tract and have a common blood supply with the adjacent segment of intestine. A completely isolated duplication cyst (CIDC) is a very rare type of gastrointestinal duplication that does not communicate with the normal bowel segment and possesses its own exclusive blood supply. Only 5 CIDC cases in adults have been reported in the English medical literature. Additionally, only 1 case of mucinous cystadenoma from an infected CIDC of the ileum has been reported. This report describes a 52-year-old male patient with a peritoneal CIDC, which upon curative excision was found to have given rise to an adenocarcinoma. The latter was lined internally with malignant glandular cells and contained a smooth muscular outer layer as determined by microscopic examination of the tissue. We believe that this is the first reported case of an adenocarcinoma originating from a CIDC in an adult.

Keyword

Adenocarcinoma; Completely isolated duplication cyst; Duplication cyst; Enteric duplications; Enterogenous cyst

MeSH Terms

Adenocarcinoma*
Adult*
Congenital Abnormalities
Cystadenoma, Mucinous
Gastrointestinal Tract
Humans
Ileum
Intestines
Male
Mesentery*
Middle Aged

Figure

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  Fig. 1 Abdominopelvic CT and EUS findings. (A) An abdominopelvic CT from an external facility showed an intraperitoneal mass near the stomach. (B) EUS revealed that the mass was in an extragastric area and had cystic features with an internal mixed echoic pattern. Furthermore, the cystic mass wall had an inner smooth hyperechoic mucosal layer (arrow a) and an outer hypoechoic muscular layer (arrow b).

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  Fig. 2 Gross finding of removed cyst (A), and feature of gross section (B). The entire cyst was excised without disturbing the normal bowel or mesenteric anatomy (A). In a gross section analysis, it was determined to be a 4×3×3-cm-sized unilocular cyst filled with dark brown necrotic material that appeared to comprise hemorrhagic contents. The cyst wall was evenly thin with a focal, ill-defined, yellowish-brown mural nodule (red arrow) (B).

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  Fig. 3 The cyst wall epithelial lining. The inner surface of the cyst was lined with neoplastic columnar epithelium that exhibited an increased nuclear/cytoplasmic ratio with a loss of polarity and nuclear hyperchromasia. Non-neoplastic epithelium was not observed in the cyst. The smooth muscle layers of the cyst resembled those of the intestinal wall (H&E, ×20) (A). The neoplastic glandular epithelium was observed to focally invade the muscle layer (red arrow) (B). There were multifocal cholesterol granulomas in the wall of the cyst, 1 of which matched the yellowish nodule observed during the gross examination (blue arrow; H&E, ×1.25). Immunohistochemical (IHC) staining. IHC staining of the neoplastic epithelial lining of the cyst was positive for cytokeratin 20 (C) and negative for cytokeratin 7 (D) (H&E, ×100).

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