An Interesting Cause of Ileal Intussusception.

A 75-year-old man with a history of deep vein thrombosis, pulmonary embolism, and nonmelanoma skin cancer was evaluated for epigastric abdominal pain and weight loss. His abdominal computed tomography scan incidentally revealed intussusception of the ileum with a 1.4-cm low-density lead point. Consequently, the patient was referred to us for endoscopic evaluation. We performed retrograde double-balloon enteroscopy. In the proximal ileum, we found 1 atypical-appearing, semi-pedunculated, submucosal polypoid lesion consistent with what was seen on imaging (Figure 1). Biopsies were obtained with cold forceps. After a single bite disrupted the surface of the lesion, a large amount of chylous discharge was noted resulting in a significant decrease in the size of the lesion, suggesting that it was an unusual chylous cyst (Figure 2). A second tunneled biopsy was obtained, which completely unroofed the surface of the lesion allowing visualization of what appeared to be a cystic cavity (Figure 2). Histopathology was consistent with leiomyoma of the muscularis mucosae (Figure 3). Given the incidental and benign nature of the lesion and the fact that it diminished significantly in size after being unroofed, no further interventions were undertaken. On follow-up, the patient's abdominal pain had improved somewhat and repeat imaging revealed median arcuate ligament syndrome—the likely etiology of his chronic symptoms—which was treated with celiac ganglion block.

Figure 1.

Atypical-appearing submucosal polyp seen endoscopically in the proximal ileum, correlating with imaging findings.

Figure 2.

(A) Chylous discharge (black arrow) from the polypoid submucosal lesion after biopsy with cold forceps, which (B) disrupted and unroofed (red arrow) the surface of the polyp revealing an underlying (C) cystic cavity (red asterisk).

Figure 3.

Representative histology slide showing leiomyoma of the muscularis mucosae.

Leiomyomas constitute approximately 20% of all benign gastrointestinal tumors, have a slight male predominance, and have a peak incidence in the sixth decade of life.[1,2] Arising from the spindle cells of the muscularis mucosae, these tumors most often present with gastrointestinal bleeding due to ulceration.[1-3] In rarer cases, they can present with obstruction due to intussusception.[2,3] Typically, these appear as solid, polypoid, intraluminal lesions that maintain their form when biopsied. Interestingly, the histopathology in this case was not different from what is typically seen on pathology for leiomyomas. To the best of our knowledge, this is the first described cystic, chylous leiomyoma in the small bowel.

DISCLOSURES

Author contributions: MG Noujaim wrote the article and subsequent revisions and prepared the final images. D. Wild performed the endoscopic procedure and diagnosed the lesion. D. Wild reviewed and proofread all versions of the article and is the article guarantor. M. Arbogast provided the pathology slides and commentary on the histology of the lesion.

Financial disclosure: None to report.

Informed consent was obtained for this case report.