Published online Apr 28, 2014. doi: 10.3748/wjg.v20.i16.4822
Revised: December 31, 2013
Accepted: January 20, 2014
Published online: April 28, 2014
Solitary Peutz-Jeghers type hamartomatous polyp is rare. It is considered to be related to a variant Peutz-Jeghers syndrome (PJS) and may be a separate disease entity. A 50-year-old man was referred to our hospital with a diagnosis of intussusception in the terminal ileum and underwent segmental ileal resection with appendectomy. We identified a 3.5-cm diameter polyp arising from the appendix with ingrowth into the terminal ileum. The polyp was confirmed to be a hamartomatous polyp of Peutz-Jeghers-type, histologically. However, the patient had no characteristic manifestations of PJS such as mucocutaneous pigmentation and family history. There are few reports of appendiceal hamartomatous polyp in PJS patients and solitary appendiceal hamartomatous polyp is even rarer. Also, rather than telescoping, ours is the first reported intussuscepted lesion, to the best of our knowledge.
Core tip: Intussusception arising from various leading points can cause intestinal obstruction. We experienced a case of solitary Peutz-Jeghers-type hamartomatous polyp in the appendix. This is an extremely rare condition with an unusual ingrowth characteristic. To the best of our knowledge, ours is the first case of a solitary appendiceal hamartomatous polyp with an ingrowing pattern. Although there are limitations, this case could serve as a reference for diagnostic and management decisions in future similar cases.