Solitary Peutz-Jeghers-type appendiceal hamartomatous polyp growing into the terminal ileum

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.