Solitary hamartomatous duodenal polyp; a different entity: Report of a case and review of the literature

Abstract

Solitary hamartomatous duodenal polyps are a clinical entity considered until now to express an incomplete or initial form of Peutz-Jeghers syndrome (PJ syndrome). Following our experience of 1 case, we analyzed the clinical characteristics of the 12 previously reported cases of solitary hamartomatous duodenal polyps. In none of these cases did we find any documentation of mucocutaneous pigmentation, a family or personal history of intestinal polyps, or local neoplastic degeneration of the disease, and all cases presented during the fifth or sixth decade of life. Thus, we conclude that solitary hamartomatous duodenal polyps constitute a different entity to PJ syndrome, the most appropriate elective treatment for which is endoscopic electrosurgical polypectomy.

Endoscopic snare papillectomy for a solitary Peutz-Jeghers-type polyp in the duodenum with ingrowth into the common bile duct: Case report

“…Solitary PJ-type polyps, defined as hamartomatous polyps without any of the clinical symptoms of PJS, are characterized histologically by tree-like branching of smooth muscle fibers, with a core of smooth muscle, covered by mucosal tissue of nearnormal appearance [4] . It is truly difficult to determine whether these solitary duodenal hamartomatous polyps are an incomplete type or initial clinical manifestation of PJS [2] . This entity may be differentiated from PJS based on information from previous studies, including: the lack of perioral or perianal mucocutaneous pigmentation and lack of family history; symptom onset usually occurring past the sixth and seventh decades of life; and lower risk of local cancer development than hamartomatous polyps in patients with PJS.…”

“…A search of case reports on the MEDLINE database up to 2014 using the terms "solitary", "duodenum", and "hamartomatous polyp", but without PJS, identified 19 patients in 13 studies published in English with solitary-PJ type polyps in the duodenum, including the patient described here [2,[5][6][7][8][9][10][11][12][13][14][15] (Table 1). Age at onset ranged from 23-89 years (mean ± SD: 60.3 ± 20.1 years).…”

“…Solitary hamartomatous polyps have been considered a variant or a separate disease entity without the features of PJS [2] . Solitary duodenal hamartomas are even less common.…”

Three cases of Solitary Peutz-Jeghers-type hamartomatous polyp in the duodenum

“…Recently, a patient with a Peutz-Jeghers polyp without either mucocutaneous pigmentation or a positive family history was described as a case of solitary Peutz-Jegherstype hamartomatous polyp. It has been suggested that this condition should be considered as a clinical entity different from Peutz-Jeghers syndrome [2] . A solitary Peutz-Jegherstype hamartomatous polyp is histologically characterized by tree-like branching of smooth muscle fibers, with a core of smooth muscle, covered by mucosal tissue of near-normal appearance [3] .…”

“…A solitary Peutz-Jegherstype hamartomatous polyp is histologically characterized by tree-like branching of smooth muscle fibers, with a core of smooth muscle, covered by mucosal tissue of near-normal appearance [3] . A solitary Peutz-Jeghers-type hamartomatous polyp in the duodenum is rare, and few reports have described its characteristic endoscopic features [2,[4][5][6][7][8][9][10] . We describe three cases of solitary Peutz-Jeghers-type hamartomatous polyp and their endoscopic findings in detail.…”