A Solitary Peutz-Jeghers-Type Hamartomatous Polyp in the Duodenum

Kitaoka, Fumio; Shiogama, Toshiaki; Mizutani, Akimasa; Tsurunaga, Yasutaka; Fukui, Hiroshi; Higami, Yoshikazu; Shimokawa, Isao; Taguchi, Takashi; Kanematsu, Takashi

doi:10.1159/000077392

Cited by 33 publications

(29 citation statements)

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“…Few of these reports have described the endoscopic characteristics of these polyps in detail. The polyp in each of our three cases showed an irregularly lobular or nodular surface, whereas adenomas often show a regularly nodular or granular surface; this finding was compatible with previous reports [2,[5][6][7][8][9] . The color of the polyp was whitish in all of our cases, whereas previous reports have described the polyps to be red to normal color [6,7,9] .…”

Section: Discussionsupporting

confidence: 93%

“…A MEDLINE search of the English language literature revealed only nine well-documented cases of a solitary Peutz-Jeghers-type hamar tomatous polyp of the duodenum (Table 1) [2,[4][5][6][7][8][9] . Few of these reports have described the endoscopic characteristics of these polyps in detail.…”

Section: Discussionmentioning

confidence: 99%

“…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.…”

Section: Introductionmentioning

confidence: 99%

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

Suzuki

Shoji

Ikeda³

et al. 2008

WJG

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A solitary Peutz-Jeghers-type hamartomatous polyp in the duodenum is rare, and few reports have described its characteristic endoscopic features. We describe three cases of solitary Peutz-Jeghers-type hamartomatous polyp and their endoscopic findings in detail. The polyp in all of our three cases showed an irregularly lobular or nodular surface, whereas adenomas often show a regularly nodular or granular surface. The color of the polyp was whitish in all of our cases. In the present cases, close observation by endoscopy revealed that the solitary Peutz-Jeghers-type hamartomatous polyps looked whitish because of the presence of diffusely scattered white spots on the surface of the polyps. Duodenal polyps that exhibit the aforementioned endoscopic characteristics may be diagnosed as Peutz-Jeghers-type hamartomatous polyps and treated by polypectomy because of the malignant potential.

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Section: Discussionsupporting

confidence: 93%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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

Suzuki

Shoji

Ikeda³

et al. 2008

WJG

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“…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).…”

Section: Discussionmentioning

confidence: 99%

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

Suzuki

Higuchi

Shimizu

et al. 2015

WJG

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Solitary duodenal Peutz-Jeghers (PJ)-type hamartomatous polyps are rare and considered a different disease entity than classic PJ syndrome. We describe the case of an 89-year-old man admitted to our emergency department with symptoms of acute cholangitis, liver dysfunction, and slight jaundice. Magnetic resonance imaging showed multiple signal voids, reflecting choledocholithiasis, and an ovalshaped tumor in the common bile duct (CBD). Following endoscopic retrograde cholangiopancreatography, the patient was diagnosed with a lower CBD tumor 20 mm in diameter. Endoscopic sphincterotomy was performed for choledocholithotomy, resulting in the expulsion of a large tumor with a stalk connected to the papilla of Vater. The tumor was successfully excised en bloc by endoscopic snare papillectomy. Histopathologic examination showed that the tumor was a PJ-type hamartomatous polyp. No mucocutaneous pigmentation of the skin was evident and the patient's family history was negative. Solitary duodenal PJ-type hamartomatous polyps are usually diagnosed incidentally during endoscopy for other indications because most of these tumors are asymptomatic or have nonspecific presentations. To our knowledge, this is the first reported solitary PJ-type polyp with intra-CBD growth treated by endoscopic snare papillectomy.

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“…A germline mutation of the STK11 gene located on chromosome 19p13.3 is responsible for development of PJS [4]. A solitary Peutz-Jeghers type hamartomatous polyp is a variant of PJS characterized by the presence of hamartomatous polyps in the absence of other manifestations of PJS [5]. These patients present with nonspecific symptoms with most diagnosed incidentally during endoscopy.…”

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Giant solitary Peutz-Jeaghers-Type Hamartomatous Polyp in the Duodenum Presenting as Gastric Outlet Obstruction

D¹,

B²,

Manohar³

et al. 2016

Surgery

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Journal of SurgeryKeywords: Peutz-jeaghers; Hamartomatous polyp; Giant polyp; Gastric outlet obstruction IntroductionPeutz-Jeghers syndrome (PJS), first described by Peutz in 1921 is a rare, autosomal dominant disorder characterized by multiple gastrointestinal hamartomatous polyps and muco cutaneous pigmentation around the lips and oral cavity [1][2][3]. A germline mutation of the STK11 gene located on chromosome 19p13.3 is responsible for development of PJS [4]. A solitary Peutz-Jeghers type hamartomatous polyp is a variant of PJS characterized by the presence of hamartomatous polyps in the absence of other manifestations of PJS [5]. These patients present with nonspecific symptoms with most diagnosed incidentally during endoscopy. We are reporting a case of giant solitary Peutz-Jeaghers-type hamartomatous polyp in the duodenum in a young girl presenting as gastric outlet obstruction. Case ReportAn 18 year old girl presented with history of recurrent non bilious vomiting of 4 month duration associated with vague upper abdominal pain. On abdominal examination, she had a mass in right hypochondrium with features suggestive of mass arising from the pancreas. She didn't have any peri oral muco cutaneous pigmentation. Oesophago gastro duodenoscopy (OGD scopy) revealed a polypoidal mass of about 10 × 6 cm arising from the medial wall of second part of duodenum close to ampulla of vater ( Figure 1A). Biopsy from the mass was reported as villous adenoma with severe dysplasia. Contrast Enhanced Computed Tomography (CECT) of the abdomen revealed mass of size arising from the second part of duodenum with loss of fat planes with pancreatic head ( Figure 1B). The mass was abutting superior mesenteric vein and right renal vein ( Figure 1C). Patient underwent pylorus preserving pancreaticoduodenectomy ( Figure 1D). Post operatively she developed post-operative pancreatic fistula (POPF) grade B which was managed conservatively. Histopathological examination of the specimen revealed solitary Peutz-Jeaghers-type hamartomatous polyp arising from the 2 nd part of duodenum involving the ampulla of vater ( Figure 1E and F). There was mild dysplasia, however no evidence of invasive malignancy. All the resection margins were free and lymph nodes were reactive. After recovery she underwent colonoscopy which was within normal limits. On retrospective enquiry, patient's parents had 2 nd degree consanguineous marriage though there was no family history of any hereditary disorders. At 8 months follow up, patient is disease free and in good health. At last follow up, patient underwent colonoscopy to rule out any other polyps. DiscussionA solitary Peutz-Jeghers type hamartomatous polyp in the duodenum was first described in 1986 by Bott et al. [6]. This disorder differs from Peutz-Jeghers Syndrome (PJS) in following characteristics: Diagnosis at a more advanced age, absence of mutation of the STK11/ LKB-1 gene and family history as well as lack of muco-cutaneous pigmentation [5]. However in many cases it is difficult to determine whether t...

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A Solitary Peutz-Jeghers-Type Hamartomatous Polyp in the Duodenum

Cited by 33 publications

References 14 publications

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

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

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

Giant solitary Peutz-Jeaghers-Type Hamartomatous Polyp in the Duodenum Presenting as Gastric Outlet Obstruction

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