J Pathol Transl Med.  2023 Mar;57(2):128-131. 10.4132/jptm.2022.11.07.

Solitary Peutz-Jeghers type harmartomatous polyp in duodenum with gastric foveolar epithelium: a case report

• Choi E
• Lee J
• Park Y

• KMID: 2540499
• DOI: http://doi.org/10.4132/jptm.2022.11.07

Abstract

Peutz-Jeghers type hamartomatous polyp is known to be associated with Peutz-Jeghers syndrome, which shows characteristic multiple hamartomatous polyp involvement in the gastrointestinal tract, combined with mucocutaneous symptom, familial history of Peutz- Jeghers syndrome or STK11/LTB1 mutation. However, some cases showing histologic appearance of the polyps discovered in Peutz- Jeghers syndrome while lacking other diagnostic criteria of the syndrome have been reported, and these are called solitary Peutz- Jeghers type polyps. Herein, we report a case of solitary Peutz-Jeghers type polyp covered with heterotopic epithelium. The patient was 47-year-old female without any mucocutaneous symptoms nor familial history of Peutz-Jeghers syndrome. Microscopic examination revealed Peutz-Jeghers type hamartomatous polyp in duodenum covered with gastric type foveolar epithelium. Considering the definition of hamartomatous polyp, which is, the abnormal overgrowth of the indigenous epithelial component, the histological feature of current case is noteworthy in a point that it shows proliferation of heterotopic component, rather than the indigenous component.

Keyword

Hamartoma; Intestinal polyps; Duodenum; Gastric mucosa; Peutz-Jeghers syndrome

Figure

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  Fig. 1 Radiologic and endoscopic findings of the duodenal polyp. (A) Abdominal computed tomography image showing 2.9-cm-sized mass in duodenal 2nd portion. (B) A pedunculated polyp occupying half of the duodenal lumen discovered in endoscopic examination.

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  Fig. 2 Histological examination of the resected polyp. (A) Hematoxylin and eosin staining of the resected polyp showing arborizing smooth muscle bundles with overgrowth of superficial foveolar epithelium. (B) Foveolar type epithelium lying over pyloric type-like mucinous glands without dysplasia.

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  Fig. 3 Immunohistochemical stainings of the resected polyp. (A) Immunohistochemisty for MUC5AC confirms the presence of the overlying gastric type foveolar epithelium. (B) Pyloric gland-like structures showing positivity for MUC6 staining.

Reference

References

1. Brosens LA, Jansen M. Peutz-Jeghers syndrome. WHO Classification of Tumours Editorial Board. Digestive system tumours. WHO classification of tumours. 5th ed. Lyon: International Agency for Research on Cancer;2019. p. 545–6.

2. Sone Y, Nakano S, Takeda I, Kumada T, Kiriyama S, Hisanaga Y. Solitary hamartomatous polyp of Peutz-Jeghers type in the jejunum resected endoscopically. Gastrointest Endosc. 2000; 51:620–2.

3. Endo K, Kawamura K, Murakami K, et al. A case of jejunal solitary Peutz-Jeghers polyp with intussusception identified by double-balloon enteroscopy. Clin J Gastroenterol. 2020; 13:1129–35.

4. Oncel M, Remzi FH, Church JM, Goldblum JR, Zutshi M, Fazio VW. Course and follow-up of solitary Peutz-Jeghers polyps: a case series. Int J Colorectal Dis. 2003; 18:33–5.

5. Itaba S, Namoto M, Somada S, et al. Two cases of solitary Peutz-Jeghers-type hamartoma of the duodenum. Endoscopy. 2006; 38(Suppl 2):E32–3.

6. Liu BL, Zhou H, Risech M, Ky A, Houldsworth J, Ward SC. Solitary Peutz-Jeghers type polyp of jejunum with gastric fundic and antral gland lining mucosa: a case report and review of literature. Int J Surg Pathol. 2022; 30:539–42.

7. Ichiyoshi Y, Yao T, Nagasaki S, Sugimachi K. Solitary Peutz-Jeghers type polyp of the duodenum containing a focus of adenocarcinoma. Ital J Gastroenterol. 1996; 28:95–7.

8. Suzuki S, Hirasaki S, Ikeda F, Yumoto E, Yamane H, Matsubara M. Three cases of solitary Peutz-Jeghers-type hamartomatous polyp in the duodenum. World J Gastroenterol. 2008; 14:944–7.

9. Kato N, Sugawara M, Maeda K, Hosoya N, Motoyama T. Pyloric gland metaplasia/differentiation in multiple organ systems in a patient with Peutz-Jegher’s syndrome. Pathol Int. 2011; 61:369–72.