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CASE REPORT Table of Contents   
Year : 2008  |  Volume : 51  |  Issue : 2  |  Page : 240-241
Gastric heterotopia of the anus: Report of two rare cases and review of the literature


1 Department of Pathology, Al Jahra Hospital, Kuwait
2 Department of Histopathology, Mubarak Al-Kabeer Hospital, Kuwait
3 Department of Surgery, Al Jahra Hospital, Kuwait

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   Abstract 

Heterotopic gastric mucosa is an extremely rare occurrence in the anorectal region, with 41 reported cases till date. Of these, in only nine cases the heterotopic tissue has been found to be located within 2 cm of the dentate line. We report two cases of gastric heterotopia in the anus - one, in a
55-year-old man; and the other, in a 35-year-old woman. Sigmoidoscopy showed presence of a single sessile anal polyp in the first patient and hemorrhoid in the other. Pathologic examination of the biopsy specimens revealed fundic-type gastric mucosa in both the cases. Both the patients had complete resolution of symptoms after the excision. We present these cases to highlight the significance of recognizing this unusual histologic entity. To the best of our knowledge, the second case represents the first reported description of gastric heterotopia in association with hemorrhoid.

Keywords: Anus, heterotopic gastric mucosa, rectum

How to cite this article:
Rifat Mannan AS, Kahvic M, Bharadwaj S, Grover VK. Gastric heterotopia of the anus: Report of two rare cases and review of the literature. Indian J Pathol Microbiol 2008;51:240-1

How to cite this URL:
Rifat Mannan AS, Kahvic M, Bharadwaj S, Grover VK. Gastric heterotopia of the anus: Report of two rare cases and review of the literature. Indian J Pathol Microbiol [serial online] 2008 [cited 2014 Sep 1];51:240-1. Available from: http://www.ijpmonline.org/text.asp?2008/51/2/240/41668



   Introduction Top


The term 'heterotopia' refers to the presence of a tissue at a site far from its natural occurrence. The presence of heterotopic gastric mucosa has been described occurring at various levels of the gastrointestinal tract. But its presence in the rectum is extremely rare. To the best of our knowledge, only 41 cases of gastric heterotopia have been reported in the anorectal region till date. [1],[2],[3],[4],[5],[6],[7] Of these, in only 9 cases, the tissue was located within 2 cm of the dentate line. [5] We report two cases of gastric heterotopia in the anus and discuss the relevant literature on the topic.


   Case Histories Top


Case 1

A 55-year-old man presented with altered bowel habits, abdominal bloating and a sense of incomplete evacuation of bowel of 4 months' duration. There was no history of rectal bleeding. He received multiple courses of antibiotics without any improvement. Colonoscopic examination revealed normal colonic mucosa, except for the presence of a single smooth, sessile polypoid lesion about 5 mm in diameter on the posterior wall of the anal canal, about 2 cm from the anal verge. The lesion was excised endoscopically. An upper GI endoscopy revealed normal results. The patient reported gradual resolution of symptoms over a period of 1 month. Repeat sigmoidoscopy 3 months after the excision did not reveal any pathology. He is currently on follow-up and is asymptomatic 1 year after the excision.

Case 2

A 35-year-old woman presented with anal pain of 1 year's duration and fresh bleeding per rectum for the preceding 1 month. Bleeding was not associated with defecation. Clinical examination revealed a hemorrhoid in 7 o'clock position, within 1.5 cm from the anal verge. A hemorrhoidectomy was performed. Colonoscopic examination and an upper GI endoscopy revealed normal results. Bleeding ceased following the excision and the patient is on follow-up for 2 years without any recurrence of symptoms.

Pathologic findings

In both the cases, the excised specimen, measuring 5 mm and 2 cm in diameter respectively, consisted of a sessile grayish white polyp. Microscopic examination demonstrated benign fundic-type gastric mucosa in both the specimens [Figure 1] and [Figure 2]. In the second case, gastric mucosa was seen sandwiched between anal mucosa at one end and colonic mucosa at the other end. Periodic acid schiff (PAS) - alcian blue stain highlighted the presence of gastric-type (neutral) mucin lining the glands.

A careful examination for Helicobacter pylori, including the use of Warthin-Starry stains, was negative in both the cases. In the second patient, multiple congested blood vessels were observed in the underlying stroma, consistent with hemorrhoid. There was no evidence of surface ulceration or epithelial dysplasia in either of the cases.


   Discussion Top


The presence of heterotopic gastric mucosa in the anorectal region is rare. Only 41 cases have been reported in international literature till date. [1],[2],[3],[4],[5],[6],[7] This is even rarer when located in the anus. Eleven cases, including the present two, described the heterotopic tissue within 2 cm of the dentate line. There was a coexisting hemorrhoid in the second patient, which to our knowledge is the first report of such unusual association.

The genesis of the heterotopic gastric mucosa at various levels of the gastrointestinal tract remains unresolved till date. Recent studies suggest that gastric differentiation (including "gastric heterotopia") in lower gastrointestinal tract might occur as a result of deregulation/reactivation of the homeobox genes which are responsible for epithelial differentiation in the gastrointestinal tract and such genetic derangement can be triggered by local inflammation as well. [8] Such an assessment suggests that gastric heterotopia-like lesions can occur due to prior inflammatory reactions as well and not necessarily be a result of epithelial displacement during the fetal developmental period.

Review of literature revealed that that men were more commonly affected (M:F, 26:15), with an average age at presentation of about 21 years (range, 1 day to 51 years). [3],[5],[6],[7] One of our patients was a woman. The most common presentation of rectal gastric heterotopia was that of painless rectal bleeding (68%), with perineal ulceration (24%), anal pain (11%), abdominal pain (11%) and melena (3%) occurring less commonly. [5] Symptoms were present from 1 day to 17 years prior to diagnosis. In our first case, the heterotopia was an incidental finding found on evaluation of persistent irritable bowel syndrome; while in the second case, heterotopic mucosa was identified on histologic workup of an excised hemorrhoid. The heterotopic tissue was most commonly identified in association with a polyp ( n = 20), followed by diverticula ( n = 7), ulcer ( n = 7) and in reddish appearing mucosal plaque, folds, or flaps ( n = 7). [5],[6],[7] Identification of the heterotopic mucosa is usually done on biopsy. Technetium-pertechnetate (99 m) scan can be helpful, which shows increased uptake in the heterotopic mucosa. [5] On microscopy, most of the cases of heterotopic mucosa have been found to be of fundic type ( n = 21), followed by mixed type ( n = 7), body type ( n = 6), not specified or definable ( n = 6). Only a single case was composed of pyloric-type mucosa and it was associated with an adenocarcinoma. [1] Both the current cases demonstrated fundic-type mucosa. Helicobacter pylori has been identified in gastric heterotopia of the rectum, [3],[4] but could not be identified in the current cases. Malignant transformation remains a possibility, though the exact incidence is not known. Adenocarcinoma arising in ectopic gastric mucosa in esophagus has been reported. [9] Vieth et al. [6] reported a case of pyloric gland adenoma within a gastric corpus heterotopia in the rectum.

Surgical excision or polypectomy is considered to be the preferred treatment modality, with complete resolution of symptoms reported in most of the patients. [3] In both of our cases, surgical excision resulted in amelioration of symptoms. Successful treatment with histamine 2 receptor antagonists or proton pump inhibitors has also been reported. [3],[4],[6] Close follow-up is mandatory, considering the reports of neoplastic transformation of the heterotopic mucosa, both in the stomach and elsewhere.

 
   References Top

1.Wolff M. Heterotopic gastric epithelium in the rectum: A report of three new cases with a review of 87 cases of gastric heterotopia in the alimentary canal. Am J Clin Pathol 1971;55:604-16.  Back to cited text no. 1  [PUBMED]  
2.Ewell GH, Jackson RH. Aberrant gastric mucosa in the rectum with ulceration and hemorrhage. Wis Med J 1939;38:641-3.  Back to cited text no. 2    
3.Devereaux CE, Devereaux RG. Heterotopic gastric mucosa of the rectum with a review of the literature. J Clin Gastroenterol 1994;19:41-5.  Back to cited text no. 3  [PUBMED]  
4.Srinavasan R, Loeweenstine H, Mayle JE. Sessile polypoid gastric heterotopia of rectum: A report of 2 cases and review of the literature. Arch Pathol Lab Med 1999;123:222-4.  Back to cited text no. 4    
5.Steele SR, Mullenix PS, Martin MJ, Ormseth E, Weppler E, Graham J, et al. Heterotopic gastric mucosa of the anus: A case report and review of the literature. Am Surg 2004;70:715-9.  Back to cited text no. 5  [PUBMED]  
6.Vieth M, Kushima R, de Jonge JP, Borchard F, Oellig F, Stolte M. Adenoma with gastric differentiation (so-called pyloric gland adenoma) in a heterotopic gastric corpus mucosa in the rectum. Virchows Arch 2005;446:542-5.  Back to cited text no. 6  [PUBMED]  [FULLTEXT]
7.Wildemore BM, Ciocca V, Infantolino A, O'Hara BJ. Gastric heterotopia of the rectum: A case report. Int J Pathol 2007;5:2.  Back to cited text no. 7    
8.Beck F, Chawengsaksophak K, Waring P, Playford RJ, Furness JB. Reprogramming of intestinal differentiation and intercalary regeneration in Cdx2 mutant mice. Proc Natl Acad Sci USA 1999;96:7318-23.  Back to cited text no. 8  [PUBMED]  [FULLTEXT]
9.Christensen WN, Sternberg SS. Adenocarcinoma of the upper esophagus arising in ectopic gastric mucosa: Two case reports and review of the literature. Am J Surg Pathol 1987;11:397-402.  Back to cited text no. 9  [PUBMED]  

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Correspondence Address:
Abul Ala Syed Rifat Mannan
PO Box 62276, Jahra - 02153
Kuwait
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DOI: 10.4103/0377-4929.41668

PMID: 18603693

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    Figures

  [Figure 1], [Figure 2]

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