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CASE REPORT
Year : 2019  |  Volume : 9  |  Issue : 2  |  Page : 115-117  

Pancreatic and gastric heterotopia in the gallbladder: A rare incidental finding


Department of Pathology, Hamdard Institute of Medical Sciences and Research, Jamia Hamdard, New Delhi, India

Date of Submission28-Mar-2018
Date of Acceptance11-Feb-2019
Date of Web Publication15-Apr-2019

Correspondence Address:
Dr. Shaan Khetrapal
Department of Pathology, Hamdard Institute of Medical Sciences and Research, Hamdard Nagar, New Delhi - 110 062
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijabmr.IJABMR_109_18

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   Abstract 


Heterotopic or ectopic tissue is a congenital anomaly, which is defined as the presence of the tissue outside its normal location, without neural, vascular, or anatomic connection with the main body of an organ in which it normally exists. This tissue is usually discovered incidentally and may be asymptomatic or may present with nonspecific gastrointestinal (GI) symptoms. Pancreatic and gastric heterotopia are the two predominantly occurring heterotopic tissues in the GI tract.[1],[2] The prevalence of ectopic pancreatic tissue in the GI tract ranges from 0.6% to 13.7% of autopsy series and it can be present anywhere in the GI tract with the most common localizations being stomach (27.5%), duodenum (25.5%), colon (15.9%), esophagus, and Meckel's diverticulum.[3],[4],[5] It is a rare finding in the gallbladder and its prevalence has not been ascertained due to lack of large-scale studies and systematic review of literature. Similarly, heterotopic gastric tissue is common throughout the GI tract from the tongue to the rectum,[6],[7] but it is extremely rare in the gallbladder with only around 34 cases reported in literature so far, while other cases of different types of heterotopic tissues in the gallbladder such as liver tissue and others such as adrenal and thyroid tissues have been described.[8] The most common presentation of ectopic tissue in the gallbladder is colicky pain in the epigastrium or right upper quadrant sometimes associated with nausea and vomiting. Here, we are presenting two incidentally detected cases, each of gastric and pancreatic heterotopias in the gallbladder.

Keywords: Chromogranin, heterotopia, pancreatic tissue


How to cite this article:
Pendharkar D, Khetrapal S, Jairajpuri ZS, Rana S, Jetley S. Pancreatic and gastric heterotopia in the gallbladder: A rare incidental finding. Int J App Basic Med Res 2019;9:115-7

How to cite this URL:
Pendharkar D, Khetrapal S, Jairajpuri ZS, Rana S, Jetley S. Pancreatic and gastric heterotopia in the gallbladder: A rare incidental finding. Int J App Basic Med Res [serial online] 2019 [cited 2019 Aug 20];9:115-7. Available from: http://www.ijabmr.org/text.asp?2019/9/2/115/256189




   Introduction Top


Heterotopic or ectopic tissue is a congenital anomaly, which is defined as the presence of the tissue outside its normal location, without neural, vascular, or anatomic connection with the main body of an organ in which it normally exists. This tissue is usually discovered incidentally and may be asymptomatic or may present with nonspecific gastrointestinal (GI) symptoms. Pancreatic and gastric heterotopia are the two predominantly occurring heterotopic tissues in the GI tract.[1],[2] The prevalence of ectopic pancreatic tissue in the GI tract ranges from 0.6% to 13.7% of autopsy series and it can be present anywhere in the GI tract with the most common localizations being stomach (27.5%), duodenum (25.5%), colon (15.9%), esophagus, and Meckel's diverticulum.[3],[4],[5] It is a rare finding in the gallbladder and its prevalence has not been ascertained due to lack of large-scale studies and systematic review of literature. Similarly, heterotopic gastric tissue is common throughout the GI tract from the tongue to the rectum,[6],[7] but it is extremely rare in the gallbladder with only around 34 cases reported in literature so far, while other cases of different types of heterotopic tissues in the gallbladder such as liver tissue and others such as adrenal and thyroid tissues have been described.[8] The most common presentation of ectopic tissue in the gallbladder is colicky pain in the epigastrium or right upper quadrant sometimes associated with nausea and vomiting. Here, we are presenting two incidentally detected cases, each of gastric and pancreatic heterotopias in the gallbladder.


   Case Reports Top


Case 1

A 35-year-old male presented with colicky pain in the abdomen. On ultrasonography, multiple stones were detected in the gallbladder with features of cholecystitis. Cholecystectomy was done, and on gross examination, the gallbladder was measured 7.5 cm × 2.5 cm with unremarkable serosal surface. On cutting open, cut surface shows partially velvety and partially hemorrhagic mucosa with multiple crushed sediments of yellow stones present in the lumen. No growth or polypoidal lesion was noted.

Microscopy revealed partially denuded and atrophic mucosa with wall which showed numerous benign looking pancreatic acini forming lobular architecture exhibiting both exocrine and endocrine components. Endocrine component was confirmed with the help of chromogranin-A immunohistochemistry and showed focal positivity [Figure 1]. The subepithelium showed acute on chronic inflammatory and muscular hypertrophy.
Figure 1: Heterotopic pancreatic tissue in the wall of the gallbladder (H and E, ×10). Inset showing few islet cells in heterotopic pancreatic tissue showing immunoreactivity to chromogranin-A (×40)

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Case 2

A 63-year-old female presented with symptoms of cholelithiasis. Cholecystectomy was done, and the sample was sent for histopathological analysis. On gross examination, the gallbladder was measured 10 cm × 2 cm with unremarkable external surface. Cut surface showed focally velvety mucosa and focal tiny sessile polypoidal areas measuring around 0.2–0.3 cm. Many small yellow stones were present in the lumen.

Microscopy revealed lining epithelium with few Rokitansky–Aschoff sinuses. The subepithelial tissue shows foci of unremarkable gastric fundic glands with surrounding tissue showing mild acute on chronic inflammation.


   Discussion Top


Heterotopia is the presence of normal tissue in an abnormal location. Heterotopic pancreatic tissue is presumed to result from an error during embryological development. The etiology of pancreatic heterotopia is not yet clear, but three theories have been proposed for the mechanism, namely (1) the separation of heterotopic pancreatic tissue from primitive pancreas during embryonic rotation; (2) pancreatic tissue is abnormally transported by the longitudinal growth of the intestine from lateral budding of the rudimentary pancreatic tissue while penetrating the intestinal wall,[9],[10] and this is the most accepted theory; and (3) another theory based on abnormalities in the Notch signaling system that leads to changes in differentiation in the developing foregut endoderm.[11]

Heterotopic pancreas is usually found incidentally during surgery or during microscopy. It is usually clinically silent and benign. In 1909, Heinrich et al. classified heterotopic pancreas into three types: Type I – with ducts, acini, and endocrine islets similar to those seen in normal pancreatic tissues; Type II – with a large number of acini, a few ducts, and no islets; and Type III – with numerous ducts, a few acini, and no islets. It was observed that 50% of ectopic tissues do not contain islet cells.[12]

Active pancreatic enzymes secreted into the gallbladder and biliary tract may lead to chronic inflammation, hyperplasia, and dysplasia of the gallbladder mucosa and even to carcinoma of the gallbladder.[13],[14] Qizilbash[15] reported a case in which the cause of acute symptoms was the inflammation of the heterotopic pancreatic tissue, resembling acute pancreatitis. It has been suggested that pancreatic heterotopia may also be the underlying cause of other cancer, for example, intraductal papillary mucinous neoplasm in the jejunum.[16] Malignant transformation of heterotopic pancreatic tissue is extremely rare. Goodarzi et al.[17] reviewed 31 documented cases of carcinoma arising in a heterotopic pancreas and mostly in the stomach and adenocarcinomas histologically. So far, neither ultrasound nor CT and magnetic resonance imaging can confirm the diagnosis of ectopic pancreas or gastric tissue preoperatively. Radical excision is the treatment of choice in pancreatic heterotopia and is aimed at avoiding potential complication.

The first case of heterotopic gastric mucosa was reported by Egyedi.[18] There are three hypotheses present regarding the etiology of heterotopic gastric mucosa:

  1. Developmental anomaly
  2. Heterotopic differentiation
  3. Metaplastic differentiation (23 hgb5).


The incidence in men was slightly higher around 20% higher than women. The age at discovery was ranging from 3 to 78 years with average age being 34 years. The finding of heterotopic gastric mucosa in the biliary tract is usually incidental, and sometimes, patients may present with right upper quadrant pain, abdominal discomfort, nausea, vomiting, and symptoms with biliary obstruction and jaundice. Under the age of 25 years, the presentation of heterotropic gastric mucosa in gall bladder is usually acute and for short duration, however, in older patients it can be associated with cholelithiasis and cholecystitis.[19] The gallbladder may be normal or may show sessile which is more common or pedunculated polypoidal lesion protruding into the lumen or may be localized thickening of the gallbladder wall. Carcinoma must be ruled out in polypoidal lesions of the gallbladder >1.0 cm. The incidence of gallbladder carcinoma is particularly high in sessile polypoidal lesion.[20] In the case presented above, the sessile polypoidal lesion present on the gallbladder mucosa was < 1cm in size which on microscopy revealed heterotopic gastric mucosa along with features of chronic cholecystitis without any features of gallbladder carcinoma.

It is necessary for a pathologist to be aware of these entities; heterotopic gastric mucosa in the biliary tract and some substantially important complications must also be considered when we deal with heterotopic gastric mucosa in the gallbladder, including ulceration of the gallbladder and possible malignant changes. Ishii et al.[21] suggested that heterotopic gastric mucosa may have the potential for carcinogenesis, but so far, no case of malignant transformation has been reported. Dysplasia in heterotopic gastric mucosa in the gallbladder has been reported.

In conclusion, surgeons must be aware of heterotopic gastric mucosa of the gallbladder, especially in young patients with cholecystitis and cholelithiasis with abdominal pain as a main complaint. As heterotopic tissue may promote carcinogenesis of the gallbladder, so close attention should be paid to any such lesions in this region.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Baudet JS, Alarcón-Fernández O, Sánchez Del Río A, Aguirre-Jaime A, León-Gómez N. Heterotopic gastric mucosa: A significant clinical entity. Scand J Gastroenterol 2006;41:1398-404.  Back to cited text no. 1
    
2.
Gokhale UA, Nanda A, Pillai R, Al-Layla D. Heterotopic pancreas in the stomach: A case report and a brief review of the literature. JOP 2010;11:255-7.  Back to cited text no. 2
    
3.
Kondi-Paphiti A, Antoniou AG, Kotsis T, Polimeneas G. Aberrant pancreas in the gallbladder wall. Eur Radiol 1997;7:1064-6.  Back to cited text no. 3
    
4.
Ben-Baruch D, Sandbank Y, Wolloch Y. Heterotopic pancreatic tissue in the gallbladder. Acta Chir Scand 1986;152:557-8.  Back to cited text no. 4
    
5.
Dolan RV, ReMine WH, Dockerty MB. The fate of heterotopic pancreatic tissue. A study of 212 cases. Arch Surg 1974;109:762-5.  Back to cited text no. 5
    
6.
Melato M, Ferlito A. Heterotopic gastric mucosa of the tongue and the oesophagus. ORL J Otorhinolaryngol Relat Spec 1975;37:244-54.  Back to cited text no. 6
    
7.
Jordan FT, Mazzeo RJ, Soiderer MH. Heterotopic gastric mucosa of the rectum. A rare cause of rectal bleeding. Arch Surg 1983;118:878-80.  Back to cited text no. 7
    
8.
Madrid C, Berrocal T, Gorospe L, Prieto C, Gámez M. Heterotopic gastric mucosa involving the gallbladder and biliary tree. Pediatr Radiol 2003;33:129-32.  Back to cited text no. 8
    
9.
Pilloni L, Cois A, Uccheddu A, Ambu R, Coni P, Faa G, et al. Complete pancreatic heterotopia of gallbladder with hypertrophic duct simulating an adenomyoma. World J Gastroenterol 2006;12:1786-7.  Back to cited text no. 9
    
10.
Elpek GO, Bozova S, Küpesiz GY, Oǧüş M. An unusual cause of cholecystitis: Heterotopic pancreatic tissue in the gallbladder. World J Gastroenterol 2007;13:313-5.  Back to cited text no. 10
    
11.
Artavanis-Tsakonas S, Rand MD, Lake RJ. Notch signaling: Cell fate control and signal integration in development. Science 1999;284:770-6.  Back to cited text no. 11
    
12.
Zinkiewicz K, Juśkiewicz W, Zgodziński W, Szumiło J, Cwik G, Furtak J, et al. Ectopic pancreas: Endoscopic, ultrasound and radiological features. Folia Morphol (Warsz) 2003;62:205-9.  Back to cited text no. 12
    
13.
Beltrán MA, Barría C. Heterotopic pancreas in the gallbladder: The importance of an uncommon condition. Pancreas 2007;34:488-91.  Back to cited text no. 13
    
14.
Lee SW, Yun SP, Seo HI. Heterotopic pancreas of the gallbladder associated with segmental adenomyomatosis of the gallbladder. J Korean Surg Soc 2013;84:309-11.  Back to cited text no. 14
    
15.
Qizilbash AH. Acute pancreatitis occurring in heterotopic pancreatic tissue in the gallbladder. Can J Surg 1976;19:413-4.  Back to cited text no. 15
    
16.
Okamoto H, Fujishima F, Ishida K, Tsuchida K, Shimizu T, Goto H, et al. Intraductal papillary mucinous neoplasm originating from a jejunal heterotopic pancreas: Report of a case. Surg Today 2014;44:349-53.  Back to cited text no. 16
    
17.
Goodarzi M, Rashid A, Maru D. Invasive ductal adenocarcinoma arising from pancreatic heterotopia in rectum: Case report and review of literature. Hum Pathol 2010;41:1809-13.  Back to cited text no. 17
    
18.
Egyedi L. Case of polypus of gallbladder containing aberrant gastricmucous membrane (in Hungarian). Gyogyaszat (Gyogyastat), 1934;74:596-599.'  Back to cited text no. 18
    
19.
Daud MS, Salomão FC, Salomão EC, Salomão BC. Gastric heterotopia together with squamous metaplasia in the gallbladder. Acta Gastroenterol Latinoam 2007;37:164-7.  Back to cited text no. 19
    
20.
Ishikawa O, Ohhigashi H, Imaoka S, Nakaizumi A, Kitamura T, Sasaki Y, et al. The difference in malignancy between pedunculated and sessile polypoid lesions of the gallbladder. Am J Gastroenterol 1989;84:1386-90.  Back to cited text no. 20
    
21.
Ishii Y, Ohmori K, Suzuki M, Hoshi T, Watanabe I, Endo S, et al. Heterotopic gastric mucosa in the wall of the gallbladder: A case report and review of the literature. Nihon Geka Gakkai Zasshi 1985;86:868-76.  Back to cited text no. 21
    


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