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CASE REPORT
Year : 2020  |  Volume : 8  |  Issue : 4  |  Page : 199-201

Heterotopic gastric mucosa in the proximal esophagus manifesting as upper gastrointestinal bleeding: An uncommon presentation


1 Department of Pathology, Hamdard Institute of Medical Sciences and Research, New Delhi, India
2 Department of Medicine, Hamdard Institute of Medical Sciences and Research, New Delhi, India

Date of Submission21-Mar-2021
Date of Acceptance22-Jun-2021
Date of Web Publication30-Dec-2021

Correspondence Address:
Sabina Khan
Department of Pathology, Hamdard Institute of Medical Sciences and Research, Jamia Hamdard, New Delhi - 110 062
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ssj.ssj_67_21

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  Abstract 


Heterotopic gastric mucosa of the proximal esophagus (HGMPE) also referred to as inlet patch is a salmon-colored patch located just distal to the upper esophageal sphincter. HGMPE is usually asymptomatic detected incidentally during endoscopy. Its clinical significance is mainly acid-related or neoplastic complications. Here, we present a case of a 58-year female presenting in the casualty with two episodes of upper gastrointestinal (UGI) bleeding. UGI Endoscopy revealed a large esophageal ulcer 1.5 cm × 1 cm just below the upper esophageal sphincter (UES). Biopsy from this area revealed a diagnosis of HGMPE. UGI bleed occurring as a complication of inlet patch has been very rarely reported. Only one case in literature is present making this case report the second one. This case highlights the importance of careful endoscopic evaluation of the UES region along with biopsy which may reveal heterotopic gastric mucosa in patients with unexplained UGI bleed.

Keywords: Endoscopy, gastrointestinal bleeding, heterotopic gastric mucosa of the proximal esophagus


How to cite this article:
Pendharkar D, Khan S, Hameed S, Vatsya S. Heterotopic gastric mucosa in the proximal esophagus manifesting as upper gastrointestinal bleeding: An uncommon presentation. Saudi Surg J 2020;8:199-201

How to cite this URL:
Pendharkar D, Khan S, Hameed S, Vatsya S. Heterotopic gastric mucosa in the proximal esophagus manifesting as upper gastrointestinal bleeding: An uncommon presentation. Saudi Surg J [serial online] 2020 [cited 2022 Jan 28];8:199-201. Available from: https://www.saudisurgj.org/text.asp?2020/8/4/199/334511




  Introduction Top


Heterotopic gastric mucosa of the proximal esophagus (HGMPE) also called “inlet patch” or “cervical inlet patch” is an island of ectopic gastric mucosa found mostly in the proximal esophagus.[1],[2] The inlet patch is thought to be a remnant of gastric mucosa from embryologic development of the gut. The incidences which are reported vary from <1% to 13.8%.[3],[4] The higher incidence has been reported in autopsy studies which is up to 70%.[1],[2]

Most of the symptoms caused by HGMPE are mild and their management depends on the severity of symptoms. The most common symptom reported is laryngopharyngeal reflux (LPR) symptoms and its prevalence has been reported to be as high as 73.1%.[5] Serious and significant complications of HGMPE have been reported in both adults and the pediatric population. HGMPE presenting as upper gastrointestinal (UGI) bleeding have been very rarely reported in the literature.

Hereby, we are presenting a case of 58-year-old female who presented with two episodes of UGI bleeding which on further investigation revealed HGMPE.


  Case Report Top


A 58-year-old female presented in the casualty with two episodes of UGI bleeding with signs of hypovolemia which warranted blood transfusion. There was no history of gastrointestinal complaints or any use of drugs/alcohol.

Upper gastroesophageal endoscopy revealed a large oesophageal ulcer 1.5 cm × 1 cm just below the upper esophageal sphincter (UES). The rest of the surrounding mucosa was unremarkable [Figure 1]. Complete esophageal gastroduodenoscopy was performed which revealed no other possible source of gastrointestinal bleeding except this ulcer in esophagus.
Figure 1: Esophageal ulcer 1.5 cm × 1 cm below the upper oesophageal sphincter

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Biopsy from the ulcerated mucosa was taken and sent to the histopathology department of our institute with suspicion of malignancy. Histopathology of this area showed gastric mucosa of cardiac type, lined by columnar mucosal cells adjacent to fragments of stratified squamous epithelium which showed no abnormality in the surrounding esophageal mucosa [Figure 2] and [Figure 3]. There was no evidence of Helicobacter pylori infection or any metaplasia/dysplasia. Based on these histological findings a diagnosis of HGMPE was made.
Figure 2: Low power microphotograph showing unremarkable esophageal mucosa with heterotopic unremarkable gastric mucosa (H and E stain, ×10) (inset image showing heterotopic gastric glands, ×40)

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Figure 3: High power showing stratified squamous mucosa with adjacent gastric (cardiac) type mucosa (H and E stain, ×40)

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  Discussion Top


HGMPE was first reported by Schmidt et al. as an aberrant gastric fundus-type epithelium present in the proximal part of esophagus.[1] There are three proposed theories on the development of HGMPE.[1],[2],[6],[7]

  1. ”HGMPE is congenital in origin” is the most widely accepted theory. At 24 weeks of gestation and during the development of esophagus, squamous lining replaces the columnar lining starting from the mid esophagus moving in both directions. The proximal part of esophagus is usually the last part to get stratification and this accounts for the common finding of heterotopic gastric mucosa in the proximal part of esophagus[1],[2]
  2. Another theory says about metaplastic transformation of the squamous lining to the columnar lining from chronic acid injury as seen in Barrett's esophagus.[6] The underlying pathogenesis which is considered is “proliferation of remnant pluripotent cells as result of acid injury”
  3. The third theory suggests rupture of proximal esophageal retention cystic glands.[7]


Von Rahden et al. proposed a clinicopathologic classification which categorized HGMPE into five distinct groups based on their clinical, endoscopic, and histological characteristics.[1]

The majority of patients having HGMPE are asymptomatic and it is detected incidentally during evaluation for other gastrointestinal complaints, this is categorized as Type-I HGMPE. Type II and Type III (nonneoplastic manifestations) have symptoms probably related to the acid produced by the patch such as LPR symptoms, strictures, webs, fistula, and bleeding.

Type IV and Type V are the least common manifestations which are histological or neoplastic changes.[1],[2] Acid-related symptoms are often seen in younger patients whereas neoplastic manifestations have been reported mainly in the elderly population.

In our case, the presenting symptom was two episodes of UGI bleeding which were serious enough to warrant blood transfusion. Bleeding is the rarest manifestation of HGMPE. In previous literature, only one case of UGI bleeding caused by HGMPE is described by Bataller et al.[8] Ours is the second case report till now in which the patient presented with UGI bleeding as a complication of HGMPE, which is very rare.

On endoscopy, HGMPE appears as a salmon-colored, ovoid-to-round patch that is distinct from the surrounding esophageal squamous mucosa.[9] HGMPE is often missed during endoscopy as the proximal esophagus is many a times neglected or briefly examined during routine endoscopic examination. Most HGMPE are located on the lateral walls, typically a few centimeters distal to the UES.[1],[2] The size can vary from microscopic to 3–5 cms. Mostly patients have a single patch while in those having multiple patches, they are small and present closely to other patches.[9] In our case, a single salmon-colored patch was found just below the esophageal sphincter measuring 1.5 cm × 1.0 cm.

Histopathologic evaluation of HGMPE demonstrates gastric-type mucosa mostly containing chief and parietal cells. However, in our case cardiac type mucosa was seen with only mucus-secreting cells. UGI bleed occurring as a complication of inlet patch has been very rarely demonstrated till now. Follow-up may need to be considered for patients with complications of HGMPE.


  Conclusion Top


This case highlights the importance of careful endoscopic evaluation of the UES region along with its biopsy which may reveal heterotopic gastric mucosa in patients with unexplained UGI bleed. The clinical significance of HGMPE lies mainly because it can result in mucosal progression to dysplasia and even frank neoplastic transformation.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
von Rahden BH, Stein HJ, Becker K, Liebermann-Meffert D, Siewert JR. Heterotopic gastric mucosa of the esophagus: Literature-review and proposal of a clinicopathologic classification. Am J Gastroenterol 2004;99:543-51.  Back to cited text no. 1
    
2.
Chong VH. Heterotopic gastric mucosal patch of the proximal esophagus. In: Pascu O, editor. Gastrointestinal Endoscopy. Croatia: InTech Publishing; 2011. p. 125-48.  Back to cited text no. 2
    
3.
Maconi G, Pace F, Vago L, Carsana L, Bargiggia S, Bianchi Porro G. Prevalence and clinical features of heterotopic gastric mucosa in the upper oesophagus (inlet patch). Eur J Gastroenterol Hepatol 2000;12:745-9.  Back to cited text no. 3
    
4.
Ohara M. Incidence of heteroptopic gastric mucosa in the upper esophagus in first time narrow banding image endoscopy of consecutive 900 patients. Gastrointest Endosc 2010;71:316-7.  Back to cited text no. 4
    
5.
Chong VH, Jalihal A. Heterotopic gastric mucosal patch of the esophagus is associated with higher prevalence of laryngopharyngeal reflux symptoms. Eur Arch Otorhinolaryngol 2010;267:1793-9.  Back to cited text no. 5
    
6.
Avidan B, Sonnenberg A, Chejfec G, Schnell TG, Sontag SJ. Is there a link between cervical inlet patch and Barrett's esophagus? Gastrointest Endosc 2001;53:717-21.  Back to cited text no. 6
    
7.
Meining A, Bajbouj M. Erupted cysts in the cervical esophagus result in gastric inlet patches. Gastrointest Endosc 2010;72:603-5.  Back to cited text no. 7
    
8.
Bataller R, Bordas JM, Ordi J, Llach J, Elizalde JI, Mondelo F. Upper gastrointestinal bleeding: A complication of “inlet patch mucosa” in the upper esophagus. Endoscopy 1995;27:282.  Back to cited text no. 8
    
9.
Chong VH. Clinical significance of heterotopic gastric mucosal patch of the proximal esophagus. World J Gastroenterol 2013;19:331-8.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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