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Journal of Pediatric Surgery (2010) 45, E15–E17

Ectopic gastric mucosa in the cervical esophagus presentingas a recurrent neck abscess: a case reportPaul Daher, Evana Francis, Lara Raffoul, Edward Riachy⁎

Department of Pediatric Surgery, Hotel Dieu de France Hospital, PO Box: 16- 6830, Beirut, Lebanon

Received 12 February 2010; revised 16 March 2010; accepted 17 March 2010

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Key words:Cervical esophagus;Ectopic gastric mucosa;Cervical abscess

Abstract We report a unique case of ectopic gastric mucosa (EGM) in the cervical esophagus. Thepatient presented with a recurrent cervical abscess communicating through a fistula with the EGM.Surgical treatment consisted of complete excision. The postoperative course was complicated by abreach in the hypopharynx, which was treated conservatively, and a stenosis of the esophagus requiringballoon dilation.© 2010 Elsevier Inc. All rights reserved.

Ectopic gastric mucosa (EGM) is found in the uppercervical esophagus in 0.1% to 10% of endoscopic studies[1,2]; it is most often asymptomatic on presentation.However, when symptoms occur, the lesions responsiblefor them include stricture, upper esophageal web, esophago-tracheal fistula, ulcer, and adenocarcinoma [1]. We present acase of EGM of the cervical esophagus manifesting as arecurrent neck abscess in a 4-year-old boy.

1. Case report

A 4-year-old boy was referred to us for a 3-cm leftcervical abscess associated with cervical lymphadenopathy,fever, and torticollis. A week before, he suffered from anupper respiratory tract infection. No signs of cellulitis,dysphonia, dyspnea, or dysphagia were noted. Ear, nose, andthroat examination was otherwise unremarkable. Result of

⁎ Corresponding author. Tel.: +961 1 513 497 1268; fax: +961 1 615295.E-mail address: eddy_riachy@msn.com (E. Riachy).

022-3468/$ – see front matter © 2010 Elsevier Inc. All rights reserved.oi:10.1016/j.jpedsurg.2010.03.022

Epstein-Barr virus serology was negative. Cervical ultraso-nography revealed the mass to be anechogenic, slightlyheterogeneous, measuring 3.2 × 2.8 cm, and located aboveand lateral to the left thyroid lobe with multiple lymph nodesin the jugulocarotid axis. The mass was incised and drained,and purulent fluid was obtained and sent for culture. APenrose drain was left in place. The cultures grew colonies ofan unspecified streptococcus with low resistance level. Thepatient was discharged receiving cefadroxil 50 mg/(kg d).

Three weeks later, the child was readmitted for arecurrence of the cervical abscess. Surgical excision of theabscess was performed. The abscessed cyst was found lateralto the trachea, medial to the cervical vessels and thesternocleidomastoid muscle, and superior to the left thyroidlobe. A third branchial arch cyst was suspected, and a morecomplete excision with fistulography was planned after theresolution of the infection.

Two weeks later, the child was hospitalized for anotherrecurrence of the abscess. Symptoms included localerythema and slight induration, without dysphagia, dys-phonia, or dyspnea. Repeat cervical ultrasonography raisedthe suspicion of a fistulous tract extending posterolaterallyto the left pyriform sinus. The child was reoperated for

Fig. 1 Histopathologic examination of the lesion showing glandular-type mucosal wall (G) abruptly alternating with a nonkeratinizedstratified squamous (S) epithelium (hematoxylin-eosin, original magnification ×35). Higher-power magnification on the right also shows thepresence of typical parietal cells (P).

E16 P. Daher et al.

excision of the abscessed cyst, and an intraoperativefistulogram was performed that showed a fistulous tractextending posterolaterally. Although the tissues were stillinflamed, the fistulous tract was dissected and was found tocommunicate with another left cyst located between thetrachea and the cervical spine. The latter was similarlyexcised and cauterized, and a drain was left in place.Histopathology of this last cyst showed gastric fundicmucosal lining with typical parietal cells consistent withEGM (Fig. 1). The fistulous tract showed fibrous andgranulation tissue.

On day 2 postoperatively, milk and saliva were noteddraining from the drain site consistent with a leak. Agastrograffin contrast swallow revealed a breach at the levelof the hypopharynx. The child was discharged with anasogastric tube in place for enteral feeding, and the breachhealed spontaneously.

Later, the boy was admitted for dysphagia and drooling;and endoscopy was performed that showed an esophagealstenosis 2.0 cm below the upper esophageal sphincter. Hewas treated with serial endoscopic balloon dilation alongwith 2 local applications of mitomycin 1 mg/mL each for 2minutes. Proximal and distal esophageal biopsies showed noresidual EGM.

2. Discussion

A recurring neck abscess in a 4-year-old child should raisethe suspicion for the presence of a congenital lesion. Thelocation of the abscess and the posterolateral fistula to thehypopharynx found on ultrasonography suggested that thismay be a third branchial arch cyst [3]. However, the pathologystudies revealed it was an EGM in the esophagus that fistulizedto the subcutaneous tissues and formed the neck abscess.

Ectopic gastric mucosa, initially described by Shmidt in1805, has been found in the esophagus; but it has also

been described in other sites of the gastrointestinal tract,such as the tongue [4], the duodenum [5], the jejunum, thegallbladder [6], and the rectum [7]. In the esophagus, it ismost commonly located just below the upper esophagealsphincter [2]. It is distinguished by a specific cytokeratinstaining pattern [2] from the metaplastic gastric epitheliumsecondary to gastroesophageal reflux that is most oftenfound in the lower esophagus [1]. Ectopic gastric mucosais regarded as a congenital condition. In the embryo, theforegut is lined by columnar epithelium. Transformation topseudostratified squamous epithelium starts in the mid-esophagus and extends cranially and caudally. A focalfailure of this transformation causes EGM [2]. Its extentcan vary from small microscopic foci to macroscopicallyvisible areas of red or salmon-colored velvety patches [2].Histologically, EGM most often has a fundic-typeepithelium along with parietal cells. Less frequently, ithas a transitional cell type with a mixture of fundic andantral glands or an antral pattern, which is defined by theabsence of chief cells and the presence of only few parietalcells [1].

Gastric parietal cells of the EGM have been proven tosecrete hydrochloric acid [8]. This acid secretion accountsfor most of the complications observed in association withthis condition such as ulcer, laryngospasm, and tracheoe-sophageal fistula [9]. The induced chronic inflammationcan also lead to formation of esophageal strictures andwebs [2]. Progression to adenocarcinoma is exceptional,with very few cases reported in the literature [2]. Thesymptoms described at presentation are dysphagia [2],stridor and dyspnea [10], recurrent episodes of sore throat,neck pain, and bronchitis.

Ectopic gastric mucosas have been associated withfistulas: tracheoesophageal fistulas in the case of esophagealEGM, considered by some authors as a perforated pepticulcer in the EGM [9], and rectovesical fistulas when it comesto EGM in the rectum [7].

E17Ectopic gastric mucosa presenting as recurrent neck abscess

To our knowledge, this is the first case in the pertinentliterature that presents with a recurrent cervical abscesscommunicating through a fistula with the esophageal EGM.

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