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Ectopic gastric mucosa in the cervical esophagus presenting as a recurrent neck abscess: a case report
Journal of Pediatric Surgery (2010) 45, E15–E17
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Ectopic gastric mucosa in the cervical esophagus presenting as a recurrent neck abscess: a case report Paul 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
Key words: Cervical esophagus; Ectopic gastric mucosa; Cervical abscess
Abstract We report a unique case of ectopic gastric mucosa (EGM) in the cervical esophagus. The patient 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 a breach in the hypopharynx, which was treated conservatively, and a stenosis of the esophagus requiring balloon dilation. © 2010 Elsevier Inc. All rights reserved.
Ectopic gastric mucosa (EGM) is found in the upper cervical esophagus in 0.1% to 10% of endoscopic studies [1,2]; it is most often asymptomatic on presentation. However, when symptoms occur, the lesions responsible for them include stricture, upper esophageal web, esophagotracheal fistula, ulcer, and adenocarcinoma [1]. We present a case of EGM of the cervical esophagus manifesting as a recurrent neck abscess in a 4-year-old boy.
1. Case report A 4-year-old boy was referred to us for a 3-cm left cervical abscess associated with cervical lymphadenopathy, fever, and torticollis. A week before, he suffered from an upper respiratory tract infection. No signs of cellulitis, dysphonia, dyspnea, or dysphagia were noted. Ear, nose, and throat examination was otherwise unremarkable. Result of
⁎ Corresponding author. Tel.: +961 1 513 497 1268; fax: +961 1 615295. E-mail address: [email protected] (E. Riachy). 0022-3468/$ – see front matter © 2010 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2010.03.022
Epstein-Barr virus serology was negative. Cervical ultrasonography revealed the mass to be anechogenic, slightly heterogeneous, measuring 3.2 × 2.8 cm, and located above and lateral to the left thyroid lobe with multiple lymph nodes in the jugulocarotid axis. The mass was incised and drained, and purulent fluid was obtained and sent for culture. A Penrose drain was left in place. The cultures grew colonies of an unspecified streptococcus with low resistance level. The patient was discharged receiving cefadroxil 50 mg/(kg d). Three weeks later, the child was readmitted for a recurrence of the cervical abscess. Surgical excision of the abscess was performed. The abscessed cyst was found lateral to the trachea, medial to the cervical vessels and the sternocleidomastoid muscle, and superior to the left thyroid lobe. A third branchial arch cyst was suspected, and a more complete excision with fistulography was planned after the resolution of the infection. Two weeks later, the child was hospitalized for another recurrence of the abscess. Symptoms included local erythema and slight induration, without dysphagia, dysphonia, or dyspnea. Repeat cervical ultrasonography raised the suspicion of a fistulous tract extending posterolaterally to the left pyriform sinus. The child was reoperated for
E16
P. Daher et al.
Fig. 1 Histopathologic examination of the lesion showing glandular-type mucosal wall (G) abruptly alternating with a nonkeratinized stratified squamous (S) epithelium (hematoxylin-eosin, original magnification ×35). Higher-power magnification on the right also shows the presence of typical parietal cells (P).
excision of the abscessed cyst, and an intraoperative fistulogram was performed that showed a fistulous tract extending posterolaterally. Although the tissues were still inflamed, the fistulous tract was dissected and was found to communicate with another left cyst located between the trachea and the cervical spine. The latter was similarly excised and cauterized, and a drain was left in place. Histopathology of this last cyst showed gastric fundic mucosal lining with typical parietal cells consistent with EGM (Fig. 1). The fistulous tract showed fibrous and granulation tissue. On day 2 postoperatively, milk and saliva were noted draining from the drain site consistent with a leak. A gastrograffin contrast swallow revealed a breach at the level of the hypopharynx. The child was discharged with a nasogastric tube in place for enteral feeding, and the breach healed spontaneously. Later, the boy was admitted for dysphagia and drooling; and endoscopy was performed that showed an esophageal stenosis 2.0 cm below the upper esophageal sphincter. He was treated with serial endoscopic balloon dilation along with 2 local applications of mitomycin 1 mg/mL each for 2 minutes. Proximal and distal esophageal biopsies showed no residual EGM.
2. Discussion A recurring neck abscess in a 4-year-old child should raise the suspicion for the presence of a congenital lesion. The location of the abscess and the posterolateral fistula to the hypopharynx found on ultrasonography suggested that this may be a third branchial arch cyst [3]. However, the pathology studies revealed it was an EGM in the esophagus that fistulized to the subcutaneous tissues and formed the neck abscess. Ectopic gastric mucosa, initially described by Shmidt in 1805, 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, the gallbladder [6], and the rectum [7]. In the esophagus, it is most commonly located just below the upper esophageal sphincter [2]. It is distinguished by a specific cytokeratin staining pattern [2] from the metaplastic gastric epithelium secondary to gastroesophageal reflux that is most often found in the lower esophagus [1]. Ectopic gastric mucosa is regarded as a congenital condition. In the embryo, the foregut is lined by columnar epithelium. Transformation to pseudostratified squamous epithelium starts in the midesophagus and extends cranially and caudally. A focal failure of this transformation causes EGM [2]. Its extent can vary from small microscopic foci to macroscopically visible areas of red or salmon-colored velvety patches [2]. Histologically, EGM most often has a fundic-type epithelium along with parietal cells. Less frequently, it has a transitional cell type with a mixture of fundic and antral glands or an antral pattern, which is defined by the absence of chief cells and the presence of only few parietal cells [1]. Gastric parietal cells of the EGM have been proven to secrete hydrochloric acid [8]. This acid secretion accounts for most of the complications observed in association with this condition such as ulcer, laryngospasm, and tracheoesophageal fistula [9]. The induced chronic inflammation can also lead to formation of esophageal strictures and webs [2]. Progression to adenocarcinoma is exceptional, with very few cases reported in the literature [2]. The symptoms 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 with fistulas: tracheoesophageal fistulas in the case of esophageal EGM, considered by some authors as a perforated peptic ulcer in the EGM [9], and rectovesical fistulas when it comes to EGM in the rectum [7].
Ectopic gastric mucosa presenting as recurrent neck abscess To our knowledge, this is the first case in the pertinent literature that presents with a recurrent cervical abscess communicating through a fistula with the esophageal EGM.
References [1] Borhan-Manesh F, Farnum J. Incidence of heterotopic gastric mucosa in the upper esophagus. Gut 1991;32:968-72. [2] Von Rahden B, Stein H, et al. Heterotopic gastric mucosa of the esophagus: literature-review and proposal of a clinicopathologic classification. Am J Gastroenterol 2004;99:543-51. [3] Batuecas Caletrío A, Blanco Pérez P, et al. Persistence of the third branchial arch. Acta Otorrinolaringol Esp 2006;57:193-5. [4] Melato M, Ferlito A. Heterotopic gastric mucosa of the tongue and the oesophagus. ORL J Otorhinolaryngol Relat Spec 1975;37:244-54.
E17 [5] Mann NS, Mann SK, Rachut E. Heterotopic gastric tissue in the duodenal bulb. J Clin Gastroenterol 2000;30:303-6. [6] Xeropotamos N, Skopelitou AS, Batsis C, et al. Heterotopic gastric mucosa together with intestinal metaplasia and moderate dysplasia in the gall bladder: report of two clinically unusual cases with literature review. Gut 2001;48:719-23. [7] Kalani BP, Vaezzadeh K, Sieber WK. Gastric heterotopia in rectum complicated by rectovesical fistula. Dig Dis Sci 1983;28:378-80. [8] Galan AR, Katzka DA, Castell DO. Acid secretion from an esophageal inlet patch demonstrated by ambulatory pH monitoring. Gastroenterology 1998;115:1574-6. [9] Kohler B, Köhler G, Riemann JF. Spontaneous esophagotracheal fistula resulting from ulcer in heterotopic gastric mucosa. Gastroenterology 1988;95:828-30. [10] Daher P, Riachy E, Zeidan S, et al. Upper airway obstructive symptoms because of ectopic gastric mucosa in a newborn: a case report. J Pediatr Surg 2006;41:7-9.
www.elsevier.com/locate/jpedsurg
Ectopic gastric mucosa in the cervical esophagus presenting as a recurrent neck abscess: a case report Paul 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
Key words: Cervical esophagus; Ectopic gastric mucosa; Cervical abscess
Abstract We report a unique case of ectopic gastric mucosa (EGM) in the cervical esophagus. The patient 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 a breach in the hypopharynx, which was treated conservatively, and a stenosis of the esophagus requiring balloon dilation. © 2010 Elsevier Inc. All rights reserved.
Ectopic gastric mucosa (EGM) is found in the upper cervical esophagus in 0.1% to 10% of endoscopic studies [1,2]; it is most often asymptomatic on presentation. However, when symptoms occur, the lesions responsible for them include stricture, upper esophageal web, esophagotracheal fistula, ulcer, and adenocarcinoma [1]. We present a case of EGM of the cervical esophagus manifesting as a recurrent neck abscess in a 4-year-old boy.
1. Case report A 4-year-old boy was referred to us for a 3-cm left cervical abscess associated with cervical lymphadenopathy, fever, and torticollis. A week before, he suffered from an upper respiratory tract infection. No signs of cellulitis, dysphonia, dyspnea, or dysphagia were noted. Ear, nose, and throat examination was otherwise unremarkable. Result of
⁎ Corresponding author. Tel.: +961 1 513 497 1268; fax: +961 1 615295. E-mail address: [email protected] (E. Riachy). 0022-3468/$ – see front matter © 2010 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2010.03.022
Epstein-Barr virus serology was negative. Cervical ultrasonography revealed the mass to be anechogenic, slightly heterogeneous, measuring 3.2 × 2.8 cm, and located above and lateral to the left thyroid lobe with multiple lymph nodes in the jugulocarotid axis. The mass was incised and drained, and purulent fluid was obtained and sent for culture. A Penrose drain was left in place. The cultures grew colonies of an unspecified streptococcus with low resistance level. The patient was discharged receiving cefadroxil 50 mg/(kg d). Three weeks later, the child was readmitted for a recurrence of the cervical abscess. Surgical excision of the abscess was performed. The abscessed cyst was found lateral to the trachea, medial to the cervical vessels and the sternocleidomastoid muscle, and superior to the left thyroid lobe. A third branchial arch cyst was suspected, and a more complete excision with fistulography was planned after the resolution of the infection. Two weeks later, the child was hospitalized for another recurrence of the abscess. Symptoms included local erythema and slight induration, without dysphagia, dysphonia, or dyspnea. Repeat cervical ultrasonography raised the suspicion of a fistulous tract extending posterolaterally to the left pyriform sinus. The child was reoperated for
E16
P. Daher et al.
Fig. 1 Histopathologic examination of the lesion showing glandular-type mucosal wall (G) abruptly alternating with a nonkeratinized stratified squamous (S) epithelium (hematoxylin-eosin, original magnification ×35). Higher-power magnification on the right also shows the presence of typical parietal cells (P).
excision of the abscessed cyst, and an intraoperative fistulogram was performed that showed a fistulous tract extending posterolaterally. Although the tissues were still inflamed, the fistulous tract was dissected and was found to communicate with another left cyst located between the trachea and the cervical spine. The latter was similarly excised and cauterized, and a drain was left in place. Histopathology of this last cyst showed gastric fundic mucosal lining with typical parietal cells consistent with EGM (Fig. 1). The fistulous tract showed fibrous and granulation tissue. On day 2 postoperatively, milk and saliva were noted draining from the drain site consistent with a leak. A gastrograffin contrast swallow revealed a breach at the level of the hypopharynx. The child was discharged with a nasogastric tube in place for enteral feeding, and the breach healed spontaneously. Later, the boy was admitted for dysphagia and drooling; and endoscopy was performed that showed an esophageal stenosis 2.0 cm below the upper esophageal sphincter. He was treated with serial endoscopic balloon dilation along with 2 local applications of mitomycin 1 mg/mL each for 2 minutes. Proximal and distal esophageal biopsies showed no residual EGM.
2. Discussion A recurring neck abscess in a 4-year-old child should raise the suspicion for the presence of a congenital lesion. The location of the abscess and the posterolateral fistula to the hypopharynx found on ultrasonography suggested that this may be a third branchial arch cyst [3]. However, the pathology studies revealed it was an EGM in the esophagus that fistulized to the subcutaneous tissues and formed the neck abscess. Ectopic gastric mucosa, initially described by Shmidt in 1805, 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, the gallbladder [6], and the rectum [7]. In the esophagus, it is most commonly located just below the upper esophageal sphincter [2]. It is distinguished by a specific cytokeratin staining pattern [2] from the metaplastic gastric epithelium secondary to gastroesophageal reflux that is most often found in the lower esophagus [1]. Ectopic gastric mucosa is regarded as a congenital condition. In the embryo, the foregut is lined by columnar epithelium. Transformation to pseudostratified squamous epithelium starts in the midesophagus and extends cranially and caudally. A focal failure of this transformation causes EGM [2]. Its extent can vary from small microscopic foci to macroscopically visible areas of red or salmon-colored velvety patches [2]. Histologically, EGM most often has a fundic-type epithelium along with parietal cells. Less frequently, it has a transitional cell type with a mixture of fundic and antral glands or an antral pattern, which is defined by the absence of chief cells and the presence of only few parietal cells [1]. Gastric parietal cells of the EGM have been proven to secrete hydrochloric acid [8]. This acid secretion accounts for most of the complications observed in association with this condition such as ulcer, laryngospasm, and tracheoesophageal fistula [9]. The induced chronic inflammation can also lead to formation of esophageal strictures and webs [2]. Progression to adenocarcinoma is exceptional, with very few cases reported in the literature [2]. The symptoms 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 with fistulas: tracheoesophageal fistulas in the case of esophageal EGM, considered by some authors as a perforated peptic ulcer in the EGM [9], and rectovesical fistulas when it comes to EGM in the rectum [7].
Ectopic gastric mucosa presenting as recurrent neck abscess To our knowledge, this is the first case in the pertinent literature that presents with a recurrent cervical abscess communicating through a fistula with the esophageal EGM.
References [1] Borhan-Manesh F, Farnum J. Incidence of heterotopic gastric mucosa in the upper esophagus. Gut 1991;32:968-72. [2] Von Rahden B, Stein H, et al. Heterotopic gastric mucosa of the esophagus: literature-review and proposal of a clinicopathologic classification. Am J Gastroenterol 2004;99:543-51. [3] Batuecas Caletrío A, Blanco Pérez P, et al. Persistence of the third branchial arch. Acta Otorrinolaringol Esp 2006;57:193-5. [4] Melato M, Ferlito A. Heterotopic gastric mucosa of the tongue and the oesophagus. ORL J Otorhinolaryngol Relat Spec 1975;37:244-54.
E17 [5] Mann NS, Mann SK, Rachut E. Heterotopic gastric tissue in the duodenal bulb. J Clin Gastroenterol 2000;30:303-6. [6] Xeropotamos N, Skopelitou AS, Batsis C, et al. Heterotopic gastric mucosa together with intestinal metaplasia and moderate dysplasia in the gall bladder: report of two clinically unusual cases with literature review. Gut 2001;48:719-23. [7] Kalani BP, Vaezzadeh K, Sieber WK. Gastric heterotopia in rectum complicated by rectovesical fistula. Dig Dis Sci 1983;28:378-80. [8] Galan AR, Katzka DA, Castell DO. Acid secretion from an esophageal inlet patch demonstrated by ambulatory pH monitoring. Gastroenterology 1998;115:1574-6. [9] Kohler B, Köhler G, Riemann JF. Spontaneous esophagotracheal fistula resulting from ulcer in heterotopic gastric mucosa. Gastroenterology 1988;95:828-30. [10] Daher P, Riachy E, Zeidan S, et al. Upper airway obstructive symptoms because of ectopic gastric mucosa in a newborn: a case report. J Pediatr Surg 2006;41:7-9.