A Novel Technique of Concurrent Esophagoscopy and Transgastrostomy Gastroscopy to Dilate a Completely Obstructed Distal Esophageal Stricture in a Child Following Fundoplication

• Introduction
• Case Report
• Discussion
• References

We report a successful dilation of a completely obstructed distal esophageal stricture in a 4-year-old boy with combined immune deficiency syndrome, at 2 and half years after fundoplication and gastrostomy tube insertion. Barium studies and esophagoscopy had revealed complete obstruction of the lower esophagus. Transgastrostomy gastroscopy demonstrated a pinhole lumen through the fundoplication wrap; a guide wire was passed into the esophagus; and the stricture was dilated with Savary dilators. We presumed that the stricture was secondary to chronic esophagitis. The stricture was identified and successfully dilated using a novel technique of concurrent esophagoscopy and transgastrostomy gastroscopy.

Introduction

Completely obstructed esophageal stricture in children is a rare complication post fundoplication and usually required surgical intervention. Here we report a novel technique of concurrent esophagoscopy and trans-gastrostomy gastroscopy to dilate a completely obstructed distal esophageal stricture in a child post-fundoplication.

Case Report

A 4-year-old boy had a past medical history of combined immunodeficiency syndrome, severe gastroesophageal reflux disease (GERD) and aspiration pneumonia. By 15 months of age, he had been admitted to hospital several times for treatment of recurrent pneumonia, failure to thrive and diarrhea. His clinical presentation and immunological work-up results were most consistent with a diagnosis of combined immunodeficiency syndrome [1]. Severe combined immunodeficiency was ruled out. Since that time, he had been receiving regular intravenous immunoglobulins plus prophylactic co-trimoxazole.

Also at that time a Nissen fundoplication with insertion of a gastrostomy tube was carried out because of severe GERD and aspiration pneumonia. Acid suppression was stopped and he had since fed through a gastrostomy tube with marked improvement of his respiratory symptoms.

At 4 years of age, he was admitted with increasingly severe coughing spells associated with cyanosis, followed by vomiting of clear fluids; this had lasted for several months. During his stay in hospital, he had a few coughing spells associated with a drop in oxygen saturation to mid 80 s. He continued to regurgitate clear fluids and was not able to tolerate oral fluids or soft solids.

Barium swallow as well as barium study via the gastrostomy tube showed dilatation of the distal esophagus with no contrast crossing the esophageal stricture. Esophagoscopy showed a dilated and scarred lower esophagus and no obvious lumen was seen. The patient was taken to the operating room. Under fluoroscopic guidance with concurrent rigid esophagoscopy, repeated attempts to pass bougies through the gastrostomy, to reach the site of the stricture, failed to cannulate a potential lumen or to perforate a possibly thin membrane separating the esophageal lumen from the stomach.

Before proceeding to a surgical option, concurrent esophagoscopy and transgastrostomy gastroscopy were done in an attempt to identify a potential lumen through the stricture from below. Also a plan was made regarding the possibility of endoscopically perforating the membrane separating the esophageal pouch from the stomach cavity, using a guide wire under direct endoscopic vision [2].

Esophagoscopy using an Olympus GIFQ 140 (10.5-mm) endoscope showed a dilated blind-ended scarred lower esophagus. Again, no distal esophageal lumen could be identified at this repeat esophagoscopy (Figure [1]).

A transgastrostomy gastroscopy was performed, using a neonatal Olympus XP160 (5.9-mm) endoscope inserted through the gastrostomy lumen; this revealed an intact fundoplication wrap. A pinhole lumen was identified when the endoscope was inserted a short distance through the center of the wrap. A 0.035-inch Zebra guide wire was easily pushed through the pinhole (Figure [2]). Esophagoscopy showed the guide wire coming through the distal esophageal stricture in an area where no lumen had been seen previously (Figure [3]). The wire was snared and pulled through the patient’s mouth.

Using the standard technique, the tight stricture was dilated using a 5-mm diameter Savary dilator over the guide wire, under direct endoscopic vision as the dilator came through the fundoplication wrap. The wire was kept taut with both ends being held, one end coming through the mouth and the other through the gastrostomy lumen. The stricture was gradually dilated up to 12.8 mm in diameter. The tip of a neonatal scope could be passed through the stricture into the lower esophagus (Figure [4]).

Initially dilation was done once every 2 weeks for the first 6 weeks, using a Savary dilator up to 14 mm in diameter. The patient was able to tolerate thickened fluid and soft foods after two dilation sessions, and his aspiration episodes resolved. Subsequently the boy underwent dilation on a monthly basis because of recurrence of his dysphagia. In the last 6 months, with the use of triamcinolone injections at the stricture site immediately following the dilation procedure, the frequency of dilation has been reduced to once every 10 weeks.

The child has now been followed up for 13 months since the first dilation. In the last 2 months he has been weaned off of gastrostomy tube feeding and has tolerated frequent oral feeding with both solid and liquid foods. At the most recent endoscopy, for the first time a 9.8-mm scope was passed through the stricture prior to dilation. Our plan is to manage the child conservatively with intermittent esophageal dilation.

Discussion

This is the first report of a completely obstructed esophageal stricture that developed following fundoplication in a child with combined immunodeficiency syndrome [3]. We presumed that the stricture was secondary to chronic reflux esophagitis, as demonstrated by the scarred lower esophagus.

Despite the introduction of H₂ blockers and proton pump inhibitors, the incidence of stricture has not changed in the past three decades and has remained at 2 % in patients with GERD [4]. In a series of 126 infants and children diagnosed with GERD and followed up for 1.5 to 3.5 years, 5 % were found to have refractory esophagitis or stricture [5]. Esophageal strictures were seen in 11 % of pediatric patients who underwent fundoplication for GERD [6]. However in a long-term outcome study, there was no statistically significant difference between the rates of stricture in patients randomly allocated to receive either surgical or medical therapy for GERD [7].

Esophageal stricture secondary to opportunistic infections in immunodeficiency is rare. There are reports of esophageal strictures in patients with AIDS who develop ulcerative esophagitis [8].

In the case of a severe esophageal stricture with complete obstruction, esophageal resection or replacement has been the standard therapy. chiefly because less aggressive methods generally have failed [9]. In recent years, combined antegrade and retrograde dilation using esophagoscopy and transgastrostomy gastroscopy has been employed in the management of completely obstructed benign and malignant esophageal strictures in ten adults [2]. Luminal patency was re-established without esophageal perforation in all ten patients. In two cases, no lumen was identified and the obstructed membrane was perforated by advancing a wire in the light provided by the distal endoscope, the wire then being grasped by a biopsy forceps from the proximal endoscope. In our case, the residual lumen was identified using this novel technique of concurrent transgastrostomy gastroscopy and esophagoscopy, despite previous barium studies and esophagoscopies having shown an absence of communication. Perforation of the obstructed membrane was not required. Bougienage dilation was achieved with the retrograde passage of a guide wire through the stricture. Potential serious surgical and anesthetic complications from major surgical procedures were avoided. As both proximal and distal endoscopic imaging of the stricture are used, this technique also ensures that the wire is not passed into a false lumen, thereby reducing the risk of esophageal perforation during dilation.

In summary, in completely obstructed esophageal stricture, it is likely that a lumen is present within the stricture, and concurrent esophagoscopy and transgastrostomy gastroscopy should be done in order to identify a residual lumen. If no lumen is identified, before proceeding to other surgical options, an attempt should be made at endoscopic perforation of the stricture membrane to establish luminal patency.

References

• 1 Cooper M D, Lanier L L, Conley M E. et al . Immunodeficiency disorders.  Hematology (Am Soc Hematol Educ Program). 2003;  314-330
  CrossrefPubMedSearch in Google Scholar
• 2 Bueno R, Swanson S J, Jaklitsch M T. Combined antegrade and retrograde dilatation: a new endoscopic technique in the management of complex esophageal obstruction.  Gastrointest Endosc. 2001;  54 368-372
  CrossrefPubMedSearch in Google Scholar
• 3 Boeck A, Buckley R H, Schiff R I. Gastroesophageal reflux and severe combined immunodeficiency.  Allergy Clin Immunol. 1997;  99 420-424
  PubMedSearch in Google Scholar
• 4 Nayyar A K, Royston C, Bardhan K D. Oesophageal acid-peptic strictures in the histamine H2 receptor antagonist and proton pump inhibitor era.  Dig Liver Dis. 2003;  35 143-150
  CrossrefPubMedSearch in Google Scholar
• 5 Shepherd R W, Wren J, Evans S. et al . Gastroesophageal reflux in children. Clinical profile, course and outcome with active therapy in 126 cases.  Clin Pediatr (Phila). 1987;  26 55-60
  CrossrefPubMedSearch in Google Scholar
• 6 Fung K P, Seagram G, Pasieka J. et al . Investigation and outcome of 121 infants and children requiring Nissen fundoplication for the management of gastroesophageal reflux.  Clin Invest Med. 1990;  13 237-246
  PubMedSearch in Google Scholar
• 7 Spechler S J, Lee E, Ahnen D. et al . Long-term outcome of medical and surgical therapies for gastroesophageal reflux disease: follow-up of a randomized controlled trial.  JAMA. 2001;  285 2331-2338
  CrossrefPubMedSearch in Google Scholar
• 8 Wilcox C M. Esophageal strictures complicating ulcerative esophagitis in patients with AIDS.  Am J Gastroenterol. 1999;  94 339-343
  CrossrefPubMedSearch in Google Scholar
• 9 Gandhi R P, Cooper A, Barlow B A. Successful management of esophageal strictures without resection or replacement.  J Pediatr Surg. 1989;  24 745-750
  CrossrefPubMedSearch in Google Scholar

H. Q. Huynh, MBBS

Department of Pediatrics · University of Alberta

2C3 Walter C Mackenzie Health Sciences Centre · Edmonton · Alberta T6G 2R7 · Canada

Fax: 1-780-407-3507

Email: hien.huynh@ualberta.ca

References

• 1 Cooper M D, Lanier L L, Conley M E. et al . Immunodeficiency disorders.  Hematology (Am Soc Hematol Educ Program). 2003;  314-330
  CrossrefPubMedSearch in Google Scholar
• 2 Bueno R, Swanson S J, Jaklitsch M T. Combined antegrade and retrograde dilatation: a new endoscopic technique in the management of complex esophageal obstruction.  Gastrointest Endosc. 2001;  54 368-372
  CrossrefPubMedSearch in Google Scholar
• 3 Boeck A, Buckley R H, Schiff R I. Gastroesophageal reflux and severe combined immunodeficiency.  Allergy Clin Immunol. 1997;  99 420-424
  PubMedSearch in Google Scholar
• 4 Nayyar A K, Royston C, Bardhan K D. Oesophageal acid-peptic strictures in the histamine H2 receptor antagonist and proton pump inhibitor era.  Dig Liver Dis. 2003;  35 143-150
  CrossrefPubMedSearch in Google Scholar
• 5 Shepherd R W, Wren J, Evans S. et al . Gastroesophageal reflux in children. Clinical profile, course and outcome with active therapy in 126 cases.  Clin Pediatr (Phila). 1987;  26 55-60
  CrossrefPubMedSearch in Google Scholar
• 6 Fung K P, Seagram G, Pasieka J. et al . Investigation and outcome of 121 infants and children requiring Nissen fundoplication for the management of gastroesophageal reflux.  Clin Invest Med. 1990;  13 237-246
  PubMedSearch in Google Scholar
• 7 Spechler S J, Lee E, Ahnen D. et al . Long-term outcome of medical and surgical therapies for gastroesophageal reflux disease: follow-up of a randomized controlled trial.  JAMA. 2001;  285 2331-2338
  CrossrefPubMedSearch in Google Scholar
• 8 Wilcox C M. Esophageal strictures complicating ulcerative esophagitis in patients with AIDS.  Am J Gastroenterol. 1999;  94 339-343
  CrossrefPubMedSearch in Google Scholar
• 9 Gandhi R P, Cooper A, Barlow B A. Successful management of esophageal strictures without resection or replacement.  J Pediatr Surg. 1989;  24 745-750
  CrossrefPubMedSearch in Google Scholar

H. Q. Huynh, MBBS

Department of Pediatrics · University of Alberta

2C3 Walter C Mackenzie Health Sciences Centre · Edmonton · Alberta T6G 2R7 · Canada

Fax: 1-780-407-3507

Email: hien.huynh@ualberta.ca