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Journal of Pediatric Gastroenterology & Nutrition:
Case Reports

Infectious Strictures Requiring Esophageal Replacement in Children

Diaz, Mercedes*; Leal, Nuria*; Olivares, Pedro*; Larrauri, Javier†; Tovar, Juan A.*

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Departments of *Pediatric Surgery and †Pathology, Hospital Universitario “La Paz,” Madrid, Spain

Received February 1, 2000; accepted January 24, 2001.

Address correspondence and reprint requests to: Prof. Dr. Juan A. Tovar, Department of Pediatric Surgery, Hospital Universitario “La Paz,” P. de la Castellana, 261, 28046 Madrid,Spain (e-mail: jatovar@hulp.insalud.es).

Esophagitis caused by herpes virus or Candida albicans infection may occur in patients with immune depression or cancer in whom the defense against these agents may be impaired, but it is rare in immunocompetent individuals. These infections seldom lead to complications, and they usually can be managed with antimycotic drugs. Esophageal strictures of these origins, like those of other causes, can be managed successfully by repeated dilatations in most patients, and esophageal replacement rarely is needed. We report two children with intractable esophageal strictures caused respectively by herpes virus and Candida infections who required esophageal replacement after failure of dilatations and medical treatment.

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CASE REPORTS

Case 1

A 14-year-old boy without previous history of disease experienced sudden severe dysphagia, odynophagia, and retrosternal pain that lasted for several months despite various treatments and led to progressive weight loss. Barium meal testing revealed severe esophageal stricture with an irregular mucosal pattern starting at the junction of the upper and middle thirds of the esophagus and extending downward all the way to the stomach. Endoscopy showed severe esophagitis (grade IV) (1) with concentric stricture and erosions. Mucosal biopsy samples taken during the endoscopic procedure after pneumatic dilatation showed multinucleated giant cells with inclusion bodies that stained positively with anti–herpes virus antibodies (2,3) (Fig. 1). Candida overinfection was also present. Extended pH metering performed after dilatation ruled out gastroesophageal reflux. A thorough immunologic workup showed a fully competent immune defense system. Six pneumatic dilatations using 15-and 18-mm balloons (50 psi, 3 minutes × 3) were performed in the ensuing 12 months and provided temporary relief, but stricture recurred in every instance after 2 or 3 weeks. Therefore, esophageal replacement was offered. This operation was performed transhiatally using an isoperistaltic ileocolonic transplant based on an ileocolic pedicle and located intramediastinally. There were no major complications. The patient immediately resumed normal swallowing ability and has remained symptom-free for the ensuing 6 years. Examination of the resected esophagus showed a thin wall and a very narrow lumen, mainly at the middle third, with scar tissue all along it. Histologic study confirmed the findings of the previous biopsies, again showing lymphocyte infiltration and multinucleated giant cells, but without inclusion bodies at that time.

Fig. 1
Fig. 1
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Figure 1
Figure 1
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Case 2

A 3-year-old girl was given a diagnosis of congenital receptive deafness and chronic mucocutaneous candidiasis resulting from repeated C. albicans infections. Her disease course started with progressive dysphagia that worsened in coincidence with mucocutaneous infections that occurred when she was 5 years old. Esophageal stenosis was shown by barium meal testing at that time, and she subsequently underwent a large number of pneumatic dilatations and eventually percutaneous gastrostomy at the hospital to which she was referred. At the time of admission to our institution, she was 8 years old and had many Candida lesions scattered on her skin and mucosae. Complete immunologic assessment showed normal humoral immunity and basically normal cellular immunity with minimal decrease of CD4/CD8 ratio. Skin reactivity to Candida antigen was absent, in vitro lymphocyte responses to Candida mitogen at 1% and 10% were negative, and responses to phytohemagluttinen, concanavalin A, pokeweed mitogens, and OKT3 mitogen were normal. Endoscopy and barium meal testing showed that she had a tight, irregular esophageal stricture with various sinuses or pseudodiverticula caused by persistent infection, previous dilatations, and possibly at least one perforation during one of the endoscopies performed elsewhere. The esophagitis was grade IV, and this led us to perform the last endoscopy through the gastrostomy. There were many Candida lesions, with reddish mucosa and easily bleeding surface. The stricture was deemed intractable (at this time it was impossible to perform endoscopic dilatation); therefore, esophageal replacement was proposed. It was performed after 3 weeks of energetic treatment with amphoterycin B (1 mg · kg −1 · 24 h −1 ) and fluconazol (2 mg · kg −1 · 48 h −1 ). Transhiatal, intramediastinal, transverse left colonic grafting based on a left colic artery pedicle was performed without complications, and the patient has been swallowing normally for the ensuing 3 years, although she always has mucocutaneous lesions.

The resected esophagus had transmural severe fibrosis and scar tissue with grayish mucosa and a lumen of less than 2 mm in diameter at the upper third. Below this level, the entire esophagus was narrowed, although to a lesser degree. Histologic studies showed diffuse lymphocyte transmural infiltration and fibrosis only.

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DISCUSSION

Herpes virus and C. albicans esophagitis are not unusual in patients with immunosuppression and neoplasia, and they are becoming more frequent since the worldwide HIV infection outbreak (4–8). However, in patients without these immune system–depressing conditions, they are rare and seldom lead to severe complications.

Herpes virus infection is common, and many individuals have type I antibodies after experiencing unspecific pseudo-flu events of fever, myalgia and eventually vesicular lesions in the pharynx. Esophageal involvement may cause dysphagia or odynophagia. In otherwise healthy patients, herpetic esophagitis can be considered to be an acute, benign, self-limited illness evolving toward complete resolution of the symptoms after 10 to 12 days (9). Herpes esophagitis has been reported occasionally in previously healthy children with intact humoral and cellular immunity (10–15). Most cases in patients of this age evolve favorably and without sequelae with solely supportive treatment. Ulceration and hemorrhage are rare in this type of infection. Rattner et al. (16) reported in 1985 one case of herpetic esophagitis in an adult with chronic leukemia and extensive bleeding. The lesions were consistent with the diagnosis of reflux esophagitis and were managed accordingly with secretory inhibitors. However, the correct diagnosis of herpetic esophagitis was possible after finding intranuclear inclusions and multinucleated cells in the biopsy samples. Bleeding persisted despite the antireflux regimen, but acyclovir treatment led to dramatic results. Spontaneous perforation of the esophagus as a complication of an intrinsic esophageal infection can occur at relatively low pressures in the presence of esophageal disease (17). Strictures seem to be exceedingly rare after herpes esophagitis (18), and we believe that no previous case of documented herpetic esophagitis leading to stricture necessitating esophageal replacement has been reported to date in a child.

The definitive diagnosis of herpes esophagitis is based on biopsy findings consisting of the presence of epithelial cells with spherical eosinophilic intranuclear bodies surrounded by a pale zone in addition to enlarged, apparently multinucleated cells and some cytoplasmic changes (19). However, the presence of multinucleated cells does not always reflect the viral origin of the disease (20), and immunohistochemical and electromicroscopic investigation may be necessary (21–23).

Esophagitis caused by C. albicans is frequent in adults with compromised immune competence (6,8,23,24) and also in children with the same deficiencies (25–27). In rare instances, these infections can lead to complications, such as esophageal perforation (28) or stricture (18,29–32). The probability of finding esophageal lesions in chronic mucocutaneous candidiasis is higher (33–36). The esophagus is narrowed often in adults with this disease (33) and less often in children (35). Kornblut (37) studied 12 patients with chronic mucocutaneous candidiasis and found that only one patient of five with esophageal involvement, a 5-year-old boy, had stricture that necessitated gastrostomy and retrograde dilatations. In case 2 of this report, the severe esophageal involvement occurred in the context of this illness. The failure of repeated dilatations accompanied by vigorous antifungal treatment led us to perform esophageal replacement, which is an interesting alternative when nonoperative measures are insufficient in this rare disease.

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© 2001 Lippincott Williams & Wilkins, Inc.

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