Gastroentérologie Clinique et Biologique (2010) 34, 329—331

CLINICAL CASE

Intramural esophageal dissection due to pharyngealabscess treated by endoscopic esophagealtransection: A case reportDissection œsophagienne intramurale en raison d’abcès pharyngeal traitépar section transversale œsophagienne endoscopique : un cas clinique

M.A. Benatta ∗, J.-C. Grimaud, M. Kaci, A. Desjeux,M. Baghdadi, A. Loumi

Universitary Military Hospital Oran, Algeria

Available online 26 May 2010

Summary Intramural esophageal dissection is a rare disorder characterized by extensivelaceration between the mucosal and submucosal layers of the esophageal wall, but withoutperforation. The etiology of intramural dissection of the esophagus remains uncertain. Conser-vative management is usually considered adequate. Only one case of circumferential intramuralesophageal dissection has been reported previously. We report here on a case due to an infec-tious cause (paryngeal abscess) that is also an unusual example of circumferential intramuralesophageal dissection, which was then treated by endoscopic transection of the true internalesophageal wall and bougienage dilation.© 2010 Elsevier Masson SAS. All rights reserved.

Résumé La dissection œsophagienne intramurale est une lésion rare caractérisée par unelongue lacération entre les couches muqueuses et sous-muqueuses de la paroi œsophagiennesans perforation. L’étiologie de la dissection intramurale de l’œsophage demeure incertaine. Letraitement Conservateur est habituellement préconisé. Un cas unique de dissection œsophagi-enne intramurale circonférentielle a été rapporté précédemment. Nous rapportons un cas dû à

une cause infectieuse (abcès pharyngé) qui est également le deuxième cas peu commun d’unedissection œsophagienne intramurale circonférentielle, traité par transection endoscopique dela paroi luminale vraie et dilatations aux bougies.© 2010 Elsevier Masson SAS. Tous droits réservés.

∗ Corresponding author.E-mail address: benattaamine@yahoo.fr (M.A. Benatta).

0399-8320/$ – see front matter © 2010 Elsevier Masson SAS. All rights reserved.doi:10.1016/j.gcb.2010.04.009

3 M.A. Benatta et al.

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ntramural esophageal dissection (IED), a rare disorder, is theesult of longitudinal separation of the submucosa and mus-le layers of the esophagus, but without perforation. Theisorder is seen predominantly in middle-aged or elderlyomen. The most common presenting symptoms are sud-en severe retrosternal pain, hematemesis, odynophagiand dysphagia. The diagnosis is made by contrast esopha-ography, esophageal endoscopy or both. The etiology ofED is uncertain, and most reports of IED advocate conser-ative management. The majority of the previously reportedases of IED were partial, with only one case of circum-erential IED reported previously [1]. Although endoscopicreatments of IED have been reported [2—6], their use inhe management of circumferential IED has been reportednly in a single case [1]. In the present report, we describesecond case of circumferential IED—–and the first to beost likely due to a pharyngeal abscess. In this patient,ysphagia did not improve with conservative management,hereby necessitating endoscopic treatment, including tran-ection (transverse resection) of the true lumen (internalsophageal mucosa/submucosa layers) and bougienage dila-ion.

ase report

32-year-old man with no previous medical or surgical his-ory was admitted because of diabetes mellitus and severecute pharyngitis. Rhinolaryngeal examination revealeddema of the larynx with epiglottal ulceration; cervicalomodensitometry confirmed that it was a non-collectingaryngeal abscess. The patient underwent antibiotherapy,lthough the pathogen was not identified. The course of theisease showed regression of general signs, but persistencef dysphagia. Upper gastrointestinal endoscopy showed aongestive muscular lumen (false lumen) due to circum-erential mucous separation that was virtually completehroughout the whole of the esophagus except for the lastcm, where dissection involved only two-thirds of the cir-umference. The true oesophageal wall appeared to beubular and lateral (Fig. 1a), with the presence of abun-ant pus in the stomach (Fig. 1b). Esophagography did nothow the classical ‘double-barreled’ esophagus with truend false lumen, but revealed an excentred thread-like nar-owing along 8 cm of the lower esophagus, with dilatationpstream (Fig. 1c). The patient was kept on conservativereatment and triple antibiotherapy and was fed throughejunostomy.

Nine weeks later, the patient was readmitted becauseotal enteral feeding had failed to bring about any improve-ent of dysphagia. Endoscopy revealed healing, with

e-epithelialization of the esophageal muscular layer. Inddition, it was possible at the same time to advance thecope into the stomach by way of a bidirectional mucosalridge 20 cm from the incisor teeth via the luminal narrowing

center) into the true lumen and via the false lumen laterallyFig. 2a). To resolve the dysphagia, endoscopic treatmentas applied, consisting of endoscopic transection of the true

nternal lumen using endoscopic diathermy snare and hotiopsy (Olympus forceps) (Fig. 2b). The procedure was easily

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ateral on endoscopy (a), and an abundance of pus is seen inhe stomach (b); esophagography reveals narrowing of the low-rmost 8 cm of the esophagus, with dilatation upstream (c).

erformed along most of the esophagus until the last 6 cm,here the procedure became more difficult because the dis-

ection involved only two-thirds of the circumference. Twoessions were necessary with a 48-h interval. However, therocedure was successful, with no complications followinghe procedure.

One week later, the patient no longer complained of anyiscomfort. Endoscopic resection was total, with easy pas-age of the endoscope except for two mucous remnants athe gastro-oesophageal junction that were resected usinghe diathermy snare. Oral intake was then permitted.

Two months later, the patient complained of recur-ence of dysphagia, and follow-up endoscopy examinationevealed an esophageal stricture lying 35 cm from thencisor teeth, which was passable only by a pediatricideoendoscope. Four sessions of endoscopic dilatation withougienage were performed, using Savary—Gilliard bougies,hich allowed easy passage of an adult-sized videoendo-

cope. Six months later, clinical improvement, in particular,f dysphagia and oral feeding with recovery of weight lossas noted.

iscussion

ED was first reported by Marks and Keet [7] in 1968.he mechanism of IED is thought to be a sudden change

n intraesophageal pressure. Predisposing factors includeoagulation defects, endoscopic instrumentation and injec-ion sclerotherapy for esophageal varices. However, in mostases, IED is the result of intramural hematoma [8]. Based onhe reported cases, most patients with IED are women in the

Intramural esophageal dissection treated by endoscopic esophageal transection 331

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Figure 2 Endoscopy shows the true lumen (center) and the falumen using hot biopsy (Olympus forceps) (b).

8th or 9th decade of life. However, younger patients havebeen reported and, occasionally, a male patient as well; thepresent case is the third report involving a young man [3,9].

The most common presenting symptoms include suddenonset chest pain, odynophagia, dysphagia and hemateme-sis. Thus, the age, gender and presenting manifestations inthe present case were highly atypical. Also, the patient wasnot taking any medications that altered blood coagulation;his clotting profile was normal. Furthermore, in the presentcase, it is believed that the IED might have been related toinfection because of the pharyngeal non-collecting abscessand the presence of abundant pus in the stomach. The pha-ryngeal abscess may have fused with and extended into thesubmucosa layer that led to separation of the submucosafrom the muscular layer of the esophagus. Also, the majorityof previously reported patients with IED had partial dissec-tion, while only a single case of circumferential dissectionhas been previously reported [1].

In the present patient, the diagnosis was made byesophageal endoscopy. The IED was circumferential alongthe entire length of the esophagus except for the last 6 cm,where dissection involved only two-thirds of the circumfer-ence. In most reported cases, management was conservativeand usually resulted in a favorable outcome. However, ourpatient was atypical not only in terms of age, gender, pre-sentation and probable infectious etiology, but also becauseof persistent symptoms despite conservative management.Surgical intervention is usually not necessary for IED. Endo-scopic treatments have been reported [2—6], but its use inthe management of circumferential IED has been reported inonly one case [1] that, for the first time, consisted of endo-scopic transection of the true internal esophageal wall. Inthe present case, because dysphagia did not improve aftera 9-week course of conservative treatment, we proceededwith several endoscopic treatments, including transectionof the true internal esophageal wall. As reported in thefirst case of such transection, a stenosis occurred in ourpatient that required four sessions of endoscopic dilation

by bougienage, using Savary—Gilliard bougies.

In conclusion, this was the first case report of an IED thatwas probably due to a laryngeal abscess that became fusedwith the esophageal wall, and the second report of a patientwith circumferential IED who received a series of endoscopic

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men (laterally) (a), endoscopic transection of the true internal

reatments. In our experience, as in the single previouslyeported case, it is difficult to expect spontaneous heal-ng with conservative treatment alone of circumferentialED, which appears to be a more severe variant of IED,iven the lack of response to conservative treatment andhe occurrence of stenosis after endoscopic treatment. Also,ndoscopic treatments should be considered early in theherapeutic strategy.

onflict of interest statement

he authors have not declared any conflict of interest.

eferences

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