Long-term outcomes of per-oral endoscopic myotomy in a patient with distal esophageal spasm followed up for 2.5 years: two sides of the same coin – A Case Report and Literature Review

Abstract

Introduction Distal esophageal spasm (DES), a relatively rare condition, is characterized by simultaneous contractions of the distal esophagus and manifested by dysphagia and chest pain. Several treatment options are recommended, such as pharmacological therapy, endoscopic interventions, and surgical myotomy. Recently, per-oral endoscopic myotomy (POEM) has been adopted as an effective and less-invasive treatment due to its excellent short-term clinical outcomes. Nevertheless, few reports describe its long-term effects.

Case presentation A 65-year-old woman complained of chest pain accompanied with dysphagia and weight loss for 4 months. A series of examinations suggested that she was suffering from DES and then POEM was performed. During the 2.5-year follow-up, we observed an exciting long-term outcome. Interestingly, hematoxylin and eosin staining verified a large number of eosinophils in the muscularis externa, which was absent in the mucosa of the esophagus of the patient.

Conclusions We herein report a case of DES who underwent POEM to eliminate persistent esophageal contractions. Eckardt score, esophageal emptying test, and high-resolution manometry were assumed to monitor the efficacy of POEM. During treatment and 2.5 years after operation, esophageal muscle biopsies and/or mucosal tissues were obtained. This case has been presented to describe that POEM could be a strategy for DES with a long-term curative effect and that eosinophils in the muscle layer of the esophagus might be involved in the pathogenesis of DES. What is more, we reviewed literature to find similar cases reported in the past.

Zusammenfassung

Einführung Diffuser Ösophagusspasmus (distal esophageal spasm DES) ist ein eher seltenes Krankheitsbild, gekennzeichnet von simultanen Kontraktionen der Muskulatur der distalen Speiseröhre, das Schluckbeschwerden und retrosternale Schmerzen verursacht. Empfohlen werden unterschiedliche Behandlungsmethoden wie medikamentöse Therapie, endoskopische Verfahren und operative Myotomie. Aufgrund der ausgezeichneten Kurzzeitergebnisse im klinischen Umfeld wird seit kurzem die perorale endoskopische Myotomie (POEM) als effektive und wenig invasive Behandlungsmethode angewandt. Es gibt jedoch nur wenige Berichte über die Langzeitergebnisse.

Fallbericht Eine 65-jährige Frau gab an, seit 4 Monaten unter Brustschmerzen mit Schluckbeschwerden und Gewichtsverlust zu leiden. Nach mehreren Untersuchungen wurde die Diagnose DES gestellt, woraufhin eine POEM durchgeführt wurde. Innerhalb des 2,5 Jahre langen Follow-up konnten wir ein ausgezeichnetes Langzeitergebnis feststellen. Interessanterweise ergab die Färbung mit Hämatoxylin und Eosin eine große Anzahl an Eosinophilen in der Muskelschicht, jedoch nicht in der Ösophagusschleimhaut der Patientin.

Schlussfolgerungen Wir berichten hier über einen Fall von DES bei einer Patientin, bei welcher eine POEM durchgeführt wurde, um persistierende Ösophaguskontraktionen zu eliminieren. Die Wirksamkeit des Eingriffs wurde mittels Eckardt-Score, Ösophagus Entleerungstest und hochauflösender Manometrie überwacht. Während der Behandlung und 2,5 Jahre nach dem Eingriff wurden Gewebeproben aus der Muskelschicht und/oder der Ösophagusschleimhaut gewonnen. Dieser Fall wurde vorgestellt, um zu zeigen, dass POEM eine Behandlungsmethode für DES mit dauerhaft heilender Wirkung sein kann und dass Eosinophile in der Muskelschicht des Ösophagus an der Pathogenese von DES beteiligt sein können. Außerdem haben wir eine Literaturübersicht durchgeführt, um ähnliche Fälle in der Vergangenheit zu finden.

Key words

distal esophageal spasm - per-oral endoscopic myotomy - eosinophils

Schlüsselwörter

distaler Ösophagusspasmus - perorale endoskopische Myotomie - Eosinophile

^# Authors share co-first authorship.

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