Endoscopic treatment for dysphagia caused by mid-esophageal diverticulum and diffuse esophageal spasm: A case report

Abstract

BACKGROUND

A mid-esophageal diverticulum is an outpouching located in the midsection of the esophagus, posterior to the bifurcation of the right and left bronchi. The condition is characterized by an outward protrusion of the inner esophageal wall. Mid-esophageal diverticula are relatively rare and may coexist with diffuse esophageal spasm. However, the potential esophageal motility disorders associated with these lesions are frequently overlooked in clinical practice. The use of endoscopic interventions may offer a novel approach for the alleviation of dysphagia associated with this condition.

CASE SUMMARY

We present a case of a 74-year-old woman with dysphagia, diagnosed with mid-esophageal diverticulum and diffuse esophageal spasm. Due to her physical condition, submucosal tunneling endoscopic septum division (STESD) was initially performed for the diverticulum. One month later, the esophageal spasm was treated using per-oral endoscopic myotomy (POEM). This combined treatment significantly improved her dysphagia, and she was discharged.

CONCLUSION

STESD and POEM are effective and safe for the treatment of dysphagia caused by mid-esophageal diverticula with diffuse esophageal spasm.

Key Words: Mid-esophageal diverticulum; Diffuse esophageal spasm; Submucosal tunneling endoscopic septum division; Per-oral endoscopic myotomy; Case report

Core Tip: A rare case of mid-esophageal diverticulum with diffuse esophageal spasm is described. The case highlights the importance of considering diffuse esophageal spasm when treating mid-esophageal diverticulum. Diagnostic tools such as upper gastrointestinal pan-glucosamine contrast, esophageal manometry, chest computed tomography, and gastroscopy can be helpful. Submucosal tunneling endoscopic septum division + per-oral endoscopic myotomy is an effective treatment approach for this condition.

INTRODUCTION

Dysphagia is classified into oropharyngeal and esophageal types based on where it occurs. Esophageal dysphagia can be caused by mechanical issues, such as tumors or diverticula, or kinetic problems, such as achalasia. Esophageal diverticulum, a rare benign condition, involves the formation of a pouch in the esophageal wall[1]. It is categorized by location into pharyngo-esophageal (Zenker's), mid-esophageal (paratracheal), and supra-diaphragmatic diverticula. Mid-esophageal diverticula are relatively uncommon, comprising approximately 10% to 20% of all esophageal diverticula. They are more prevalent in developing countries[2], where they predominantly affect middle-aged and elderly individuals These diverticula are frequently associated with inflammatory processes and adhesions. Asymptomatic esophageal diverticula typically do not require treatment, while mild symptoms can be managed with medication. Larger, symptomatic diverticula may need surgery. Surgical options include open and minimally invasive procedures such as submucosal tunneling endoscopic septum division (STESD), which is associated with lower recurrence and complication rates. Diffuse esophageal spasm, characterized by uncoordinated contractions, may be associated with impaired neuroinhibitory mechanisms, increased muscle thickness, and issues linked to relaxation of the gastroesophageal junction. It can be treated with medication, endoscopy, and surgery, with peroral endoscopic myotomy (POEM) being a well-established option for esophageal dyskinesia.

Supradiaphragmatic diverticula are thought to result from congenital developmental abnormalities or esophageal motility disorders, and frequently co-occur with diffuse esophageal spasm[3]. Mid-esophageal diverticula are commonly associated with inflammation and adhesions. However, Kaye[4] observed, when using a perfused catheter manometric system, that diffuse esophageal spasm can also manifest within the mid-esophageal diverticulum. The present report describes a case of dysphagia caused by esophageal diverticulum and spasm, which was successfully treated with STESD and POEM, demonstrating the safety and effectiveness of this combined endoscopic approach.

CASE PRESENTATION

Chief complaints

A 74-year-old woman visited our hospital on July 8, 2023, with a one-year history of dysphagia, nausea, and vomiting.

History of present illness

Approximately one year before, she had begun experiencing dysphagia of unknown etiology, which was accompanied by nausea and vomiting.

History of past illness

The patient reported a history of good health.

Personal and family history

The patient denied any familial history of similar illnesses.

Physical examination

On physical examination, the vital signs were as follows: Body temperature, 36 °C; blood pressure, 18.8/12.3 kPa; heart rate, 68 beats per minute; respiratory rate, 19 breaths per minute. No abdominal tenderness or palpable mass was observed, nor was there palpable enlargement of superficial lymph nodes throughout the body.

Laboratory examinations

No abnormalities were found in routine blood and biochemical tests. The levels of serum tumor markers were normal.

Imaging examinations

A CT scan revealed a right-sided cystic bulge in the middle esophagus, identified as an esophageal diverticulum, accompanied by localized thickening of the lower esophagus (Figure 1). Upper gastrointestinal contrast imaging showed the presence of a 3.0 cm × 3.0 cm cystic shadow in the mid esophagus connected to the esophageal lumen, associated with barium movement (Figure 2A). Gastroscopy confirmed a large esophageal diverticulum and slight tension in the lower and middle esophagus (Figure 3), leading to a diagnosis of mid-esophageal diverticulum with diffuse esophageal spasm. After discussing the condition with the patient and her family, ultrasonographic gastroscopy was used to assess the thickness of the diverticulum wall, and STESD was subsequently performed to treat the diverticulum.

Figure 1 Computed tomography image of the whole abdomen. The cystic bulge on the right side of the middle esophagus was considered an esophageal diverticulum, and local thickening of the lower esophagus was observed.

Figure 2 Upper gastrointestinal pan-glucosamine contrast. A: A cystic shadow is visible on the right side of the middle esophagus, measuring approximately 3.0 cm × 3.0 cm. This was connected to the lumen of the esophagus, and barium could be seen entering and exiting the esophagus; B: A saccular pouching exophthalmos is visible on the right side of the esophagus, with rigidity of the esophageal wall and narrowing of the lumen in the lower part of the esophagus; C: A cystic shadow measuring approximately 2.1 cm × 2.6 cm was seen in the right side of the middle esophagus. String-like changes in the body of the esophagus were visible, accompanied by narrowing of the lumen in the lower and middle esophagus.

Figure 3 Gastroscopy. A: A large diverticulum was seen in the esophagus; B: Slight tension in the lower and middle esophagus.

After surgery, the patient received gastric protection, pain relief, anti-infection treatment, cardiac monitoring, oxygen, and rehydration. Three days later, a contrast test revealed a saccular pouch on the right side of the esophagus, with a rigid wall (Figure 2B) and narrowing of the lumen in the lower esophagus. The patient's swallowing difficulties improved, allowing a semi-liquid diet.

On August 16, 2023, the patient returned to our hospital with dysphagia after healing of the esophageal mucosa. An upper gastrointestinal angiogram revealed a 2.1 cm × 2.6 cm cystic shadow on the right side of the middle esophagus, with string-like changes and narrowing of the lumen of the lower and middle esophagus (Figure 2C). Esophageal manometry indicated normal peristalsis (Figure 4). We then performed POEM for diffuse esophageal spasm.

Figure 4 Esophageal manometry. A: Static pressure: At rest, the upper esophageal sphincter pressure was low (8 mmHg in this patient, normal value 33-180 mmHg) and the low esophageal sphincter pressure was normal (27 mmHg in this patient, normal value 10-45 mmHg); B: 5 mL wet swallow: While normal 4s integrated relaxation pressure and normal distal contractile integral suggest normal peristalsis in the body of the esophagus, an interruption of the contraction wave on the 20 mmHg isobaric line with a defect length greater than 5 cm (6.5 cm) suggests segmental contraction; this swallow was ineffective; C: 5 mL wet pharynx: Normal 4s integrated relaxation pressure, normal distal contractile integral, distal latency, and maximal interruptions are in the normal range, suggesting normal esophageal motility; D: Comprehensive analysis: The patient performed 10 5-mL wet swallows, with 6 (60%) normal contractions (450 mmHg.s.cm < distal contractile integral < 8000 mmHg.s.cm) and 6 (60%) ineffective swallows (including 2 peristaltic failures, 2 weak contractions, and 2 fragmentary contractions). This suggests an indeterminate ineffective esophageal motility. Performed 3 multiple rapid swallows, with the distal contractile integral after performing multiple rapid swallows being less than the mean distal contractile integral of a single swallow, suggesting poor esophageal contractile reserve function.

FINAL DIAGNOSIS

Combined with the patient’s medical history, the final diagnosis was a mid-esophageal diverticulum with diffuse esophageal spasm.

TREATMENT

Postoperatively, the patient recovered well. The STESD procedure (Figure 5): The patient, under tracheal intubation and intravenous anesthesia, was positioned on her side. A large esophageal diverticulum, 30 cm from the incisors, and measuring 3 cm × 5 cm, was identified. The diverticulum had a small opening and smooth mucosa, and while there was some resistance to entry, the fallout sensation was minimal. A submucosal injection of saline and indigo carmine was administered 3 cm above the diverticulum to facilitate tunnel creation. The submucosa was carefully peeled back, and the muscle at the crest of the diverticulum was incised to its base. Upon re-entry, the diverticular opening appeared flaccid and was closed using 13 titanium clips. A 55 cm gastrointestinal decompression tube was then inserted. The POEM procedure (Figure 6): The patient was positioned on her left side and underwent tracheal intubation with intravenous anesthesia. A large esophageal diverticulum was observed 30 cm from the incisors. The angle of the diverticulum was reduced, accompanied by a large opening. A narrowing ring was noted 30-36 cm from the incisors that caused resistance to scope entry. Indigo carmine saline was injected submucosally 30 cm from the incisors (opposite to the diverticulum), followed by mucosal incision to form a tunnel. The circumferential and longitudinal muscles were incised from 30 cm to 2 cm above the cardia, and the wound was closed with 5 harmonic clips. Upon re-entry, the lumen of the esophagus was enlarged, enabling smooth passage of the endoscope and preventing resistance to stenosis. No change in the cardia was observed when the mirror was flipped, and the dentate line was clearly visible.

Figure 5 The submucosal tunneling endoscopic septum division surgical procedure. A and B: Ultrasonic gastroscopy of the layers of the diverticulum; C: Giant esophageal diverticulum; D: Construction of the tunnel; E: Incision of the muscle; F: Closure of the wound with titanium clips.

Figure 6 The per-oral endoscopic myotomy surgical procedure. A: Ultrasonic gastroscopy of the layers of the esophagus; B: Construction of a tunnel; C: Incision of the muscle; D: Closure of the wound with titanium clips.

OUTCOME AND FOLLOW-UP

By six months, there was no return of the patient's dysphagia symptoms, as confirmed by phone follow-ups, and her quality of life was significantly improved.

DISCUSSION

Dysphagia is classified into oropharyngeal and esophageal types. This report describes a case of esophageal dysphagia, caused by mechanical and kinetic issues. Esophageal diverticulum is a rare benign condition. Mid-segment diverticula, often linked with peripheral inflammation and adhesions, tend to be more common in the Chinese population[2]. Supradiaphragmatic diverticula are believed to result from abnormal esophageal motility[5] with 80%-100% of cases involving motor dysfunction, such as pancreatic achalasia and diffuse esophageal spasm. Clinical trials have indicated that diffuse esophageal spasm can also manifest in conjunction with mid-esophageal diverticula[4]. Nevertheless, the co-occurrence of mid-esophageal diverticula and diffuse esophageal spasm is frequently underrecognized. For instance, a case report published in the journal of Endoscopy[6] described the effectiveness of ESD utilizing metal clips for treating mid-esophageal diverticula. However, esophageal manometry was not performed to evaluate esophageal motility function, potentially not identifying esophageal motility disorders. In the present case, we identified a diverticulum in the mid-esophageal region, performed esophageal manometry, followed by subsequent POME surgery, which successfully alleviated the patient's dysphagia symptoms.

Asymptomatic esophageal diverticula require no treatment, while severe cases are typically treated with invasive procedures such as diverticulotomy, which carry risks of trauma, recurrence, and complications[7]. STESD, developed by Li et al[8], offers a minimally invasive alternative. The procedure involves submucosal tunneling to preserve mucosal integrity and minimize complications such as perforation and mediastinitis. In contrast to conventional methods, STESD enables a more thorough incision of the interstitial ridge, reducing the likelihood of recurrence[9]. STESD has been shown to be both safe and effective for treating diverticula[10] but its benefits for esophageal dyskinesia remain under-researched.

POEM is a well-established treatment for esophageal dyskinesia and is now also used for treating mid-esophageal and supradiaphragmatic diverticula. While the extension of myotomy to the lower esophageal sphincter in patients with combined conditions alleviates symptoms[11], there are limited data on its long-term effectiveness and outcomes long-term outcomes.

The present patient was hospitalized with dysphagia and diagnosed with mid-esophageal diverticulum and diffuse esophageal spasm. It is essential to distinguish this condition from achalasia, esophageal cancer, and non-esophageal diseases. The primary diagnostic modalities include esophageal manometry, gastroscopy, and imaging examinations. Traditional STESD was ineffective, leading to the use of the POEM procedure, which was successful in alleviating the patient's symptoms, thereby offering a treatment reference for similar conditions.

CONCLUSION

Based on our experience and reports in the literature, diffuse esophageal spasm should be considered when treating mid-esophageal diverticula. Imaging and gastroscopy can help identify esophageal diverticula as well as other esophageal conditions. The combined use of STESD and POEM significantly improves dysphagia caused by mid-esophageal diverticulum and diffuse esophageal spasm, demonstrating both its safety and efficacy.