Difficult To Treat Recurrent Esophageal Dysphagia Due To Secondary Esophageal Motility Disorders: A 24 Weeks Follow Up Case Report

Received: November 24, 2018 Peer-review started: November 26, 2018 First decision: November 29, 2018 Revised: December 27, 2018 The second round of peer review (by journal editors): January 15, 2019 Accepted: February 7, 2019 Article in press: February 15, 2019 First online: February 16, 2019. Informed consent statement: Informed consent was obtained from the patient. Conflict-of-interest statement: The author declares no conflict-of-interest related to this article. Abstract


Introduction
Dysphagia is an alarming symptom that warrants prompt evaluation to define the exact cause and initiate appropriate therapy. It may be due to a structural or motility abnormality in the passage of solids or liquids. Dysphagia in older adults should not be attributed to aging. Aging alone causes mild esophageal motility abnormalities, rarely symptomatic (1).
Dysphagia to solids and liquids may be related to either an esophageal motility disorder (OMD) or a functional disorder that can be differentiated by investigations like upper endoscopy, barium image, and manometry studies.
Symptoms of dysphagia may be intermittent or present after each meal (2).
Nonspecific esophageal dysmotility (including diffuse esophageal spasm, hypertensive peristalsis "nutcracker or corkscrew esophagus," hyper or hypotensive lower esophageal sphincter, and ineffective esophageal motility) is a type of OMD, which is considered if motility findings exceed two standard deviations from those found in a large group of healthy subjects (3).
OMD may occur as a primary or secondary to other diseases (like systemic sclerosis, Chagas' disease, diabetes mellitus, and chronic gastroesophageal reflux disease), with limited data on the prevalence that is ranging, in some studies, from 4% up to 12% in other reviews, which were carried on individuals referred for esophageal manometry for evaluation of dysphagia or unexplained chest pain (4)(5)(6).
Although the underlying pathology is unknown, the supposed pathophysiology of OMD range from impairment of inhibitory innervation, leading to premature and rapidly propagated or simultaneous contractions and overactivity of There is considerable controversy concerning the clinical implications of these abnormalities and whether they cause or explain the patient's symptoms, contrary to other clear ones like achalasia.

Presenting Concerns
Seventy-year-old male, a previous farmer, married and has four offspring with a medical history of diabetes mellitus for 20 years on insulin therapy, ischemic cardiomyopathy with moderate diastolic dysfunction, and preserved systolic function. For five years, the patient gave a vague history of endoscopic esophageal dilatation (without available documentation) for the same condition, recurrent intermittent dysphagia for solids and liquids with a gradual increase in daily attacks over the last three months from the presentation.
There was no history of psychiatric illness.

Clinical Findings
There is unremarkable general and local examination except for mild oral thrush, bilateral lower limb edema up to mid-leg, and body mass index of 36.7.

Diagnostic Focus and Assessment
Diabetes was controlled in the past three months as glycated hemoglobin (HbA1c) was 7.2%. Complete blood count, liver chemistry tests, and kidney functions were within the normal range. There are normal chest X-ray findings and abdominal ultrasonography except for fatty liver. Echocardiography revealed moderate diastolic dysfunction with preserved systolic function.
Diagnostic upper endoscopy showed multiple circular narrowing in the mid and lower esophagus with the complex proceeding of the endoscope (fig 1). Multiple biopsies were taken, and the histopathological report revealed esophagitis, mild keratosis, and parakeratosis. Barium swallow imaging of the esophagus revealed the corkscrew appearance (Fig 2).

African journal of gastroenterology and hepatology
Sadek AM.2019 25

Therapeutic Focus and Assessment
We started with a twice-daily proton pump inhibitor Omeprazole 20mg before the meal for one hour for two months without changing the patient complaint.
Then we tried a prokinetic agent, Itopride 50mg tid before the dinner by one hour for 15 days but no response. Then we proceed to the first session of dilatation with Savary-Gilliard® Dilator size 9-10mm (27-30Fr), and we add a calcium channel blocker Lercanidipine 10mg daily after the cardiologist consultation. The patient still had no response, so we performed the second dilatation session with a size 11-12mm (33-36Fr) plus changing the Isosorbide Mononitrate to Sildenafil 25mg daily and adding Sodium Alginate 10ml tid before meals.

Follow-up and Outcomes
The patient, after one month, gave us positive feedback of a gradual improvement of dysphagia, especially to fluids with decreasing in daily attacks.

Discussion
One of the strengths of this report was the ability to manage this complex case associated with significant cardiac comorbidity that limits our choices in drugs and doses. Although, a considerable limitation was the absence of esophageal manometry study due to patient refusal and the unique treatment modality that needs validation on a large scale of similar conditions. Our approach is consistent with the American Gastroenterological Association, the American Society for Gastrointestinal Endoscopy, and the World Gastroenterology guidelines (11)(12)(13)(14). The principal concern, in this case, was esophageal cancer due to the age of the patient. However, the intermittent nature of dysphagia and the absence of alarming signs like significant weight loss and anemia make it unlikely (15). Also, complications of gastroesophageal reflux disease, such as erosive esophagitis, peptic stricture, or adenocarcinoma of the esophagus, were precluded by the absence of chronic heartburn (16,17). eosinophilic esophagitis or lymphocytic one should be excluded; we take multiple biopsies from the esophagus (15,16). Lastly, we must not ignore the cardiovascular abnormalities in this age, like severe atherosclerosis or a giant aneurysm of the thoracic aorta can result in impingement on the esophagus (17). However, the endoscopic picture, chest X-ray, and echocardiography did not raise suspicion of this possibility.

Conclusion
We learn from this case that a different patient-to-patient response necessitates other treatment modalities, even switching between each patient's nitric oxide scavengers.

Informed Consent
The patient provided his informed consent for the publication of this case report.

Conflicts of interest
The author declares no conflicts of interest.

Funding support
Zagazig University, Faculty of Human Medicine.