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Dysphagia

https://doi.org/10.1007/s00455-019-10044-7

CLINICAL CONUNDRUM

Management of an Unusual Case of Dysphagia


R. S. Shah1 · K. A. Mayuga2 · S. Gabbard3

Received: 22 May 2019 / Accepted: 20 July 2019


© Springer Science+Business Media, LLC, part of Springer Nature 2019

To the Editors: (Fig. 1). While he experienced dysphagia and associated


presyncopal symptoms, Holter monitoring showed sinus
The case report has never been published elsewhere, pre- bradycardia followed by three sinus pauses up to four sec-
sented elsewhere, or is being considered elsewhere. onds and resumption to normal sinus rhythm. What is the
diagnosis and management of this patient?

Clinical Conundrum
Diagnosis and Management
A 58-year-old male presented to the gastroenterology clinic
with syncope while eating. His medical history was signifi- Based on the history and evaluation, a diagnosis of deglu-
cant for hypertension and type 2 diabetes with peripheral tition syncope with combined cardioinhibition and vaso-
neuropathy. Over the past year, he experienced four syn- depression was made. In addition to avoidance of bulky
copal episodes while eating. He reported diaphoresis and food, carbonated beverages, and medications delaying
lightheadedness before losing consciousness for less than a cardiac conduction, the patient was recommended to have
minute. In two of these episodes, the patient experienced a pacemaker implantation to prevent bradyarrhythmia; how-
globus sensation at the sternal notch level while swallow- ever, the patient refused this intervention. Calcium channel
ing solid foods. He reports solid food dysphagia requiring blockade was contraindicated due to bradycardia [1]. Due to
self-induced regurgitation with larger swallows. Denies limited therapeutic options for underlying esophageal and
odynophagia, abdominal pain, nausea, vomiting, heartburn, cardiac disorders, low-dose 10 mg amitriptyline was recom-
voice hoarseness, morning cough, chest pain, palpitations, mended for its vagolytic activity [2]. At his one-year follow-
and shortness of breath. Physical exam was unremarkable. up, the patient denied any episodes of deglutition-induced
Laboratory values were unremarkable. Electroencephalo- presyncope or syncope. In this patient with deglutition syn-
gram, electrocardiogram, and echocardiography were unre- cope, it is proposed that cardioinhibition and vasodepression
markable. The patient’s tilt table test was positive, suggest- are induced by the vagovagal reflex secondary to esophageal
ing vasovagal syncope. For evaluation of his dysphagia, he spasm. Findings of esophageal spasm on manometry and
had an unremarkable esophagogastroduodenoscopy (EGD) bradycardia with sinus pauses on Holter monitoring sup-
and esophagram. Esophageal manometry indicated normal port this diagnosis. Exhausted treatment options lead to a
liquid swallows, but was remarkable for multiple dry swal- trial of amitriptyline due to its anticholinergic effects on
lows followed by premature contraction (distal latency 2.2 s) the vagus nerve to prevent bradycardia and AV dissociation
in the distal esophagus suggesting distal esophageal spasm and its neuromodulator effect for pain in hypercontractile
esophageal dysmotility. Clinically, Oto et al. showed a 91%
prevention rate of vasovagal syncope in 74 patients using
* R. S. Shah amitriptyline [2]. However, there are no reports of using
Shahr4@ccf.org amitriptyline specifically for deglutition syncope.
1
Department of Internal Medicine, Cleveland Clinic,
Cleveland, OH, USA
2
Department of Cardiovascular Medicine, Cleveland Clinic,
Heart and Vascular Institute, Cleveland, OH, USA
3
Department of Digestive Diseases Institute, Cleveland Clinic,
Cleveland, OH, USA

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Management of an Unusual Case of Dysphagia

Fig. 1  Esophageal manometry

Discussion

Deglutition syncope is a rare entity with approximately one- Compliance with Ethical Standards 
hundred cases reported[3]. It is a subtype of neurocardio-
genic syncope triggered by deglutition secondary to organic Conflict of interest  No reported conflicts of interest.
and/or functional esophageal pathology. Although contro- Informed Consent  Patient consent was obtained.
versial, one proposed mechanism is the vagovagal reflex
in which the vagal afferent signal of the esophagus relays
an efferent signal to the AV node [3, 4]. Reduction in car-
diac output or sympathetic withdrawal results in decreased References
cerebral perfusion and brief loss of consciousness [4]. The
1. Kahn A, Koepke LM, Umar SB. Deglutition syncope: a case report
vagovagal mechanism is supported by the prevention of and review of the literature. ACG Case Rep J. 2015;3(1):20–2.
syncope with pre-administration of atropine [4]. Triggers of 2. Barış Kaya E, Abalı G, Aytemir K, Köse S, Kocabaş U,
deglutition syncope include hiatal hernia, acid reflux, acha- Tokgözoğlu L, Kabakçı G, Amasyalı B, Özkutlu H, Nazlı N, Oto
lasia, esophageal spasm, stricture, diverticula, and malig- A. Preliminary observations on the effect of amitriptyline treat-
ment in preventing syncope recurrence in patients with vasovagel
nancy or in patients with normal esophageal function [4]. syncope. Ann Noninvasive Electrocardiol. 2007;12:15–157.
Management of this condition has limited options. The 3. Mitra S, Ludka T, Rezkalla SH, Sharma PP, Luo J. Swallow syn-
initial management includes treating the underlying trig- cope: a case report and review of the literature. Clin Med Res.
ger and avoiding bulky foods, carbonated beverages, and 2011;9(3–4):125–9.
4. Bhogal S, Sethi P, Taha Y, et al. Deglutition syncope: two case
medications that delay cardiac conduction. Calcium chan- reports attributed to vagal hyperactivity. Case Rep Cardiol.
nel blockers have been reported to be effective in cases [1]. 2017;2017:1–3.
In this case, we propose the use of 10 mg of amitriptyline
as a preventative treatment for deglutition syncope. Further Publisher’s Note Springer Nature remains neutral with regard to
jurisdictional claims in published maps and institutional affiliations.
studies may be valuable in the treatment of deglutition syn-
cope; however, prospective, randomized controlled trials are
unlikely to be feasible.

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