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Clinical Case Report

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Hiccups triggered by bladder filling after bilateral
pontine hemorrhage
A case report

∗

Jinmann Chon, MD, PhD, Seung Don Yoo, MD, PhD, Seung Ah Lee, MD, PhD

Abstract
Introduction: A hiccup is myoclonus of a sudden involuntary contraction of the diaphragm. Hiccups have various causes, and

brain stem stroke is one of the causes of central hiccups. Certain types of hiccups are caused by diseases that can be fatal. Therefore,
it is beneficial for physicians to be familiar with the various cases of unusual hiccups. We report a case of hiccups triggered by urinary
bladder filling in a brain stem stroke patient. To the best of our knowledge, previous reports have not described a similar case.
Patient concerns: We describe the case of a 54-year-old patient who had acute bilateral pontine hemorrhage. The patient had
intermittent hiccups in the early stages of the stroke onset. The hiccups ceased by the administration of medication or stimulation of
the pharyngeal or tracheal wall. Two months after the onset, the Foley catheter was removed to check if the patient could void the
bladder voluntarily. Hiccups occurred whenever the bladder was filled with some amount of urine.
Diagnosis: Pontine hemorrhage, neurogenic bladder, and quadriplegia.
Interventions: When the hiccups occurred, the amount of urine in the bladder was checked using a transabdominal bladder
ultrasonography scanner. After clean intermittent catheterization for bladder emptying, the hiccups subsided.
Outcomes: The hiccups occurred 5 or 6 times a day, as often as the bladder was filling. He was unable to void the urine voluntarily
for 5 days after the removal of the Foley catheter. Percutaneous suprapubic cystostomy was performed finally to remove the
stimulation of bladder filling and the hiccups disappeared.
Conclusion: Bladder filling is suspected to increase the sympathetic tone and cause a hiccup reflex. Bladder filling could be a factor

triggering hiccups in pontine hemorrhage.
Abbreviations: CIC = clean intermittent catheterization, CT = computed tomography, PMC = pontine micturition center.
Keywords: hiccup, neurogenic, stroke, urinary bladder

distension (e.g., overeating, eating too fast, drinking carbonated
drinks, and aerophagia), a sudden change in temperature (e.g.,
very hot or cold food or drinks, and a cold shower), drinking
alcohol, excessive smoking, or psychogenic (sudden excitement
or emotional stress).[2] To date, no report has discussed that
bladder filling causes hiccups.
The pathophysiology of hiccupping is poorly understood, but
it may be mediated through a hiccup reflex arc. A reflex arc is
involved with phrenic nerve, vagus nerve, and sympathetic
pathways and central modulation are likely responsible for
hiccups. Accordingly, any irritant, such as physical/chemical
factors, inflammation, and neoplasia invading the arc leads to
hiccups. The central causes of hiccups include stroke, spaceoccupying lesions, injury, and others. Among the types of stroke,
brain stem stroke is the most common cause of hiccups. We
present the case of a middle-aged man who had hiccups
repeatedly every time his bladder was filling, after bilateral
pontine hemorrhage. We performed a literature review of the
previous cases and aimed to elucidate a possible mechanism for
our case. The patient’s guardian provided informed consent for
participation in the study.

1. Introduction
A hiccup is abrupt erratic diaphragmatic muscular contraction,
and is followed by laryngeal closure immediately.[1]
Hiccup is usually a self-limited disorder; hence, many episodes
of hiccups may subside spontaneously without any clinical
significance. Most hiccups are brief self-limiting episodes of no
clinical significance. The most frequent benign causes are gastric
Editor: N/A.
The authors have no conflicts of interest to disclose.
Department of Physical Medicine and Rehabilitation, Kyung Hee University
College of Medicine, Seoul, Republic of Korea.
∗

Correspondence: Seung Ah Lee, Department of Physical Medicine and
Rehabilitation, Kyung Hee University College of Medicine, 7-13, Kyungheedae-ro
6-gil, Dongdaemun-gu, Seoul 02453, Republic of Korea
(e-mail: lsarang80@gmail.com).

Copyright © 2020 the Author(s). Published by Wolters Kluwer Health, Inc.
This is an open access article distributed under the Creative Commons
Attribution License 4.0 (CCBY), which permits unrestricted use, distribution, and
reproduction in any medium, provided the original work is properly cited.
How to cite this article: Chon J, Yoo SD, Lee SA. Hiccups triggered by bladder
filling after bilateral pontine hemorrhage: a case report. Medicine 2020;99:14
(e19338).

2. Case report

Received: 14 May 2019 / Received in final form: 11 December 2019 / Accepted:
29 January 2020

A 54-year-old man without any premedical history had suddenonset left-sided weakness at home. A few minutes later, right-

http://dx.doi.org/10.1097/MD.0000000000019338

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Chon et al. Medicine (2020) 99:14

Medicine

suprapubic cystostomy was performed. He discharged from
hospital 5 days after suprapubic cystostomy procedure. One and
a half years after a stroke, motor power did not recover and
urinary drainage maintained through the suprapubic cystostomy
without hiccups.

3. Discussion
We report a case of a hiccup induced by bladder filling after
pontine hemorrhage. Approximately 4000 hospital admissions
due to hiccups are reported every year in the United States.[1]
Hiccups are more common in central nervous system and
gastrointestinal diseases. Persistent or intractable hiccup is a
common disorder that can occur due to brain stem lesions.
Keane’s[3] analysis revealed that 56% of his experience with
central hiccups were secondary to lateral medullary infarcts.
However, any ischemic insult to the brainstem or pons[4] will
place an individual at risk of developing intractable hiccups. If
hiccups persist, aspiration, during episodes of regurgitation
secondary to hiccups, and loss of airway control would occur. It
is more fatal to stroke patients. Therefore, prompt and
appropriate management is required.
The production of hiccups is a complex mechanism that involves
multiple neurotransmitters and anatomical structures within the
central and peripheral nervous systems. The reflex arc has afferents
from the phrenic, vagus, and sympathetic nerves (T6–T12) to the
central processing area in the brain stem, with efferents in the
motor fibers of the phrenic nerve to the diaphragm and accessory
nerve to the intercostal musculature.[2,5,6] Brain stem infarction
may cause disintegration of inhibitory connections with a higher
central pathway, which could make hiccups persist.
Considering the bladder neurophysiology, connections between various brain areas and extensive tracts in the spinal cord
including sympathetic, parasympathetic, and somatic systems are
necessary for the neural control of micturition. In the brain, the
pontine micturition center (PMC, Barrington nucleus) is specific
for the micturition center.[7] In an intact central circuit, excitation
of the PMC activates the descending pathways that control
urethral sphincter relaxation and activation of the sacral
parasympathetic outflow. This results in bladder contraction
and increased intravesical pressure and urinary flow.
During the bladder filling phase, the sympathetic afferent fibers
are activated with somatic afferent fibers owing to the mechanical
distension of the bladder wall. In this case, we postulated that the
hiccups were caused by the irritative sympathetic fibers of the
hiccup reflex arc that were full-blown at the end of the filling
phase. If the PMC was intact and the bladder emptying phase was
initiated, the parasympathetic tone would be activated, and the
sympathetic tone would be turned off. As the PMC was injured
due to the stroke, the sympathetic tone subsided, and in turn, the
hiccups subsided after artificial bladder emptying.
If hiccups in pontine stroke patients are persistent, physicians
must check for intra-abdominal stimuli, especially bladder
distension.

Figure 1. Computed tomography (CT) scan shows high attenuation in the
acute pontine hemorrhage with intraventricular hemorrhage at the fourth
ventricle.

sided weakness also developed, and he could not speak. He was
admitted to the emergency department in a stuporous state. The
initial Glasgow coma scale score was 4 (extensor posturing to
pain, no eye opening, and no verbal response). The initial brain
computed tomography scan revealed acute pontine hemorrhage
with intraventricular hemorrhage at the fourth ventricle (Fig. 1).
In the intensive care unit, he remained in a semicoma, and
central high fever and apnea developed, which could occur in
pontine hemorrhage. A Foley catheter, Levin tube, and
endotracheal tube were inserted. After 7 days in the hospital,
he opened his eyes spontaneously but was not aware of his
surroundings. Making eye contact was impossible, and he could
not move his extremities at all. Hiccups occurred once every few
days. In addition, the hiccups subsided after administration of
metoclopramide HCl or stimulation of the pharyngeal and
tracheal walls with a suction catheter.
On the 70th day of hospitalization, the Foley catheter was
removed to check if he could urinate voluntarily. Six hours after
the removal, hiccups began again. Stimulation of the pharyngeal
and tracheal walls was not successful to cease the hiccups. At that
time, urine volume was assessed using a transabdominal
ultrasonography bladder scan. The urine volume was 260 mL.
Two hours later, although the urine volume increased up to 360
mL, he could not void voluntarily. Bladder emptying was
performed through clean intermittent catheterization (CIC).
Within a few minutes after the CIC, the hiccups that lasted for 2
hours spontaneously subsided. Ever since, whenever his bladder
was filled with urine at a volume of as much as 175 to 350 mL,
hiccups occurred. After the CIC, the hiccups were terminated. He
could not void urine voluntarily for 5 days after the removal of
the Foley catheter. Thus, the physician decided to reinsert a Foley
catheter for continuous drainage of urine. Hiccups did not occur
thereafter. In consultation with a urologist, percutaneous
suprapubic cystostomy was the final decision to remove the
stimulation of bladder filling. Three months after a stroke,

Author contributions
Conceptualization: Jinmann Chon, Seung Ah Lee, Seung Don
Yoo.
Data curation: Jinmann Chon, Seung Ah Lee.
Investigation: Seung Ah Lee.
Methodology: Seung Ah Lee.
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[2] Wilcox SK, Garry A, Johnson MJ. Novel use of amantadine: to treat
hiccups. J Pain Symptom Manage 2009;38:460–5.
[3] Keane JR. Hiccups due to central nervous system disease: analysis of 71
inpatients. Can J Neurol Sci 2010;37:870–2.
[4] Kumar A, Dromerick AW. Intractable hiccups during stroke rehabilitation. Arch Phys Med Rehabil 1998;79:697–9.
[5] Nausheen F, Mohsin H, Lakhan SE. Neurotransmitters in hiccups.
Springerplus 2016;5:1357.
[6] Steger M, Schneemann M, Fox M. Systemic review: the pathogenesis and
pharmacological treatment of hiccups. Aliment Pharmacol Ther
2015;42:1037–50.
[7] Holstege G. Micturition and the soul. J Comp Neurol 2005;493:
15–20.

Supervision: Seung Ah Lee.
Writing – original draft: Jinmann Chon, Seung Ah Lee, Seung
Don Yoo.
Writing – review & editing: Jinmann Chon, Seung Ah Lee, Seung
Don Yoo.
Seung Ah Lee orcid: 0000-0002-3426-6259.

References
[1] Cymet TC. Retrospective analysis of hiccups in patients at a community
hospital from 1995–2000. J Natl Med Assoc 2002;94:480–3.

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