Ann Thorac Surg 2008;85:328-330. doi:10.1016/j.athoracsur.2007.07.059
© 2008 The Society of Thoracic Surgeons
Case Reports
Use of Baclofen in the Treatment of Esophageal Stent-Related Hiccups
Atila Turkyilmaz, MD*,
Atilla Eroglu, MD
Department of Thoracic Surgery, Medical Faculty, Ataturk University, Erzurum, Turkey
Accepted for publication July 23, 2007.
* Address correspondence to Dr Turkyilmaz, Department of Thoracic Surgery, Faculty of Medicine, Ataturk University, Erzurum, 25240, Turkey (Email: atilat{at}atauni.edu.tr).
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Abstract
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Hiccupping is a characteristic noise caused by a sudden closure of the glottis after repeated, involuntary, spasmodic contraction of the respiratory muscles. Hiccupping caused by gastric distention, spicy foods, and neural dysfunction often resolves itself without any treatment. Some hiccups are associated with certain diseases or occur postsurgically, and life-restricting intractable hiccups should be treated. The cause of hiccups should be expressly stated for treatment. We report a case of inoperable esophagogastric junction tumor with hiccupping after esophageal stent that could only be treated with baclofen.
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Introduction
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Hiccups are an involuntarily powerful spasm of the diaphragm, followed by a sudden inspiration with a closure of the glottis. Hiccups often remit spontaneously within a short period of time, but may also occur without remission for a prolonged period in some cases [1]. Chronic or recurrent hiccups are troublesome for the patient as well as for the clinician and may lead to dehydration, insomnia, depression, and albeit rarely, even death [2].
A 68-year-old man presented with a 4-month history of dysphagia and weight loss. Clinical examination was unremarkable. The results of all laboratory tests on admission were within normal ranges. Multiple biopsies were taken when esophagogastroduodenoscopy revealed an ulcerated, friable, and strictured distal esophagus and thickened mucosal folds throughout the stomach. After histopathologic examination, this was determined as adenocarcinoma and considered inoperable because chest and abdominal computed tomography (CT) scanning showed a thickening of the lower esophagus and the gastroesophageal junction and revealed liver metastasis (Figs 1a, b).
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Fig 1. Computed tomography scans show (a) a well-defined homogeneous, large mass in the lower esophagus and (b) liver metastasis.
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A self-expandable metallic stent was inserted with the aim of achieving a temporary oral diet and nutritional improvement (Fig 2). Because hiccupping was present on the same day, a nasogastric tube was inserted for gastric decompression. Oral treatments with metoclopramide (40 mg daily for 3 days) and chlorpromazine (50 mg daily for 2 days) were sequentially administered, but the hiccups did not improve.
After 72 hours the hiccupping had become intolerable, so treatment with baclofen was initiated. The hiccups ceased 1 day after baclofen treatment commenced. The patient was discharged on baclofen therapy at a dose of 5 mg orally every 6 hours. A gradually tapering baclofen schedule was initiated over the next month, and by 1 month after discharge, baclofen had been gradually stopped altogether. There was no hiccups recurrence over the next few months.
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Comment
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The exact mechanism of hiccups is not completely understood [3]. The pathophysiology is thought to be a "respiratory reflex" of neurogenic origin. The reflex arc consists of afferent stimulation from the phrenic, vagus, and T6-T12 sympathetic fibers; a hiccup central integrator located in the cervical cord between C3 and C5, the brain stem, or midbrain area; and the main efferent limbs being the phrenic nerves [1, 3, 4]. Hiccups can result from direct injury to the reflex arc or any underlying disease, including injury, irritation, or inflammation affecting one of the nerves involved in the reflex arc.
Hiccups may be classified depending on their duration. Persistent hiccups are defined as those lasting longer than 48 hours, whereas intractable hiccups are those lasting more than 1 month [2]. One of the longest cases of continuous hiccups is reported to have lasted for more than 60 years [5].
Multiple causes have been attributed to the etiology of hiccups. The most common are those of gastrointestinal origin, such as gastric distention or gastroesophageal reflux disease [3]. Metabolic derangements and drugs are also frequently implicated [2, 6]. More than 100 organic causes of hiccups have been identified [2, 3]. Surgical procedures involving the cranium, thorax, abdomen, and urinary tract have also been implicated [2]. Furthermore, hiccups may suggest a lesion in the distal portion of the esophagus. Esophageal endoprosthesis and malignancies irritate the afferent limb. To date, one case of intractable hiccups induced by esophageal endoprosthesis placement has been published [7]. Because the hiccups occurred in our patient after insertion of the self-expandable metallic stent, we believe that stent expansion and gastroesophageal reflux provoked excitation of the afferent limb of the hiccup reflex.
Persistent hiccups should be carefully investigated because of their significant impact on quality of life and on patient rehabilitation [1]. They are annoying and uncomfortable to patients and restrict day-to-day activities. They may lead to fatigue, sleep disturbances, dehydration, depression, wound dehiscence in the perioperative period, and albeit rarely, even death [3]. They are often due to an underlying disease process. An organic cause must always be sought and, if possible, treated.
Treatment options for hiccups can include both pharmacologic and nonpharmacologic agents [5]. Nonpharmacologic agents such as breath-holding, drinking cold water, and the application of a nasal or nasogastric catheter are not usually effective in persistent hiccups [2]. Various pharmacologic agents for hiccups have included metoclopramide, chlorpromazine, baclofen, amitriptyline, phenytoin, valproic acid, carbamazepine, haloperidol, and nifedipine [2]. Treatment frequently requires a combination of pharmacologic agents in persistent hiccups.
The efficacy of baclofen in the treatment of chronic hiccups has been demonstrated in a wide number of studies. Recently, a consensus is emerging that presents baclofen as the most effective treatment for chronic hiccups [4]. Launois and colleagues [8] have personal experience with 9 patients started on baclofen therapy—5 were totally relieved of their symptom and 2 had significant improvement—leading them to state that baclofen was their drug of choice.
Baclofen, an analog of 
-aminobutyric, acid, is emerging as a potentially successful treatment for intractable hiccups. It is thought to reduce excitability and depress reflex hiccup activity. The drug is believed to exert its antispastic effects by increasing the threshold for excitation (cell hyperpolarization), resulting in depression of synaptic transmission in the spinal cord. The oral route provides rapid absorption, half-life of 3 to 4 hours, and renal excretion. It is commonly used in patients with multiple sclerosis and spinal cord injury.
The baclofen dosage should start with 5 mg orally every 8 hours, increasing each dose by 5 mg every 3 days until effective, with a maximum dose of 80 mg/d. Its most common side effects are sedation, insomnia, dizziness, weakness, and ataxia, and confusion may also occur. Abrupt discontinuation after regular use can lead to withdrawal symptoms, such as seizure, and gradual discontinuation is recommended. In our case, baclofen appears to be the most efficacious agent in the treatment of chronic hiccups.
Hiccups are usually benign and self-resolving; however, chronic or recurrent hiccups are troublesome for the patient as well as for the clinician. Physicians should bear in mind that intractable hiccups are a potential adverse effect in the stenting of the esophagus and that baclofen is highly effective in the treatment of stent-induced hiccups.
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References
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- Launois S, Bizec JL, Whitelaw WA, Cabane J, Derenne JP. Hiccup in adults: an overview Eur Respir J 1993;6:563-575.[Abstract]
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Abstract
Introduction
