Objective:

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Background:

Hiccups occur due to an involuntary, spasmodic contraction of the diaphragm involving a reflex arc with afferent contributions from phrenic and vagus nerves, a poorly defined central mediator, and efferent contributions involving the phrenic nerve. While self-limited hiccups are a benign experience, intractable hiccups, i.e., those lasting longer than one month, can impair quality of life and cause severe medical complications. Few case reports have described relief from intractable hiccups after vagal nerve stimulator (VNS) implantation. We present an alternative case where a patient with epilepsy developed intractable hiccups correlating with VNS activity.

Design/Methods:

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Results:

A 26-year-old male with epilepsy and VNS therapy developed intractable hiccups in the absence of any changes in the VNS settings. Hiccups persisted for 18 months and were associated with constant coughing, dyspepsia, and vomiting. He suffered several complications requiring emergency medical care including iron deficiency anemia requiring blood transfusions and iron therapy, upper and lower GI endoscopies with biopsy demonstrating grade B esophagitis, H. pylori stool antigen positivity requiring treatment, and video capsule endoscopy. He eventually trialed proton-pump inhibitors, H2-receptor antagonists, bismuth, calcium carbonate, and baclofen. Despite this, his hiccups continued and remained intractable. He presented for neurology follow-up care where VNS current was lowered, leading to immediate and complete cessation of hiccups.

Conclusions:

Incidence of hiccups after VNS insertion is rarely reported in literature. Our case demonstrates VNS as the culprit leading to intractable hiccups. We hypothesize vagabond current leaks downstream from the point of insertion of the VNS leads, causing aberrant stimulation of the reflex arc in hiccups production. The etiology of such an aberrant discharge is not well understood. This case report highlights a rare but significant adverse effect of the VNS. This knowledge should guide providers in the evaluation and management of intractable hiccups in patients with an implanted VNS.