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Year : 2012  |  Volume : 60  |  Issue : 1  |  Page : 108--109

Vertebral artery dissection and intractable hiccups: An uncommon presentation

Sujit Meher, Pramod Churasia, Monica Tandon, Daljit Singh 
 Department of Neurosurgery, GB Pant Hospital, New Delhi, India

Correspondence Address:
Daljit Singh
Department of Neurosurgery, GB Pant Hospital, New Delhi
India




How to cite this article:
Meher S, Churasia P, Tandon M, Singh D. Vertebral artery dissection and intractable hiccups: An uncommon presentation.Neurol India 2012;60:108-109


How to cite this URL:
Meher S, Churasia P, Tandon M, Singh D. Vertebral artery dissection and intractable hiccups: An uncommon presentation. Neurol India [serial online] 2012 [cited 2021 Jun 16 ];60:108-109
Available from: https://www.neurologyindia.com/text.asp?2012/60/1/108/93604


Full Text

Sir,

Hiccup (singultus - act of catching one's breath while sobbing) is a benign, frequent and usually a transient phenomenon associated with gastrointestinal disorders. Intractable hiccup, with bouts lasting for more than 48 h, is uncommon. [1] Most neurological lesions for hiccup are located in the vicinity of the brainstem. [2] We describe a patient with vertebral artery dissection and pseudoaneurysm with acute-onset intractable hiccup.

A 26-year-old male presented with acute-onset pain in the right neck followed by persistent hiccups of three years' duration. The pain was severe, unbearable, tearing in character. The hiccups were initially two to four per day and rapidly progressed to 10-12 per min lasting throughout the day. On examination he was fully conscious with right lower cranial nerve paresis without motor or sensory deficit. Magnetic resonance imaging (MRI) revealed a large mass in the right cerebello-pontine angle lifting and tenting the brainstem [Figure 1]. The lesion had layering effect suggesting a thrombus in a vessel. Angiography showed a large pseudoaneurysm measuring 35 × 20 mm [Figure 2]. The vertebral artery was obliterated with coils [Figure 3] resulting in complete non-visualization. There was no retrograde flow from the contralateral vertebral artery [Figure 4]. Following the procedure he had complete relief from hiccups.{Figure 1}{Figure 2}{Figure 3}{Figure 4}

Persistent intractable hiccups can be unpleasant and debilitating and may indicate organic disease and should be further investigated. Intractable hiccups affect male subjects (80-90%) more frequently than females. [3] Neurological causes of intractable hiccup include medullary lesions, multiple sclerosis, periaqueductal lesions, neuromyelitis optica, Arnold-Chiari malformation, and cavernous angioma of the medulla oblongata. Rare etiologies include craniovertebral injury, spinal cord disease, arteriovenous malformation, and hemangioblastoma. [4],[5],[6],[7]

In the vast majority, hiccups are treatable with home remedies, various psychosomatic measures, swallowing, breathing and eating techniques. Various pharmacological agents used in the treatment of hiccups include gastrointestinal stimulants, intravenous steroids, and antipsychotic medications. Sodium valproate and gabapentin have been shown to be effective. [7] Surgical management involves removing the offending lesion. Microvascular decompression for hiccups secondary to medullary and vagal nerve irritation showed encouraging results. [8]

The pathophysiology of hiccups remains unclear; a hiccup is supposed to be related to a stimulation of one or more portions of the so-called hiccup reflex arc. The brainstem hiccup center is near the respiratory center in the medulla. Other supratentorial regions likely to play a role in the genesis of a hiccup are the hypothalamus, temporal areas, and the reticular activating substance. Afferent pathway of the hiccup reflex consists of pharyngeal plexus, the thoracic sympathetic chain T6-T12, the vagus and phrenic nerves. The principal efferent limb and diaphragmatic spasms are mediated by motor fibers of the phrenic and vagus nerves. [9]

Our patient is probably the first case of intractable hiccups due to vertebral artery dissection and pseudoaneurysm formation. Patient had a good response with complete vertebral artery occlusion, supporting the hypothesis that besides direct mechanical compression of the brainstem, continuous pulsatile pressure might have a role to play in triggering the hiccup. A similar explanation has been proposed by Farin et al.,[8] who reported the role of microvascular decompression in intractable hiccups.

References

1Marsot-Dupuch K, Bousson V, Cabane J, Tubiana JM. Intractable Hiccups; The role of cerebral MR in cases without systemic cause. AJNR Am J Neuroradiol 1995;16:2093-100.
2Souadjian JV, Cain JC. Intractable hiccup. Etiologic factors in 220 cases. Postgrad Med 1968;43:72-7.
3Chang YY, Chen WH, Liu JS, Shih PY, Chen SS. Intractable hiccup caused by medulla oblongata lesions. J Formos Med Assoc 1993;92:926-8.
4Misu T, Fujihara K, Nakashima I, Sato S. Intractable hiccup and and nausea with periaqueductal lesions in neuromyelitis optica. Neurology 2005;65:1479-82.
5Musumeci A, Cristofori L, Bricolo A. Persistent hiccup as presenting symptoms in medulla oblongata cavernoma: A case report and review of literature. Clin Neurol Neurosurg 2000;102:13-7.
6Nagayama T, Kaji M, Hirano H, Niiro M, Kuratsu J. Intractable hiccups as a presenting symptom of Chiary 1 malformation J Neurosurg 2004;100:1107-10.
7Alonso-Navarro H, Rubio L, Jimenez FJ. Refractory hiccup: Successful treatment with gabapentin. Clin Neuropharmacol 2007;30:186-7.
8Farin A, Chakrabarti I, Giannotta SL, Vaynamn S, Samudrala S. Microvascular decompression for intractable singulutus; technical case report. Neurosurg 2008;62:E1180-1.
9Newson-Davis J. An experimental study of hiccups. Brain 1970;93:851-72.