On September 2, 2013, a 70-year-old man presented with persistent vertigo and vomiting for 1 week, and intractable hiccups of about 3 years’ duration. The frequency of hiccups was about 15-20 per minute, and it was not affected by eating and drinking. His hiccupping was persistent during the day, and it had not responded to metoclopramide, domperidone, omeprazole, or even traditional Chinese medicine. However, it stopped spontaneously while he was asleep. The physical and general medical examinations were normal, as was B-mode ultrasound of the abdomen and radiograph of the chest. Computed tomography of the head showed a single hyperintense space-occupying lesion (approximately 6.2 × 6.3 cm2) in the right frontotemporal lobe but mostly located in the frontal lobe, which was referred to as meningioma. Further contrast-enhanced scanning of the lesion showed that its signal intensity was obviously enhanced. A hypointense edema zone was found around the space-occupying lesion. Moreover, the right ventricle and left ventricle anterior horn became narrow, and the midline deviated about 1.5 cm to the left side (Figure). The space-occupying lesion was resected, and histologic examination confirmed the diagnosis of fibroblastic meningioma, World Health Organization grade I. Furthermore, the intractable hiccupping disappeared right after the operation without any antihiccup treatment drugs. As of now, no recurrences of persistent hiccups have been reported.
Discussion
Meningioma is a relatively common neoplasm of the central nervous system that has a predilection to arise from the parasagittal region, cerebral convexity, sphenoidal ridge, olfactory groove, and spinal canal. The frontotemporal lobe is a common region for the origin of meningioma, but meningioma on the right frontotemporal lobe is an unreported cause of intractable hiccups. The etiology of intractable hiccups is varied. Gastroesophageal illness causes most bouts of hiccups,1 while central nervous system illnesses, such as cerebrovascular accidents and brain tumor, that lead to intractable hiccups have recently become an advanced research area. The pathophysiology of intractable hiccups is not fully understood. The accurate central genesis of hiccups, in particular, is still under study. According to the past case reports, hiccups may be mediated through the cerebral center located in the hypothalamus, medullary reticular formation, brain stem near the respiratory centers, medial and dorsal medullary nuclei, and supratentorial areas.2, 3, 4
Irritative or destructive lesions in frontal areas mostly lead to mental disorders, temporal lobe epilepsy, aphemia, and paralysis. As temporal lesions, Wernicke aphasia, anomic aphasia, and mental illness are the common clinical manifestations. Hiccupping caused by temporal space-occupying lesions is rare. The present case of persistent hiccups resolved shortly following resection of a right frontotemporal lobe (mostly in the frontal lobe) meningioma. Therefore, it was speculated that the intractable hiccups arose from the frontotemporal lobe, especially the frontal lobe. It was suggested that the temporal area might be involved in the control mechanisms of hiccups because of invasive tumors.5, 6 Moreover, it was postulated that the genesis of hiccups in the cerebral center was as extensive as in the peripheral regions. On the contrary, hiccups may be generated by abnormal neural discharge stimulated by frontal or temporal space-occupying lesions, which reaches the hiccup-mediating center such as the brainstem.
When hiccups have no causative extracranial pathology and antihiccup drugs do not work, central nervous system illness should be considered as the etiology of persistent hiccups. Appropriate imaging techniques such as computed tomography and magnetic resonance imaging are valuable for diagnosing lesions in the central nervous system, although the incidence of such lesions is relatively low. Undoubtedly, resecting the space-occupying lesions as soon as possible is the best treatment for similar cases.
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-Zhuoqing Hu, PhD, Liao Cui, MD, HPD, Wei Li, MD, Ziji Liang, MD, Minqun Du, MD
This article originally appeared in the January 2017 issue of The American Journal of Medicine.
