Intractable Hiccup as the Presenting Symptom of Cavernous Hemangioma in the Medulla Oblongata: A Case Report and Literature Review

Abstract

A case of intractable hiccup developed by cavernous hemangioma in the medulla oblongata is reported. There have been only five previously reported cases of medullary cavernoma that triggered intractable hiccup. The patient was a 28-year-old man who was presented with intractable hiccup for 15 days. It developed suddenly, then aggravated progressively and did not respond to any types of medication. On magnetic resonance images, a well-demarcated and non-enhancing mass with hemorrhagic changes was noted in the left medulla oblongata. Intraoperative findings showed that the lesion was fully embedded within the brain stem and pathology confirmed the diagnosis of cavernous hemangioma. The hiccup resolved completely after the operation. Based on the presumption that the medullary cavernoma may trigger intractable hiccup by displacing or compression the hiccup arc of the dorsolateral medulla, surgical excision can eliminate the symptoms, even in the case totally buried in brainstem.

Rapidly Expanding Pediatric Post Radiation Brainstem Cavernoma Presenting with Singultus

“…Brainstem cavernomas can present with a variety of symptoms, rarely including singultus. Spontaneous and familial medullary cavernomas resulting in singultus, while not common, have also been described with one review by Lee et al reporting six patients with a mean age of 34 years (26-40) undergoing surgery [2]. This review found an incidence of hiccups ranging from 3 to 27% with medullary lesions, lesion size ranged from under 1 cm to 2.2 cm and hiccup duration ranged from 15 days to three years, with all patients having a sudden onset.…”

“…The mechanism of medullary cavernoma and hiccups is debated, but many theories exist including (1) disruption of the GABA inhibitory control via the raphe magnus nucleus of the hiccup arc [7], (2) irritation of the medullary reticular activating system, near nucleus ambiguus and the obex [8], and lastly (3) involvement of Mollaret’s myoclonic triangle and the olive [2]. Our surgical approach was planned with regards to this anatomy.…”

“…There is a well-described link between early radiation therapy in children (prior to 10 years) and future cavernoma development with a median time to development of post radiation cavernoma of ten and a half years (<3000 cGy dose), and about five years (>3000 cGy) [1]. Medullary cavernomas have been previously reported to be associated with singultus [2]. Here we describe a pediatric patient with an intramedullary cavernoma presenting with singultus, and discuss why post-radiation brainstem lesions may follow a natural history different from that of incidental brainstem cavernous malformations.…”

Risk Factors for Cancer Chemotherapy-Induced Hiccups (CIH)

“…The medulla oblongata has the vomiting center, and nausea and vomiting result from afferent stimulation. It is known that the reaction center of the hiccup reflex is also in the medulla oblongata [18]. Serotonin and dopamine are neurotransmitters involved in nausea, vomiting, and hiccups as well.…”

Hiccups amelioration following a transcranial direct current stimulation protocol targeting central structures

“…Different central nervous system structures have been hypothesized to underlie the pathological mechanisms of hiccups, such as deep brainstem sites and the medulla oblongata (Tiedt and Wenzel, 2013). Neurological disorders, including medullary infarction and hemorrhage, have reportedly been mechanisms to generate hiccups (Lee et al, 2014). Regarding technical difficulties for accessing brainstem structures, there is growing evidence for the use of novel brain stimulation strategies such as transcranial direct current stimulation (tDCS) for modulating both cortical and subcortical areas.…”