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

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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.

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Cavernous malformations of the brainstem: experience with 100 patients.

Lorreta Lawton, Robert Spetzler, Jonathan Baskin … (1998)

In this study the authors review surgical experience with cavernous malformations of the brainstem (CMBs) in an attempt to define more clearly the natural history, indications, and risks of surgical management of these lesions. The authors retrospectively reviewed the cases of 100 patients (38 males and 62 females; mean age 37 years) harboring 103 lesions at treated a single institution between 1984 and 1997. Clinical histories, radiographs, pathology records, and operative reports were evaluated. The brainstem lesions were distributed as follows: pons in 39 patients, medulla in 16, midbrain in 16, pontomesencephalic junction in 15, pontomedullary junction in 10, midbrain-hypothalamus/thalamus region in two patients, and more than two brainstem levels in five. The retrospective annual hemorrhage rate was most conservatively estimated at 5% per lesion per year. Standard skull base approaches were used to resect lesions in 86 of the 100 patients. Intraoperatively, all 86 patients were found to have a venous anomaly in association with the CMB. Follow up was available in 98% (84 of 86) of the surgical patients. Of these, 73 (87%) were the same or better after surgical intervention, eight (10%) were worse, and three (4%) died. Two surgical patients were lost to follow-up review. Incidences of permanent or severe morbidity occurred in 10 (12%) of the surgically treated patients. The average postoperative Glasgow Outcome Scale score for surgically treated patients was 4.5, with a mean follow-up period of 35 months. The natural history of CMBs is worse than that of cavernous malformations in other locations. These CMBs can be resected using skull base approaches, which should be considered in patients with symptomatic hemorrhage who harbor lesions that approach the pial surface. Venous anomalies are always associated with CMBs and must be preserved.

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Advances in the treatment and outcome of brainstem cavernous malformation surgery: a single-center case series of 300 surgically treated patients.

Adib A. Abla, Gregory P. Lekovic, Jay Turner … (2011)

Brainstem cavernous malformations (BSCMs) are relatively uncommon, low-flow vascular lesions. Because of their relative rarity, relatively little data on their natural history and on the efficacy and durability of their treatment.

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Surgical management of brain-stem cavernous malformations: report of 137 cases.

Kristyn A Liu, Cheng-Bo Sun, Li Zhao … (2003)

With the improvement in neuroimaging and microsurgical techniques, brain stem cavernous malformations are no longer considered inoperable. Surgical indications for brainstem cavernoma are evolving, with better understanding of its natural history and decreasing surgical complications. During 1986 through 1998, a series of 137 patients (4 patients each with two brain stem lesions, total number of lesions, 141) with brain stem cavernous malformations were treated microsurgically at Beijing Neurosurgery Institute. The age distribution, lesion location, and clinical presentations were analyzed. The bleeding rate, surgical indications and microsurgical techniques were also discussed. In our series, 92 of 137 cases (67.2%) suffered more than one hemorrhage. Female patients had a higher risk of recurrent hemorrhage than that of male patients. Unlike cavernomas malformations from other locations, repeated hemorrhages from brain stem malformations are much more common and usually lead to new neurologic deficits. Among all 137 surgically treated patients, there was no operative mortality. Ninety-nine patients (72.3%) either improved or remained clinically stable postoperatively. The size of the cavernoma/hematoma does not necessarily correlate with the surgical result. While the acute hematoma can facilitate the surgical dissection, longer clinical history with multiple hemorrhages often makes total surgical resection difficult, partially because of the firmer capsule that may not shrink or collapse after hematoma is released. Pathologically those capsules were associated with more hyaline degeneration, fibrous proliferation and even calcifications. During the follow-up period between 0.5 to 11 years in 129 cases, 115 patients (89.2%) have been working, studying, or doing house work. Three patients (2.3%) suffered recurrent hemorrhages. Surgical indications of brain stem cavernoma include (1) progressive neurologic deficits; (2) overt acute or subacute hemorrhage on MRI either inside or outside cavernous malformations with mass effect; (3) cavernoma/hematoma reaching brainstem surface (<2 mm brain tissue between cavernoma /hematoma and pial surface). Grave clinical presentations like coma, respiratory, or cardiac instability are not surgical contraindications. Emergent surgical evacuation may lead to satisfactory outcome. Repeated hemorrhages will worsen the pre-existing neurologic deficits and possibly make the surgical dissections more difficult. Patients with minimum, stable neurologic deficits and lesion/hematoma that has not reached the brain stem surface should be followed conservatively.

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Author and article information

Journal

Journal ID (nlm-ta): J Korean Neurosurg Soc

Journal ID (iso-abbrev): J Korean Neurosurg Soc

Journal ID (publisher-id): JKNS

Title: Journal of Korean Neurosurgical Society

Publisher: The Korean Neurosurgical Society

ISSN (Print): 2005-3711

ISSN (Electronic): 1598-7876

Publication date (Print): June 2014

Publication date (Electronic): 30 June 2014

Volume: 55

Issue: 6

Pages: 379-382

Affiliations

[1 ]Department of Pathology, Chonnam National University Research Institute of Medical Sciences, Chonnam National University Hwasun Hospital & Medical School, Hwasun, Korea.

[2 ]Department of Neurosurgery, Chonnam National University Research Institute of Medical Sciences, Chonnam National University Hwasun Hospital & Medical School, Hwasun, Korea.

[3 ]Department of Radiology, Chonnam National University Research Institute of Medical Sciences, Chonnam National University Hwasun Hospital & Medical School, Hwasun, Korea.

Author notes

Address for reprints: Kyung-Sub Moon, M.D., Ph.D. Department of Neurosurgery, Chonnam National University Research Institute of Medical Sciences, Chonnam National University Hwasun Hospital and Medical School, 322 Seoyang-ro, Hwasun-eup, Hwasun 519-763, Korea. Tel: +82-61-379-7666, Fax: +82-61-379-7673, moonks@ 123456chonnam.ac.kr

Article

DOI: 10.3340/jkns.2014.55.6.379

PMC ID: 4166338

PubMed ID: 25237438

SO-VID: 7a7f6b4c-aa86-4de3-b733-d700632b4db5

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This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

History

Date received : 14 October 2013

Date revision received : 09 February 2014

Date accepted : 11 June 2014

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

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International Journal of Applied Technology in Medical Sciences

Most cited references 21

Cavernous malformations of the brainstem: experience with 100 patients.

Advances in the treatment and outcome of brainstem cavernous malformation surgery: a single-center case series of 300 surgically treated patients.

Surgical management of brain-stem cavernous malformations: report of 137 cases.

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