Hiccups Attributable to Syringobulbia and/or Syringomyelia Associated with a Chiari I Malformation: Case Report

Seki, Takahiro; Hida, Kazutoshi; Lee, Jangbo; Iwasaki, Yoshinobu

doi:10.1227/01.neu.0000097557.77978.5e

Cited by 18 publications

(11 citation statements)

References 25 publications

(10 reference statements)

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“…2,3,5,6 Other common presentations include weakness, numbness, unsteadiness, and loss of temperature sensation secondary to syringomyelia and syringobulbia. 8 The subsequent posterior fossa decompression leading to complete resolution of the syringobulbia and hiccups provided support for this theory and possible localization for a so-called hiccup center. 2 The theorized pathophysiology involves the mass effect produced by tonsillar ectopia and a derangement of normal CSF dynamics within the posterior fossa.…”

Section: Discussionmentioning

confidence: 81%

Chronic Cough in an Adult Patient With Chiari Type-I Malformation

Chang¹,

Goshgarian

Simoff

et al. 2010

Neurosurgery Quarterly

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Objective and Importance: We report on a patient who had a chronic cough as the sole symptom of a Chiari I malformation that resolved with posterior fossa decompression.Clinical Presentation: The patient was earlier diagnosed with Chiari I malformation and underwent posterior fossa decompression. She subsequently developed a chronic cough that was not explained by an extensive pulmonary work-up.Intervention or Technique: The patient underwent reoperation, and the earlier suboccipital craniectomy was broadened by 2 cm. In addition, a tonsillectomy with dissection of arachnoid adhesions was also carried out, exposing the obex. A wide pericranial graft of the craniocervical junction was also carried out.Conclusions: The patient's cough resolved after reoperation. We speculate that the pathophysiology of her case may stem from irritation or compression of the cough reflex circuitry in the dorsal medulla. This case represents an unusual manifestation of symptoms from a Chiari I malformation.

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Section: Discussionmentioning

confidence: 81%

Chronic Cough in an Adult Patient With Chiari Type-I Malformation

Chang¹,

Goshgarian

Simoff

et al. 2010

Neurosurgery Quarterly

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“…10,13,17,23,27 Giant SB in childhood and fatal hemorrhage have been reported. 31,34,35 Intractable hiccups 4,33 have been observed in SB and SM, and in 1 case the hiccups resolved after posterior fossa surgery. 33 Another consistent observation we report relates to SB location: SB in the medulla was present in all of our 13 cases.…”

Section: Discussionmentioning

confidence: 99%

Syringobulbia in pediatric patients with Chiari malformation type I

Menezes

Greenlee

Dlouhy

2018

Journal of Neurosurgery: Pediatrics

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OBJECTIVESyringobulbia (SB) is a rare entity, with few cases associated with Chiari malformation type I (CM-I) in the pediatric population. The authors reviewed all pediatric cases of CM-I–associated SB managed at their institution in order to better understand the presentation, treatment, and surgical outcomes of this condition.METHODSA prospectively maintained institutional database of craniovertebral junction abnormalities was analyzed to identify all cases of CM-I and SB from the MRI era (i.e., after 1984). The authors recorded presenting symptoms, physical examination findings, radiological findings, surgical treatment strategy, intraoperative findings, and outcomes. SB cases associated with tumors, infections, or type II Chiari malformations were excluded.RESULTSThe authors identified 326 pediatric patients with CM-I who were surgically treated. SB was identified in 13 (4%) of these 326 patients. Headache and neck pain were noted in all 13 cases. Cranial nerve abnormalities were common: vagus and glossopharyngeal nerve dysfunction was the most frequent observation. Other cranial nerves affected included the trigeminal, abducens, and hypoglossal nerves. Several patients exhibited multiple cranial nerve palsies at presentation. Central sleep apnea was present in 6 patients.Syringomyelia (SM) was present in all 13 patients. SB involved the medulla in all cases, and extended rostrally into the pons and midbrain in 2 patients; in 1 of these 2 cases the cavity extended further rostrally to the cerebrum (syringocephaly). SB communicated with the fourth ventricle in 7 of the 13 cases.All 13 patients were treated with posterior fossa decompression with intradural exploration to ensure CSF egress out of the fourth ventricle and through the foramen magnum. The foramen of Magendie was found to be occluded by an arachnoid veil in 9 cases. Follow-up evaluation revealed that SB improved before SM. Cranial nerve palsies regressed in 11 of the 13 patients, and SB improved in all 13.CONCLUSIONSThe incidence of SB in our surgical series of pediatric patients with CM-I was 4%, and all of these patients had accompanying SM. The SB cavity involved the medulla in all cases and was found to communicate with the fourth ventricle in 54% of cases. Posterior fossa decompression with intradural exploration and duraplasty is an effective treatment for these patients.

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“…Besides neoplastic or inflammatory diseases, Chiari I deformation or vascular anomalies such as aneurysm or dolichoectasia have caused hiccups by compression of the involved centers. 10,[13][14][15] To date, however, only one patient with a cavernous angioma triggering intractable hiccup has been reported to our knowledge: Musumeci et al describe a cavernous angioma located near the obex of the medulla oblongata, with dorsal exophytic growth in the fourth ventricle. Intractable hiccups was the main symptom.…”

Section: Discussionmentioning

confidence: 99%