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      Dorsal medullary cavernous hemangioma presenting as obstinate hiccups and its surgical treatment: illustrative case

      case-report

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          Abstract

          BACKGROUND

          Hiccups are characterized by involuntary, intermittent, repetitive, myoclonic, and spasmodic contractions of the diaphragm. Hiccups are termed “intractable” when they last for over 1 month.

          OBSERVATIONS

          A rare case of intractable hiccups due to an uncommon location of cavernous hemangioma in the dorsal medulla is illustrated. With respect to the management, surgical excision was performed, and postsurgical complete recovery was witnessed, which has been reported only in six cases worldwide to date.

          LESSONS

          A mechanism of the hiccups reflex arc is discussed in detail with special reference to the need for equal emphasis on evaluating central nervous system causes and peripheral etiologies for pertinent hiccups.

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          Most cited references13

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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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            Hiccups (singultus): review and approach to management.

            K Eilers, P Kolodzik (1991)
            Hiccups are a common, and fortunately usually transient, benign malady. Occasionally, however, hiccups fail to resolve spontaneously, resulting in patient fatigue and incapacitation and the need for the affected individual to seek medical care for resolution of the problem. The approach to the management of these patients consists of the identification and treatment of serious underlying causes of the episode as well as therapeutic interventions to achieve hiccup resolution.
            Article 0 comments Cited 33 times – based on 0 reviews     Review now
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              Diagnosis and management of hiccups in the patient with advanced cancer.

              Mark A Marinella (2015)
              Benign, self-limited hiccups are more of a nuisance, but persistent and intractable hiccups lasting more than 48 hours and 1 month, respectively, are a source of significant morbidity in the patient with advanced malignancy.The hiccup reflex is complex, but stimulation of vagal afferents followed by activation of efferent phrenic and intercostal nerve pathways results in contraction of the diaphragm and intercostal muscles, respectively.The etiology of hiccups in the cancer and palliative care population may include chemotherapy, electrolyte derangements, esophagitis, and neoplastic involvement of the central nervous system (CNS), thorax, and abdominal cavity. Prolonged hiccups can result in depression, fatigue, impaired sleep, dehydration, weight loss, malnutrition, and aspiration syndromes. Evaluation should be symptom-directed, focusing mainly upon the CNS and thoracoabdominal cavities as well as assessment of medications and serum chemistries. Most patients with ongoing hiccups require pharmacotherapy, with chlorpromazine being the only US Food and Drug Administration-approved agent. However, numerous other medications have been reported to be efficacious for treating intractable hiccups. Gabapentin has recently been shown to terminate hiccups effecitvely in cancer patients and may emerge as a therapy of choice in the palliative setting due to favorable tolerability, pain-modulating effects, minimal adverse events, and lack of drug interactions.
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                Author and article information

                Journal
                Journal ID (nlm-ta): J Neurosurg Case Lessons
                Journal ID (iso-abbrev): J Neurosurg Case Lessons
                Journal ID (publisher-id): J Neurosurg Case Lessons
                Title: Journal of Neurosurgery: Case Lessons
                Publisher: American Association of Neurological Surgeons
                ISSN (Electronic): 2694-1902
                Publication date Collection: 6 March 2023
                Publication date (Electronic): 6 March 2023
                Volume: 5
                Issue: 10
                Electronic Location Identifier: CASE22336
                Affiliations
                Departments of [1 ]Neurology and
                [2 ]Neurosurgery, IMS & SUM Hospital, Bhubaneswar, Odisha, India
                Author notes
                Correspondence Atmaranjan Dash: IMS & SUM Hospital, Bhubaneswar, Odisha, India. atmanranjan@ 123456gmail.com .
                [*]

                S.P. and A.A. contributed equally to this work.

                INCLUDE WHEN CITING Published March 6, 2023; DOI: 10.3171/CASE22336.

                Disclosures The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

                Article
                Publisher ID: CASE22336
                DOI: 10.3171/CASE22336
                PMC ID: 10550667
                PubMed ID: 36880512
                SO-VID: 92fa135c-d8e4-4485-9f3c-f093332daabc
                Copyright © © 2023 The authors
                License:

                CC BY-NC-ND 4.0 ( http://creativecommons.org/licenses/by-nc-nd/4.0/).

                History
                Date received : 10 October 2022
                Date accepted : 10 November 2022
                Page count
                Figures: 4, Tables: 1, References: 13, Pages: 4
                Categories
                Compound subject: Anatomy, Anatomy
                Compound subject: Epilepsy, Epilepsy
                Compound subject: Functional-Neurosurgery, Functional Neurosurgery
                Compound subject: Stereotactic-Radiosurgery, Stereotactic Radiosurgery
                Compound subject: Vascular-Disorders, Vascular Disorders
                Compound subject: Technique, Technique
                Compound subject: Surgical-Technique, Surgical Technique
                Subject: Case Lesson

                Keywords: myoclonus,cavernoma,hiccups,excision,cns = central nervous system,mri = magnetic resonance imaging
                Data availability:
                Keywords: myoclonus, cavernoma, hiccups, excision, cns = central nervous system, mri = magnetic resonance imaging

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