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      Management of an intracranial hypotension patient with diplopia as the primary symptom: A case report

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          Abstract

          BACKGROUND

          Intracranial hypotension (IH) is a disorder involving cerebrospinal fluid (CSF) hypovolemia due to spontaneous or traumatic spinal CSF leakage and is easily being misdiagnosed or missed, especially in these patients without the prototypical manifestation of an orthostatic headache. At present, the management of IH with both cranial nerve VI palsy and bilateral subdural hematomas (SDHs) is still unclear.

          CASE SUMMARY

          A 67-year-old male Chinese patient complained of diplopia on the left side for one and a half mo. Computed tomography revealed bilateral SDHs and a midline shift. However, neurotrophic drugs were not effective, and 3 d after admission, he developed a non-orthostatic headache and neck stiffness. Enhanced magnetic resonance imaging revealed dural enhancement as an additional feature, and IH was suspected. Magnetic resonance myelography was then adopted and showed CSF leakage at multiple sites in the spine, confirming the diagnosis of having IH. The patient fully recovered following multiple targeted epidural blood patch (EBP) procedures.

          CONCLUSION

          IH is a rare disease, and to the best of our knowledge, IH with diplopia as its initial and primary symptom has never been reported. In this study, we also elucidated that it could be safe and effective to treat IH patients with associated cranial nerve VI palsy and bilateral SDHs using repeated EBP therapy.

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          Most cited references22

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          Spontaneous spinal cerebrospinal fluid leaks and intracranial hypotension.

          Spontaneous intracranial hypotension is caused by spontaneous spinal cerebrospinal fluid (CSF) leaks and is known for causing orthostatic headaches. It is an important cause of new headaches in young and middle-aged individuals, but initial misdiagnosis is common. To summarize existing evidence regarding the epidemiology, pathophysiology, diagnosis, and management of spontaneous spinal CSF leaks and intracranial hypotension. MEDLINE (1966-2005) and OLDMEDLINE (1950-1965) were searched using the terms intracranial hypotension, CSF leak, low pressure headache, and CSF hypovolemia. Reference lists of these articles and ongoing investigations in this area were used as well. Spontaneous intracranial hypotension is caused by single or multiple spinal CSF leaks. The incidence has been estimated at 5 per 100,000 per year, with a peak around age 40 years. Women are affected more commonly than men. Mechanical factors combine with an underlying connective tissue disorder to cause the CSF leaks. An orthostatic headache is the prototypical manifestation but other headache patterns occur as well, and associated symptoms are common. Typical magnetic resonance imaging findings include subdural fluid collections, enhancement of the pachymeninges, engorgement of venous structures, pituitary hyperemia, and sagging of the brain (mnemonic: SEEPS). Myelography is the study of choice to identify the spinal CSF leak. Treatments include bed rest, epidural blood patching, percutaneous placement of fibrin sealant, and surgical CSF leak repair, but outcomes have been poorly studied and no management strategies have been studied in properly controlled randomized trials. Spontaneous intracranial hypotension is not rare but it remains underdiagnosed. The spectrum of clinical and radiographic manifestations is varied, with diagnosis largely based on clinical suspicion, cranial magnetic resonance imaging, and myelography. Numerous treatment options are available, but much remains to be learned about this disorder.
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            Surgical management of acute subdural hematomas.

            An acute subdural hematoma (SDH) with a thickness greater than 10 mm or a midline shift greater than 5 mm on computed tomographic (CT) scan should be surgically evacuated, regardless of the patient's Glasgow Coma Scale (GCS) score. All patients with acute SDH in coma (GCS score less than 9) should undergo intracranial pressure (ICP) monitoring. A comatose patient (GCS score less than 9) with an SDH less than 10-mm thick and a midline shift less than 5 mm should undergo surgical evacuation of the lesion if the GCS score decreased between the time of injury and hospital admission by 2 or more points on the GCS and/or the patient presents with asymmetric or fixed and dilated pupils and/or the ICP exceeds 20 mm Hg. In patients with acute SDH and indications for surgery, surgical evacuation should be performed as soon as possible. If surgical evacuation of an acute SDH in a comatose patient (GCS < 9) is indicated, it should be performed using a craniotomy with or without bone flap removal and duraplasty.
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              Spontaneous spinal cerebrospinal fluid leaks.

              Spontaneous intracranial hypotension is an uncommon but not rare cause of new onset daily persistent headaches. A delay in diagnosis is the norm. Women are affected more commonly than men and most are in the fifth or sixth decade of life. The underlying cause is a spontaneous spinal cerebrospinal fluid (CSF) leak. Typically the headache is orthostatic in nature but other headache patterns occur as well. Associated symptoms are common and include neck pain, a change in hearing, diplopia, facial numbness, cognitive abnormalities and even coma. Typical imaging findings consist of subdural fluid collections, pachymeningeal enhancement, pituitary hyperaemia and brain sagging, but magnetic resonance imaging may be normal. Myelography is the study of choice to identify the CSF leak but is not always necessary to make the diagnosis. Treatment consists of bedrest, abdominal binder, epidural blood patching, percutaneous fibrin glue injection or surgical CSF leak repair. Outcomes have been poorly studied.
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                Author and article information

                Contributors
                Journal
                World J Clin Cases
                WJCC
                World Journal of Clinical Cases
                Baishideng Publishing Group Inc
                2307-8960
                6 August 2021
                6 August 2021
                : 9
                : 22
                : 6544-6551
                Affiliations
                Department of Anesthesiology, Sir Run Run Shaw Hospital, School of Medicine, Zhejiang University, Hangzhou 310003, Zhejiang Province, China
                Department of Pain Management, Sir Run Run Shaw Hospital, School of Medicine, Zhejiang University, Hangzhou 310003, Zhejiang Province, China
                Department of Anesthesiology, Sir Run Run Shaw Hospital, School of Medicine, Zhejiang University, Hangzhou 310003, Zhejiang Province, China. chengang120@ 123456zju.edu.cn
                Department of Pain Management, Sir Run Run Shaw Hospital, School of Medicine, Zhejiang University, Hangzhou 310003, Zhejiang Province, China
                Author notes

                Author contributions: Wei TT and Chen G reviewed the literature and contributed to manuscript drafting; Huang H and He FF analyzed, interpreted the imaging findings, and drafted the manuscript; Wei TT, Huang H, He FF, and Chen G were responsible for the revision of the manuscript for important intellectual content; All authors issued final approval for the version to be submitted.

                Corresponding author: Gang Chen, PhD, Chief Doctor, Department of Anesthesiology, Sir Run Run Shaw Hospital, School of Medicine, Zhejiang University, No. 3 Qingchun Road, Hangzhou 310003, Zhejiang Province, China. chengang120@ 123456zju.edu.cn

                Article
                jWJCC.v9.i22.pg6544
                10.12998/wjcc.v9.i22.6544
                8362549
                34435024
                dca720cb-3e47-4137-85ef-354235272244
                ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.

                This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/Licenses/by-nc/4.0/

                History
                : 11 April 2021
                : 18 May 2021
                : 26 May 2021
                Categories
                Case Report

                cranial nerve vi palsy,epidural blood patch,intracranial hypotension,subdural hematoma,case report

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