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      Acquired Chiari I Malformation Secondary to Spontaneous Intracranial Hypotension Syndrome and Persistent Hypoglycemia: A Case Report

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          Abstract

          Spontaneous intracranial hypotension (SIH) is a rare and potentially serious condition in childhood. Cerebrospinal fluid (CSF) volume depletion is thought to be the main causative feature for intracranial hypotension and results from a spontaneous CSF leak, often at the spine level. SIH is increasingly diagnosed in clinical practice, although it manifests a varied symptomatology. The downward displacement of the brain, sometimes mimicking a Chiari I malformation, has rarely been reported. We present a case of a SIH with Chiari I malformation accompanied by an unusual clinical presentation of persistent hypoglycemia.

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          Chiari I malformations: clinical and radiologic reappraisal.

          Eric A. Elster, Hong Chen (1992)
          Clinical findings and magnetic resonance (MR) images in 68 patients with Chiari I malformations were retrospectively analyzed to identify those radiologic features that correlated best with clinical symptoms. A statistically significant (P = .03) female predominance of the malformation was observed, with a female: male ratio of approximately 3:2. Associated skeletal anomalies were seen in 24% of patients. Syringomyelia was detected in 40% of patients, most commonly between the C-4 and C-6 levels. Of the 25 patients who presented with spinal symptoms, 23 (92%) proved to have a syrinx at MR imaging. When the syrinx extended into the medulla (n = 3), however, brain stem symptoms predominated. Patients with objective brain stem or cerebellar signs had the largest mean tonsillar herniations. Patients with tonsillar herniations greater than 12 mm were invariably symptomatic, but approximately 30% of patients with tonsils herniating 5-10 mm below the foramen magnum were asymptomatic at MR imaging. "Incidental" Chiari I malformations are thus much more common than previously recognized, and careful clinical assessment remains the cornerstone for proper diagnosis and management.
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            Diagnostic criteria for headache due to spontaneous intracranial hypotension: a perspective.

            Bahram Mokri, Marie-Germaine Bousser, Simone Schievink … (2011)
            The clinical and radiographic manifestations of spontaneous intracranial hypotension are highly variable and many patients do not satisfy the 2004 International Classification of Headache Disorders criteria. We developed new diagnostic criteria for spontaneous intracranial hypotension based on cases we have seen reflecting the variable manifestations of the disorder. These criteria provide a basis for change when the classification criteria are next revised. The diagnostic criteria consist of A, orthostatic headache; B, the presence of at least one of the following: low opening pressure (≤ 60 mm H(2) O), sustained improvement of symptoms after epidural blood patching, demonstration of an active spinal cerebrospinal fluid leak, cranial magnetic resonance imaging changes of intracranial hypotension (eg, brain sagging or pachymeningeal enhancement); C, no recent history of dural puncture; and D, not attributable to another disorder. © 2011 American Headache Society.
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              Syndrome of cerebral spinal fluid hypovolemia: clinical and imaging features and outcome.

              M Pursch, SONG-JA KIM, Roger S. Chung (2000)
              To investigate clinical, MRI, and radioisotope findings and therapeutic outcome of the syndrome of CSF hypovolemia. Retrospective review was performed of 30 consecutive patients (10 men, 20 women; mean age 37 years) with the syndrome of CSF hypovolemia. All patients had an orthostatic headache, which was alleviated to a variable extent on recumbency. Additional clinical symptoms included nausea, dizziness, neck stiffness, blurring of vision, tinnitus, plugged ear, hearing difficulties and radicular pain of the arm. Eighty-two percent of the patients had CSF opening pressure less than 60 mm H2O, 59% had CSF pleocytosis, and 95% had increased CSF protein. Brain MRI showed diffuse pachymeningeal gadolinium enhancement on T1-weighted image in 83%, which was seen as hyperintense signals on T2-weighted imaging. Other features included subdural hematoma/hygroma in 17% and descent of the brain in 48% of the patients. Radioisotope cisternographic results identified CSF leakage sites in 52%, most often at the lumbar region. Also observed were limited ascent of the tracer to the cerebral convexity (91%), early appearance of radioisotope in the bladder (65%), and early soft tissue uptake of radioisotope (43%). Epidural blood patches were performed in 23 patients, which produced complete resolution of headaches in 70%. Two patients underwent drainage of subdural hematoma. None died or were disabled during hospitalization. Patients with CSF hypovolemia frequently have distinct MRI and radioisotope cisternographic abnormalities and often respond favorably to an epidural blood patch.
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                Author and article information

                Journal
                Journal ID (nlm-ta): J Clin Res Pediatr Endocrinol
                Journal ID (iso-abbrev): J Clin Res Pediatr Endocrinol
                Journal ID (publisher-id): JCRPE
                Title: Journal of Clinical Research in Pediatric Endocrinology
                Publisher: Galenos Publishing
                ISSN (Print): 1308-5727
                ISSN (Electronic): 1308-5735
                Publication date (Print): December 2018
                Publication date (Electronic): 29 November 2018
                Volume: 10
                Issue: 4
                Pages: 391-394
                Affiliations
                [1 ]University of Health Sciences, Kanuni Sultan Süleyman Training and Research Hospital, Clinic of Pediatric Endocrinology and Metabolism, İstanbul, Turkey
                [2 ]University of Health Sciences, Okmeydanı Training and Research Hospital, Clinic of Pediatrics, İstanbul, Turkey
                [3 ]University of Health Sciences, Kanuni Sultan Süleyman Training and Research Hospital, Clinic of Pediatrics, İstanbul, Turkey
                [4 ]Bezmialem Vakıf University Faculty of Medicine, Department of Pediatric Neurology, İstanbul, Turkey
                [5 ]University of Health Sciences, Kanuni Sultan Süleyman Training and Research Hospital, Clinic of Pediatric Surgery, İstanbul, Turkey
                [6 ]İstanbul University Cerrahpaşa Faculty of Medicine, Department of Radiology, İstanbul, Turkey
                Author notes
                * Address for Correspondence: University of Health Sciences, Kanuni Sultan Süleyman Training and Research Hospital, Clinic of Pediatric Endocrinology and Metabolism, İstanbul, Turkey Phone: +90 532 509 29 88 E-mail: hasanonal@ 123456hotmail.com
                Author information
                http://orcid.org/0000-0001-9676-7086
                http://orcid.org/0000-0002-2404-170X
                http://orcid.org/0000-0002-9264-8482
                http://orcid.org/0000-0003-4930-8447
                http://orcid.org/0000-0002-8965-6668
                http://orcid.org/0000-0001-6987-5946
                http://orcid.org/0000-0001-9111-6815
                Article
                Publisher ID: 16767
                DOI: 10.4274/jcrpe.0042
                PMC ID: 6280325
                PubMed ID: 29513222
                SO-VID: cf54e22f-7efe-4125-819a-30af9f686460
                Copyright © © Copyright 2018, Journal of Clinical Research in Pediatric Endocrinology, Published by Galenos Publishing.
                License:

                This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                Date received : 31 January 2018
                Date accepted : 25 February 2018
                Categories
                Subject: Case Report

                ScienceOpen disciplines: Pediatrics
                Keywords: intracranial hypotension,hypoglycemia,vagotomy
                Data availability:
                ScienceOpen disciplines: Pediatrics
                Keywords: intracranial hypotension, hypoglycemia, vagotomy

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