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A new hypothesis for the pathophysiology of symptomatic adult Chiari malformation Type I

Chiari malformation Type I (CMI) is characterized by herniation of the cerebellar tonsils through the foramen magnum. The pathophysiology of CMI is not well elucidated; however, the prevailing theory focuses on the underdevelopment of the posterior cranial fossa which results in tonsillar herniation...

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Autores principales: Labuda, Rick, Nwotchouang, Blaise Simplice Talla, Ibrahimy, Alaaddin, Allen, Philip A., Oshinski, John N., Klinge, Petra, Loth, Francis
Formato: Online Artículo Texto
Lenguaje:English
Publicado: 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8730378/
https://www.ncbi.nlm.nih.gov/pubmed/34992329
http://dx.doi.org/10.1016/j.mehy.2021.110740
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author Labuda, Rick
Nwotchouang, Blaise Simplice Talla
Ibrahimy, Alaaddin
Allen, Philip A.
Oshinski, John N.
Klinge, Petra
Loth, Francis
author_facet Labuda, Rick
Nwotchouang, Blaise Simplice Talla
Ibrahimy, Alaaddin
Allen, Philip A.
Oshinski, John N.
Klinge, Petra
Loth, Francis
author_sort Labuda, Rick
collection PubMed
description Chiari malformation Type I (CMI) is characterized by herniation of the cerebellar tonsils through the foramen magnum. The pathophysiology of CMI is not well elucidated; however, the prevailing theory focuses on the underdevelopment of the posterior cranial fossa which results in tonsillar herniation. Symptoms are believed to be due to the herniation causing resistance to the natural flow of cerebrospinal fluid (CSF) and exerting a mass effect on nearby neural tissue. However, asymptomatic cases vastly outnumber symptomatic ones and it is not known why some people become symptomatic. Recently, it has been proposed that CMI symptoms are primarily due to instability of either the atlanto-axial (AA) or the atlanto-occipital (AO) joint and the cerebellar tonsils herniate to prevent mechanical pinching. However, only a small percentage of patients exhibit clinical instability and these theories do not account for asymptomatic herniations. We propose that the pathophysiology of adult CMI involves a combination of craniocervical abnormalities which leads to tonsillar herniation and reduced compliance of the cervical spinal canal. Specifically, abnormal AO and/or AA joint morphology leads to chronic cervical instability, often subclinical, in a large portion of CMI patients. This in turn causes overwork of the suboccipital muscles as they try to compensate for the instability. Over time, the repeated, involuntary activation of these muscles leads to mechanical overload of the myodural bridge complex, altering the mechanical properties of the dura it merges with. As a result, the dura becomes stiffer, reducing the overall compliance of the cervical region. This lower compliance, combined with CSF resistance at the same level, leads to intracranial pressure peaks during the cardiac cycle (pulse pressure) that are amplified during activities such as coughing, sneezing, and physical exertion. This increase in pulse pressure reduces the compliance of the cervical subarachnoid space which increases the CSF wave speed in the spinal canal, and further increases pulse pressure in a feedback loop. Finally, the abnormal pressure environment induces greater neural tissue motion and strain, causing microstructural damage to the cerebellum, brainstem, and cervical spinal cord, and leading to symptoms. This hypothesis explains how the combination of craniocervical bony abnormalities, anatomic CSF restriction, and reduced compliance leads to symptoms in adult CMI.
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spelling pubmed-87303782022-01-05 A new hypothesis for the pathophysiology of symptomatic adult Chiari malformation Type I Labuda, Rick Nwotchouang, Blaise Simplice Talla Ibrahimy, Alaaddin Allen, Philip A. Oshinski, John N. Klinge, Petra Loth, Francis Med Hypotheses Article Chiari malformation Type I (CMI) is characterized by herniation of the cerebellar tonsils through the foramen magnum. The pathophysiology of CMI is not well elucidated; however, the prevailing theory focuses on the underdevelopment of the posterior cranial fossa which results in tonsillar herniation. Symptoms are believed to be due to the herniation causing resistance to the natural flow of cerebrospinal fluid (CSF) and exerting a mass effect on nearby neural tissue. However, asymptomatic cases vastly outnumber symptomatic ones and it is not known why some people become symptomatic. Recently, it has been proposed that CMI symptoms are primarily due to instability of either the atlanto-axial (AA) or the atlanto-occipital (AO) joint and the cerebellar tonsils herniate to prevent mechanical pinching. However, only a small percentage of patients exhibit clinical instability and these theories do not account for asymptomatic herniations. We propose that the pathophysiology of adult CMI involves a combination of craniocervical abnormalities which leads to tonsillar herniation and reduced compliance of the cervical spinal canal. Specifically, abnormal AO and/or AA joint morphology leads to chronic cervical instability, often subclinical, in a large portion of CMI patients. This in turn causes overwork of the suboccipital muscles as they try to compensate for the instability. Over time, the repeated, involuntary activation of these muscles leads to mechanical overload of the myodural bridge complex, altering the mechanical properties of the dura it merges with. As a result, the dura becomes stiffer, reducing the overall compliance of the cervical region. This lower compliance, combined with CSF resistance at the same level, leads to intracranial pressure peaks during the cardiac cycle (pulse pressure) that are amplified during activities such as coughing, sneezing, and physical exertion. This increase in pulse pressure reduces the compliance of the cervical subarachnoid space which increases the CSF wave speed in the spinal canal, and further increases pulse pressure in a feedback loop. Finally, the abnormal pressure environment induces greater neural tissue motion and strain, causing microstructural damage to the cerebellum, brainstem, and cervical spinal cord, and leading to symptoms. This hypothesis explains how the combination of craniocervical bony abnormalities, anatomic CSF restriction, and reduced compliance leads to symptoms in adult CMI. 2021-12-14 2022-01 /pmc/articles/PMC8730378/ /pubmed/34992329 http://dx.doi.org/10.1016/j.mehy.2021.110740 Text en https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/ (https://creativecommons.org/licenses/by-nc-nd/4.0/) ).
spellingShingle Article
Labuda, Rick
Nwotchouang, Blaise Simplice Talla
Ibrahimy, Alaaddin
Allen, Philip A.
Oshinski, John N.
Klinge, Petra
Loth, Francis
A new hypothesis for the pathophysiology of symptomatic adult Chiari malformation Type I
title A new hypothesis for the pathophysiology of symptomatic adult Chiari malformation Type I
title_full A new hypothesis for the pathophysiology of symptomatic adult Chiari malformation Type I
title_fullStr A new hypothesis for the pathophysiology of symptomatic adult Chiari malformation Type I
title_full_unstemmed A new hypothesis for the pathophysiology of symptomatic adult Chiari malformation Type I
title_short A new hypothesis for the pathophysiology of symptomatic adult Chiari malformation Type I
title_sort new hypothesis for the pathophysiology of symptomatic adult chiari malformation type i
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8730378/
https://www.ncbi.nlm.nih.gov/pubmed/34992329
http://dx.doi.org/10.1016/j.mehy.2021.110740
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