Multicenter Prospective Analysis of Hypertrophic Olivary Degeneration Following Infratentorial Stroke (HOD-IS): Evaluation of Disease Epidemiology, Clinical Presentation, and MR-Imaging Aspects
Introduction: Ischemic and hemorrhagic strokes in the brainstem and cerebellum with injury to the functional loop of the Guillain-Mollaret triangle (GMT) can trigger a series of events that result in secondary trans-synaptic neurodegeneration of the inferior olivary nucleus. In an unknown percentage...
Main Authors: | , , , , , , , , , , , , |
---|---|
Format: | Article |
Language: | English |
Published: |
Frontiers Media S.A.
2021-07-01
|
Series: | Frontiers in Neurology |
Subjects: | |
Online Access: | https://www.frontiersin.org/articles/10.3389/fneur.2021.675123/full |
id |
doaj-f90ed04807e047ec9641d72374d48bd8 |
---|---|
record_format |
Article |
collection |
DOAJ |
language |
English |
format |
Article |
sources |
DOAJ |
author |
Martin A. Schaller-Paule Eike Steidl Manoj Shrestha Ralf Deichmann Helmuth Steinmetz Alexander Seiler Sriramya Lapa Thorsten Steiner Thorsten Steiner Sven Thonke Stefan Weidauer Juergen Konczalla Elke Hattingen Christian Foerch |
spellingShingle |
Martin A. Schaller-Paule Eike Steidl Manoj Shrestha Ralf Deichmann Helmuth Steinmetz Alexander Seiler Sriramya Lapa Thorsten Steiner Thorsten Steiner Sven Thonke Stefan Weidauer Juergen Konczalla Elke Hattingen Christian Foerch Multicenter Prospective Analysis of Hypertrophic Olivary Degeneration Following Infratentorial Stroke (HOD-IS): Evaluation of Disease Epidemiology, Clinical Presentation, and MR-Imaging Aspects Frontiers in Neurology neurodegeneration cerebellum palatal tremor brainstem connectivity tractography |
author_facet |
Martin A. Schaller-Paule Eike Steidl Manoj Shrestha Ralf Deichmann Helmuth Steinmetz Alexander Seiler Sriramya Lapa Thorsten Steiner Thorsten Steiner Sven Thonke Stefan Weidauer Juergen Konczalla Elke Hattingen Christian Foerch |
author_sort |
Martin A. Schaller-Paule |
title |
Multicenter Prospective Analysis of Hypertrophic Olivary Degeneration Following Infratentorial Stroke (HOD-IS): Evaluation of Disease Epidemiology, Clinical Presentation, and MR-Imaging Aspects |
title_short |
Multicenter Prospective Analysis of Hypertrophic Olivary Degeneration Following Infratentorial Stroke (HOD-IS): Evaluation of Disease Epidemiology, Clinical Presentation, and MR-Imaging Aspects |
title_full |
Multicenter Prospective Analysis of Hypertrophic Olivary Degeneration Following Infratentorial Stroke (HOD-IS): Evaluation of Disease Epidemiology, Clinical Presentation, and MR-Imaging Aspects |
title_fullStr |
Multicenter Prospective Analysis of Hypertrophic Olivary Degeneration Following Infratentorial Stroke (HOD-IS): Evaluation of Disease Epidemiology, Clinical Presentation, and MR-Imaging Aspects |
title_full_unstemmed |
Multicenter Prospective Analysis of Hypertrophic Olivary Degeneration Following Infratentorial Stroke (HOD-IS): Evaluation of Disease Epidemiology, Clinical Presentation, and MR-Imaging Aspects |
title_sort |
multicenter prospective analysis of hypertrophic olivary degeneration following infratentorial stroke (hod-is): evaluation of disease epidemiology, clinical presentation, and mr-imaging aspects |
publisher |
Frontiers Media S.A. |
series |
Frontiers in Neurology |
issn |
1664-2295 |
publishDate |
2021-07-01 |
description |
Introduction: Ischemic and hemorrhagic strokes in the brainstem and cerebellum with injury to the functional loop of the Guillain-Mollaret triangle (GMT) can trigger a series of events that result in secondary trans-synaptic neurodegeneration of the inferior olivary nucleus. In an unknown percentage of patients, this leads to a condition called hypertrophic olivary degeneration (HOD). Characteristic clinical symptoms of HOD progress slowly over months and consist of a rhythmic palatal tremor, vertical pendular nystagmus, and Holmes tremor of the upper limbs. Diffusion Tensor Imaging (DTI) with tractography is a promising method to identify functional pathway lesions along the cerebello-thalamo-cortical connectivity and to generate a deeper understanding of the HOD pathophysiology. The incidence of HOD development following stroke and the timeline of clinical symptoms have not yet been determined in prospective studies—a prerequisite for the surveillance of patients at risk.Methods and Analysis: Patients with ischemic and hemorrhagic strokes in the brainstem and cerebellum with a topo-anatomical relation to the GMT are recruited within certified stroke units of the Interdisciplinary Neurovascular Network of the Rhine-Main. Matching lesions are identified using a predefined MRI template. Eligible patients are prospectively followed up and present at 4 and 8 months after the index event. During study visits, a clinical neurological examination and brain MRI, including high-resolution T2-, proton-density-weighted imaging, and DTI tractography, are performed. Fiberoptic endoscopic evaluation of swallowing is optional if palatal tremor is encountered.Study Outcomes: The primary endpoint of this prospective clinical multicenter study is to determine the frequency of radiological HOD development in patients with a posterior fossa stroke affecting the GMT at 8 months after the index event. Secondary endpoints are identification of (1) the timeline and relevance of clinical symptoms, (2) lesion localizations more prone to HOD occurrence, and (3) the best MR-imaging regimen for HOD identification. Additionally, (4) DTI tractography data are used to analyze individual pathway lesions. The aim is to contribute to the epidemiological and pathophysiological understanding of HOD and hereby facilitate future research on therapeutic and prophylactic measures.Clinical Trial Registration: HOD-IS is a registered trial at https://www.drks.de/drks_web/navigate.do?navigationId=trial.HTML&TRIAL_ID=DRKS00020549. |
topic |
neurodegeneration cerebellum palatal tremor brainstem connectivity tractography |
url |
https://www.frontiersin.org/articles/10.3389/fneur.2021.675123/full |
work_keys_str_mv |
AT martinaschallerpaule multicenterprospectiveanalysisofhypertrophicolivarydegenerationfollowinginfratentorialstrokehodisevaluationofdiseaseepidemiologyclinicalpresentationandmrimagingaspects AT eikesteidl multicenterprospectiveanalysisofhypertrophicolivarydegenerationfollowinginfratentorialstrokehodisevaluationofdiseaseepidemiologyclinicalpresentationandmrimagingaspects AT manojshrestha multicenterprospectiveanalysisofhypertrophicolivarydegenerationfollowinginfratentorialstrokehodisevaluationofdiseaseepidemiologyclinicalpresentationandmrimagingaspects AT ralfdeichmann multicenterprospectiveanalysisofhypertrophicolivarydegenerationfollowinginfratentorialstrokehodisevaluationofdiseaseepidemiologyclinicalpresentationandmrimagingaspects AT helmuthsteinmetz multicenterprospectiveanalysisofhypertrophicolivarydegenerationfollowinginfratentorialstrokehodisevaluationofdiseaseepidemiologyclinicalpresentationandmrimagingaspects AT alexanderseiler multicenterprospectiveanalysisofhypertrophicolivarydegenerationfollowinginfratentorialstrokehodisevaluationofdiseaseepidemiologyclinicalpresentationandmrimagingaspects AT sriramyalapa multicenterprospectiveanalysisofhypertrophicolivarydegenerationfollowinginfratentorialstrokehodisevaluationofdiseaseepidemiologyclinicalpresentationandmrimagingaspects AT thorstensteiner multicenterprospectiveanalysisofhypertrophicolivarydegenerationfollowinginfratentorialstrokehodisevaluationofdiseaseepidemiologyclinicalpresentationandmrimagingaspects AT thorstensteiner multicenterprospectiveanalysisofhypertrophicolivarydegenerationfollowinginfratentorialstrokehodisevaluationofdiseaseepidemiologyclinicalpresentationandmrimagingaspects AT sventhonke multicenterprospectiveanalysisofhypertrophicolivarydegenerationfollowinginfratentorialstrokehodisevaluationofdiseaseepidemiologyclinicalpresentationandmrimagingaspects AT stefanweidauer multicenterprospectiveanalysisofhypertrophicolivarydegenerationfollowinginfratentorialstrokehodisevaluationofdiseaseepidemiologyclinicalpresentationandmrimagingaspects AT juergenkonczalla multicenterprospectiveanalysisofhypertrophicolivarydegenerationfollowinginfratentorialstrokehodisevaluationofdiseaseepidemiologyclinicalpresentationandmrimagingaspects AT elkehattingen multicenterprospectiveanalysisofhypertrophicolivarydegenerationfollowinginfratentorialstrokehodisevaluationofdiseaseepidemiologyclinicalpresentationandmrimagingaspects AT christianfoerch multicenterprospectiveanalysisofhypertrophicolivarydegenerationfollowinginfratentorialstrokehodisevaluationofdiseaseepidemiologyclinicalpresentationandmrimagingaspects |
_version_ |
1721297810809159680 |
spelling |
doaj-f90ed04807e047ec9641d72374d48bd82021-07-16T10:02:36ZengFrontiers Media S.A.Frontiers in Neurology1664-22952021-07-011210.3389/fneur.2021.675123675123Multicenter Prospective Analysis of Hypertrophic Olivary Degeneration Following Infratentorial Stroke (HOD-IS): Evaluation of Disease Epidemiology, Clinical Presentation, and MR-Imaging AspectsMartin A. Schaller-Paule0Eike Steidl1Manoj Shrestha2Ralf Deichmann3Helmuth Steinmetz4Alexander Seiler5Sriramya Lapa6Thorsten Steiner7Thorsten Steiner8Sven Thonke9Stefan Weidauer10Juergen Konczalla11Elke Hattingen12Christian Foerch13Department of Neurology, University Hospital Frankfurt, Goethe-University, Frankfurt, GermanyInstitute of Neuroradiology, University Hospital Frankfurt, Goethe-University, Frankfurt, GermanyBrain Imaging Center (BIC), Goethe-University, Frankfurt, GermanyBrain Imaging Center (BIC), Goethe-University, Frankfurt, GermanyDepartment of Neurology, University Hospital Frankfurt, Goethe-University, Frankfurt, GermanyDepartment of Neurology, University Hospital Frankfurt, Goethe-University, Frankfurt, GermanyDepartment of Neurology, University Hospital Frankfurt, Goethe-University, Frankfurt, GermanyDepartment of Neurology, Klinikum Frankfurt Höchst, Teaching Hospital of the Goethe-University, Frankfurt, GermanyDepartment of Neurology, Heidelberg University Hospital, Heidelberg, GermanyDepartment of Neurology, Klinikum Hanau, Teaching Hospital of the Goethe-University, Frankfurt, GermanyDepartment of Neurology, Sankt Katharinen Hospital, Teaching Hospital of the Goethe-University Frankfurt, Frankfurt, GermanyDepartment of Neurosurgery, University Hospital Frankfurt, Goethe-University, Frankfurt, GermanyInstitute of Neuroradiology, University Hospital Frankfurt, Goethe-University, Frankfurt, GermanyDepartment of Neurology, University Hospital Frankfurt, Goethe-University, Frankfurt, GermanyIntroduction: Ischemic and hemorrhagic strokes in the brainstem and cerebellum with injury to the functional loop of the Guillain-Mollaret triangle (GMT) can trigger a series of events that result in secondary trans-synaptic neurodegeneration of the inferior olivary nucleus. In an unknown percentage of patients, this leads to a condition called hypertrophic olivary degeneration (HOD). Characteristic clinical symptoms of HOD progress slowly over months and consist of a rhythmic palatal tremor, vertical pendular nystagmus, and Holmes tremor of the upper limbs. Diffusion Tensor Imaging (DTI) with tractography is a promising method to identify functional pathway lesions along the cerebello-thalamo-cortical connectivity and to generate a deeper understanding of the HOD pathophysiology. The incidence of HOD development following stroke and the timeline of clinical symptoms have not yet been determined in prospective studies—a prerequisite for the surveillance of patients at risk.Methods and Analysis: Patients with ischemic and hemorrhagic strokes in the brainstem and cerebellum with a topo-anatomical relation to the GMT are recruited within certified stroke units of the Interdisciplinary Neurovascular Network of the Rhine-Main. Matching lesions are identified using a predefined MRI template. Eligible patients are prospectively followed up and present at 4 and 8 months after the index event. During study visits, a clinical neurological examination and brain MRI, including high-resolution T2-, proton-density-weighted imaging, and DTI tractography, are performed. Fiberoptic endoscopic evaluation of swallowing is optional if palatal tremor is encountered.Study Outcomes: The primary endpoint of this prospective clinical multicenter study is to determine the frequency of radiological HOD development in patients with a posterior fossa stroke affecting the GMT at 8 months after the index event. Secondary endpoints are identification of (1) the timeline and relevance of clinical symptoms, (2) lesion localizations more prone to HOD occurrence, and (3) the best MR-imaging regimen for HOD identification. Additionally, (4) DTI tractography data are used to analyze individual pathway lesions. The aim is to contribute to the epidemiological and pathophysiological understanding of HOD and hereby facilitate future research on therapeutic and prophylactic measures.Clinical Trial Registration: HOD-IS is a registered trial at https://www.drks.de/drks_web/navigate.do?navigationId=trial.HTML&TRIAL_ID=DRKS00020549.https://www.frontiersin.org/articles/10.3389/fneur.2021.675123/fullneurodegenerationcerebellumpalatal tremorbrainstemconnectivitytractography |