Case Report: Globus Pallidus Internus (GPi) Deep Brain Stimulation Induced Keyboard Typing Dysfunction

Joshua K. Wong; Melissa J. Armstrong; Leonardo Almeida; Aparna Wagle Shukla; Addie Patterson; Michael S. Okun; Irene A. Malaty

doi:10.3389/fnhum.2020.583441

Case Report: Globus Pallidus Internus (GPi) Deep Brain Stimulation Induced Keyboard Typing Dysfunction

Joshua K. Wong
, Melissa J. Armstrong
, Leonardo Almeida
, Aparna Wagle Shukla
, Addie Patterson
, Michael S. Okun
, Irene A. Malaty

Research output: Contribution to journal › Article › peer-review

3 Scopus citations

Abstract

Introduction:Typing on a keyboard requires complex collaboration between visuospatial/procedural memory, language, and motor function. The impaired ability to type, independent of motor deficits, apraxia, or aphasia has been coined “dystypia.” Case Presentation: A 68-year-old woman with a history of blepharospasm, oromandibular, and segmental dystonia underwent bilateral pallidal deep brain stimulation (DBS) because of a waning response to botulinum toxin therapy. Following DBS, she discovered she no longer “remembered” how to type fluidly and had to “hunt and peck” for letters on the keyboard. This issue persisted at a 2-year follow-up. The patient underwent serial typing tests with the DBS ON vs. OFF. Post-operative lead reconstruction was performed using Lead-DBS. The volume of tissue activation (VTA) modeling was combined with whole-brain tractography. Results: Typing improved when the device was switched to the DBS OFF state. Cortical mapping revealed strong modulation of the right angular gyrus, left calcarine fissure, and left cuneus. There was also activation of bilateral supplemental motor areas and superior parietal gyri. Discussion: Shared lesion topography analysis of dystypia cases in the literature has suggested the involvement of the superior longitudinal fasciculus (SLF). The SLF involves the superior parietal lobe, angular gyrus, supramarginal gyrus, and arcuate fasciculus. Our patient’s connectivity pattern suggested SLF involvement. The improvement in OFF state typing and her imaging together suggested that the dystypia in her case was a stimulation-induced side effect. Conclusion: Dystypia is a rare side effect of Globus Pallidus Internus (GPi) DBS therapy and may be associated with SLF involvement.

Original language	English (US)
Article number	583441
Journal	Frontiers in Human Neuroscience
Volume	14
DOIs	https://doi.org/10.3389/fnhum.2020.583441
State	Published - Nov 9 2020
Externally published	Yes

Bibliographical note

Funding Information:
Funding. This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. JW’s DBS research is supported by R25 NS108939. MA reports receipt of personal fees from the American Academy of Neurology (consultancy services); grants from the Lewy Body Dementia Association; the Michael J. Fox Foundation; funding from the Agency of Healthcare Research and Quality; and others from Oxford University Press (royalties) outside the submitted work. LA serves as a consultant and has received honoraria from Medtronic and Boston Scientific. AW reports grants from the NIH and has received grant support from Benign Essential Blepharospasm Research Foundation, Dystonia Coalition, Dystonia Medical Research Foundation, National Organization for Rare Disorders, and grant support from NIH (KL2 and K23 NS092957-01A1). Reports receiving honoraria from Acadia, Cavion, Elsevier, and MJFF in past; participates as a co-investigator for several NIH, foundations, and industry-sponsored trials over the years but has not received honoraria. MO serves as a consultant for the Parkinson’s Foundation and has received research grants from NIH, Parkinson’s Foundation, the Michael J. Fox Foundation, the Parkinson Alliance, Smallwood Foundation, the Bachmann-Strauss Foundation, the Tourette Syndrome Association, and the UF Foundation. MO’s DBS research is supported by R01 NR014852 and R01NS096008. MO has received royalties for publications with Demos, Manson, Amazon, Smashwords, Books4Patients, Perseus, Robert Rose, Oxford, and Cambridge (movement disorders books). MO is an associate editor for the New England Journal of Medicine Journal Watch Neurology. MO has participated in CME and educational activities on movement disorders sponsored by the Academy for Healthcare Learning, PeerView, Prime, QuantiaMD, WebMD/Medscape, Medicus, MedNet, Einstein, MedNet, Henry Stewart, American Academy of Neurology, Movement Disorders Society and by Vanderbilt University. The institution and not MO receives grants from Medtronic, Abbvie, Abbott, and Allergan and the principal investigator (PI) has no financial interest in these grants. MO has participated as a site PI and/or co-investigator for several NIH, foundations, and industry-sponsored trials over the years but has not received honoraria.

Funding Information:
This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. JW’s DBS research is supported by R25 NS108939. MA reports receipt of personal fees from the American Academy of Neurology (consultancy services); grants from the Lewy Body Dementia Association; the Michael J. Fox Foundation; funding from the Agency of Healthcare Research and Quality; and others from Oxford University Press (royalties) outside the submitted work. LA serves as a consultant and has received honoraria from Medtronic and Boston Scientific. AW reports grants from the NIH and has received grant support from Benign Essential Blepharospasm Research Foundation, Dystonia Coalition, Dystonia Medical Research Foundation, National Organization for Rare Disorders, and grant support from NIH (KL2 and K23 NS092957-01A1). Reports receiving honoraria from Acadia, Cavion, Elsevier, and MJFF in past; participates as a co-investigator for several NIH, foundations, and industry-sponsored trials over the years but has not received honoraria. MO serves as a consultant for the Parkinson’s Foundation and has received research grants from NIH, Parkinson’s Foundation, the Michael J. Fox Foundation, the Parkinson Alliance, Smallwood Foundation, the Bachmann-Strauss Foundation, the Tourette Syndrome Association, and the UF Foundation. MO’s DBS research is supported by R01 NR014852 and R01NS096008. MO has received royalties for publications with Demos, Manson, Amazon, Smashwords, Books4Patients, Perseus, Robert Rose, Oxford, and Cambridge (movement disorders books). MO is an associate editor for the New England Journal of Medicine Journal Watch Neurology. MO has participated in CME and educational activities on movement disorders sponsored by the Academy for Healthcare Learning, PeerView, Prime, QuantiaMD, WebMD/Medscape, Medicus, MedNet, Einstein, MedNet, Henry Stewart, American Academy of Neurology, Movement Disorders Society and by Vanderbilt University. The institution and not MO receives grants from Medtronic, Abbvie, Abbott, and Allergan and the principal investigator (PI) has no financial interest in these grants. MO has participated as a site PI and/or co-investigator for several NIH, foundations, and industry-sponsored trials over the years but has not received honoraria.

Publisher Copyright:
© Copyright © 2020 Wong, Armstrong, Almeida, Wagle Shukla, Patterson, Okun and Malaty.

Keywords

connectomics
deep brain stimulation
dystonia
dystypia
stimulation induced side effect

Publisher link

10.3389/fnhum.2020.583441

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@article{6fc83722da1b42f6a7fd08e23e3177f1,

title = "Case Report: Globus Pallidus Internus (GPi) Deep Brain Stimulation Induced Keyboard Typing Dysfunction",

abstract = "Introduction:Typing on a keyboard requires complex collaboration between visuospatial/procedural memory, language, and motor function. The impaired ability to type, independent of motor deficits, apraxia, or aphasia has been coined “dystypia.” Case Presentation: A 68-year-old woman with a history of blepharospasm, oromandibular, and segmental dystonia underwent bilateral pallidal deep brain stimulation (DBS) because of a waning response to botulinum toxin therapy. Following DBS, she discovered she no longer “remembered” how to type fluidly and had to “hunt and peck” for letters on the keyboard. This issue persisted at a 2-year follow-up. The patient underwent serial typing tests with the DBS ON vs. OFF. Post-operative lead reconstruction was performed using Lead-DBS. The volume of tissue activation (VTA) modeling was combined with whole-brain tractography. Results: Typing improved when the device was switched to the DBS OFF state. Cortical mapping revealed strong modulation of the right angular gyrus, left calcarine fissure, and left cuneus. There was also activation of bilateral supplemental motor areas and superior parietal gyri. Discussion: Shared lesion topography analysis of dystypia cases in the literature has suggested the involvement of the superior longitudinal fasciculus (SLF). The SLF involves the superior parietal lobe, angular gyrus, supramarginal gyrus, and arcuate fasciculus. Our patient{\textquoteright}s connectivity pattern suggested SLF involvement. The improvement in OFF state typing and her imaging together suggested that the dystypia in her case was a stimulation-induced side effect. Conclusion: Dystypia is a rare side effect of Globus Pallidus Internus (GPi) DBS therapy and may be associated with SLF involvement.",

keywords = "connectomics, deep brain stimulation, dystonia, dystypia, stimulation induced side effect",

author = "Wong, \{Joshua K.\} and Armstrong, \{Melissa J.\} and Leonardo Almeida and \{Wagle Shukla\}, Aparna and Addie Patterson and Okun, \{Michael S.\} and Malaty, \{Irene A.\}",

note = "Funding Information: Funding. This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. JW{\textquoteright}s DBS research is supported by R25 NS108939. MA reports receipt of personal fees from the American Academy of Neurology (consultancy services); grants from the Lewy Body Dementia Association; the Michael J. Fox Foundation; funding from the Agency of Healthcare Research and Quality; and others from Oxford University Press (royalties) outside the submitted work. LA serves as a consultant and has received honoraria from Medtronic and Boston Scientific. AW reports grants from the NIH and has received grant support from Benign Essential Blepharospasm Research Foundation, Dystonia Coalition, Dystonia Medical Research Foundation, National Organization for Rare Disorders, and grant support from NIH (KL2 and K23 NS092957-01A1). Reports receiving honoraria from Acadia, Cavion, Elsevier, and MJFF in past; participates as a co-investigator for several NIH, foundations, and industry-sponsored trials over the years but has not received honoraria. MO serves as a consultant for the Parkinson{\textquoteright}s Foundation and has received research grants from NIH, Parkinson{\textquoteright}s Foundation, the Michael J. Fox Foundation, the Parkinson Alliance, Smallwood Foundation, the Bachmann-Strauss Foundation, the Tourette Syndrome Association, and the UF Foundation. MO{\textquoteright}s DBS research is supported by R01 NR014852 and R01NS096008. MO has received royalties for publications with Demos, Manson, Amazon, Smashwords, Books4Patients, Perseus, Robert Rose, Oxford, and Cambridge (movement disorders books). MO is an associate editor for the New England Journal of Medicine Journal Watch Neurology. MO has participated in CME and educational activities on movement disorders sponsored by the Academy for Healthcare Learning, PeerView, Prime, QuantiaMD, WebMD/Medscape, Medicus, MedNet, Einstein, MedNet, Henry Stewart, American Academy of Neurology, Movement Disorders Society and by Vanderbilt University. The institution and not MO receives grants from Medtronic, Abbvie, Abbott, and Allergan and the principal investigator (PI) has no financial interest in these grants. MO has participated as a site PI and/or co-investigator for several NIH, foundations, and industry-sponsored trials over the years but has not received honoraria. Funding Information: This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. JW{\textquoteright}s DBS research is supported by R25 NS108939. MA reports receipt of personal fees from the American Academy of Neurology (consultancy services); grants from the Lewy Body Dementia Association; the Michael J. Fox Foundation; funding from the Agency of Healthcare Research and Quality; and others from Oxford University Press (royalties) outside the submitted work. LA serves as a consultant and has received honoraria from Medtronic and Boston Scientific. AW reports grants from the NIH and has received grant support from Benign Essential Blepharospasm Research Foundation, Dystonia Coalition, Dystonia Medical Research Foundation, National Organization for Rare Disorders, and grant support from NIH (KL2 and K23 NS092957-01A1). Reports receiving honoraria from Acadia, Cavion, Elsevier, and MJFF in past; participates as a co-investigator for several NIH, foundations, and industry-sponsored trials over the years but has not received honoraria. MO serves as a consultant for the Parkinson{\textquoteright}s Foundation and has received research grants from NIH, Parkinson{\textquoteright}s Foundation, the Michael J. Fox Foundation, the Parkinson Alliance, Smallwood Foundation, the Bachmann-Strauss Foundation, the Tourette Syndrome Association, and the UF Foundation. MO{\textquoteright}s DBS research is supported by R01 NR014852 and R01NS096008. MO has received royalties for publications with Demos, Manson, Amazon, Smashwords, Books4Patients, Perseus, Robert Rose, Oxford, and Cambridge (movement disorders books). MO is an associate editor for the New England Journal of Medicine Journal Watch Neurology. MO has participated in CME and educational activities on movement disorders sponsored by the Academy for Healthcare Learning, PeerView, Prime, QuantiaMD, WebMD/Medscape, Medicus, MedNet, Einstein, MedNet, Henry Stewart, American Academy of Neurology, Movement Disorders Society and by Vanderbilt University. The institution and not MO receives grants from Medtronic, Abbvie, Abbott, and Allergan and the principal investigator (PI) has no financial interest in these grants. MO has participated as a site PI and/or co-investigator for several NIH, foundations, and industry-sponsored trials over the years but has not received honoraria. Publisher Copyright: {\textcopyright} Copyright {\textcopyright} 2020 Wong, Armstrong, Almeida, Wagle Shukla, Patterson, Okun and Malaty.",

year = "2020",

month = nov,

day = "9",

doi = "10.3389/fnhum.2020.583441",

language = "English (US)",

volume = "14",

journal = "Frontiers in Human Neuroscience",

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TY - JOUR

T1 - Case Report

T2 - Globus Pallidus Internus (GPi) Deep Brain Stimulation Induced Keyboard Typing Dysfunction

AU - Wong, Joshua K.

AU - Armstrong, Melissa J.

AU - Almeida, Leonardo

AU - Wagle Shukla, Aparna

AU - Patterson, Addie

AU - Okun, Michael S.

AU - Malaty, Irene A.

N1 - Funding Information: Funding. This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. JW’s DBS research is supported by R25 NS108939. MA reports receipt of personal fees from the American Academy of Neurology (consultancy services); grants from the Lewy Body Dementia Association; the Michael J. Fox Foundation; funding from the Agency of Healthcare Research and Quality; and others from Oxford University Press (royalties) outside the submitted work. LA serves as a consultant and has received honoraria from Medtronic and Boston Scientific. AW reports grants from the NIH and has received grant support from Benign Essential Blepharospasm Research Foundation, Dystonia Coalition, Dystonia Medical Research Foundation, National Organization for Rare Disorders, and grant support from NIH (KL2 and K23 NS092957-01A1). Reports receiving honoraria from Acadia, Cavion, Elsevier, and MJFF in past; participates as a co-investigator for several NIH, foundations, and industry-sponsored trials over the years but has not received honoraria. MO serves as a consultant for the Parkinson’s Foundation and has received research grants from NIH, Parkinson’s Foundation, the Michael J. Fox Foundation, the Parkinson Alliance, Smallwood Foundation, the Bachmann-Strauss Foundation, the Tourette Syndrome Association, and the UF Foundation. MO’s DBS research is supported by R01 NR014852 and R01NS096008. MO has received royalties for publications with Demos, Manson, Amazon, Smashwords, Books4Patients, Perseus, Robert Rose, Oxford, and Cambridge (movement disorders books). MO is an associate editor for the New England Journal of Medicine Journal Watch Neurology. MO has participated in CME and educational activities on movement disorders sponsored by the Academy for Healthcare Learning, PeerView, Prime, QuantiaMD, WebMD/Medscape, Medicus, MedNet, Einstein, MedNet, Henry Stewart, American Academy of Neurology, Movement Disorders Society and by Vanderbilt University. The institution and not MO receives grants from Medtronic, Abbvie, Abbott, and Allergan and the principal investigator (PI) has no financial interest in these grants. MO has participated as a site PI and/or co-investigator for several NIH, foundations, and industry-sponsored trials over the years but has not received honoraria. Funding Information: This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. JW’s DBS research is supported by R25 NS108939. MA reports receipt of personal fees from the American Academy of Neurology (consultancy services); grants from the Lewy Body Dementia Association; the Michael J. Fox Foundation; funding from the Agency of Healthcare Research and Quality; and others from Oxford University Press (royalties) outside the submitted work. LA serves as a consultant and has received honoraria from Medtronic and Boston Scientific. AW reports grants from the NIH and has received grant support from Benign Essential Blepharospasm Research Foundation, Dystonia Coalition, Dystonia Medical Research Foundation, National Organization for Rare Disorders, and grant support from NIH (KL2 and K23 NS092957-01A1). Reports receiving honoraria from Acadia, Cavion, Elsevier, and MJFF in past; participates as a co-investigator for several NIH, foundations, and industry-sponsored trials over the years but has not received honoraria. MO serves as a consultant for the Parkinson’s Foundation and has received research grants from NIH, Parkinson’s Foundation, the Michael J. Fox Foundation, the Parkinson Alliance, Smallwood Foundation, the Bachmann-Strauss Foundation, the Tourette Syndrome Association, and the UF Foundation. MO’s DBS research is supported by R01 NR014852 and R01NS096008. MO has received royalties for publications with Demos, Manson, Amazon, Smashwords, Books4Patients, Perseus, Robert Rose, Oxford, and Cambridge (movement disorders books). MO is an associate editor for the New England Journal of Medicine Journal Watch Neurology. MO has participated in CME and educational activities on movement disorders sponsored by the Academy for Healthcare Learning, PeerView, Prime, QuantiaMD, WebMD/Medscape, Medicus, MedNet, Einstein, MedNet, Henry Stewart, American Academy of Neurology, Movement Disorders Society and by Vanderbilt University. The institution and not MO receives grants from Medtronic, Abbvie, Abbott, and Allergan and the principal investigator (PI) has no financial interest in these grants. MO has participated as a site PI and/or co-investigator for several NIH, foundations, and industry-sponsored trials over the years but has not received honoraria. Publisher Copyright: © Copyright © 2020 Wong, Armstrong, Almeida, Wagle Shukla, Patterson, Okun and Malaty.

PY - 2020/11/9

Y1 - 2020/11/9

N2 - Introduction:Typing on a keyboard requires complex collaboration between visuospatial/procedural memory, language, and motor function. The impaired ability to type, independent of motor deficits, apraxia, or aphasia has been coined “dystypia.” Case Presentation: A 68-year-old woman with a history of blepharospasm, oromandibular, and segmental dystonia underwent bilateral pallidal deep brain stimulation (DBS) because of a waning response to botulinum toxin therapy. Following DBS, she discovered she no longer “remembered” how to type fluidly and had to “hunt and peck” for letters on the keyboard. This issue persisted at a 2-year follow-up. The patient underwent serial typing tests with the DBS ON vs. OFF. Post-operative lead reconstruction was performed using Lead-DBS. The volume of tissue activation (VTA) modeling was combined with whole-brain tractography. Results: Typing improved when the device was switched to the DBS OFF state. Cortical mapping revealed strong modulation of the right angular gyrus, left calcarine fissure, and left cuneus. There was also activation of bilateral supplemental motor areas and superior parietal gyri. Discussion: Shared lesion topography analysis of dystypia cases in the literature has suggested the involvement of the superior longitudinal fasciculus (SLF). The SLF involves the superior parietal lobe, angular gyrus, supramarginal gyrus, and arcuate fasciculus. Our patient’s connectivity pattern suggested SLF involvement. The improvement in OFF state typing and her imaging together suggested that the dystypia in her case was a stimulation-induced side effect. Conclusion: Dystypia is a rare side effect of Globus Pallidus Internus (GPi) DBS therapy and may be associated with SLF involvement.

AB - Introduction:Typing on a keyboard requires complex collaboration between visuospatial/procedural memory, language, and motor function. The impaired ability to type, independent of motor deficits, apraxia, or aphasia has been coined “dystypia.” Case Presentation: A 68-year-old woman with a history of blepharospasm, oromandibular, and segmental dystonia underwent bilateral pallidal deep brain stimulation (DBS) because of a waning response to botulinum toxin therapy. Following DBS, she discovered she no longer “remembered” how to type fluidly and had to “hunt and peck” for letters on the keyboard. This issue persisted at a 2-year follow-up. The patient underwent serial typing tests with the DBS ON vs. OFF. Post-operative lead reconstruction was performed using Lead-DBS. The volume of tissue activation (VTA) modeling was combined with whole-brain tractography. Results: Typing improved when the device was switched to the DBS OFF state. Cortical mapping revealed strong modulation of the right angular gyrus, left calcarine fissure, and left cuneus. There was also activation of bilateral supplemental motor areas and superior parietal gyri. Discussion: Shared lesion topography analysis of dystypia cases in the literature has suggested the involvement of the superior longitudinal fasciculus (SLF). The SLF involves the superior parietal lobe, angular gyrus, supramarginal gyrus, and arcuate fasciculus. Our patient’s connectivity pattern suggested SLF involvement. The improvement in OFF state typing and her imaging together suggested that the dystypia in her case was a stimulation-induced side effect. Conclusion: Dystypia is a rare side effect of Globus Pallidus Internus (GPi) DBS therapy and may be associated with SLF involvement.

KW - connectomics

KW - deep brain stimulation

KW - dystonia

KW - dystypia

KW - stimulation induced side effect

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UR - https://www.scopus.com/pages/publications/85096449441#tab=citedBy

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DO - 10.3389/fnhum.2020.583441

M3 - Article

AN - SCOPUS:85096449441

SN - 1662-5161

VL - 14

JO - Frontiers in Human Neuroscience

JF - Frontiers in Human Neuroscience

M1 - 583441

ER -

Case Report: Globus Pallidus Internus (GPi) Deep Brain Stimulation Induced Keyboard Typing Dysfunction

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