Thymectomy may not be associated with clinical improvement in MuSK myasthenia gravis

Katherine M. Clifford; Lisa D. Hobson-Webb; Michael Benatar; Ted M. Burns; Carolina Barnett; Nicholas J. Silvestri; James F. Howard; Amy Visser; Brian A. Crum; Richard Nowak; Rachel Beekman; Aditya Kumar; Katherine Ruzhansky; I. Hweii Amy Chen; Michael T. Pulley; Shannon M. Laboy; Melissa A. Fellman; Diantha B. Howard; Noah A. Kolb; Shane M. Greene; Mamatha Pasnoor; Mazen M. Dimachkie; Richard J. Barohn; Michael K. Hehir

doi:10.1002/mus.26404

Thymectomy may not be associated with clinical improvement in MuSK myasthenia gravis

Katherine M. Clifford, Lisa D. Hobson-Webb, Michael Benatar, Ted M. Burns, Carolina Barnett, Nicholas J. Silvestri, James F. Howard, Amy Visser, Brian A. Crum, Richard Nowak, Rachel Beekman, Aditya Kumar, Katherine Ruzhansky, I. Hweii Amy Chen, Michael T. Pulley, Shannon M. Laboy, Melissa A. Fellman, Diantha B. Howard, Noah A. Kolb, Shane M. GreeneMamatha Pasnoor, Mazen M. Dimachkie, Richard J. Barohn, Michael K. Hehir

Neurology

Research output: Contribution to journal › Review article › peer-review

53 Scopus citations

Abstract

Introduction: A randomized trial demonstrated benefit from thymectomy in nonthymomatous acetylcholine receptor (AChR)-antibody positive myasthenia gravis (MG). Uncontrolled observational and histologic studies suggest thymectomy may not be efficacious in anti–muscle-specific kinase (MuSK)-MG. Methods: The therapeutic impact of thymectomy was evaluated from data collected for a multicenter, retrospective blinded review of rituximab in MuSK-MG. Results: Baseline characteristics were similar between thymectomy (n = 26) and nonthymectomy (n = 29) groups, including treatment with rituximab (42% vs. 45%). At last visit, 35% of thymectomy subjects reached the primary endpoint, a Myasthenia Gravis Foundation of America (MGFA) post-intervention status (PIS) score of minimal manifestations (MM) or better, compared with 55% of controls (P = 0.17). After controlling for age at onset of MG, rituximab, prednisone, and intravenous immunoglobulin/plasma exchange treatment, thymectomy was not associated with greater likelihood of favorable clinical outcome (odds ratio = 0.43, 95% confidence interval 0.12–1.53, P = 0.19). Discussion: Thymectomy was not associated with additional clinical improvement in this multicenter cohort of MuSK-MG patients. Muscle Nerve 59:404–410, 2019.

Original language	English (US)
Pages (from-to)	404-410
Number of pages	7
Journal	Muscle and Nerve
Volume	59
Issue number	4
DOIs	https://doi.org/10.1002/mus.26404
State	Published - Apr 2019

Keywords

immunosuppressant therapy
muscle-specific kinase (MuSK) antibodies
myasthenia gravis
rituximab
thymectomy

ASJC Scopus subject areas

Physiology
Clinical Neurology
Cellular and Molecular Neuroscience
Physiology (medical)

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10.1002/mus.26404

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Clifford, K. M., Hobson-Webb, L. D., Benatar, M., Burns, T. M., Barnett, C., Silvestri, N. J., Howard, J. F., Visser, A., Crum, B. A., Nowak, R., Beekman, R., Kumar, A., Ruzhansky, K., Chen, I. H. A., Pulley, M. T., Laboy, S. M., Fellman, M. A., Howard, D. B., Kolb, N. A., ... Hehir, M. K. (2019). Thymectomy may not be associated with clinical improvement in MuSK myasthenia gravis. Muscle and Nerve, 59(4), 404-410. https://doi.org/10.1002/mus.26404

Clifford, KM, Hobson-Webb, LD, Benatar, M, Burns, TM, Barnett, C, Silvestri, NJ, Howard, JF, Visser, A, Crum, BA, Nowak, R, Beekman, R, Kumar, A, Ruzhansky, K, Chen, IHA, Pulley, MT, Laboy, SM, Fellman, MA, Howard, DB, Kolb, NA, Greene, SM, Pasnoor, M, Dimachkie, MM, Barohn, RJ & Hehir, MK 2019, 'Thymectomy may not be associated with clinical improvement in MuSK myasthenia gravis', Muscle and Nerve, vol. 59, no. 4, pp. 404-410. https://doi.org/10.1002/mus.26404

@article{49d0bb555236440db27cbb4b5d9ff3fa,

title = "Thymectomy may not be associated with clinical improvement in MuSK myasthenia gravis",

abstract = "Introduction: A randomized trial demonstrated benefit from thymectomy in nonthymomatous acetylcholine receptor (AChR)-antibody positive myasthenia gravis (MG). Uncontrolled observational and histologic studies suggest thymectomy may not be efficacious in anti–muscle-specific kinase (MuSK)-MG. Methods: The therapeutic impact of thymectomy was evaluated from data collected for a multicenter, retrospective blinded review of rituximab in MuSK-MG. Results: Baseline characteristics were similar between thymectomy (n = 26) and nonthymectomy (n = 29) groups, including treatment with rituximab (42% vs. 45%). At last visit, 35% of thymectomy subjects reached the primary endpoint, a Myasthenia Gravis Foundation of America (MGFA) post-intervention status (PIS) score of minimal manifestations (MM) or better, compared with 55% of controls (P = 0.17). After controlling for age at onset of MG, rituximab, prednisone, and intravenous immunoglobulin/plasma exchange treatment, thymectomy was not associated with greater likelihood of favorable clinical outcome (odds ratio = 0.43, 95% confidence interval 0.12–1.53, P = 0.19). Discussion: Thymectomy was not associated with additional clinical improvement in this multicenter cohort of MuSK-MG patients. Muscle Nerve 59:404–410, 2019.",

keywords = "immunosuppressant therapy, muscle-specific kinase (MuSK) antibodies, myasthenia gravis, rituximab, thymectomy",

author = "Clifford, {Katherine M.} and Hobson-Webb, {Lisa D.} and Michael Benatar and Burns, {Ted M.} and Carolina Barnett and Silvestri, {Nicholas J.} and Howard, {James F.} and Amy Visser and Crum, {Brian A.} and Richard Nowak and Rachel Beekman and Aditya Kumar and Katherine Ruzhansky and Chen, {I. Hweii Amy} and Pulley, {Michael T.} and Laboy, {Shannon M.} and Fellman, {Melissa A.} and Howard, {Diantha B.} and Kolb, {Noah A.} and Greene, {Shane M.} and Mamatha Pasnoor and Dimachkie, {Mazen M.} and Barohn, {Richard J.} and Hehir, {Michael K.}",

note = "Publisher Copyright: {\textcopyright} 2019 Wiley Periodicals, Inc.",

year = "2019",

month = apr,

doi = "10.1002/mus.26404",

language = "English (US)",

volume = "59",

pages = "404--410",

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TY - JOUR

T1 - Thymectomy may not be associated with clinical improvement in MuSK myasthenia gravis

AU - Clifford, Katherine M.

AU - Hobson-Webb, Lisa D.

AU - Benatar, Michael

AU - Burns, Ted M.

AU - Barnett, Carolina

AU - Silvestri, Nicholas J.

AU - Howard, James F.

AU - Visser, Amy

AU - Crum, Brian A.

AU - Nowak, Richard

AU - Beekman, Rachel

AU - Kumar, Aditya

AU - Ruzhansky, Katherine

AU - Chen, I. Hweii Amy

AU - Pulley, Michael T.

AU - Laboy, Shannon M.

AU - Fellman, Melissa A.

AU - Howard, Diantha B.

AU - Kolb, Noah A.

AU - Greene, Shane M.

AU - Pasnoor, Mamatha

AU - Dimachkie, Mazen M.

AU - Barohn, Richard J.

AU - Hehir, Michael K.

PY - 2019/4

Y1 - 2019/4

N2 - Introduction: A randomized trial demonstrated benefit from thymectomy in nonthymomatous acetylcholine receptor (AChR)-antibody positive myasthenia gravis (MG). Uncontrolled observational and histologic studies suggest thymectomy may not be efficacious in anti–muscle-specific kinase (MuSK)-MG. Methods: The therapeutic impact of thymectomy was evaluated from data collected for a multicenter, retrospective blinded review of rituximab in MuSK-MG. Results: Baseline characteristics were similar between thymectomy (n = 26) and nonthymectomy (n = 29) groups, including treatment with rituximab (42% vs. 45%). At last visit, 35% of thymectomy subjects reached the primary endpoint, a Myasthenia Gravis Foundation of America (MGFA) post-intervention status (PIS) score of minimal manifestations (MM) or better, compared with 55% of controls (P = 0.17). After controlling for age at onset of MG, rituximab, prednisone, and intravenous immunoglobulin/plasma exchange treatment, thymectomy was not associated with greater likelihood of favorable clinical outcome (odds ratio = 0.43, 95% confidence interval 0.12–1.53, P = 0.19). Discussion: Thymectomy was not associated with additional clinical improvement in this multicenter cohort of MuSK-MG patients. Muscle Nerve 59:404–410, 2019.

AB - Introduction: A randomized trial demonstrated benefit from thymectomy in nonthymomatous acetylcholine receptor (AChR)-antibody positive myasthenia gravis (MG). Uncontrolled observational and histologic studies suggest thymectomy may not be efficacious in anti–muscle-specific kinase (MuSK)-MG. Methods: The therapeutic impact of thymectomy was evaluated from data collected for a multicenter, retrospective blinded review of rituximab in MuSK-MG. Results: Baseline characteristics were similar between thymectomy (n = 26) and nonthymectomy (n = 29) groups, including treatment with rituximab (42% vs. 45%). At last visit, 35% of thymectomy subjects reached the primary endpoint, a Myasthenia Gravis Foundation of America (MGFA) post-intervention status (PIS) score of minimal manifestations (MM) or better, compared with 55% of controls (P = 0.17). After controlling for age at onset of MG, rituximab, prednisone, and intravenous immunoglobulin/plasma exchange treatment, thymectomy was not associated with greater likelihood of favorable clinical outcome (odds ratio = 0.43, 95% confidence interval 0.12–1.53, P = 0.19). Discussion: Thymectomy was not associated with additional clinical improvement in this multicenter cohort of MuSK-MG patients. Muscle Nerve 59:404–410, 2019.

KW - immunosuppressant therapy

KW - muscle-specific kinase (MuSK) antibodies

KW - myasthenia gravis

KW - rituximab

KW - thymectomy

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U2 - 10.1002/mus.26404

DO - 10.1002/mus.26404

M3 - Review article

C2 - 30575980

AN - SCOPUS:85059931126

SN - 0148-639X

VL - 59

SP - 404

EP - 410

JO - Muscle and Nerve

JF - Muscle and Nerve

IS - 4

ER -

Thymectomy may not be associated with clinical improvement in MuSK myasthenia gravis

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