Literature DB >> 26905909

Primary Lateral Sclerosis and Early Upper Motor Neuron Disease: Characteristics of a Cross-Sectional Population.

Christina N Fournier1, Alyssa Murphy2, Lorena Loci2, Hiroshi Mitsumoto3, Catherine Lomen-Hoerth4, Yasushi Kisanuki5, Zachary Simmons6, Nicholas J Maragakis7, April L McVey8, Tawfiq Al-Lahham9, Terry D Heiman-Patterson10, Jinsy Andrews11, Erin McDonnell2, Merit Cudkowicz2, Nazem Atassi2.   

Abstract

OBJECTIVES: The goals of this study were to characterize clinical and electrophysiologic findings of subjects with upper motor neuron disease and to explore feasibility of clinical trials in this population.
METHODS: Twenty northeast amyotrophic lateral sclerosis consortium (northeast amyotrophic lateral sclerosis) sites performed chart reviews to identify active clinical pure upper motor neuron disease patients. Patients with hereditary spastic paraplegia or meeting revised El Escorial electrodiagnostic criteria for amyotrophic lateral sclerosis were excluded. Patients were classified into 2 groups according to the presence or absence of minor electromyography (EMG) abnormalities.
RESULTS: Two hundred thirty-three subjects with upper motor neuron disease were identified; 217 had available EMG data. Normal EMGs were seen in 140 subjects, and 77 had minor denervation. Mean disease duration was 84 (±80) months for the entire cohort with no difference seen between the 2 groups. No difference was seen in clinical symptoms, disability, or outcome measures between the 2 groups after correcting for multiple comparisons.
CONCLUSIONS: Minor EMG abnormalities were not associated with phenotypic differences in a clinical upper motor neuron disease population. These findings suggest that subtle EMG abnormalities can not necessarily be used as a prognostic tool in patients with clinical upper motor neuron disease. This study also demonstrates the availability of a large number of patients with upper motor neuron diseases within the northeast amyotrophic lateral sclerosis network and suggests feasibility for conducting clinical trials in this population.

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Year:  2016        PMID: 26905909      PMCID: PMC4770823          DOI: 10.1097/CND.0000000000000102

Source DB:  PubMed          Journal:  J Clin Neuromuscul Dis        ISSN: 1522-0443


  11 in total

Review 1.  El Escorial revisited: revised criteria for the diagnosis of amyotrophic lateral sclerosis.

Authors:  B R Brooks; R G Miller; M Swash; T L Munsat
Journal:  Amyotroph Lateral Scler Other Motor Neuron Disord       Date:  2000-12

2.  The ALSFRS-R: a revised ALS functional rating scale that incorporates assessments of respiratory function. BDNF ALS Study Group (Phase III).

Authors:  J M Cedarbaum; N Stambler; E Malta; C Fuller; D Hilt; B Thurmond; A Nakanishi
Journal:  J Neurol Sci       Date:  1999-10-31       Impact factor: 3.181

3.  El Escorial World Federation of Neurology criteria for the diagnosis of amyotrophic lateral sclerosis. Subcommittee on Motor Neuron Diseases/Amyotrophic Lateral Sclerosis of the World Federation of Neurology Research Group on Neuromuscular Diseases and the El Escorial "Clinical limits of amyotrophic lateral sclerosis" workshop contributors.

Authors:  B R Brooks
Journal:  J Neurol Sci       Date:  1994-07       Impact factor: 3.181

4.  Clinical evolution of pure upper motor neuron disease/dysfunction (PUMMD).

Authors:  Emanuele D'Amico; Meredith Pasmantier; Yei-Won Lee; Louis Weimer; Hiroshi Mitsumoto
Journal:  Muscle Nerve       Date:  2012-11-21       Impact factor: 3.217

5.  Disease burden in upper motor neuron syndromes: a survey of patient perspectives.

Authors:  J A Andrews; Sabrina Paganoni; Corey Braastad; Merit Cudkowicz; Nazem Atassi
Journal:  J Clin Neuromuscul Dis       Date:  2014-12

6.  Differentiation between primary lateral sclerosis and amyotrophic lateral sclerosis: examination of symptoms and signs at disease onset and during follow-up.

Authors:  Maria Carmela Tartaglia; Ann Rowe; Karen Findlater; J B Orange; Gloria Grace; Michael J Strong
Journal:  Arch Neurol       Date:  2007-02

7.  Amyotrophic lateral sclerosis in Olmsted County, Minnesota, 1925 to 1998.

Authors:  Eric J Sorenson; Andrew P Stalker; Leonard T Kurland; Anthony J Windebank
Journal:  Neurology       Date:  2002-07-23       Impact factor: 9.910

8.  Clinical phenotypes and natural progression for motor neuron disease: analysis from an Australian database.

Authors:  Paul Talman; Andrew Forbes; Susan Mathers
Journal:  Amyotroph Lateral Scler       Date:  2009-04

9.  Clinical features that distinguish PLS, upper motor neuron-dominant ALS, and typical ALS.

Authors:  P H Gordon; B Cheng; I B Katz; H Mitsumoto; L P Rowland
Journal:  Neurology       Date:  2009-06-02       Impact factor: 9.910

10.  Age-period-cohort analysis of trends in amyotrophic lateral sclerosis in Denmark, 1970-2009.

Authors:  Ryan M Seals; Johnni Hansen; Ole Gredal; Marc G Weisskopf
Journal:  Am J Epidemiol       Date:  2013-09-24       Impact factor: 4.897

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  4 in total

1.  Better understanding the neurobiology of primary lateral sclerosis.

Authors:  P Hande Ozdinler; Mukesh Gautam; Oge Gozutok; Csaba Konrad; Giovanni Manfredi; Estela Area Gomez; Hiroshi Mitsumoto; Marcella L Erb; Zheng Tian; Georg Haase
Journal:  Amyotroph Lateral Scler Frontotemporal Degener       Date:  2020-11       Impact factor: 4.092

2.  Natural History of "Pure" Primary Lateral Sclerosis.

Authors:  Anhar Hassan; Shivam Om Mittal; William T Hu; Keith A Josephs; Eric J Sorenson; J Eric Ahlskog
Journal:  Neurology       Date:  2021-02-26       Impact factor: 9.910

3.  Primary lateral sclerosis: consensus diagnostic criteria.

Authors:  Martin R Turner; Richard J Barohn; Philippe Corcia; John K Fink; Matthew B Harms; Matthew C Kiernan; John Ravits; Vincenzo Silani; Zachary Simmons; Jeffrey Statland; Leonard H van den Berg; Hiroshi Mitsumoto
Journal:  J Neurol Neurosurg Psychiatry       Date:  2020-02-06       Impact factor: 10.154

Review 4.  Upper Motor Neuron Disorders: Primary Lateral Sclerosis, Upper Motor Neuron Dominant Amyotrophic Lateral Sclerosis, and Hereditary Spastic Paraplegia.

Authors:  Timothy Fullam; Jeffrey Statland
Journal:  Brain Sci       Date:  2021-05-11
  4 in total

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