Cognitive and motor function in long-duration PARKIN-associated parkinson disease

Roy N. Alcalay, Elise Caccappolo, Helen Mejia-Santana, Ming Xin Tang, Llency Rosado, Martha Orbe Reilly, Diana Ruiz, Elan D. Louis, Cynthia L. Comella, Martha A. Nance, Susan B. Bressman, William K. Scott, Caroline M. Tanner, Susan F. Mickel, Cheryl H. Waters, Stanley Fahn, Lucien J. Cote, Steven J. Frucht, Blair Ford, Michael RezakKevin E. Novak, Joseph H. Friedman, Ronald Pfeiffer, Laura Marsh, Bradley Hiner, Haydeh Payami, Eric Molho, Stewart A. Factor, John Nutt, Carmen Serrano, Maritza Arroyo, Ruth Ottman, Michael W. Pauciulo, William C. Nichols, Lorraine N. Clark, Karen S. Marder

Research output: Contribution to journalArticle

26 Citations (Scopus)

Abstract

IMPORTANCE: Data on the long-term cognitive outcomes of patients with PARKIN-associated Parkinson disease (PD) are unknown but may be useful when counseling these patients. OBJECTIVE: Among patients with early-onset PD of long duration, we assessed cognitive and motor performances, comparing homozygotes and compound heterozygotes who carry 2 PARKIN mutations with noncarriers. DESIGN, SETTING, AND PARTICIPANTS: Cross-sectional study of 44 participants at 17 different movement disorder centers who were in the Consortium on Risk for Early-Onset PD study with a duration of PD greater than the median duration (>14 years): 4 homozygotes and 17 compound heterozygotes (hereafter referred to as carriers) and 23 noncarriers. MAINOUTCOMESAND MEASURES: Unified Parkinson Disease Rating Scale Part III (UPDRS-III) and Clinical Dementia Rating scores and neuropsychological performance. Linear regression models were applied to assess the association between PARKIN mutation status and cognitive domain scores and UPDRS-III scores. Models were adjusted for age, education, disease duration, language, and levodopa equivalent daily dose. RESULTS: Carriers had an earlier age at onset of PD (P <.001) and were younger (P = .004) at time of examination than noncarriers. They performed better than noncarriers on the Mini-Mental State Examination (P = .010) and were more likely to receive lower scores on the Clinical Dementia Rating (P = .003). In multivariate analyses, carriers performed better than noncarriers on the UPDRS-III (P = .02) and on tests of attention (P = .03), memory (P = .03), and visuospatial (P = .02) cognitive domains. CONCLUSIONS AND RELEVANCE: In cross-sectional analyses, carriers demonstrated better cognitive and motor performance than did noncarriers with long disease duration, suggesting slower disease progression. A longitudinal follow-up study is required to confirm these findings.

Original languageEnglish (US)
Pages (from-to)62-67
Number of pages6
JournalJAMA Neurology
Volume71
Issue number1
DOIs
StatePublished - 2014

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Cognition
Parkinson Disease
Homozygote
Heterozygote
Dementia
Linear Models
Cross-Sectional Studies
Mutation
Movement Disorders
Levodopa
Disease Progression
Counseling
Language
Multivariate Analysis
Education

ASJC Scopus subject areas

  • Clinical Neurology
  • Medicine(all)

Cite this

Alcalay, R. N., Caccappolo, E., Mejia-Santana, H., Tang, M. X., Rosado, L., Reilly, M. O., ... Marder, K. S. (2014). Cognitive and motor function in long-duration PARKIN-associated parkinson disease. JAMA Neurology, 71(1), 62-67. https://doi.org/10.1001/jamaneurol.2013.4498

Cognitive and motor function in long-duration PARKIN-associated parkinson disease. / Alcalay, Roy N.; Caccappolo, Elise; Mejia-Santana, Helen; Tang, Ming Xin; Rosado, Llency; Reilly, Martha Orbe; Ruiz, Diana; Louis, Elan D.; Comella, Cynthia L.; Nance, Martha A.; Bressman, Susan B.; Scott, William K.; Tanner, Caroline M.; Mickel, Susan F.; Waters, Cheryl H.; Fahn, Stanley; Cote, Lucien J.; Frucht, Steven J.; Ford, Blair; Rezak, Michael; Novak, Kevin E.; Friedman, Joseph H.; Pfeiffer, Ronald; Marsh, Laura; Hiner, Bradley; Payami, Haydeh; Molho, Eric; Factor, Stewart A.; Nutt, John; Serrano, Carmen; Arroyo, Maritza; Ottman, Ruth; Pauciulo, Michael W.; Nichols, William C.; Clark, Lorraine N.; Marder, Karen S.

In: JAMA Neurology, Vol. 71, No. 1, 2014, p. 62-67.

Research output: Contribution to journalArticle

Alcalay, RN, Caccappolo, E, Mejia-Santana, H, Tang, MX, Rosado, L, Reilly, MO, Ruiz, D, Louis, ED, Comella, CL, Nance, MA, Bressman, SB, Scott, WK, Tanner, CM, Mickel, SF, Waters, CH, Fahn, S, Cote, LJ, Frucht, SJ, Ford, B, Rezak, M, Novak, KE, Friedman, JH, Pfeiffer, R, Marsh, L, Hiner, B, Payami, H, Molho, E, Factor, SA, Nutt, J, Serrano, C, Arroyo, M, Ottman, R, Pauciulo, MW, Nichols, WC, Clark, LN & Marder, KS 2014, 'Cognitive and motor function in long-duration PARKIN-associated parkinson disease', JAMA Neurology, vol. 71, no. 1, pp. 62-67. https://doi.org/10.1001/jamaneurol.2013.4498
Alcalay RN, Caccappolo E, Mejia-Santana H, Tang MX, Rosado L, Reilly MO et al. Cognitive and motor function in long-duration PARKIN-associated parkinson disease. JAMA Neurology. 2014;71(1):62-67. https://doi.org/10.1001/jamaneurol.2013.4498
Alcalay, Roy N. ; Caccappolo, Elise ; Mejia-Santana, Helen ; Tang, Ming Xin ; Rosado, Llency ; Reilly, Martha Orbe ; Ruiz, Diana ; Louis, Elan D. ; Comella, Cynthia L. ; Nance, Martha A. ; Bressman, Susan B. ; Scott, William K. ; Tanner, Caroline M. ; Mickel, Susan F. ; Waters, Cheryl H. ; Fahn, Stanley ; Cote, Lucien J. ; Frucht, Steven J. ; Ford, Blair ; Rezak, Michael ; Novak, Kevin E. ; Friedman, Joseph H. ; Pfeiffer, Ronald ; Marsh, Laura ; Hiner, Bradley ; Payami, Haydeh ; Molho, Eric ; Factor, Stewart A. ; Nutt, John ; Serrano, Carmen ; Arroyo, Maritza ; Ottman, Ruth ; Pauciulo, Michael W. ; Nichols, William C. ; Clark, Lorraine N. ; Marder, Karen S. / Cognitive and motor function in long-duration PARKIN-associated parkinson disease. In: JAMA Neurology. 2014 ; Vol. 71, No. 1. pp. 62-67.
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abstract = "IMPORTANCE: Data on the long-term cognitive outcomes of patients with PARKIN-associated Parkinson disease (PD) are unknown but may be useful when counseling these patients. OBJECTIVE: Among patients with early-onset PD of long duration, we assessed cognitive and motor performances, comparing homozygotes and compound heterozygotes who carry 2 PARKIN mutations with noncarriers. DESIGN, SETTING, AND PARTICIPANTS: Cross-sectional study of 44 participants at 17 different movement disorder centers who were in the Consortium on Risk for Early-Onset PD study with a duration of PD greater than the median duration (>14 years): 4 homozygotes and 17 compound heterozygotes (hereafter referred to as carriers) and 23 noncarriers. MAINOUTCOMESAND MEASURES: Unified Parkinson Disease Rating Scale Part III (UPDRS-III) and Clinical Dementia Rating scores and neuropsychological performance. Linear regression models were applied to assess the association between PARKIN mutation status and cognitive domain scores and UPDRS-III scores. Models were adjusted for age, education, disease duration, language, and levodopa equivalent daily dose. RESULTS: Carriers had an earlier age at onset of PD (P <.001) and were younger (P = .004) at time of examination than noncarriers. They performed better than noncarriers on the Mini-Mental State Examination (P = .010) and were more likely to receive lower scores on the Clinical Dementia Rating (P = .003). In multivariate analyses, carriers performed better than noncarriers on the UPDRS-III (P = .02) and on tests of attention (P = .03), memory (P = .03), and visuospatial (P = .02) cognitive domains. CONCLUSIONS AND RELEVANCE: In cross-sectional analyses, carriers demonstrated better cognitive and motor performance than did noncarriers with long disease duration, suggesting slower disease progression. A longitudinal follow-up study is required to confirm these findings.",
author = "Alcalay, {Roy N.} and Elise Caccappolo and Helen Mejia-Santana and Tang, {Ming Xin} and Llency Rosado and Reilly, {Martha Orbe} and Diana Ruiz and Louis, {Elan D.} and Comella, {Cynthia L.} and Nance, {Martha A.} and Bressman, {Susan B.} and Scott, {William K.} and Tanner, {Caroline M.} and Mickel, {Susan F.} and Waters, {Cheryl H.} and Stanley Fahn and Cote, {Lucien J.} and Frucht, {Steven J.} and Blair Ford and Michael Rezak and Novak, {Kevin E.} and Friedman, {Joseph H.} and Ronald Pfeiffer and Laura Marsh and Bradley Hiner and Haydeh Payami and Eric Molho and Factor, {Stewart A.} and John Nutt and Carmen Serrano and Maritza Arroyo and Ruth Ottman and Pauciulo, {Michael W.} and Nichols, {William C.} and Clark, {Lorraine N.} and Marder, {Karen S.}",
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T1 - Cognitive and motor function in long-duration PARKIN-associated parkinson disease

AU - Alcalay, Roy N.

AU - Caccappolo, Elise

AU - Mejia-Santana, Helen

AU - Tang, Ming Xin

AU - Rosado, Llency

AU - Reilly, Martha Orbe

AU - Ruiz, Diana

AU - Louis, Elan D.

AU - Comella, Cynthia L.

AU - Nance, Martha A.

AU - Bressman, Susan B.

AU - Scott, William K.

AU - Tanner, Caroline M.

AU - Mickel, Susan F.

AU - Waters, Cheryl H.

AU - Fahn, Stanley

AU - Cote, Lucien J.

AU - Frucht, Steven J.

AU - Ford, Blair

AU - Rezak, Michael

AU - Novak, Kevin E.

AU - Friedman, Joseph H.

AU - Pfeiffer, Ronald

AU - Marsh, Laura

AU - Hiner, Bradley

AU - Payami, Haydeh

AU - Molho, Eric

AU - Factor, Stewart A.

AU - Nutt, John

AU - Serrano, Carmen

AU - Arroyo, Maritza

AU - Ottman, Ruth

AU - Pauciulo, Michael W.

AU - Nichols, William C.

AU - Clark, Lorraine N.

AU - Marder, Karen S.

PY - 2014

Y1 - 2014

N2 - IMPORTANCE: Data on the long-term cognitive outcomes of patients with PARKIN-associated Parkinson disease (PD) are unknown but may be useful when counseling these patients. OBJECTIVE: Among patients with early-onset PD of long duration, we assessed cognitive and motor performances, comparing homozygotes and compound heterozygotes who carry 2 PARKIN mutations with noncarriers. DESIGN, SETTING, AND PARTICIPANTS: Cross-sectional study of 44 participants at 17 different movement disorder centers who were in the Consortium on Risk for Early-Onset PD study with a duration of PD greater than the median duration (>14 years): 4 homozygotes and 17 compound heterozygotes (hereafter referred to as carriers) and 23 noncarriers. MAINOUTCOMESAND MEASURES: Unified Parkinson Disease Rating Scale Part III (UPDRS-III) and Clinical Dementia Rating scores and neuropsychological performance. Linear regression models were applied to assess the association between PARKIN mutation status and cognitive domain scores and UPDRS-III scores. Models were adjusted for age, education, disease duration, language, and levodopa equivalent daily dose. RESULTS: Carriers had an earlier age at onset of PD (P <.001) and were younger (P = .004) at time of examination than noncarriers. They performed better than noncarriers on the Mini-Mental State Examination (P = .010) and were more likely to receive lower scores on the Clinical Dementia Rating (P = .003). In multivariate analyses, carriers performed better than noncarriers on the UPDRS-III (P = .02) and on tests of attention (P = .03), memory (P = .03), and visuospatial (P = .02) cognitive domains. CONCLUSIONS AND RELEVANCE: In cross-sectional analyses, carriers demonstrated better cognitive and motor performance than did noncarriers with long disease duration, suggesting slower disease progression. A longitudinal follow-up study is required to confirm these findings.

AB - IMPORTANCE: Data on the long-term cognitive outcomes of patients with PARKIN-associated Parkinson disease (PD) are unknown but may be useful when counseling these patients. OBJECTIVE: Among patients with early-onset PD of long duration, we assessed cognitive and motor performances, comparing homozygotes and compound heterozygotes who carry 2 PARKIN mutations with noncarriers. DESIGN, SETTING, AND PARTICIPANTS: Cross-sectional study of 44 participants at 17 different movement disorder centers who were in the Consortium on Risk for Early-Onset PD study with a duration of PD greater than the median duration (>14 years): 4 homozygotes and 17 compound heterozygotes (hereafter referred to as carriers) and 23 noncarriers. MAINOUTCOMESAND MEASURES: Unified Parkinson Disease Rating Scale Part III (UPDRS-III) and Clinical Dementia Rating scores and neuropsychological performance. Linear regression models were applied to assess the association between PARKIN mutation status and cognitive domain scores and UPDRS-III scores. Models were adjusted for age, education, disease duration, language, and levodopa equivalent daily dose. RESULTS: Carriers had an earlier age at onset of PD (P <.001) and were younger (P = .004) at time of examination than noncarriers. They performed better than noncarriers on the Mini-Mental State Examination (P = .010) and were more likely to receive lower scores on the Clinical Dementia Rating (P = .003). In multivariate analyses, carriers performed better than noncarriers on the UPDRS-III (P = .02) and on tests of attention (P = .03), memory (P = .03), and visuospatial (P = .02) cognitive domains. CONCLUSIONS AND RELEVANCE: In cross-sectional analyses, carriers demonstrated better cognitive and motor performance than did noncarriers with long disease duration, suggesting slower disease progression. A longitudinal follow-up study is required to confirm these findings.

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