Publications by authors named "Edward A Hurvitz"

70 Publications

Critical periods of bone health across the lifespan for individuals with cerebral palsy: Informing clinical guidelines for fracture prevention and monitoring.

Bone 2021 Sep 18;150:116009. Epub 2021 May 18.

Department of Orthopaedic Surgery, University of Michigan, Ann Arbor, MI, USA.

Background: Skeletal fragility is a major burden for individuals with cerebral palsy (CP), but little is known clinically about when to prevent fractures or monitor bone health for this population. Critical periods of bone health (CPBH) are important windows for intervention to augment bone growth or mitigate bone loss. However, CPBH from the general population may not align with the needs or timing of skeletal fragility for individuals with CP. The objective of this study was to identify discrepancies when evaluating individuals with CP using CPBH across the lifespan from the general population, and propose new CP-specific CPBH.

Methods: Data from 2016 administrative claims databases were used, including the Optum's De-identified Clinformatics® Data Mart Database and a random 20% sample of the Medicare fee-for-service database from the Centers for Medicare and Medicaid Services. Sex-stratified fracture prevalence was compared between individuals with and without CP across the lifespan starting at 2 years of age using age groups to capture important stages of development and 3-4-year age bands in adulthood (up to >80 years). Sex-specific CPBH from the general population included: rapid bone accrual, peak bone mass, menopause, and elderly.

Results: There were 23,861 individuals with CP and 9,976,161 individuals without CP. CPBH from the general population did not align with the timing of skeletal fragility for CP. For example, fractures were rare and decreased throughout the CPBH of peak bone mass for males without CP, but males with CP had a greater relative fracture risk (2.9-5.6-fold higher) and a substantially increased rate of fracture (CP-slope 14× higher than non-CP-slope). For females with CP, fracture risk was increased by 18-21 years, with additional inflection points (e.g., decade before menopause and again by 57-60 years). For males with CP, fracture risk in mid-life exhibited a pattern similar to elderly males without CP.

Conclusions: This study identified discrepancies in evaluating fracture risk for individuals with CP if using established CPBH from the general population. We therefore propose new CP- and sex-specific CPBH for fracture monitoring and prevention.
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http://dx.doi.org/10.1016/j.bone.2021.116009DOI Listing
September 2021

Psychological, cardiometabolic, musculoskeletal morbidity and multimorbidity among adults with cerebral palsy and spina bifida: a retrospective cross-sectional study.

Am J Phys Med Rehabil 2021 May 17. Epub 2021 May 17.

Department of Physical Medicine and Rehabilitation, Michigan Medicine, University of Michigan, Ann Arbor, MI, USA Institute for Healthcare Policy and Innovation, Michigan Medicine, University of Michigan, Ann Arbor, MI, USA Department of Obstetrics and Gynecology, Michigan Medicine, University of Michigan Department of Emergency Medicine, Michigan Medicine, University of Michigan Department of Surgery, Michigan Medicine, University of Michigan Department of Family Medicine, Michigan Medicine, University of Michigan.

Background: Individuals living with cerebral palsy (CP) or spina bifida (SB) are at heightened risk for a number of chronic health conditions such as secondary comorbidities, that may develop or be influenced by the disability, the presence of impairment, and/or the process of aging. However, very little is known about the prevalence and/or risk of developing secondary-comorbidities among individuals living with CP or SB throughout adulthood. The objective of this study was to compare the prevalence of psychological, cardiometabolic, musculoskeletal morbidity, and multimorbidity among adults with and without CP or SB.

Methods: Privately-insured beneficiaries were included if they had an ICD-9-CM diagnostic code for CP or SB (n = 29,841). Adults without CP or SB were also included (n = 5,384,849). Prevalence estimates of common psychological, cardiometabolic, and musculoskeletal morbidity and multimorbidity (≥2 conditions) were compared.

Results: Adults living with CP or SB had a higher prevalence of all psychological disorders and psychological multimorbidity (14.6% vs 5.4%), all cardiometabolic disorders and cardiometabolic multimorbidity (22.4% vs. 15.0%), and all musculoskeletal disorders and musculoskeletal multimorbidity (12.2% vs. 5.4%), as compared to adults without CP or SB, and differences were to a clinically meaningful extent.

Conclusions: Adults with CP or SB have a significantly higher prevalence of common psychological, cardiometabolic, and musculoskeletal morbidity and multimorbidity, as compared to adults without CP or SB. Efforts are needed to facilitate the development of improved clinical screening algorithms and early interventions to reduce risk of disease onset/progression in these higher risk populations.
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http://dx.doi.org/10.1097/PHM.0000000000001787DOI Listing
May 2021

The effect of age when initiating anti-seizure medication therapy on fragility fracture risk for children with epilepsy.

Bone 2021 Aug 5;149:115996. Epub 2021 May 5.

Department of Orthopaedic Surgery, University of Michigan, Ann Arbor, MI, USA.

Background: Anti-seizure medication (ASM) is necessary to manage epilepsy and often prescribed to children and adolescents, but can lead to iatrogenic effects, including bone fragility by altering bone metabolism. Disrupting bone metabolism during crucial developmental stages could have a lasting adverse effect on bone health. Therefore, the objective of this propensity score-matched, observational cohort study was to determine if age when initiating ASM therapy across developmental stages (from pre- to post-puberty) for individuals with epilepsy was associated with an increased risk of fragility fracture.

Methods: Data from 01/01/2011 to 12/31/2018 were extracted from Optum Clinformatics® Data Mart. Children aged 4-21 years at baseline with at least 5 years of continuous health plan enrollment were included to allow for a 1-year baseline and 4-years of follow-up. The primary group of interest included new ASM users (i.e., treatment naïve) with epilepsy. The comparison group, no ASM users without epilepsy, was matched 1:14 to new ASM users with epilepsy for demographics and baseline fracture. To provide a proxy for developmental stages, age was categorized as 4-6 (pre-puberty), 7-10 (early puberty), 11-13 (mid-puberty), 14-17 (late puberty), and 18-21 (post-puberty). Crude incidence rate (IR; per 1000 person years) and IR ratio (IRR and 95% confidence intervals [CI]) were estimated for non-trauma fracture (NTFx) for up to 4-years of follow-up.

Results: Prior to stratifying by age group, the crude NTFx IR (95% CI) of 20.6 (16.5-24.8) for new ASM users with epilepsy (n = 1205) was 34% higher (IRR = 1.34; 95% CI = 1.09-1.66) than the crude NTFx IR (95% CI) of 15.4 (14.4-16.3) for no ASM users without epilepsy. The groups exhibited a different pattern of NTFx incidence with age, with new ASM users showing a more dramatic increase and peaking at 11-13 years, then decreasing with the older age groups. The crude IR and IRR were elevated for new ASM users with epilepsy compared to no ASM users without epilepsy for each age group (10% to 55% higher), but was only statistically significant for 11-13 years (IRR = 1.55; 95% CI = 1.02-2.36).

Conclusions: Children with epilepsy initiating ASM therapy may be vulnerable to fragility fracture, especially when initiating ASM around the time of puberty. Clinicians should be aware of this age-related association and consider age-appropriate adjunct bone fragility therapies.
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http://dx.doi.org/10.1016/j.bone.2021.115996DOI Listing
August 2021

The long-term view for cerebral palsy research and care.

Authors:
Edward A Hurvitz

Dev Med Child Neurol 2021 Jul 11;63(7):765. Epub 2021 Apr 11.

Department of Physical Medicine and Rehabilitation, Michigan Medicine/University of Michigan Health, Ann Arbor, MI, USA.

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http://dx.doi.org/10.1111/dmcn.14886DOI Listing
July 2021

Timecourse of Morbidity Onset Among Adults Living With Cerebral Palsy.

Am J Prev Med 2021 Jul 13;61(1):37-43. Epub 2021 Mar 13.

Department of Physical Medicine and Rehabilitation, Michigan Medicine, University of Michigan, Ann Arbor, Michigan; Institute for Healthcare Policy & Innovation, University of Michigan, Ann Arbor, Michigan.

Introduction: Despite the greater risk of an array of morbidities, little is known about when morbidities occur for adults with cerebral palsy. The objective of this study is to determine the timecourse of morbidity risk/development for adults with cerebral palsy and the effect by patient-level factors.

Methods: Cross-sectional data from 2016 were used from a random 20% sample from the fee-for-service Medicare database. Diagnosis codes identified adults aged ≥18 years with cerebral palsy and 16 clinically relevant morbidities. Qualitative and quantitative approaches identified the age where each morbidity became exceedingly prevalent. The effect of the timecourse by sex, race, and co-occurring intellectual disabilities and epilepsy was examined. Data were sequestered and analyzed in 2020.

Results: Among 16,818 adults with cerebral palsy, the prevalence of most morbidities was already high among those aged 18-30 years, and all morbidities increased with age except liver disease and anxiety. Hypertension and diabetes exhibited a positive linear trend with age. Of the morbidities that did not exhibit a linear trend, the qualitative and quantitative approaches were consistent considering that the cardiorespiratory diseases, osteoarthritis, renal disease, and dementia became exceedingly more prevalent at age >50 years, whereas the threshold was >60 years for depression, cancer, and metastatic cancer. There were interactions with sex, race, and co-occurring intellectual disabilities and epilepsy for some of the morbidities.

Conclusions: Morbidity prevalence is already elevated early in adulthood among individuals living with cerebral palsy, with an abrupt increase by age 50 years. Preventive efforts should be adopted early in the lifespan and not later than age 50 years for adults with cerebral palsy.
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http://dx.doi.org/10.1016/j.amepre.2021.01.020DOI Listing
July 2021

The paradoxical relationship between severity of cerebral palsy and renal function in middle-aged adults: better renal function or inappropriate clinical assessment?

Disabil Rehabil 2021 Feb 26:1-7. Epub 2021 Feb 26.

Department of Physical Medicine and Rehabilitation, University of Michigan, Ann Arbor, MI, USA.

Purpose: To determine the association between severity of cerebral palsy with serum creatinine (sCr) and sCr-based equations to estimate glomerular filtration rate (eGFR), a marker of renal function.

Methods: A clinic-based sample of 30-64 year-olds with cerebral palsy was examined and stratified by motor impairment: gross motor function classification system (GMFCS) I/II ( = 79), GMFCS III ( = 78), and GMFCS IV/V ( = 137). sCr, which is influenced by muscle mass, was obtained and sCr-based eGFR was calculated using the Modification of Diet in Renal Disease (MDRD) and Chronic Kidney Disease Epidemiology Collaboration (CKD-EPI) equations.

Results: sCr was lower with increasing GMFCS. The opposite pattern was observed for eGFR: GMFCS IV/V had significantly higher eGFR derived from MDRD compared to other GMFCS groups; GMFCS III had significantly higher eGFR compared to GMFCS I/II. The pattern was similar for CKD-EPI derived eGFR.

Conclusions: According to widely used clinical assessment methods for renal function, higher severity of cerebral palsy among adults is associated with better renal function, which is incongruent with their other biological systems. This paradoxical relationship is likely driven by lower muscle rather than true renal function, and thus, sCr-based eGFR may overestimate renal function, especially for GMFCS IV/V. Further prospective studies are needed.Implications for rehabilitationCommon methods of clinical assessment may over-estimate renal function for adults with cerebral palsy (CP), potentially giving a false positive for normal renal health due to their reliance on muscle mass.This study of a clinic-based sample of middle-aged adults with CP highlights the paradoxical relationship between severity of CP and renal function, which is likely driven by methodological limitations in the presence of low muscle mass rather than actual better renal function.It is recommended that clinicians have a high suspicion of abnormal renal function and the need for a nephrology consultation, especially with changes in creatinine levels, even within the normal range.Rehabilitation for adults with CP must have a strong focus on muscle and kidney health, especially for patients with more severe forms of CP.
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http://dx.doi.org/10.1080/09638288.2021.1890841DOI Listing
February 2021

The Effect of Osteoporosis Medication on Risk Attenuation of Non-Trauma Fracture Among Adults with Cerebral Palsy: A Propensity Score-Matched Observational Study.

Clin Epidemiol 2021 12;13:91-102. Epub 2021 Feb 12.

Department of Orthopaedic Surgery, University of Michigan, Ann Arbor, MI, USA.

Purpose: The efficacy of osteoporosis medication on reducing the risk of non-trauma fracture (NTFx) among adults with cerebral palsy (CP) has not been comprehensively investigated. There are many logistical and biological factors that may reduce this efficacy, and therefore requires attention. The purpose of this propensity score-matched, observational cohort study was to determine if osteoporosis medication was associated with NTFx risk attenuation among adults with CP and compared to adults without CP.

Materials And Methods: Data from 07/01/2011 to 09/30/2015 were extracted from Optum Clinformatics Data Mart. Claims identified adults (≥18 years), CP, osteoporosis medication, pre-index NTFx (6-months), and post-index NTFx (12-months). CP without osteoporosis medication (CP) and without CP with Meds (non-CP; reflects "background" population) served as controls and were matched (6:1 ratio) to adults with CP with Meds (CP; n=306). The Meds groups were further stratified by the initiation of their medication as new users or consistent users. Changes in the prevalence of NTFx from pre- to post-index periods were examined with risk ratios (RR) and the change was compared among groups using the ratio of the RR (RRR) via difference-in-difference analysis.

Results: New users with CP had: a larger risk attenuation of any NTFx compared to CP (RRR=0.39; 95% CI=0.22-0.71), which was consistent for vertebral column/hip and lower extremities; a larger risk attenuation for NTFx of the lower extremities compared to consistent users with CP (RRR=0.22; 95% CI=0.05-0.93); and a similar risk attenuation of any NTFx compared to new users without CP (RRR=0.81; 95% CI=0.45-1.43), which was consistent for vertebral column/hip and lower extremities.

Conclusion: The findings suggest that osteoporosis medication is associated with clinically meaningful risk attenuation of NTFx, especially for new users with CP.
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http://dx.doi.org/10.2147/CLEP.S294202DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7886102PMC
February 2021

Adults with Cerebral Palsy Require Ongoing Neurologic Care: A Systematic Review.

Ann Neurol 2021 05 26;89(5):860-871. Epub 2021 Feb 26.

Department of Neurology, Division of Pediatric Neurology, Washington University School of Medicine, St Louis, MO, USA.

Cerebral palsy (CP) neurologic care and research efforts typically focus on children. However, most people with CP are adults. Adults with CP are at increased risk of new neurologic conditions, such as stroke and myelopathy, that require ongoing neurologic surveillance to distinguish them from baseline motor impairments. Neurologic factors could also contribute to the motor function decline, chronic pain, and chronic fatigue that are commonly experienced by adults with CP. Based on a systematic literature review, we suggest (1) guidelines for neurologic surveillance and neurologist referral and (2) clinical research questions regarding the evolving neurologic risks for adults with CP. ANN NEUROL 2021;89:860-871.
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http://dx.doi.org/10.1002/ana.26040DOI Listing
May 2021

Cerebral Palsy Grows Up.

Mayo Clin Proc 2021 06 12;96(6):1404-1406. Epub 2021 Jan 12.

Department of Physical Medicine and Rehabilitation, Michigan Medicine, University of Michigan, Ann Arbor, MI.

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http://dx.doi.org/10.1016/j.mayocp.2020.10.006DOI Listing
June 2021

The respiratory disease burden of non-traumatic fractures for adults with cerebral palsy.

Bone Rep 2020 Dec 27;13:100730. Epub 2020 Oct 27.

Department of Physical Medicine and Rehabilitation, University of Michigan, Ann Arbor, MI, USA.

Background: Individuals with cerebral palsy (CP) are vulnerable to non-trauma fracture (NTFx) and premature mortality due to respiratory disease (RD); however, very little is known about the contribution of NTFx to RD risk among adults with CP. The purpose of this study was to determine if NTFx is a risk factor for incident RD and if NTFx exacerbates RD risk in the adult CP population.

Methods: Data from 2011 to 2016 Optum Clinformatics® Data Mart and a random 20% sample Medicare fee-for-service were used for this retrospective cohort study. Diagnosis codes were used to identify adults (18+ years) with and without CP, NTFx, incident RD at 3-, 6-, 12-, and 24-month time points (pneumonia, chronic obstructive pulmonary disease, interstitial/pleura disease), and comorbidities. Crude incidence rates per 100 person years of RD were estimated. Cox regression estimated hazard ratios (HR and 95% confidence interval [CI]) for RD measures, comparing: (1) CP and NTFx (CP + NTFx); (2) CP without NTFx (CP w/o NTFx); (3) without CP and with NTFx (w/o CP + NTFx); and (4) without CP and without NTFx (w/o CP w/o NTFx) after adjusting for demographics and comorbidities.

Results: The crude incidence rate was elevated for CP + NTFx vs. CP w/o NTFx and w/o CP + NTFx for each RD measure. After adjustments, the HR was elevated for CP + NTFx vs. CP w/o NTFx for pneumonia and interstitial/pleura disease at all time points (all  < 0.05), but not chronic obstructive pulmonary disease (e.g., 24-month HR = 1.07; 95%CI = 0.88-1.31). The adjusted HR was elevated for CP + NTFx vs. w/o CP + NTFx for pneumonia at all time points, interstitial/pleura disease at 12- and 24-month time points, and chronic obstructive pulmonary disease at 24-months (all  < 0.05). There is evidence of a time-dependent effect of NTFx on pneumonia and interstitial/pleura disease for CP + NTFx as compared to CP w/o NTFx.

Conclusions: Study findings suggest that NTFx is a risk factor for incident RD, including pneumonia and interstitial/pleura disease, among adults with CP and that NTFx exacerbates RD risk for adults with vs. without CP.
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http://dx.doi.org/10.1016/j.bonr.2020.100730DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7645631PMC
December 2020

The mortality burden of non-trauma fracture for adults with cerebral palsy.

Bone Rep 2020 Dec 7;13:100725. Epub 2020 Oct 7.

Department of Orthopaedic Surgery, University of Michigan, 1540 E Hospital Dr., Ann Arbor, MI 48109, United States of America.

Background: Individuals with cerebral palsy (CP) manifest skeletal fragility problems early in life, are vulnerable to non-trauma fracture (NTFx), and have a high burden of premature mortality. No studies have examined the contribution of NTFx to mortality among adults with CP. The purpose of this study was to determine if NTFx is a risk factor for mortality among adults with CP and if NTFx exacerbates mortality risk compared to adults without CP.

Methods: Data from 2011 to 2016 Optum Clinformatics® Data Mart and a random 20% sample Medicare fee-for-service were used for this retrospective cohort study. Diagnosis codes were used to identify adults (18+ years) with and without CP, NTFx, and pre-NTFx comorbidities. Crude mortality rates per 100 person years were estimated. Cox regression estimated hazard ratios (HR and 95% confidence interval [CI]) for mortality, comparing: (1) CP and NTFx (CP + NTFx;  = 1777); (2) CP without NTFx (CP w/o NTFx;  = 12,933); (3) without CP and with NTFx (w/o CP + NTFx;  = 433,560); and (4) without CP and without NTFx (w/o CP w/o NTFx;  = 6.8 M) after adjusting for demographics and pre-NTFx comorbidities.

Results: The 3-, 6-, and 12-month crude mortality rates were highest among CP + NTFx (12-month mortality rate = 6.80), followed by w/o CP + NTFx (12-month mortality rate = 4.91), CP w/o NTFx (12-month mortality rate = 2.15), and w/o CP w/o NTFx (12-month mortality rate = 0.49). After adjustments, the mortality rate was elevated for CP + NTFx for all time points compared to CP w/o NTFx (e.g., 12-month HR = 1.61; 95%CI = 1.29-2.01), w/o CP + NTFx (e.g., 12-month HR = 1.49; 95%CI = 1.24-1.80), and w/o CP w/o NTFx (e.g., 12-month HR = 5.33; 95%CI = 4.42-6.44). There were site-specific effects (vertebral column, lower extremities) on 12-month mortality.

Conclusions: NTFx is associated with an increase of 12-month mortality risk among adults with CP and compared to adults without CP. Findings suggest that NTFx may be a robust risk factor for mortality among adults with CP.
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http://dx.doi.org/10.1016/j.bonr.2020.100725DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7560646PMC
December 2020

Psychological morbidity among adults with cerebral palsy and spina bifida.

Psychol Med 2021 Mar 27;51(4):694-701. Epub 2020 Jul 27.

Department of Physical Medicine and Rehabilitation, Michigan Medicine, University of Michigan, Ann Arbor, MI, USA.

Background: Very little is known about the risk of developing psychological morbidities among adults living with cerebral palsy (CP) or spina bifida (SB). The objective of this study was to compare the incidence of and adjusted hazards for psychological morbidities among adults with and without CP or SB.

Methods: Privately insured beneficiaries were included if they had an International Classification of Diseases, Ninth revision, Clinical Modification diagnostic code for CP or SB (n = 15 302). Adults without CP or SB were also included (n = 1 935 480). Incidence estimates of common psychological morbidities were compared at 4-years of enrollment. Survival models were used to quantify unadjusted and adjusted hazard ratios for incident psychological morbidities.

Results: Adults living with CP or SB had a higher 4-year incidence of any psychological morbidity (38.8% v. 24.2%) as compared to adults without CP or SB, and differences were to a clinically meaningful extent. Fully adjusted survival models demonstrated that adults with CP or SB had a greater hazard for any psychological morbidity [hazard ratio (HR): 1.60; 95% CI 1.55-1.65], and all but one psychological disorder (alcohol-related disorders), and ranged from HR: 1.32 (1.23, 1.42) for substance disorders, to HR: 4.12 (3.24, 5.25) for impulse control disorders.

Conclusions: Adults with CP or SB have a significantly higher incidence of and risk for common psychological morbidities, as compared to adults without CP or SB. Efforts are needed to facilitate the development of improved clinical screening algorithms and early interventions to reduce the risk of disease onset/progression in these higher-risk populations.
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http://dx.doi.org/10.1017/S0033291720001981DOI Listing
March 2021

Cardiometabolic Morbidity in Adults With Cerebral Palsy and Spina Bifida.

Am J Med 2020 12 17;133(12):e695-e705. Epub 2020 Jul 17.

Department of Physical Medicine and Rehabilitation.

Purpose: The purpose of this study was to compare the incidence of, and adjusted hazards for, cardiometabolic morbidities among adults with and without cerebral palsy or spina bifida.

Methods: Privately insured beneficiaries were included if they had an International Classification of Diseases, Ninth Revision, Clinical Modification (ICD-9-CM) diagnostic code for cerebral palsy or spina bifida (n = 15,302). Adults without cerebral palsy or spina bifida were also included (n = 1,935,480). Incidence estimates of common cardiometabolic morbidities were compared at 4 years of enrollment. Survival models were used to quantify unadjusted and adjusted hazard ratios (HRs) for incident cardiometabolic morbidities.

Results: Adults living with cerebral palsy or spina bifida had a higher 4-year incidence of any cardiometabolic morbidity (41.5% vs 30.6%) as compared to adults without cerebral palsy or spina bifida, and differences were to a clinically meaningful extent. Fully adjusted survival models demonstrated that adults with cerebral palsy or spina bifida had a greater hazard for any cardiometabolic morbidity (HR: 1.52; 95% confidence interval [CI]: 1.47, 1.57), and all but 1 cardiometabolic disorder (nonalcoholic fatty liver disease) and ranged from HR: 1.20 (1.15, 1.25) for hypercholesterolemia to HR: 1.86 (1.74, 1.98) for heart failure.

Conclusions: Adults with cerebral palsy or spina bifida have a significantly higher incidence of, and risk for, common cardiometabolic morbidities, as compared to adults without cerebral palsy or spina bifida. Efforts are needed to facilitate the development of improved clinical screening algorithms and early interventions to reduce risk of cardiometabolic disease onset and progression in these higher-risk populations.
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http://dx.doi.org/10.1016/j.amjmed.2020.05.032DOI Listing
December 2020

Clinical factors associated with mood affective disorders among adults with cerebral palsy.

Neurol Clin Pract 2020 Jun;10(3):206-213

Department of Physical Medicine and Rehabilitation (DGW, SAW, DW, AK, SLM, EAH, MDP), University of Michigan; University of Michigan (DGW, MDP), Institute for Healthcare Policy and Innovation; University of Michigan (DGW), University of Michigan Depression Center, Ann Arbor, MI; and Epidemiology Group (DW), University of Aberdeen, School of Medicine, Medical Sciences and Nutrition, Scotland, UK.

Objective: To determine individual and aggregated associations of cerebral palsy (CP)-related symptoms and the effect of comorbid neurodevelopmental conditions on mood (affective) disorders among adults with CP.

Methods: Cross-sectional data from 2016 were extracted from a random 20% sample of the Medicare fee-for-service database. diagnosis codes were used to identify 18- to -64-year-old beneficiaries with CP, as well as mood (affective) disorders, pain, sleep disorders, fatigue, and comorbid neurodevelopmental conditions (intellectual disabilities [ID], autism spectrum disorders [ASD], and epilepsy).

Results: Four thousand eight hundred twenty-three of the 17,212 adults with CP had mood (affective) disorders (28.0%). After adjusting for age, sex, and race, pain (odds ratio [OR] = 2.15; 99.5% confidence interval [CI] = 1.94-2.39), sleep disorders (OR = 2.43; 99.5% CI = 2.13-2.77), fatigue (OR = 1.38; 99.5% CI = 1.18-1.60), ID (OR = 1.47; 99.5% CI = 1.31-1.63), ASD (OR = 1.44; 99.5% CI = 1.16-1.80), and epilepsy (OR = 0.81; 99.5% CI = 0.73-0.91) were each associated with mood (affective) disorders. When pain, sleep disorders, and fatigue were presented as a count variable, the adjusted odds of mood (affective) disorders increased with the number of factors: 1 factor (OR = 1.99; 99.5% CI = 1.79-2.22), 2 factors (OR = 4.18; 99.5% CI = 3.58-4.89), and all 3 factors (OR = 7.38; 99.5% CI = 5.17-10.53).

Conclusions: Among young and middle-aged adults with CP, mood (affective) disorders were associated with pain, sleep disorders, and fatigue, and increasing co-occurrence of these factors further increased the likelihood of mood (affective) disorders. Further, comorbid neurodevelopmental conditions were also associated with mood (affective) disorders among adults with CP. Study findings could be used to improve screening strategies for mood (affective) disorders among adults with CP in the clinical setting.
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http://dx.doi.org/10.1212/CPJ.0000000000000721DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7292558PMC
June 2020

Effect of pain on mood affective disorders in adults with cerebral palsy.

Dev Med Child Neurol 2020 08 10;62(8):926-932. Epub 2020 May 10.

Department of Physical Medicine and Rehabilitation, University of Michigan, Ann Arbor, MI, USA.

Aim: To determine if pain is associated with 12-month incidence of mood affective disorders (MAD) in adults with cerebral palsy (CP).

Method: Data from Optum Clinformatics Data Mart (2013-2016) were used for this retrospective cohort study. Diagnostic codes were used to identify adults (≥18y) with CP, incident cases of MAD, and covariates (other neurodevelopmental conditions, sleep disorders, arthritis). Pain (any type, location) was identified between 1st October 2014 and 30th September 2015. The pain group was divided into new or consistent pain if they had a history of pain (i.e. consistent) in the 12 months before their first pain claim date. Crude incidence rates of MAD (expressed per 100 person-years) were estimated. Cox regression was used to estimate hazard ratio (95% confidence interval [CI]) of MAD after adjusting for covariates.

Results: Adults that had new pain (n=859; incidence rate=15.5) or consistent pain (n=1303; incidence rate=17.9) had greater crude incidence rate of MAD compared to adults without pain (n=3726; incidence rate=5.9). The elevated rate of MAD remained after adjusting for covariates, for new pain (hazard ratio=2.4; 95% CI=1.9-3.0) and consistent pain (hazard ratio=2.1; 95% CI=1.7-2.7).

Interpretation: Pain is associated with greater incidence of MAD in adults with CP. This association remained after accounting for potential confounding factors.

What This Paper Adds: What this paper adds Pain was associated with higher 12-month incidence of mood affective disorders (MAD). The 12-month MAD incidence was similar between new and consistent pain groups. The MAD incidence remained higher adjusting for neurodevelopmental comorbidities, sleep disorders, and arthritis.
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http://dx.doi.org/10.1111/dmcn.14559DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7955588PMC
August 2020

The mortality burden attributable to nontrauma fracture for privately insured adults with epilepsy.

Epilepsia 2020 04 27;61(4):714-724. Epub 2020 Feb 27.

Department of Physical Medicine and Rehabilitation, University of Michigan, Ann Arbor, Michigan.

Objective: Individuals with epilepsy have poor bone development and preservation throughout the lifespan and are vulnerable to nontrauma fracture (NTFx) and post-NTFx complications. However, no studies have examined the contribution of NTFx to mortality among adults with epilepsy. The objective was to determine whether NTFx is a risk factor for mortality among adults with epilepsy.

Methods: Data from 2011 to 2016 were obtained from Optum Clinformatics Data Mart, a nationwide claims database from a single private payer in the United States. Diagnosis codes were used to identify adults (≥18 years old) with epilepsy, NTFx, and covariates (demographics and pre-NTFx cardiovascular disease, respiratory disease, diabetes, chronic kidney disease, cancer). Crude mortality rate per 100 person-years was estimated. Cox regression estimated hazard ratios (HRs) and 95% confidence intervals (CIs) were determined for mortality, comparing epilepsy and NTFx (EP + NTFx; n = 11 471), epilepsy without NTFx (EP without NTFx; n = 50 384), without epilepsy and with NTFx (without EP + NTFx; n = 423 041), and without epilepsy and without NTFx (without EP without NTFx; n = 6.8 million) after adjusting for covariates.

Results: The 3-, 6-, and 12-month crude mortality rates were highest among EP + NTFx (12-month mortality rate = 8.79), followed by without EP + NTFx (12-month mortality rate = 4.80), EP without NTFx (12-month mortality rate = 3.06), and without EP without NTFx (12-month mortality rate = 0.47). After adjustments, the mortality rate was elevated for EP + NTFx for all time points compared to EP without NTFx (eg, 12-month HR = 1.70, 95% CI = 1.58-1.85), without EP + NTFx (eg, 12-month HR = 1.41, 95% CI = 1.32-1.51), and without EP without NTFx (eg, 12-month HR = 5.23, 95% CI = 4.88-5.60). Stratified analyses showed higher adjusted HRs of 12-month mortality for EP + NTFx for all NTFx sites (ie, vertebral column, hip, extremities), all age categories (young, middle-aged, older), and for both women and men.

Significance: Among adults with epilepsy and compared to adults without epilepsy, NTFx is associated with a higher 12-month mortality rate. Findings suggest that NTFx may be a robust risk factor for mortality among adults with epilepsy.
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http://dx.doi.org/10.1111/epi.16465DOI Listing
April 2020

Early-Onset Noncommunicable Disease and Multimorbidity Among Adults With Pediatric-Onset Disabilities.

Mayo Clin Proc 2020 02 3;95(2):274-282. Epub 2019 Dec 3.

Department of Physical Medicine and Rehabilitation, Michigan Medicine, University of Michigan, Ann Arbor; Institute for Healthcare Policy and Innovation, University of Michigan, Ann Arbor. Electronic address:

Objective: To determine the prevalence of major noncommunicable diseases among young adults with pediatric-onset disabilities (PoDs) compared with young adults without PoDs.

Patients And Methods: Data were obtained from the Optum Clinformatics Data Mart, a de-identified nationwide claims database of beneficiaries from a single private payer in the United States. Beneficiaries were included if they were 18 to 40 years old and had an International Classification of Diseases, Ninth Revision, Clinical Modification diagnostic code for a PoD known to originate in childhood. Diagnostic codes were used to identify high-burden noncommunicable diseases: ischemic heart disease, cerebrovascular disease, hypertensive and other cardiovascular disease, type 2 diabetes, malignant cancer, osteoporosis, mood affective disorders, chronic obstructive pulmonary disease, chronic kidney disease, and liver disease. The prevalence of noncommunicable diseases and multimorbidity (≥2 diseases) was compared between adults with (N=47,077) and without (N=2,180,250) PoDs, before and after adjusting for sociodemographic characteristics. This study was conducted between July 1, 2018, and February 1, 2019.

Results: Adults with PoDs had higher prevalences and adjusted odds of all noncommunicable diseases (odds ratio, 2.1-9.0; all P<.05) and multimorbidity (odds ratio, 3.8; 95% CI, 3.7-3.9) compared with adults without PoDs. After stratifying by the type of PoD (eg, musculoskeletal, circulatory), all PoD categories had higher prevalence of all noncommunicable diseases and multimorbidity compared with young adults without PoDs, except for ischemic heart disease and cerebrovascular disease among adults with PoDs of the genital organs.

Conclusion: Young adults with PoDs have an early onset of several noncommunicable diseases that represent major contributors to the global and national burden of disease and mortality.
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http://dx.doi.org/10.1016/j.mayocp.2019.07.010DOI Listing
February 2020

Registry-based Research in Cerebral Palsy: The Cerebral Palsy Research Network.

Phys Med Rehabil Clin N Am 2020 02 7;31(1):185-194. Epub 2019 Nov 7.

Health System Innovation and Research Division, Department of Population Health Sciences, University of Utah School of Medicine, Williams Building, Room 1N474, 295 Chipeta Way, Salt Lake City, UT 84108, USA.

Registries are a powerful tool for clinical research. Clinical registries for cerebral palsy can aid in comparative effectiveness research, especially using the practice-based evidence model. The Cerebral Palsy Research Network (CPRN) was initiated in 2014 as a patient-centered, multidisciplinary registry. The leadership group initiated a 4-stage participatory action research process: listen, reflect, plan/analyze, and take action. CPRN also joined with CP NOW, an advocacy group, to create a research agenda for cerebral palsy. With more than 20 centers and growing, CPRN hopes to generate evidence for developing best practices and measure their implementation and impact for individuals with cerebral palsy throughout North America.
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http://dx.doi.org/10.1016/j.pmr.2019.09.005DOI Listing
February 2020

Elevated fracture risk for adults with neurodevelopmental disabilities.

Bone 2020 01 24;130:115080. Epub 2019 Oct 24.

Department of Physical Medicine and Rehabilitation, University of Michigan, 325 E. Eisenhower, Ann Arbor, MI 48108, USA; Institute for Healthcare Policy and Innovation, University of Michigan, 2800 Plymouth Rd., Ann Arbor, MI 48109, USA.

Background: Fracture is a high-burden condition that accelerates unhealthful aging and represents a considerable economic burden. Adults with neurodevelopmental disabilities (NDDs) may be susceptible for fracture at younger ages compared to adults without NDDs; and yet, very little is known about the burden of fracture for these underserved populations. The purpose of this study was to determine the sex-stratified prevalence of all-cause fracture among adults with NDDs, as compared to adults without NDDs, and if comorbidity of NDDs is associated with greater risk of fracture.

Methods: Data from 2016 were extracted from Optum Clinformatics® Data Mart (private insurance) and a random 20% sample from Medicare fee-for-service (public insurance). ICD-10-CM diagnosis codes were used to identify adults with NDDs, including intellectual disabilities, autism spectrum disorders, and cerebral palsy. Age-standardized prevalence of any fracture and fracture by anatomical location was compared between adults with and without NDDs, and then for adults with 1 NDD vs. 2 and 3 NDDs.

Results: Adults with intellectual disabilities (n=69,456), autism spectrum disorders (n=21,844), and cerebral palsy (n=29,255) had a higher prevalence of any fracture compared to adults without NDDs (n=8.7 million). For women, it was 8.3%, 8.1%, and 8.5% vs. 3.5%, respectively. For men, it was 6.6%, 5.9%, and 6.7% vs. 3.0%, respectively. Women with NDDs had a higher prevalence of fracture of the head/neck, thoracic, lumbar/pelvis, upper extremities, and lower extremities compared to women without NDDs. A similar pattern was observed for men, except for no difference for lumbar/pelvis for all NDDs and thoracic for autism spectrum disorders. For women and men, increasing comorbidity of NDDs was associated with a higher prevalence of any fracture: 1 NDD (women, 7.7%; men, 5.7%); 2 NDDs (women, 9.4%; men, 7.2%); all 3 NDDs (women, 11.3%; men, 13.7%).

Conclusions: Study findings suggest that adults with NDDs have an elevated prevalence of fracture compared to adults without NDDs, with the fracture risk being higher with greater numbers of comorbid NDD conditions for most anatomical locations. Our study findings indicate a need for earlier screening and preventive services for musculoskeletal frailty for adults with NDDs.
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http://dx.doi.org/10.1016/j.bone.2019.115080DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8065344PMC
January 2020

Economic burden of paediatric-onset disabilities among young and middle-aged adults in the USA: a cohort study of privately insured beneficiaries.

BMJ Open 2019 09 3;9(9):e030490. Epub 2019 Sep 3.

Department of Physical Medicine and Rehabilitation, University of Michigan, Ann Arbor, Michigan, USA.

Objective: Individuals with paediatric-onset disabilities (PoDs) have complex healthcare needs and are susceptible to adverse health outcomes, which may impose a higher strain on healthcare resources. The burden of healthcare resource utilisation and costs attributed to the population of adults with PoDs is not clearly established. The objective here was to compare healthcare resource utilisation and costs between adults with versus without PoDs.

Design: Cohort.

Setting: Data were from the 2016 Optum Clinformatics Data Mart, a de-identified nationwide claims database of beneficiaries from a single private payer in the USA.

Participants: International Classification of Diseases, Tenth Revision, Clinical Modification diagnosis codes were used to identify beneficiaries with PoDs that were between 18 and 64 years of age.

Primary And Secondary Outcome Measures: Annual all-cause healthcare resource utilisation and total healthcare costs were compared between adults with and without PoDs before and after adjusting for sociodemographics and several costly non-communicable diseases.

Results: Adults with PoDs (n=121 446) had greater annual mean counts of service utilisation for all service types (eg, inpatient, outpatient, emergency visits) compared with adults without PoDs (n=5 415 475) before and after adjustments (all p<0.001). Adults with PoDs had greater unadjusted total standardised reimbursement costs (US$26 702 vs US$8464; mean difference=US$18 238; cost ratio (CR)=3.16; 95% CI=3.13 to 3.18) and total patient out-of-pocket costs (US$2226 vs US$1157; mean difference=US$1069; CR=1.88; 95%CI=1.86 to 1.89). After adjustments, total standardised reimbursement costs were 2.32 times higher (95% CI=2.30 to 2.34) and total patient out-of-pocket costs were 1.65 times higher (95% CI=1.64 to 1.66) compared with adults without PoDs.

Conclusion: Adults with PoDs had greater healthcare utilisation and costs, even after accounting for costly diseases. Future research is needed to identify the cost drivers for adults with PoDs.
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http://dx.doi.org/10.1136/bmjopen-2019-030490DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6731834PMC
September 2019

Prevalence of high-burden medical conditions and health care resource utilization and costs among adults with cerebral palsy.

Clin Epidemiol 2019 19;11:469-481. Epub 2019 Jun 19.

Department of Physical Medicine and Rehabilitation, University of Michigan, Ann Arbor, MI, USA.

Individuals with cerebral palsy (CP) are susceptible to early development of high-burden medical conditions, which may place a considerable strain on health care resources. However, little is known about the prevalence of high-burden medical conditions or health care resource utilization among adults with CP. The purpose of this study was to determine the prevalence of high-burden medical conditions and health care resource utilization and costs among adults with CP, as compared to adults without CP. Cross-sectional data from the 2016 Optum Clinformatics Data Mart, a de-identified nationwide claims database of beneficiaries from a single private payer in the US. ICD-10-CM diagnosis codes were used to identify all medical conditions among beneficiaries with and without CP who were between 18 and 64 years of age. Medical and outpatient pharmacy claims were used to identify annual all-cause health care resource utilization and health care costs as standardized reimbursement and patient out-of-pocket costs. Adults with CP (n=5,555) had higher prevalence and odds of all medical conditions compared to adults without CP (OR=1.3-5.8; all <0.05), except cancer (OR=1.1; 95% CI=0.9-1.3). Adults with CP had greater annual mean counts of all health care service types (eg, inpatient, emergency department) compared to adults without CP (all <0.01). Adults with CP had higher unadjusted standardized reimbursement (mean difference=$16,288; cost ratio [CR]=3.0; 95% CI=2.9-3.1) and patient out-of-pocket (mean difference=$778; CR=1.7; 95% CI=1.6-1.7) costs compared to adults without CP. After adjusting for all prevalent medical conditions, adults with CP still had higher standardized reimbursement (CR=2.5; 95% CI=2.5-2.6) and patient out-of-pocket (CR=1.8; 95% CI=1.7-1.8) costs. Adults with CP have a higher prevalence of high-burden medical conditions, health care resource utilization, and health care costs compared to adults without CP. Study findings suggest the need for earlier screening strategies and preventive medical services to quell the disease and economic burden attributable to adults with CP.
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http://dx.doi.org/10.2147/CLEP.S205839DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6592066PMC
June 2019

Prevalence of Mental Health Disorders Among Adults With Cerebral Palsy: A Cross-sectional Analysis.

Ann Intern Med 2019 09 6;171(5):328-333. Epub 2019 Aug 6.

Michigan Medicine and the Institute for Healthcare Policy and Innovation, University of Michigan, Ann Arbor, Michigan (M.D.P.).

Background: Persons with cerebral palsy (CP) have an increased risk for secondary chronic conditions during childhood, including mental health disorders. However, little is known about how these disorders affect adults with CP.

Objective: To determine the prevalence of mental health disorders among adults with CP compared with those without CP.

Design: Cross-sectional.

Setting: 2016 Optum Clinformatics Data Mart.

Patients: 8.7 million adults (including 7348 adults with CP).

Measurements: Other neurodevelopmental comorbid conditions (intellectual disabilities, autism spectrum disorders, epilepsy) and 37 mental health disorders (as 6 categories) were identified on the basis of diagnosis codes. Direct age-standardized prevalence of the mental health disorder categories was estimated by sex for adults with CP alone, adults with CP and neurodevelopmental disorders, and adults without CP.

Results: Men with CP alone had higher age-standardized prevalence than men without CP for schizophrenic disorders (2.8% [95% CI, 2.2% to 3.4%] vs. 0.7%), mood affective disorders (19.5% [CI, 18.0% to 21.0%] vs. 8.1%), anxiety disorders (19.5% [CI, 18.0% to 21.0%] vs. 11.1%), disorders of adult personality and behavior (1.2% [CI, 0.8% to 1.6%] vs. 0.3%), and alcohol- and opioid-related disorders (4.7% [CI, 3.9% to 5.5%] vs. 3.0%). The same pattern was observed for women. Compared with adults with CP alone, those with CP and neurodevelopmental disorders had similar or higher age-standardized prevalence of the 6 mental health disorder categories, except for the lower prevalence of alcohol- and opioid-related disorders in men.

Limitations: Single claims code was used to define the cohort of interest. Information on the severity of CP was not available.

Conclusion: Compared with adults without CP, those with CP have an elevated prevalence of mental health disorders, some of which may be more pronounced in patients with comorbid neurodevelopmental disorders.

Primary Funding Source: National Institute on Disability, Independent Living, and Rehabilitation Research.
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http://dx.doi.org/10.7326/M18-3420DOI Listing
September 2019

Total and regional body fat status among children and young people with cerebral palsy: A scoping review.

Clin Obes 2019 Oct 24;9(5):e12327. Epub 2019 Jun 24.

Department of Physical Medicine and Rehabilitation, University of Michigan, Ann Arbor, Michigan.

The purpose of our scoping review was to determine if children and young people with cerebral palsy (CP) have elevated total or regional body fat compared to children and young people without CP. Databases (Ovid MEDLINE, Embase Ovid, CINHAL and Scopus) were systematically searched from 1 January 1993 to 7 December 2018 in order to identify articles that compared weight status, total body fat or regional body fat (eg, abdominal) between children and young people (0-21 years) with and without CP. Extracted data included country, subject characteristics, group sample sizes and matching strategies, methods/measures for weight status/fat depot, fat depot(s) assessed and key findings. Twenty-two studies were included. Of these, 19 studies examined total body fat; the most common method was use of anthropometrics and the more common measures were body mass index and skin-fold thickness. Twelve studies examined at least one regional fat depot; the most common method was use of anthropometrics and the most common measure was skin-fold thickness. Findings were inconsistent across studies. Further, among 10 studies that examined total and regional body fat depots, 8 found differences across fat depots within the same children and young people (eg, no difference in total body fat but higher abdominal fat). This review provides a summary of inconsistent findings from published studies on body fat comparisons between children and young people with vs without CP, highlights limitations for evaluating body fat for children with CP and discusses future research directions.
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http://dx.doi.org/10.1111/cob.12327DOI Listing
October 2019

Focus on Risk Factors for Cardiometabolic Disease in Cerebral Palsy: Toward a Core Set of Outcome Measurement Instruments.

Arch Phys Med Rehabil 2019 12 23;100(12):2389-2398. Epub 2019 May 23.

Department of Rehabilitation Medicine, Erasmus University Medical Center, Rotterdam, The Netherlands; Rijndam Rehabilitation, Rotterdam, The Netherlands.

Objective: To identify existing outcome measurement instruments (OMIs) assessing risk factors for cardiometabolic disease in adolescents and adults with cerebral palsy (CP) reported on in the literature or used in the field.

Data Sources: The COnsensus-based Standards for the selection of health Measurement Instruments database of systematic reviews and 4 electronic databases (Embase, MEDLINE/Ovid, MEDLINE/Pubmed, PsychINFO) were searched up to June 19, 2017, that yielded 2594 articles. Experts in the field were consulted to identify any additional OMIs.

Study Selection: Two reviewers independently applied inclusion criteria to select eligible studies using or evaluating measurement properties of OMIs assessing 1 of 8 outcomes: cardiorespiratory endurance, body size, body composition, physical behavior, sleep, nutrition, blood pressure, and blood lipids and glucose. Studies with an experimental or observational design including ≥10 adolescents or adults with CP were included.

Data Extraction: One reviewer extracted data that were summarized for study and sample characteristics, outcomes, OMIs used, and if applicable data on measurement properties. Two reviewers rated the methodological quality and the quality of the OMIs. Feasibility for clinical practice and research was rated by experts in the field.

Data Synthesis: Ninety OMIs were identified from 56 included articles and by the experts. Seventy OMIs pertained to cardiorespiratory endurance, body size, body composition, and physical behavior, whereas only 5 were identified for sleep and nutrition. Overall synthesis revealed that there is moderate to poor evidence for good quality of OMIs in this population. Based on feasibility for clinical practice, experts agreed on a single OMI per outcome (and 2 for cardiorespiratory endurance) to be included in a core set.

Conclusion: Despite the range of available OMIs to assess risk factors for cardiometabolic disease in adolescents and adults with CP, evidence of good quality is often lacking. Nonetheless, a preliminary core set of 9 OMIs was systematically developed.
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http://dx.doi.org/10.1016/j.apmr.2019.04.012DOI Listing
December 2019

Multimorbidity risk assessment in adolescents and adults with cerebral palsy: a protocol for establishing a core outcome set for clinical research and practice.

Trials 2019 Mar 19;20(1):176. Epub 2019 Mar 19.

Department of Pediatrics, McMaster University, Institute for Applied Health Sciences, 1400 Main Street West, Hamilton, ON, L8S 1C7, Canada.

Background: Estimates of multimorbidity, defined as the presence of at least two chronic conditions, some of which attributable to modifiable behaviours, are high in adults with cerebral palsy (CP). An assessment protocol evaluating multimorbidity risk is needed in order to develop and evaluate effective interventions to optimize lifelong health in individuals with CP. The aim of this protocol paper is to describe the development of a core outcome set (COS) for assessing multimorbidity risk in adolescents and adults with CP, to be used in clinic and research.

Methods: The expert consortium will first define the target population and outcomes to be measured. Through a process of literature review and an international Delphi survey with expert clinicians and researchers, we will then determine which outcome measurement instruments (OMIs) can best measure those outcomes. The resulting OMIs will be used in a feasibility study with adolescents and adults with CP from an international clinical research network. Finally, a face-to-face stakeholder meeting with adolescents and adults with CP, their families/caregivers and researchers and clinicians who are experts in CP, will be organized to reach final agreement on the COS.

Discussion: This COS will guide clinicians and researchers in assessing multimorbidity risk in adolescents and adults with CP. The inclusion of experts and individuals with CP from international locations for establishing the COS lends strong support to its generalizability. Evidence of its feasibility and approval from all stakeholders will enable implementation in clinical practice, and guide future research using the COS in individuals with CP.
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http://dx.doi.org/10.1186/s13063-019-3265-zDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6425572PMC
March 2019

Adults with Cerebral Palsy have Higher Prevalence of Fracture Compared with Adults Without Cerebral Palsy Independent of Osteoporosis and Cardiometabolic Diseases.

J Bone Miner Res 2019 07 6;34(7):1240-1247. Epub 2019 Mar 6.

Department of Physical Medicine and Rehabilitation, Michigan Medicine, University of Michigan, Ann Arbor, MI, USA.

Individuals with cerebral palsy (CP) have an increased risk of fracture throughout their lifespan based on an underdeveloped musculoskeletal system, excess body fat, diminished mechanical loading, and early development of noncommunicable diseases. However, the epidemiology of fracture among adults with CP is unknown. The purpose of this cross-sectional study was to determine the prevalence of fracture among a large sample of privately insured adults with CP, as compared with adults without CP. Data were from the Optum Clinformatics Data Mart (Eden Prairie, MN, USA), a deidentified nationwide claims database of beneficiaries from a single private payer. Diagnostic codes were used to identify 18- to 64-year-old beneficiaries with and without CP and any fracture that consisted of osteoporotic pathological fracture as well as any type of fracture of the head/neck, thoracic, lumbar/pelvic, upper extremity, and lower extremity regions. The prevalence of any fracture was compared between adults with (n = 5,555) and without (n = 5.5 million) CP. Multivariable logistic regression was performed with all-cause fracture as the outcome and CP group as the primary exposure. Adults with CP had a higher prevalence of all-cause fracture (6.3% and 2.7%, respectively) and fracture of the head/neck, thoracic, lumbar/pelvic, upper extremity, and lower extremity regions compared with adults without CP (all p < 0.01). After adjusting for sociodemographic and socioeconomic variables, adults with CP had higher odds of all-cause fracture compared with adults without CP (OR 2.5; 95% CI, 2.2 to 2.7). After further adjusting for cardiometabolic diseases, adults with CP had higher odds of all-cause fracture compared with adults without CP (OR 2.2; 95% CI, 2.0 to 2.5). After further adjusting for osteoporosis, adults with CP still had higher odds of all-cause fracture compared with adults without CP (OR 2.0; 95% CI, 1.8 to 2.2). These findings suggest that young and middle-aged adults with CP have an elevated prevalence of all-cause fracture compared with adults without CP, which was present even after accounting for cardiometabolic diseases and osteoporosis. © 2019 American Society for Bone and Mineral Research.
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http://dx.doi.org/10.1002/jbmr.3694DOI Listing
July 2019

Cardiometabolic disease, depressive symptoms, and sleep disorders in middle-aged adults with functional disabilities: NHANES 2007-2014.

Disabil Rehabil 2020 07 17;42(15):2186-2191. Epub 2019 Jan 17.

Department of Physical Medicine and Rehabilitation, University of Michigan, Ann Arbor, MI, USA.

This study examined whether depressive symptoms and sleep disorders modified the association between functional disabilities and cardiometabolic disease profiles in middle-aged adults (40-64 years). Participants came from the 2007-2014 NHANES. Information regarding cardiometabolic diseases, demographics, depressive symptoms, and sleep disorders were obtained. Logistic regression analyses were performed with group as the exposure and cardiometabolic diseases as the response. Adults with moderate ( = 550) and severe ( = 556) functional disabilities had a higher prevalence of cardiometabolic diseases, depressive symptoms, and sleep disorders compared to adults without functional disabilities ( = 3765;  < 0.05). After adjusting for demographics, the odds of cardiovascular disease and diabetes were higher in adults with severe functional disabilities (OR: 1.47 and 1.76,  < 0.05), but not in adults with moderate functional disabilities (OR: 1.21 and 1.22,  > 0.05). With further adjustment for depressive symptoms and sleep disorders, the odds of cardiovascular disease (OR: 1.47) and diabetes (OR: 1.76) remained increased ( < 0.05) in adults with severe functional disabilities. By middle-age, adults with functional disabilities have an elevated prevalence of cardiometabolic diseases, depressive symptoms, and sleep disorders compared to adults without functional disabilities. The elevated cardiometabolic disease profiles are present in adults with severe functional disabilities even after adjusting depressive symptoms and sleep disorders.IMPLICATIONS FOR REHABILITATIONIn the elderly population, cardiometabolic diseases, depression, and sleep disorders are prevalent conditions and are often co-morbid.In a nationally representative sample of middle-aged adults, study findings found that those with severe functional disabilities had an elevated cardiometabolic disease prevalence compared to adults without functional disabilities, even after accounting for the higher prevalence of depressive symptoms and sleep disorders.Earlier screening for cardiometabolic diseases, depression, and sleep disorders in adults with functional disabilities, or those who are at risk for developing functional disabilities, are warranted.Interventions pertaining to physical, pharmacological, or care coordination focused on improving cardiometabolic disease profiles among adults with functional disabilities are needed.
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http://dx.doi.org/10.1080/09638288.2018.1555720DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6640075PMC
July 2020

The contribution of neurologic disorders to the national prevalence of depression and anxiety problems among children and adolescents.

Ann Epidemiol 2019 01 15;29:81-84.e2. Epub 2018 Nov 15.

Department of Physical Medicine and Rehabilitation, University of Michigan, Ann Arbor.

Purpose: Depression and anxiety are growing global public health issues and affect millions of children and adolescents in the United States. Although individuals with neurologic disorders (NDs) are at increased risk of adverse mental health disorders, they represent a minority of the population. The purpose of this study was to characterize the national prevalence of depression and anxiety problems in children and adolescents by the presence of various NDs.

Methods: Parent-reported data from the 2016 National Survey of Children's Health were analyzed in children and adolescents with and without NDs aged 6-17 years.

Results: The prevalence of depression and anxiety problems varied by the type of ND (0%-18.5% and 2.8%-62.5%, respectively). In the combined group of children and adolescents with NDs (weighted estimate: 1,998,654), the prevalence of depression and anxiety problems was 15.3% and 37.9%, respectively, whereas in children and adolescents without NDs (weighted estimate: 47,644,055), the prevalence was 3.4% and 7.3%, respectively. Children and adolescents with NDs represented 4.0% of the total sample, but 15.7% and 17.7% of the overall sample with depression and anxiety problems, respectively.

Conclusions: Children and adolescents with NDs contribute to a considerable portion of the overall prevalence of depression and anxiety problems despite only representing 4% of the population.
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http://dx.doi.org/10.1016/j.annepidem.2018.11.003DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6344250PMC
January 2019

Setting a patient-centered research agenda for cerebral palsy: a participatory action research initiative.

Dev Med Child Neurol 2018 12 21;60(12):1278-1284. Epub 2018 Aug 21.

CP NOW, Greenville, SC, USA.

Aim: To establish a patient-centered research agenda for cerebral palsy (CP).

Method: We engaged a large cross-section of the extended community of people living with CP and those providing healthcare to people with CP ('the community') in an educational series and collaborative survey platform to establish an initial list of prioritized research ideas. After online workshops, a facilitated Delphi process was used to select the 20 highest priorities. Select participants attended an in-person workshop to provide comment and work toward consensus of research priorities.

Results: A research agenda for CP was developed by the community, which included consumers, clinicians, and researchers interested in advancing the established research agenda. The results included the top 16 research concepts produced by the process to shape and steward the research agenda, and an engaged cross-section of the community.

Interpretation: It has been shown that proactively engaging consumers with clinical researchers may provide more meaningful research for the community. This study suggests that future research should have more focus on interventions and outcomes across the lifespan with increased emphasis on the following outcome measures: function, quality of life, and participation.

What This Paper Adds: A patient-centered research agenda for cerebral palsy was established. Comparative effectiveness of interventions, physical activity, and understanding ageing were leading themes. Longitudinal studies across the lifespan, clinical spectrum, and ages were highly ranked. Participants reported high value for participation outcomes. Participants reported great appreciation for the engagement between consumers and clinician researchers.
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http://dx.doi.org/10.1111/dmcn.13984DOI Listing
December 2018

Age trajectories of musculoskeletal morbidities in adults with cerebral palsy.

Bone 2018 09 5;114:285-291. Epub 2018 Jul 5.

Department of Physical Medicine and Rehabilitation, Michigan Medicine, University of Michigan, 325 E. Eisenhower, Ann Arbor, MI 48108, United States. Electronic address:

Background: Individuals with cerebral palsy (CP) are at an increased risk for age-related morbidities due to functional impairments, maladapted growth, and altered body composition. While musculoskeletal (MSK) deficits are present in children, little is understood about MSK morbidity throughout the lifespan in those with CP. The purpose of this study was to examine the age-related trajectories of MSK morbidity and multimorbidity throughout adulthood in those with CP.

Methods: A clinic-based sample of adults with CP (n = 1395; ≥18 years) was examined to determine prevalence of MSK morbidities at the University of Michigan Medical Center. Logistic regression was used to determine the effects of age on individual MSK morbidities and multimorbidity (i.e., ≥2 morbidities) after adjusting for sex, race, weight, and smoking.

Results: With the 18-30 year age group as the reference, the adjusted odds of osteopenia was lower in the 41-50 and >50 year age groups, the odds of osteoporosis and rheumatoid arthritis was higher in 41-50 and >50 year age groups, and the odds of osteoarthritis was higher in 31-40, 41-50, and >50 year age groups. The adjusted odds of MSK multimorbidity increased substantially with increasing age for 31-40 year olds (OR: 1.919; 95% CI 1.05-3.52), 41-50 year olds (OR: 4.30; 95% CI 2.40-7.69), and >50 year olds (OR: 6.05; 95% CI 3.56-10.27).

Conclusions: Adults with CP are at high risk for MSK morbidities across all ages. Future studies are needed to examine the global aging trajectories of MSK health among adults with CP. Study findings highlight the importance of maximizing MSK accretion, and developing programs to assist individuals with CP and their caregivers to maintain MSK mass and function throughout the lifespan.
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http://dx.doi.org/10.1016/j.bone.2018.07.002DOI Listing
September 2018