Comorbidities and Complications of Cerebral Palsy

A special issue of Journal of Clinical Medicine (ISSN 2077-0383). This special issue belongs to the section "Clinical Neurology".

Deadline for manuscript submissions: closed (20 November 2022) | Viewed by 41090

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Special Issue Editors


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Guest Editor
1. Department of Pediatrics, The University of Melbourne, Melbourne, VIC, Australia
2. Murdoch Children’s Research Institute, Melbourne, VIC, Australia
Interests: occupational therapy; child-onset disability; participation; consumer-engaged research; longitudinal outcomes; intervention research; measurement

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Guest Editor
1. Department of Paediatrics, The University of Melbourne, Melbourne, VIC, Australia
2. Murdoch Children’s Research Institute, Melbourne, VIC, Australia
3. Department of Neurodevelopment & Disability Royal Children's Hospital, Melbourne, Melbourne, Australia
Interests: cerebral palsy; neurodisability; drooling; bioethics

Special Issue Information

Dear Colleagues,

Cerebral palsy is the most common cause of physical disability in childhood that, despite evidence of falling prevalence in some Western countries, remains a global concern. Cerebral palsy is a clinical description, rather than a discrete diagnosis. Despite the aetiological heterogeneity, it is thought that many of the associated comorbidities and complications in cerebral palsy share more similarities than differences.

The aim of this Special Issue is to provide an overview of recent advances in the treatment of the associated comorbidities and complications of cerebral palsy. Increased prescription of technology means that we are treating some comorbidities of cerebral palsy earlier or even pre-emptively. Increased life expectancy, compared with 20 years ago, means that we are also seeing even later complications of cerebral palsy. There is increased access to intensive medical care at home, non-invasive ventilation and total parenteral nutrition. There is also the rise of the parent/carer voice and expectations. As society begins to discuss mental health more openly, we are also starting to recognise the significant two-way associations between disability and mental health. Ethical consultation may be warranted when harm with treatment exceeds benefit. As we prepare children with cerebral palsy for adulthood, there is more thought and discussion around issues such as pain, mental health, quality of life and independence. Researchers in the field of cerebral palsy are encouraged to submit original articles or reviews to this Special Issue (case reports and short reviews are not accepted), so that we can build our evidence base to guide decision making.

Prof. Dr. Christine Imms
Dr. Monica S. Cooper
Guest Editors

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Keywords

  • cerebral palsy
  • family factors
  • physical health
  • mental health
  • prevention (primary and secondary)
  • interventions
  • management
  • quality of life

Published Papers (20 papers)

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Editorial

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3 pages, 165 KiB  
Editorial
Editorial Highlights from the Comorbidities and Complications of Cerebral Palsy Special Issue
by Monica S Cooper and Christine Imms
J. Clin. Med. 2023, 12(16), 5329; https://doi.org/10.3390/jcm12165329 - 16 Aug 2023
Viewed by 580
Abstract
Cerebral palsy is a life-long condition and the most common cause of physical disability in childhood [...] Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)

Research

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11 pages, 697 KiB  
Article
Examining the Role of Sublingual Atropine for the Treatment of Sialorrhea in Patients with Neurodevelopmental Disabilities: A Retrospective Review
by Kayla Durkin Petkus, Garey Noritz and Laurie Glader
J. Clin. Med. 2023, 12(16), 5238; https://doi.org/10.3390/jcm12165238 - 11 Aug 2023
Cited by 1 | Viewed by 1274
Abstract
Sialorrhea is common in children with neurodevelopmental disabilities (NDD) and is reported in >40% of children with cerebral palsy (CP). It causes a range of complications, including significant respiratory morbidity. This single-center retrospective chart review aims to document sublingual atropine (SLA) utilization to [...] Read more.
Sialorrhea is common in children with neurodevelopmental disabilities (NDD) and is reported in >40% of children with cerebral palsy (CP). It causes a range of complications, including significant respiratory morbidity. This single-center retrospective chart review aims to document sublingual atropine (SLA) utilization to guide further study in establishing its role in secretion management for children with NDD. A chart review was completed for patients with NDD ≤ 22 years of age treated with SLA at a free-standing children’s hospital between 1 January 2016 and 1 June 2021. Descriptive statistics were generated to summarize findings. In total, 190 patients were identified, of which 178 met inclusion criteria. The average starting dose for SLA was 1.5 mg/day, or 0.09 mg/kg/day when adjusted for patient weight. Eighty-nine (50%) patients were prescribed SLA first line for secretion management while 85 (48%) patients tried glycopyrrolate prior to SLA. SLA was used after salivary Botox, ablation, and/or surgery in 16 (9%) patients. This study investigates SLA as a potential pharmacologic agent to treat sialorrhea in children with NDD. We identify a range of prescribing patterns regarding dosing, schedule, and place in therapy, highlighting the need for further evidence to support and guide its safe and efficacious use. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
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12 pages, 1149 KiB  
Article
Emotion Regulation Is Associated with Anxiety, Depression and Stress in Adults with Cerebral Palsy
by Ingrid Honan, Emma Waight, Joan Bratel, Fiona Given, Nadia Badawi, Sarah McIntyre and Hayley Smithers-Sheedy
J. Clin. Med. 2023, 12(7), 2527; https://doi.org/10.3390/jcm12072527 - 28 Mar 2023
Cited by 1 | Viewed by 2186
Abstract
Emotion regulation difficulties are associated with many neurological conditions and negatively impact daily function. Yet little is known about emotion regulation in adults with cerebral palsy (CP). Our aim was to investigate emotion regulation in adults with CP and its relationship with condition-related [...] Read more.
Emotion regulation difficulties are associated with many neurological conditions and negatively impact daily function. Yet little is known about emotion regulation in adults with cerebral palsy (CP). Our aim was to investigate emotion regulation in adults with CP and its relationship with condition-related and/or socio-demographic factors. In a cross-sectional study of adults with CP, participants completed a survey containing the Difficulties in Emotion Regulation Scale (DERS), Depression Anxiety and Stress Scale-21 (DASS-21), and socio-demographic and condition-related questions. Descriptive statistics, chi-squared and Mann–Whitney tests were performed. Of the 42 adults with CP (x31.5 years, SD13.5) that were tested, 38 had within normal limits DERS total scores; however, a significantly higher proportion of participants experienced elevated scores (i.e., more difficulties with emotion regulation) than would be expected in the general population across five of the six DERs subdomains. Moderate–extremely severe depression and anxiety symptoms were reported by 33% and 60% of participants, respectively. The DERS total scores for participants with elevated depression, anxiety, and stress scores were significantly higher than the DERS totals score for those without elevated depression, anxiety, and stress scores. DERS and DASS-21 scores did not differ significantly by condition-related nor socio-demographic characteristics. In conclusion, emotion regulation difficulties were associated with elevated symptoms of depression and anxiety, which were overrepresented in the adults with CP participating in this study. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
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13 pages, 249 KiB  
Article
Associated Impairments among Children with Cerebral Palsy in Rural Bangladesh—Findings from the Bangladesh Cerebral Palsy Register
by Aditya Narayan, Mohammad Muhit, John Whitehall, Iskander Hossain, Nadia Badawi, Gulam Khandaker and Israt Jahan
J. Clin. Med. 2023, 12(4), 1597; https://doi.org/10.3390/jcm12041597 - 17 Feb 2023
Cited by 3 | Viewed by 1935
Abstract
Background: We aimed to describe the burden, severity, and underlying factors of associated impairments among children with cerebral palsy (CP) in rural Bangladesh. Methods: This study reports findings from the Bangladesh Cerebral Palsy Register—the first population-based surveillance of children with CP in any [...] Read more.
Background: We aimed to describe the burden, severity, and underlying factors of associated impairments among children with cerebral palsy (CP) in rural Bangladesh. Methods: This study reports findings from the Bangladesh Cerebral Palsy Register—the first population-based surveillance of children with CP in any LMIC, where children with confirmed CP aged < 18 years are registered by a multidisciplinary team following a standard protocol. Associated impairments were documented based on clinical assessment, available medical records, and a detailed clinical history provided by the primary caregivers. Descriptive analysis, as well as unadjusted and adjusted logistic regression, were completed using R. Results: Between January 2015 and February 2022, 3820 children with CP were registered (mean (SD) age at assessment: 7.6 (5.0) y; 39% female). Overall, 81% of children had ≥1 associated impairment; hearing: 18%, speech: 74%, intellectual: 40%, visual: 14%, epilepsy: 33%. The presence of a history of CP acquired post-neonatally and having a gross motor function classification system levels III–V significantly increased the odds of different types of associated impairments in these children. Most of the children had never received any rehabilitation services and were not enrolled in any mainstream or special education system. Conclusions: The burden of associated impairments was high among children with CP, with comparatively low receipt of rehabilitation and educational services in rural Bangladesh. Comprehensive intervention could improve their functional outcome, participation, and quality of life. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
14 pages, 1340 KiB  
Article
The Effect of Bimanual Intensive Functional Training on Somatosensory Hand Function in Children with Unilateral Spastic Cerebral Palsy: An Observational Study
by Catherine V. M. Steinbusch, Anke Defesche, Bertie van der Leij, Eugene A. A. Rameckers, Annemarie C. S. Knijnenburg, Jeroen R. J. Vermeulen and Yvonne J. M. Janssen-Potten
J. Clin. Med. 2023, 12(4), 1595; https://doi.org/10.3390/jcm12041595 - 17 Feb 2023
Cited by 1 | Viewed by 1853
Abstract
(1) Background: Next to motor impairments, children with unilateral spastic cerebral palsy (CP) often experience sensory impairments. Intensive bimanual training is well known for improving motor abilities, though its effect on sensory impairments is less known. (2) Objective: To investigate whether bimanual intensive [...] Read more.
(1) Background: Next to motor impairments, children with unilateral spastic cerebral palsy (CP) often experience sensory impairments. Intensive bimanual training is well known for improving motor abilities, though its effect on sensory impairments is less known. (2) Objective: To investigate whether bimanual intensive functional therapy without using enriched sensory materials improves somatosensory hand function. (3) Methods: A total of twenty-four participants with CP (12–17 years of age) received 80–90 h of intensive functional training aimed at improving bimanual performance in daily life. Somatosensory hand function was measured before training, directly after training, and at six months follow-up. Outcome measures were: proprioception, measured by thumb and wrist position tasks and thumb localization tasks; vibration sensation; tactile perception; and stereognosis. (4) Results: Next to improving on their individual treatment goals, after training, participants also showed significant improvements in the perception of thumb and wrist position, vibration sensation, tactile perception, and stereognosis of the more affected hand. Improvements were retained at six months follow-up. Conversely, proprioception measured by the thumb localization tasks did not improve after training. (5) Conclusions: Intensive functional bimanual training without environmental tactile enrichment may improve the somatosensory function of the more affected hand in children with unilateral spastic CP. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
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17 pages, 1512 KiB  
Article
Morphological Medial Gastrocnemius Muscle Growth in Ambulant Children with Spastic Cerebral Palsy: A Prospective Longitudinal Study
by Nathalie De Beukelaer, Ines Vandekerckhove, Ester Huyghe, Geert Molenberghs, Nicky Peeters, Britta Hanssen, Els Ortibus, Anja Van Campenhout and Kaat Desloovere
J. Clin. Med. 2023, 12(4), 1564; https://doi.org/10.3390/jcm12041564 - 16 Feb 2023
Cited by 5 | Viewed by 1749
Abstract
Only cross-sectional studies have demonstrated muscle deficits in children with spastic cerebral palsy (SCP). The impact of gross motor functional limitations on altered muscle growth remains unclear. This prospective longitudinal study modelled morphological muscle growth in 87 children with SCP (age range 6 [...] Read more.
Only cross-sectional studies have demonstrated muscle deficits in children with spastic cerebral palsy (SCP). The impact of gross motor functional limitations on altered muscle growth remains unclear. This prospective longitudinal study modelled morphological muscle growth in 87 children with SCP (age range 6 months to 11 years, Gross Motor Function Classification System [GMFCS] level I/II/III = 47/22/18). Ultrasound assessments were performed during 2-year follow-up and repeated for a minimal interval of 6 months. Three-dimensional freehand ultrasound was applied to assess medial gastrocnemius muscle volume (MV), mid-belly cross-sectional area (CSA) and muscle belly length (ML). Non-linear mixed models compared trajectories of (normalized) muscle growth between GMFCS-I and GMFCS-II&III. MV and CSA growth trajectories showed a piecewise model with two breakpoints, with the highest growth before 2 years and negative growth rates after 6–9 years. Before 2 years, children with GMFCS-II&III already showed lower growth rates compared to GMFCS-I. From 2 to 9 years, the growth rates did not differ between GMFCS levels. After 9 years, a more pronounced reduction in normalized CSA was observed in GMFCS-II&III. Different trajectories in ML growth were shown between the GMFCS level subgroups. These longitudinal trajectories highlight monitoring of SCP muscle pathology from early ages and related to motor mobility. Treatment planning and goals should stimulate muscle growth. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
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7 pages, 414 KiB  
Article
Postoperative Airway Management after Submandibular Duct Relocation in 96 Drooling Children and Adolescents
by Saskia E. Kok, Joris Lemson and Frank J. A. van den Hoogen
J. Clin. Med. 2023, 12(4), 1473; https://doi.org/10.3390/jcm12041473 - 12 Feb 2023
Viewed by 1089
Abstract
The aim of this study was to evaluate our institutions airway management and complications after submandibular duct relocation (SMDR). We analysed a historic cohort of children and adolescents who were examined at the Multidisciplinary Saliva Control Centre between March 2005 and April 2016. [...] Read more.
The aim of this study was to evaluate our institutions airway management and complications after submandibular duct relocation (SMDR). We analysed a historic cohort of children and adolescents who were examined at the Multidisciplinary Saliva Control Centre between March 2005 and April 2016. Ninety-six patients underwent SMDR for excessive drooling. We studied details of the surgical procedure, postoperative swelling and other complications. Ninety-six patients, 62 males and 34 females, were treated consecutively by SMDR. Mean age at time of surgery was 14 years and 11 months. The ASA physical status was 2 in most patients. The majority of children were diagnosed with cerebral palsy (67.7%). Postoperative swelling of the floor of the mouth or tongue was reported in 31 patients (32.3%). The swelling was mild and transient in 22 patients (22.9%) but profound swelling was seen in nine patients (9.4%). In 4.2% of the patients the airway was compromised. In general, SMDR is a well-tolerated procedure, but we should be aware of swelling of the tongue and floor of the mouth. This may lead to a prolonged period of endotracheal intubation or a need for reintubation which can be challenging. After extensive intra-oral surgery such as SMDR we strongly recommend a extended perioperative intubation and extubation after the airway is checked and secure. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
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11 pages, 531 KiB  
Article
Characteristics and Challenges of Epilepsy in Children with Cerebral Palsy—A Population-Based Study
by Ana Dos Santos Rufino, Magnus Påhlman, Ingrid Olsson and Kate Himmelmann
J. Clin. Med. 2023, 12(1), 346; https://doi.org/10.3390/jcm12010346 - 01 Jan 2023
Cited by 3 | Viewed by 2085
Abstract
The aim of this population-based study was to describe the prevalence and characteristics of epilepsy in children with cerebral palsy (CP), focusing on antiseizure medication (ASM) and seizure outcome. Findings were related to CP type, gross motor function and associated impairments. Data on [...] Read more.
The aim of this population-based study was to describe the prevalence and characteristics of epilepsy in children with cerebral palsy (CP), focusing on antiseizure medication (ASM) and seizure outcome. Findings were related to CP type, gross motor function and associated impairments. Data on all 140 children with CP born in 2003–2006 were taken from the CP register of Western Sweden. Medical records were reviewed at ages 9–12 and 13–16 years. In total 43% had a diagnosis of epilepsy. Epilepsy was more common in children with dyskinetic CP, who more often had a history of infantile spasms, continuous spike-and-wave during sleep and status epilepticus. Neonatal seizures, severe intellectual disability, severe motor disability and autism were associated with a higher risk of epilepsy. Many children were on polytherapy, and valproate was frequently used, even in girls. At age 13–16 years, 45% of the children with epilepsy were seizure free for at least one year. Onset after 2 years of age, female sex and white matter injury were associated with good seizure outcome. Despite the risk of relapse, reduction or discontinuation of ASM could be an option in selected cases. It is important to optimize ASM and to consider the possibility of epilepsy surgery. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
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16 pages, 510 KiB  
Article
Parental Coping, Representations, and Interactions with Their Infants at High Risk of Cerebral Palsy
by Silja Berg Kårstad, Åse Bjørseth, Johanna Lindstedt, Anne Synnøve Brenne, Helene Steihaug and Ann-Kristin Gunnes Elvrum
J. Clin. Med. 2023, 12(1), 277; https://doi.org/10.3390/jcm12010277 - 29 Dec 2022
Cited by 2 | Viewed by 2005
Abstract
The aim of this study is to describe parental coping, representations, and interactions during the time of inclusion in the Small Step early intervention program for infants at high risk of cerebral palsy (CP) in Norway (ClinicalTrials.gov: NCT03264339). Altogether, 11 infants (mean age [...] Read more.
The aim of this study is to describe parental coping, representations, and interactions during the time of inclusion in the Small Step early intervention program for infants at high risk of cerebral palsy (CP) in Norway (ClinicalTrials.gov: NCT03264339). Altogether, 11 infants (mean age 4.8 months, SD: 1.5) and their parents (mothers: n = 10, fathers: n = 9) were included. Parental coping was assessed using the Parenting Stress Index-Short Form (PSI-SF) and the Hospital Anxiety and Depression Scale (HADS). Parental representations and parent–infant interactions were assessed using the Working Model of the Child Interview (WMCI) and the Parent–Child Early Relational Assessment (PCERA). Parents’ PSI-SF and HADS scores were within normal range; however, 26.7% showed symptoms of stress, 52.6% showed symptoms of anxiety, and 31.6% showed symptoms of depression above the cut-off. WMCI results indicate that 73.7% of the parents had balanced representations. For PCERA, the subscale Dyadic Mutuality and Reciprocity was of concern, while two other subscales were in areas of strength and three subscales in some concern areas. There were no differences between mothers and fathers. Most of the parents had balanced representations, some had mental or stress symptoms and many were struggling with aspects of the parent–infant interaction. This knowledge could be useful when developing more family-centered interventions. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
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26 pages, 3565 KiB  
Article
Implementation of an Early Communication Intervention for Young Children with Cerebral Palsy Using Single-Subject Research Design
by Roslyn Ward, Elizabeth Barty, Neville Hennessey, Catherine Elliott and Jane Valentine
J. Clin. Med. 2023, 12(1), 232; https://doi.org/10.3390/jcm12010232 - 28 Dec 2022
Cited by 1 | Viewed by 1866
Abstract
The implementation of an intervention protocol aimed at increasing vocal complexity in three pre-linguistic children with cerebral palsy (two males, starting age 15 months, and one female, starting age 16 months) was evaluated utilising a repeated ABA case series design. The study progressed [...] Read more.
The implementation of an intervention protocol aimed at increasing vocal complexity in three pre-linguistic children with cerebral palsy (two males, starting age 15 months, and one female, starting age 16 months) was evaluated utilising a repeated ABA case series design. The study progressed until the children were 36 months of age. Weekly probes with trained and untrained items were administered across each of three intervention blocks. Successive blocks targeted more advanced protophone production and speech movement patterns, individualised for each participant. Positive treatment effects were seen for all participants in terms of a greater rate of achievement of target protophone categories and speech movement patterns. Tau coefficients for trained items demonstrated overall moderate to large AB phase contrast effect sizes, with limited evidence of generalisation to untrained items. Control items featuring protophones and speech movements not targeted for intervention showed no change across phases for any participant. Our data suggest that emerging speech-production skills in prelinguistic infants with CP can be positively influenced through a multimodal intervention focused on capitalising on early periods of plasticity when language learning is most sensitive. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
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12 pages, 548 KiB  
Article
Respiratory Health Inequities among Children and Young Adults with Cerebral Palsy in Aotearoa New Zealand: A Data Linkage Study
by Alexandra Sorhage, Samantha Keenan, Jimmy Chong, Cass Byrnes, Amanda Marie Blackmore, Anna Mackey, Timothy Hill, Dug Yeo Han and Ngaire Susan Stott
J. Clin. Med. 2022, 11(23), 6968; https://doi.org/10.3390/jcm11236968 - 25 Nov 2022
Cited by 2 | Viewed by 1787
Abstract
(1) Background: Respiratory disease is a leading cause of morbidity, mortality, and poor quality of life in children with cerebral palsy (CP). This study describes the prevalence of CP-related respiratory disease and the non-modifiable risk factors for respiratory-related hospital admissions in the Aotearoa [...] Read more.
(1) Background: Respiratory disease is a leading cause of morbidity, mortality, and poor quality of life in children with cerebral palsy (CP). This study describes the prevalence of CP-related respiratory disease and the non-modifiable risk factors for respiratory-related hospital admissions in the Aotearoa New Zealand population. (2) Methods: New Zealand Cerebral Palsy Register (NZCPR) participant data and de-identified data from the National Minimum Dataset and Pharmaceutical Dispensing Collections were linked to identify all respiratory-related hospital admissions and respiratory illness-related antibiotic exposure over 5 years in individuals with CP (0–26 years). (3) Results: Risk factors for respiratory-related hospital admissions included being classified Gross Motor Function Classification System (GMFCS) IV or V compared to GMFCS I [OR = 4.37 (2.90–6.58), p < 0.0001; OR = 11.8 (7.69–18.10), p < 0.0001, respectively,]; having ≥2 antibiotics dispensed per year [OR = 4.42 (3.01–6.48), p < 0.0001]; and being of Māori ethnicity [OR = 1.47 (1.13–1.93), p < 0.0047]. Māori experienced health inequities compared to non-Māori, with greater functional disability, and also experienced greater antibiotic dispensing than the general population. (4) Conclusion: Māori children and young adults have a higher risk of respiratory-related illness. Priority should be given to the screening for potentially modifiable risk factors for all children with CP from diagnosis onwards in a way that ensures Māori health equity. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
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6 pages, 225 KiB  
Article
Underlying Causes of Death among Adults with Cerebral Palsy
by Mark D. Peterson, Allecia M. Wilson and Edward A. Hurvitz
J. Clin. Med. 2022, 11(21), 6333; https://doi.org/10.3390/jcm11216333 - 27 Oct 2022
Cited by 2 | Viewed by 2891
Abstract
Background: Adults with cerebral palsy (CP) represent a growing population whose healthcare needs are poorly understood. The purpose of this study was to examine trends in the underlying causes of death (UCOD) among adults with CP in the United States. Methods: A national [...] Read more.
Background: Adults with cerebral palsy (CP) represent a growing population whose healthcare needs are poorly understood. The purpose of this study was to examine trends in the underlying causes of death (UCOD) among adults with CP in the United States. Methods: A national cohort was created from the Centers for Disease Control and Prevention Wide-ranging Online Data for Epidemiologic Research (WONDER) database from 1999 to 2019. The UCOD was determined using the International Statistical Classification of Diseases and Related Health Problems, Tenth Revision (ICD-10 code G80x, Infantile CP) based on death certificate adjudication. Crude and age-adjusted mortality rates (AAMRs), as well as 95% confidence intervals (CIs) were calculated for adults with CP. Results: There were 25,138 deaths where CP was listed as the UCOD between 1999–2019. There was a steady increase in the UCOD attributable to CP in both crude mortality rates and AAMRs, with the highest rates occurring in 2019. The highest co-occurring secondary causes of death were other diseases of the nervous system (e.g., epilepsy), diseases of the respiratory system (e.g., pneumonia), symptoms, signs, and abnormal clinical and laboratory findings, not elsewhere classified (e.g., dysphagia), and diseases of the circulatory system (e.g., cardiovascular disease). Conclusions: Listing the UCOD as CP should be accompanied by other mechanisms leading to mortality in this population. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
13 pages, 1027 KiB  
Article
Post-Fracture Inpatient and Outpatient Physical/Occupational Therapy and Its Association with Survival among Adults with Cerebral Palsy
by Daniel G. Whitney, Tao Xu, Daniel Whibley, Dayna Ryan, Michelle S. Caird, Edward A. Hurvitz and Heidi Haapala
J. Clin. Med. 2022, 11(19), 5561; https://doi.org/10.3390/jcm11195561 - 22 Sep 2022
Cited by 1 | Viewed by 1279
Abstract
Physical and/or occupational therapy (PT/OT) may improve post-fracture health and survival among adults with cerebral palsy (CP), but this has not been studied in the inpatient setting. The objective was to quantify the association between acute inpatient and outpatient PT/OT use with 1-year [...] Read more.
Physical and/or occupational therapy (PT/OT) may improve post-fracture health and survival among adults with cerebral palsy (CP), but this has not been studied in the inpatient setting. The objective was to quantify the association between acute inpatient and outpatient PT/OT use with 1-year mortality among adults with CP. This was a retrospective cohort study of adults with CP with an incident fragility fracture admitted to an acute care or rehabilitation facility using a random 20% Medicare fee-for-service dataset. Acute care/rehabilitation PT/OT was measured as the average PT/OT cost/day for the length of stay (LOS). Weekly exposure to outpatient PT/OT was examined up to 6 months post-fracture. Cox regression examined the adjusted association between the interaction of acute care/rehabilitation average PT/OT cost/day and LOS with 1-year mortality. A separate Cox model added time-varying outpatient PT/OT. Of 649 adults with CP, average PT/OT cost/day was associated with lower mortality rate for LOS < 17 days (HR range = 0.78–0.93), and increased mortality rate for LOS > 27 days (HR ≥ 1.08) (all, p < 0.05). After acute care/rehabilitation, 44.5% initiated outpatient PT/OT, which was associated with lower mortality rate (HR = 0.52; 95% CI = 0.27–1.01). Post-fracture inpatient and outpatient PT/OT were associated with improved 1-year survival among adults with CP admitted to acute care/rehabilitation facilities. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
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10 pages, 229 KiB  
Article
Unmet Health Needs among Young Adults with Cerebral Palsy in Ireland: A Cross-Sectional Study
by Jennifer M. Ryan, Michael Walsh, Mary Owens, Michael Byrne, Thilo Kroll, Owen Hensey, Claire Kerr, Meriel Norris, Aisling Walsh, Grace Lavelle and Jennifer Fortune
J. Clin. Med. 2022, 11(16), 4847; https://doi.org/10.3390/jcm11164847 - 18 Aug 2022
Cited by 5 | Viewed by 1834
Abstract
Data describing the unmet health needs of young adults with cerebral palsy (CP) may support the development of appropriate health services. This study aimed to describe unmet health needs among young adults with CP in Ireland and examine if these differed between young [...] Read more.
Data describing the unmet health needs of young adults with cerebral palsy (CP) may support the development of appropriate health services. This study aimed to describe unmet health needs among young adults with CP in Ireland and examine if these differed between young adults who were and were not yet discharged from children’s services. In this cross-sectional study, young adults with CP aged 16–22 years completed a questionnaire assessing unmet health needs. Logistic regression was used to examine the association between discharge status and unmet health needs. Seventy-five young adults (mean age 18.4 yr; 41% female; 60% in GMFCS levels I-III) were included in the study. Forty (53%) had been discharged from children’s services. Unmet health need, as a proportion of those with needs, was highest for speech (0.64), followed by epilepsy (0.50) and equipment, mobility, control of movement and bone or joint problems (0.39 or 0.38). After adjusting for ambulatory status, unmet health needs did not differ according to discharge status. The proportion of young adults with unmet health needs highlights the importance of taking a life-course approach to CP and providing appropriate services to people with CP regardless of age. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
15 pages, 310 KiB  
Article
Correlates of Mental Health in Adolescents and Young Adults with Cerebral Palsy: A Cross-Sectional Analysis of the MyStory Project
by Jan Willem Gorter, Darcy Fehlings, Mark A. Ferro, Andrea Gonzalez, Amanda D. Green, Sarah N. Hopmans, Dayle McCauley, Robert J. Palisano, Peter Rosenbaum, Brittany Speller and on behalf of the MyStory Study Group
J. Clin. Med. 2022, 11(11), 3060; https://doi.org/10.3390/jcm11113060 - 29 May 2022
Cited by 3 | Viewed by 3059
Abstract
Background: It is important to gain a better understanding of mental health issues in adolescents and young adults (AYA) with cerebral palsy (CP). In this cross-sectional study, we explore if demographics, social and clinical questionnaire scores, and cortisol levels in hair samples from [...] Read more.
Background: It is important to gain a better understanding of mental health issues in adolescents and young adults (AYA) with cerebral palsy (CP). In this cross-sectional study, we explore if demographics, social and clinical questionnaire scores, and cortisol levels in hair samples from AYA with CP are associated with higher scores on anxiety and/or depression questionnaires. Methods: Data from a community-based sample of 63 AYA with CP (30 females; ages 16 to 30 (median age of 25)) were analyzed. Forty-one (65%) participants (20 females) provided a hair sample. Outcomes were assessed using bivariate linear regression analyses and hierarchical regression analyses. Results: Clinical depressive and anxiety symptoms were present in 33% and 31% of participants, respectively. Family functioning, B = 9.62 (95%CI: 5.49–13.74), fatigue, B = 0.15 (95%CI: 0.05–0.25), and pain, B = 1.53 (95%CI: 0.48–2.58) were statistically significant predictors of depressive symptoms. Fatigue, B = 0.24 (95%CI: 0.12–0.35) and pain, B = 1.63 (95%CI: 0.33–2.94) were statistically significant predictors of anxiety. Cortisol levels from hair samples were not found to be associated with depressive symptoms or anxiety. Conclusions: A high prevalence of mental health problems and co-occurring physical problems was found in AYA with CP. Integrating mental support into regular care for AYA with CP is recommended. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)

Review

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18 pages, 463 KiB  
Review
Persistent Inflammation in Cerebral Palsy: Pathogenic Mediator or Comorbidity? A Scoping Review
by Madison C. B. Paton, Megan Finch-Edmondson, Russell C. Dale, Michael C. Fahey, Claudia A. Nold-Petry, Marcel F. Nold, Alexandra R. Griffin and Iona Novak
J. Clin. Med. 2022, 11(24), 7368; https://doi.org/10.3390/jcm11247368 - 12 Dec 2022
Cited by 5 | Viewed by 1933
Abstract
Research has established inflammation in the pathogenesis of brain injury and the risk of developing cerebral palsy (CP). However, it is unclear if inflammation is solely pathogenic and primarily contributes to the acute phase of injury, or if inflammation persists with consequence in [...] Read more.
Research has established inflammation in the pathogenesis of brain injury and the risk of developing cerebral palsy (CP). However, it is unclear if inflammation is solely pathogenic and primarily contributes to the acute phase of injury, or if inflammation persists with consequence in CP and may therefore be considered a comorbidity. We conducted a scoping review to identify studies that analyzed inflammatory biomarkers in CP and discuss the role of inflammation in the pathogenesis of CP and/or as a comorbidity. Twelve included studies reported a range of analytes, methods and biomarkers, including indicators of inflammatory status, immune function and genetic changes. The majority of controlled studies concluded that one or more systemic biomarkers of inflammation were significantly different in CP versus controls; most commonly serum or plasma cytokines such as tumor necrosis factor, Interleukin (IL)-6 and IL-10. In addition, differences in inflammation were noted in distinct subgroups of CP (e.g., those with varying severity). The available evidence supports the pathogenic role of inflammation and its ongoing role as a comorbidity of CP. This review shows that inflammation may persist for decades, driving functional impairment across development and into adulthood. However, inflammation is complex, thus further research will increase our understanding. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
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28 pages, 1348 KiB  
Review
Are We Getting It Right? A Scoping Review of Outcomes Reported in Cell Therapy Clinical Studies for Cerebral Palsy
by Megan Finch-Edmondson, Madison C. B. Paton, Ingrid Honan, Petra Karlsson, Candice Stephenson, Darryl Chiu, Sarah Reedman, Alexandra R. Griffin, Catherine Morgan and Iona Novak
J. Clin. Med. 2022, 11(24), 7319; https://doi.org/10.3390/jcm11247319 - 09 Dec 2022
Cited by 4 | Viewed by 1971
Abstract
Cell therapies are an emergent treatment for cerebral palsy (CP) with promising evidence demonstrating efficacy for improving gross motor function. However, families value improvements in a range of domains following intervention and the non-motor symptoms, comorbidities and complications of CP can potentially be [...] Read more.
Cell therapies are an emergent treatment for cerebral palsy (CP) with promising evidence demonstrating efficacy for improving gross motor function. However, families value improvements in a range of domains following intervention and the non-motor symptoms, comorbidities and complications of CP can potentially be targeted by cell therapies. We conducted a scoping review to describe all outcomes that have been reported in cell therapy studies for CP to date, and to examine what instruments were used to capture these. Through a systematic search we identified 54 studies comprising 2066 participants that were treated with a range of cell therapy interventions. We categorized the reported 53 unique outcome instruments and additional descriptive measures into 10 categories and 12 sub-categories. Movement and Posture was the most frequently reported outcome category, followed by Safety, however Quality of Life, and various prevalent comorbidities and complications of CP were infrequently reported. Notably, many outcome instruments used do not have evaluative properties and thus are not suitable for measuring change following intervention. We provide a number of recommendations to ensure that future trials generate high-quality outcome data that is aligned with the priorities of the CP community. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
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10 pages, 375 KiB  
Review
Perioperative Care of Children with Severe Neurological Impairment and Neuromuscular Scoliosis—A Practical Pathway to Optimize Perioperative Health and Guide Decision Making
by Giuliana C. Antolovich, Monica S. Cooper, Michael B. Johnson, Kris Lundine, Yi Yang, Katherine Frayman, Moya Vandeleur, Ingrid Sutherland, Donna Peachey, Tali Gadish, Ben Turner and Adrienne Harvey
J. Clin. Med. 2022, 11(22), 6769; https://doi.org/10.3390/jcm11226769 - 16 Nov 2022
Cited by 4 | Viewed by 1808
Abstract
Neuromuscular scoliosis is a common feature in children with severe neurological impairment (SNI), including those with severe cerebral palsy. Surgical correction of scoliosis is the mainstay of treatment. This group of patients also have associated medical complexity. The complication rates post-surgery are high, [...] Read more.
Neuromuscular scoliosis is a common feature in children with severe neurological impairment (SNI), including those with severe cerebral palsy. Surgical correction of scoliosis is the mainstay of treatment. This group of patients also have associated medical complexity. The complication rates post-surgery are high, although, for many, they are worth the risk. There are currently no published practice guidelines or care pathways for children with SNI who are undergoing scoliosis corrective surgery. In response to the high uptake of this surgery, coupled with the expected complication rates, our hospital established a perioperative clinic. The purpose of this paper is to describe our perioperative approach. This clinic has developed into a service beyond perioperative care and, with the collaborative meeting, enables shared decision-making to identify the right candidate for surgery. The process involves surgical expertise, understanding the family and child at the centre, and optimisation of medical care pre- and post-surgery. In this paper, we describe the process in a step-by-step manner. We provide clinical vignettes, as well as the proformas that we use, and we highlight the benefits of the team-based process. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
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Other

19 pages, 2950 KiB  
Perspective
Hip Surveillance and Management of Hip Displacement in Children with Cerebral Palsy: Clinical and Ethical Dilemmas
by Jason J. Howard, Kate Willoughby, Pam Thomason, Benjamin J. Shore, Kerr Graham and Erich Rutz
J. Clin. Med. 2023, 12(4), 1651; https://doi.org/10.3390/jcm12041651 - 19 Feb 2023
Cited by 10 | Viewed by 3228
Abstract
Hip displacement is the second most common musculoskeletal deformity in children with cerebral palsy. Hip surveillance programs have been implemented in many countries to detect hip displacement early when it is usually asymptomatic. The aim of hip surveillance is to monitor hip development [...] Read more.
Hip displacement is the second most common musculoskeletal deformity in children with cerebral palsy. Hip surveillance programs have been implemented in many countries to detect hip displacement early when it is usually asymptomatic. The aim of hip surveillance is to monitor hip development to offer management options to slow or reverse hip displacement, and to provide the best opportunity for good hip health at skeletal maturity. The long-term goal is to avoid the sequelae of late hip dislocation which may include pain, fixed deformity, loss of function and impaired quality of life. The focus of this review is on areas of disagreement, areas where evidence is lacking, ethical dilemmas and areas for future research. There is already broad agreement on how to conduct hip surveillance, using a combination of standardised physical examination measures and radiographic examination of the hips. The frequency is dictated by the risk of hip displacement according to the child’s ambulatory status. Management of both early and late hip displacement is more controversial and the evidence base in key areas is relatively weak. In this review, we summarise the recent literature on hip surveillance and highlight the management dilemmas and controversies. Better understanding of the causes of hip displacement may lead to interventions which target the pathophysiology of hip displacement and the pathological anatomy of the hip in children with cerebral palsy. We have identified the need for more effective and integrated management from early childhood to skeletal maturity. Areas for future research are highlighted and a range of ethical and management dilemmas are discussed. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
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16 pages, 3381 KiB  
Protocol
Co-Design of an Intervention to Increase the Participation in Leisure Activities Including Adolescents with Cerebral Palsy with GMFCS Levels IV and V: A Study Protocol
by Rocío Palomo-Carrión, Caline Cristine De Araújo Ferreira Jesus, Camila Araújo Santos Santana, Raquel Lindquist, Roselene Alencar, Helena Romay-Barrero, Elena Contell-Gonzalo, Karolinne Souza Monteiro, Elena Pinero-Pinto and Egmar Longo
J. Clin. Med. 2023, 12(1), 182; https://doi.org/10.3390/jcm12010182 - 26 Dec 2022
Cited by 2 | Viewed by 2457
Abstract
The participation of adolescents with cerebral palsy (CP) within the community is reduced compared to their peers and is a barrier to their socialization, self-determination and quality of life. Patient and Public Involvement (PPI) is a key strategy for successful interventions, especially when [...] Read more.
The participation of adolescents with cerebral palsy (CP) within the community is reduced compared to their peers and is a barrier to their socialization, self-determination and quality of life. Patient and Public Involvement (PPI) is a key strategy for successful interventions, especially when involvement of the stakeholders takes place at all stages of the research. Co-design can be crucial for success as researchers, patients with CP and their families work together to bring the necessary elements to the interventions to be designed. The objectives will be: (1) To co-design an intervention aimed at improving the participation of adolescents with significant motor disabilities within the community in partnership with adolescents with CP, families and rehabilitation professionals. (2) To assess the feasibility of the co-design process in partnership with interested parties. The study will be based on Participatory Action Research (PAR) and will be held in Spain and Brazil. In both countries, the study will be carried out remotely with nine adolescents aged 12 to 17 years with CP, Gross Motor Function Classification System (GMFCS) levels IV–V, their families and six health professionals (physiotherapists and occupational therapists). Different dialogue groups will be created to involve adolescents, families and health professionals to the research’s project. To manage their involvement in the co-design process, the Involvement Matrix (IM) will be used, and according to the IM phases, four steps will be included in the research: (1) Preparation; (2) Co-design; (3) Analysis: results of the intervention protocol and the study’s feasibility and (4) Dissemination of results. Partnering with the public to design an intervention to improve participation can bring better results compared to protocols designed only by health professionals. In addition, it will allow for knowing the needs of adolescents with CP in terms of participation within the community. The study will also explore which roles were chosen by all participants and how they felt while actively participating in the process of co-designing an intervention protocol and their own perspectives on the use of the involvement matrix. Full article
(This article belongs to the Special Issue Comorbidities and Complications of Cerebral Palsy)
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