case study child with cerebral palsy

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CHAPTER 19:  Case Study: Cerebral Palsy

Donna Cech, PT, DHS, PCS

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Introduction.

• Examination: Age 6 Years
• Evaluation, Diagnosis, and Prognosis Including Plan of Care
• Intervention
• Termination of Episode of Care
• Examination: Age 13 Years
• Kayla: 20 Years of Age
• Interventions
• Recommended Readings
• Full Chapter
• Supplementary Content

This case study focuses on the physical therapy management of Kayla, a young woman with spastic, diplegic cerebral palsy (CP). Kayla is now 20 years old and a sophomore in college. She was born prematurely and has received physical therapy services in a variety of settings since infancy. She has been followed for early intervention, early childhood, school-based, outpatient, and home health physical therapy services. At this time she does not regularly see a physical therapist, but does continue with occasional sessions to monitor adaptive equipment and to address episodes of foot pain or back pain. Kayla walks in her home/dormitory settings and on campus using bilateral forearm crutches. For longer distances, she uses a motorized cart.

Children and young adults with CP are reportedly less socially and physically active than their peers without a physical disability ( Shikako-Thomas, Majnemer, Law, & Lach, 2008 ; Engel-Yeger, Jarus, Anaby, & Law, 2009 ; Maher, Williams, Olds, & Lane, 2007 ). Individuals with CP frequently present with impairments of range of motion (ROM), soft tissue mobility, strength, coordination, and balance, resulting in motor control difficulties. CP implies damage to the immature cortex, involving the sensorimotor system. Associated problems with vision, seizures, perception, and cognition may be seen if areas of the cortex associated with these functions are also damaged. Although the cortical lesion is nonprogressive, as the infant grows and strives to become more independent, functional limitations become more apparent, as do restrictions in activities and community participation. Secondary impairments in body structures and function, such as ROM limitations, disuse atrophy, and impaired aerobic capacity, may further limit functional motor skills and ability for activities and participation. Multiple episodes of physical therapy management are frequently warranted as the child attempts more complex functional skills and as the risk for secondary impairments increases. The goal of physical therapy intervention for children and young adults with CP is to maximize the individual's ability to participate in age-appropriate activities within the home, school, and community settings.

Children with CP present with a variety of functional abilities, reflecting the location and severity of their original neurological insult. Distribution of motor involvement varies and may include hemiplegia, diplegia, or quadriplegia. The degree to which the neurological insult impacts motor ability and function also varies. The Gross Motor Function Classification System (GMFCS) provides a mechanism to classify these children, based on their gross motor abilities and limitations ( Palisano, Rosenbaum, Bartlett, & Livingston, 2008 ; Palisano et al., 1997 ). Based on Kayla's ability to ambulate with an assistive device and need to use power mobility for community mobility, she would be classified as functioning at the GMFCS level III through elementary and high school.

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Client background:

Tom is a 3 year old boy, born at 28 weeks. He has a diagnosis of evolving dyskinetic Cerebral Palsy, GMFCS V. Tom has a history of seizures.

Pia met Tom while teaching a therapist course about the Key to CP approach. Tom was a demo child, meaning he only spent about an hour with Pia.

Tom was not receiving direct Physical Therapy at home and he did not have any positioning equipment at the time. He was spending his days held by caregivers or on the floor.

Tom’s journey

See how doing the right thing, at the right time, in the right order helped set the stage for Tom to develop play and communication skills during an hour therapy session at Key to CP in this case study, or continue reading below.

Goals before treatment:

Family goals for Tom were to achieve better trunk and head control, to gain strength, and to achieve a level of independence.

It was quickly clear that Tom need support of his trunk in order to improve his head and trunk control for sitting and standing. At Key to CP we often use trunk orthoses to help the child gain upright control. For Tom, a TheraTogs garment was the obvious choice.

As soon as Tom was fitted with TheraTogs there was an immediate improvement in his head and trunk control. He became much more animated and was easily interacting with his parents and with me. He gained a whole new perspective of the world in just the 15 minutes it took to fit him with TheraTogs.

Tom’s parents were surprised to see the changes and they immediately ordered TheraTogs for him. They were also able to try a corner seat and a low table and Tom was happy and interactive.

What does this case teach us? If a child with poor trunk and head control is given the right trunk support, not only does their body control improve, but also their ability to interact with their environment. For Tom, TheraTogs brought his trunk muscles into mid-range alignment (where they are strongest) and the compression gave him sensory input. This tool allowed Tom to better experience his body in relation to his environment. 

It also teaches us that when children have to struggle less to maintain body control, they can focus on communication and learning more.

Tom is now able to develop communication and play skills, and he can participate in activities with his family. 

And it took less than an hour to bring about this transformation.

Doing the RIGHT thing

Adding TheraTogs and appropriate seating, and most importantly, abundant parent coaching

At the RIGHT time

Giving Tom an opportunity to PARTICIPATE and create positive neuroplastic changes during the first window of abundant brain growth and development

In the RIGHT order

Alignment, Awareness, Activation and Strength

Tom feels much more stable in TheraTogs. We have noticed lately how his head control is coming along and he is really looking up and engaging with everyone, especially his sister. He has been a lot more vocal too. And he is being much more aggressive with telling me he is hungry by sticking his tongue out. He is so content in his new chair that I almost cannot believe it. We feel energized and grateful to have found you. Tom's mother

A child kicks a virtual ball in one of the augmented reality games as a friend looks on. Photo used with permission from Augment Therapy.

Can Augmented Reality Improve Exercise for Children With Cerebral Palsy?

Exercise plays a key role in helping children with cerebral palsy to improve or maintain their mobility, including the ability to walk. But research has shown that many of these kids don’t get the physical activity they need.

Now, a new pilot study at Children’s Hospital Los Angeles is testing whether an augmented reality exergaming program can help these children better adhere to home therapy.

Principal Investigator Melissa Bent, MD , a pediatric orthopedist in the Jackie and Gene Autry Orthopedic Center at Children’s Hospital Los Angeles, presented results from the trial’s first phase at the 2024 Association for Clinical and Translational Science meeting, held April 3-5 in Las Vegas.

“Issues with access and usability often prevent children with cerebral palsy from participating in exercise,” Dr. Bent says. “The hope is that augmented reality will help improve that access and be more engaging for kids.”

Flying in a virtual world

The study is a partnership with Augment Therapy, which develops gamified exercise programs for pediatric rehabilitation. In 2021, the startup was one of 13 companies selected for the inaugural KidsX Pediatric Digital Health Accelerator, which is anchored by a consortium of children’s hospitals, including CHLA.

The randomized trial, which launched this spring, will enroll 30 children with cerebral palsy between the ages of 5 and 10. All children must be able to walk in most settings and be classified at Levels I or II on the Gross Motor Function Classification System (GMFCS) scale.

Participants will be randomized into two groups for a three-month at-home exercise program. One group will exercise with the augmented reality games, and the other group will be assigned to a standard home exercise program.

Unlike with virtual reality, the games do not use any headsets or wearables. Instead, children see a digital overlay of themselves in a virtual 3D world on the screen. An iPad tracks children’s movements as they play the game, controlling their avatar character on the screen with their body.

For example, in one game, children navigate an obstacle course—which requires them to bend down, squat, jump and more. In another game, children must make certain arm movements and maintain their balance as they make their avatar fly on screen.

“We don’t give them specific exercises,” Dr. Bent says. “We just put them in the game.”

Participants in both groups will log their time and answer a survey. The app also tracks the time the child spends with the games.

Early findings

At the conference, Dr. Bent presented findings from the study’s preliminary phase, which enrolled six children under the same criteria and took place last summer.

The goal of that phase was to gather user-experience data in the clinic to inform the randomized trial. Although the sample size was small, the team found that the games were well-accepted, with 83% of participants reporting that the avatar matched their child’s movements and the game was easy for their child to play.

All participants also expressed interest in enrolling in the randomized at-home trial.

If results from the trial are positive, Dr. Bent hopes to eventually launch a larger clinical trial and test the app with children who have more severe motor impairments.

“Cerebral palsy is the most common motor disability in childhood, so finding ways to move the needle in terms of engagement with rehabilitation is important,” she says. “We want to see if gamification can help us do that.”

Additional study authors were Abigail Padilla BS; Susan A. Rethlefsen, PT, DPT; Alison Hanson, PT, DPT; Eva Ciccodicola, PT, DPT; and Tishya AL Wren, PhD.

Learn more about cerebral palsy care at Children’s Hospital Los Angeles.

Related content.

What Are the Barriers to Access to Pediatric Gait Analysis?

Fracture Risk Is Low After Femoral Osteotomy in Children With Cerebral Palsy

Biomechanics Study Aims to Help Prevent Youth Sports Injuries

Power mobility and socialization in preschool: follow-up case study of a child with cerebral palsy

Affiliation.

• 1 Infant Motor Behavior Laboratory, Department of Physical Therapy, University of Delaware, Newark, Delaware 19716, USA. [email protected]
• PMID: 22090084
• PMCID: PMC3266169
• DOI: 10.1097/PEP.0b013e318235266a

Purpose: Our previous study found it feasible for a preschooler with cerebral palsy (CP) to use a power mobility device in his classroom but noted a lack of typical socialization. The purpose of this follow-up study was to determine the feasibility of providing mobility and socialization training for this child.

Methods: Will, a 3-year-old with CP, 1 comparison peer, 2 preschool teachers, and 2 therapists were filmed daily during a training and posttraining phase. Adult-directed training was provided in the classroom by therapists and teachers during the training phase. Mobility and socialization measures were coded from video.

Outcomes: During training, Will demonstrated greater socialization but less mobility than the comparison peer. Posttraining, Will socialized less but was more mobile, though less mobile than the comparison peer.

Discussion: Short-term, adult-directed power mobility and socialization training appear feasible for the preschool classroom. Important issues regarding socialization and power mobility are discussed.

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Adaptive Standing for Contracture Management in Cerebral Palsy

Determinants of Contracture in Cerebral Palsy

Muscles in cerebral palsy are notably reduced in volume, and although the tendons may present as longer, the overall muscle is often shorter in length. Further differences at the cellular level contribute toward muscles that are not only smaller and weaker but are stiffer with more tightness and less extensibility than typical muscle (Handsfield 2022). These muscle deficits can result in a cycle of inactivity: as children with CP participate in less physical activity, they have decreased opportunity for mechanical loading, which in turn results in further muscle weakness and decreased muscle flexibility.

Structural abnormalities frequently increase with age and are evident beyond the growth years into adulthood (Ágústsson 2019). Children with cerebral palsy do not have any skeletal distortion at birth: their muscles and joints appear typical. However, during development, we observe the widely recognized combination of knee and hip flexion contractures, with progressively worsening pelvic obliquity and hip rotation. These contribute to the lower extremity pattern known as wind-swept deformity, along with hip dislocation and scoliosis (Sato, 2022). These bodily distortions are more evident in children with postural asymmetry and decreased motor function (Nordmark 2009, Casey 2022, Casey 2022).

Knee contracture occurs in children at all GMFCS levels (Cloodt 2021, Cloodt 2002), with knee flexion contracture being of primary concern. There is an association between the severity of knee flexion contracture and reduced functional mobility, including decreased ability for standing and transfer (Pantzar-Castilla 2021). Recently, a scoliosis risk score has determined limited knee extension to be one of four predictors for scoliosis (Pettersson 2020). Knee contracture is notably associated with higher GMFCS levels, increasing age, and hamstring muscle shortness. Muscle spasticity appears to have a smaller effect on contracture development (Cloodt 2018).

Although this topic is beyond the scope of this article, it is interesting to consider that perhaps the crouched gait pattern contributes to knee flexion contracture, more so than knee flexion contracture contributes to crouched gait. Factors such as muscle weakness, postural stability, and balance have an influence on crouched gait. In stance, knee extensor weakness contributes to knee flexion, whereas in the swing phase of gait, passive knee flexion range-of-motion modulates the knee pattern. As for muscle spasticity, it does have a recognized role in knee flexion during gait: knee extensor hypertonia is the greatest contributor to knee flexion in both the stance and swing phases. (Manikowska 2022.)

While we recognize that muscle shortening occurs in cerebral palsy (Cloodt 2021, Cloodt 2022) and contributes to deformity and a cycle of inactivity, the mechanisms of the structural changes that result in muscle contracture are not fully understood. Additional research is needed to determine effectiveness of interventions that to date have included passive manual stretching, serial casting, orthoses, and the use of standing devices. A Cochrane review published in 2010 with an abridged republication in 2017, concludes that stretch does not have clinically important effects on joint mobility (Katalinic 2010, Harvey 2017). Earlier publications stated that evidence is inconclusive to either support or refute stretching for preventing contractures (Wiart 2008, Craig 2016, Eldridge 2016). Therefore, passive manual stretching in isolation is not considered an effective intervention for contracture management (Novak 2020).

Adaptive Standing for Contracture Management

Published systematic reviews, however, offer a positive outlook on the potential of supported standing to either prevent or ameliorate contractures (Paleg 2013, Occhipintti 2018, McLean 2023). And the following experimental studies included in these reviews describe positive outcomes with children placed in adaptive standers.

In a 2009 study, five non-mobile children were placed in standing for one hour, five days a week, for six weeks (Gibson 2009). This was followed by six weeks of no standing. These phases were repeated. The hamstring muscles lengthened (reducing knee flexion contracture) during the standing phase, and shortened during the non-standing phase.

A 2020 study compared static standing to dynamic movement in the upright position (Tornberg, 2020). Twenty non-ambulatory children between five and seven years of age were randomly assigned to either static standing or dynamic standing intervention for a minimum of thirty minutes daily for four months. After a two-week washout period, the children were placed in the other device for thirty minutes daily for four more months. Both interventions improved range of motion in hip abduction, hip external rotation, and hip extension. Interestingly, the dynamic movement opportunity also increased the range of movement in the opposite directions - ie, flexion, adduction, and internal rotation. This may be less relevant for contracture but certainly is interesting to see.

A longitudinal retrospective case-control study took measurements of children’s hip abduction and knee extension (Martinsson 2021). It is interesting to notice that the children began the supported standing at a fairly young age, ranging from a little over one year old to eleven years old, and data was collected over seven years. Children stood for ten hours per week, and the study groups stood in fifteen to thirty degrees of hip abduction. Children in the study groups did not develop any contractures. This 2021 contribution to research strongly indicates that adaptive standing in abduction can prevent contractures.

Further, a preliminary randomized control trial with ten children, published in 2022, was set up in the United Kingdom as a feasibility study in preparation for a future larger RCT (Rapson 2022). This twelve month study recruited children with CP at GMFCS levels III, IV, and V. The researchers determined the baseline average standing time of the children, and then doubled their standing time for the intervention group. The participants used their existing standing frames and orthotics. The average daily standing time of the intervention group was fifty-eight minutes daily on weekdays and forty-nine minutes daily on average overall. The researchers measured ankle, knee, and hip range of motion. The children who spent more time standing showed statistically significant improvement in knee and hip range, whereas the children who continued their usual standing showed statistically significant improvement in knee and ankle range. Both groups showed increased knee ROM with the knee extension position in standing.

Results from an individual case study published in 2020 are also promising. A sixteen year-old young man, GMFCS Level V, was placed in a stander that accommodated his hip and knee flexion contractures. The average standing frequency was three times a week for approximately one hour per session. Over fifteen months, his knee extension and hip extension were increased intentionally and gradually every week. At the same time, the overall angle of the stander was gradually moved into a more upright position.

Table 1 shows that the subject’s hip extension increased bilaterally, and his right knee extension also increased. Only the left knee flexion remained essentially the same. Since this individual is sixteen years old, his contractures may be long standing. Since this an individual case study it does not hold a lot of weight in terms of data. However, the results do effectively support the concept of the use of adaptive standing for improving range of motion.

Table 1 shows the improvement in range of motion measurements. 

Early Intervention and Standing

It is exciting to consider the possible results if we could begin this intervention earlier. In Paleg and Livingstone’s 2022 evidence-informed clinical perspectives on postural management, they encourage “supported standing in ten to fifteen degrees hip abduction bilaterally (twenty to thirty degrees total) at least one hour daily as an important change of position from sitting or lying” beginning at nine months of age. (Paleg 2022).

Similarly, Morgan’s 2021 international clinical practice guideline based on systematic reviews, suggests regular use of standing equipment for positioning as part of an active intervention program. This is recommended particularly for children with or at high risk of CP, at the age when weight-bearing in standing would begin in children who are non-weight bearing (Morgan 2021).

Cerebral palsy affects numerous individuals worldwide, many of whom experience range of motion limitations that negatively impact function. Recent research strengthens the available evidence supporting the use of adaptive standing for contracture management. With regular standing opportunity at any age, joint range of motion can be maintained and potentially improved. And with adaptive standing in early intervention, particularly in the abduction position, the development of contracture may be avoided completely, enabling children with cerebral palsy to experience better musculoskeletal health, function, and quality of life.

• Ágústsson A, Sveinsson T, Pope P, Rodby-Bousquet E. Preferred posture in lying and its association with scoliosis and windswept hips in adults with cerebral palsy.  Disabil Rehabil . 2019;41(26):3198-3202. doi:10.1080/09638288.2018.1492032
• Capati V, Covert SY, Paleg G. Stander Use for an Adolescent with Cerebral Palsy at GMFCS Level with Hip and Knee Contractures.  Assist Technol . 2020;32(6):335-341. doi:10.1080/10400435.2019.1579268
• Casey J, Agustsson A, Rosenblad A, Rodby-Bousquet E. Relationship between scoliosis, windswept hips and contractures with pain and asymmetries in sitting and supine in 2450 children with cerebral palsy.  Disabil Rehabil . 2022;44(22):6738-6743. doi:10.1080/09638288.2021.1971308
• Casey J, Rosenblad A, Rodby-Bousquet E. Postural asymmetries, pain, and ability to change position of children with cerebral palsy in sitting and supine: a cross-sectional study.  Disabil Rehabil . 2022;44(11):2363-2371. doi:10.1080/09638288.2020.1834628
• Cloodt E, Lindgren A, Lauge-Pedersen H, Rodby-Bousquet E. Sequence of flexion contracture development in the lower limb: a longitudinal analysis of 1,071 children with cerebral palsy.  BMC Musculoskelet Disord . 2022;23(1):629.. doi:10.1186/s12891-022-05548-7
• Cloodt E, Rosenblad A, Rodby-Bousquet E. Demographic and modifiable factors associated with knee contracture in children with cerebral palsy.  Dev Med Child Neurol . 2018;60(4):391-396. doi:10.1111/dmcn.13659
• Cloodt E, Wagner P, Lauge-Pedersen H, Rodby-Bousquet E. Knee and foot contracture occur earliest in children with cerebral palsy: a longitudinal analysis of 2,693 children.  Acta Orthop . 2021;92(2):222-227. doi:10.1080/17453674.2020.1848154
• Craig J, Hilderman C, Wilson G, Misovic R. Effectiveness of Stretch Interventions for Children With Neuromuscular Disabilities: Evidence-Based Recommendations.  Pediatr Phys Ther . 2016;28(3):262-275. doi:10.1097/PEP.0000000000000269
• Eldridge F, Lavin N. How effective is stretching in maintaining range of movement for children with cerebral palsy? A critical review.  Int J Ther Rehabil.  2016; 23 (8):386–395. http://dx.doi.org/10.12968/ijtr.2016.23.8.386
• Gibson SK, Sprod JA, Maher CA. The use of standing frames for contracture management for nonmobile children with cerebral palsy.  Int J Rehabil Res . 2009;32(4):316-323. doi:10.1097/MRR.0b013e32831e4501
• Handsfield GG, Williams S, Khuu S, Lichtwark G, Stott NS. Muscle architecture, growth, and biological Remodelling in cerebral palsy: a narrative review.  BMC Musculoskelet Disord . 2022;23(1):233. doi:10.1186/s12891-022-05110-5
• Harvey LA, Katalinic OM, Herbert RD, Moseley AM, Lannin NA, Schurr K. Stretch for the treatment and prevention of contracture: an abridged republication of a Cochrane Systematic Review.  J Physiother . 2017;63(2):67-75. doi:10.1016/j.jphys.2017.02.014
• Katalinic OM, Harvey LA, Herbert RD, Moseley AM, Lannin NA, Schurr K. Stretch for the treatment and prevention of contractures.  Cochrane Database Syst Rev . 2010;(9):CD007455. doi:10.1002/14651858.CD007455.pub2
• Manikowska F, Brazevič S, Jóźwiak M, Lebiedowska MK. Contribution of Different Impairments to Restricted Knee Flexion during Gait in Individuals with Cerebral Palsy.  J Pers Med . 2022;12(10):1568. doi:10.3390/jpm12101568
• Martinsson C, Himmelmann K. Abducted Standing in Children With Cerebral Palsy: Effects on Hip Development After 7 Years.  Pediatr Phys Ther . 2021;33(2):101-107. doi:10.1097/PEP.0000000000000789
• McLean LJ, Paleg GS, Livingstone RW. Supported-standing interventions for children and young adults with non-ambulant cerebral palsy: A scoping review. Dev Med Child Neurol. 2023;65(6):754-772. doi: 10.1111/dmcn.15435.
• Morgan C, Fetters L, Adde L, et al. Early Intervention for Children Aged 0 to 2 Years With or at High Risk of Cerebral Palsy: International Clinical Practice Guideline Based on Systematic Reviews.  JAMA Pediatr . 2021;175(8):846-858. doi:10.1001/jamapediatrics.2021.0878
• Nordmark E, Hägglund G, Lauge-Pedersen H, Wagner P, Westbom L. Development of lower limb range of motion from early childhood to adolescence in cerebral palsy: a population-based study [published correction appears in BMC Med. 2010;8:49].  BMC Med . 2009;7:65. Published 2009 Oct 28. doi:10.1186/1741-7015-7-65
• Novak I, Morgan C, Fahey M, et al. State of the Evidence Traffic Lights 2019: Systematic Review of Interventions for Preventing and Treating Children with Cerebral Palsy. Curr Neurol Neurosci Rep. 2020 Feb 21;20(2):3. doi: 10.1007/s11910-020-1022-z.
• Occhipintti A, Mendoza SM. Efficacy of passive standing programmes in children with cerebral palsy: a systematic review. Fisioterapia. 2018;40(3):153–164. https://doi.org/10.1016/j.ft.2018.02.004
• Paleg G, Livingstone R. Evidence-informed clinical perspectives on postural management for hip health in children and adults with non-ambulant cerebral palsy. J Pediatr Rehabil Med. 2022;15(1):39-48. doi: 10.3233/PRM-220002. PMID: 35275575.
• Paleg GS, Smith BA, Glickman LB. Systematic review and evidence-based clinical recommendations for dosing of pediatric supported standing programs.  Pediatr Phys Ther . 2013;25(3):232-247. doi:10.1097/PEP.0b013e318299d5e7
• Pantzar-Castilla EHS, Wretenberg P, Riad J. Knee flexion contracture impacts functional mobility in children with cerebral palsy with various degree of involvement: a cross-sectional register study of 2,838 individuals [published correction appears in Acta Orthop. 2021 Aug;92(4):500].  Acta Orthop . 2021;92(4):472-478. doi:10.1080/17453674.2021.1912941
• Pettersson K, Wagner P, Rodby-Bousquet E. Development of a risk score for scoliosis in children with cerebral palsy.  Acta Orthop . 2020;91(2):203-208. doi:10.1080/17453674.2020.1711621
• Rapson R, King T, Morris C, et al. Effect of different durations of using a standing frame on the rate of hip migration in children with moderate to severe cerebral palsy: a feasibility study for a randomised controlled trial.  Physiotherapy . 2022;116:42-49. doi:10.1016/j.physio.2022.01.001
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SYSTEMATIC REVIEW article

Diagnosis and therapies for patients with cerebral palsy over the past 30 years: a bibliometric analysis.

• 1 Department of Outpatient, Hangzhou Children's Hospital, Hangzhou Normal University, Hangzhou, Zhejiang, China
• 2 Department of Radiology, Zhejiang Hospital of Integrated Traditional Chinese and Western Medicine, Hangzhou, Zhejiang, China
• 3 Department of Neurosurgery, Zhejiang Hospital of Integrated Traditional Chinese and Western Medicine, Hangzhou, Zhejiang, China
• 4 Department of Neurosurgery, People's Hospital of Haimen District, Nantong, Jiangsu, China
• 5 Department of Neurosurgery, Hangzhou Children's Hospital, Hangzhou Normal University, Hangzhou, Zhejiang, China

Background: Currently, the incidence of cerebral palsy is high in newborns. However, the current methods for diagnosing and treating patients with cerebral palsy are complex and poorly targeted. Moreover, these studies lack the support of bibliometric analysis results.

Objective: Our study focused on a bibliometric analysis of published papers on the diagnosis and treatment of patients with cerebral palsy. This study identified the primary authors, institutions, and countries involved in analyzing the status and trends of research on the diagnosis and treatment of patients with cerebral palsy. Additionally, the study also involved screening pathways related to cerebral palsy.

Methods: The PubMed database was searched for publications on the diagnosis and treatment of patients with cerebral palsy between 1990 and 2023. R v4.2.2 and VOSviewer v1.6.18 software tools were utilized to perform bibliometric analysis and visualization.

Results: There were 1,965 publications on cerebral palsy diagnosis and 5,418 articles on the qualified treatment strategies, and the annual number of publications also increased. The United States dominated in this field of research. Gregory Y.H. Lip and Patrizio Lancellotti published the most number of papers. The Cleveland Clinic published the most number of papers in the field. According to the analysis of the co-occurrence of keywords, we found that the main research directions were age, sex, disease diagnosis, and treatment. Newly emerging research has focused mainly on heart failure, which is related to valvular heart disease.

Conclusion: The findings presented in this study offer valuable insights into ongoing research and potential future directions pertaining to cerebral palsy. These insights can assist researchers in identifying suitable collaborators and enhancing their investigations aimed at identifying the underlying molecular mechanisms associated with cerebral palsy, encompassing its etiology, preventive measures, and therapeutic interventions.

Introduction

Cerebral palsy (CP) is not a clear, separate disease classification but rather an umbrella term that includes miscellaneous signs and symptoms that change with age. Cerebral palsy is diagnosed primarily through motor function and posture disorders and typically develops in early childhood and persists throughout life. These disorders are not progressive but change with age ( 1 , 2 ). Motor dysfunction is a fundamental manifestation of cerebral palsy and frequently co-occurs with additional impairments encompassing sensory, perceptual, cognitive, communicative, and behavioral disorders; epilepsy; and secondary musculoskeletal complications ( 3 ). Cerebral palsy occurs in 2–3 infants out of 1,000 live births ( 4 ). There are multiple causes of brain damage that affect movement, posture, and balance. Motor disorders associated with cerebral palsy can be classified as spasms, motor disorders, ataxia, or mixed/other disorders ( 5 ). The symptoms of cerebral palsy include movement disorders, hip dislocations, balance difficulties, and hand dysfunction. In cases of cerebral palsy without a clear cause, magnetic resonance imaging may help diagnose brain damage when there is no clinical diagnosis. Once cerebral palsy is diagnosed, instruments such as the hair motor function classification system can be used to assess its severity and treatment response. The treatment of motor disorders associated with cerebral palsy includes intramuscular botulinum toxin A, medications, selective dorsal rhizotomy, and physiotherapy could be used to treat a wide range of muscle disorders. Patients with cerebral palsy often experience problems that are not related to movement and that need to be addressed in adulthood; these problems include cognitive impairment, seizures, pressure sores, osteoporosis, behavioral or emotional problems, and speech and hearing impairment ( 6 ).

The visual analysis software used for literature analysis included R v4.2.2 and VOSviewer, which play important roles in analyzing the current state of scientific research, detecting disciplinary frontiers, and selecting research directions. Bibliometrics can provide us with information on the most influential factors (including countries, institutions, and authors) in the field we want to study through relevant publications. In a manner, this approach could provide researchers with potentially favorable directions for their research. Currently, there is no research summarizing the treatment and diagnosis of cerebral palsy.

The identification of the relationships between the molecular and pathological levels of pathways and between the diagnosis and therapies used in patients with cerebral palsy can provide a basis for understanding the pathogenesis of cerebral palsy to some extent. However, there have been few bibliometric studies on the diagnosis/therapies of patients with cerebral palsy. Based on our bibliometric analysis, we generated data on cerebral palsy to provide insights for researchers who seek to discover new topics and directions.

Materials and methods

Data selection.

We screened the PubMed database for the period from 1 January 1990 to 20 January 2023 to identify publications related to the diagnosis and treatment of patients with cerebral palsy. We considered only English-language publications. The title and abstract were reviewed and screened by two independent reviewers (Lili Jiang and Song Zhang), and disagreements were resolved.

Data analysis and visualization

Key information (e.g., title, author, country/region, institution, keywords, and the year of publication) was derived from our included articles that met the inclusion criteria. The above variables were processed and visualized using Python v3.10.8, R v4.2.2, and VOSviewer v1.6.18.

VOSviewer was applied to perform the network analysis of the authors and frequent keywords. The minimum number of occurrences of a keyword was seven, which was the parameter of VOSviewer. A country collaboration map and various chart drawings based on the bibliometrix R package were constructed.

Pathway enrichment analysis

We downloaded canonical pathway gene sets based on the Reactome pathway database from the GSEA database ( 7 ). The pathway enrichment analysis of the GSE183021 ( 8 ) dataset was performed using the GSVA R package ( 9 ). The differential gene expression analysis of pathway scores was performed based on the limma R package ( 10 ).

General information

A total of 7,383 publications were identified from PubMed. Of these, 1,965 publications were based on diagnosis, and 5,418 publications were based own treatment. We created two boxplots ( Figure 1 ) to represent the number and ratio of annual publications over the last 33 years, which indicated the development trend of related research in this field. This field continues to attract the attention and interest of researchers, as evidenced by the growing number of publications. Research on the promotion of different diseases has shown differences in the associations between diagnosis and treatment.

Figure 1 . The number of annual publications relating to research on the diagnosis and treatment of cerebral palsy from 1990 to 2023. (A) The number of studies related to cerebral palsy diagnosis from 1990 to 2023. (B) The number of studies related to cerebral palsy treatment from 1990 to 2023. (C) A comparison of the annual journal publications related to cerebral palsy diagnosis and treatment.

Compared to diagnostic research, the treatment of cerebral palsy received more attention from 1990 to 2023. In summary, our results showed that the diagnosis and treatment of patients with cerebral palsy have gained widespread attention.

Distribution of authors

The top 10 authors were involved in more than 10 papers on the diagnosis and treatment of patients with cerebral palsy, most of which were published in Europe and Australia ( Tables 1 , 2 ). The bar chart shows that most of the studies originated from the United States and were mainly single-country studies ( Figure 2 ). The author collaboration network visualization ( Figure 3 ) was performed on VOSviewer.

Table 1 . The top 10 authors published articles on the diagnosis of cerebral palsy.

Table 2 . The top 10 authors published articles on the therapy for cerebral palsy.

Figure 2 . Boxplot of countries engaged in research on the diagnosis (A) and treatment (B) of cerebral palsy.

Figure 3 . The network of authors who contributed to the research on the diagnosis (A) and treatment (B) of cerebral palsy. In the network, author contributions are reflected by node size. The connection strength is reflected by the thickness of the line.

Distribution of countries/territories and institutions

The top 10 institutions had over 937 ( Figure 4A ) and 1,105 ( Figure 4B ) articles on the diagnosis and treatment of patients with cerebral palsy, respectively. According to the country collaboration map, we found that the research was mostly distributed in Europe and was closely related to many regions ( Figure 5 ).

Figure 4 . The treemap of institutions engaged in research on the diagnosis (A) and treatment (B) of cerebral palsy.

Figure 5 . A country collaboration map of the research on the diagnosis (A) and treatment (B) of cerebral palsy.

Analysis of keywords and research direction analysis

The results of keyword analysis not only help us understand the topic of the article but also help us understand the hot spots in a research area. The top 20 keywords with the highest frequency are listed in Tables 3 , 4 . As shown in Figure 6 , we used VOSviewer to display the network of keywords.

Table 3 . The top 20 keywords for the diagnosis of cerebral palsy.

Table 4 . The top 20 keywords related to therapy for cerebral palsy.

Figure 6 . The network visualization of keywords related to the diagnosis (A) and treatment (B) of cerebral palsy. A keyword's weight is represented by the size of its circle. The distance between two circles indicates the relatedness between the two circles. Shorter distances are associated with stronger relatedness. The different colors of the circles represent the cluster classes.

The heat map did not reveal relatively important results in the field of diagnosis, but, during treatment, we found that the individual may be more important in the clinical treatment of cerebral palsy ( Figure 7 ).

Figure 7 . Thematic map of the research on the diagnosis (A) and treatment (B) of cerebral palsy. In the thematic map, the horizontal axis represents the centrality and the vertical axis represents the density. Motor themes: these themes are important and well-developed. Niche themes: these themes are very specialized/niche themes that are well-developed but are not important for the current field. Emerging or declining themes: these themes are not well-developed, may just emerge, or may soon disappear. Basic themes: these themes are very important to the field and have not achieved good development.

We downloaded the Reactome pathway dataset, subjected it to enrichment analysis to obtain the fractional matrix of the pathways, and conducted differential analysis to obtain the differential heatmap of cerebral palsy-related pathways ( Figure 8 ).

Figure 8 . Pathway enrichment analysis of the expression matrix in cerebral palsy patients.

We identified 3,089 pathways enriched in cerebral palsy, including 56 upregulated pathways (logFC > 0, P < 0.01) and 10 downregulated pathways (logFC < 0, P < 0.01).

In relation to publishing patterns, our analysis reveals a steady upward trajectory in the number of publications pertaining to this domain since 1990. This trend suggests that both fields will continue to garner significant interest from researchers in the coming years. Moreover, the authors' collaborative network analysis offers a valuable tool for researchers to foster collaborative associations, as it enables the identification of influential research groups and potential partners.

Most of the authors listed in Tables 1 , 2 are located in Europe/Australia, and the relationships between European scholars are much closer than those between scholars in other regions. As shown in Figure 2 , many authors have made contributions to this field, but their cooperation needs to be strengthened. Figure 4 shows that most well-published institutions are located in the United States. For the current research, the active cooperation and exchange of ideas between researchers/research institutions in various countries will be beneficial to the development of this field.

Study hotspots

We have not mined important research directions from the diagnosis section, but we have found that the basic themes for the diagnosis section are similar to the motor themes for the treatment section. The basic criteria for diagnoses included studies on humans (1,653), female (1,047), male (944), child, preschool (623), infant (515), infant or newborn (445) factors; retrospective studies (307); information on risk factor (200); pregnancy (176); and follow-up studies (133).

The motor themes for the treatment included studies on humans (4,474), child (2,790), female (2,641), male (2,483), child, preschools (1,614), adolescent (1,537), treatment outcome (1,160), adult (687), retrospective studies (591), and cerebral palsy/complications (514). However, to some extent, these results do not explain some of the details, thus we combined them with keyword analysis.

Research has shown that cerebral palsy or “high risk of cerebral palsy” can be accurately predicted before the corrected age of 6 months, and detecting cerebral palsy before the corrected age, including magnetic resonance imaging (MRI), has the best predictive effectiveness, with a sensitivity of 86–89% ( 11 ).

Magnetic resonance imaging (MRI) can help assess the duration of brain damage during the development of cerebral palsy (CP) ( 12 ). MRI can also be used to assess white matter damage in premature infants and is therefore more common in the field of cerebral palsy diagnosis ( 13 ).

Therefore, the development of MRI technology and the discovery of cerebral palsy monitoring indicators have become increasingly important. In the biological and molecular research fields, finding more accurate monitoring indicators is particularly important ( 14 , 15 ).

The keywords used for the treatment of cerebral palsy include muscle spasticity and physical therapy modifications. The treatment of cerebral palsy (CP) includes physical therapy and complementary therapies to various standard clinical treatments ( 16 ).

The prenatal diagnosis of cerebral palsy relies on routine ultrasound examinations, which mainly detect congenital malformations and other diseases, but there is currently no specific method for treating cerebral palsy ( 17 ). Research ( 18 ) has summarized the risk factors for cerebral palsy, such as chorioamnionitis, maternal infection, neurotropic virus infection, any virus from Herpes Group B for hemiplegia, and infection of the mother with cytomegalovirus during the first trimester.

Therefore, in the absence of significant progress in imaging, biochemistry, and other related research fields, the discovery of key genes and therapeutic targets in genetics for cerebral palsy will promote the development of methods for diagnosing and treating cerebral palsy.

Future frontiers

Due to the extensive screening of diagnosis and treatment, we observed a relatively significant clinical situation. Imaging is used for the diagnosis of cerebral palsy, while physical therapy and some auxiliary treatments are used for the clinical treatment of cerebral palsy. At the molecular level, research on cerebral palsy

is relatively scarce. Currently, relatively few studies on neurons, muscle contractures, and genes exist; these studies have not been fully examined through bibliometric analysis of the literature. Research on the underlying mechanism of cerebral palsy is not particularly abundant, but, in combination with other research studies on diseases and a small amount of basic research, we speculate that research on the molecular mechanisms will provide evidence for the diagnosis and treatment of cerebral palsy to some extent.

The “Activation of the Ap 1 Family of Transcription Factors” was the most significantly differentially expressed pathway. A previous study showed that gene mutations in AP-1-related transcription factor complexes can affect brain development ( 19 ).

The “Akt Phosphorylates Targets in the Cytosol” represents the presence of phosphorylated Akt in the cytoplasm. Studies have shown that the phosphoinositide 3-kinase (PI3K)-protein kinase B (Akt) signaling pathway enhances neurogenesis ( 20 ). Our study showed that Akt phosphorylation was downregulated in patients with NAFLD compared to healthy individuals ( Figure 8 ).

This study examined a comprehensive collection of 7,383 publications spanning from 1990 to 2023 that specifically address valvular diagnosis and therapies in patients with cerebral palsy. The objective of this research is to identify the countries, institutions, and authors that have had significant impacts on this field. Furthermore, our investigation focuses on specific topics to discern prevailing research patterns. Additionally, we employed GSVA R package to explore common pathways associated with cerebral palsy. Although cerebral palsy is a congenital disease, clinical detection has been less common but should be the focus of future research. Although the molecular mechanisms involved have not been the focus of current research, these mechanisms will provide a basis for the drug treatment to some extent.

Data availability statement

The original contributions presented in the study are included in the article/ Supplementary material , further inquiries can be directed to the corresponding author.

Author contributions

LJ: Project administration, Writing – original draft. WY: Writing – original draft. HC: Data curation, Writing – review & editing. HS: Data curation, Writing – review & editing. SZ: Conceptualization, Data curation, Formal analysis, Funding acquisition, Investigation, Methodology, Project administration, Resources, Software, Supervision, Validation, Visualization, Writing – original draft, Writing – review & editing.

The author(s) declare that financial support was received for the research, authorship, and/or publication of this article. This study was supported by Hangzhou, Zhejiang Province, Biomedical and Health Industry Development Support Science and Technology Special Project (2023WJC326).

Conflict of interest

The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

Publisher's note

All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.

Supplementary material

The Supplementary Material for this article can be found online at: https://www.frontiersin.org/articles/10.3389/fneur.2024.1354311/full#supplementary-material

Figure S1. A flowchart of literature identification and selection.

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19. Jamra RA, Philippe O, Raas-Rothschild A, Eck SH, Graf E, Buchert R, et al. Adaptor protein complex 4 deficiency causes severe autosomal-recessive intellectual disability, progressive spastic paraplegia, shy character, and short stature. Am J Hum Genet. (2011) 88:788–95. doi: 10.1016/j.ajhg.2011.04.019

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Keywords: cerebral palsy, bibliometric analysis, visual analysis, diagnosis, therapy

Citation: Jiang L, Yang W, Chen H, Song H and Zhang S (2024) Diagnosis and therapies for patients with cerebral palsy over the past 30 years: a bibliometric analysis. Front. Neurol. 15:1354311. doi: 10.3389/fneur.2024.1354311

Received: 12 December 2023; Accepted: 20 March 2024; Published: 17 April 2024.

Reviewed by:

Copyright © 2024 Jiang, Yang, Chen, Song and Zhang. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY) . The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

*Correspondence: Song Zhang, zhsg1049@163.com

† These authors have contributed equally to this work and share first authorship

Disclaimer: All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article or claim that may be made by its manufacturer is not guaranteed or endorsed by the publisher.

Cerebral Palsy Heterogeneity: Clinical Characteristics and Diagnostic Significance from a Large Sample Analysis

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Junying Yuan , Mengli Cui , Qiongqiong Liang , Dengna Zhu , Jie Liu , Jiefeng Hu , Shijie Ma , Dong Li , Jing Wang , Xuejie Wang , Deyou Ma , Kate Himmelmann , Xiaoyang Wang , Yiran Xu , Changlian Zhu; Cerebral Palsy Heterogeneity: Clinical Characteristics and Diagnostic Significance from a Large Sample Analysis. Neuroepidemiology 2024; https://doi.org/10.1159/000539002

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Introduction: Cerebral palsy (CP) is a nonprogressive movement disorder resulting from prenatal or perinatal brain injury that benefits from early diagnosis and intervention. The timing of early CP diagnosis remains controversial, necessitating analysis of clinical features in a substantial cohort. Methods: We retrospectively reviewed medical records from a university hospital, focusing on children aged >24 months or followed up for ≥24 months, and adhered to the International classification of diseases-10 for diagnosis and Subtyping. Results: Among the 2012 confirmed CP cases, 68.8% were male and 51.44% had spastic diplegia. Based on the Gross Motor Function Classification System (GMFCS), 62.38% were levels I and II, and 19.88% were levels IV and V. Hemiplegic and diplegic subtypes predominantly fell into levels I and II, while quadriplegic and mixed types were mainly levels IV and V. White matter injuries appeared in 46.58% of cranial MRI findings, while maldevelopment was rare (7.05%). Intellectual disability co-occurred in 43.44% of the CP cases, with hemiplegia having the lowest (20.28%, 58/286) and mixed types (73.85%, 48/65). Additionally, 51.67% (697/1349) of the children with CP aged ≥48 months had comorbidities. Conclusions: This study underscores white matter injury as the primary CP pathology and identifies intellectual disability as a common comorbidity. Although CP can be identified in infants under one year old, precision in diagnosis improves with development. These insights inform early detection and tailored interventios, emphasizing their crucial role in CP management.

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The Effect of Eight Weeks of Aquatic Exercises on Muscle Strength in Children with Cerebral Palsy: A Case Study

Mehrnoosh esmailiyan.

1 Faculty of Sport Sciences, Ragheb Isfahani Higher Education Institute, Isfahan, Iran

Seyed Mohamad Marandi

2 Department of Exercise Physiology, Faculty of Sport Sciences, University of Isfahan, Isfahan, Iran

Maryam Darvishi

3 Sama Technical and Vocational Training College, Islamic Azad University, Esfahan (Khorasgan) Branch, Esfahan, Iran

Shaghayegh Haghjooy Javanmard

4 Applied Physiology Research Center, Cardiovascular Research Institute, Isfahan University of Medical Sciences, Isfahan, Iran

Atefeh Amerizadeh

5 Isfahan Cardiovascular Research Center, Cardiovascular Research Institute, Isfahan University of Medical Sciences, Isfahan, Iran

Background:

Cerebral palsy in children is considered a non-progressive brain injury due to abnormal brain development. The aim of this study was to investigate the effect of eight weeks of aquatic exercises on muscle strength in children with cerebral palsy.

Materials and Methods:

This study was performed on three boys with cerebral palsy with a mean age of 6.5 years. In this research, a single case study method with A1-B-A2 design has been used. After determining the position of the baseline, the intervention began and during 24 sessions of individual intervention, aquatic exercises were presented to the subjects and all three subjects were followed up for 2 consecutive weeks and one month after the end of the intervention. The strength of the flexor muscles of the arms and legs was measured by a power track dynamometer made by JTECK with a threshold of 4.4 N.

Based on the indicators of descriptive statistics and visual analysis, the intervention was effective for all three participants in muscle strength, and the strength of individuals after the intervention has improved compared to the baseline stage (percentage). Information overlap for the first and second participant in the strength of right thigh flexors was 75% and for the third participant was 100%. The strength of the upper and lower torso muscles improved after the end of the training compared to the basic stage.

Conclusion:

Aquatic exercises can increase the strength of children with cerebral palsy and provide a favorable environment for children with cerebral palsy.

I NTRODUCTION

Cerebral palsy is a group of developmental-motor disorders that are non-progressive and occur in a developing fetus or infant brain and continue throughout life.[ 1 ] Its prevalence in Iran is 2.06[ 2 ] and in other countries is about 2 per 1000 live births.[ 3 ] The rate has been stable in Western countries for the past two decades.[ 4 ] In this group of children, problems in posture and movement cause limitations in various life activities and the level of participation. On the other hand, defects in the proper functioning of the muscular system lead to conditions such as abnormal muscle tone, reduced control in selective movements.[ 1 ]

Dodd in 2003 considered muscle weakness to be a major problem for most patients with spastic cerebral palsy.[ 5 ] Andersson[ 6 ] clinically demonstrated in 2003 that reducing spasticity causes muscle weakness and abnormal movement patterns in most children, and therefore recommended muscle strengthening and coordination to improve motor function.

Researchers are always looking for appropriate strategies to support patients with cerebral palsy and reduce the various complications associated with it. In recent years, the effect of various sports exercises on this very common sensory-motor disorder has attracted the attention of many researchers. Among the main goals of exercise interventions for children with cerebral palsy are to reduce the effects of disorders in gross motor function, gait, aerobic capacity, functional strength, balance, andconsequently, reduce their limitations for activity and increase sports participation.[ 7 ] Children with a cerebral palsy score lower than healthy children in terms of physical and motor fitness, including strength and endurance.[ 8 ] Exercising in water is one of the methods that its use has grown significantly in the last two decades due to its benefits and has become a form of exercise therapy. Hydrotherapy can relieve the symptoms of the disease, as well as improve motor and cognitive abilities in many Help diseases.[ 9 ] Different training conditions in an aqueous environment reduce overload on the joints and prevent injury. Floating also allows one to perform exercises that one cannot do on the ground.[ 9 ] Water sports are safer and safer than land sports for such people because the anti-gravity buoyancy force in the water environment acts as a resistance force. The water environment is suitable for comfortable and easy movement of people with mobility problems on the ground. In addition, hydrostatic pressure during water immersion exerts an equal resistance on all active muscle groups. Therefore, the water environment can be suitable for resistance activities,[ 10 ] and it is recommended as an immune and complementary treatment in children with cerebral palsy.[ 11 ]

However, due to the fact that few studies have studied the effects of hydrotherapy exercises on the muscular strength of children with cerebral palsy on a case-by-case basis, and on the other hand, these studies have evaluated more young and elderly people and less studied children; Therefore, the present study was designed and conducted to investigate the effect of hydrotherapy exercises on muscle strength in children with cerebral palsy.

M ATERIALS AND M ETHODS

Study design and population.

The method of the present study was a case-study based on individual analysis with purposive sampling. Three boys with cerebral palsy (two 7 years old and one 6 years old) from “Farda Physical-Mental Center” in Isfahan province using a medical record and consent of their parents, observing the entry and exit criteria as follows were chosen.

Inclusion and exclusion criteria

Inclusion criteria: 1- 6-7 years old age range, 2- being male, 3- ability to understand verbal commands, 4- lack of regular participation in water sports sessions at least 3 months before the intervention, 5- no orthopedic surgery in 1 year before the intervention, 6- do not inject botulism toxin for 6 months before the intervention, 7- no cardiovascular problems, 8- do not use sedatives during the intervention.

Exclusion criteria: 1- lack of cooperation between parents and children; 2- occurrence of orthopedic accidents during the intervention time.

Baseline measurements

The strength of the shoulder and thigh flexor muscles was measured by a JTECK Power Track Õ dynamometer with a threshold of 4.4 N. The strength of shoulder and thigh muscles in three hands was measured with 10-12 repetitions, which after 60 seconds of rest between each repetition; the average was calculated and recorded. The measurement was that the subject was sitting, the instrument was placed on the flexor muscles of her shoulder and thigh, and then the subject was asked to bend the shoulder and thigh according to a previously documented method.[ 12 ]

Intervention protocol

The design used in this study was A1-B-A2. Stage A1 involves gathering information before the intervention for four weeks; Phase B, the intervention phase consisted of eight weeks of training in water, and phase A2 included the follow-up phase or the secondary baseline phase, which included a period of two weeks.[ 13 ]

Children with cerebral palsy followed eight weeks of water training. This program replaced their usual treatment program. The exercise plan consisted of eight weeks of exercise, three sessions per week, and 60 minutes per session (with a day off between each exercise day), which was performed in the pool of the University of Isfahan under the supervision of a researcher. The training sessions were performed according to the American College of Sports Medicine (ACSM) guidelines, consisted of four sections, which included 5 to 10 minutes of warm-up, 15 to 20 minutes of stretching exercises for people with cerebral palsy, and 25 to 30 minutes of torso strengthening exercises, respectively and at the end of each session, people cooled for about 5 minutes.

In this study, to analyze the data, first, the raw data were plotted (for each subject, the data related to the three baseline positions, intervention, and follow-up were plotted on a graph, respectively). Then, the stability and trend compartment was plotted for the data diagram of all three subjects in the baseline and intervention positions. Then, using the trend and stability index, the degree of stability and direction of the data trend was determined. Between situational, the effectiveness of the independent and dependent variables was evaluated.

The results of visual analysis of the graphs show that training in water in the intervention position compared to the baseline has increased the strength of the flexor muscles of the superior shoulder. The percentage of information overlap (PND) is 100% for the first and second participants and 75% for the third participant. Also, the findings of visual analysis of the data graphs of all three subjects showed an increase in the strength of the superior thigh flexor muscles (PND was 75% for the first and third participants and 100% for the second participant). Thus, the results of the degree of overlap between two adjacent positions (PND) showed that hydrotherapy exercises increased the strength of superior thigh flexors in the research sample in the intervention position relative to the baseline [Figures ​ [Figures1 1 and ​ and2 2 ].

Superior shoulder flexor muscle strength for all three participants

Superior thigh flexor muscle strength for all three participants

Table 1 shows the individual characteristics of the subjects. The findings of in-situ and inter-situ analysis are shown in Tables ​ Tables2 2 and ​ and3 3 .

Individual characteristics

Within and between position analysis for right hip flexion for all three participants

Within and between position analysis for right shoulder flexion for all three participants

D ISCUSSION

The aim of this study was to evaluate the effect of a course of hydrotherapy exercises on muscle strength in children with cerebral palsy. The results showed that the strength of the shoulder flexor muscles in the baseline stage has a steady trend, but with the start of hydrotherapy, the strength of the shoulder flexor muscles in the first participant at the end of the second week is the same as the basal strength level and from the end of the second week to the end of the eighth week it was increased. The third participant experienced a significant increase in shoulder flexor muscle strength during the first three weeks of training, but it almost remained constant from the end of the sixth week until the end of the intervention phase. PND is 100% for the first and second participant and 75% for the third participant. Also, the findings of visual analysis of all three subjects showed an increase in the strength of the superior thigh flexor muscles (PND was 75% for the first and third participants and 100% for the second participant). Thus, the results of the degree of overlap between two adjacent positions (PND) showed that hydrotherapy exercises increased the strength of superior thigh flexors in the research sample in the intervention position relative to the baseline.

The strength of the flexor muscles at baseline was almost constant in all three subjects. Starting the exercises every two weeks showed that the strength of the flexor muscles of the thighs, subjects number one and two, decreased from the second week to the end of the fourth week and then increased until the end of the exercises. Also, in subject number three, muscle strength increased by the end of the eighth week, starting with the exercises. The strength of the flexor muscles of the shoulders and thighs decreased in the follow-up phase in all three subjects, but was still greater than in the baseline phase and the initial weeks of training.

In their review study, Fregala reported that water exercise for children with cerebral palsy could improve other factors by reducing the negative effects of poor balance.[ 14 ] In a study by Kelly et al .,[ 15 ] exercise in water increased muscle strength and improved the aerobic status of children with cerebral palsy, but conflicting results were obtained in terms of balance. While according to the research of Wadu et al .[ 16 ] and Shinost,[ 17 ] the nature of the water environment and its buoyancy effect, in addition to reducing the force of gravity and greater freedom of muscles to move and increase deep inputs, improve range of motion, and better maintain posture settings; the latter occurs especially due to water temperature. In hot water, the activity of the gamma nerve decreases with increasing body temperature, and this reduces the activity of the spindle, and as a result, facilitates muscle relaxation and reduces its stiffness, which in turn can affect the balance and muscle strength of people with cerebral palsy. Have a positive effect.[ 18 ] Dollings stated that the muscles of people with cerebral palsy suffer from a secondary muscle injury in which type b and IIa fibers decrease but type I fibers increase. However, the muscle strength caused by type II fibers is more prominent. Therefore, increasing strength can somehow indicate an increase in muscle volume and conversion of type II to I fibers.[ 19 ]

Similarly, Getz et al .[ 20 ] showed that children with more severe motor dysfunction, as classified by the GMFCS, may display superior performance in aquatic environments compared to their performance on land, which is consistent with our findings, who are restricted in their ability to perform many activities on land. One possible reason for this may be the thermal and mechanical effects of aquatic exercise.[ 21 ] The mechanical properties of the aquatic environment offer benefits by decreasing the effect of gravity and joint loading, optimizing postural control, and muscle strength. The viscosity of the water allows for fluid movement patterns to be experienced. Clapham et al .[ 22 ] have argued that these factors improve neuromuscular coordination, muscle endurance, and aerobic capacity. In addition, the increased unloading of body weight may facilitate an increase in muscle strength, thus allowing children to initiate movements that are more restricted on land.[ 23 , 24 ]

Daly et al .[ 25 ] showed in their study that the type of exercise is very important in improving the muscular strength of patients with cerebral palsy, and this issue should be considered when prescribing exercise. Also, children with CP are more likely to have lower levels of physical activity than their peers, which has negative implications for their health. However, aquatic exercise can be used to improve levels of fitness among children with CP.[ 26 ]

So far, there is little evidence to identify the best exercise protocol that includes the intensity, repetition, and duration of activity for children with cerebral palsy.[ 27 ] However, the response of age groups and different types of cerebral palsy to resistance training has not been fully studied. In the past, strength training was prohibited in patients with cerebral palsy because it was thought that these exercises increased muscle stiffness and thus increased spasms and range of motion of the joint.[ 28 ] Recent studies, however, show that there is no change in muscle spasms during and after strength training, and there is not even a ban on using these exercises in people with spasms.[ 29 ]

A study by Retarekar et al . evaluated the effects of a water-based aerobic exercise program on a child with cerebral palsy.[ 29 ] Significant improvements in participation, activity, functional abilities, endurance, and gait were observed. These findings indicate that an aerobic exercise program in water is effective for a child with cerebral palsy and supports the need for further research in this area.[ 30 ]

In children with cerebral palsy, the combination and use of controlled reflex movement patterns and muscle activity may cause shortening of muscles, tendons, and ligaments. Therefore, their compensatory movements create an abnormal physical posture that prevents the development of motor skills in the long run. Due to muscle weakness and spasms, this group of people have difficulty performing activities such as walking and running independently and their participation in physical activity is reduced.[ 31 ] The benefits of using hydrotherapy depend on its anti-gravity and buoyancy position. Therefore, it can help to lose weight and reduce the compressive forces on the joints. As a result, the treatment promotes active and easier movements for children who are unable to perform some ground activities. Finally, due to the effects of hydrostatic water pressure, hydrotherapy can reduce muscle spasticity and improve the endurance of multisensory stimuli and increase blood circulation.[ 32 ]

The hydrotherapy program had positive effects on the body function and structure of children and adolescents with cerebral palsy. It can also strengthen the function of the heart, arteries, and muscles and reduce energy consumption while walking. Hydrotherapy program reduces spasticity of hip joints and knee flexors.[ 33 ] It was also found that the hydrotherapy program causes a significant increase in parameters related to walking speed, stride length, increase in strength, range of motion, and cardiovascular endurance in children with cerebral palsy.[ 32 ] Therefore, it can be used as an alternative but safe treatment even in children with cerebral palsy, whose ability to perform ground training is limited.[ 34 ] Group hydrotherapy exercises help to improve the ability to walk in adolescents with cerebral palsy by creating a balance of functions between the heart and the respiratory system and reducing the rate and number of heartbeats.[ 28 ] The most important effects of hydrotherapy in patients with cerebral palsy are increased concentration, muscle strength, balance, the increased threshold of touch, onset, and maintenance of eye contact.[ 35 ] In addition, hydrotherapy also has positive effects on gross motor functions such as jumping, running, and walking.[ 35 ] The use of hydrotherapy exercises along with resistance training was also useful in strengthening endurance and muscle strength.[ 36 ]

Improving the important components of muscle strength and balance, which are the primary disorders in children with cerebral palsy, seems to increase self-confidence, strengthen muscles, and children's participation in daily activities and social relationships. However, this study, with some limitations, such as the small number of samples, was a more accurate assessment of the need for more advanced instruments such as electromyography, which unfortunately could not be used due to instrumental and costly conditions. In addition, each participant, due to their specific circumstances, had to have their own occupational therapist, which was also not possible due to limited staff.

The results showed that hydrotherapy exercises increased muscle strength in children with CP. Part of the increase in strength can be attributed to an increase in muscle mass and part to anabolic hormones. However, the increase in strength due to muscle volume and filament conversion has not been well demonstrated and these factors were not investigated in this study. It seems that by examining muscle volume using the magnetic resonance imaging (MRI) method, it is possible to determine the effect of muscle volume on muscle strength.

C ONCLUSION

The results of the present study showed that hydrotherapy exercises can be effective in improving the muscle strength of the flexor muscles of the shoulders and thighs of children with cerebral palsy, so these exercises can be considered as an important strategy to increase muscle strength due to increased volume. Muscle and other anabolic hormone stimulants are recommended for people with cerebral palsy who have weak muscles and reduced flexibility. They concluded that hydrotherapy could be used in children and adolescents. Exercises, their duration, and intensity should be determined based on the physical and cognitive condition of patients. Hence, it is suggested that further studies in this field are very necessary.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Conflicts of interest.

There are no conflicts of interest.

Acknowledgment

This research has been done in “Farda Physical-Mental Center” in Isfahan; therefore, the cooperation of this center and the subjects is fully appreciated.

R EFERENCES