case study of cerebral palsy child

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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

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Case report article, case report: perspective of a caregiver on functional outcomes following bilateral lateral pectoral nerve cryoneurotomy to treat spasticity in a pediatric patient with cerebral palsy.

• 1 Island Medical Program, University of British Columbia, Victoria, BC, Canada
• 2 Division of Physical Medicine and Rehabilitation, University of British Columbia, Victoria, BC, Canada
• 3 Canadian Advances in Neuro-Orthopedics for Spasticity Congress, Victoria, BC, Canada

Spasticity is common and difficult to manage complication of cerebral palsy that significantly affects the function and quality of life of patients. This case study reports a 15-year-old male with quadriplegic cerebral palsy, Gross Motor Function Classification System 5 (GMFCS 5), who presented with significant bilateral adducted and internally rotated shoulders as a component of generalized spasticity. Spasticity in the lower limb of the patient had been treated with botulinum toxin A (BoNT-A) injections; however, the shoulder region was spared due to concerns of toxin spread and aspiration risk. Following diagnostic nerve blocks, the patient underwent bilateral cryoneurotomies of the right and left lateral pectoral nerves (LPNs) lasting 3.5 min for each lesion. One month after the cryoneurotomies, the range of motion (ROM) had improved from 86° to 133° on the right and 90° to 139° on the left. Improvements in ROM were retained at 9 months post-procedure. At 8.5 months following the cryoneurotomies, the caregiver reported improvements in upper body dressing, upper body washing, transferring, and the ability of the patient to remain sitting in his wheelchair for extended periods. Cryoneurotomy may be an effective procedure for improving shoulder ROM and specific functional outcomes for caregivers of patients with spasticity arising from cerebral palsy.

Introduction

Spasticity is a common and important complication of cerebral palsy that has a significant impact on the quality of life and functional capacity of patients ( 1 ). Targeted management of muscle spasticity is a key aspect of patient care. Various therapeutic options are available for managing spasticity, typically consisting of a combination of pharmacological treatments and surgical or injectable modalities ( 2 ). The use of a mini-invasive percutaneous cryoneurotomy to induce disruption of the axon and myelin is an emerging technique for managing spasticity ( 3 ). There is substantial evidence of the efficacy of cryoanalgesia in the pain literature; however, literature outlining the use of cryoneurotomy for spasticity treatment is limited ( 3 , 4 ). There is no literature available on functional outcomes of cryoneurotomy in the pediatric population ( 3 ). This sentinel case demonstrates quantitative improvements in range of motion (ROM) and qualitative improvements that have been reported from the caregiver of a patient who underwent bilateral pectoral cryoneurotomy to manage spasticity arising from cerebral palsy.

Case Report

This study conforms to all Case Reports (CARE) guidelines and reports the required information accordingly (refer to Supplementary Material 2 ). The parent/caregiver provided informed consent for the publication of this study. A 14-year-old male with quadriplegic cerebral palsy, Gross Motor Function Classification System 5 (GMFCS), presented with problematic bilateral adducted and internally rotated shoulders as a component of generalized spasticity in the upper and lower limbs and cervical spasticity/dystonia. He had been treated with botulinum toxin A (BoNT-A) injections to various arm and leg muscles based on symptomatology. No BoNT-A had been injected into the shoulder girdle due to fears of toxin spread and aspiration risk. He had undergone surgical release of his hip adductors. The patient had repeated admissions to hospital intensive care for recurrent pneumonia and gastrointestinal bleeding with hematemesis, which affected his BoNT-A regimen. He had a generalized seizure disorder. He was referred to the multidisciplinary spasticity clinic for consideration of a novel cryoneurotomy procedure to counteract problematic tone in the upper extremities.

The physical examination revealed a greatly reduced shoulder ROM with abduction passively to 85° on the right and 90° on the left, with no active abduction. His Modified Ashworth Scores (MAS) were four, with a fixed end point. The elbows had minimal spasticity, and the wrists and fingers were held in a fist, but flexible, with contracture noted at the metacarpophalangeal joints. His parent/caregiver reported that the painful shoulder positions greatly affected his daily care needs, such as dressing, bathing, and sitting. Diagnostic anesthetic motor nerve blocks (DNBs) were performed to each of the right and left lateral pectoral nerves (LPNs). The DNB causes temporary nerve conduction cessation to differentiate between the presence of shoulder girdle muscle contracture necessitating surgical release due to musculotendinous retraction (an unsuccessful block) vs. a reducible deformity due to spastic muscle overactivity (a successful block). Under ultrasound guidance, the neurovascular bundles of the LPN were identified using a longitudinal orientation along the chest, four fingerbreadths below the coracoid process. Lidocaine (1.5 ml of 2%) was injected juxtaposed to each of the right and left LPNs at the undersurface of the pectoral major muscle (PMM). After the DNB, there was an improvement in passive ROM in shoulder abduction to 120° bilaterally and a reduction in spasticity on the MAS (refer to Supplementary Material 1 ). There was an observed reduction in facial grimacing and easing of heavy respirations with passive abduction.

The decision was then made to proceed to percutaneous cryoneurotomies of both the LPNs. The procedure was delayed until the medical stability of the patient improved. He was then 15-year old. The procedures were performed 10 days apart in an outpatient interventional suite. An aseptic technique was used with 2% chlorhexidine and betadine. The ultrasound-guided cryoneurotomy was performed using a Lloyd SL 2000 Neurostat (San Diego, CA, USA) with a 1.2-mm cryoprobe at −60°C placed through a #16 angio guide. E-stimulation was performed to confirm nerve contact at 0.8 mV. The ice ball was repositioned to contact the LPN at two spots along the nerve. Each lesion was treated for 3.5 min. Hemostasis was achieved using skin glue and a plaster bandage. There were no surgical complications with the procedure or complications reported by the caregiver following the procedure.

Quantitative Results

One month after the bilateral cryoneurotomies, the ROM in abduction had improved from 86 to 133° on the right and 90 to 139° on the left ( Figure 1 and Supplementary Material 1 ). The MAS was reduced to two. He was next seen for follow-up at 9 months. The improvement in ROM noted a gain on the right to 146° and a reduction to 125° left ( Figure 1 and Supplementary Material 1 ). The reduction in tone was to MAS 1+ within the available ROM.

Figure 1 . Abduction and arm position of the right arm prior to cryoneurotomy (A) , at 1 month following cryoneurotomy (B) and 9 months following cryoneurotomy (C) .

Qualitative Results

For this assessment, the Care and Comfort Caregiver Questionnaire (CareQ) was used. The CareQ is an assessment tool that has been adapted from the Caregiver Questionnaire (CQ), a questionnaire created in 1990 to assess children with spastic quadriplegic cerebral palsy prior to and following selective posterior rhizotomy ( 5 , 6 ). In creating the CareQ, the CQ was modified to emphasize caregiver experience and goal setting for the child ( 5 ). The CareQ focuses on three functional areas, namely, personal care, position/transfers, and comfort ( 5 ). Functional outcomes are compared in these three areas prior to and following the procedure in a retrospective manner ( Table 1 ). The caregiver of the patient was administered the CareQ over the phone following the procedure for 8.5 months. The patient is dependent on the caregiver to undertake all personal care tasks outlined in the questionnaire. At 8.5 months following the cryoneurotomies, there was an improvement in putting on shirts, taking off shirts, and washing the upper body of the patient. There was an improvement in the ability of the patient to remain sitting in a wheelchair for 3 h, the ease of transferring the patient into and out of the wheelchair, and the ease of applying orthotics. There were improvements in comfort levels during position changes while sitting in his wheelchair and while participating in school programs and physiotherapy.

Table 1 . Results of CareQ were completed by the caregiver of a 15-year-old patient with cryoneurotomy who underwent bilateral pectoral cryoneurotomy.

Spasticity arising from cerebral palsy is challenging to treat, and clinical approaches to management vary due to a lack of strong evidence to inform pharmacological therapy regimens ( 7 ). The field of spasticity management for patients with cerebral palsy may be amenable to novel therapies that have a clinical benefit. Traditional approaches to spasticity management consist of pharmacologic, surgical or neurolytic, and injectable options, such as botulinum toxin ( 2 , 7 – 12 ). Pharmacological regimens may consist of diazepam, baclofen, or trihexyphenidyl ( 7 – 12 ). Cryoneurotomy percutaneously induces selective neurolysis of a motor nerve to manage spasticity, similar to other injectable and surgical modalities, such as partial neurotomy and chemodenervation by alcohol or phenol ( 2 , 3 ). In cryoneurotomy, the axons and myelin of peripheral nerves are disrupted by the tip of the cryoprobe which may reach −70°; however, the epineurium is maintained allowing for nerve regeneration ( 3 , 4 , 13 ). Cryoneurotomy carries less risk of damage to surrounding tissue than phenol or alcohol chemodenervation ( 3 ). This procedure has been shown to have a clinically significant impact on spasticity reduction, even in cases refractory to other therapeutic strategies ( 3 ). Cryoneurotomy for the flexed elbow spasticity was shown to maintain the improved ROM and reduced MAS at a mean follow-up interval of 12.5 months in 11 patients including maintenance in the longest follow at over 2 years ( 14 ).

The LPN is the dominant innervation to the pectoralis major muscle (PMM). Anatomical studies have demonstrated that the LPN was found to have a highly consistent course after leaving the lateral trunk of the brachial plexus alongside the blood vessels on the undersurface of the pectoralis major in 100 consecutive patients ( 15 , 16 ). The PMM is the largest muscle implicated in shoulder adduction and internal rotation ( 17 ). The LPN was shown to innervate both heads of the pectoralis major, ( 15 ) while the lower portion of the PMM has innervation from the medial pectoral and also from intercostal nerves ( 17 ). In contrast, the medial pectoral nerve is also thought to have a far less consistent course and is harder to consistently target ( 16 , 17 ). It has been shown to dive below the pectoralis minor before rising along with the pectoralis major ( 18 ). The consistency of the LPN renders it the more easily identifiable nerve with ultrasound, a target for cryoneurotomy, and less deep and further away from the chest cavity ( Figure 2 ) ( 18 ).

Figure 2 . Ultrasound image of the lateral pectoral nerve (LPN), the dominant nerve of the pectoralis major muscle (PMM) ( 15 , 16 ).

The passive abduction of the patient improved by an average of 55% within 6 weeks. At 9 months, the right nerve continued to show improvements in ROM. The left side reduced in ROM but maintained a 30° improvement compared to the measurement prior to the cryoneurotomy. The MAS remained reduced at 9 months with much greater ease in passive ROM of the shoulder. This is consistent with findings of cryoneurotomy for flexed elbow spasticity ( 14 ) and the tibial nerve ( 3 ).

In a non-verbal patient, discussion with the caregiver is necessary to identify goals and expectations. The frequent hospitalizations of this young patient for infection, respiratory compromise, seizures, and gastrointestinal bleeding made it challenging to attend the routine 3-month intervals for botulinum toxin, hence a longer-lasting procedure was the desired option. Due to the comorbidities of the patient, surgical interventions that carry a risk of toxin spread and respiratory compromise, such as BoNT-A injections, were avoided.

This case demonstrates the impact that the emerging therapeutic procedure cryoneurotomy has on the LPN to reduce spasticity in a GMFCS 5 patient. Outcomes of cryoneurotomy were measured not only through improvement in spasticity and ROM but also through functional outcomes reported by the caregiver of the patient. Targeted cryoneurotomy to address spasticity in specific muscles may improve ROM and functionality in a variety of tasks, such as dressing, hygiene, transferring, and physiotherapy programs. Given the lack of standardized management in treating spasticity, there is a benefit to exploring novel procedures that may be efficacious in many patients, including those who are resistant to more traditional therapeutic options. Further research is necessary to determine how cryoneurotomy fits into the current practice of spasticity management.

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/s.

Ethics Statement

Written informed consent was obtained from the individual(s), and minor(s)' legal guardian/next of kin, for the publication of any potentially identifiable images or data included in this article.

Author Contributions

PW provided the case study and patient care. JS provided the patient interview and outcome measures.

Conflict of Interest

PW has funding for a clinical trial in adults with spasticity for cryoneurotomy, provided by Abbvie Allergan and Pacira.

The remaining author declares 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/fresc.2021.719054/full#supplementary-material

Supplementary Material 1. (Multimedia) Demonstration of diagnostic nerve block on the lateral pectoral nerve (LPN) under ultrasound imaging followed by a demonstration of shoulder range of motion (ROM) prior to cryoneurotomy, at 1 month following cryoneurotomy, and 9 months following cryoneurotomy.

Supplementary Material 2. CARE checklist.

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Keywords: cryoneurotomy, spasticity, cerebral palsy, caregiver perspective, pediatrics

Citation: Scobie J and Winston P (2021) Case Report: Perspective of a Caregiver on Functional Outcomes Following Bilateral Lateral Pectoral Nerve Cryoneurotomy to Treat Spasticity in a Pediatric Patient With Cerebral Palsy. Front. Rehabilit. Sci. 2:719054. doi: 10.3389/fresc.2021.719054

Received: 02 July 2021; Accepted: 09 August 2021; Published: 06 September 2021.

Reviewed by:

Copyright © 2021 Scobie and Winston. 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: Paul Winston, paul.winston@viha.ca ; orcid.org/0000-0002-8403-6988

† These authors have contributed equally to this work

This article is part of the Research Topic

Emerging Concepts and Evidence in Novel Approaches for Spasticity Management

• Second Opinion

Cerebral Palsy in Children

What is cerebral palsy in children.

Cerebral palsy (CP) is a brain (neurological) disorder that causes problems with normal motor function. It is a lifelong condition that affects how the brain and muscles communicate. CP affects body movement, muscle control, coordination, reflexes, posture, and balance. These problems are caused by damage to or abnormal development of certain brain areas. But many children with CP have normal intelligence. CP can range in severity, but it doesn’t get worse over time. CP does not get better over time either. With diagnosis and treatment, children can learn how to manage their condition.

What causes CP in a child?

CP occurs when there is abnormal development of or damage to areas of the brain that control motor function. This can happen before or during birth (congenital CP). Most CP cases are congenital. Less commonly, CP can happen after birth. This is called acquired CP and usually happens from an infection or head injury.

In many cases, the exact cause of CP is not known. It may be the result of a problem such as:

Lack of oxygen to the brain

Genetic condition

Bleeding in the brain

Severe jaundice

Head injury

Which children are at risk for CP?

A child is more at risk for CP because of any of the following:

Preterm birth

Inflammation of the placenta or amniotic fluid from an infection (chorioamnionitis)

Blood clotting disorder

Very low birthweight, especially under 3.3 pounds

Infection with a virus

Chemical or substance abuse during pregnancy

Complications of labor and delivery, in rare cases

What are the symptoms of CP in a child?

Symptoms can occur a bit differently in each child. A child may have muscle weakness, poor motor control, or shaking (spasticity) of the arms or legs. A child may also have muscle stiffness in the form of stiff legs or clenched fists.

The symptoms depend on what type of CP a child has. The types and symptoms include:

Spastic diplegia. Di means 2.This is spasticity of the legs in most cases, but sometimes the arms. Diplegia is also called paraplegia.

Spastic quadriplegia. This is also called tetraplegia. Quad and tetra mean 4. This is spasticity of all arms and legs.

Spastic hemiplegia. Hemi means half. This is spasticity that affects 1 side of the body, such as the right arm and right leg.

Spastic double hemiplegia.  This is spasticity on both sides of the body. The amount of spasticity is different on each side.

Athetoid CP. This is also called dyskinetic CP. This is movement that can’t be controlled (involuntary). The movement is usually twisting and rigid.

Ataxic CP.  This affects balance, leading to unsteady walking. It also affects fine motor coordination. This makes it hard to do things such as writing.

Babies with CP are often slow to reach developmental motor milestones. These may include learning to roll over, sit, crawl, or walk. They may also keep certain reflexes that normally disappear in early infancy.

Children with CP may have additional problems. But these are not signs or symptoms of CP. CP refers only to the motor dysfunction. The additional problems may include:

Vision, hearing, or speech problems

Learning disabilities and behavior problems

Intellectual disability

Respiratory problems

Bowel and bladder problems

Bone problems including scoliosis, a sideways curvature of the spine

The symptoms of CP can be like other health conditions. Make sure your child sees his or her healthcare provider for a diagnosis.

How is CP diagnosed in a child?

A diagnosis of CP is not usually made until a child is at least 6 to 12 months old. This is when a child should be reaching developmental milestones. These include sitting, standing, and walking, plus hand and head control. The healthcare provider will ask about your child’s symptoms and health history. He or she will give your child a physical exam.

Your child may also have tests, such as:

Neurological exam.  This checks reflexes and brain and motor function.

MRI. This imaging test uses large magnets and a computer to make detailed images of organs and tissues in the body. This imaging test is often used to assess CP.

Feeding studies.  These tests use X-rays or videos to see what happens from the time food enters your child’s mouth until after your child swallows.

Electroencephalogram (EEG).  This checks electrical activity in the brain.

Gait lab analysis.  This looks at your child’s walking pattern.

CT scan.  This test uses X-rays and a computer to make detailed images of the body. A CT scan shows detailed images of any part of the body, including the bones, muscles, fat, and organs. CT scans are more detailed than standard X-rays.

Genetic studies.  These tests look for health conditions that can run in families.

Metabolic tests.  These tests check for the lack of specific enzymes that are needed to maintain the normal function of the body.

How is cerebral palsy treated in a child?

Treatment will depend on your child’s symptoms, age, and general health. It will also depend on how severe the condition is. CP is a lifelong condition that has no cure. Because of this, your child’s healthcare providers will work to:

Prevent or lessen defects and problems

Make the most of a child's abilities

A child is treated by a healthcare team that may include:

Pediatrician or family doctor. This is a child’s primary healthcare provider.

Orthopedic surgeon.  This is a surgeon who treats muscles, ligaments, tendons, and bones.

Neurologist.  This is a healthcare provider who treats conditions of the brain, spinal cord, and nerves.

Neurosurgeon.  This is a healthcare provider who treats the brain and spinal cord.

Ophthalmologist.  This is a healthcare provider who treats eye problems.

Dentist. This is a healthcare provider who treats mouth and teeth problems.

Nurse. This is a healthcare provider who often works with other healthcare providers.

Physiatrist.  This is a healthcare provider who specializes in physical medicine and rehabilitation.

Orthotist.  This is a professional who makes braces and splints.

Rehabilitation team.  These include physical, occupational, speech, and audiology therapists.

Management of CP may include:

Rehabilitation

Positioning aids to help a child sit, lie down, or stand

Braces and splints to prevent deformity and to give support or protection

Medicines given by mouth or injection to help decrease spasticity in the muscles

Surgery to treat orthopedic problems such as curvature in the back, hip dislocation, ankle and foot deformities, and contracted muscles

Surgery to treat spasticity

Talk with your child’s healthcare providers about the risks, benefits, and possible side effects of all treatments.

What are possible complications of CP in a child?

Possible complications vary widely from child to child. Treatment for complications will depend on your child’s symptoms, age, and general health. It will also depend on how serious the condition is. Your child’s healthcare provider will discuss treatment choices with you.

How can I help prevent CP in my child?

Because healthcare providers don’t know fully what causes congenital CP, little can be done to prevent it. CP related to gene problems can’t be prevented. But you can do certain things that might help reduce the risk:

Be as healthy as possible before and during pregnancy. Get early and regular prenatal care.

Don't smoke during pregnancy.

Get vaccinated for certain diseases that can harm a developing baby.

After birth, acquired CP is often caused by an infection or injury. Some of these cases can be prevented by helping keep your baby healthy and safe:

Keep your baby’s vaccines up to date.

Take steps to prevent injuries by always using a car seat, babyproofing your living areas, and watching your young child when near water.

How can I help my child live with CP?

CP is a lifelong condition that has no cure. It can range in severity, but it doesn’t get worse over time. The full extent of CP is usually not fully known right after birth. It can become clearer as a child grows and develops. With diagnosis and treatment, children can learn how to manage their condition.

Your child’s healthcare providers will work to prevent deformities or keep them to a minimum. They will also work to help your child make the most of his or her capabilities. You can help your child strengthen his or her self-esteem and be as independent as possible. Physical and occupational rehabilitation, plus extra support in school, can help a child function as well as possible.

When should I call my child’s healthcare provider?

Call the healthcare provider if your child has:

Symptoms that don’t get better, or get worse

New symptoms

Key points about cerebral palsy in children

Cerebral palsy (CP) is a brain (neurological) disorder that causes problems with normal motor function. It affects body movement, muscle control, coordination, reflexes, posture, and balance. 

In many cases, the exact cause of CP is not known. Most cases happen before or during birth (congenital CP). CP that occurs after birth usually happens from an infection or head injury.

CP can range in severity, but it doesn’t get worse over time. With diagnosis and treatment, children can learn how to manage their condition.

Symptoms can vary in each child and depend on the severity of CP. Many children with CP have normal intelligence. A child may have muscle weakness, poor motor control, or shaking (spasticity) of the arms or legs. A child may also have stiff legs or clenched fists.

CP is a lifelong condition that has no cure. A child is treated by a healthcare team. Treatment may include rehab, positioning aids, braces, splints, medicines, or surgery.

Tips to help you get the most from a visit to your child’s healthcare provider:

Know the reason for the visit and what you want to happen.

Before your visit, write down questions you want answered.

At the visit, write down the name of a new diagnosis, and any new medicines, treatments, or tests. Also write down any new instructions your provider gives you for your child.

Know why a new medicine or treatment is prescribed and how it will help your child. Also know what the side effects are.

Ask if your child’s condition can be treated in other ways.

Know why a test or procedure is recommended and what the results could mean.

Know what to expect if your child does not take the medicine or have the test or procedure.

If your child has a follow-up appointment, write down the date, time, and purpose for that visit.

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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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Robotic lower extremity exoskeleton use in a non-ambulatory child with cerebral palsy: a case study

Affiliations.

• 1 Alberta Children's Hospital, Calgary, Canada.
• 2 Hotchkiss Brain Institute, Calgary, Canada.
• 3 Swiss Children's Rehab, University Children's Hospital Zurich, Affoltern am Albis, Switzerland.
• 4 Departments of Clinical Neurosciences and Pediatrics, University of Calgary, Calgary, Canada.
• PMID: 33539714
• DOI: 10.1080/17483107.2021.1878296

Purpose: With few treatment options available for non-ambulatory children with cerebral palsy (CP), a robotic lower extremity gait trainer may provide a non-invasive addition to conventional treatment options. This case study investigates the usage and impact of robotic lower extremity gait trainer use in a participant with CP over the initial 3 months of use.

Materials and methods: This prospective case study involves a 7-year old female (GMFCS V) with CP (registered clinical trial: NCT04251390 ). The participant used a Trexo Home robotic gait trainer (Trexo) in the community with assessments occurring in the home and school. Trexo usage and bowel movements (BMs) were tracked daily. Postural control and lower extremity range of motion (ROM) and spasticity were evaluated prior to Trexo use and weekly to biweekly thereafter.

Results: The participant used the device an average of 46 min/week, over 3.3 d/week. BM frequency increased from 0.4/d at baseline, to 1.2 (±0.5)/d during Trexo use. There were no diffuse systematic changes in postural stability, ROM or muscle spasticity, but specifically head control and spasticity in the knee flexors had improvements.

Conclusions: Data and anecdotal reports suggest that regular use of the Trexo Home robotic gait trainer has positive outcomes on frequency and quality of BMs, and may improve head control, and knee flexor spasticity. Larger controlled studies are needed to evaluate the impacts of Trexo use in children with CP.Implications for RehabilitationNon-ambulatory children with CP can use and may experience benefits from using a robot-assisted gait trainer (RAGT).Constipation, aspects of balance and focal spasticity may improve.

Keywords: Robotics; cerebral palsy; constipation; locomotion; muscle spasticity; neurological rehabilitation; postural balance.

Publication types

• Case Reports
• Research Support, Non-U.S. Gov't
• Cerebral Palsy*
• Exoskeleton Device*
• Gait / physiology
• Lower Extremity
• Robotic Surgical Procedures*

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• Published: 01 May 2024

Exome sequencing reveals genetic heterogeneity and clinically actionable findings in children with cerebral palsy

• Yangong Wang 1   na1 ,
• Yiran Xu 2   na1 ,
• Chongchen Zhou 3   na1 ,
• Ye Cheng   ORCID: orcid.org/0000-0002-7380-8233 1 , 4   na1 ,
• Niu Qiao 5 ,
• Qing Shang 3 ,
• Lei Xia 2 ,
• Juan Song 2 ,
• Chao Gao 3 ,
• Yimeng Qiao 1 , 2 ,
• Xiaoli Zhang 2 ,
• Ming Li 2 ,
• Caiyun Ma 3 ,
• Yangyi Fan 1 ,
• Xirui Peng 2 ,
• Silin Wu   ORCID: orcid.org/0000-0002-4599-1851 6 ,
• Bingbing Li 2 ,
• Yanyan Sun 2 ,
• Bohao Zhang 2 ,
• Tongchuan Li 2 ,
• Hongwei Li 2 ,
• Jin Zhang 1 , 4 ,
• Qiaoli Li 1 ,
• Junying Yuan 2 ,
• Lei Liu 1 ,
• Andres Moreno-De-Luca   ORCID: orcid.org/0000-0002-2732-4043 7 ,
• Alastair H. MacLennan 8 ,
• Jozef Gecz   ORCID: orcid.org/0000-0002-7884-6861 8 ,
• Dengna Zhu 2 ,
• Xiaoyang Wang 9 ,
• Changlian Zhu   ORCID: orcid.org/0000-0002-5029-6730 2 &
• Qinghe Xing   ORCID: orcid.org/0000-0002-9916-8254 1 , 4  

Nature Medicine ( 2024 ) Cite this article

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• Developmental disorders
• Neurodevelopmental disorders

Cerebral palsy (CP) is the most common motor disability in children. To ascertain the role of major genetic variants in the etiology of CP, we conducted exome sequencing on a large-scale cohort with clinical manifestations of CP. The study cohort comprised 505 girls and 1,073 boys. Utilizing the current gold standard in genetic diagnostics, 387 of these 1,578 children (24.5%) received genetic diagnoses. We identified 412 pathogenic and likely pathogenic (P/LP) variants across 219 genes associated with neurodevelopmental disorders, and 59 P/LP copy number variants. The genetic diagnostic rate of children with CP labeled at birth with perinatal asphyxia was higher than the rate in children without asphyxia ( P  = 0.0033). Also, 33 children with CP manifestations (8.5%, 33 of 387) had findings that were clinically actionable. These results highlight the need for early genetic testing in children with CP, especially those with risk factors like perinatal asphyxia, to enable evidence-based medical decision-making.

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Comprehensive whole-genome sequence analyses provide insights into the genomic architecture of cerebral palsy

Hidden etiology of cerebral palsy: genetic and clinical heterogeneity and efficient diagnosis by next-generation sequencing

Yield of clinically reportable genetic variants in unselected cerebral palsy by whole genome sequencing

Data availability.

The sequencing data from our patients have been deposited securely in the Genome Sequence Archive (GSA) housed within the National Genomics Data Center at the China National Center for Bioinformation/Beijing Institute of Genomics, Chinese Academy of Sciences. The raw sequencing data are available under restricted access in GSA-Human (BioProject accession no.: PRJCA023830 ( https://ngdc.cncb.ac.cn/gsa-human/ )) due to patient privacy and Regulations on the Management of Human Genetics Resources of China, following the GSA guidelines ( https://ngdc.cncb.ac.cn/gsa-human/document ). VCF files of 1,578 children with clinical manifestations of CP have been deposited in the National Human Genetic Resources system (Record no.: *BF2023081113944). For access to data, please email [email protected] or [email protected]. The committee reviews data access requests on a monthly basis. This statement pertains to the distribution of sequencing data produced by our research. In addition, the P/LP/VUS variants within our CP cohort and previously reported P/LP/VUS variants in other CP cohorts are accessible at http://81.70.179.157/ all the time. The databases used in our analysis are all publicly available and can be obtained from the following links: ClinVar: https://www.ncbi.nlm.nih.gov/clinvar ; Gene Ontology: http://geneontology.org ; gnomAD: http://www.gnomad-sg.org ; and Kyoto Encyclopedia of Genes and Genomes (KEGG): https://www.genome.jp/kegg .

Code availability

All codes used in this manuscript have been deposited and are publicly available at https://github.com/YeCheng57/CerebralPalsy_pipeline .

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Acknowledgements

We thank B. Han and X. Chen from the Institute of Pediatrics, Children’s Hospital of Fudan University for their help with the nematode Caenorhabditis elegans experiment, and Y. Ping from the Bio-X Institutes, Key Laboratory for the Genetics of Developmental and Neuropsychiatric Disorders (Ministry of Education), Shanghai Jiao Tong University for his help in the fly Drosophila melanogaster experiment. We also thank the Core Facility of Shanghai Medical College, Fudan University for use of instruments. This work was supported by the Shanghai Municipal Commission of Science and Technology Research Project (19JC1411000 to Q.X.), the National Natural Science Foundation of China (U21A20347 to C. Zhu; 31972880, 32170615 and 31371274 to Q.X. and 82203969 to X.W.), the National Key Research and Development Program from the Ministry of Science and Technology of the People’s Republic of China (2021YFC2700800 to Q.X. and 2022YFC2704800 to C. Zhu), the National Key Research and Development Plan for Stem Cell and Translational Research (2017YFA0104202 to Q.X.), the collaborative innovation center project construction for Shanghai Women and Children’s Health (to Q.X.), a grant from Department of Science and Technology of Henan Province for international collaboration (GZS2023003 to X.W.), the Health Department of Henan Province (SBGJ202301009 to C. Zhu), Swedish governmental grants to scientists working in healthcare (ALFGBG-965197 to C. Zhu, ALFGBG-966034 to X.W.), the Swedish Research Council (2018-02267 and 2022-01019 to C. Zhu, and 2015-06276 and 2021-01950 to X.W.) and the Brain Foundation (FO2022-0120 to C. Zhu).

Author information

These authors contributed equally: Yangong Wang, Yiran Xu, Chongchen Zhou, Ye Cheng.

Authors and Affiliations

Children’s Hospital of Fudan University and Institutes of Biomedical Sciences of Fudan University, Shanghai, China

Yangong Wang, Ye Cheng, Yimeng Qiao, Yangyi Fan, Jin Zhang, Yu Su, Qiaoli Li, Lei Liu & Qinghe Xing

Department of Pediatrics, Henan Key Laboratory of Child Brain Injury and Henan Pediatric Clinical Research Center, The Third Affiliated Hospital and Institute of Neuroscience of Zhengzhou University, Zhengzhou, China

Yiran Xu, Lei Xia, Juan Song, Yimeng Qiao, Xiaoli Zhang, Ming Li, Xirui Peng, Bingbing Li, Yanyan Sun, Bohao Zhang, Tongchuan Li, Hongwei Li, Junying Yuan, Dengna Zhu & Changlian Zhu

Rehabilitation Department, Henan Key Laboratory of Child Genetics and Metabolism, Children’s Hospital of Zhengzhou University, Zhengzhou, China

Chongchen Zhou, Qing Shang, Chao Gao, Caiyun Ma & Nan Lv

Shanghai Center for Women and Children’s Health, Shanghai, China

Ye Cheng, Jin Zhang & Qinghe Xing

State Key Laboratory of Medical Genomics, Shanghai Institute of Hematology, National Research Center for Translational Medicine (Shanghai), and School of Life Sciences and Biotechnology, Shanghai Jiao Tong University, Shanghai, China

Department of Neurosurgery, The Affiliated Zhongshan Hospital of Fudan University, Shanghai, China

Department of Radiology, Neuroradiology Section, Kingston Health Sciences Centre, Queen’s University Faculty of Health Sciences, Kingston, Ontario, Canada

Andres Moreno-De-Luca

Robinson Research Institute and Adelaide Medical School, The University of Adelaide, Adelaide, South Australia, Australia

Alastair H. MacLennan & Jozef Gecz

Centre for Perinatal Medicine and Health, Institute of Clinical Science, Sahlgrenska Academy, University of Gothenburg, Gothenburg, Sweden

Xiaoyang Wang

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Contributions

Q.X., C. Zhu and X.W. were responsible for study conception, design, supervision and funding as well as analysis and interpretation of the data. Y.W., Y.C. and N.Q. were responsible for performing the ES, data acquisition, analysis and interpretation. Y.X., C. Zhou, Q.S., L.X., J.S., C.G., M.L. and D.Z. were responsible for cohort ascertainment, recruitment and phenotypic characterization. Y.W. drafted the manuscript and Q.X., C. Zhu, X.W., A.H.M., A.M.-D.-L. and J.G. revised the manuscript critically for important intellectual content. N.Q., Y.Q., X.Z., C.M., Y.F., X.P., S.W., N.L., B.L., Y. Sun, B.Z., T.L., H.L., J.Z., Y. Su, Q.L., J.Y., L.L. and D.Z. provided data, developed models, reviewed results and provided guidance on methods. All authors contributed and critically reviewed the final version of the manuscript.

Corresponding authors

Correspondence to Changlian Zhu or Qinghe Xing .

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Extended data

Extended data fig. 1 the correlation between cp phenotypes and genetic findings..

In order to determine the relationship between the different clinical phenotypes and the molecular diagnosis of individuals with CP, we performed clinical grouping of individuals, including classification, developmental profiles, and morphological examination. Global developmental delay/Intellectual Disability(GDD/ID), termed intellectual disability, was identified based on a score of less than 70 on the Bayley scales for measurement of the mental development index. The classification of CP shown in a includes groups with a large number of individuals: spastic quadriplegia, spastic diplegia, dyskinesia CP, and mixed types. The developmental profiles shown in b show complications of individuals with CP, such as speech disability. Shown in c are some imaging findings of CP, such as white matter injury and hydrocephalus. The results show that children with GDD/ID were closely related to heredity. The green is genetic diagnosis positive rate, and the gray is genetic diagnosis negative rate. The numbers on the top of the column represent the corresponding number of people. A two-side Fisher’s exact test was used in comparison of genetic diagnostic rate in specific CP subgroup. Bonferroni was used to handle multiple test problem. ‘*’ means P  < 0.05, ‘**’ means P  < 0.01, and ‘***’ means P  < 0.001.

Extended Data Fig. 2 Phenotype detection of nematode motility development.

After the strains L4440 and Escherichia coli containing target gene RNAi were fed to rrf-3 mutant nematode and TU3311 strain, we measured and analyzed the body length, body width, moving distance and speed. The differences were compared using a statistical method called the Log-rank(Mantel-Cox) Test. n = 50 worms for each case. ‘*’ means P  < 0.05, ‘**’ means P  < 0.01, and ‘***’ means P  < 0.001. Data are presented as mean values +/− SEM. a . Mean Worm Length (um): The body length showed no significant difference in nmpg-1 or tom-1 target gene RNAi bacteria feeding rrf-3 mutant nematode ( P  = 0.22 and 0.075, respectively), but the body length was increased when feeding TU3311 strain nematode ( P  = 0.044 and 0.0011 in TU3311). b . Mean Width (um): There was no significant difference in the body width of the mutant strains ( P  = 0.53 and 0.18 in rrf-3, and P  = 0.72 and 0.10 in TU3311). c . Speed (um/s): When the tom-1 target gene RNAi bacteria were fed to rrf-3 mutant nematode, the movement speed was significantly slower than that of the nematode fed by L4440 bacteria ( P  = 0.0002). When feeding tom-1 target gene RNAi bacteria to TU3311 strain nematode, the movement speed was faster than that fed by L4440 strain nematode ( P  = 0.011). For nmgp-1 RNAi bacteria fed rrf-3 mutant nematode and TU3311 strain nematode, there was no significant difference ( P  = 0.88 and 0.21, respectively). d . Track Length (um): When feeding tom-1 target gene RNAi bacteria to rrf-3 nematode, the distance was shorter than that fed by L4440 bacteria ( P  = 0.0002). When feeding tom-1 target gene RNAi bacteria to TU3311 strain nematode, the track length was longer than that fed by L4440 strain nematode ( P  = 0.011). There was no significant difference in distance when feeding nmgp-1 target gene RNAi bacteria ( P  = 0.88 and 0.21 in rrf-3 and TU3311 strain, respectively).

Extended Data Fig. 3 Knockdown of Tom and Bsg affects larval and adult locomotion.

a . Relative crawling speed of a single 3rd instar larval was measured as the number of squares (sqr) crossed during test for each genotype as indicated ( P  = 0.019 and 0.0002 for Bsg knockdown). n = 15 larvae for each case. b . Adult climbing locomotion was measured as the time for 50% flies to reach the threshold line (8 cm from the bottom) for different genotypes as indicated ( P  = 0.0062 and 0.0010 for Tom knockdown; P  = 0.0022 and 0.0043 for Bsg knockdown). n = 6 flies for each case. The differences were compared using a statistical method called the Log-rank (Mantel-Cox) Test. ‘*’ means P  < 0.05, ‘**’ means P  < 0.01, and ‘***’ means P  < 0.001. Data are presented as mean values +/− SEM.

Extended Data Fig. 4 Filtering process for annotated files.

The screening procedure according to the ACMG for (likely) pathogenic variants through annotated files is shown for a single sample. After deletion of some sites like high-frequency sites and synonymous sites, the preliminary analysis of the sites was performed through prediction software scoring. The remaining variants were further divided into categories based on the literature. All variants were verified by Sanger sequencing.

Extended Data Fig. 5 The KEGG pathway of the P/LP-variants-related genes and VUS-related genes.

a showed the KEGG pathway using P/ LP-variants-related genes (n = 218). And b is the KEGG pathway using VUS-related genes (n = 299). The size of the circle represents the number of genes in the pathway. Hypergeometric test was used in the pathway enrichment analysis and Benjamin and Hochberg method was applied to deal with the multiple correction problem. The color is the corrected p-value, and the darker the color, the more significant the p-value.

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Wang, Y., Xu, Y., Zhou, C. et al. Exome sequencing reveals genetic heterogeneity and clinically actionable findings in children with cerebral palsy. Nat Med (2024). https://doi.org/10.1038/s41591-024-02912-z

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Genetics causes 1 in 4 cerebral palsy cases

Imma Perfetto

Imma Perfetto is a science journalist at Cosmos. She has a Bachelor of Science with Honours in Science Communication from the University of Adelaide.

A large Chinese study has re-affirmed growing evidence that genetics and not birth asphyxia is often responsible for cerebral palsy.

“Birth asphyxia is a label that’s never been proven,” says Alastair MacLennan AO, Head of the Australian Collaborative Cerebral Palsy Research Group at the Robinson Research Institute at University of Adelaide, and co-author of the study published overnight in Nature Medicine .

“We are showing birth asphyxia is a myth.”

The world’s largest study of cerebral palsy (CP) genetics was conducted in China which had more than 1000 children in a studiable cohort.

The research found the mutations were significantly higher in CP cases labelled at birth with perinatal asphyxia – a lack of blood flow to a baby’s brain before, during, or right after birth.

The findings, which are consistent with smaller studies globally, indicate that “at least some portion of the perinatal asphyxia events seen in individuals with CP are very probably related to improper brain development due to underlying genetic variants,” according to the authors.

“24.5% of Chinese children in the study had rare genetic variations linked to cerebral palsy,” says co-author Jozef Gecz, Head of Neurogenetics at the University of Adelaide Medical School and the Robinson Research Institute.

“This revelation mirrors our earlier findings in our Australian cerebral palsy cohort, where up to one third of cases have genetic causes.

“Our research shows at least some babies who experience birth asphyxia and are diagnosed with CP may have improper brain development as a result of the underlying genetic variants rather than a lack of oxygen.

“Crucially, clinically actionable treatments were found in 8.5% of cases with a genetic cause. It is exciting to see how genetic pathways to cerebral palsy inform tailored treatments for these individuals.”

Cerebral palsy is the most common physical motor disability in children. It is estimated to affect 1.6 in every 1,000 live births globally.

In CP, damage or abnormalities inside the developing brain disrupt its ability to control movement and maintain posture and balance.

Symptoms often emerge during infancy and early childhood and can range from mild to severe. They also commonly occur in association with epilepsy, autism, and intellectual disability.

In a collaboration between Fudan  and Zhengzhou Universities in China, researchers conducted genetic sequencing on 1,578 Chinese children (505 girls and 1,073 boys) with CP. In the children who had experienced birth asphyxia they identified 81 genes with causative mutations.

All the genes are known to play important roles in neural and embryonic development. For example, 5 are associated with spastic paraplegia, 3 have been associated with neonatal encephalopathy; and 3 are involved in molecular pathways relevant to central nervous system development.  

“A lack of oxygen at birth is often claimed to be the cause of CP in medical litigation following a diagnosis and this has led to the presumption that the condition is preventable with better obstetrics or midwifery. This is simply not the case,” says MacLennan.

“These results highlight the need for early genetic testing in children with cerebral palsy, especially those with risk factors like birth asphyxia, to ensure they receive the right medical care and treatment.

“All children with cerebral palsy merit modern genetic screening as early and customised interventions really can make a difference and improve their long-term outcomes.”

Originally published by Cosmos as Genetics causes 1 in 4 cerebral palsy cases

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a Upper straps were designed to protect hip joints from displacement.

b Lower straps were designed to prevent coxa valga.

c Thigh straps were designed to prevent hip adduction.

d To maximize the preventive effect on hip joint displacement, the greater trochanter (d) should be located between the upper and lower straps.

e The round design was applied at the buttock area of the fabric to allow comfort when lying or sitting and to prevent movement of the orthosis.

f The hip brace compresses the capsule and ligaments around the hip joints where displacement occurs, thereby helping with normal alignment.

After 12 months, the migration index for both sides was significantly decreased from 37.4% to 34.6% in the intervention group and significantly increased from 30.6% to 40.1% in the control group ( P  < .001). Whiskers indicate range; top and bottom of the boxes, IQR values; dark line, median; diamond, mean.

Trial Protocol and Statistical Analysis Plan

eFigure. Clothing Pressure Measurements

eTable. Results of the Linear Mixed Model

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• Errors in Figures JAMA Network Open Correction December 1, 2022

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Kim BR , Yoon JA , Han HJ, et al. Efficacy of a Hip Brace for Hip Displacement in Children With Cerebral Palsy : A Randomized Clinical Trial . JAMA Netw Open. 2022;5(11):e2240383. doi:10.1001/jamanetworkopen.2022.40383

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Efficacy of a Hip Brace for Hip Displacement in Children With Cerebral Palsy : A Randomized Clinical Trial

• 1 Department of Physical Medicine and Rehabilitation, Anam Hospital, Korea University College of Medicine, Seoul, South Korea
• 2 Department of Rehabilitation Medicine, Pusan National University Hospital, Biomedical Research Institute, Pusan National University School of Medicine, Busan, South Korea
• 3 Research Institute of Human Ecology, Chungbuk National University, Chungju-si, South Korea
• 4 Chungbuk Technopark, Biocenter, Medical Device Health Team, Chungju-si, South Korea
• 5 Department of Rehabilitation Medicine, Seoul National University Bundang Hospital, Seoul National University College of Medicine, Seongnam, South Korea
• 6 Department of Rehabilitation Medicine, Jeju National University Hospital, Jeju National University College of Medicine, Jeju, South Korea
• 7 Department of Rehabilitation Medicine, Ewha Women’s University medical center, Ewha Woman’s University School of Medicine, Seoul, South Korea
• 8 SRC Rehabilitation Hospital, Gwangju-si, Gyeonggi-do, South Korea
• Correction Errors in Figures JAMA Network Open

Question   Can a newly designed hip brace prevent progressive hip displacement in children with nonambulatory cerebral palsy?

Findings   In this prospective, single-blinded randomized clinical trial including 66 patients with nonambulatory cerebral palsy (Gross Motor Function Classification System level IV or V), the Reimers migration index at 12-month follow-ups was significantly decreased in the group wearing hip brace but significantly increased in the control group.

Meaning   These findings suggest that this newly developed hip brace could be used more widely as a nonsurgical treatment option to prevent hip displacement in children with nonambulatory cerebral palsy.

Importance   There is no consensus on interventions to slow the progress of hip displacement in patients with cerebral palsy.

Objective   To investigate the efficacy of a novel hip brace in preventing progressive hip displacement in patients with cerebral palsy.

Design, Setting, and Participants   This 2-group randomized clinical trial was conducted at 4 tertiary hospitals in South Korea from July 2019 to November 2021. Participants included children aged 1 to 10 years with nonambulatory cerebral palsy (Gross Motor Function Classification System level IV or V). Block randomization was used to assign an equal number of patients to the study and control groups via computerized random allocation sequences. Data were analyzed from November to December 2021.

Interventions   The intervention group wore the hip brace for at least 12 hours a day for the study duration (ie, 12 months). Follow-up evaluations were performed after 6 and 12 months of wearing the brace. Both groups proceeded with conventional rehabilitation therapy during the trial.

Main Outcomes and Measures   The primary outcome was the Reimers migration index (MI) on radiography, as assessed by 3 blinded investigators. Primary outcome variables were analyzed using linear mixed models. Secondary outcomes include change in the Caregiver Priorities & Child Health Index of Life with Disabilities, on which lower scores indicate better quality of life.

Results   A total of 66 patients were included, with 33 patients (mean [SD] age, 68.7 [31.6] months; 25 [75.8%] boys) randomized to the intervention group and 33 patients (mean [SD] age, 60.7 [24.9] months; 20 [60.6%] boys) randomized to the control group. The baseline mean (SD) MI was 37.4% (19.3%) in the intervention group and 30.6% (16.3%) in the control group. The mean difference of the MI between the intervention group and control group was −8.7 (95% CI, −10.2 to −7.1) percentage points at 6 months and −12.7 (95% CI, −14.7 to −10.7) percentage points at 12 months. The changes in the Caregiver Priorities & Child Health Index of Life with Disabilities were favorable in the study group and reached statistical significance at the 6-month follow-up compared with the control group (difference, −14.2; 95% CI, −25.2 to −3.3).

Conclusions and Relevance   In this randomized clinical trial, the novel hip brace was significantly effective in preventing the progression of hip displacement, compared with the control group. It effectively improved quality of life in patients with nonambulatory cerebral palsy. Therefore, hip brace use could be a promising treatment method to delay hip surgery and improve the quality of life of patients with nonambulatory cerebral palsy.

Trial Registration   ClinicalTrials.gov Identifier: NCT04033289

Many children with cerebral palsy present with various musculoskeletal deformities associated with poor biomechanical alignment during growth. 1 - 3 Among them, progressive hip displacement (Reimers migration index (MI) >30%-33%) is the second most common musculoskeletal deformity. 4 , 5 Gradual deterioration of MI is seen in these children, from 3.9% per year at Gross Motor Function Classification System (GMFCS) level IV to 9.5% at level V. 4 - 8 Hip displacement may cause pain; fixed deformity; pelvic obliquity; scoliosis; loss of ability to sit, stand, and walk; and difficulty with dressing, bathing, and perineal care; these significantly impact function and quality of life (QOL). 9

Several hip surveillance programs for the early identification of and intervention for children with hips at risk have been established to prevent hip displacement and the need for complex salvage surgery. 6 , 10 - 13 However, there is conflicting evidence for conservative management to prevent hip displacement in patients with nonambulatory cerebral palsy. 9 Therefore, effective treatment for hip displacement in children with cerebral palsy has mainly focused on surgery. 10 , 12 , 14

Meanwhile, several studies regarding nonsurgical treatment for hip displacement, including various types of hip abduction braces, postural alignment seating systems, and botulinum toxin injection, have reported inconsistent results. 15 - 20 In previous research, we showed that a seating system with medial knee support could act as a fulcrum, thereby accelerating progressive hip displacement in patients with nonambulatory spastic cerebral palsy. 21 In a follow-up study, the electromyographic activity of adductor muscles was significantly decreased after introducing a dynamic hip compression bandage, suggesting potential benefits. 22 Theoretically, hip compression bandages biomechanically stabilize and assist in the protective function of the ligament and capsule around the hip joints.

Our hypothesis is that a hip brace can stabilize the hip joints, reduce hip adductor activation, and assist the ligaments and capsule in protecting the hip joint, thereby decreasing the progression of hip displacement. Therefore, we aimed to investigate the efficacy of a newly designed hip brace in preventing progressive hip displacement in patients with nonambulatory cerebral palsy.

This study was a multicenter, prospective, single-blinded randomized clinical trial, conducted from July 26, 2019, to November 30, 2021, at the rehabilitation units of 4 teaching hospitals in South Korea. The study protocol was approved by the institutional review boards of each hospital, and all methods were performed in accordance with relevant guidelines and regulations. The trial protocol and statistical analysis plan are shown in Supplement 1 . All patients or their representatives provided written informed consent prior to participation. This study is reported following the Consolidated Standards of Reporting Trials ( CONSORT ) reporting guideline.

The inclusion criteria for participation were (1) diagnosis of cerebral palsy, (2) age 1 to 10 years, (2) GMFCS 23 levels IV or V, (3) quadriplegia or diplegia, and (4) written consent with permission of the child and caregiver. The exclusion criteria were patients who had undergone a hip joint surgery, were scheduled to receive surgery during the trial, or had received botulinum toxin injections in their hip muscles within the 3 months before the study commencement or within the study duration.

Figure 1 shows the flow of participants through the trial. In this study, 1 participant refused to undergo the baseline evaluation after being assigned to the control group. Therefore, 66 participants (33 in the intervention group and 33 in the control group) were initially included. In the intervention group, 8 participants dropped out at the 6-month follow-up due to brace sizing issues (3 participants), visit problem (3 participants), and surgery (2 participants). At the 12-month follow-up, 4 more patients dropped out for the same reasons. In the control group, 1 participant dropped out due to a visit problem at the 6-month follow-up and 3 more participants dropped out due to visit problems (1 participant) and refusal (2 participants) at the 12-month follow-up ( Figure 1 ).

Block randomization was used to randomly allocate participants in a 1:1 ratio to the intervention or control group via computerized random allocation sequences prepared by a statistician (random block size, 4). Eligible participants were randomly assigned immediately after baseline assessment. The randomization schedule and group allocation could only be accessed by the statistician and physical therapist (S.L.). Investigators involved in outcome assessment were blinded to the group allocation (B.R.K., J.A.Y., and J.L.).

The hip brace was developed for research and approved by the South Korean Ministry of Food and Drug Safety as a Class 1 medical device. The brace is composed of inner pants and outer fabric bands. The inner pants and Velcro make the brace easy to put on and take off. The outer fabric bands comprise of 3 elements (upper, lower, and thigh straps). The upper straps were designed to protect hip joints from displacement, and the lower straps were designed to prevent coxa valga. The thigh straps were designed to prevent hip adduction ( Figure 2 A). To maximize the preventive effect on hip joint displacement, the greater trochanter should be located between the upper and lower straps. Figure 2 B and C shows radiographs of the hip before and after wearing the brace. The hip brace compresses the capsule and ligaments around the hip joints where displacement occurs, thereby helping with alignment.

Using the hip brace prototype (based on the age 5 years), the mean value of the 3 measurements of clothing pressure in 7 pressure areas were evaluated. The clothing pressure measurement method was based on the European Committee for Standardization, 2001, and the pressure class is divided into 4 grades (I, II, III, and IV) according to the compression force. Pressure was measured using an air-injected clothing pressure sensor and measuring instrument TNL-AMI 3037 (AMI Techno) (Pressure Class III or higher can be applied as a medical device). 24 The results showed compression pressure higher than level III class on upper, lower, and thigh straps (eFigure in Supplement 2 ).

After enrollment, all participants received clinical and radiographic evaluation. Clinical evaluation included hip abduction range of motion (ROM) at 0° and 90° hip flexion. For radiographic evaluations, total hip anterior posterior (AP), bilateral femur lateral, and whole-spine AP radiographs were obtained in the supine position, with bilateral hip and knee extension, without wearing the brace. MI was measured using picture archiving and communication system (PACS) image analysis. The MI is the percentage of the femoral head that lies outside the acetabulum. 21 , 25 The radiographic evaluations were performed by 3 blinded examiners, and the mean values were used for our study. 25 After randomization, the study group wore the hip brace for at least 12 hours a day during the study period (ie, 12 months). Follow-up evaluations were performed after 6 and 12 months of wearing the brace. Both groups proceeded with conventional rehabilitation therapy during the trial.

The primary outcome was the MI at 12 months. The secondary outcomes included MI at 6 months, hip ROM at 0° and 90° hip flexion, Cobb angle, pain intensity, QOL of the patients and their caregivers, and their satisfaction scores for the brace. Cobb angle was measured to assess the effect of hip brace on scoliosis. A visual analog scale (VAS; range, 0-10, with 0 indicating no pain and 10, worst pain) was used to measure the pain intensity and the Caregiver Priorities & Child Health Index of Life with Disabilities (CPCHILD; lower values indicate better QOL) was used to evaluate the change in QOL. 26 A Likert scale was used to assess the satisfaction scores for the hip brace (range, 1-5, with 1 indicating very satisfied and 5, very unsatisfied). 27 Because patients with nonambulatory cerebral palsy may find it difficult to communicate, the VAS and CPCHILD were evaluated by asking the caregivers’ perceptions of what the patients were feeling.

The calculation of the sample size was based on a previous epidemiological study. 21 In this study, the annual progression of MI in patients with nonambulatory cerebral palsy was measured to be a mean (SD) of 7.83% (8.73%) per year. Assuming that the MI decreased by 80% when using the hip brace, the MI of the study group was set to a mean (SD) of 1.57% (8.73%). With an α < .05 in the 2-tailed tests and a power of 80%, the target sample size of each group was 64 patients (32 in each group). Considering a dropout rate of 5%, the final sample size was determined to be a total of 68 patients.

Descriptive statistics were used to summarize participant characteristics using mean (SD) or number (percentage) as appropriate. Continuous data were assessed for skewness by visual inspection and using a normality test. Unadjusted mean (SD) values were computed for the primary and secondary continuous variables at baseline and 6 and 12 months of follow up.

There was excellent agreement for MI among the 3 outcome assessors (interclass correlation coefficient: 0.989). The t test and χ 2 tests were used to examine the baseline group differences. Analyses of the primary outcome variables were undertaken using linear mixed models, as specified in the study protocol, with age, baseline MI, groups, and time as fixed variables and patients, center, and side as a random effect using an unstructured covariance structure. In the case of dropout due to surgery and other reasons, data after dropout were excluded from the analysis. For the intention-to-treat analyses, all available data at baseline and 6 and 12 months were used. Mean differences at 6 and 12 months were estimated by the group by time interaction term, with associated 95% CIs and P values. A negative mean difference was indicative of better outcome values in terms of the MI. As there were interactions between group and time factors ( P  < .05), the model including the interaction was used as the final model. All data were analyzed using R statistical software version 4.1.1 (R Project for Statistical Computing). A 2-sided 5% level of significance was used throughout the analyses. Data were analyzed from November to December 2021.

Table 1 shows the baseline characteristics of all participants. A total of 66 patients were included, with 33 patients (mean [SD] age, 68.7 [31.6] months; 25 [75.8%] boys) randomized to the intervention group and 33 patients (mean [SD] age, 60.7 [24.9] months; 20 [60.6%] boys) randomized to the control group. The mean (SD) baseline MI (mean value between the right and left sides) was 37.4 (19.3) in the intervention group and 30.6 (16.3) in the control group. There were no statistically significant differences in the demographic data, except the baseline MI between the groups.

The changes in the evaluated variables between groups are presented in Table 2 and Figure 3 . The MI of the intervention group was significantly decreased by a mean (SD) −2.7 (6.9) percentage points at 6 months and −3.3 (6.9) percentage points at 12 months (mean [SD] annual progression rate: 6 months, −5.4 [13.8] percentage points; 12 months, −3.3 [6.9] percentage points; P  < .001). However, the MI of the control group was significantly increased by a mean (SD) of 5.9 (7.4) percentage points at 6 months and 9.4 (10.9) percentage points at 12 months (mean [SD] annual progression rate: 6 months, 11.8 [14.8] percentage points; 12 months, 9.4 [10.9] percentage points; P  < .001). The mean differences in the MI between groups was −8.7 (95% CI, −10.2 to −7.1) percentage points at 6 months and −12.7 (95% CI, −14.7 to −10.7) percentage points at 12 months.

Based on a linear mixed model, the regression coefficient for the MI in the intervention group was −2.7 at 6 months and −3.7 at 12 months. In the control group, the MI regression coefficient was 5.8 at 6 months and 9.4 at 12 months (P < .001) (eTable in Supplement 2 ).

The CPCHILD was favorable in the intervention group and reached statistically significant levels at the 6-month follow up compared with the control group (difference, −14.2; 95% CI, −25.2 to −3.3; P  = .01) ( Table 2 ). Although the statistical significance of the CPCHILD disappeared, the change was still favorable for the study group at the 12-month follow-up ( Table 2 ). Although not statistically significant, the overall pain score was more favorable for the study group at the 6- and 12-month follow-ups than for the control group ( Table 2 ). The mean (SD) Likert scale score for satisfaction for the brace was 2.9 (0.8) at 6 months and 2.7 (0.8) at 12 months. Other clinical parameters, such as hip and knee ROM, were not significantly different between groups at the 6- and 12-month follow-ups ( Table 2 ).

This randomized clinical trial found significant improvement of hip displacement in the intervention group compared with the control group. The results of this study are meaningful because the hip brace used in this study can provide significant treatment to delay surgery and improve quality of life, in relation to hip displacement in patients with nonambulatory cerebral palsy.

As risk factors of hip displacement, it is well established that hip displacement is more frequent in quadriplegia, presence of spasticity, GMFCS levels IV and V, anomalous femoral geometry (increased femoral anteversion and neck-shaft angle), and acetabular dysplasia. 4 , 7 , 13 , 28 , 29 In the presence of hip adductor spasticity, hip displacement was shown to be aggravated nearly 2-fold by the hip abduction bar due to leverage effects and length tension. 21 , 22 Biomechanically, increased adduction forces on the hip joint with an abduction bar are thought to create the torque on the femoral head, shifting it laterally out of the acetabulum. 21 , 22 , 30 In previous studies, a direct relationship has been established between hip displacement and GMFCS levels. In other words, patients with nonambulatory cerebral palsy without spasticity also develop hip displacement. 4 , 13 , 14 Since the ligaments and capsule, which act to protect hip joints in children, are very weak, they may loosen if forces continuously act to stretch these structures in various daily activities, and displacement of the hip joint occurs. If the displacement state persists, the condition is not reversed by pulvinar formation. 31 The hip brace in this study was developed to reinforce the protection for the ligaments and capsule to prevent progression of hip displacement.

Interestingly, the displacement of the study group significantly improved. This may be because the laxity of the ligaments and capsule precedes the displacement, but the laxity is reversible, and the ligaments and capsule are tightened when compressed. Owing to this laxity, the MI changed every time plain radiographs were acquired and even improved after botulinum toxin injection. 16 , 32 Therefore, hip braces can be used as a promising treatment method.

Until now, there has been no definite recommendation for hip brace to slow the progression of hip displacement. 9 A hip abduction brace was used to prevent hip displacement. 33 However, the origin of this brace started with an explanation of the concept and it was used in clinical practice without clear study. 34 , 35 However, in previous studies, the hip abduction brace combined with botulinum toxin injection was shown to be ineffective in preventing hip displacement. 15 , 16 , 19 These results may be attributed to the inclusion of patients with lower risk of displacement (GMFCS levels I-III), the selection of incidence of surgery as the primary outcome, ineffectiveness of the previous abduction brace, and botulinum toxin injection to hamstring rather than the hip adductors muscles. 15 , 16 , 19 The spasticity of the adductor muscles at hip joints should be distinguished from the spasticity of the hamstring muscle at the knee joints, but it was not distinguished in previous studies. 15 , 16 , 19 These studies 15 , 16 , 19 used an unverified abduction brace together with botulinum toxin, which resulted in an erroneous conclusion that both were ineffective.

As multiple factors are associated with hip displacement, complex treatments targeting the diverse mechanisms of hip displacement can maximize the efficacy of hip protection and reduce complications or the need for surgery. 28 , 32 , 36 Theoretically, we can apply weight-bearing exercises in a standing position to activate the hip abductors and stimulate the acetabulum. These methods might additionally be effective to prevent coxa valga, femoral anteversion, and acetabular dysplasia. 28 , 36 To control adductor spasticity, we can apply botulinum toxin injection at the adductors or adductor tenotomy. A 2021 study 20 reported that a botulinum toxin injection repeated at 6 months in these muscles significantly reduced muscle tone by 40% at 1, 2, 3, and 7 months, which remained below baseline levels at 12 months, and the progression of the hip MI was significantly lower than that of the control group. Furthermore, neurogenic denervation after repetitive botulinum toxin injection can result in permanent decrease in muscle contractility. 37 Wearing a hip brace during ambulation or vibration therapy can maximize the efficacy of and minimize the complication rate due to excessive force at the hip joints.

Hip displacement and related surgery can significantly impact function and QOL in patients and their caregivers. 38 , 39 However, after applying the brace, the patients in the intervention group had higher QOL scores at 6 months compared with the control group. Prevention of surgery can further relieve the burden on patients and caregivers. Based on these results, it is necessary to amend the research on hip displacement with cerebral palsy, which previously focused on surgical treatment, to new conservative treatments focused on prevention. In addition, further study with long-term follow up is needed to determine whether using a hip brace can delay surgery and improve quality of life.

This study has some limitations. First, the dropout rate was higher than expected. We started with the representative brace size for patients aged 3, 5, and 6 years, but some participants grew faster than expected and could not wear the brace. In addition, there were difficulties in patient enrollment and follow-up due to COVID-19. Although 3 patients dropped out after undergoing surgery, the MI values of these 3 patients did not change, and surgery was performed by the guardians’ decisions. Second, hip ROM and pain did not show a significant change. Meaningful outcomes may have been observed if a longer follow-up was performed. Third, during the block randomization, MI was not included as a factor; therefore, there were statistically significant differences in baseline data between the groups. Fourth, although it was recommended that the braces be worn for at least 12 hours every day, the actual wearing time was not measured.

In this randomized clinical trial, the hip brace was effective in preventing hip displacement aggravation. It effectively slowed and improved displacement and improved QOL in patients with cerebral palsy. Therefore, brace use could comprise a promising treatment method to delay hip surgery in patients with cerebral palsy.

Accepted for Publication: September 20, 2022.

Published: November 4, 2022. doi:10.1001/jamanetworkopen.2022.40383

Correction: This article was corrected on December 1, 2022, to fix errors in Figure 1 and Figure 2C.

Open Access: This is an open access article distributed under the terms of the CC-BY License . © 2022 Kim BR et al. JAMA Network Open .

Corresponding Author: Ju Seok Ryu, MD, PhD, Department of Rehabilitation Medicine, Seoul National University Bundang Hospital, Seoul National University College of Medicine, 82 Gumi-ro 173 Beon-gil, Bundang-gu, Seongnam-si, Gyeonggi-do 13620, South Korea ( [email protected] ).

Author Contributions: Ms Cho and Dr Beom had full access to all of the data in the study and take responsibility for the integrity of the data and the accuracy of the data analysis. Drs Kim and J. A. Yoon contributed equally to this paper and should therefore be regarded as equivalent first authors.

Concept and design: Han, Y. I. Yoon, Park, Ryu.

Acquisition, analysis, or interpretation of data: Kim, J. A. Yoon, Lim, S. Lee, Cho, Shin, H. J. Lee, Suh, Jang, Beom, Choi.

Drafting of the manuscript: Kim, J. A. Yoon, Han, Y. I. Yoon, S. Lee, Suh, Jang, Park, Choi, Ryu.

Critical revision of the manuscript for important intellectual content: Kim, Lim, Cho, Shin, H. J. Lee, Beom.

Statistical analysis: Lim.

Obtained funding: Cho, Ryu.

Administrative, technical, or material support: Han, Y. I. Yoon, Lim, H. J. Lee, Jang, Beom, Park, Choi.

Supervision: Suh, Ryu.

Conflict of Interest Disclosures: Drs Han, Yoon, and Ryu reported owning patent No. 1023060520000 and patent No. PCT/KR2020/015622. Dr Ryu reported owning patent No. 1019812070000. After conducting research and submitting a manuscript, the patents were transferred to RS Rehab, and compensation for the patent was received. No other disclosures were reported.

Funding/Support: This research was supported by grant a from the Korea Health Technology R&D Project through the Korea Health Industry Development Institute, funded by the Ministry of Health & Welfare, Republic of Korea (grant No. HI18C1169).

Role of the Funder/Sponsor: The funders had no role in the design and conduct of the study; collection, management, analysis, and interpretation of the data; preparation, review, or approval of the manuscript; and decision to submit the manuscript for publication.

Disclaimer: No commercial party having a direct financial interest in the results of the research supporting this article has or will confer a benefit upon the authors or on any organization with which the authors are associated.

Data Sharing Statement: See Supplement 3 .

Additional Contributions: We thank the Medical Research Collaborating Center team of Seoul National University Bundang Hospital for their work in the area of statistics in this study.

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Sporadic pediatric vestibular schwannoma: a case report in a 4-year-old boy

• Case Report
• Published: 06 May 2024

Cite this article

• Cheng-Chieh Tsai 1 ,
• Chia-Lang Fang 2 , 3 ,
• Minhua Liao 4 ,
• YiShan Yang 5 ,
• Kevin Li-Chun Hsieh 1 , 4 , 6   na1 &
• Tai-Tong Wong 4 , 5 , 7   na1  

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Sporadic vestibular schwannomas (VSs) are rare in children. When occurred in the pediatric population, they usually appear bilaterally and are related to neurofibromatosis type 2 (NF2). The current study reports a 4-year-old boy without family history of VS or NF2 who presented with a large (5.7-cm) VS involving the right cerebellopontine angle and internal auditory canal. Through seven-staged surgical interventions and two stereotactic γ‑knife radiosurgery, the disease was stabilized. At 2-year follow-up, the child had right ear hearing loss, grade IV facial palsy, and normal motor function and gait. No definite evidence of gene mutation regarding NF2 can be identified after sequence analysis and deletion/duplication testing. This case highlights the significance of considering the possibility of sporadic VSs, even in very young children. It emphasizes the importance of not overlooking initial symptoms, as they may indicate the presence of a large tumor and could potentially result in delayed diagnosis.

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Authors Kevin Li-Chun Hsieh and Tai-Tong Wong contributed equally to this work.

Authors and Affiliations

Department of Medical Imaging, Taipei Medical University Hospital, Taipei City, Taiwan

Cheng-Chieh Tsai & Kevin Li-Chun Hsieh

Department of Pathology, School of Medicine, College of Medicine, Taipei Medical University, Taipei City, Taiwan

Chia-Lang Fang

Department of Pathology, Taipei Medical University Hospital, Taipei Medical University, Taipei City, Taiwan

Pediatric Brain Tumor Program, Taipei Cancer Center and Taipei Neurological Institute, Taipei Medical University, Taipei City, Taiwan

Minhua Liao, Kevin Li-Chun Hsieh & Tai-Tong Wong

Division of Pediatric Neurosurgery, Department of Neurosurgery, Taipei Medical University Hospital, Taipei City, Taiwan

YiShan Yang & Tai-Tong Wong

Department of Radiology, School of Medicine, College of Medicine, Taipei Medical University, Taipei City, 110301, Taiwan

Kevin Li-Chun Hsieh

Graduate Institute of Clinical Medicine, College of Medicine, Taipei Medical University, Taipei City, Taiwan

Tai-Tong Wong

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KH and TTW conceptualized and designed the study. KH, TTW, CLF, and ML analyzed and interpreted the data. CLF and ML performed the sample processing, extracting, and sequencing experiments. CCT, CLF, and KH drafted the initial manuscript. CCT, YY, KH, and TTW reviewed and revised the manuscript. All authors approved the final manuscript as submitted and agreed to be accountable for all aspects of the work.

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Tsai, CC., Fang, CL., Liao, M. et al. Sporadic pediatric vestibular schwannoma: a case report in a 4-year-old boy. Childs Nerv Syst (2024). https://doi.org/10.1007/s00381-024-06398-5

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DOI : https://doi.org/10.1007/s00381-024-06398-5

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Genetic defects, not lack of oxygen cause cerebral palsy in children: Study

Cerebral palsy is a disorder affecting one's ability to move. it is the most common motor disability in children, with symptoms emerging in infancy and early childhood.

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