Cerebral palsy (CP) is an umbrella term used to describe a group of permanent, fluctuating disorders of movement, posture and motor function caused by a non- progressive lesion or abnormality in the immature brain (Sadowska et al 2020).

Motor functions are core symptoms; however, they are frequently accompanied by epileptic, sensory, perceptual, cognitive, communicative and behavioural disorders (Sadowska et al 2020).

The International Classification of Functioning, Disability and Health (ICF) model can be applied as a framework for management of a person with CP.

This model emphasises the person through categorising by participation role and by how people interact with their health, context and community activities rather than by disease.

****In 2020, an ICF Core Set was developed for adults with CP by global experts (Limsakul et al 2020), as depicted in the ICF Core Set Table BELOW****.

The ICF model has been further explored using the experiences of adults with CP to increase our understanding of areas of concern.

Pain and movement were cited as the most frequent concerns for body structure and function, while mobility, employment and self-care were cited as the most frequent for activity and participation (Noten et al 2021).

Several factors may affect movement control in an individual with CP.

Movement strategies and control are altered because of primary motor-neural control dysfunction (eg, activation capacity and strategies) (Rose & McGill 2005, Stackhouse et al 2005) and secondary motor-neural control dysfunction (eg, disuse, reduced muscular reserve) from delayed developmental milestones (Shortland 2009).

Both lead to altered muscle activation and reserve and increase the development of premature sarcopaenia (Skoutelis et al 2020, Himuro et al 2018).

Children with CP also show morphological changes in their skeletal muscle—increased collagen and reduced muscle volume, cross-sectional area and muscle belly length (Skoutelis et al 2020, Howard & Herzog 2021)—which make muscles inelastic, thinner and shorter, with longer tendons.

Consequently, as bone length elongates with human growth, contractures may occur.

This altered movement control leads individuals with CP to have an increased risk of mobility decline, particularly in their third or fourth decade of life (Himuro et al 2018, Opheim et al 2013).

While muscle weakness is a strong predictor of overall motor function (Himuro et al 2018, Gjesdal et al 2020), it is less clear whether strengthening programs produce clinically relevant change (Ross et al 2016, Fleeton et al 2020).

This may be due to the multifaceted factors and heterogeneity of people with CP, necessitating a broader framework and multimodal approach to improve outcomes.

Case introduction and consent

This case report illustrates a typical case of an adult with CP and the impact of neurological physiotherapy on key ICF domains.

It proposes an intensive and multimodal intervention approach that includes body structure and activity-based interventions to influence change in balance, walking and confidence.

Patient consent was obtained to use specific information and details in this case study.

Clinical findings—subjective

BK is a 54-year-old woman with spastic diplegic CP, classified as Gross Motor Function Classification System Level II.

She self-referred for decline in mobility (self-reported slower walking speeds, reduced balance and use of walking poles with low confidence).

She reported that her mobility decline was gradual, from using a walking stick 10 years ago to her current use of two walking poles when walking indoors and outdoors.

BK received surgery as a child for bilateral equinovarus contracture and in 2011 underwent bilateral rectus femoris and Achilles tendon lengthening.

BK is independent with all activities of daily living, including walking three times weekly for 500 metres, averaging 2.5 kilometres per hour.

BK does not experience pain or physical fatigue while walking; however, she is concerned about falls and reports falling once every two to three months, resulting in lower limb bruising.

Her 24-month goals are:
•    to be able to walk independently, without walking poles, indoors at home and at work for two hours a day
•    to be able to walk independently up and down stairs without walking poles and without a rail
•    to be able to walk independently with her two walking poles at an average speed greater than three kilometres per hour for two kilometres.

Clinical findings—objective

Body structure function and impairments Strength was measured with the Medical Research Council’s manual muscle testing, while range of movement (ROM) was measured with goniometry in supine.

The Modified Tardieu Scale is considered a reliable measure of spasticity (Ben-Shabat et al 2013) and was used as per the Olveret al testing protocol (Olver et al 2019).

****Her presentation is characterised by globallower limb weakness, reduced active ROM bilaterally and low levels of spasticity (see Tables 1 and 2).

Lower limb light touch localisation and pressure sensation were normal, with severe static joint position proprioceptive loss in the feet only.

Activity and participation measures

Valid and reliable measures specific to people with CP have been used where available.

****Outcome measures were captured in non-modifiable footwear and reported in Table 3.

1.    Timed single-leg stance was captured on each leg without gait aids, recorded in seconds. This measure correlates with the biomechanics of gait and provides useful information relating to anticipatory motor control (Bonora et al 2017).
2.    The Mini-BESTest is a 14-item test that captures quiet and dynamic balance with a maximum score of 32—a higher score indicating better balance (Saether et al 2013). It provides information about components influencing motor control: anticipatory and reactive, sensory orientation and dynamic gait.
3.    The timed 10-metre walk test (fast) is a measure of walking speed over 10
metres, captured with gait aids (Bahrami et al 2017). A 14-metre path was used, with the time captured over the middle 10 metres in seconds.
4.    The timed six-minute walk test is a measure of walking distance over six minutes, used with gait aids. An even
footpath was used and distance was captured to the nearest metre.
Patient-reported outcomes (questionnaires available from authors on request):
5.    The Ambulatory Self-Confidence Questionnaire, available at tinyurl.com/ ymk8tkxw, is a patient-reported 22-item questionnaire that captures ambulatory confidence in different environments and contexts. Each question is rated out of 10, with higher scores indicating greater confidence.
6.    The Falls Efficacy Scale International is a patient-reported 16-item questionnaire
(with a maximum score of 64) that captures an individual’s concern about falling during general activities and participation. Higher scores indicate greater concern (16–19
low concern, 20–27 moderate concern and 28–64 high concern).

Working hypothesis and clinical reasoning

Current evidence on clinical decision frameworks in CP highlight the need for health professionals to screen, identify, manage and refer on for a breadth of issues (Morgan et al 2016) and seek to adopt a hypothesis-oriented algorithm within the ICF model to solve problems (Franki et al 2014).

Understanding the biomechanical requirements of the functional task in BK’s goals enables a hypothesis about body structure dysfunctions to be explored.

Gait and stair climbing require the ability to create a reciprocal single-leg stance with sufficient joint/muscle active range and control.

BK’s movement diagnosis and analysis revealed significant muscle weakness, reduced motor control and altered anticipatory and reactive balance reactions.

The altered motor control may be influenced by spasticity, reduced proprioception and limited ROM.

Keegan Bow.

Gait speed can be increased by increasing cadence and stride length.

To achieve improvements in speed, body structure must be adequate to allow larger ranges of dynamic joint motion along with higher levels of muscle power and motor control with sequencing of relevant muscles.

Therefore, a treatment priority would be to increase BK’s ROM and potentially therefore stride length.

Gait training, while important in addressing hip musculature for stability, also requires a specific emphasis on key muscle groups such as the dorsiflexors and plantarflexors, trained to lengthen and contract at high power output within the context of their phase in the gait cycle (Williams et al 2019).

This is consistent with effective gait training and more modern approaches to gait training in individuals with CP (Booth et al 2018, Torberntsson 2020, Kalkman et al 2019).

However, given the patient’s profound and longstanding impairments, it was hypothesised that a highly specific, intensive and multimodal approach would address the layers of complexity to effect change at body structure and activity levels and create a window of opportunity to target motor behaviour and confidence at participation levels.

Therapeutic intervention

A two-week intensive treatment period was implemented to create a burst of body structure adaption and motor relearning, spanning 15 hours of intervention.

Assessments occurred at day 0, 14 and 35 (three weeks post intervention completion).

Week 1 had five days of 90-minute sessions per day. Week 2 had three days of twice-daily 90-minute sessions, separated by a 45-minute break.

A home exercise program was scripted for independent continuance at the end of the two-week block.

The physiotherapy interventions included:
•    joint mobilisation (Youn et al 2020) and musculotendinous stretching in the lower limbs (Deschenes & Kraemer 2002) to increase hip, knee and ankle joint range, prior to progressive strength training in lying, sitting and standing positions
•    activities to increase sensory and proprioceptive input: trampoline bouncing, heel drops on decline board and backward stepping with minimal arm support (with manual guidance as needed)
•    activities in standing that challenged limits of stability and reactive balance 
•    ballistic and plyometric training of key muscle groups such as the hip abductors and extensors, knee flexors and extensors and ankle plantarflexors
•    part-practice of phases of gait sequences (with manual guidance as needed) with reduced visual feedback and fading verbal cues to increase implicit motor learning
•    task-specific gait practice on a treadmill and overground walking.

The environmental set-up was considered to reduce the effort required of very weak and disused muscles, gradually adding load, tempo and multi-joint sequencing to increase synergies of movement contextual to gait.

Rests were minimised to enhance fatigue resistance.

A home exercise program of standing balance, core-based and walking exercises, matched to the highest level of motor performance achieved in the final session, was provided at the conclusion of the two-week period, with a summary of progress, performance and results.

Results

Overall strength improved in the lower limbs, with the greatest increases noted in ankle plantarflexors (grade 4 from grade 2), invertors (grade 2–3 from grade 1–2), evertors (grade 2–3 from grade 0–2) and first metatarsophalangeal flexors and extensor (grade 4 and 3 respectively from grade 1–2). Active ROM improved most notably at the hips for flexion (36–63 degrees) and external rotation (21–32 degrees) and at the ankles for plantarflexion (12–20 degrees).

Mainly left R2 and R1 scores in psoas major, adductor longus, rectus femoris, semitendinosus/semimembranosus, gastrocnemius and soleus increased. There were small to no changes to spasticity muscle reactions.

Using the Ambulatory Self-Confidence Questionnaire, activity outcomes noted improvements for ambulatory confidence without poles post intervention (+1.05 mean from 0) and timed single-leg stance (+1.27 seconds from unable). Other outcomes had noted improvements at the three-week follow-up: the Mini-BESTest (+5), the timed 10-metre walk test (+1.1 metres per second) and the timed six- minute walk test (+34 metres).

There were no adverse reactions reported.

Discussion

Intensive, neuro-adaptive and multimodal physical intervention improved balance and gait function in an adult with CP, but not falls confidence. BK regained the ability to walk indoors without aids, a motor skill lost 10 years earlier.

A minimally clinically important difference of 0.1 metres per second for gait speed was only achieved at the three-week follow-up under both walking conditions.

Walking faster provides evidence of improved biomechanical movement efficiency through strength and motor control (Deschenes & Kraemer 2002).

Walking without an aid also has a clinical impact as it liberates the upper limbs for function and introduces the possibility of upper and lower limb motor tasks.

These clinical translations are congruent with sub-analysis of the Ambulatory Self-Confidence Questionnaire data, which demonstrated significant increases in confidence when walking without poles in item 15 (carrying small items while walking—score 0, now 7), item 20 (walking from one room to another in the house—score 0, now 9) and item 21 (walking a short distance without stopping—score 0, now 7).

All other items of the questionnaire pertaining to higher-level mobility showed no change.

This may be due to the limitations of this short intervention period or to the level of interventions being functionally too low to affect performance in other items.

Over the two-week period, strength and active ROM improved, follow-up scores of walking and balance improved and there were minimal effects on low pre-intervention levels of spasticity.

Due to the short time frame, immediate improvements in strength are unlikely to be related to hypertrophy (Hryvniak et al 2021) but rather perhaps to changes in motor recruitment and movement pattern consistency (Rahlin et al 2020).

Increases in walking and balance outcomes at follow-up were larger than those noted in the post-intervention scores.

This result was surprising because it was hypothesised that motor performance would peak immediately after the intervention, when motor recruitment and active range had been maximised during intensive motor training, and reduce over time due to the reduced intensity of input.

This may be due to the post-intervention home exercises program providing ongoing practice of motor skills acquisition (walking speed, balance and endurance) and the opportunity for motor planning practice, consolidation and transfer of skills to real-life conditions.

It highlights the importance of reflecting on all aspects of clinical practice and on whether the intensity was a necessary precursor to the enhanced home program effects.

While results are promising, it is unclear whether the functional gains will be sustained in the longer term or if such intensity is of further benefit for rehabilitation to achieve higher mobility goals.

However, this case report does present an alternative model to what is traditionally explored and delivered, pertaining to a dose–response effect that may shorten the typical time frame needed to achieve goals.

A recently published protocol (Valadão et al 2021) using a similar multimodal model of dosed delivery in a randomised controlled trial may provide further clarity about the benefits of this treatment model and highlight the need for trials with larger sample sizes.

A limitation of this report is its ability to be generalised. Because individuals with CP present with a wide range of disability levels, these results cannot be generalised to all people with spastic diplegia.

In this case report, a neuro-adaptive, rehabilitative approach was undertaken; however, a compensatory approach may be more appropriate for some individuals with more severe CP.

In the absence of detailed decision-making algorithms and more precise evidence-based guidance specific to CP, current physiotherapy management of other adults with CP will require individualised and holistic assessment to establish a set of priorities to effect change in all domains of the ICF, while considering the needs and desires of the individual.

Conclusion

This case report showcases how an evidence-informed, intensive, neuro-adaptive, multimodal approach can significantly enhance walking and balance in an adult with CP and mobility decline in a relatively short period.

However, carryover into confidence in walking and concerns about falls is limited and it is unclear whether further gains require such intensity.

Further research is warranted into multimodal interventions to inform future clinical practice.

>> Keegan Bow APAM MACP is an APA Neurological Physiotherapist and a first- year registrar in the clinical specialisation program at the Australian College of Physiotherapists. Keegan is the director of Klint Intensive Neuro Therapies and Klint Kids, providing adult and paediatric neurological physiotherapy services across Melbourne, Victoria. He is a sessional educator at Swinburne University in neuroscience and complex care and he provides expert opinion on medicolegal cases.

References

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Appendix A: The Ambulatory Self-Confidence Questionnaire (ASCQ) 

Asano, M., Miller, W. C., & Eng, J. J. (2007). Development and psychometric properties of the ambulatory self-confidence questionnaire. Gerontology, 53(6), 373-381.

This questionnaire measures how confident you are in your ability to walk. If you normally walk with a walker or cane, assume you have your walking aid with you when answering each question. Please answer all items. If activities do not apply to you please guess how you would feel to perform the activity. 
Please answer each question using the following 0 – 10 scale: 0 1 2 3 4 5 6 7 8 9 10 
0=Not at all confident, 10=Completely Confident
On a scale of 0 – 10, how confident are you that you are able to… 
ITEMS
a. step up onto a curb? 
b. step down off a curb? 
c. walk up a ramp (mild incline)? 
d. walk down a ramp (mild incline)? 
e. walk up a flight of stairs (4 steps or more) with a handrail? 
f. walk down a flight of stairs (4 steps or more) with a handrail? 
g. cross a street with a timed cross walk (walk signal)? 
h. cross a street without a timed cross walk (walk signal)? 
i. walk on an uneven sidewalk? 
j. walk on grass? 
k. walk on slippery ground: for example icy or wet surfaces? 
l. walk in the dark or at night when it is difficult to see your feet? 
m. walk through a crowded place: for example a busy street? 
n. walk and talk to a companion at the same time? 
o. carry small items while walking: for example a carton of milk? 
p. stop walking suddenly to avoid an oncoming vehicle? 
q. uses an escalator ? 
r. use a moving sidewalk (one at an airport)? 
s. walk on a moving bus? 
t. walk from one room to another in your home? 
u. walk a short distance without stopping: for example from your home to a car? 
v. walk a long distance without stopping: for example from your home to a bus stop?
 
Appendix B:  Falls Efficacy Scale

Yardley, L., Beyer, N., Hauer, K., Kempen, G., Piot-Ziegler, C., & Todd, C. (2005). Development and initial validation of the Falls Efficacy Scale-International (FES-I). Age and ageing, 34(6), 614-619.

The text of the FES-I below is the final version agreed by the authors on completion of the development study, prior to subsequent translation and validation in different languages. It became clear during the process of translation that there was no wording of the questionnaire that would translate easily into every EC language using exactly the same words and phrases. Consequently, these notes are intended to assist translators of the FES-I to express the same meaning of items, even if they cannot use quite the same words in their language. They may also assist interviewers who are asked for clarification of the meaning of items when the FES-I is administered by interview.

Instructions
Participants should answer items thinking about how they usually do the activity – for example, if they usually walk with an aid they should answer items about walking to show how concerned they would be about falling when using that aid. Some translators may find it helpful to clarify in the instructions (after the sentence on circling an opinion) ‘The opinions you can choose from are: 1 = not at all concerned 2 = somewhat concerned 3 = fairly concerned 4 = very concerned.’ In some languages it is better to translate the word ‘opinion’ as ‘statement’.

Response categories
The word ‘concerned’ expresses a cognitive or rational disquiet about the possibility of falling, but does not express the emotional distress that would be expressed by terms such as ‘worried’, ‘anxious’ or ‘fearful’. It is important to use a similar unemotional term, as respondents may be less willing to admit to emotions, which might be viewed as signs of weakness.

Item 3. In some EC languages ‘simple’ meals are best translated as ‘everyday’ meals, but the intention is to refer to a meal that does not require complex preparation, rather than one that is prepared every day.

Item 5. This item is intended to refer to shopping that is not extensive or recreational. In some languages the best translation is ‘shopping for  groceries’.

Item 7.  This item refers to any stairs, not necessarily the flight of stairs in one’s own house.

Item 8.  In some languages ‘neighbourhood’ may be difficult to translate, and so ‘walking around outside’ can be used instead.

Item 12. In some languages it is necessary to add the term ‘acquaintances’ to friends and relatives, since this is a more common and casual 
category of relationship than friends. (See also comment on items 12, 13 and 16 below)

Item 13. ‘Crowds’ can be translated as ‘many people’ if necessary. (See also comment on items 12,13 and 16 below)

Item 14. It was found to be necessary to give examples of what is meant by uneven ground, but no examples could be found that were appropriate for all countries. Consequently, translators should choose any TWO examples from the following: cobblestones; poorly maintained pavement; rocky ground; unpaved surface.

Items 12, 13, 16. These items contain a greater element of ambiguity than many of the items assessing functional capabilities, because the physical activities involved in these social events may differ greatly for different respondents. However, it was decided that this ambiguity was acceptable because it is important to assess effects of fear of falling on social activities.