PROFILE OF ANKLE CONTRACTURE SURGERY IN CEREBRAL PALSY PATIENTS IN A SINGLE CENTER

NATIONAL REFERRAL ORTHOPAEDIC HOSPITAL IN INDONESIA

Hendra Cahya Kumara1, Anung Budi Satriadi1, Musa Fasa Roshada2

1
Orthopaedic Surgeon, Pediatric Orthopaedic Department, Prof. Dr. R. Soeharso Orthopaedic Hospital –
Faculty of Medicine Sebelas Maret University, Surakarta
2
Orthopaedic Resident, Faculty of Medicine Sebelas Maret University, Surakarta

ABSTRACT
Background : Children with Cerebral Palsy (CP) mostly present with spasticity of the extremities. Ankle
contracture has been known to be the most common finding. This condition adversely affect standing
and walking ability. With the preserved cognitive function in both spastic diplegic and hemiplegic CP
patient, independent ambulation is expected. Thus, surgical treatment is expected to provide significant
benefit to the patients. This article provides the demography of ankle contracture soft tissue surgery in
CP patients in a single center orthopaedic hospital in Indonesia to provide the magnitude and
distribution of this condition.
Methods : This was a retrospective descriptive study. Data was taken from the hospital registry since
January 2019 to May 2021. All CP patients with ankle contracture soft tissue surgery were included.
Clinical type of CP, age at the time of surgery, procedure taken, and recurrence was taken into
description.
Results : There was a total of 152 CP patients who had ankle contracture soft tissue surgery. The average
number of cases per month was 8,1 cases in 2019, 3,5 cases in 2020, and 2,4 cases in 2021 until May.
There were 141 cases of diplegic CP (92,8%) compared to 11 cases of hemiplegic CP (7,2%). All patient
presenting for surgery had contracture in hip and knee as well and the release was performed to the
same side hip and knee contracture. The surgical treatment that is performed is achilles tendon
lengthening (74%), gastrocnemius recession (13%), and achilles tendon lengthening with split posterior
tibial tendon transfer (13%). There was no recurrence of ankle contracture after surgery in 2 years.
Conclusion : Diplegic CP patient who had ankle contracture surgery is more common than hemiplegic CP
patient. Ankle contracture is always present with contracture of hip and knee thus the surgery is
performed at the same time for the same side. There was a significant decrease of number of surgery
starting from 2020 that was probably due to the COVID-19 pandemic had emerged.
Keywords : Cerebral pasy, ankle contracture, surgical release, diplegic CP, hemiplegic CP.

INTRODUCTION

Cerebral palsy is a group of disorder that manifest as abnormal muscle tone, posture and movement.
Based on an international consensus, CP is described as a group of permanent disorders of movement
and posture causing any activity limitation that are attributed to nonprogressive disturbance that
occurred in the developing fetal or immature brain. Its classification is based on the predominantly
motoric syndrome and the extremities in which it involved, they are spastic diplegic, spastic hemiplegic,
spastic quadriplegic, and extrapyramidal. The incidence of CP is 2-3 in 1.000 live births. The diagnosis of
CP is mainly from the presenting signs of the patients although some findings may also be found in
normal infants making diagnosis more reliable when the children is at least 2 years old. Early diagnosis
can be supported with the use of standardized neuromotor assessment and findings on Magnetic
Resonance Imaging (MRI). MRI is useful in assessing the scope of the brain lesion and detect the
presence of brain malformation.1

Spastic cerebral palsy children have a good chance to walk independently as many as 75%. However,
their gait may be affected by the spasticity of the muscle and the contractures of the joints. 2 Muscle
from patients with cerebral palsy shows functional deficits such as decreased force production and
range of motion. Muscle is altered at a structural level, with decreased muscle body size, smaller-
diameter fibers, and highly stretched sarcomeres (the force-producing unit of muscle). 3 While CP
contractures are often thought to be stiff because of muscle overactivation, there are also critical
contributions to stiffness that simply result from increased intrinsic passive stiffness of the tissue.
Muscle from patients with cerebral palsy has altered extracellular matrix, connective tissue, decreased
muscle stem cell numbers and altered gene expression. Capillary density was 30% lower in patients with
CP. Lipid content has been shown to increase in some cases. Collagen content also appears to be
increased, and this has been correlated with increased stiffness of muscle fiber bundle. 4,5
Equinus deformity is the most common finding of CP children. The general spasticity of the muscle of the
leg results in a significantly greater plantar flexor force of gastrocnemius and soleus muscle in
comparison to the dorsiflexor muscle such as tibialis anterior and extensor digitorum longus, resulting in
ankle contracture. The treatment for this deformity is a stepwise approach depending on age and
severity with the objective of optimizing the child’s function to stand and walk. As the child started to
stand, an Ankle Foot Orthotic can be applied. The caregivers should also be encouraged to stretch the
plantar flexor periodically everyday. Botulinum toxin can also be injected periodically in conjunction
with the orthotics. Most children will be able to tolerate this conservative measure until the age of 6-8
years. This is the age when surgical lengthening is considered as the contracture become more
prominent that the ankle could not dorsiflex to neutral with the knee extended. The surgical lengthening
includes an open Z-lengthening, recession of gastrocnemius muscle, cutting the whole tendon of
gastrocnemius, and additional plication of tibialis anterior.6,7

Considering the magnitude and importance of the deformity in a relatively common cases presenting in
our hospital, this study was brought to describe the demographic distribution of ankle contracture
surgery in CP patient.

METHODS

This was a retrospective descriptive study. Data was taken from the hospital registry since January 2019
to May 2021. All CP patients with ankle contracture surgery was included in this study. Clinical type of
CP, age at the time of surgery, and surgical procedure was taken into description.

RESULTS

The period taken into this research was 29 months starting from January 2019 to May 2021. There were
a total of 152 CP patients who had ankle contracture surgery at this time. The total number of surgery in
2019 was 98, whereas the number declines to 42 in 2020. The average number of cases per month was
8,1 cases in 2019, 3,5 cases in 2020, and 2,4 cases in 2021 until May. There were 141 cases of diplegic CP
(92,8%) compared to 11 cases of hemiplegic CP (7,2%).
 18

16

14

12

10

0
3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25

Diplegic CP Hemiplegic CP

Figure 1. Number of Ankle Contracture Surgery based on Patient's Age

The earliest surgery performed was at the age of 3 years old in diplegic CP patients. Surgical procedure is
most commonly performed at the age of 6 years old and started to decline after 13 years old in diplegic
patients. In hemipegic patients, the case is relatively rare. However, surgical procedure was performed
from the age of 7 years old until 19 years old. All patient presenting for surgery had contracture in hip
and knee as well and the release was performed to the same side hip and knee contracture. The surgical
treatment that is performed is achilles tendon lengthening (74%), gastrocnemius recession (13%), and
achilles tendon lengthening with split posterior tibial tendon transfer (13%). No recurrence after surgery
was found in 2 years interval.

DISCUSSION

From this research it was known that CP patient presents with contracture of joints including hip, knee,
and ankle. This was mainly because the pathophysiology of the disease involving all groups of muscles
affected in the central nervous system. Whereas for the ankle to become equinus, it is because the
plantar flexors of the ankle are five to six times stronger than the dorsiflexor so that even when all the
muscles are equally spastic in CP, there will always an overpowering of dorsiflexors, which is tibialis
anterior, extensor digitorum longus, and extensor halluces longus, by the plantar flexor groups which
consisted of soleus, gastrocnemius, tibialis posterior, flexor halluces longus, flexor digitorum longus,
peroneus longus, and peroneus brevis.7

Diplegic CP patients outnumbered those with hemiplegia. Based on the predominantly motoric
syndrome, diplegic CP was known as the most common type of CP, as many as 35% of all children with
CP. It was known to be the result of immature oligodendroglia between 20 and 34 weeks gestation with
both motoric corticospinal and the thalamocortical pathways affected. These patients commonly
preserve normal prefrontal cortex function for cognition and better prognosis for ambulation. Thus, a
surgery of this group of patient is preferred.1

The number of surgery was seen to decline significantly in 2021. This was thought to be as a cause of
COVID-19 cases that emerged early in 2020 and was declared as pandemic in March 2020. The overall
visits of outpatient clinic were seen to be decreasing as patients were afraid of getting COVID-19
infection at the hospital. Moreover, the surgical contracture release is considered as an elective surgery
that is limited at that time.8,9

Of all surgeries performed, the ankle was released together with knee and ankle at the same side of the
lower limb. This was called Single-Event Multilevel Surgery (SEMLS). This procedure offered several
advantages including single hospital admission, decreased need of anaesthetics, single stay in inpatient
rehabilitation, and prevention of deformity resulting secondary to untreated spasticity at another joints
nearby. This procedure was proven to be more beneficial performed in children than in adults. 10

Soft tissue procedure that may be performed for ankle contracture is proximal gastrocnemius tenotomy,
gastrocnemius myofascial lengthening, complete release of gastrocnemius allowing it to slide
proximally, myofascial lengthening of combined gastrocnemius and soleus tendons, tenotomy of achilles
tendon, anterior transfer of tendon achilles, sliding lengthening of achilles tendon, and Z-lengthening of
achilles tendon.7 In this study group, achilles tendon lengthening was most commonly performed as this
was relatively easy to perform and has provided good outcome with low recurrence rate. A tendon
transfer procedure may be performed by splitting the tibialis posterior tendon to the dorsum of the foot
converting the tendon as ankle dorsiflexor. This provided good result especially for those with varus
hindfoot.11 When the cause of ankle contracture is an isolated gastrocnemius contracture that is tested
by limitation of ankle dorsiflexion with full knee extension, gastrocnemius recession is performed. This
recession procedure should not involve the soleus to minimize the potential plantarflexion strength
loss.12

In this more than two years data, no recurrence of ankle contracture was recorded. The period of this
research showed that one year recurrence rate was zero. It was mentioned that recurrence rate varies
from 0 – 43% and may increase to 62% in specific group such as hemiplegic CP. From the previous
researches, the recurrence rate was described in at least 5 years follow up. Thus, in this research the
period involved was not long enough to detect recurrence cases. Moreover, it was implicated by the
pandemic time that hinders the patients visiting the hospital. Other risk factors of recurrence rate
following surgical ankle release was the age of the first surgery. Thus, it is essential to start with
conservative treatment before proceeding with surgical release. 7,13

CONCLUSION

Diplegic CP patient who had ankle contracture surgery is more common than hemiplegic CP patient.
Ankle contracture is always present with contracture of hip and knee thus the surgery is performed at
the same time for the same side. There was a significant decrease of number of surgery starting from
2020 that was probably due to the COVID-19 pandemic had emerged.

REFERENCES
1. Patel DR, Neelakantan M, Pandher K, Merrick J. Cerebral palsy in children: a clinical overview. Transl
Pediatr. 2020 Feb;9(S1):S125–35.

2. Park CI, Park ES, Kim HW, Rha D. Soft Tissue Surgery for Equinus Deformity in Spastic Hemiplegic
Cerebral Palsy: Effects on Kinematic and Kinetic Parameters. Yonsei Med J. 2006;47(5):657.

3. Fridén J, Lieber RL. Spastic muscle cells are shorter and stiffer than normal cells: Spastic Muscle Cell
Properties. Muscle Nerve. 2003 Feb;27(2):157–64.

4. Mathewson MA, Lieber RL. Pathophysiology of Muscle Contractures in Cerebral Palsy. Physical
Medicine and Rehabilitation Clinics of North America. 2015 Feb;26(1):57–67.

5. Smith LR, Lee KS, Ward SR, Chambers HG, Lieber RL. Hamstring contractures in children with spastic
cerebral palsy result from a stiffer extracellular matrix and increased in vivo sarcomere length:
Passive mechanical properties of muscle contracture. The Journal of Physiology. 2011 May
15;589(10):2625–39.
6. Sees JP, Miller F. Overview of foot deformity management in children with cerebral palsy. J Child
Orthop. 2013 Nov 1;7(5):373–7.

7. Miller F. Ankle Equinus in Cerebral Palsy. In: Miller F, Bachrach S, Lennon N, O’Neil M, editors.
Cerebral Palsy [Internet]. Cham: Springer International Publishing; 2019 [cited 2021 Sep 5]. p. 1–24.
Available from: http://link.springer.com/10.1007/978-3-319-50592-3_199-1

8. Massey PA, McClary K, Zhang AS, Savoie FH, Barton RS. Orthopaedic Surgical Selection and Inpatient
Paradigms During the Coronavirus (COVID-19) Pandemic. J Am Acad Orthop Surg. 2020 Jun
1;28(11):436–50.

9. Petrone B, Iturriaga CR, Mauri T, Sgaglione N. COVID-19 and Orthopaedics: Recovery After the
Pandemic Surge. Arthroscopy, Sports Medicine, and Rehabilitation. 2020 Oct;2(5):e677–82.

10. Jea A, Dormans J. Single-Event Multilevel Surgery: Contender or Pretender. Pediatrics. 2019
Apr;143(4):e20190102.

11. VlAChOu M, BEriS A, DiMiTriADiS D. Split tibialis posterior tendon transfer for correction of spastic
equinovarus hindfoot deformity. 2010;76:7.

12. Chimera NJ, Castro M, Manal K. Function and Strength Following Gastrocnemius Recession for
Isolated Gastrocnemius Contracture. Foot Ankle Int. 2010 May;31(5):377–84.

13. Shore BJ, White N, Kerr Graham H. Surgical correction of equinus deformity in children with cerebral
palsy: a systematic review. J Child Orthop. 2010 Aug 1;4(4):277–90.