Firth Et Al. - 2013 - Multilevel Surgery For Equinus Gait in Children With Spastic Diplegic Cerebral Palsy
Firth Et Al. - 2013 - Multilevel Surgery For Equinus Gait in Children With Spastic Diplegic Cerebral Palsy
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C OPYRIGHT Ó 2013 BY T HE J OURNAL OF B ONE AND J OINT S URGERY, I NCORPORATED
Investigation performed at the Orthopaedic Department, The Royal Children’s Hospital, Parkville, Victoria, Australia
Background: In children with spastic diplegia, surgery for ankle equinus contracture is associated with a high prevalence
of both overcorrection, which may result in a calcaneal deformity and crouch gait, and recurrent equinus contracture, which
may require revision surgery. We sought to determine if conservative surgery for equinus gait, in the context of multilevel
surgery, could result in the avoidance of overcorrection and crouch gait as well as an acceptable rate of recurrent equinus
contracture at the time of medium-term follow-up.
Methods: This was a retrospective, consecutive cohort study of children with spastic diplegia who had had surgery for
equinus gait between 1996 and 2006. All children had distal gastrocnemius recession or differential gastrocnemius-
soleus complex lengthening, on one or both sides, as part of single-event multilevel surgery. The primary outcome
measures were the Gait Variable Scores (GVS) and Gait Profile Score (GPS) at two time points after surgery.
Results: Forty children with spastic diplegia, Gross Motor Function Classification System (GMFCS) level II or III, were
included in this study. There were twenty-five boys and fifteen girls. The mean age was ten years at the time of surgery and
seventeen years at the time of final follow-up. The mean postoperative follow-up period was 7.5 years. The mean ankle
GVS improved from 18.5° before surgery to 8.7° at the time of short-term follow-up (p < 0.005) and 7.8° at the time of
medium-term follow-up. The equinus gait was successfully corrected in the majority of children, with a low rate of
overcorrection (2.5%) and a high rate of recurrent equinus (35%), as determined by sagittal ankle kinematics. Mild
recurrent equinus was usually well tolerated and conferred some advantages, including contributing to strong coupling at
the knee and independence from using an ankle-foot orthosis.
Conclusions: Surgical treatment for equinus gait in children with spastic diplegia was successful, at a mean of seven
years, in the majority of cases when combined with multilevel surgery, orthoses, and rehabilitation. No patient developed
crouch gait, and the rate of revision surgery for recurrent equinus was 12.5%.
Level of Evidence: Therapeutic Level IV. See Instructions for Authors for a complete description of levels of evidence.
W
hen children with spastic diplegia begin walking, of the gastrocnemius-soleus complex, especially percutaneous
they usually walk on tiptoe with an equinus gait1. lengthening of the Achilles tendon4-9. The standard of care for
Older children and adolescents more often have the management of gait dysfunction in children with spastic
flexed-knee patterns and crouch gait1-3. Crouch gait may be diplegia is single-event multilevel surgery in which all con-
part of the natural history of gait evolution in children with tractures and osseous deformities are dealt with during a single
spastic diplegia but may be precipitated by isolated lengthening operative session, commonly on the basis of three-dimensional
Disclosure: None of the authors received payments or services, either directly or indirectly (i.e., via his or her institution), from a third party in support of
any aspect of this work. One or more of the authors, or his or her institution, has had a financial relationship, in the thirty-six months prior to submission of
this work, with an entity in the biomedical arena that could be perceived to influence or have the potential to influence what is written in this work. No
author has had any other relationships, or has engaged in any other activities, that could be perceived to influence or have the potential to influence what
is written in this work. The complete Disclosures of Potential Conflicts of Interest submitted by authors are always provided with the online version of the
article.
gait analysis10-23. In this context, the correction of equinus de- The inclusion criteria were (1) a diagnosis of cerebral palsy according to
formity is important to the overall outcome because of its the 2005 international definition and registration in the Statewide Cerebral
43,44
Palsy Register ; (2) a spastic movement disorder with bilateral involvement
contribution to sagittal plane balance24.
(spastic diplegia) and Gross Motor Function Classification System (GMFCS)
Many procedures for the correction of equinus deformity 45
level II or III ; (3) single-event multilevel surgery between 1996 and 2006 ; (4)
46
spastic diplegia than the soleus, which spans just one joint31,39,40. Three different ‘‘surgical doses’’ for equinus were utilized according to the severity
Given the importance of the soleus muscle in maintaining of the equinus contracture as described in the following sections.
plantar flexion-knee extension coupling with gait, intramus- Distal Gastrocnemius Recession: Strayer Procedure (Fig. 1): The distal
cular gastrocnemius lengthening or gastrocnemius recession limit of the gastrocnemius was identified by inspection and palpation. The
junction of the gastrocnemius aponeurosis with the soleus fascia was explored
might be preferable to more commonly used procedures in
from a 2 to 3-cm vertically oriented posteromedial skin incision. The interval
which both the gastrocnemius and the soleus are length- between the gastrocnemius and soleus fascia was identified and was opened by
ened9,32,33. Some studies suggest that calcaneal gait leading to blunt dissection on the medial side. The sural nerve was protected. The gas-
crouch gait in spastic diplegia is more common after Achilles trocnemius aponeurosis was divided transversely from medial to lateral and
tendon lengthening than after gastrocnemius recession4,5,9.
However, some short-term studies demonstrated no difference
based on the surgical procedure or the zone of the procedure9,17.
Biomechanical studies of human cadavers have provided evi-
dence to suggest that procedures performed proximally in a
muscle-tendon unit, where the cross-sectional area is greater,
may provide more biomechanically stable results than distal
procedures41. Given that both calcaneus/crouch gait and re-
current equinus may not be apparent for many years after
surgery, studies with long-term follow-up and objective out-
come measures may be superior to randomized trials to in-
vestigate these important outcomes4,5,9,30. The purpose of this
study was to report the medium-term outcomes of conserva-
tive (mainly Zone-1) surgery for the management of equinus
gait in the context of single-event multilevel surgery in children
with spastic diplegia. Specifically, we wanted to determine if
conservative surgery resulted in the avoidance of crouch gait at
the expense of recurrent equinus and the need for revision
surgery.
Fig. 1
Gastrocnemius recession with the Strayer procedure (Fig. 1-A) and the
Materials and Methods modified Strayer procedure, which combines distal gastrocnemius re-
T his was a retrospective, consecutive cohort study of data that were pro-
spectively gathered according to standardized gait laboratory protocols,
42
which have good reliability . The study was approved under the audit provi-
cession with soleal fascial lengthening (S.F.L.) (Fig. 1-B). Both are Zone-
1 procedures. Recurrent equinus was managed with lengthening of the
sions of our institution’s Ethics in Human Research Committee. Parents pro- gastrocnemius-soleus complex fascia, according to the method described
34
vided written informed consent for the children to undergo the surgery. by Vulpius and Stoffel (Zone-2 procedure) (Fig. 1-C).
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*There were forty patients (twenty-five boys and fifteen girls). The mean interval between Time 1 and Time 3 was 7.5 years (range, 4.4 to 14.6
years). †The values are given as the mean with the range in parentheses.
allowed to retract proximally. The Silfverskiöld test was performed and if 5° of length and strength. Repeat three-dimensional gait analysis was performed at
ankle dorsiflexion with the knee extended was achieved no additional surgery twelve to eighteen months after surgery (prior to removal of implants [Time 2])
was performed. The gastrocnemius aponeurosis was sutured distally to the and again at four to fourteen years after surgery (Time 3).
52
soleus fascia at the level at which it rested when the knee was in full extension GMFCS level, Functional Mobility Scale (FMS) score , height, weight,
33,51
and the foot was at 90° to the leg . and body mass index (BMI) were all available at Time 1 (in the six months
Distal Gastrocnemius Recession Plus Soleal Fascial Lengthening: Modified before surgery), Time 2, and Time 3 (Table I). Physical examination was per-
Strayer Procedure (Fig. 1): When the Silfverskiöld test revealed residual soleus formed by an experienced physical therapist using standardized protocols,
22,42,52,53
contracture after the Strayer procedure, a single transverse stripe/division of the which have good reliability . Quantitative three-dimensional gait-
51
soleus fascia was performed . The Silfverskiöld test was repeated to confirm 5° analysis data were collected with a state-of-the-art Vicon system (Oxford
of dorsiflexion with the knee extended. The combination of distal gastrocne- Metrics Group, Oxford, United Kingdom) and two AMTI force plates
mius recession and soleal fascial lengthening is called a modified Strayer (Watertown, Massachusetts). Reflective markers were applied to the osseous
51
procedure . landmarks with use of a standardized procedure. Kinematic data were cal-
White Slide Lengthening of the Achilles Tendon: Lengthening of the culated with use of Plug In Gait (Oxford Metrics Group) and uploaded to
54
Achilles tendon was used only when there was a contracture of both the gas- GaitaBase, a web-interfaced repository for gait data .
28
trocnemius and the soleus of >30° and no improvement with knee flexion . The Gait Profile Score (GPS) as well as the Gait Variable Scores (GVS)
In children with symmetric equinus deformities, both sides were treated for nine kinematic parameters (pelvic tilt, pelvic obliquity, pelvic rotation, hip
with either a Strayer procedure or a modified Strayer procedure. Asymmetric flexion, hip abduction, hip rotation, knee flexion, ankle dorsiflexion, and foot
equinus contractures were managed with various combinations, including the progression) were calculated for each child. The nine GVS values and the GPS
Strayer procedure on one side plus Botox injection on the contralateral side, the value formed the Movement Analysis Profile (MAP), which is a graphical
55
Strayer procedure plus the modified Strayer procedure, or slide lengthening of representation of these data (Fig. 2). The GVS was also calculated for
the Achilles tendon by double hemisection (White slide procedure) accom- two kinetic parameters: sagittal ankle moment and ankle power. Each GVS is
28,51
panied by a contralateral Strayer or modified Strayer procedure . The final a measure of the distance of the patient’s curve from the corresponding
47,50,51
choice was always based on an intraoperative Silfverskiöld test .
Recurrent equinus was corrected with a Vulpius gastrocnemius-soleus
complex recession with use of a single transverse division of both the gas-
34,51
trocnemius aponeurosis and the soleus fascia, in Zone 2 (Fig. 1). Indica-
tions for revision surgery were identical to those for the index surgery.
Postoperative management was uniform for all of the equinus surgical
procedures. A below-the-knee plaster cast was applied with the foot in a neutral
position of dorsiflexion. Concomitant hamstring surgery was protected by knee
immobilizers. The children were discharged from the hospital four to seven
days after surgery, at which time they used knee immobilizers with elevating
22,46
footrests on their wheelchair . Unrestricted weight-bearing beginning the
first postoperative day was prescribed for children who had had soft-tissue
surgery only. Children treated with osteotomies were encouraged to begin full
weight-bearing at one to three weeks after surgery, according to the surgeon’s
46
assessment of the stability of fixation and osteotomy site healing .
Casts were removed after three weeks to check healing and to cast molds
for ankle-foot orthoses. The standard postoperative ankle-foot orthosis was
46
solid with hinges inserted but not activated . Fiberglass casts were applied and Fig. 2
worn for an additional three weeks. These were removed at six weeks after The Movement Analysis Profile (MAP) for nine kinematic Gait Variable
surgery and replaced by ankle-foot orthoses. Gait and function were checked in Scores (GVS) at Time 1 (T1), Time 2 (T2), and Time 3 (T3) in comparison
the gait laboratory with use of a standardized protocol for two-dimensional with laboratory normal values. In addition, the Gait Profile Scores (GPS) at
22,46
video-based gait analysis at three, six, and nine months after surgery . The
decision to activate the ankle-foot-orthosis hinges was contingent on stable Time 1, Time 2, and Time 3 are shown, also with reference to the laboratory
plantar flexion-knee extension coupling, full knee extension in stance phase, normal range. Mean values with standard deviation (error bars) are shown.
and a clinical assessment showing sufficient gastrocnemius-soleus complex The units of the y axis are degrees for each of the parameters.
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TABLE II Physical Examination Results of Silfverskiöld Test at Time 1, Time 2, and Time 3
three, for recurrent equinus deformity before Time 3. The re- the mean height increased from 129 to 162 cm, the mean
vision lengthenings were performed at a mean of 3.7 years weight increased from 30 to 59 kg, and the mean BMI increased
(range, 1.7 to 5.6 years) after the initial surgery. The indications from 17.6 to 22.7 kg/m2 between Time 1 and Time 3 (Table I).
for revision surgery were identical to those for the index sur- By Time 3, the majority of adolescents had reached skeletal
gery. The mean age at the index surgery for those who required maturity.
revision surgery was 8.5 years compared with 10.2 years for The results of equinus contracture surgery may dete-
those who did not. riorate with time, and long-term follow-up with objective
measures offers the best opportunity to report meaningful
Movement Analysis Profile (MAP) (Fig. 2 and Appendix) outcomes4,5,9,30,59. To the best of our knowledge, the present
The MAP improved significantly between Time 1 and Time 2 study has the second longest follow-up among studies in which
and between Time 1 and Time 3 for six five of the nine GVS gait analysis has been utilized9,30. It is relatively easy to deter-
(sagittal hip, sagittal knee, sagittal ankle, coronal pelvis, trans- mine the rate of recurrent equinus requiring revision surgery
verse hip, and transverse foot progression). The mean sagittal from a chart review27,59. However, only three-dimensional
ankle GVS was 18.5° at Time 1 and decreased to 8.7° at Time 2 gait analysis permits precise documentation of more minor
and 7.8° at Time 3. The sagittal ankle GVS improved for 81% of degrees of both overcorrection and undercorrection and re-
the subjects at Time 2, and this percentage increased to 92% of current equinus deformity4,30.
the subjects at Time 3 (see Appendix). Significant improvements In this study, we report the outcomes of a conservative
in the ankle moment and power GVS were also noted (see approach to equinus contracture surgery in the context of
Appendix). single-event multilevel surgery, with the aim being to preserve
Mean gait parameters do not reveal individual patterns of strong coupling between the ankle and knee and to prevent the
outcomes. Three cases are illustrated with serial sagittal ankle cascade of events that can lead to progressive crouch gait4,5,7,8.
and knee kinematics at Time 1, Time 2, and Time 3 in the The potential danger of this approach is undercorrection, re-
Appendix. current deformity, and the need for repeat surgery30,59. This
study demonstrated satisfactory correction at the ankle level in
First Ankle Rocker most children as well as a clinically relevant and statistically
At Time 1, first rocker was not present at either ankle in any significant correction of the overall gait pattern. The im-
subject. At Time 2, first rocker was present on one or both sides provement in GPS was clinically relevant and statistically sig-
in seven subjects and this increased to ten subjects at Time 3. nificant, equating to a 38% improvement in gait function
between Time 1 and Time 3—more than four times the min-
Discussion imum clinically important difference56. The ankle also showed
with spastic diplegia and 50% of patients with quadriplegia We believe that we are the first to report on the presence
developed crouch gait and required bracing5. Dietz et al. also of first rocker at the time of medium-term follow-up after
noted that ground-reaction ankle-foot orthoses were difficult surgery for equinus gait. First rocker is a hallmark of normal
to don and doff, were poorly tolerated, and were relatively gait and is rarely present in children with cerebral palsy57. The
ineffective5. presence of first rocker is associated with a gait pattern that
In contrast, Dreher and colleagues recently reported ex- appears more ‘‘normal.’’ Conversely, the absence of first rocker,
cellent long-term results after gastrocnemius-soleus intramus- even when equinus has been corrected, is associated with an
cular aponeurotic recession as part of multilevel surgery in abnormal appearance of gait, or ‘‘limping.’’ In this study, no
patients with spastic diplegic cerebral palsy30. The patients in patient had first rocker before surgery and 25% had first rocker
their study had less severe cerebral palsy than the patients in our at Time 3. Equinus deformity precludes first rocker in children
study, according to the GMFCS levels, and the equinus con- with cerebral palsy. Effective surgery for equinus gait may re-
tracture was less severe. The mean ages of the patients (9.8 years store first rocker but only when the correction is precise and
versus ten years), the concomitant procedures in the multilevel underlying strength and selective motor control are adequate5
surgery (both osseous and soft-tissue), and the changes in the (see Appendix).
height, weight, and BMI of the patients during the follow-up Five children had surgery for recurrent equinus con-
period were similar30. Dreher et al. used the same kinematic tracture between Time 2 and Time 3. No overcorrection or
criteria for overcorrection and undercorrection as were utilized recurrent equinus had been recorded in these children at
in our study, permitting a direct comparison between the Time 3, at a mean of 4.8 years (range, 1.1 to 10.0 years). Two
studies. In their study, fixed equinus was corrected by intra- children were offered additional surgery and declined. The
muscular aponeurotic recession of the gastrocnemius or in- equinus in the remaining nineteen adolescents was asymp-
tramuscular aponeurotic recession of the gastrocnemius and tomatic and well tolerated at the time of writing. The equinus
soleus, with three legs (in the group of forty-four patients) not could be considered to be an advantage for some in that it
requiring a lengthening procedure. The Baumann procedure preserved extension at the knee and allowed them to walk
consists of intramuscular aponeurotic lengthening of the gas- without the need for ankle-foot orthoses5,10 (see Appendix).
trocnemius (and soleus when indicated) and is performed in The results of the medium-term gait analysis (Time 3) were
Zone 113. In the current study, distal gastrocnemius recession used to determine which children and adolescents could safely
(the Strayer procedure) or distal gastrocnemius recession with discontinue the use of ankle-foot orthoses; it was possible for
soleal fascial lengthening (the modified Strayer procedure) 65% to do so.
were the principal procedures utilized, and they are Zone-1 The strengths of this study were a standardized approach
surgical procedures that yield stable results. An important to surgery, rehabilitation and orthotic prescription, and long-
difference between the two studies was the intraoperative goal. term follow-up with objective outcome measures. A rigorous
Dreher et al. aimed for 15° to 20° of dorsiflexion with knee kinematic definition of both overcorrection (calcaneus gait)
extension and knee flexion whereas our goal was 5° of dorsi- and undercorrection/recurrence (recurrent equinus) was uti-
flexion with the knee extended. Overcorrection with calcaneal lized, allowing for direct comparison with previous studies in
gait and severe crouch gait was more common in their study30. which the same outcome criteria had been used4,30,61. The
It would seem that both Zone-1 procedures are associated with weaknesses of the study were variability in the single-event
a significant rate of recurrent equinus, as determined by kine- multilevel surgery prescription and in the duration of follow-
matics, and a relatively low rate of overcorrection. However, up. In addition, the variable surgical prescription, including
aiming for 15° to 20° of dorsiflexion intraoperatively may be both soft-tissue and osseous procedures at multiple levels, has
overly aggressive, and our data suggest that a more conservative both known and unknown impacts on the ankle level and
goal of 5° may be better. dynamic ankle function. However, given the documented
The categorical classification of dynamic gastrocnemius- poor results of single-level surgery for equinus, it is likely
soleus complex length is a useful method for summarizing that a multilevel surgical approach will remain the treatment
outcomes and comparing results between series4,9,30. How- paradigm for the foreseeable future4,30,61. Clinical researchers
ever, dichotomous outcomes that are quantitatively similar and surgeons will continue to have to grapple with the
are not of equal clinical relevance. A gastrocnemius-soleus complexity of the local and distant effects of multilevel sur-
complex length that is two standard deviations above the gical procedures when they evaluate outcomes. In addition,
mean may, in the long term, result in severe crouch gait with given that patients cannot ethically remain in randomized
severe disability10,30. There are currently no reliable methods trials for long periods of time, prospective studies with long-
with which to shorten an overlengthened gastrocnemius- term objective outcome measures are likely to offer the best
soleus complex. Surgery for severe crouch gait is invasive and opportunity for meaningful clinical research in this complex
requires prolonged rehabilitation7,8. In contrast, recurrent area9,30,61.
equinus can be easily managed with revision surgery59. Most
cases of equinus gait at Time 3 in our study were the result of Appendix
recurrent equinus deformity and not undercorrection at the A table showing comparisons of GPS and GVS among
index surgery59,60. the evaluation times and figures demonstrating sagittal
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kinematic traces of the knee and ankle of specific patients are Elyse Passmore, BEng, MEng
available with the online version of this article as a data sup- Morgan Sangeux, MSc, PhD
plement at jbjs.org. n Pam Thomason, BPhty, MPT
Jill Rodda, BAppSc(PT), PhD
Hugh Williamson Gait Laboratory,
The Royal Children’s Hospital,
Gregory B. Firth, MBBCh, FCS(Orth)SA, MMed(Orth) Parkville, Victoria 3052, Australia
Paulo Selber, MD, FRACS
H. Kerr Graham, MD, FRCS(Ed), FRACS Susan Donath, MA, BSc
Orthopaedic Department, The University of Melbourne,
The Royal Children’s Hospital, Flemington Road, Parkville, Grattan Street, Parkville,
Victoria 3052, Australia Victoria 3010, Australia
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