Study reference

Study characteristics

Patient characteristics

Intervention (I)

Comparison / control (C)

Follow-up

Outcome measures and effect size

Study quality

Research question: What are the effects of surgical treatment of gait disorders in children and adolescents with spina bifida?

Studies on prenatal surgical treatment

Danzer, 2009

Type of study:

Retrospective study

Country:

USA

Source of funding:

NIH (National Institute of Health)

Inclusion criteria:

-          Less then 26 weeks of gestation

-          Confirmed normal karyotype

-          Absence of associated congenital malformations

-          Maximum lateral ventricular malformations

-          Severe Arnold-Chiari II malformation

-          S1-level lesion or higher

-          Normal leg movement and absence of talipes deformity.

Exclusion criteria:

Not stated

N= 54 (14 eligble patients excluded from fetal surgery due to maternal issues)

Intervention group:

Sex: Not stated

Gestational age fetal surgery: 23.1 ± 1.4 weeks

Control group:

Not applicable

Groups comparable at baseline?

Not applicable

N= 54

Describe intervention:

Fetal myelomeningocele (fMMC) closure

N= 0

Describe control intervention:

Not applicable

Endpoint of follow-up:

Mean follow-up 45 months (range 30-72 months)

For how many participants were no complete outcome data available?

Detailed neurodevelopmental assessment incompleet in 37%. Other measurements complete.

Control group: Not applicable

N (%):%

Reasons for incomplete outcome data described?

Not stated

Outcome measures and effect size (include 95%CI and p-value if available):

Ambulation: 69% walk independently, 24% assisted walkers and 7% wheelchair dependent

Lower Extremity Neuromotor Function: 57.4% better then predicted, 24.1% same as predicted and 18.5% worse as predicted

Predictors of independent walking after fMMC repair:

Lower likelihood to walk independent:

-          Higher-level lesion (>L4, p=0.001)

-          Development of clubfoot deformity (p=0.026)

Not significant associated:

-          Early fetal intervention (<22 weeks’ gestation)

-          Higher birth weight

-          LENF at birth

-          Need for V-P shunt placement

-          Improved LENF at birth

C

No control group

Higly selective study population

Young population, results may not reflect long-term LENF and ambulatory status.

Adzick et al. 2011

Type of study:

Randomized Trial

Country:

USA

Source of funding:

-          Eunice Kennedy Shriver National Institute of Child Health and Human Development

-          Clinical and Translational Science Awards, National Institutes of Health.

Inclusion criteria:

-          Singleton pregnancy

-          Myelomeningocele with the upper boundary located between T1 and S1

-          Evidence of hindbrain herniation

-          Gestational age of at least 19.0 to 25.9 weeks at randomization

-          Normal karyotype

-          U.S. residency

-          Maternal age of at least 18 years

Exclusion criteria:

-          Fetal anomaly unrelated to myelomeningocele

-          Servere kyphosis

-          Risk of preterm birth (including short cervix and previous preterm birth)

-          Placental abruption

-          Body-mass index of 35 or more

-          Contraindication to surgery (including previous hysterotomy in the active uterine segment)

N= 158

Intervention group:

Sex: 55% M/45% F

Gestational age at randomization ± SD: 23.6 ± 1.4 weeks

Control group:

Sex: 36% M/64% F

Gestational age at randomization ± SD: 23.9 ± 1.3 weeks

Groups comparable at baseline?

Yes, except fewer female fetuses and lesion level more severe in prenatal-surgery group (p=0.02 for both comparisons).

N=78

Describe intervention:

Prenatal surgery before 26 weeks of gastation

N=80

Describe control intervention:

Standard postnatal repair

Endpoint of follow-up: age children of 30 months

For how many participants were no complete outcome data available?

First primary outcome:

Intervention group:

12 (15%)

Control group:

13 (16.7%)

Second primary outcome:

22 (31.4%)

Control group:

27 (42.2%)

Reasons for incomplete outcome data described?

Termination of the trial by data and safety monitoring committee, data of 158 patients whose children were evaluated at 12 months.

Outcome measures and effect size (include 95%CI and p-value if available):

-          Deaths: 2 perinatal in each group.

Prenatal vs. Postnatal surgery at 12 months:

-          Fetal or neonatal death, cerebrospinal fluid shunt: 68% vs. 98% (RR: 0.70, 95% CI 0.58-0.84, p<0.001)

-          Shunt placement: 40% vs. 82% (p<0.001)

-          Hindbrain hernation: 64% vs. 96% (RR: 0.67, 95% CI: 0.56-0.81, p<0.001)

-          No significant differences in rates of identification of epidermoid cysts

Prenatal vs. Postnatal surgery at 30 months:

-          No significant difference in Bayley Mental Development Index (p=0.53)

-          Difference between motorfunction and anatomical levels (positive values: indicate function is better than expected): 0.58 ±1.94 and -0.69±1.99 (p=0.001)

-          Able to walk independently: 42% vs. 21% (RR: 2.01, 95CI: 1.16-3.48, p=0.01)

-          Better motor function

-          Better parent-reported selfcare(p=0.02) and mobility (0.003)

-          No significant differences in cognitive scores (p=0.67)

B

Termination of the trial by data and safety monitoring committee.

Young population, results may not reflect long-term ambulatory status

Ambulation secondary outcome

Studies on orthopaedic surgical treatment

Bowman, 2009

Type of study:

Prospective cohort

Country: USA

Source of funding: Not stated

Inclusion criteria:

Newborns with myelomeningocele undergone back closure (tethered cord release)

Exclusion criteria: Not stated

N= 114 (502 newborns with myelomeningocele, 114 patients with symptomatic tethered spinal cord)

Intervention group:

Sex: 49% M /51% F

Mean age ± SD: 7 years (range 7 months – 21.8 years) at time of first untethering

Ambulation:

Control group: Not applicable

Sex: M/F

Mean age ± SD:

Ambulation:

Groups comparable at baseline?

Not applicable

N= 114

Describe intervention:

Untethering procedure

N= 0

Describe control intervention:

Not applicable

Endpoint of follow-up:

Not stated.

Mean follow up after initial untethetering (TCR 1) was 12 years (range: 1 month – 21.8 years), after second release (TCR 2) 4.6 years (1.1 - 11.0 years), third (TCR 3) 3.6 years (0.3 - 7.1 years) and fourth (TCR 4) 3.3 years (2.6 – 4.3 years)

For how many participants were no complete outcome data available?

All children underwent full evaluation 3 months postoperative and follow-up at yearly basis.

Intervention group:

N (%):

Control group:

N (%):%

Reasons for incomplete outcome data described?

Not applicable.

Outcome measures and effect size (include 95%CI and p-value if available):

Scoliosis:

17% showed improvement (mean curve improvement 18˚, range 5-30), 9% stable (curve range 15-46˚) and 24% progreesed (range 44-74˚)

Manual Muscle Test changes:

70% improvement, 28% stable and 2% decline

Lower extremity contracture:

22% improvement, 78% stable

Lower extremity spasticity:

63% improvement, 37% stable

Gait change:

79% improvement, 19% stable and 3% worsened

Back pain:

100% improvement

Urinary changes:

64% improvement, 36% stabilization.

C

No control group

No statistical analysis

Schoenmakers et al. 2003

Type of study:

Prospecitive cohort

Country:

The Netherlands

Source of funding:

Not stated

Inclusion criteria:

-          Neurosurgical treatment for tethered cord syndrome

-          Younger than 18 years of age

Exclusion criteria:

Not stated

N=44

Intervention group:

Sex: 17 M/ 27 F

Mean age ± SD: 6 years and 2 months ± 5 years

Ambulation: 22 patients

Control group: not applicable

Sex: M/F

Mean age ± SD:

Ambulation:

Groups comparable at baseline?

Not applicable

N= 44

Describe intervention:

Neurosurgical untethering

N=0

Describe control intervention:

Not applicable

Endpoint of follow-up:

Mean duration 7 years and 1 month ± 1 year and 8 months.

For how many participants were no complete outcome data available?

Not stated

Intervention group:

N (%): 0

Control group:

N (%):% Not applicable

Reasons for incomplete outcome data described?

No

Outcome measures and effect size (include 95%CI and p-value if available):

26 patients, 6 weeks to 6 months after surgery, ambulation level stable compared to preoperative

Late deterioration of ambulatory status in 5 patients

Obesity (OR: 16,7 95%CI 2,1-133,0) and retethering (OR:20,6 95%CI 2,9-143,6) strongly associated with deterioration of ambulatory status, age had no statistically significant influence

C

No control group

Herman et al., 1993

Type of study:

Retrospective cohort

Country:

USA

Source of funding:

Not stated

Inclusion criteria:

-          Children with myelomeningocele or lipomyelomeningocele

-          Operated for release of tethered spinal cord between 1981 and 1988

Exclusion criteria:

Not stated

N= 153 patients

Intervention group:

Lipomyelomeningocele patients (N=53)

Sex ratio: 1 M: 1.2 F

Mean age (range): 8 years (3-30 years)

Ambulation: not stated

Myelomeningocele patients (N=100)

Sex ratio: 1 M: 1 F

Mean age (range): 6 years (1-19 years)

Ambulation: not stated

Control group: not applicable

Sex: M/F

Mean age ± SD:

Ambulation:

Groups comparable at baseline?

Not applicable

N= 153

Describe intervention:

Reoperation for a tethered cord

N=0

Describe control intervention:

Not applicable

Endpoint of follow-up:

Average of 4 years follow-up

For how many participants were no complete outcome data available?

Intervention group:

N (%): Not stated

Control group:

N (%): not applicable

Reasons for incomplete outcome data described?

Not applicable

Outcome measures and effect size (include 95%CI and p-value if available):

Improvement in gait, increased walking endurance and improved stance noted in 72% of myelomeningocele and 59% of spinal lipoma patients.

C

No control group

Most patients were initial operated at another institution.

Cochrane et al., 1998

Type of study:

Retrospective cohort

Country:

Canada

Source of funding:

Not stated

Inclusion criteria:

-          Undergoing secondary cord untethering

-          Original spina malformation myelomeningecele or hemimyelomengocele

-          Progressive neurological, urological and/or musculoskeletal deterioration

-          Etiology of the deterioration was thought to be due to cord tethering

-          Other etiological possibilities were excluded based on CT myelography or magnetic resonance of the brain and cord.

Exclusion criteria:

Not stated

N= 24 patients

Intervention group:

Sex: Not stated

Mean age (range): 7.8 years (0.72 – 18.2 years)

Ambulation: not stated

Control group: not applicable

Sex: M/F

Mean age ± SD:

Ambulation:

Groups comparable at baseline?

Not applicable

N= 24

Describe intervention:

Placode untethering (secondary cord untethering)

N= 0

Describe control intervention:

Not applicable

Endpoint of follow-up:

3, 12 and 24 months following placode untethering.

For how many participants were no complete outcome data available?

Intervention group:

N (%): Not stated

Control group:

N (%): Not applicable

Reasons for incomplete outcome data described?

No.

Outcome measures and effect size (include 95%CI and p-value if available):

Preoperative:

Changes in ambulatory support requirements and/or the pattern of gait occured in 9 patients

Postoperative:

Changes in muscle power, tone and range of movement translated into alterations in mobility and ambulatory support requirements in 9 patients of whom 5 were initially improved and 4 were unchanged.

C

No control group

Small sample size

No statistical analysis

Stott et al. 1996

Type of study:

Retrospective study

Country: USA

Source of funding:

Not stated

Inclusion criteria:

-          Low-lumbar-level spina bifida

-          Undergone bilateral transfer of the tibialis anterior to the calcaneus

-          Agree to undergo clinical and radiographic evaluation and gait analysis as part of the study

Exclusion criteria:

Not stated

N=7 (respons: 88%)

Intervention group:

Sex: 2 M/ 5 F

Mean age and range: 8 years (3-12 years)

Ambulation: 100% community ambular

Control group: not applicable

Sex: M/F

Mean age ± SD:

Ambulation:

Groups comparable at baseline?

not applicable

N= 7

Describe intervention:

Bilateral transfer of the tibialis anterior to the calcaneus

N= 0

Describe control intervention:

Not applicable

Endpoint of follow-up: average of 40 months (range 24-60 months)

For how many participants were no complete outcome data available?

Intervention group:

N (%): 7 (100%)

Control group:

N (%): not applicable

Reasons for incomplete outcome data described?

Not applicable

Outcome measures and effect size (include 95%CI and p-value if available):

-          All patients remained community walkers

-          Improved velocity (compared to laboratory data, age- and sex-matched controls) P<0.01

-          Increased stride length (compared to laboratory data, age- and sex-matched controls) P<0.01

-          Decreased quadriceps activity at terminal stance with use of braces in free walking (p<0.03)

-          Decreased energy expenditure when braces were worn (p<0.05)

D

Very small sample size

(N=7)

Unclear defined control group.

Park et al. 2008

Type of study:

Retrospective review

Country:

South Korea

Source of funding:

Not stated

Inclusion criteria:

Not stated

Exclusion criteria:

Not stated

N=18

Intervention group:

Sex: 7 M/11 F

Mean age and range: 7 years, 4 months (4 years 11 months – 14 years and 4 months)

Ambulation: 100%

Control group: not applicable

Sex: M/F

Mean age ± SD:

Ambulation:

Groups comparable at baseline?

not applicable

N= 18

Describe intervention:

Surgical treatment of calcaneal deformity

N= 0

Describe control intervention:

Not applicable

Endpoint of follow-up: average of 47 months

For how many participants were no complete outcome data available?

Intervention group:

Not stated

Control group:

N (%): not applicable

Reasons for incomplete outcome data described?

No

Outcome measures and effect size (include 95%CI and p-value if available):

-          No recurrence or worsening of the deformity was observed in any of the patients, and no other types of foot deformity developed after surgery.

-          Postoperative kinematic studies showed a significant (p < 0.0001) increase in peak plantar flexion and a significant decrease in peak dorsiflexion force of the ankle in the stance phase of gait.

-          Peak pressures under the forefoot and midfoot were increased after surgery, and the relative amount of weight-bearing on the heel as compared with the forefoot was shifted toward more equal weight-bearing.

-          Lless improvement in foot-pressure distribution was observed in patients with increased pelvic rotation before surgery.

-          Those patients also had decreased knee extension in stance phase and increased hip abduction and pelvic obliquity both before and after surgery in comparison with patients who had normal pelvic rotation.

C

Small sample size

Unclear defined study population.

Georgiadis et al. 1990

Type of study:

Retrospective study

Country:

USA

Source of funding:

No funding

Inclusion criteria:

Myelomeningocle patients

Had posterior transfer of the anterior tibial tendon between 1969 and 1985.

Exclusion criteria:

Moved away and could not be contacted

N= 20

Intervention group:

Sex: 7 M/13 F

Mean age: 4.6 (1.1 – 10.2) years

Ambulation: 90% community walkers postoperative

Control group:

Not applicable

Groups comparable at baseline?

Not applicable

N= 20

Describe intervention:

Posterior transfer of the anterior tibial tendon

N= 0

Describe control intervention:

Not applicable

Endpoint of follow-up:

6 (2.2 – 17) years

For how many participants were no complete outcome data available?

Operative reports were available for 19 of the 20 patients.

Reasons for incomplete outcome data described?

No

Outcome measures and effect size (include 95%CI and p-value if available):

-          During follow up: 18 of 20 patients community walkers

C

Small study population

Preoperative ambulation was not reported

Schoenmakers et al. 2005

Type of study:

Prospective cohort

Country:

The Netherlands

Source of funding:

Inclusion criteria:

-          Patients with myelomeningocele

-          Younger than 18 years of age

-          Surgical treatment for spinal deformities

-          Willing to participate in study

-          Informed consent signed by parents

Exclusion criteria:

Not stated

N=10

Intervention group:

Sex: 3 M/ 7 F patients

Mean age ± SD: 9.3 ± 3.4

Ambulation: 4 patients

Control group: not applicable

Sex: M/F

Mean age ± SD:

Ambulation:

Groups comparable at baseline?

Not applicable

N= 10

Describe intervention:

Spinal fusion

N=0

Describe control intervention:

Not applicable

Endpoint of follow-up:

18 months after surgery

For how many participants were no complete outcome data available?

Intervention group:

N (%): 0

Reasons for incomplete outcome data described?

Not applicable

Outcome measures and effect size (include 95%CI and p-value if available):

-          3 out of 4 patients ambulation level deteriorated after surgery (no longer able to walk)

-          2 out of 3 exercise walkers lost ablility to walk

-          Mean PEDI-scaled scores on functional skills self-care showed nonsignificant tendency towards deteriorate form pre-surgery to 6 months after surgery

-          18 months after surgery functional skills on self-care borderline improved (p=0,07)

-          Caregiver assistence improved on selfcare (p=0,2) and borderline on mobility (p=0,06) from baseline to 18 months

-          Complications in 8 out of 10 patients

C

Small sample size

No control group

Müller et al. 1992

Type of study:

Prospective cohort

Country:

Sweden

Source of funding:

Bräcke Östergård Research Foundation

Folke Bernadotte Foundation

Bertha and Félix Neubergh Foundation

Inclusion criteria:

-          Patients with myelomeningocele

-          Born between 1964 and 1979

-          Undergone spinal surgery for scoliosis

Exclusion criteria:

Not stated

N=14

Intervention group:

Sex: 5 M/ 9 F

Mean age (range): 12,4 (9-16)

Ambulation: presurgery mean score 2 (non functional ambulators)

Control group: not applicable

Sex: M/F

Mean age ± SD:

Ambulation:

Groups comparable at baseline?

No applicable

N=14

Describe intervention:

Spinal surgery for scoliosis (Harrington, Dwyer or combination)

N=0

Describe control intervention:

Not applicable

Endpoint of follow-up:

Mean age of children at time of follow up 15,6 years (10­20) years

Mean time of 3,4 years (1-8 years) between time of surgery and latest clinical examination

For how many participants were no complete outcome data available?

Not stated

Intervention group:

N (%):

Control group:

N (%): Not applicable

Reasons for incomplete outcome data described?

No

Outcome measures and effect size (include 95%CI and p-value if available):

57% lost some of their ambulation capacity

No influence on activity of daily life

C

Small sample size

No control group

Incomplete outcome data not described

Mazur et al. 1986

Type of study:

Retrospective cohort

Country:

Australia

Source of funding:

Not stated

Inclusion criteria:

Spina bifida cystica

Underwent surgical treatment for paralytic scoliosis between 1974 and 1983

Exclusion criteria:

Not stated

N=49

Intervention group A:

Sex: 5 M/10 F

Mean age ± SD: 11 years and 3 months

Ambulation:

Intervention group B:

Sex: 4 M/3 F

Mean age ± SD: 10 years and 8 months

Ambulation:

Intervention group C:

Sex: 14 M/13 F

Mean age ± SD: 12 years

Ambulation:

Control group: Not applicable

Groups comparable at baseline?

All 3 groups seemed nearly equivalent in terms of deformity, level of lesion, functional capabilities, age and sex distribution

Group A:

N=15

Describe intervention:

Posterior Harrington instrumentation and fusion

Group B:

N=7

Describe intervention:

Anterior Dwyer fusions

Group C:

N=28

Describe intervention:

Combined anterior Dwyer and posterior Harrington fusions

N=0

Describe control intervention:

Not applicable

Endpoint of follow-up:

Group A: 7 years follow up

Group B: 4 years and 9 months follow up

Group C: 4 years and 1 month follow up

For how many participants were no complete outcome data available?

Intervention group:

N (%): 4 (8% loss to follow up)

Control group: not applicable

N (%):%

Reasons for incomplete outcome data described?

Family relocation or death unrelated to surgery

Outcome measures and effect size (include 95%CI and p-value if available):

Group A:

Ability to ambulate or to me mobile in wheelchair deterioted in 27%

No patient improved ambulatory status

Group B:

Ability to ambulate or to me mobile in wheelchair deterioted in 57%

No patient improved ambulatory status

Group C:

Ability to ambulate or to me mobile in wheelchair deterioted in 67%

No patient improved ambulatory status

C

Allocation to treatment groups not random

Small number of patients in treatment groups

No control group

Kahanovitz et al. 1981

Type of study:

Retrospective study

Country:

USA

Source of funding:

Not stated

Inclusion criteria:

-          Patients with myelomeningocele and scoliosis

-          Minimum age 16 years

Exclusion criteria:

-          Primary lordosis or kyphosis

N= 39

Intervention group:

Sex: Not stated

Mean age: 19.9 (range 16.5 – 38) years

Ambulation: 48,7 %

Control group:

Not applicable

Groups comparable at baseline?

Not applicable

N= 15

Describe intervention:

N=12: posterior fusion in situ followed by postoperative immobilization in a Risser cast

N=2: posterior fusion and Harrington instrumentation

N=1: anterior fusion with Dwyer followed by posterior and Harrington.

N=0

Describe control intervention:

Not applicable

Endpoint of follow-up:

Not stated.

For how many participants were no complete outcome data available?

Intervention group:

N (%): Unknown, not stated

Control group:

N (%): Not applicable

Reasons for incomplete outcome data described?

No.

Outcome measures and effect size (include 95%CI and p-value if available):

Preoperative vs. Postoperative:

-          8 patients maintained functional levels

-          1 community ambulator à household ambulator

-          4 household ambulators à balanced sitters

-          2 balanced sitters à unbalanced sitters

C

Unknown follow-up duration

For 8 patients preoperative function unknown

Small sample size

No randomization.

Wai et al. 2005

Type of study:

Retrospective study

Country:

Canada

Source of funding:

Trainee’s Starter Grant from the Hospital for Sick Children’s Research Institute

Inclusion criteria:

-          English speaking

-          Aged 7 to 16 years

-          Spina bifida cystica and scoliosis

Exclusion criteria:

Not had a radiograph of their spine within 6 months or had had an orthopeaedic operative procedure within previous year

N=80

Intervention group:

Sex: 38 M/ 42F

Mean age: 12.5 (7-16) years

Ambulation: 57% non-walker, 10.1% exercise walker, 6.3% household walker, 26.6 community walker

Control group:

Not applicable

Groups comparable at baseline?

Not applicable

N= 24

Describe intervention:

Spinal stabilization surgery consisting of anterior release and discectomy and posterior instrumentation to the pelvis with segmental unit rod fixation

N= 0

Describe control intervention:

Not applicable

Endpoint of follow-up:

Not stated

For how many participants were no complete outcome data available?

19 of 24 children had adequate preoperative radiographs available for comparison (79%).

Reasons for incomplete outcome data described?

No

Outcome measures and effect size (include 95%CI and p-value if available):

Preoperative vs. Postoperative:

-          Cobb angle: 73.7° (SD: 18) vs. 39.0° (SD: 24.8) (P<0.001)

-          Sitting Scale: -1,3 (P=0.003)

-          SBSQ: -11.0 (P=0.002)

-          Ask: -9.3 (P=0.01)

C

No control group

Small sample size

Limted study population: data not extrapolatable to scoliosis beyond 110°

Marshall et al. 1996

Type of study:

Prospective study

Country:

Australia

Source of funding:

Not stated

Inclusion criteria:

Surgical release of knee flexion contractures

Exclusion criteria:

Not stated

N=28

Intervention group:

Sex: Not stated

Mean age (range): 6,4 (3-21)

Ambulation: 5 community walkers

ntrol group: Not applicable

Sex: M/F

Mean age ± SD:

Ambulation:

Groups comparable at baseline?

Not applicable

N=28

Describe intervention:

Surgical release of knee flexion contractures

N=0

Describe control intervention:

Not applicable

Endpoint of follow-up:

Mean period of follow up 13 years (4-20 years)

For how many participants were no complete outcome data available?

Intervention group: Not stated

N (%):

Control group: Not applicable

N (%):%

Reasons for incomplete outcome data described?

No

Outcome measures and effect size (include 95%CI and p-value if available):

The thoracic level group remained non ambulators or non functional ambulators

L3/L4 group: 4 non walkers became non functional walkers, 1 non functional walker progessed to household ambulator, 3 household ambulators became community ambulators

C

Small sample size

No control group

No statistical analysis

Abraham et al. 1977

Type of study:

Retrospective cohort

Country:

Great Brittian

Source of funding:

Not stated

Inclusion criteria:

Patients with myelomeningocele treated surgcally for knee flexion contracture

Exclusion criteria:

Not stated

N= 76

Intervention group:

Sex: 35 M/ 41 F

Age range: 2 – 14 years

Ambulation: 24%

Control group: not applicable

Sex: M/F

Mean age ± SD:

Ambulation:

Groups comparable at baseline?

Not applicable

N= 76

Describe intervention:

Surgical treatment for flexion contracture of the knee

N= 0

Describe control intervention:

Not applicable

Endpoint of follow-up:

Average follow up 4 years with minimum of 1 year

For how many participants were no complete outcome data available?

Intervention group:

N (%): Not stated

Control group:

N (%): Not applicable

Reasons for incomplete outcome data described?

No.

Outcome measures and effect size (include 95%CI and p-value if available):

45 patients were active walkers at follow up compared to 18 before.

C

No control group

Snela et al. 2000

Type of study:

Retrospective cohort

Country:

Germany

Source of funding:

Not stated

Inclusion criteria:

Sought consultation between 1980 and 1995

Flexion contracture suitable for correction

Exclusion criteria:

Not stated

N= 80

Intervention group:

Sex: not stated

Age range: 2 – 19 years

Ambulation: not stated

Control group: not applicable

Sex: M/F

Mean age ± SD:

Ambulation:

Groups comparable at baseline?

Not applicable

N= 80

Describe intervention:

Surgical treatment for flexion contracture of the knee

N= 0

Describe control intervention:

Not applicable

Endpoint of follow-up:

Average follow up 5.1 (1.3 – 17 years)

For how many participants were no complete outcome data available?

Intervention group:

N (%): Not stated

Control group:

N (%): Not applicable

Reasons for incomplete outcome data described?

No.

Outcome measures and effect size (include 95%CI and p-value if available):

-          74% very good long term results (full knee extension, flexion contracture <10˚, bracing without problems, daily use braces for standing or walking, walking without braces)

-          20% good long term results (knee flexion contracture recurrence up to 30˚, posibility to wear brace, no pressure sore, limited use of brace for standing or walking exercise)

-          6% unsatifactory long term result (recurrence of more than 30˚, inability to wear braces for standing and walking exercises)

C

No control group

Dunteman et al. 2000

Type of study:

Retrospective review

Country:

USA

Source of funding:

Not stated

Inclusion criteria:

All patients with high sacral level myelomeninggocele who had pre- and postoperative gait analysis with both kinectic and kinematic data as well as surgical correction of external tibial torsion form October 1993 to February 1999.

Exclusion criteria:

Not stated

N= 8

Intervention group:

Sex: 4 M/ 4 F

Mean age ± SD: 12.9 (range 8-20.2) years

Ambulation: all ambulatory

Control group:

Not applicable

Groups comparable at baseline?

Not applicable

N= 8

Describe intervention:

Distal tibia and fibular internal derotation osteotomies

N= 0

Describe control intervention:

Not applicable

Endpoint of follow-up:

37 months (11-49 months)

For how many participants were no complete outcome data available?

Intervention group:

Not stated

Reasons for incomplete outcome data described?

Not stated

Outcome measures and effect size (include 95%CI and p-value if available):

-          Improvement in the abnormal internal knee varus moment (P<0.005)

-          Increase in stance phase knee extension (P<0.01)

-          Resolution of preoperative knee pain in 3 patiens.

D

Very small sample size (N=8)

No control group

Alman et al., 1996

Type of study:

Retrospective cohort

Country:

Canada

Source of funding:

Medical Research Council Scholarship

Inclusion criteria:

-          Patients with spina bifida born between 1974 and 1985

-          Bilateral lesions at L3 and L4

-          L3 involvement on one side and L4 on the other

Exclusion criteria:

Undergone operations in the previous four months

N= 52 (respons 88%)

Intervention group:

Sex: 11 M/ 19 F

Mean age ± SD: 14.5 (5.8)

Ambulation: 26

Control group:

Sex: 12 M/ 10 F

Mean age ± SD: 16.4 (5.4)

Ambulation: 20

Groups comparable at baseline?

Trend towards higher levels of motor involvement in conservative group

N=30

Describe intervention:

Operative relocation of the hip

N=22

Describe control intervention:

Conservative

Endpoint of follow-up:

Not stated

For how many participants were no complete outcome data available?

Intervention group:

N (%): Not stated

Control group:

N (%): Not stated

Reasons for incomplete outcome data described?

No.

Outcome measures and effect size (include 95%CI and p-value if available):

Functional level of walking did not differ significantly (p = 0.05)

C

No random allocation of patients to treatment or control group

Small number of patients

Lorente Moltó et al., 2005

Type of study:

Retrospectively cohort

Country:

Spain

Source of funding:

Not stated

Inclusion criteria:

-          Myelomeningocele patients

-          Diagnosed and treated from neonatal period between october 1975 and march 1992

-          L3 paralysis

Exclusion criteria:

-          Death because neurological complications before adolescence

N= 29

Intervention group:

Sex: 17 M/ 12 F

Mean age (range): 26.5 months (11 months – 6 years)

Ambulation: 11 non-walkers (38%)

Control group: not applicable

Sex: M/F

Mean age ± SD:

Ambulation:

Groups comparable at baseline?

Not applicable

N= 29

Describe intervention:

Posterolateral iliopsoas transfer. Surgical technique as described by Sharrard.

N= 0

Describe control intervention:

Not applicable

Endpoint of follow-up:

Mean follow up 21 years (range 11-28 years)

For how many participants were no complete outcome data available?

Intervention group:

N (%): Not stated

Control group:

N (%): Not applicable

Reasons for incomplete outcome data described?

No.

Outcome measures and effect size (include 95%CI and p-value if available):

-          At preschool age and in adolescence proportion of effective walkers did not differ significantly (86.2 vs. 75.8%, p=0.315)

-          All adolescence community ambulators kept ambulatory status in adulthood.

C

No control group

Small number of patients included

Selective population (only L3 paralysis).

Bazih et al., 1981

Type of study:

Retrospective cohort

Country:

USA

Source of funding:

Not stated

Inclusion criteria:

-          Patients with myelomeningocele

-          4 years or older

Exclusion criteria:

Not stated

N= 74 patients

Intervention and control group:

Sex: 29 M/ 45 F

Mean age (range): 10 years and 5 months (4 years and 1 month – 20 years)

Ambulation: 28 patients

Control group:

Sex: M/F

Mean age ± SD:

Ambulation:

Groups comparable at baseline?

No neurologic features distinguishing operated and nonoperated groups. Operated group older.

N= 18

Describe intervention:

Surgery for specific purpose of attaining reduction in an unstable hip

N= 56

Describe control intervention:

No operation

Endpoint of follow-up:

Unclear, not stated

For how many participants were no complete outcome data available?

Intervention group: unclear, not stated

N (%):

Control group: unclear, not stated

N (%):%

Reasons for incomplete outcome data described?

No.

Outcome measures and effect size (include 95%CI and p-value if available):

-          16 hips successfully reduced, 13 hips (45%) were redislocated or subluxed.

-          Little difference between nonoperated and operated groups regarding ambulatory capabilities or the percentage of ambuators with dislocated hip.

C

Small intervention group

Use of historic controls

No random allocation to intervention or control group