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Table 1 Characteristics of the included studies

From: Orthopedic management of myelomeningocele with a multidisciplinary approach: a systematic review of the literature

Study

Study type

Sample

Age, gender, follow-up

Management

Orthopedics

Urology

Neurosurgery

Neurology

Gastrointestinal

Multidisciplinary

Torosian 2000 [42],

USA

Retrospective cohort

- N = 27 (38 feet)

- 38 feet in 27 patients,17 right and 21 left

- Thoracic: 1 (4%)

- Low lumbar: 18 (63%)

- Sacral: 10 (33%)

- Mean age at surgery: 10.88 ± 2.50

- Gender: 16 (59.2%) males

- Mean FU: > 5 years

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Sponseller 2000 [41], USA

Retrospective cohort

- N = 48

- Children and adolescents who underwent Scoliosis surgery from 1986 to 1996

- Mean age at surgery: Infected 14.1 years, uninfected matched group 14 years

- Note: CP patients also included

- Gender: N/A

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Frischhut 2000 [12], Austria

Retrospective cohort

- N = primarily 107 (final 91 patients and 182 feet)

- Thoracic: 51 (28%)

- Lumbar: 73 (40%)

- Sacral: 58 (32%)

- Patients with SB born before 1982

- Patients older than 15 years old

- Gender: N/A

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Parsch 2001 [13], Germany

Retrospective cohort

- N = 54

- Patients with surgical management for paralytic scoliosis between 1984 and 1996

- Mean age at surgery: 13.24 ± 2.09 years

- Gender: 45/75 males (initial population before exclusion)

- Mean FU: 3.3 years

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Crawford 2003 [20], USA

Retrospective cohort

- N = 11

- Neonates with MM who had a kyphectomy in conjunction with dural sac closure between 1980 and 2000

- Mean gestational age: 38 weeks

- Mean age at surgery: 36 h

- Gender: 9 (81.8%) males

- Mean FU: 7 years and 4 months (range 44–174 months)

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Ozerdemoglu 2003 [43], USA

Retrospective cohort

- N = 105 (26 patients with scoliosis and MMC)

- Patients with scoliosis and syringomyelia treated between 1941 and 1998

- Age: N/A

- Gender: 47% males

- Mean FU: 12.8 ± 9.6 years (range: 2–45.2 years)

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Ulsenheimer 2004 [44], Brazil

Retrospective cohort

- N = 31

- MMC neonates born between 1990 and 2000

- Median age at MMC correction: 4 days (range 1–44 days)

- Mean gestational age: 39.5 ± 1 week

- Gender: 16/31 (51.6%) males

- Median FU: 24 months (range 6–68 months)

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Erol 2005 [26], Turkey

Prospective cohort

- N = 26 (28 hips)

- Patients underwent surgery for hip instability between 1998 and 2001

- Patients previously underwent various surgical interventions other than orthopedic procedures, mainly urological and neurosurgical interventions (mean number of previous operations 2; range 1–4)

- Mean age at surgery: 4.5 years (range 3–6 years)

- Gender: 16/26 males

- Mean FU: 38 months (range 30–48 months)

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Lemelle 2006 [45], France

Retrospective cohort

- N = 769 (421 were finally included)

- Patients with at least 10 years of age who underwent surgery between 2003 and 2004

- All presented with MMC at the neonatal period

- Mean age: 22.1 years (range 10–47.5 years)

- Gender: 191/421 (45%) males

- Medically treated for urinary incontinence (n = 191)

o Age: 21.7 ± 7.9

o Gender: 104/191 (54.4%) males

o FU: N/A

- Surgically treated for urinary incontinence (n = 230)

o Age: 23.0 ± 7.6

o Gender: 126/230 (54.7%) males

o Mean FU after initial surgery: 9.25 years

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Gerlach 2009 [46], USA

Prospective cohort

- N = 36

- 18 patients (28 feet) with MMC and clubfoot between 2001 and 2006

- 20 patients (35 feet) with idiopathic clubfoot prospectively gathered

- Both groups managed with the Ponseti method

- Mean age at first cast for MMC patients: 12.4 weeks (range 1.2–25.9 weeks)

- Gender: 9/16 males in MMC group

- Mean FU for MMC patients:33.8 months (range 25.0–42.7 months)

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Huang 2010 [49], China

Retrospective cohort

- N = 10

- Cervical MMC patients

- Median age at management: 3 months (range 9 days–8 years)

- Gender: 6 (60%) males

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Akbar 2010 [48], Germany

Retrospective cross-sectional

- N = 862

- Patients with MMC

- 92 (10.7%) patients suffered from fractures

- Mean age: 10.2 ± 7.0 years

- Gender: 391 (45.4%) males

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Akbar 2010 [47], Germany

Retrospective cross-sectional

- N = 32

- MMC patients with Lumbar Kyphosis operated between 1994 and 2008

- Mean age at surgery: 7.3 ± 3.4 years

- Gender: 13/32 (40.6%) males

- Mean FU: 7.6 years (range 1.5–14.3 years)

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Yildirim 2015 [14], Turkey

Retrospective cohort

- N = 13

- Patients with hip subluxation or dislocation secondary to lower lumbar MMC, surgically treated between 2004 and 2013

- Mean age at surgery: 6 years (range 21 months–12 years)

- Gender: 8/13 (61.5%) males

- Median FU:

o Muscle transfer group: 36 months (range 14–122 months)

o Non-muscle transfer group: 40 months (range 19–66 months)

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Wang 2015 [50], USA

Case-control

- N = 226,709 cases and 888,774 controls

- Nationwide Emergency Department Sample (NEDS) data between 2006 and 2010

- Mean age: 28.23 ± 0.58 years cases and 28.39 ± 0.13 years controls

- Gender: 98,365 (43%) males in case group and 386,181 (43%) males in control group

- FU: N/A

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Januschek 2016 [53], Germany

Prospective cohort

- N = 48

- Meningocele:1 case

- MMC: 29 cases

- Myeloschisis:18 cases

- Mean gestational age at birth: 38 weeks

- Gender: 25 (52%) males

- FU: 9 weeks to 8 years

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Altiok 2016 [51], USA

Retrospective cohort

- N = 65 patients (MMC: 43 and Lipomeningocele: 22)

- Thoracic and upper levels: 12

- L3–L4: 42

- Sacral: 11

- Mean age

o Tethered cord release: 6.2 years

o Definitive spine surgery: 11.6 years

- Gender (TCR): 9 (43%) males

- Mean FU: 3.8 years

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El-fadl 2016 [52], Egypt

Prospective cohort

- N = 27 (54 feet)

- MMC patients with clubfoot (Talipes equinovarus)

- Mean age at first cast: 5.9 weeks (range: 3–8 weeks)

- Gender: 9/24 (37.5%) males

- Mean FU: 27 months (range 24–34)

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Matar 2017 [54], UK

Retrospective cohort

- N = 11 (18 feet)

- 18 MMC-associated clubfoot (7 bilateral deformities)

- Mean age: 4.7 weeks (range 2–8 weeks)

- Gender: 4 (36.3%) males

- Mean FU: 4.5 years (range 3–9)

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Ryabykh 2018 [21], Russia

Retrospective cohort

- N = 20

- Patients with MMC-related spinal deformity

- Mean age at treatment: 6.3 years

- Gender: 12 (60%) males

- Mean FU: 34.5 months

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Spazzapan 2018 [56], Slovenia

Retrospective cohort

- N = 20

- Patients with MMC treated between 2007 and 2017

- All patients were operated within the first 48 h of life

- Gender: 9/20 (45%) males

- Mean follow-up: 7.7 years

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North 2018 [55], Canada

Retrospective two-time cohorts

- N = 101 (C1) + 51 (C2)

- Two cohorts: C1) born with MMC between 1971 and 1981 (data available from previously published work); C2) born with MMC between 1996 and 2006

- Delivery (C2): 51% caesarean section, 49% spontaneous vaginal delivery

- Mean gestational age: 37.8 weeks

C1

- Mean age at last FU (83 patients): 13.4 years (range 8.6–19.6 years)

- Gender: 56/101 (55%) females

C2

- Mean age at last FU (46 patients): 15.7 years (range 10.5–20.3 years)

- Gender: 18/46 (39%) females

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Beuriat 2018 [9], France

Retrospective cohort

- N = 46

- MMC children

- Sacral or lower lumbar (≤ L4): 27

- Higher lumbar (L1-L3): 5

- Thoracic: 14

- Delivery: 20 cesarean sections and 26 vaginal delivery

- Mean gestational age at birth: 38 weeks and 6 days (range 33 weeks and 3 days–41 weeks)

- Gender: 24/46 (52.2%) females

- Mean duration of FU: 8.1 ± 4.6 years

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Goldstein 2019 [10], USA

Multidisciplinary, retrospective cohort

- N = 208

- Patients undergoing scoliosis corrective surgery

- Prophylactic untethering: 32 concomitant untethering, 21 untethering within 3 months prior, 155 without untethering

- Mean age at treatment: 9.4 years (range: 1–17 years)

- Gender: 99 (47.6%) males

- FU: 90 days

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Sileo 2019 [57], UK

Retrospective cohort

- N = 241

- Prenatally diagnosed isolated spina bifida

- 67 underwent postnatal surgery

Maternal parameters:

- Mean maternal age: 30.1 ± 5.9 years

- Mean gestational age at diagnosis: 20.8 ± 1.9 weeks

- Mean gestational age at birth: 38 ± 2.0 weeks

Child parameters

- Age at surgery: first few days

- Mean age of children at the time of study: 6 years (range 0–18 years)

- Mean duration of FU: 8.1 ± 4.1 years

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Beuriat 2019 [11], France

Retrospective Cohort

- N = 29

- MMC with an anatomical level of L4 and below

- 12 sacral

- 17 lumbar

- Age range at surgery: 0–97 days

- Mean age at delivery:

o Cesarean section: 38 weeks and 2 days (range 37 weeks and 6 days–39 weeks)

o Vaginal delivery: 39 weeks and 2 days (range 37 week and 7 days–41 weeks)

- Gender: 13 (44.8%) females

- Mean FU: 9.1 ± 4.9 year

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  1. FU follow-up, N/A not available