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Surgical Management of Myelomeningocele-Related Spinal Deformities
Original Article
Surgical Management of Myelomeningocele-Related Spinal Deformities Sergey O. Ryabykh, Olga M. Pavlova, Dmitry M. Savin, Alexander V. Burtsev, Alexander V. Gubin
OBJECTIVE: To evaluate the optimal timing and type of surgical treatment of myelomeningocele (MMC)-related spinal deformities and long-term follow-up of surgical treatment.
-
METHODS: We reviewed and presented clinical pictures, treatment strategies and results of 20 patients with MMC-related spinal deformities treated at our center between 2010 and 2017.
problems, but there are no alternative procedures for cases of heavy rigid kyphosis.
-
RESULTS: The average patient age was 6.3 years. The average preoperative neurologic status according to a modified Japan Orthopedic Association (mJOA) scale was 7.3 points (Benzel’s modification). Average functional status was 41 points according to a functional independent measure scale (FIM). The average angle of kyphosis was 83.7 , that of scoliosis was 36.7 , and that of lordosis was 67 (Cobb angles). The average duration of surgery was 234 minutes, and the average total blood loss was 175 mL. The average angle of kyphosis correction was 61 , that of scoliosis correction was 25 , and that of lordosis correction was 25 (Cobb angles). The average duration of hospitalization was 16.6 days, and the average follow-up was 34.5 months. The total number of complications was 13. Reoperation was required in 9 cases. Neurologic status according to the mJOA scale improved by 0.6 point on average. Functional status according to the FIM increased by 6.6 points on average.
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CONCLUSIONS: Early surgical correction of MMCrelated spinal deformities improves body balance and quality of life. The dual growing rod technique is safe and effective in cases of moderate neuromuscular spinal deformities at an early age. Kyphectomy is a challenging procedure with high complication rates, especially skin
-
INTRODUCTION
S
pinal deformities are common in patients with myelomeningocele (MMC).1-20 Scoliosis occurs in about 94% of thoracolumbar MMC21 and in about 52% overall.22 The frequency of kyphosis is 10%e20%,8,9,20 and that of lumbar hyperlordosis is 1.5%.23 Progressive spinal deformity is a significant comorbidity associated with MMC. It leads to loss of truncal height and causes sitting, breathing, eating, and urination difficulties due to increased chest and abdominal pressures.3-8,10,13,24-27 These deformities are caused by neuromuscular disturbances and vertebral abnormalities.1,4,7,9,12,26 Spinal deformities associated with MMC can be divided into 2 major groups: predominantly neuromuscular kyphoscoliosis or lordoscoliosis2,12,13,20,28,29 and severe rigid kyphosis or sharp-angled kyphosis.3-10,13-15,19,25 The dual growing rod technique is safe and effective in cases of moderate neuromuscular kyphoscoliosis or lordoscoliosis at an early age, but associated with large number of rod fractures.30-32 Kyphectomy is the surgery of choice in cases of rigid MMC-related kyphosis but is associated with numerous complications, including skin and wound infection, nonfusion, and others.6,7,13,16,26,33,34 Here we present our experience with 20 consecutive patients with different types of MMC-related spinal deformities. This experience focuses on early deformity correction using the growing dual rod technique and instrumented fusion with or without vertebrectomy. To date, only a few studies have described successful treatment of MMC-related spinal deformities and longterm outcomes. We explored the following questions: Is early surgical correction of MMC-related spinal deformities safe and effective? Can it reduce skin breakdown, instability, and
Key words Myelomeningocele-related spinal deformity - Post-myelomeningocele syndrome - Sharp-angled kyphosis
Russian Ilizarov Scientific Center for Restorative Traumatology and Orthopaedics, Neurosurgical Department, Kurgan, Russia
Abbreviations and Acronyms FIM: Functional Independent Measure mJOA: Modified Japanese Orthopedic Association MMC: Myelomeningocele MRI: Magnetic resonance imaging VAS: Visual analog scale
Citation: World Neurosurg. (2018). https://doi.org/10.1016/j.wneu.2018.01.058
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To whom correspondence should be addressed: Olga M. Pavlova, M.D. [E-mail: [email protected]]
Journal homepage: www.WORLDNEUROSURGERY.org Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2018 Elsevier Inc. All rights reserved.
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SURGICAL MANAGEMENT OF MYELOMENINGOCELE-RELATED SPINAL DEFORMITIES
paradoxical pain without a major loss of functional abilities? Is it effective in the long-term follow-up? Can early surgical correction of MMC-related spinal deformities improve body balance and quality of life? METHODS We evaluated a group of patients with MMC-related spinal deformity treated at our center between 2010 and 2017. Inclusion criteria and indications for surgery were progressive spinal deformity >20 (Cobb angle) in any plane, accompanied by spinal imbalance, low Functional Independent Measure (FIM) score, restricted verticalization, presence of pressure sores, signs of spinal instability, neurologic deterioration, including urodynamic studies (post-void residual volume, urine culture, uroflowmetry), and persistent back pain. We assessed neurologic symptoms using the modified Japanese Orthopedic Association (mJOA) scale (Benzel’s modification),35 the FIM scale, and a visual analog scale (VAS) for pain. All patients underwent spinal and craniovertebral X-ray, computed tomography scan, and magnetic resonance imaging (MRI) preoperatively; spinal computed tomography scan and X-ray at 48 hours postoperatively; and spinal X-ray at 6 months, 1 year, and 2 years after surgery. All operations were performed by 2 surgeons, S.O.R. and/or D.M.S. A literature review was performed in English and Russian in PubMed, Google Scholar, and eLibrary databases. Search queries included myelomeningocele-related spinal deformity, sharpangled kyphosis, myelomeningocele kyphosis, kyphectomy, and myelomeningocele spinal deformity. RESULTS Preoperative Clinical and Radiologic Study Data The 20 patients included 8 females (40%) and 12 males (60%), with an average age of 6.3 years (median, 4.3 years; range, 2e26 years). There were 12 toddlers, 7 grade schoolers, and 1 adult (age 26 years) (Table 1). All patients had undergone previous MMC repair. All patients had normal upper extremity muscle strength and sensation (mJOA score 6) and lower extremity paraparesis with micturition disturbance. The average preoperative neurologic status according to the mJOA was 7.3 points (median, 7.0; range, 6e11). The average FIM score was 41 points (median, 34.5; range, 18e108). Five patients (25%) had intermittent focal back pain (VAS score 2e4), and 4 (20%) had psychomotor retardation. All patients experienced chronic recurrent urinary tract infections, 2e3 times per year. Two patients had cardiac pathology, one with moderate tachycardia with mitral valve prolapse and the other with bradycardia due to severe sinus node dysfunction, requiring a pacemaker. No patients had significant respiratory issues. Eleven patients (55%) had kyphoscoliosis (Figure 1A), 4 (20%) had lordoscoliosis (Figure 1B), 4 (20%) had lumbar kyphosis only (Figure 1C), and 1 (5%) had a “flat” lower spine (Figure 1D). Rigid sharp-angled lumbar kyphosis (>90 Cobb angle) was present in 6 patients (30%), and skin ulcerations was present in 3 of these patients (Figure 2). Moderate lumbar or thoracolumbar kyphotic deformity (30e90 Cobb) was present in 9 patients (45%).
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The average angle of kyphosis was 83.7 Cobb (median, 75 ; range, 45 e134 ), that of scoliosis was 36.7 Cobb (median, 36 ; range, 11 e77 ), and that of lordosis was 67 Cobb (median, 71 ; range, 43 e84 ). Brain MRI demonstrated Chiari malformation and cerebral atrophy in 19 patients (95%). Cervicothoracic MRI revealed syringomyelia in 7 patients (35%). Thoracolumbar MRI unveiled marked hypoplasia of the thoracolumbar spinal cord in 11 patients (55%), of the lumbar spinal cord in 4 patients (20%), and of the conus medullaris/cauda equina in 5 patients (25%) (Table 1). Spina bifida was present in all patients, and 12 patients had accompanying vertebral anomalies (60%). Seven patients (35%) had rib anomalies. A ventriculoperitoneal shunt at an early age was performed in 12 patients (60%), and an endoscopic third ventriculostomy was performed in 1 patient (5%). Chiari decompression surgery was performed in 4 patients (20%). External spinal distraction was performed in 2 patients with sharp-angled kyphosis with apical ulcer, with 35 days of external distraction in one patient (Figure 3) and 25 days of external distraction in the other. One patient had undergone surgery for deformity, including kyphectomy with additional laminar-hook fixation, in another hospital 5 years before presentation to our center. Surgery All 20 patients underwent surgery, which included instrumented fusion by pedicle screw-rod constructs (Table 2). Pedicle screw placement was performed by a freehand technique, standard in “normal” vertebrae and modified in spina bifida vertebrae (Figure 4). Dual growing rod constructs were implanted in 6 patients (30%) with mobile kyphoscoliosis and lordoscoliosis. In 1 patient with local rigid kyphosis, growing rod implantation and bone-diskbone osteotomy was performed at 2 levels (L3-4 and L4-5) simultaneously. Another patient underwent 2-stage surgery with dual rib-iliacebased hook growing rod construct implantation at age 3 years with staged distractions over a 2-year period, followed by kyphectomy with pedicle screw fixation at age 5 years. Kyphectomy with pedicle screw fixation was performed in 11 patients with rigid kyphosis (55%; Figure 4). One of these patients underwent additional bone-disk-bone osteotomies at the 2 lower levels (L1-2 and L3-4) to reduce pressure on the scar tissue of the back. Spondylodesis with local pedicle screw fixation without osteotomy was performed in 4 patients (20%), including moderate local lordoscoliosis in 3 and moderate local kyphoscoliosis in 1. The atretic thecal sac was preserved in all patients to prevent disruption of cerebrospinal fluid dynamics and shunt malfunction. Screw malposition was seen in 6 patients (15 screws total), including 5 (12 screws) with permissible malposition and 1 (3 screws) with inadmissible malposition, necessitating reoperation. The average duration of surgery was 234 minutes (median, 225 minutes; range, 110e390 minutes). The average total blood loss was 175 mL (median, 100 mL; range, 20e650 mL). Postoperative Clinical and Radiologic Study Data The average angle of kyphosis correction was 57 Cobb (median, 56 ; range, 19 e96 ), that of scoliosis correction was 25 Cobb
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2018.01.058
Patient, Sex
Level of Cord Hydrocephalus Dysplasia Syringomyelia ArnoldeChiari II
Other Vertebral Abnormalities
1, f
C5, Th1-3, T7-S5
Block laminae C6-7
VPS
LE
Th
C3
2, m
Th3-S5
BV Th3, HV Th6, Th8, Th11, block Th9-10
-
ThL
-
C3 (PFD)
3, f
Th12-S5
-
VPS
LE
Th
C3
4, m
Th8-S5
-
VPS
ThL
-
C2 (PFD)
5, m
Th5-S5
HV Th8, BV L1-2
VPS
ThL
-
C3 (PFD)
RUI, PMR
Comorbidities
Lumbar/Thoracolumbar Lumbar Lordosis Scoliosis Kyphosis (Cobb Angle) (Cobb Angle) (Cobb Angle)
RUI, PMR
49
-
21
Mitral valve prolapse, RUI, PMR
60
-
70
RUI, PMR
130
-
35
Epilepsy, sinus node dysfunction required pacemaker, RUI, PMR
73
-
49
75
-
10
Th6-S5
Block L1-2
-
ThL
-
C2
RUI, PMR
61
-
28
7, f
Th8-S5
Block laminae Th1-4, HV Th10, block Th11-L1
ETVS
ThL
-
C3
RUI, PMR
101
-
28
8, m
L2-S4
-
-
CM CE
-
C2
RUI, PMR
-
60
77
9, m
Th10-S4
-
VPS
ThL
-
C3
RUI, PMR
121
-
11
10, m
Th6-S5
-
VPS
ThL
-
C3
RUI
83
-
-
11, m
Th7-S5
-
VPS
ThL
C
C3
RUI, PMR
134
-
24
12, f
L2-S5
-
VPS
CM CE
-
C1
RUI
-
82
38
13, f
L3-S5
BV L5
-
CM CE
-
-
RUI
-
84
53
14, m
Th12-S5
BV L2-3, diastema L3
-
CE
-
C2
RUI
-
43
37
15, f
Th12-S5
BV Th11, HVTh12 dex, block Th3-5, diastema S1-2, sacral hypoplasia
VPS
LE
CTh
C2
RUI, PMR
45
-
45
16, m
Th10-S5
-
VPS
LE
-
C2
RUI, PMR
47
-
22
17, f
L5-S5
-
-
CM CE
Th
C2 (PFD)
RUI, PMR
3
-
40
18, m
Th8-S5
Block Th5-6
-
ThL
Th
C2
RUI, PMR
98
-
-
19, f
Th10-S5
-
VPS
ThL
-
C3
RUI, PMR
71
-
-
20, m
Th5-S5
Block Th5-8
VPS
ThL
Th
C6
RUI, PMR
107
-
-
f, female; m, male; C, cervical; Th, thoracic; L, lumbar; S, sacral; BV, butterfly vertebrae; HV, hemivertebrae; VPS, ventriculoperitoneal shunt; LE, lumbar enlargement; CM, conus medullaris; CE, cauda equina; PFD, posterior fossa decompression; RUI, recurrent urinal infection; PMR, psychomotor retardation; dex, dexter.
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6, m
SURGICAL MANAGEMENT OF MYELOMENINGOCELE-RELATED SPINAL DEFORMITIES
Spina Bifida
SERGEY O. RYABYKH ET AL.
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Table 1. Clinical and Radiologic Features of 20 Patients with MMC-related Spinal Deformity
ORIGINAL ARTICLE SERGEY O. RYABYKH ET AL.
SURGICAL MANAGEMENT OF MYELOMENINGOCELE-RELATED SPINAL DEFORMITIES
Figure 1. Lateral and anteroposterior X-rays of 4 patients with different types of myelomeningocele-related spinal deformity: (A)
(median, 23 ; range, 1 e62 ), and that of lordosis correction was 25 Cobb (median, 28 ; range, 9 e35 ) (Table 2). The postoperative period was complicated by marginal wound skin necrosis requiring a cutaneous plastic by a graft in 1 patient (Figure 5A). An impermissible screw malposition necessitated reoperation in 1 patient (Figure 5B), and a superficial wound healing problem was seen in 1 patient. The average length of hospital stay was 16.6 days (median, 13 days; range, 7e36 days). Implant-associated long-term complications were present in 10 patients (50%) (Table 3). Eight patients had skin problems above screw heads (40%), 5 of whom required surgery (25%). One
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kyphoscoliosis; (B) lordoscoliosis; (C) severe lumbar kyphosis; and (D) scoliosis and “flat” back.
patient experienced a rod fracture at 3 years after surgery (Figure 5C), and 1 patient had an iliac screw fracture at 2 years after surgery (Figure 5D). All 6 patients with growing rod systems were followed closely, with staged distraction performed every 6 months. The average follow-up was 34.5 months (median, 31 months; range, 6e67 months). Neurologic status according to the mJOA improved by an average of 0.6 point (median, 0; range, 0e2). Average functional status improved by 6.6 points on the FIM score (median, 6; range, 0e18). All patients were free of back pain at follow-up (0 points on the VAS).
WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2018.01.058
ORIGINAL ARTICLE SERGEY O. RYABYKH ET AL.
SURGICAL MANAGEMENT OF MYELOMENINGOCELE-RELATED SPINAL DEFORMITIES
Figure 2. Primary local back view of an 8-year-old girl with rigid sharp-angled myelomeningocele-related kyphosis with skin ulceration.
DISCUSSION Surgical treatment of MMC-related spinal deformities is often required for correction of deformity and establishment of truncal balance, reduction of degenerative spinal changes that can trigger pain in older age, expansion of respiratory capacity and abdominal space, and minimization of skin ulcers over a gibbus.14,36-39 Predictors of Various Types of MMC-Related Spinal Deformities A high incidence of spinal deformity is associated with thoracolumbar MMC21; scoliosis occurs in approximately 94% of these patients, compared with approximately 52% overall.22 Almost all of our patients with kyphoscoliosis had thoracolumbar MMC with thoracic spinal cord and/or lumbar enlargement hypoplasia. Lordoscoliosis was associated with lumbosacral MMC and conus
Figure 3. Local status and X-ray of an 8-year-old girl with rigid sharp-angled myelomeningocele-related kyphosis after implantation of external
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medullaris and/or cauda equina dysplasia. Severe rigid sharpangled kyphosis was associated with a greater frequency of a syringomyelia and additional spinal congenital malformations (Table 1). Dual Growing Rod Technique in Mobile MMC-Related Kyphoscoliosis and Lordoscoliosis The peak of spinal growth is during the first 5 years of life (2 cm/ year). The growth rate subsequently diminishes over the next 5e10 years before a second growth spurt is observed.40 Traditional casting and orthotic treatment are difficult and sometimes ineffective in young children, possibly causing immature rib cage deformities, pulmonary problems, and skin problems (i.e., pressure sores).
distraction device. (A) Local view of applied external distraction device. (B and C) Anteroposterior and lateral X-rays on day 34 after surgery.
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Patient, Sex
Age (Years)
Kyphectomy
Level and Type of Instrumental Fusion
Duration of Surgery (minutes)
Blood Loss (mL)
Lumbar Kyphosis Correction (Cobb Angle)
Lumbar Hyperlordosis Correction (Cobb Angle)
Scoliosis Correction (Cobb Angle)
Short-Term Complications
Days of Postoperative Treatment
4.2
-
DGRS Th5-S2 adult
155
20
41
15
11
2, m
6.8
Th10-12
Final Th2-S1, pediatric
260
250
35
62
23
3, f
8.0
L3-L4
Final,Th3-S1-iliac, pediatric
240
500
70
25
4, m
3.7
-
DGRS Th5-L5, pediatric
190
100
37
38
5, m
3.1
L1-L3
Final Th6-S1, pediatric
235
100
60
10
6, m
9.0
L1
Final Th11-S1, adult
385
200
29
5
7, f
4.9
L1
Final Th8-S1, pediatric
200
100
73
24
13
8, m
10.0
-
DGRS Th5-S1, adult
270
200
50
16
35
Postoperative skin problems/skin plasty
31 7 11
Screw malposition/replacement
1
24
9, m
2.0
L3
Final Th10-S1-iliac, pediatric
185
50
96
10, m
3.3
L3-L4
Final Th7-L5-iliac, cervical
275
100
71
25
11, m
4.3
L2-L5
Final Th7-S1-iliac, cervical
310
180
94
12, f
10.0
-
Final Th12-S1-iliac, adult
240
650
32
20
13, f
26.0
-
Final L1-S1, adult
390
300
24
41
36
14, m
4.1
-
Final, Th11-S1-iliac, pediatric
215
150
9
21
9
15, f
6.5
-
Final Th7-S1-iliac, pediatric
200
100
51
29
10
16, m
5.8
-
DGRS Th3-S1, pediatric
250
50
22
22
12
17, f
3.8
-
DGRS Th5-L5, adult
110
100
19
28
12
18, m
3.5
L1-L2
Final Th5-S1-iliac, cervical
210
150
71
16
19, f
3.1
L2
Final Th8-L5, pediatric
150
100
52
19
20, m
3.0
L3-L4
DGRS; final Th9-S1, adult
200
100
87
10
10 16
13 Superficial wound infection
23
ORIGINAL ARTICLE
f, female; m, male; C, cervical; Th, thoracic; L, lumbar; S, sacral; DGRS, dual growing rod system; pediatric, pediatric system (3.5e4.5 screw diameter, 3.5 rod diameter); adult, adult system (4.5e6.5 screw diameter, 5.5 rod diameter); cervical, cervical system (2.5 screw diameter, 2.5 rod diameter).
SURGICAL MANAGEMENT OF MYELOMENINGOCELE-RELATED SPINAL DEFORMITIES
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1, f
SERGEY O. RYABYKH ET AL.
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ORIGINAL ARTICLE SERGEY O. RYABYKH ET AL.
SURGICAL MANAGEMENT OF MYELOMENINGOCELE-RELATED SPINAL DEFORMITIES
Figure 4. (A and B) Scheme of the kyphectomy zone. (CeE) Pedicle screw placement in a patient with spina
The dual growing rod technique is safe and effective in cases of moderate neuromuscular spinal deformities in early age.30,31 We implanted dual growing rods in 6 patients, including 4 with moderate kyphoscoliosis (from 3 to 73 Cobb), 1 with moderate lumbar scoliosis (60 lordosis and 77 scoliosis Cobb) and 1 with severe lumbar kyphosis (107 Cobb) with skin ulceration. One patient with kyphosis 83 (Cobb) was treated by the dual growing rod technique owing to low weight and thinning of the skin,
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bifida (C and D), compared with healthy vertebrae (E).
which preclude the performance of kyphectomy. After 2 years of distraction, vertebrotomy was performed. Instrumented Spondylodesis with or without Osteotomy in Rigid MMC-Related Kyphoscoliosis and Kyphosis In 1968, Sharrard41 described kyphectomy for MMC-related kyphosis. Various types of instrumented fixation for achieving spondylodesis in kyphectomy have been described, including anterior plating, posterior
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Figure 5. (A) Skin problems arising early after kyphectomy in an 8-year-old girl with rigid sharp-angled myelomeningocele (MMC) -related kyphosis. A 6.0 4.0 cm area of necrosis required skin plasty, performed on day 12 after kyphectomy. (B) Impermissible screw malposition necessitated reoperation in a 9-year-old
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boy with moderate (MMC)-related kyphoscoliosis, treated by final fusion with kyphectomy. (C) Anteroposterior X-ray showing a right rod fracture in an 11-year-old girl at 3 years after kyphectomy. (D) Lateral X-ray showing a fractured iliac screw in a 6-year-old boy at 2 years after final fusion.
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SURGICAL MANAGEMENT OF MYELOMENINGOCELE-RELATED SPINAL DEFORMITIES
Table 3. Remote Results of Treatment in 20 Patients with MMC-Related Spinal Deformity Follow-Up (Months)
mJOA Improvement
FIM Improvement
VAS Improvement
Long-Term Complications
1, f
15
2
15
0
6 months; SUUS
Skin plasty
2, m
12
0
0
2
6 months; STUS
Skin plasty, removal of right part of the screw-rod construction
3, f
45
2
18
0
3 years; rod fracture
4, m
16
0
0
0
6 months; STUS
5, m
34
0
11
0
3 years; STUS
6, m
60
0
2
2
7, f
14
0
5
0
8, m
31
0
2
0
9, m
6
0
5
0
10, m
55
1
8
0
3 years; STUS
11, m
54
0
9
0
12 months; SUUS
12, f
14
0
3
2
13, f
67
1
2
4
14, m
58
1
6
0
15, f
29
1
3
2
16, m
25
1
7
0
17, f
31
1
7
0
18, m
54
0
16
0
19, f
12
0
6
0
20, m
58
1
7
0
Patient, Sex
Reoperations
Rod replacement
6 months; STUS
Iliac screw replacement Wound revision, in-and-out draining
2 years; iliac screw fracture
Iliac screw replacement
2 years; iliac screw resorbtion, STUS
Iliac screw replasement
MMC, myelomeningocele; mJOA, modified Japanese Orthopedic Association Scale; FIM, Functional Independence Measure scale; VAS, visual analog scale; f, female; m, male; STUS, skin thinning under screw head; SUUS, skin ulceration under screw head.
plating, Harrington rods, Dwyer instrumentation, Luque rods, cables, hooks, wiring, Gardner bottle screws, Hartshill rectangle, pedicle screws, transcorporeal screws, Isola rods, and CotreleDubousset instrumentation.13,14,24,33,36,37,42-44 Kyphectomy is the surgical technique of choice in cases of rigid MMC-related kyphosis, but is associated with numerous complications, including skin and wound infection, nonfusion, and others.6,7,13,16,26,33,34 The use of additional posterior fixation promotes greater correction of the kyphosis and higher frequency of spondylodesis,7,13,15,19,20,24,34,45,46 but also leads to an additional skin tension.6,7,13,16,33 A number of authors have recommended anterior instrumented fixation to prevent skin problems.6,19,29,47,48 This option is convenient in cases of severe paralytic lordoscoliosis requiring anterior release, but generally is a highly invasive procedure. Moreover, anterior surgery for rigid MMC-related kyphosis, moderate lordoscoliosis, or scoliosis does not provide access to pelvic fixation and is less reliable biomechanically.3,6,7,9,11,12,14,20,33 Four of the 11 patients who underwent kyphectomy with instrumented fixation developed a skin problem under a crew head at 3e6 months after surgery. This required an additional intervention, either removing or replacing the screws. All such
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problems occurred over heads of distal screws (iliac, lumbar, or sacral), likely due to a greater load on a lumbosacral junction, soft tissue scarring, and impaired innervation. Surgical Treatment of MMC-Related Lordoscoliosis Lordoscoliosis most commonly develops in children with cerebral palsy; however, lumbar hyperlordosis is also associated with MMC.23 Surgical treatment is indicated when lumbar hyperlordosis causes sitting limitations, low back pain, and eating disorders due to high abdominal pressure.23 All of our patients with lumbar hyperlordosis had lumbosacral spina bifida, as well as thoracolumbar myelodysplasia or dysplasia of the conus medullaris and cauda equina (Table 1). At a spinal deformity up to 70 (Cobb), we did not perform vertebrectomy/ release in our patients with lumbar hyperlordosis. Three of these patients underwent local instrumented spondylodesis, and 1 had dual growing rod insertion. One patient experienced a complication (iliac screw fracture) 3 years after the procedure. Limitations and Further Prospects This study has several limitations. We used the FIM score to evaluate the results of surgical treatment, but FIM improvement is
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expected in growing children. Moreover, larger series are needed to draw more comprehensive conclusions regarding the safety and efficacy of the surgical techniques. CONCLUSIONS Kyphoscoliosis is a common finding in patients with thoracolumbar myelodysplasia, whereas lordoscoliosis is more common in patients with cauda equina and conus medullaris dysplasia. Bracing in patients with MMC and spinal deformities is
REFERENCES 1. Biedermann R. Orthopedic management of spina bifida. Orthopade. 2014;43:603-610 [in German]. 2. Ahmad AA. Treatment of spinal deformity associated with myelomeningocele in young children with the use of the four-rib construct. J Pediatr Orthop B. 2013;22:595-601. 3. Altiok H, Finlayson C, Hassani S, Sturm P. Kyphectomy in children with myelomeningocele. Clin Orthop Relat Res. 2011;469:1272-1278. 4. Bas CE, Preminger J, Olgun ZD, Demirkiran G, Sponseller P, Yazici M. Safety and efficacy of apical resection following growth-friendly instrumentation in myelomeningocele patients with gibbus: growing rod versus Luque trolley. J Pediatr Orthop. 2015;35:e98-e103. 5. Cho FN, Kan YY, Yang TL, Huang HP, Hsu PH, Tai MH. Prenatal diagnosis of sharp-angled lumbosacral kyphosis with myelomeningocele and spina bifida in a fetus. J Ultrasound Med. 2004; 23:1115-1119. 6. Comstock SA, Cook PC, Leahey JL, El-Hawary R, Hyndman JC. Posterior kyphectomy for myelomeningocele with anterior placement of fixation: a retrospective review. Clin Orthop Relat Res. 2011; 469:1265-1271. 7. de Amoreira Gepp R, Quiroga MR, Gomes CR, de Araújo HJ. Kyphectomy in meningomyelocele children: surgical technique, risk analysis, and improvement of kyphosis. Childs Nerv Syst. 2013;29: 1137-1141. 8. Duddy JC, Caird J, Connolly P. Repair of a large thoracolumbar myelomeningocele with associated lumbar kyphosis. Acta Neurochir (Wien). 2013;155: 1965-1968. 9. Dunn RN, Bomela LN. Kyphectomy in children with severe myelomeningocele-related kyphosis. Spine Deform. 2016;4:230-236. 10. Ganjeifar B, Zabihyan S, Baharvahdat H, Baradaran A. Five-level posterior total en bloc spondylectomy of severe myelomeningocele kyphosis. World Neurosurg. 2016;90:705.e1-705.e3. 11. Garg S, Oetgen M, Rathjen K, Richards BS. Kyphectomy improves sitting and skin problems in patients with myelomeningocele. Clin Orthop Relat Res. 2011;469:1279-1285. 12. Greggi T, Lolli F, Di Silvestre M, Martikos K, Vommaro F, Maredi E, et al. Surgical treatment of
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usually ineffective and can lead to rib deformation, respiratory volume reduction, and neuropathic skin ulcers. Early surgical correction improves body balance and quality of life and helps reduce the aggressiveness of surgical intervention. The dual growing rod technique is safe and effective in cases of moderate neuromuscular spinal deformities at an early age. Kyphectomy is a challenging procedure with high rates of complications, especially skin problems, but in patients with significant rigid kyphosis, there are no viable alternative procedures.
neuromuscular scoliosis: current techniques. Stud Health Technol Inform. 2012;176:315-318. 13. Hwang SW, Thomas JG, Blumberg TJ, Whitehead WE, Curry DJ, Dauser RC, et al. Kyphectomy in patients with myelomeningocele treated with pedicle screw-only constructs: case reports and review. J Neurosurg Pediatr. 2011;8: 63-70.
24. Martin J Jr, Kumar SJ, Guille JT, Ger D, Gibbs M. Congenital kyphosis in myelomeningocele: results following operative and nonoperative treatment. J Pediatr Orthop. 1994;14:323-328. 25. Yoshioka K, Watanabe K, Toyama Y, Chiba K, Matsumoto M. Kyphectomy for severe kyphosis with pyogenic spondylitis associated with myelomeningocele: a case report. Scoliosis. 2011;6:5.
14. Kaplan SÇ, Eks¸i MS¸, Bayri Y, Toktas¸ ZO, Konya D. Kyphectomy and pedicular screw fixation with posterior-only approach in pediatric patients with myelomeningocele. Pediatr Neurosurg. 2015;50:133-144.
26. Keessen W, van Ooy A, Pavlov P, Pruijs JE, Scheers MM, Slot G, et al. Treatment of spinal deformity in myelomeningocele: a retrospective study in four hospitals. Eur J Pediatr Surg. 1992; 2(Suppl 1):18-22.
15. Karlin LI. Kyphectomy for myelodysplasia. Neurosurg Clin N Am. 2007;18:357-364.
27. Olegovich RS, Mikhailovich SD, Nikolayevna TA. Surgical treatment of severe combined kyphosis secondary to myelocele: first homeland experience. Hir Pozvonoc. 2014;1:65-70 [in Russian].
16. Ko AL, Song K, Ellenbogen RG, Avellino AM. Retrospective review of multilevel spinal fusion combined with spinal cord transection for treatment of kyphoscoliosis in pediatric myelomeningocele patients. Spine (Phila Pa 1976). 2007;32: 2493-2501. 17. Kose KC, Inanmaz ME, Uslu M, Bal E, Caliskan I. Kyphectomy for congenital kyphosis due to meningomyelocele: a case treated with a modified approach to skin healing. Int Wound J. 2012;9: 311-315. 18. Portillo S. How I do it: surgical treatment of myelomeningocele. Surg Neurol Int. 2014;5(Suppl 1):S29-S38 [in Spanish]. 19. Samagh SP, Cheng I, Elzik M, Kondrashov DG, Rinsky LA. Kyphectomy in the treatment of patients with myelomeningocele. Spine J. 2011;11: e5-e11. 20. Schroeder JE, Barzilay Y, Hasharoni A, Kaplan L. Long-term outcome of surgical correction of congenital kyphosis in patients with myelomeningocele (MMC) with segmental spino-pelvic fixation. Evid Based Spine Care J. 2011;2:17-22. 21. Müller EB, Nordwall A. Prevalence of scoliosis in children with myelomeningocele in Western Sweden. Spine (Phila Pa 1976). 1992;17:1097-1102. 22. Trivedi J, Thomson JD, Slakey JB, Banta JV, Jones PW. Clinical and radiographic predictors of scoliosis in patients with myelomeningocele. J Bone Joint Surg Am. 2002;84-A:1389-1394. 23. Sato T, Yonezawa I, Onda S, Yoshikawa K, Takano H, Shimamura Y, et al. Surgical treatment for lumbar hyperlordosis after resection of a spinal lipoma associated with spina bifida: a case report. Medicine (Baltimore). 2017;96:e7895.
28. Smith JT, Novais E. Treatment of gibbus deformity associated with myelomeningocele in the young child with use of the vertical expandable prosthetic titanium rib (VEPTR): a case report. J Bone Joint Surg Am. 2010;92:2211-2215. 29. Osebold WR. Stability of myelomeningocele spines treated with the Mayfield two-stage anterior and posterior fusion technique. Spine (Phila Pa 1976). 2000;25:1344-1351. 30. Akbarnia BA, Marks DS, Boachie-Adjei O, Thompson AG, Asher MA. Dual growing rod technique for the treatment of progressive earlyonset scoliosis: a multicenter study. Spine (Phila Pa 1976). 2005;30(17 Suppl):S46-S57. 31. White KK, Song KM, Frost N, Daines BK. VEPTR growing rods for early-onset neuromuscular scoliosis: feasible and effective. Clin Orthop Relat Res. 2011;469:1335-1341. 32. Hill G, Nagaraja S, Akbarnia BA, Pawelek J, Sponseller P, Sturm P, et al. Retrieval and clinical analysis of distraction-based dual growing rod constructs for early-onset scoliosis. Spine J. 2017; 17:1506-1518. 33. Niall DM, Dowling FE, Fogarty EE, Moore DP, Goldberg C. Kyphectomy in children with myelomeningocele: a long-term outcome study. J Pediatr Orthop. 2004;24:37-44. 34. Odent T, Arlet V, Ouellet J, Bitan F. Kyphectomy in myelomeningocele with a modified DunnMcCarthy technique followed by an anterior inlayed strut graft. Eur Spine J. 2004;13:206-212. 35. Benzel EC, Lancon J, Kesterson L, Hadden T. Cervical laminectomy and dentate ligament
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section for cervical spondylotic myelopathy. J Spinal Disord. 1991;4:286-295. 36. Heydemann JS, Gillespie R. Management of myelomeningocele kyphosis in the older child by kyphectomy and segmental spinal instrumentation. Spine (Phila Pa 1976). 1987;12:37-41. 37. Lowe GP, Menelaus MB. The surgical management of kyphosis in older children with myelomeningocele. J Bone Joint Surg Br. 1978;60:40-45. 38. Eckstein HB, Vora RM. Spinal osteotomy for severe kyphosis in children with myelomeningocele. J Bone Joint Surg Br. 1972;54:328-333. 39. Fürderer S, Eysel P, Hopf C, Heine J. Sagittal static imbalance in myelomeningocele patients: improvement in sitting ability by partial and total gibbus resection. Eur Spine J. 1999;8:451-457. 40. Dimeglio A. Growth of the spine before age 5 years. J Pediatr Orthop B. 1992;1:102-107. 41. Sharrard WJ. Spinal osteotomy for congenital kyphosis in myelomeningocele. J Bone Joint Surg Br. 1968;50:466-471.
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SURGICAL MANAGEMENT OF MYELOMENINGOCELE-RELATED SPINAL DEFORMITIES
42. Banta JV. Combined anterior and posterior fusion for spinal deformity in myelomeningocele. Spine (Phila Pa 1976). 1990;15:946-952. 43. Fürderer S, Hopf C, Schwarz M, Voth D. Orthopedic and neurosurgical treatment of severe kyphosis in myelomeningocele. Neurosurg Rev. 1999;22:45-49. 44. McCall RE. Modified Luque instrumentation after myelomeningocele kyphectomy. Spine (Phila Pa 1976). 1998;23:1406-1411. 45. Huang TJ, Lubicky JP. Kyphectomy and segmental spinal instrumentation in young children with myelomeningocele kyphosis. J Formos Med Assoc. 1994;93:503-508. 46. Savini R, Cervellati S, Bettini N, Palmisani M, Bianco T. Surgical treatment of vertebral deformity due to myelomeningocele. Ital J Orthop Traumatol. 1991;17:55-63. 47. Sponseller PD, Young AT, Sarwark JF, Lim R. Anterior only fusion for scoliosis in patients with
myelomeningocele. Clin Orthop Relat Res. 1999: 117-124. 48. Hopf CG, Eysel P, Dubousset J. Operative treatment of scoliosis with Cotrel-Dubousset-Hopf instrumentation. New anterior spinal device. Spine (Phila Pa 1976). 1997;22:618-627 [discussion: 627-628].
Conflict of interest statement: The authors declare that the article content was composed in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. Received 25 September 2017; accepted 11 January 2018 Citation: World Neurosurg. (2018). https://doi.org/10.1016/j.wneu.2018.01.058 Journal homepage: www.WORLDNEUROSURGERY.org Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2018 Elsevier Inc. All rights reserved.
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Surgical Management of Myelomeningocele-Related Spinal Deformities Sergey O. Ryabykh, Olga M. Pavlova, Dmitry M. Savin, Alexander V. Burtsev, Alexander V. Gubin
OBJECTIVE: To evaluate the optimal timing and type of surgical treatment of myelomeningocele (MMC)-related spinal deformities and long-term follow-up of surgical treatment.
-
METHODS: We reviewed and presented clinical pictures, treatment strategies and results of 20 patients with MMC-related spinal deformities treated at our center between 2010 and 2017.
problems, but there are no alternative procedures for cases of heavy rigid kyphosis.
-
RESULTS: The average patient age was 6.3 years. The average preoperative neurologic status according to a modified Japan Orthopedic Association (mJOA) scale was 7.3 points (Benzel’s modification). Average functional status was 41 points according to a functional independent measure scale (FIM). The average angle of kyphosis was 83.7 , that of scoliosis was 36.7 , and that of lordosis was 67 (Cobb angles). The average duration of surgery was 234 minutes, and the average total blood loss was 175 mL. The average angle of kyphosis correction was 61 , that of scoliosis correction was 25 , and that of lordosis correction was 25 (Cobb angles). The average duration of hospitalization was 16.6 days, and the average follow-up was 34.5 months. The total number of complications was 13. Reoperation was required in 9 cases. Neurologic status according to the mJOA scale improved by 0.6 point on average. Functional status according to the FIM increased by 6.6 points on average.
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CONCLUSIONS: Early surgical correction of MMCrelated spinal deformities improves body balance and quality of life. The dual growing rod technique is safe and effective in cases of moderate neuromuscular spinal deformities at an early age. Kyphectomy is a challenging procedure with high complication rates, especially skin
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INTRODUCTION
S
pinal deformities are common in patients with myelomeningocele (MMC).1-20 Scoliosis occurs in about 94% of thoracolumbar MMC21 and in about 52% overall.22 The frequency of kyphosis is 10%e20%,8,9,20 and that of lumbar hyperlordosis is 1.5%.23 Progressive spinal deformity is a significant comorbidity associated with MMC. It leads to loss of truncal height and causes sitting, breathing, eating, and urination difficulties due to increased chest and abdominal pressures.3-8,10,13,24-27 These deformities are caused by neuromuscular disturbances and vertebral abnormalities.1,4,7,9,12,26 Spinal deformities associated with MMC can be divided into 2 major groups: predominantly neuromuscular kyphoscoliosis or lordoscoliosis2,12,13,20,28,29 and severe rigid kyphosis or sharp-angled kyphosis.3-10,13-15,19,25 The dual growing rod technique is safe and effective in cases of moderate neuromuscular kyphoscoliosis or lordoscoliosis at an early age, but associated with large number of rod fractures.30-32 Kyphectomy is the surgery of choice in cases of rigid MMC-related kyphosis but is associated with numerous complications, including skin and wound infection, nonfusion, and others.6,7,13,16,26,33,34 Here we present our experience with 20 consecutive patients with different types of MMC-related spinal deformities. This experience focuses on early deformity correction using the growing dual rod technique and instrumented fusion with or without vertebrectomy. To date, only a few studies have described successful treatment of MMC-related spinal deformities and longterm outcomes. We explored the following questions: Is early surgical correction of MMC-related spinal deformities safe and effective? Can it reduce skin breakdown, instability, and
Key words Myelomeningocele-related spinal deformity - Post-myelomeningocele syndrome - Sharp-angled kyphosis
Russian Ilizarov Scientific Center for Restorative Traumatology and Orthopaedics, Neurosurgical Department, Kurgan, Russia
Abbreviations and Acronyms FIM: Functional Independent Measure mJOA: Modified Japanese Orthopedic Association MMC: Myelomeningocele MRI: Magnetic resonance imaging VAS: Visual analog scale
Citation: World Neurosurg. (2018). https://doi.org/10.1016/j.wneu.2018.01.058
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To whom correspondence should be addressed: Olga M. Pavlova, M.D. [E-mail: [email protected]]
Journal homepage: www.WORLDNEUROSURGERY.org Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2018 Elsevier Inc. All rights reserved.
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paradoxical pain without a major loss of functional abilities? Is it effective in the long-term follow-up? Can early surgical correction of MMC-related spinal deformities improve body balance and quality of life? METHODS We evaluated a group of patients with MMC-related spinal deformity treated at our center between 2010 and 2017. Inclusion criteria and indications for surgery were progressive spinal deformity >20 (Cobb angle) in any plane, accompanied by spinal imbalance, low Functional Independent Measure (FIM) score, restricted verticalization, presence of pressure sores, signs of spinal instability, neurologic deterioration, including urodynamic studies (post-void residual volume, urine culture, uroflowmetry), and persistent back pain. We assessed neurologic symptoms using the modified Japanese Orthopedic Association (mJOA) scale (Benzel’s modification),35 the FIM scale, and a visual analog scale (VAS) for pain. All patients underwent spinal and craniovertebral X-ray, computed tomography scan, and magnetic resonance imaging (MRI) preoperatively; spinal computed tomography scan and X-ray at 48 hours postoperatively; and spinal X-ray at 6 months, 1 year, and 2 years after surgery. All operations were performed by 2 surgeons, S.O.R. and/or D.M.S. A literature review was performed in English and Russian in PubMed, Google Scholar, and eLibrary databases. Search queries included myelomeningocele-related spinal deformity, sharpangled kyphosis, myelomeningocele kyphosis, kyphectomy, and myelomeningocele spinal deformity. RESULTS Preoperative Clinical and Radiologic Study Data The 20 patients included 8 females (40%) and 12 males (60%), with an average age of 6.3 years (median, 4.3 years; range, 2e26 years). There were 12 toddlers, 7 grade schoolers, and 1 adult (age 26 years) (Table 1). All patients had undergone previous MMC repair. All patients had normal upper extremity muscle strength and sensation (mJOA score 6) and lower extremity paraparesis with micturition disturbance. The average preoperative neurologic status according to the mJOA was 7.3 points (median, 7.0; range, 6e11). The average FIM score was 41 points (median, 34.5; range, 18e108). Five patients (25%) had intermittent focal back pain (VAS score 2e4), and 4 (20%) had psychomotor retardation. All patients experienced chronic recurrent urinary tract infections, 2e3 times per year. Two patients had cardiac pathology, one with moderate tachycardia with mitral valve prolapse and the other with bradycardia due to severe sinus node dysfunction, requiring a pacemaker. No patients had significant respiratory issues. Eleven patients (55%) had kyphoscoliosis (Figure 1A), 4 (20%) had lordoscoliosis (Figure 1B), 4 (20%) had lumbar kyphosis only (Figure 1C), and 1 (5%) had a “flat” lower spine (Figure 1D). Rigid sharp-angled lumbar kyphosis (>90 Cobb angle) was present in 6 patients (30%), and skin ulcerations was present in 3 of these patients (Figure 2). Moderate lumbar or thoracolumbar kyphotic deformity (30e90 Cobb) was present in 9 patients (45%).
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The average angle of kyphosis was 83.7 Cobb (median, 75 ; range, 45 e134 ), that of scoliosis was 36.7 Cobb (median, 36 ; range, 11 e77 ), and that of lordosis was 67 Cobb (median, 71 ; range, 43 e84 ). Brain MRI demonstrated Chiari malformation and cerebral atrophy in 19 patients (95%). Cervicothoracic MRI revealed syringomyelia in 7 patients (35%). Thoracolumbar MRI unveiled marked hypoplasia of the thoracolumbar spinal cord in 11 patients (55%), of the lumbar spinal cord in 4 patients (20%), and of the conus medullaris/cauda equina in 5 patients (25%) (Table 1). Spina bifida was present in all patients, and 12 patients had accompanying vertebral anomalies (60%). Seven patients (35%) had rib anomalies. A ventriculoperitoneal shunt at an early age was performed in 12 patients (60%), and an endoscopic third ventriculostomy was performed in 1 patient (5%). Chiari decompression surgery was performed in 4 patients (20%). External spinal distraction was performed in 2 patients with sharp-angled kyphosis with apical ulcer, with 35 days of external distraction in one patient (Figure 3) and 25 days of external distraction in the other. One patient had undergone surgery for deformity, including kyphectomy with additional laminar-hook fixation, in another hospital 5 years before presentation to our center. Surgery All 20 patients underwent surgery, which included instrumented fusion by pedicle screw-rod constructs (Table 2). Pedicle screw placement was performed by a freehand technique, standard in “normal” vertebrae and modified in spina bifida vertebrae (Figure 4). Dual growing rod constructs were implanted in 6 patients (30%) with mobile kyphoscoliosis and lordoscoliosis. In 1 patient with local rigid kyphosis, growing rod implantation and bone-diskbone osteotomy was performed at 2 levels (L3-4 and L4-5) simultaneously. Another patient underwent 2-stage surgery with dual rib-iliacebased hook growing rod construct implantation at age 3 years with staged distractions over a 2-year period, followed by kyphectomy with pedicle screw fixation at age 5 years. Kyphectomy with pedicle screw fixation was performed in 11 patients with rigid kyphosis (55%; Figure 4). One of these patients underwent additional bone-disk-bone osteotomies at the 2 lower levels (L1-2 and L3-4) to reduce pressure on the scar tissue of the back. Spondylodesis with local pedicle screw fixation without osteotomy was performed in 4 patients (20%), including moderate local lordoscoliosis in 3 and moderate local kyphoscoliosis in 1. The atretic thecal sac was preserved in all patients to prevent disruption of cerebrospinal fluid dynamics and shunt malfunction. Screw malposition was seen in 6 patients (15 screws total), including 5 (12 screws) with permissible malposition and 1 (3 screws) with inadmissible malposition, necessitating reoperation. The average duration of surgery was 234 minutes (median, 225 minutes; range, 110e390 minutes). The average total blood loss was 175 mL (median, 100 mL; range, 20e650 mL). Postoperative Clinical and Radiologic Study Data The average angle of kyphosis correction was 57 Cobb (median, 56 ; range, 19 e96 ), that of scoliosis correction was 25 Cobb
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Patient, Sex
Level of Cord Hydrocephalus Dysplasia Syringomyelia ArnoldeChiari II
Other Vertebral Abnormalities
1, f
C5, Th1-3, T7-S5
Block laminae C6-7
VPS
LE
Th
C3
2, m
Th3-S5
BV Th3, HV Th6, Th8, Th11, block Th9-10
-
ThL
-
C3 (PFD)
3, f
Th12-S5
-
VPS
LE
Th
C3
4, m
Th8-S5
-
VPS
ThL
-
C2 (PFD)
5, m
Th5-S5
HV Th8, BV L1-2
VPS
ThL
-
C3 (PFD)
RUI, PMR
Comorbidities
Lumbar/Thoracolumbar Lumbar Lordosis Scoliosis Kyphosis (Cobb Angle) (Cobb Angle) (Cobb Angle)
RUI, PMR
49
-
21
Mitral valve prolapse, RUI, PMR
60
-
70
RUI, PMR
130
-
35
Epilepsy, sinus node dysfunction required pacemaker, RUI, PMR
73
-
49
75
-
10
Th6-S5
Block L1-2
-
ThL
-
C2
RUI, PMR
61
-
28
7, f
Th8-S5
Block laminae Th1-4, HV Th10, block Th11-L1
ETVS
ThL
-
C3
RUI, PMR
101
-
28
8, m
L2-S4
-
-
CM CE
-
C2
RUI, PMR
-
60
77
9, m
Th10-S4
-
VPS
ThL
-
C3
RUI, PMR
121
-
11
10, m
Th6-S5
-
VPS
ThL
-
C3
RUI
83
-
-
11, m
Th7-S5
-
VPS
ThL
C
C3
RUI, PMR
134
-
24
12, f
L2-S5
-
VPS
CM CE
-
C1
RUI
-
82
38
13, f
L3-S5
BV L5
-
CM CE
-
-
RUI
-
84
53
14, m
Th12-S5
BV L2-3, diastema L3
-
CE
-
C2
RUI
-
43
37
15, f
Th12-S5
BV Th11, HVTh12 dex, block Th3-5, diastema S1-2, sacral hypoplasia
VPS
LE
CTh
C2
RUI, PMR
45
-
45
16, m
Th10-S5
-
VPS
LE
-
C2
RUI, PMR
47
-
22
17, f
L5-S5
-
-
CM CE
Th
C2 (PFD)
RUI, PMR
3
-
40
18, m
Th8-S5
Block Th5-6
-
ThL
Th
C2
RUI, PMR
98
-
-
19, f
Th10-S5
-
VPS
ThL
-
C3
RUI, PMR
71
-
-
20, m
Th5-S5
Block Th5-8
VPS
ThL
Th
C6
RUI, PMR
107
-
-
f, female; m, male; C, cervical; Th, thoracic; L, lumbar; S, sacral; BV, butterfly vertebrae; HV, hemivertebrae; VPS, ventriculoperitoneal shunt; LE, lumbar enlargement; CM, conus medullaris; CE, cauda equina; PFD, posterior fossa decompression; RUI, recurrent urinal infection; PMR, psychomotor retardation; dex, dexter.
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6, m
SURGICAL MANAGEMENT OF MYELOMENINGOCELE-RELATED SPINAL DEFORMITIES
Spina Bifida
SERGEY O. RYABYKH ET AL.
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Table 1. Clinical and Radiologic Features of 20 Patients with MMC-related Spinal Deformity
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SURGICAL MANAGEMENT OF MYELOMENINGOCELE-RELATED SPINAL DEFORMITIES
Figure 1. Lateral and anteroposterior X-rays of 4 patients with different types of myelomeningocele-related spinal deformity: (A)
(median, 23 ; range, 1 e62 ), and that of lordosis correction was 25 Cobb (median, 28 ; range, 9 e35 ) (Table 2). The postoperative period was complicated by marginal wound skin necrosis requiring a cutaneous plastic by a graft in 1 patient (Figure 5A). An impermissible screw malposition necessitated reoperation in 1 patient (Figure 5B), and a superficial wound healing problem was seen in 1 patient. The average length of hospital stay was 16.6 days (median, 13 days; range, 7e36 days). Implant-associated long-term complications were present in 10 patients (50%) (Table 3). Eight patients had skin problems above screw heads (40%), 5 of whom required surgery (25%). One
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kyphoscoliosis; (B) lordoscoliosis; (C) severe lumbar kyphosis; and (D) scoliosis and “flat” back.
patient experienced a rod fracture at 3 years after surgery (Figure 5C), and 1 patient had an iliac screw fracture at 2 years after surgery (Figure 5D). All 6 patients with growing rod systems were followed closely, with staged distraction performed every 6 months. The average follow-up was 34.5 months (median, 31 months; range, 6e67 months). Neurologic status according to the mJOA improved by an average of 0.6 point (median, 0; range, 0e2). Average functional status improved by 6.6 points on the FIM score (median, 6; range, 0e18). All patients were free of back pain at follow-up (0 points on the VAS).
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ORIGINAL ARTICLE SERGEY O. RYABYKH ET AL.
SURGICAL MANAGEMENT OF MYELOMENINGOCELE-RELATED SPINAL DEFORMITIES
Figure 2. Primary local back view of an 8-year-old girl with rigid sharp-angled myelomeningocele-related kyphosis with skin ulceration.
DISCUSSION Surgical treatment of MMC-related spinal deformities is often required for correction of deformity and establishment of truncal balance, reduction of degenerative spinal changes that can trigger pain in older age, expansion of respiratory capacity and abdominal space, and minimization of skin ulcers over a gibbus.14,36-39 Predictors of Various Types of MMC-Related Spinal Deformities A high incidence of spinal deformity is associated with thoracolumbar MMC21; scoliosis occurs in approximately 94% of these patients, compared with approximately 52% overall.22 Almost all of our patients with kyphoscoliosis had thoracolumbar MMC with thoracic spinal cord and/or lumbar enlargement hypoplasia. Lordoscoliosis was associated with lumbosacral MMC and conus
Figure 3. Local status and X-ray of an 8-year-old girl with rigid sharp-angled myelomeningocele-related kyphosis after implantation of external
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medullaris and/or cauda equina dysplasia. Severe rigid sharpangled kyphosis was associated with a greater frequency of a syringomyelia and additional spinal congenital malformations (Table 1). Dual Growing Rod Technique in Mobile MMC-Related Kyphoscoliosis and Lordoscoliosis The peak of spinal growth is during the first 5 years of life (2 cm/ year). The growth rate subsequently diminishes over the next 5e10 years before a second growth spurt is observed.40 Traditional casting and orthotic treatment are difficult and sometimes ineffective in young children, possibly causing immature rib cage deformities, pulmonary problems, and skin problems (i.e., pressure sores).
distraction device. (A) Local view of applied external distraction device. (B and C) Anteroposterior and lateral X-rays on day 34 after surgery.
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Patient, Sex
Age (Years)
Kyphectomy
Level and Type of Instrumental Fusion
Duration of Surgery (minutes)
Blood Loss (mL)
Lumbar Kyphosis Correction (Cobb Angle)
Lumbar Hyperlordosis Correction (Cobb Angle)
Scoliosis Correction (Cobb Angle)
Short-Term Complications
Days of Postoperative Treatment
4.2
-
DGRS Th5-S2 adult
155
20
41
15
11
2, m
6.8
Th10-12
Final Th2-S1, pediatric
260
250
35
62
23
3, f
8.0
L3-L4
Final,Th3-S1-iliac, pediatric
240
500
70
25
4, m
3.7
-
DGRS Th5-L5, pediatric
190
100
37
38
5, m
3.1
L1-L3
Final Th6-S1, pediatric
235
100
60
10
6, m
9.0
L1
Final Th11-S1, adult
385
200
29
5
7, f
4.9
L1
Final Th8-S1, pediatric
200
100
73
24
13
8, m
10.0
-
DGRS Th5-S1, adult
270
200
50
16
35
Postoperative skin problems/skin plasty
31 7 11
Screw malposition/replacement
1
24
9, m
2.0
L3
Final Th10-S1-iliac, pediatric
185
50
96
10, m
3.3
L3-L4
Final Th7-L5-iliac, cervical
275
100
71
25
11, m
4.3
L2-L5
Final Th7-S1-iliac, cervical
310
180
94
12, f
10.0
-
Final Th12-S1-iliac, adult
240
650
32
20
13, f
26.0
-
Final L1-S1, adult
390
300
24
41
36
14, m
4.1
-
Final, Th11-S1-iliac, pediatric
215
150
9
21
9
15, f
6.5
-
Final Th7-S1-iliac, pediatric
200
100
51
29
10
16, m
5.8
-
DGRS Th3-S1, pediatric
250
50
22
22
12
17, f
3.8
-
DGRS Th5-L5, adult
110
100
19
28
12
18, m
3.5
L1-L2
Final Th5-S1-iliac, cervical
210
150
71
16
19, f
3.1
L2
Final Th8-L5, pediatric
150
100
52
19
20, m
3.0
L3-L4
DGRS; final Th9-S1, adult
200
100
87
10
10 16
13 Superficial wound infection
23
ORIGINAL ARTICLE
f, female; m, male; C, cervical; Th, thoracic; L, lumbar; S, sacral; DGRS, dual growing rod system; pediatric, pediatric system (3.5e4.5 screw diameter, 3.5 rod diameter); adult, adult system (4.5e6.5 screw diameter, 5.5 rod diameter); cervical, cervical system (2.5 screw diameter, 2.5 rod diameter).
SURGICAL MANAGEMENT OF MYELOMENINGOCELE-RELATED SPINAL DEFORMITIES
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1, f
SERGEY O. RYABYKH ET AL.
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ORIGINAL ARTICLE SERGEY O. RYABYKH ET AL.
SURGICAL MANAGEMENT OF MYELOMENINGOCELE-RELATED SPINAL DEFORMITIES
Figure 4. (A and B) Scheme of the kyphectomy zone. (CeE) Pedicle screw placement in a patient with spina
The dual growing rod technique is safe and effective in cases of moderate neuromuscular spinal deformities in early age.30,31 We implanted dual growing rods in 6 patients, including 4 with moderate kyphoscoliosis (from 3 to 73 Cobb), 1 with moderate lumbar scoliosis (60 lordosis and 77 scoliosis Cobb) and 1 with severe lumbar kyphosis (107 Cobb) with skin ulceration. One patient with kyphosis 83 (Cobb) was treated by the dual growing rod technique owing to low weight and thinning of the skin,
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bifida (C and D), compared with healthy vertebrae (E).
which preclude the performance of kyphectomy. After 2 years of distraction, vertebrotomy was performed. Instrumented Spondylodesis with or without Osteotomy in Rigid MMC-Related Kyphoscoliosis and Kyphosis In 1968, Sharrard41 described kyphectomy for MMC-related kyphosis. Various types of instrumented fixation for achieving spondylodesis in kyphectomy have been described, including anterior plating, posterior
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SURGICAL MANAGEMENT OF MYELOMENINGOCELE-RELATED SPINAL DEFORMITIES
Figure 5. (A) Skin problems arising early after kyphectomy in an 8-year-old girl with rigid sharp-angled myelomeningocele (MMC) -related kyphosis. A 6.0 4.0 cm area of necrosis required skin plasty, performed on day 12 after kyphectomy. (B) Impermissible screw malposition necessitated reoperation in a 9-year-old
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boy with moderate (MMC)-related kyphoscoliosis, treated by final fusion with kyphectomy. (C) Anteroposterior X-ray showing a right rod fracture in an 11-year-old girl at 3 years after kyphectomy. (D) Lateral X-ray showing a fractured iliac screw in a 6-year-old boy at 2 years after final fusion.
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ORIGINAL ARTICLE SERGEY O. RYABYKH ET AL.
SURGICAL MANAGEMENT OF MYELOMENINGOCELE-RELATED SPINAL DEFORMITIES
Table 3. Remote Results of Treatment in 20 Patients with MMC-Related Spinal Deformity Follow-Up (Months)
mJOA Improvement
FIM Improvement
VAS Improvement
Long-Term Complications
1, f
15
2
15
0
6 months; SUUS
Skin plasty
2, m
12
0
0
2
6 months; STUS
Skin plasty, removal of right part of the screw-rod construction
3, f
45
2
18
0
3 years; rod fracture
4, m
16
0
0
0
6 months; STUS
5, m
34
0
11
0
3 years; STUS
6, m
60
0
2
2
7, f
14
0
5
0
8, m
31
0
2
0
9, m
6
0
5
0
10, m
55
1
8
0
3 years; STUS
11, m
54
0
9
0
12 months; SUUS
12, f
14
0
3
2
13, f
67
1
2
4
14, m
58
1
6
0
15, f
29
1
3
2
16, m
25
1
7
0
17, f
31
1
7
0
18, m
54
0
16
0
19, f
12
0
6
0
20, m
58
1
7
0
Patient, Sex
Reoperations
Rod replacement
6 months; STUS
Iliac screw replacement Wound revision, in-and-out draining
2 years; iliac screw fracture
Iliac screw replacement
2 years; iliac screw resorbtion, STUS
Iliac screw replasement
MMC, myelomeningocele; mJOA, modified Japanese Orthopedic Association Scale; FIM, Functional Independence Measure scale; VAS, visual analog scale; f, female; m, male; STUS, skin thinning under screw head; SUUS, skin ulceration under screw head.
plating, Harrington rods, Dwyer instrumentation, Luque rods, cables, hooks, wiring, Gardner bottle screws, Hartshill rectangle, pedicle screws, transcorporeal screws, Isola rods, and CotreleDubousset instrumentation.13,14,24,33,36,37,42-44 Kyphectomy is the surgical technique of choice in cases of rigid MMC-related kyphosis, but is associated with numerous complications, including skin and wound infection, nonfusion, and others.6,7,13,16,26,33,34 The use of additional posterior fixation promotes greater correction of the kyphosis and higher frequency of spondylodesis,7,13,15,19,20,24,34,45,46 but also leads to an additional skin tension.6,7,13,16,33 A number of authors have recommended anterior instrumented fixation to prevent skin problems.6,19,29,47,48 This option is convenient in cases of severe paralytic lordoscoliosis requiring anterior release, but generally is a highly invasive procedure. Moreover, anterior surgery for rigid MMC-related kyphosis, moderate lordoscoliosis, or scoliosis does not provide access to pelvic fixation and is less reliable biomechanically.3,6,7,9,11,12,14,20,33 Four of the 11 patients who underwent kyphectomy with instrumented fixation developed a skin problem under a crew head at 3e6 months after surgery. This required an additional intervention, either removing or replacing the screws. All such
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problems occurred over heads of distal screws (iliac, lumbar, or sacral), likely due to a greater load on a lumbosacral junction, soft tissue scarring, and impaired innervation. Surgical Treatment of MMC-Related Lordoscoliosis Lordoscoliosis most commonly develops in children with cerebral palsy; however, lumbar hyperlordosis is also associated with MMC.23 Surgical treatment is indicated when lumbar hyperlordosis causes sitting limitations, low back pain, and eating disorders due to high abdominal pressure.23 All of our patients with lumbar hyperlordosis had lumbosacral spina bifida, as well as thoracolumbar myelodysplasia or dysplasia of the conus medullaris and cauda equina (Table 1). At a spinal deformity up to 70 (Cobb), we did not perform vertebrectomy/ release in our patients with lumbar hyperlordosis. Three of these patients underwent local instrumented spondylodesis, and 1 had dual growing rod insertion. One patient experienced a complication (iliac screw fracture) 3 years after the procedure. Limitations and Further Prospects This study has several limitations. We used the FIM score to evaluate the results of surgical treatment, but FIM improvement is
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ORIGINAL ARTICLE SERGEY O. RYABYKH ET AL.
SURGICAL MANAGEMENT OF MYELOMENINGOCELE-RELATED SPINAL DEFORMITIES
expected in growing children. Moreover, larger series are needed to draw more comprehensive conclusions regarding the safety and efficacy of the surgical techniques. CONCLUSIONS Kyphoscoliosis is a common finding in patients with thoracolumbar myelodysplasia, whereas lordoscoliosis is more common in patients with cauda equina and conus medullaris dysplasia. Bracing in patients with MMC and spinal deformities is
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usually ineffective and can lead to rib deformation, respiratory volume reduction, and neuropathic skin ulcers. Early surgical correction improves body balance and quality of life and helps reduce the aggressiveness of surgical intervention. The dual growing rod technique is safe and effective in cases of moderate neuromuscular spinal deformities at an early age. Kyphectomy is a challenging procedure with high rates of complications, especially skin problems, but in patients with significant rigid kyphosis, there are no viable alternative procedures.
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42. Banta JV. Combined anterior and posterior fusion for spinal deformity in myelomeningocele. Spine (Phila Pa 1976). 1990;15:946-952. 43. Fürderer S, Hopf C, Schwarz M, Voth D. Orthopedic and neurosurgical treatment of severe kyphosis in myelomeningocele. Neurosurg Rev. 1999;22:45-49. 44. McCall RE. Modified Luque instrumentation after myelomeningocele kyphectomy. Spine (Phila Pa 1976). 1998;23:1406-1411. 45. Huang TJ, Lubicky JP. Kyphectomy and segmental spinal instrumentation in young children with myelomeningocele kyphosis. J Formos Med Assoc. 1994;93:503-508. 46. Savini R, Cervellati S, Bettini N, Palmisani M, Bianco T. Surgical treatment of vertebral deformity due to myelomeningocele. Ital J Orthop Traumatol. 1991;17:55-63. 47. Sponseller PD, Young AT, Sarwark JF, Lim R. Anterior only fusion for scoliosis in patients with
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Conflict of interest statement: The authors declare that the article content was composed in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. Received 25 September 2017; accepted 11 January 2018 Citation: World Neurosurg. (2018). https://doi.org/10.1016/j.wneu.2018.01.058 Journal homepage: www.WORLDNEUROSURGERY.org Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2018 Elsevier Inc. All rights reserved.
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