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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
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Title: Factors Influencing the Evaluation and Management of Neuromuscular
Scoliosis: A Review of the Literature
Type of Article: Review article
Key words: Neuromuscular diseases, cerebral palsy, Duchenne muscular dystrophy,
scoliosis, surgery, bracing, orthoses.
Word Count: 3556 words
Ethical approval: None required.
Number of Figures: 10 (Fig. 1, Fig. 2a-d, Fig. 3a-d, Fig. 4)
Number of Tables: 2
Number of References: 83
Authors: 1. Simon B Roberts ECAT Fellow/Specialist Registrar Trauma and
Orthopaedics,
2. Athanasios I Tsirikos Consultant Orthopaedic and Spinal Surgeon
Authors’ Institutional Address/Affiliations: University of Edinburgh/Scottish
National Spinal Deformity Centre, Royal Hospital for Sick Children, Edinburgh, EH9
1LF, UK
First Author: S B Roberts MBChB, BSc, MSc, MRCS(Ed); postal address - 12 Littlejohn
Avenue, Edinburgh, EH10 5TG, UK; email - [email protected]; tel –
07832084911.
Corresponding author: Mr Athanasios I Tsirikos; postal address - Scottish National
Spinal Deformity Centre, Royal Hospital for Sick Children, Edinburgh, EH9 1LF, UK;
email – [email protected]; tel – 0131 536 0784; fax – 0131 536
0924.
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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
Abstract
Neuromuscular scoliosis (NMS) is the second most prevalent spinal deformity (after
idiopathic scoliosis) and is usually first identified during early childhood. Cerebral
palsy (CP) is the most common cause of NMS, followed by Duchenne muscular
dystrophy (DMD). Progressive spinal deformity causes difficulty with daily care,
walking and sitting, and can lead to back and rib pain, cardiac and pulmonary
complications, altered seizure thresholds, and skin compromise. Early referral to
specialist spinal services and early diagnosis of NMS is essential to ensure appropriate
multidisciplinary patient management. The most important goals for patients are
preservation of function, facilitation of daily care, and alleviation of pain. Nonoperative management includes observation or bracing for less severe and flexible
deformity in young patients as a temporising measure to provide postural support.
Surgical correction and stabilisation of NMS is considered for patients with a deformity
>40-50o, but may be performed for less severe deformity in patients with DMD. Postoperative intensive care, and early mobilisation and nutritional supplementation aim to
minimise the rate of post-surgical complications, which are relatively common in this
patient group. However, surgical management of NMS is associated with good longterm outcomes and high satisfaction rates for patients, their relatives and carers.
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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
1. Introduction
Scoliosis is defined as a coronal plane spinal curvature of at least 10 degrees. A scoliotic
deformity arising in the clinical setting of muscle imbalance due to an underlying
neuropathic or myopathic disease can be diagnosed as neuromuscular scoliosis.
Neuromuscular scoliosis is the second most prevalent spinal deformity, after idiopathic
scoliosis. Patients with neuromuscular scoliosis frequently also have cardiac,
gastrointestinal, and respiratory compromise in addition to other musculoskeletal and
neurological disorders. Management of the scoliosis is therefore complex and requires
careful multidisciplinary care. This review will focus on the diagnosis, management,
and outcomes following the treatment of scoliosis in patients with neuromuscular
conditions.
2. Epidemiology and Aetiology
The incidence of scoliosis in the most common neuromuscular conditions is shown in
Table 1. Cerebral palsy is the most prevalent cause of neuromuscular scoliosis,
followed by Duchenne muscular dystrophy (DMD). Children with neuromuscular
conditions develop spinal deformities due to a combination of poor muscular control
across the trunk and pelvis, inherent muscle weakness, and occasionally spasticity. The
abnormal curvature develops as a consequence of the effect of gravity on the vertebral
column, which is inadequately supported due to generalised weakness of the posterior
spinal and abdominal muscles during a period of accelerated skeletal growth. The
degree of spinal deformity in these patients is therefore directly related to the degree of
neuromuscular involvement and the child’s ambulatory ability. Wheelchair-bound
growing patients with any underlying aetiology are much more likely to develop
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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
scoliosis that demonstrates a high risk of rapid progression, and which often also affects
the pelvis, than children who still retain walking capacity [1].
3. Clinical Manifestations and Presentation
3.1. Types of Deformity
Neuromuscular scoliotic deformities are generally first identified during early
childhood. Initially, a flexible postural curve develops which is correctable with the
patient lying flat or on suspension. This often progresses into a torsional rigid deformity
during late child life and the pubertal growth and finally into a stiff curve of larger
magnitude before growth is complete [1]. The typical curvature of neuromuscular
scoliosis is a long, collapsing C-shaped curve affecting the thoracic and lumbar spine
(Figure 1) [2]. The deformity often extends to include the pelvis, producing pelvic
obliquity with the elevated one-half of the pelvis corresponding to the concave side
(inside) of the curve. Asymmetric hip positioning due to hip contractures, subluxation
or dislocation, as well as unbalanced function of the trunk and pelvic muscles may also
cause pelvic deformity and accentuate the underlying scoliosis. If a severe scoliosis and
pelvic obliquity co-exist with associated hip subluxation/dislocation, it is generally
preferable to treat the spino-pelvic deformity first in order to produce a horizontal pelvis
before surgical treatment to relocate the displaced femoral head within the dysplastic
hip joint is planned. Finally, a kyphotic deformity with collapse of the trunk often in
conjunction to the scoliosis is also common in neuromuscular conditions [3]. In
contrast, isolated lumbar hyperlordosis or a severe lordoscoliosis is less frequent but
produces major trunk imbalance, sitting intolerance and persistent pain, which
necessitates surgical treatment [4].
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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
3.2. Clinical signs and symptoms
Deformity progression leads to significant trunk and spino-pelvic imbalance with
consequent loss of function and increasing difficulty with daily care. Walking and
sitting ability may become difficult, leading to pain or the patient becoming confined
to bed. Rotation and shortening of the trunk occurs and can lead to compression of the
heart and lungs, reduced mobility of the ribs and restriction of breathing capacity. If
tilting of the pelvis develops, mechanical back or rib impingement pain can result, as
well as cardiac or pulmonary complications. Feeding can also be affected and
exacerbation of pre-existing swallowing disorders or gastro-oesophageal reflux can
occur. Seizure activity may become worse due to uncontrolled pain. Asymmetrical
loading in the area of the buttocks due to pelvic obliquity can cause pressure sores,
especially in patients with impaired skin sensitivity (myelomeningocele, occasionally
cerebral palsy) [1]. Many patients are also unable to articulate their symptoms, which
can lead to a delay in presentation and diagnosis. Due to the universal progressive
nature of neuromuscular scoliosis, early diagnosis of deformity is essential to optimise
the multidisciplinary management. Early referral to specialist spinal centres of any
patient with a neuromuscular condition and suspected spinal deformity is
recommended.
4. Clinical Evaluation
Patients with a neuromuscular scoliosis are initially assessed by clinical review in order
to identify the severity of the spinal deformity followed by a systemic examination to
identify associated co-morbidities or medical problems related to the development of
the curve. Upper and lower limb function is assessed and evaluation of lower limb
deformities is performed as these may contribute to the development of spino-pelvic
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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
imbalance. Whole-spine postero-anterior and lateral radiographs are then performed.
Sitting radiographs may be obtained if the patient is unable to stand. Supine x-rays in
maximum traction can be useful to assess curve flexibility. Curve magnitude (as
measured by the Cobb method) [5], spinal balance, pelvic obliquity, curve flexibility
and curve progression are all closely monitored. An MRI scan of the whole spine is
required if there is suspicion of intraspinal pathology. A patient with established
neuromuscular scoliosis requires at least annual follow-up examination to assess curve
progression, but more frequent reviews, usually every 6 months, are required during
periods of rapid skeletal growth and for more severe or progressive curves.
5. Risk Factors for Disease Progression
In general, there is a proportional relationship between the severity of neurological or
myopathic involvement and curve severity, as well as the risk for deformity
progression. For example, quadriplegic cerebral palsy typically causes a more severe
and progressive scoliosis compared to hemiplegic or diplegic cerebral palsy. The
progression of scoliosis in patients with cerebral palsy treated with intrathecal baclofen
is not significantly different from cerebral palsy patients not receiving baclofen therapy
[6]. For patients with DMD, later age at loss of walking ability and greater duration of
corticosteroid therapy are related to later onset of scoliosis, whereas asymmetry of the
hips and forced vital capacity at age 11-12 years are directly correlated with subsequent
severity of scoliosis [7]. While the rate of curve progression is highly variable, the
average progression for children with cerebral palsy has been reported as 0.8 degrees
each year for curves that are less than 50 degrees and 1.4 degrees each year for curves
that are more than 50 degrees [8]. During periods of rapid growth such as the pubertal
growth spurt, much more rapid progression can occur, with curves increasing as much
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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
as 2-4 degrees each month, resulting in rapid deterioration of spinal deformity and the
patient’s function [9, 10].
6. Nonoperative Management
6.1. Aims of Treatment
Several factors need to be considered during the global planning of disease management
in patients with neuromuscular scoliosis. The most important goals for patients are
preservation of function, facilitation of daily care, and alleviation of pain. Nonoperative management of patients with neuromuscular spinal deformities should be
directed at maximising sitting ability and postural control in order to facilitate motor
function and interaction to the environment. Due to the lack of randomised controlled
trials investigating treatment modalities for spinal deformity in neuromuscular patients,
much of the management of neuromuscular scoliosis derives from results of cohort
studies, comparative cohort studies, anecdotal evidence and expert opinion. Growing
patients with mild, flexible curves can be managed by observation only. For patients
with DMD, corticosteroids may delay the development and progression of scoliosis and
may be considered for this purpose, though the optimal dosing regimen remains to be
determined [11-14].
6.2. Bracing
If progression of neuromuscular scoliosis occurs, bracing may be considered. It is
generally accepted that spinal bracing is ineffective in significantly altering progression
of deformity in patients with neuromuscular disease. Although bracing may not
ultimately prevent the need for surgical intervention, bi-valved or soft full-body contact
thoraco-lumbo-sacral braces can be used in patients in whom surgery is contra-
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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
indicated or declined, and in young children with small, flexible curves. In these
circumstances, bracing can help to manage the spinal deformity by providing postural
support, maintaining sitting position, improving muscle balance, and augmenting
function, while permitting further growth to occur prior to surgical intervention [1517]. Furthermore, bracing may limit complications such as impaired pulmonary
development, and therefore reduce the risks associated with any future surgery [3].
However, bracing is often ineffective due to shortening duration of brace wear, skin
irritation, and respiratory problems, as well as the aggressive nature of the deformity.
Cognitive impairment associated with cerebral palsy may also reduce a patient’s
tolerance of bracing. It is important that the potential advantages, disadvantages, and
expected outcomes of bracing are clearly understood by the patients, their family and
carers if it is used [9, 18].
6.3. Orthotics
Wheelchair adaptations can be used in addition to, or as an alternative to, bracing. These
adaptations include offset lateral chest supports, shoulder/waist harnesses, modular
seating systems, and moulded shells. The latter permit the patient to lie and be tilted,
and may be better tolerated than braces, are simple to use for carers, permit free access
to the abdomen if gastrostomy feeding is required, and do not restrict respiratory
movements, which is particularly important for children at high risk for chest infections
[15]. There is conflicting evidence regarding the role of knee-ankle-foot orthoses in
prolonging functional walking, and any association with subsequent progression of
scoliosis. Therefore knee-ankle-foot orthoses cannot be recommended for the purpose
of preventing development or progression of scoliosis in neuromuscular patients [19,
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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
20]. Braces and orthoses that protect all functions are not available, and therefore their
selection and use must be tailored to the needs of each individual patient.
6.4. Physiotherapy
Physiotherapy, including regular stretching, is widely performed and important in the
general care of neuromuscular patients, but does not improve any aetiological factors
for neuromuscular scoliosis or prevent progression of an established scoliosis.
Physiotherapy can be helpful to prevent any adverse effects from prolonged brace use,
prevent the onset of joint contractures, and maintain both chest mobility and respiratory
excursion [21].
6.5. Other Non-operative Treatments
There is no evidence to support electrical stimulation or botulinum toxin injections to
manage spinal deformity in patients with neuromuscular conditions. The use of lycra
suits may be beneficial in patients who retain some degree of muscle function and who
develop small and flexible postural curves as an alternative to a conventional brace due
to the fact that they are usually better tolerated and in order to stimulate upright posture
and preserve function [22].
7. Operative Management
7.1. Aims of Surgical Intervention
The decision to proceed with operative correction and stabilisation of neuromuscular
scoliosis is dependent on individual patient-specific factors, in particular the nature of
the underlying neuromuscular condition and the patient’s current level of function. The
patient’s individual needs, overall medical state and long-term prognosis of the
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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
underlying neuromuscular condition have to be taken into account and the decision for
scoliosis surgery has to be individualised and discussed thoroughly with the patients
and their families. For higher-functioning patients, operative intervention aims to
provide a more normal spinal balance and alter the progression of disease with the goal
of preserving function with respect to ambulatory capacity. In wheelchair-dependent
patients, the aim is to maintain sitting independence, promote more physiological
respiratory and gastrointestinal functioning, and facilitate overall care [23].
7.2. Indications for Surgery and Surgical Techniques
In general, surgical intervention is considered for patients with a curve greater than 40
or 50 degrees and significant deterioration in function (Figure 2). Such curves will
deteriorate even after the patients have completed their skeletal growth. An exception
to this is DMD scoliosis in which, due to the natural history of the disease, surgery is
considered for progressive spinal curvatures greater than 25 degrees in order to prevent
respiratory compromise produced by the deformity. Surgical treatment will aim to
correct the spinal deformity and pelvic obliquity, restore coronal and sagittal spinal
balance, and achieve a solid spinal fusion. This is usually performed using a posterior
surgical approach to the spine, with instrumentation to correct the curvatures, and
autologous and allograft bone to achieve a bony fusion [24-26]. This typically results
in up to 75-80% overall correction of deformity [27-29]. For patients with severe
curves, which may be permitted to develop in patients with cerebral palsy, the challenge
is to manage spinal growth while controlling the deformity. For curves of 60-90 degrees
magnitude, surgery is considered when the deformity becomes stiff on clinical and
radiographic examination. If the spine remains flexible, surgery may be delayed until
the curve reaches approximately 90 degrees of magnitude, and this can usually be
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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
corrected by posterior spinal fusion in the advent of modern segmental pedicle screw
instrumentation techniques (Figure 3). For stiff curves or those greater than 90 degrees
with fixed pelvic obliquity, surgical release and fusion of the anterior column of the
spine may be required to increase flexibility of the deformity and allow for better
correction in addition to posterior spinal fusion; this combined procedure increases the
potential morbidity and complications associated with surgery.
The instrumentation and fusion of the deformity in neuromuscular scoliosis usually
extends from the upper thoracic spine (T2 vertebra) proximally to the sacrum/pelvis
distally as this prevents the development of recurrent deformities above and below the
instrumentation that can occur with further skeletal growth if a shorter fusion is
performed [26, 30-33]. In the absence of significant pelvic deformity the lower lumbar
spine may be selected as the lower extent of the spinal fusion to preserve the patient’s
lumbosacral flexibility [34, 35].
Growth preservation techniques (growing rods) may be considered in young patients
with severe but flexible curves as a temporising measure spanning the levels of the
deformity across the spine and often extending down to the pelvis. The benefits of
preserving spinal growth and delaying the need of the definitive spinal fusion for a later
age have to be balanced over the considerable risk of major complications in the need
of serial operations which is increased due to the underlying medical co-morbidities
and inherent bone weakness. Control of scoliosis and spinal growth with growing rod
surgery has been reported as an effective strategy in patients with spinal muscular
atrophy [36].
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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
8. Perioperative Care
8.1. Perioperative Risks and Preoperative Planning
The risks associated with surgery for neuromuscular scoliosis are correlated with the
severity of neurological impairment and associated co-morbidities. Factors that are
strongly associated with increased risk of postoperative complications include non-oral
feeding, severe cognitive impairment, inability to communicate vocally, presence of
seizures, and non-independent sitting ability. As patients with neuromuscular disease
often have varied and complex medical conditions, pre-operative assessment requires
a multidisciplinary involvement. In particular, it is important to recognise and optimise
any seizure activity, cardio-respiratory problems, gastro-intestinal reflux or
dysmotility, feeding and nutritional difficulties. As the risk of surgery is greatly
increased by cardiac dysfunction or arrhythmias associated with DMD, pre-operative
assessment by a paediatric cardiologist and anaesthesiologist is mandatory [37]. Preoperative pulmonary function tests positively correlate with the incidence of
postoperative pulmonary complications, with values below 30-40% of predicted vital
capacity providing the best indication of a high risk of major postoperative respiratory
complications [38]. Spinal stabilisation can, however, be performed safely in patients
with low vital capacity and those with pre-existing respiratory failure requiring
nocturnal non-invasive ventilation [39, 40]. The use of preoperative non-invasive
ventilation may be helpful for patients with forced vital capacity less than 40% of
predicted values or dependent on arterial blood gas results.
Further specific anaesthetic measures beneficial in the peri-operative management of
patients with neuromuscular scoliosis include hypotensive anaesthesia, use of cell
salvage, tranexamic acid, haemodilution, haemodynamic monitoring, and careful
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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
maintenance of normothermia [41-44]. A summary of preoperative investigations for
neuromuscular patients undergoing spinal stabilisation is provided in Table 2. To
protect function, spinal cord monitoring is performed intra-operatively with
transcranial motor and somatosensory evoked potentials for patients who are
ambulatory, can stand, or have purposeful lower extremity movement or for those
patients with normal bladder and bowel function. Spinal cord monitoring is therefore
utilised intra-operatively for patients with muscular causes and mild cerebral palsy, but
may not be appropriate, for example, in patients with severe quadriplegic cerebral palsy
or severe spinal cord dysfunction. When performed together, transcranial motor and
somatosensory evoked potentials have 95-100% reliability and permit motor function
testing intra-operatively and adaptation of recording sites to the relevant motor function
for each patient [45-47]. Patients, family and caregivers should be fully informed
regarding the potential for prolonged and complex intensive care and in-patient stay
postoperatively (additional education resources are shown in Figure 4). Furthermore,
resuscitation decision-making should also be discussed preoperatively with the patient
and their family as appropriate.
8.2. Postoperative Care
Patients may remain in an intensive care unit, often still intubated, for several days
postoperatively, for close monitoring of cardio-respiratory and haemodynamic status,
before returning to standard ward care. The required duration of postoperative
ventilation is determined individually and is usually no more than 36 hours. Early reinstigation of nutritional intake, often with the use of nasogastric, nasojejunal or
gastrostomy tubes and nutritional supplementation, is important in the postoperative
period. This is particularly important perioperatively for patients with cerebral palsy
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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
who are often malnourished prior to surgery [48]. Patients are encouraged to mobilise
out of bed or in a wheelchair as soon as medically safe following surgery. Patients are
usually able to mobilise using a wheelchair by day 2-5 postoperatively [42, 49].
Adjustments to patients’ wheelchairs can also be made during this period as necessary
and by assessment of an orthotist. Patients are usually advised to avoid rotation of the
spine or flexion of the hips beyond 90 degrees for 4-6 months postoperatively, and
appropriate techniques are learnt with the assistance of a physiotherapist. Consideration
should be given to the need of a hoist for patient transfers during subsequent
rehabilitation. Postoperative trunk support in the form of a brace is usually not required
following the use of modern spinal stabilisation techniques. Following discharge,
children can usually return to school within 3-4 weeks.
9. Outcomes of Treatment
9.1. Patient Satisfaction and Quality of Life
With modern medical care, the life expectancy of patients with complex neuromuscular
conditions has been considerably prolonged. Therefore, treatment of their spinal
deformities is now becoming a greater necessity in order to maintain functional abilities
and preserve quality of life. For patients with severe neuromuscular disease, careful
consideration must be given on whether or not the risks of an extensive surgical
procedure in the presence of complex medical problems outweigh any anticipated
benefits. Surveys following surgical correction for patients with total-bodyinvolvement spastic cerebral palsy and scoliosis demonstrated that 85-96% of parents
and caregivers are satisfied with the outcomes for the patient after surgery and would
choose the procedure again [29, 50, 51]. Surgery resulted in improved sitting ability,
function, activities of daily living, physical appearance, comfort, ease of care, and self-
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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
image [52-55]. Such improvements may positively affect quality of life for these
patients, which is important as the mean patient survival following surgical correction
for scoliosis secondary to severe cerebral palsy has been reported as 11.2 years [56].
Similar satisfaction rates have been demonstrated in surveys of patients treated for
DMD scoliosis, who also report improvements in breathing, digestion and posture
postoperatively [57]. In DMD scoliosis, early surgical correction improves the
deformity, and may have a positive impact on survival [58, 59].
9.2. Effect of Surgical Intervention on Respiratory Function
Prior to surgery, patients with neuromuscular disease and scoliosis have lower
pulmonary function parameters than those without scoliosis and healthy controls [60].
In a prospective study including neuromuscular patients, pulmonary function declined
up to 60% immediately following surgery, reaching a nadir at 3 days postoperatively,
but returning to preoperative values between 1 and 2 months after surgery [61]. One
year following spinal stabilisation, peak cough flow and end tidal CO2 measurements
are reportedly well maintained in neuromuscular patients [62]. However, the long-term
effect of surgical stabilisation of scoliosis on respiratory function is incompletely
understood and existing evidence is conflicting, with studies reporting both no
difference in subsequent rate of decline and other studies indicating a reduced rate of
decline in respiratory function [63-69]. The effect of spinal stabilisation on gastric
emptying, upper gastrointestinal symptoms and clinical nutritional state has been
reported as minimal [70].
9.3. Complications of Treatment
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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
The incidence of complications following surgical correction of neuromuscular
scoliosis has been estimated as ranging from 18% to 68%. Complications should
therefore be actively anticipated, especially in patients with DMD who have higher
overall rates [71, 72]. Non-ambulatory status and severe preoperative curve magnitude
greater than 60o are associated with increased risk for major postoperative
complications [73]. The most common complications include superficial and deep
wound infections (13.1%), non-surgical site infections (10%), pseudoarthrosis, (1.9%),
problematic instrumentation (3.4%), and pulmonary/respiratory complications (19.4%)
[42, 74-76].
The most frequent postoperative complication in patients with cerebral palsy is
pulmonary infection, with a particular risk of aspiration pneumonia. Pulmonary
complications are also common following surgical correction in patients with DMD;
postoperative atelectasis is relatively frequent in DMD patients, and the incidence of
pulmonary infection has been estimated at 7-8% [62, 72]. Wound infections also occur
at a similar frequency in patients with DMD, but are less common in patients with
cerebral palsy [72, 77]. In patients with spinal cord dysfunction, particular care must be
given to surgical site and general skin pressure management due to insensate skin, and
also to care of the urinary system to avoid urosepsis.
Other recognised complications include postoperative ileus, pancreatitis, and superior
mesenteric artery syndrome, while neurological complications are rare [78]. However,
surgical complications rarely cause any permanent problem for the patient or
caregivers, but rather extend the need for inpatient care. Indeed, the mean hospital stay
16
Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
for surgical correction of neuromuscular scoliosis can be prolonged and this has been
previously reported as between 14 and 19.6 days [42, 56].
10. Conclusion
Scoliosis is common in patients with neuromuscular conditions. The spinal deformity
is progressive and can lead to loss of ambulation, difficulty sitting, feeding disorders,
pain, poor self-image, and increasing difficulty with daily care. Non-operative
treatment cannot control the deformity but may be used while significant spinal growth
remains. Early referral of patients with neuromuscular conditions and suspected spinal
deformity to specialist spinal centres is essential to optimise the multidisciplinary
management and long-term outcomes. Surgical correction and stabilisation of the spinal
deformity is effective, but it is associated with significant risks that are usually
manageable. Surgical management of neuromuscular scoliosis is associated with good
long-term outcomes and high satisfaction rates among patients, their families and
caregivers.
Acknowledgements
Both authors contributed to the design and writing of the article. The authors have no
relevant interests to declare. This study has no conflict of interest. No funding was
received for this work.
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Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
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Bulman WA, Dormans JP, Ecker ML, Drummond DS. Posterior spinal fusion
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28
Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
Table 1. Scoliosis Research Society classification and incidence of spinal deformity
in neuromuscular conditions.
Classification
Diagnosis
Co-incidence
of Scoliosis
Central neurological
Cerebral palsy
25-74%
Central motor neuron
Friedreich’s ataxia
80%
involvement
Syringomyelia
25-85% [79]
cause
Other (encephalopathy, Rett’s
syndrome)
Peripheral neurological
Acute anterior poliomyelitis
17-65% [80]
Peripheral motor
Spinal muscular atrophy
67%
neuron involvement
Hereditary motor and sensory
26% [81]
causes
neuropathy
Hereditary sensory and vegetative
86% [82]
neuropathy (familial dysautonomia)
Mixed central and
Traumatic paralysis (before 10
100%
peripheral neurological
years of age)
causes
Myelodysplasia
60%
Myelomeningocele
52-89% [83]
Neuromuscular junction
Myasthenia
Unknown
Muscular causes
Duchenne Muscular Dystrophy
90%
Arthrogryposis
30-67% [80]
Other muscular dystrophies
29
Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
Table 2. Preoperative clinical and radiological investigations for patients undergoing
surgical correction of neuromuscular scoliosis
Role of
Investigation for Neuromuscular Scoliosis
Investigation
Assessment of Spinal

Pathology
Whole spine radiographs (PA, lateral & supine
maximum traction)

Whole spine MRI (if any suspicion of intraspinal
pathology)

Hips & pelvic radiographs
Anaesthetic &

FBCs, U&Es, LFTs
Medical Assessment

Blood cross-match

Albumin/calcium/vitamin D

Coagulation screen

Chest radiograph

ECG, echocardiogram

PFTs, ABGs, sleep studies, spirometry

Nutritional assessment

Urinalysis, urine culture & sensitivities

Neurological assessment
Keys: FBC (full blood count), MRI (magnetic resonance imaging), U&Es (urea &
electrolytes), PFTs (pulmonary function tests), ABG (arterial blood gases), ECG
(electrocardiogram), LFTs (liver function tests), PA (posteroanterior).
30
Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
Figure Captions
Figure 1. Spinal deformity patterns in cerebral palsy associated neuromuscular
scoliosis. In types A and B (double curves of the thoracic and lumbar spine), the spine
remains well balanced with little or no pelvic obliquity. In types C and D (large
thoracolumbar or lumbar curves), the deformity extends into the sacrum and often
causes significant pelvic obliquity.
Figure 2. Radiographs of a patient with Friedrich’s ataxia showing pre-operative a)
posteroanterior (PA) and b) lateral radiographs of the spine at age 14 years and 6
months. A primary thoracolumbar scoliosis measuring 50o with a compensatory
thoracic curve measuring 35o and no pelvic deformity can be seen. Post-operative c)
PA and d) lateral radiographs at age 17 years and 2 months following posterior spinal
fusion demonstrate excellent correction of the thoracolumbar and thoracic curves to 8o
and 5o, respectively, with restoration of global spinal balance.
Figure 3. Radiographs of a patient with quadriplegic cerebral palsy showing preoperative a) posteroanterior (PA) and b) lateral radiographs of the spine at age 14 years
and 8 months. A collapsing thoracolumbar scoliosis measuring 88o with associated
thoracic hyperkyphosis measuring 98o and severe pelvic obliquity can be seen. The pain
had severe back and left costo-pelvic impingement pain (bony pain produced due to the
lower ribs sitting against the elevated left side of the pelvis) which did not allow him to
sit on his wheelchair. Post-operative c) PA and d) lateral radiographs at age 17 years
and 1 month following posterior spinal fusion demonstrate correction of the
thoracolumbar curve to 25o, levelling of the pelvis and normalisation of thoracic
kyphosis to 40o. An intrathecal Baclofen pump may also be seen in the radiographs and
this was preserved in place during scoliosis surgery.
31
Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
Figure 4. Additional education resources for patients and healthcare professionals.
32
Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
Figure 1
33
Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
Figure 2a
34
Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
Figure 2b
35
Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
Figure 2c
36
Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
Figure 2d
37
Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
Figure 3a
38
Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
Figure 3b
39
Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
Figure 3c
40
Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
Figure 3d
41
Factors Influencing the Evaluation and Management of Neuromuscular Scoliosis
Figure 4

Scoliosis Association (UK): charity supporting patients with scoliosis;
www.sauk.org.uk

Scottish National Spine Deformity Service: information regarding the
service, spinal deformities, and their treatment;
http://www.nhslothian.scot.nhs.uk/Services/AZ/ScottishNationalSpineDeformityService/Pages/default.aspx

British Scoliosis Society: society supporting surgeons, healthcare workers
and researchers regarding scoliosis; http://www.britscoliosissoc.org.uk

British Scoliosis Research Foundation: charity promoting research into
scoliosis and allied health conditions in the UK; http://www.bsrf.co.uk

Scoliosis Research Society: society supporting education for healthcare
workers and research in spinal deformities; http://www.srs.org
42