Child's Nervous System (2021) 37:1983–1989

https://doi.org/10.1007/s00381-020-05018-2

ORIGINAL ARTICLE

Long-term upper extremity performance in children with cerebral

palsy following selective dorsal rhizotomy
Patricia Mortenson 1,2 & Nishanth Sadashiva 3 & Mandeep S. Tamber 4 & Paul Steinbok 4

Received: 4 June 2020 / Accepted: 17 December 2020 / Published online: 2 January 2021
# The Author(s), under exclusive licence to Springer-Verlag GmbH, DE part of Springer Nature 2021

Abstract
Purpose In children with spastic cerebral palsy, selective dorsal rhizotomy (SDR) is conducted to improve lower limb spasticity.
Improvements in upper extremity function have also been noted in early follow-up. The purpose of this study was to determine if
upper extremity improvements are sustained in the long term.
Methods A retrospective review of prospectively collected data on children who underwent SDR was conducted. Quality of
Upper Extremities Skill Test (QUEST) scores for dissociated movement, grasp and total scores were compared using repeated
measures ANOVA for individual patients at three time points: preoperatively, early post-operatively (≤ 2 years) and late post-
operatively (9+ years).
Results Out of 200+ patients having SDR, 32 had QUEST assessment at all three time points. Significant improvements in
QUEST dissociated movement (F = 3.665, p = 0.045), grasp (F = 7.995, p = 0.001) and total scores (F = 9.471, p = 0.001) were
found. Pairwise comparisons were significant from pre-operative to early post-operative times for all QUEST scores (p = 0.001,
0.003, 0.001), and this was maintained at late post-operative assessment for grasp and total scores (p = 0.02, p = 0.02). There was
no significant change in scores between early and late post-operative assessment time points.
Conclusion Early improvements in upper extremity QUEST total scores are sustained in the long term following SDR.

Keywords Upper limb . CP . Function . Suprasegmental

Introduction early in life and leading to motor and activity limitations [1,
2]. Interventions for management include physical and occu-
Cerebral palsy (CP) is a term for disorders caused by non- pational therapy, orthoses, oral medications, botulinum toxin
progressive cerebral abnormalities occurring before birth or type A injections, intrathecal baclofen, orthopaedic surgery
and selective dorsal rhizotomy (SDR) [3].
Used since the 1980s to alleviate lower limb spasticity in
What this paper adds
• Improvements in upper extremity movement following SDR are children with CP [4], SDR reduces the excitatory sensory
maintained in the long term input from the legs entering the spinal cord by selectively
transecting the posterior lumbosacral sensory nerve rootlets
* Patricia Mortenson from L1 or L2 to S1 or S2, based on EMG and/or clinical data
pmortenson@cw.bc.ca [5]. SDR is typically used to reduce spasticity in ambulatory
children (Gross Motor Function Classification System
1
Department of Occupational Therapy, British Columbia Children’s (GMFCS) levels I–III) and results in improvements in lower
Hospital, 4480 Oak Street, Room K3-130, Vancouver, BC V6H limb spasticity and movement, performance of activities of
3V4, Canada daily living and quality of life [4, 6–9]. Although SDR tran-
2
Department of Occupational Science and Occupational Therapy, section is limited to the lumbosacral nerve rootlets, early re-
University of British Columbia, Vancouver, Canada ports described unexpected improvements above the level of
3
Department of Neurosurgery, National Institute of Mental Health and the lower extremities, termed ‘supra-segmental effects’ [10].
Neurosciences, Bengaluru, India In a previous study, upper extremity function, as measured by
4
Division of Pediatric Neurosurgery, Department of Surgery, the Quality of Upper Extremity Skills Test (QUEST), im-
University of British Columbia and British Columbia Children’s proved significantly 1 year following surgery [10]. Other case
Hospital, Vancouver, Canada
1984
series described further anecdotal improvement in upper ex-
tremity function or tone following SDR [8–14].
The objective of this study was to evaluate long-term
changes in upper extremity function post-SDR.
Methods
After obtaining ethics approval from the local university clin-
ical research ethics board (certificate # H17-02783), a retro-
spective review of prospectively collected data was conduct-
ed. This included all patients with CP who underwent SDR
from 1987. Our centre follows an SDR protocol, which in-
cludes assessments conducted pre-operatively, at 1 year post-
operatively and at intervals thereafter by a neurosurgeon, or-
thopaedic surgeon, physiotherapists and occupational thera-
pists. Assessment of upper extremity performance began in
late 1992 using the QUEST, chosen as it was the only upper
extremity measure designed specifically for a paediatric
neuromotor population at that time. Occupational therapists
experienced in working with children with CP and familiar
with the QUEST conduct the assessments. The therapists are
not involved with the ongoing treatment of children, but are
not blinded to the children’s operative status and previous
QUEST performances. The collected data is entered into an
ongoing database. For this study, only patients with preoper-
ative and long-term (close to 10+ years) follow-up QUEST
assessment data were included.
All SDR operations were performed by a single group of
paediatric neurosurgeons and consisted of either L1–S1
laminoplasties (1987–2005) or single-level laminectomy
(L1) at the conus medullaris–cauda equina junction (from
2005), with L1–S1 nerve rootlet transection guided by clinical
findings and electrophysiological monitoring. The goal was
approximately half to two-third transection of rootlets. Post-
operatively, children received intensive physiotherapy inter-
vention focussed on mobility, but no interventional therapy
specifically directed at upper extremity function.
The QUEST is a criterion-referenced tool developed by
and for therapists to look at patterns of upper extremity move-
ment and to measure changes in upper extremity performance
in children with neuro-motor dysfunction with spasticity [15,
16]. The tool has good intra- and inter-rater and test-retest
reliability [15, 17] and Gilmore et al. (2010) recommended
the QUEST as an outcome assessment to measure change in
uni-manual function following spasticity management or sur-
gery [18]. The QUEST measures performance in four do-
mains: dissociated movements, grasp, weight bearing and pro-
tective extension. Only the dissociated movements and grasp
domains were administered, because the other two domains
are difficult to assess in children with severe motor limitations
[19]. The number of items and raw point scores vary by do-
main, but domain subscores and QUEST total score are
Childs Nerv Syst (2021) 37:1983–1989
calculated with weighted formulas and reported in percentage
scores, with a maximum possible score of 100%. Thorley
et al. recommend reporting domain scores for improved va-
lidity, rather than combined total scores [19].
The primary outcome measures for this study were the
QUEST dissociated movements (DM) and grasp domains;
however, to be consistent with our previous 1-year follow-
up study, the combined total score of the QUEST was used
as a secondary outcome. A significance value of p = 0.05 was
pre-set as being statistically significant. Repeated measures
ANOVA analyses were conducted to determine the change
in the QUEST between the pre-operative, early post-
operative and long-term follow-up time points. Pairwise post
hoc comparisons were conducted between time points to as-
certain when significant changes occurred. Descriptive analy-
ses were calculated on the Excel database. All other statistical
analyses were conducted using the Statistical Package for
Social Sciences version 25.
In the absence of published data to guide the interpretation
of clinical significance of the change in QUEST scores, a 3%
change in score was used following personal communication
with one of the QUEST authors (Carol De Matteo, personal e-
mail communication, February 19, 2019), calculations based
on Norman et al. [20] and the clinical experience of the first
author. The influences of age at surgery, average percentage
of L2-S1 rootlets cut and degree of cerebral palsy involvement
(as described by pre-operative GMFCS level) to impact a
change score of greater than three were analysed using logistic
regression, with p value adjusted for multiple comparisons
(p < 0.05/3 = 0.017).
Results
Of the 150 patients who underwent SDR during the study time
frame, 39 were excluded as they missed an initial pre-
operative QUEST assessment. Reasons included therapist un-
available or appointment missed by family. Return for a long-
term assessment was voluntary, and 75 did not or were lost to
follow-up. Of the remaining, 36 had both a preoperative and a
long-term post-operative follow-up QUEST assessment at 9
years or more following surgery. Of this group, 32 had an
additional ‘early’ post-operative assessment. Due to variations
in family availability to attend voluntary follow-up, this ‘ear-
ly’ time point ranged between 3 and 24 months post-SDR, but
provided an intermediary time point to look at the trend over-
time. In addition, 17 were part of an earlier cohort of 36 in an
analysis of 1-year follow-up upper extremity changes [10].
Table 1 details the age ranges and means at the assessment
time points and the extent of the SDR procedure, reported as
average percentage of rootlets cut. One subject had surgery
after the 2005 change in surgical technique. Patients were at
Childs Nerv Syst (2021) 37:1983–1989
Table 1 Ages at assessment time points and average percentage of rootlet cut
Age at pre-op Age at early Time from surgery
assessment post-op assessment to early post-op assess-
ment
Range 3–7 years and 4–8 years and 3–23 months
6 months 5 months
Mean 4 years and 9 5 years and 8 13 months
(SD) months months (4 months)
(14 months) (16 months)
pre-operative GMFCS level II (5 patients), level III (15 pa-
tients), level IV (13 patients) and level V (3 participants).
The mean scores for the QUEST dissociated movements
(DM), grasp and total scores at the three time points are listed
in Table 2. Four patients missed early post-operative assess-
ment and were excluded from the repeated measures ANOVA
but included in later analyses. All data for the remaining 32
patients were analysed. Sphericity was violated for the
QUEST DM scores and Greenhouse-Geisser correction was
applied. The findings indicate that the mean QUEST DM,
grasp and total scores all improved over time. There were
significant improvements in all outcomes at the early preop-
erative assessment and these changes remained significant
long term for QUEST grasp and total score. For the QUEST
DM, there was a small decrease in mean scores at the late post-
operative assessment (0.7), but unlike other outcomes, the
change did not remain significant between baseline and late
post-operative assessments. Floor and ceiling effects were un-
likely as < 15% of patients had the worst (48%) or best (100%)
percentage scores.
Most, but not all, patients experienced either clinically sig-
nificant improvements or stability in upper extremity function
dissociated movement and grasp scores in the long term as
determined by a change of 3% or greater in score (see
Table 3). Findings from the logistic regression analyses indi-
cated younger age at surgery was predictive for better im-
provement in upper extremity dissociated movement at early
pre-op (p = 0.01) but not in the long term (see Table 4).
Overall, lower GMFCS level was predictive for better im-
provement in grasp score in the long term (p = 0.01). A sepa-
rate analysis by level was not conducted due to limited num-
bers in each category. Average percentage of rootlet cut was
not predictive at either time point.
Discussion
The results of this long-term study extend the findings of a
prior study on upper extremity function following SDR,
1985
Age at long-term Time from surgery to Average %
assessment long-term assessment of
L2 to S1
rootlet cut
12 years and 4 months 9–18 years and 46–65%
–26 years and 10 months
2 months
17 years and 2 months 12 years and 6 55.4%
(3 years and months
2 months) (2 years and
9 months)
which reported an improvement in the mean QUEST score
of 3.2 from preoperatively to 1-year postoperatively [10]. The
improvements in upper extremity function observed by pro-
fessionals correlated well with positive gains in motor self-
care skills parents observed at home. Our findings years after
SDR demonstrate that upper extremity ‘supra-segmental ef-
fects’ for the most part are maintained over time, but with
variability typical of the CP population.
Although SDR is limited to the lumbosacral region, upper
extremity improvements can be explained from findings of
intraoperative electrophysiological stimulation [21]. When
dorsal lumbosacral roots are stimulated electrically, an abnor-
mal response spread is observed in children with CP, involv-
ing the contralateral side and throughout the spinal system,
including to the level of the upper extremities, instead of a
response primarily limited to the ipsilateral level of stimula-
tion [22]. This spread is thought to be due to the lack of normal
spinal interneuron inhibition that occurs with damage to the
descending neurons seen in CP. Similarly, when dorsal roots
are cut in SDR, overall sensory input to the spinal cord inter-
neurons is reduced. It is postulated that this reduction con-
tinues in the spinal cord interneurons, up the spinal cord and
even into the brainstem and cerebral cortex [10]. This may
impact the upper pathways positively, creating a reduction in
upper extremity spasticity, among other ‘supra-segmental ef-
fects’ [10].
There are several reports investigating ‘supra-segmental
effects’ of the upper extremities in the early post-operative
period following SDR, in addition to Loewen et al. [10].
Gigante et al. (2013) compared upper extremity tone
(Modified Ashworth Scale (MAS)) and functional outcomes
of upper limbs pre and post-operatively in patients undergoing
SDR [12]. The mean MAS decreased significantly at a mean
follow-up period of 16 months. The average improvement in
upper extremity MAS score suggested that those with moder-
ately elevated upper extremity tone are most likely to experi-
ence the greatest reduction. Improvement in upper extremity
tone did not correlate with preoperative lower extremity tone,
but improvement in lower extremity tone predicted upper-
1986 Childs Nerv Syst (2021) 37:1983–1989

F(1.496, 46.370) = 3.665** (0.045)

extremity tone improvement. Buckon et al. (1995) reported no
change in spasticity, range of movement and muscle strength
in upper extremities 1 year following SDR [23]. Lewin et al.

F(2, 62) = 7.995 (0.001)

F(2, 62) = 9.471 (0.001)

(1994) showed minimal changes in upper extremity mobility,
measures ANOVA

as measured with the MAS, range of motion and reach and

grasp tasks [24]. However, Beck et al. (1993) and Kinghorn
F value (p)
Effect size
Pre-op to 10+ year f/u Early f/u to 10 + year f/u Repeated

(1992) both reported improvement in block stacking skills 1

0.234
0.106
year after SPR [11, 14], and Kinghorn and Mittal et al. (2002)

0.21
both reported fine motor skill improvement at 1-year post-
surgery, with Mittal et al. using a developmental measure
[25]. In addition, Mittal et al. reported the improvements held
at 3 and 5 years following surgery.
The majority of these early studies did not use standard-
ized, valid and reliable assessments of upper extremity func-
tion or used measures potentially less sensitive to changes in
1.0
1.0

1.0
(p)

populations with neuromotor dysfunction or developmental

changes. In addition, others used physiologic spasticity mea-
sures which may not capture changes in function. These mea-
surement limitations may explain the variety and contradicto-
ry nature of early findings. A previous study discussed how
the QUEST was designed with minimal developmental se-
0.28

0.02

0.02
(p)

quencing and has lower correlations with age [10, 26]. For
our study, this means that the measured long-term improve-
Pre-op to Long-term post-op assess-

88.0% (13.4)

80.6% (17.0)

84.3% (14.9)
Mean (SD)

ments are unlikely to be due to maturation over time.

Our findings demonstrate variability in long-term out-
comes with little predictability for the factors accounted for
(10+ years post)

in this study. All assessment data showed widening in the

53.1–100%

38.9–100%

standard deviation of scores in the long term. In addition,

46–100%
Range

although the average QUEST DM change score of 3.5 was

early f/u ment

above the 3 points pre-determined for clinical significance,

eight patients had a clinically significant decline representing
variability in long-term upper extremity dissociated move-
0.001

0.003

0.001
(p)

ment. This variability likely limited the statistical significance

of long-term improvement for the QUEST DM. The percent-
age of rootlets cut in surgery did not influence the occurrence
88.7% (10.1)

81.3% (16.3)

85.0% (12.8)
Early post-op assessment (3–

Mean (SD)

of clinical improvement, but for this sample, there was limited

variation in this factor. Age at surgery appeared to have an
impact in the short term, with younger age at surgery resulting
QUEST scores and repeated measures ANOVA

24 months post)

in greater short-term improvement in dissociated movements

(not grasp), perhaps explainable by greater potential for
85.3% (11.2) 60.2–100%

38.9–96.3% 76.8% (14.8) 38.9–100%

81.1% (12.6) 49.6–100%

italicized values are significant p < 0.05/3 = 0.017

neuroplasticity and/or developmental potential at a younger

Range

*QUEST scores are reported as percentages

age. At long-term follow-up, this advantage disappeared.

**Greenhouse-Geisser correction applied

Less CP involvement as described using the GMFCS predict-

Mean (SD)

ed improvement in grasp skills, but not dissociated movement.

It might be that for those with little involvement, greater im-
Pre-op assessment

provements can be gained with maturation and use over time.

It is important to put our findings in the context of expected
48–98.2%
57–100%

long-term trends for individuals with CP [27]. For children at

Range

GMFCS levels III to V (the majority of our study partici-

pants), Hanna et al. (2009) described a “peak and decline”
trajectory of gross motor function dependent on the GMFCS
grasp*

total*
DM*
QUEST

QUEST
QUEST
Table 2

level, with a peak at between ages 7 years and 11 months and

6 years and 11 months, followed by clinically significant
Childs Nerv Syst (2021) 37:1983–1989 1987

Table 3 Percentage change score

at long-term follow-up Average % change score Number improved Number declined
≥ 3% (overall % of sample) < 3% (overall % of sample)

QUEST DM 3.5 17 (47.2%) 8 (22.2%)

QUEST grasp 5.3 20 (55.6%) 5 (13.9%)
QUEST total 4.1 17 (47.2%) 5 (13.9%)

declines [28]. By contrast, using these reference centiles as a
comparison, Bolster et al. found no long-term gross motor
deterioration for their sample of GMFCS level III children
post-SDR, but rather more improvement than expected, al-
though additional treatment was needed following surgery
[6]. Children at levels I and II also did not decline and they
did not include children at levels IV and V [6]. From our study
analysis, we cannot comment on performance improvements
by separate or groupings of the GMFCS level due to limited
numbers, but the overall trend fits with the Bolster et al. find-
ings of sustained improvement following SDR.
Information of long-term trends for upper extremity func-
tion in CP is limited and uncertain. Upper extremity move-
ment growth curves (until age 70 months) follow a peak at
about 46 months of age and decline trend as measured using
the QUEST [29]. Nordstrand and Eliasson (2013) conducted a
follow-up study of children with unilateral CP 6 years follow-
ing constraint-induced movement therapy and found stability
in hand function but also a declining trend in the quality of
upper extremity movement [30]. For hand skill specifically,
children with bilateral CP involvement tend to have lower and
earlier (at 30 months of age) skill development peak than those
with unilateral CP [31] and tend to hold the improvement in
the long term [27]. In addition, children who have greater
hand development at younger ages tend to have higher skills
in the long term. However, these reports all describe individ-
ual variability, as we found in our results. Over time, a pattern
of upper extremity contracture emerges, more so, but not ex-
clusively, for children with higher GMFCS levels and at a
faster rate in early adolescent years [32]. Upper extremity
movement trends may be different than for hand function
specifically [29] and the impact of contractures over the long
term is not understood. Given this picture of general decline,
our findings of “peak and hold” for upper extremity grasp
skills and overall QUEST scores in the long term post-SDR
may be of significance.

Table 4 Predictors of greater than three percentage improvement (logistic regression analyses)

B SE Exp(B) p value

Early post 10+ years post Early post 10+ years post Early post 10+ years post Early post op 10+-years
op op op op op op n = 32 post op
n = 32 n = 36 n = 32 n = 36 n = 32 n = 36 n = 36

QUEST DM
Age at − 1.648 − 0.609 0.651 0.341 0.192 0.544 0.01* (younger 0.08
surgery better)
% rootlet cut − 0.016 − 0.178 0.078 0.089 0.984 0.837 0.83 0.05
GMFCS 0.886 0.774 0.499 0.453 2.425 2.167 0.08 0.09
level
QUEST Grasp
Age at 0.227 = − 0.445 0.312 0.311 1.255 0.641 0.47 0.15
surgery
% rootlet cut 0.05 − 0.028 0.081 0.076 1.051 0.973 0.54 0.71
GMFCS − 0.882 − 1.419 0.506 0.557 0.414 0.242 0.08 0.01* (lower
level better)
QUEST total
Age at − 0.565 0.609 0.323 0.347 0.568 0.544 0.08 0.08
surgery
% rootlet cut − 0.035 − 0.004 0.079 0.076 0.966 0.996 0.66 0.96
GMFCS − 0.137 − 0.554 0.438 0.432 0.872 0.575 0.75 0.20
level

*p value adjusted for multiple comparisons (p < 0.05/3 = 0.017)

1988
Future research is needed to explore the factors that may
limit and enhance upper extremity function in children with
CP following SDR. Specifically, upper extremity movement
developmental trends may be different than for hand function
specifically and compounding factors, such as the impact of
contractures over the long term, are not well understood. In
addition, there is often little intervention directed at maximiz-
ing upper extremity function specifically for children follow-
ing SDR and the efficacy of potential intervention strategies
could be explored.
The limitations of our findings are consistent with the chal-
lenges of performing long-term outcome research, including
the number of patients lost to follow-up, changes in clinical
care over time (changes in selection of children for SDR and
in surgical procedures), difficulty controlling for subsequent
health changes, medical events, therapeutic interventions and
changes in data collection personnel. There may be a positive
selection bias of those that pursued SDR as well as those who
attended long-term follow-up. The challenges of following a
cohort over a long time (up to 20+ years for some of our
participants) resulted in a small sample size and necessitated
the use of a single observational cohort design. In addition, at
our study start, the QUEST was the only available upper ex-
tremity CP-specific outcome measure, but it was not designed
for use into adolescence. In a recent study, Thorley et al. de-
scribed the QUEST score to be strongly reliable in children
with CP aged 2–12 years [17]. As the mean age at surgery for
children in our study was 4 years and 9 months and the mean
age at long-term follow-up was 17 years, the reliability of
QUEST at late follow-up may be questionable. While the
QUEST continues to be an appropriate outcome measure for
CP, additional measures and classifications of upper extremity
function are now available that cover longer age spans (e.g.
the Shriners Hospital for Children Upper Extremity
Evaluation and the Manual Ability Classification System).
Further limitations include a lack of blinded assessment and
of a minimum clinically significant change score specific for
the QUEST necessitating a reliance on expert opinion and
extrapolations. However, it is precisely because of these chal-
lenges and the rarity in long-term outcome research which
make our results of interest.
Conclusion
The findings of this study suggest that early improvements in
upper extremity function post-SDR are largely, but not uni-
versally maintained in the long-term, with variability. To our
knowledge, this is the first study tracking the long-term ben-
efits of a supra-segmental effect of SDR. While sustained
improvement of upper extremity function is not the primary
consideration for SDR, the potential for this benefit is not
inconsequential and these findings may be beneficial to
Childs Nerv Syst (2021) 37:1983–1989
discuss with parents and team members regarding what
changes to expect and what individual outcomes to track.
Our findings can be generalised only to children with CP
who have undergone SDR at similar ages, with the cautions
of the limitations discussed. There may also be implications
for the delivery of therapy following surgery, to maximize
outcomes not just for lower extremity mobility but also for
upper extremity function.
Code availability Not applicable.
Authors’ contributions All authors contributed to the study conception
and design. Material preparation and data collection were performed by
Patricia Mortenson and Paul Steinbok and analyses were performed by
Nishanth Sadashiva, Mandeep Tamber and Patricia Mortenson. The first
draft of the manuscript was written by Patricia Mortenson and all authors
commented on previous versions of the manuscript. All authors read and
approved the final manuscript.
Data availability The anonymized datasets analysed during the current
study are available from the corresponding author on reasonable request.
Compliance with ethical standards
Conflict of interest On behalf of all authors, the corresponding author
states that there is no conflict of interest.
Ethics approval The methodology for this study was approved by the
Clinical Research Ethics Board of the University of British Columbia
(certificate # H17-02783).
Consent to participate and consent for publication Not applicable (ret-
rospective study).
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