PEDIATRIC/CRANIOFACIAL

Effectiveness of Surgical Treatment for

Neglected Congenital Muscular Torticollis:
A Systematic Review and Meta-Analysis
Hyun Jung Kim, M.P.H.,
Ph.D.
Hyeong Sik Ahn, M.D.,
Ph.D.
Shin-Young Yim, M.D.,
Ph.D.
Seoul and Suwon, Republic of Korea
Background: Findings on the effectiveness of surgical release for neglected
congenital muscular torticollis have been conflicting. This systematic review
aims to examine the literature describing the effectiveness of surgery for ne-
glected congenital muscular torticollis.
Methods: The authors conducted a systematic review to examine the effec-
tiveness of surgical treatment for neglected congenital muscular torticollis by
means of meta-analysis. The authors searched MEDLINE, EMBASE, the Co-
chrane library, and KoreaMed for all articles published before May of 2014.
Studies with quantitative data describing the effectiveness of surgery for con-
genital muscular torticollis were included. The primary outcomes after surgery
were (1) clinical outcome measured by scoring systems; (2) change in range of
motion of the neck; and (3) change in skeletal deformities, measured by Cobb
angle and the cervicomandibular angle. Effect estimates were pooled using a
random-effects meta-analysis.
Results: Twelve studies were included in the review (published between 1976
and 2014), including a total of 220 patients with neglected congenital mus-
cular torticollis. All studies used an interrupted time-series design. Surgical
treatment was successful in 81 percent of cases, based on the scoring systems
for surgical outcome. Outcomes of surgery performed in patients older than
15 years did not differ significantly from those of surgery performed in pa-
tients aged 15 years or younger. The range of motion of the neck and skeletal
deformities showed significant improvement after surgery.
Conclusions: Surgical treatment for neglected congenital muscular torticollis
produced satisfactorily successful results, with improvement in both the range
of motion of the neck and secondary skeletal deformities.  (Plast. Reconstr.
Surg. 136: 67e, 2015.)
CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, IV.

C
ongenital muscular torticollis is one of the This leads to limited neck motion and secondary
most common musculoskeletal problems in musculoskeletal complications such as craniofa-
children. The incidence of congenital mus- cial deformity and scoliosis.3 Approximately 80 to
cular torticollis has been reported to be as high 90 percent of congenital muscular torticollis cases
as 3.92 percent in neonates.1,2 Congenital muscu- resolve with stretching exercises without evident
lar torticollis might be defined as a developmen-
tal disorder of the sternocleidomastoid muscle
Disclosure: The authors have no financial interests
characterized by fibrosis, which results in the
regarding the content of this article.
shortening of the sternocleidomastoid muscle.3

From the Department of Preventive Medicine, College of
Medicine, Korea University; and The Center for Torticol-
lis, Physical Medicine and Rehabilitation, Ajou University
School of Medicine.
Received for publication December 18, 2014; accepted
­January 27, 2015.
Copyright © 2015 by the American Society of Plastic Surgeons
DOI: 10.1097/PRS.0000000000001373
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musculoskeletal complications.4 However, approxi-
mately 10 to 20 percent of patients with congenital
muscular torticollis have a large amount of fibro-
sis composed of dense connective tissue within
the sternocleidomastoid muscle.5 This subgroup
of individuals with congenital muscular torticol-
lis does not respond well to stretching exercises.5
Therefore, surgical treatment is needed for this
subgroup of patients to minimize the secondary
musculoskeletal complications of congenital mus-
cular torticollis.3
The ideal age for surgical treatment of congen-
ital muscular torticollis is younger than 5 years.6,7
However, not all patients who need surgery for
congenital muscular torticollis are able to have
surgical treatment for various reasons, and these
individuals are said to have neglected congenital
muscular torticollis. Patients with neglected con-
genital muscular torticollis may develop chronic
pain and limited range of motion of the neck with
advancing age. They also develop secondary mus-
culoskeletal deformities such as craniofacial asym-
metry and scoliosis.8–10
Because surgical outcome is largely influ-
enced by the timing of surgery,6,7 findings on the
effectiveness of surgical treatment for neglected
congenital muscular torticollis have been conflict-
ing.11 Some studies suggest that surgical treatment
of the sternocleidomastoid muscle for neglected
congenital muscular torticollis is still effective for
cosmetic purposes, pain reduction, and functional
improvement.8 Other studies have reported that
improvement in secondary musculoskeletal defor-
mities after surgery is uncertain in patients aged
older than 5 years, and late complications such
as a disfiguring scar and the presence of lateral
bands were common.9,10 Therefore, the postoper-
ative prognosis is influenced by the timing of sur-
gery, and it has been recommended that the ideal
time for surgical treatment of congenital muscu-
lar torticollis is before 5 years of age.6,7 The aim of
this systematic review is to analyze the literature
exploring the effectiveness of surgical treatment
for neglected congenital muscular torticollis.
PATIENTS AND METHODS
This study is based on the Cochrane Review
Methods, and reporting follows the Meta-anal-
ysis Of Observational Studies in Epidemiology
statements.12
Data Sources
In May of 2014, we performed a comprehen-
sive literature search. We conducted electronic
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Plastic and Reconstructive Surgery • July 2015
searches in the MEDLINE, EMBASE, and Cochrane
Library databases. We also conducted searches in a
regional electronic bibliographic database (Kore-
aMed). No restrictions were imposed in terms
of the publication language, time, or status. The
search strategy was designed for searching MED-
LINE through the PubMed interface. The follow-
ing keywords were used: “torticollis,” “wryneck,”
and “surgery.” Electronic database searches used
both free-text words and Medical Subject Head-
ings. The search strategy was adapted as appropri-
ate for all other databases searched, taking into
account differences in indexing terms and search
syntax for each database. The comprehensive
search strategies are described in the supplemental
file. (See Figure, Supplemental Digital Content 1,
which shows the effectiveness of surgical treat-
ment for neglected congenital muscular torticol-
lis: a systematic review and meta-analysis, http://
links.lww.com/PRS/B335.) We identified further
relevant studies for possible inclusion in our
review by reviewing the reference lists of the stud-
ies identified by our initial search strategies.
Study Selection
The inclusion of all studies was decided inde-
pendently by two reviewers (S.Y.Y. and H.J.K.)
based on predefined inclusion and exclusion cri-
teria. Discrepancies were resolved by discussion
between the reviewers. Study selection included
two levels of screening. Two reviewers indepen-
dently screened the titles and abstracts of inden-
tified studies. We retrieved the articles of any
citation identified by the reviewers for full-text
review. Both reviewers then assessed the reports
to ensure that they met the inclusion criteria
detailed below.
Studies were included in our systematic
review if they met all of the following four inclu-
sion criteria: (1) the patients underwent surgery
for congenital muscular torticollis; (2) the age of
the patients at the time of surgery for congenital
muscular torticollis was 5 years or older; (3) quan-
titative data on the effectiveness of surgery for
congenital muscular torticollis were available; and
(4) the study was not a case report.
Data Extraction
The two reviewers independently performed
data extraction using a predefined data extraction
form. Any disagreement unresolved by discussion
was reviewed by a third author. The following vari-
ables were extracted from the studies: (1) demo-
graphic characteristics such as the number and
 Volume 136, Number 1 • Neglected Congenital Muscular Torticollis

Table 1.  Cheng and Tang’s Scoring System* for Assessment of Surgical Outcome of Congenital Muscular
Torticollis Used in This Review
Excellent Good
Overall Results (3 Points) (2 Points) Fair (1 Point) Poor (0 Points)
Rotational deficits, degrees <5 6-10 11–15 >15
Side flexion deficits, degrees <5 6-10 11–15 >15
Craniofacial asymmetry (no, mild,
moderate, severe) No to mild Mild Moderate Severe
Scar (no, mild, moderate, severe) No to mild Mild Moderate Severe
Band (no, lateral, clavicular, sternal) No Lateral Lateral, clavicular Clavicular, sternal
Head tilt (no, mild, moderate, severe) No Mild Moderate Severe
Subjective assessment (cosmetic and
functional) Excellent Good Fair Poor
Scores 17–21 12–16 7–11 <7
Dichotomous scale Successful Successful Successful Unsuccessful
*Cheng JC, Tang SP. Outcome of surgical treatment of congenital muscular torticollis. Clin Orthop Relat Res. 1999;362:190–200.

Table 2.   Lee and Kang’s Scoring System* for Assessment of Surgical Outcome of Congenital Muscular
Torticollis Used in This Review
Excellent Good
Overall Results (3 Points) (2 Points) Fair (1 Point) Poor (0 Points)
Limitation of rotation or limitation
of side flexion, degrees 0 <10 10–25 >25
Head tilt No Mild Moderate Severe
Scar Fine Slight Moderate Unacceptable
Obvious but cosmetically
Loss of column None Slight acceptable Unacceptable
Obvious but cosmetically
Lateral band None Slight acceptable Unacceptable
Facial asymmetry None Slight Moderate Severe
Scores 17–18 15–16 13–14 ≤12
Dichotomous scale Successful Successful Successful Unsuccessful
*Lee EH, Kang YK, Bose K. Surgical correction of muscular torticollis in the older child. J Pediatr Orthop. 1986;6:585–589.

sex of the patients; (2) age at the time of surgery;
(3) follow-up period after surgery; (4) interven-
tion protocol; (5) outcomes of surgical treatment;
and (6) postoperative complications.
The outcomes of surgical treatment for
neglected congenital muscular torticollis that
were used in the meta-analysis were as follows: (1)
the results of the scoring systems that quantitated
the outcomes; (2) the change in deficits of rota-
tion of the neck; (3) the change in the deficits
of lateral flexion of the neck; (4) the change in
Cobb angle; and (5) the change in cervicoman-
dibular angle.
Of 12 studies, 11 reported the scoring system–
based results after surgery, whereas Minamitani et
al. did not use a scoring system.13 Both Cheng and
Tang’s scoring system14 and Lee and Kang’s scor-
ing system15 quantified the outcomes of surgery
by dividing the potential results into four levels:
excellent, good, fair, and poor. Both Canale’s scor-
ing system6 and Ling’s scoring system9 used three
levels: good, fair, and poor. The results of the four-
level scoring systems were converted into a dichot-
omous scale: successful and unsuccessful. For this
scale, a poor response on the four-level scoring
systems was rated as unsuccessful; and excellent,
good, and fair responses were considered success-
ful. The four-level scoring systems for the assess-
ment of congenital muscular torticollis and the
corresponding dichotomous scale are presented
in Tables 1 through 4.
Along with the meta-analysis of the results of
the scoring system, we conducted subgroup analy-
ses that compared the surgical success between
patients who underwent surgery before 15 years
of age and patients who underwent surgery after
15 years of age.
Whereas the results of the surgical outcome
scoring system comprised a single measurement,
the other four outcomes of surgical treatment
measured the difference before and after sur-
gery. In these cases, the postsurgery measure-
ment used was the last measurement taken after
surgery. The deficits in the rotation and lateral
flexion of the neck before and after surgery refer
to the deficits compared to those of the con-
tralateral, non–congenital muscular torticollis
side, and were measured with a goniometer. A

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 Plastic and Reconstructive Surgery • July 2015

Table 3.  Canale’s Scoring System* for Assessment of Surgical Outcome of Congenital Muscular Torticollis Used
in This Review
Dichotomous
Cosmetic Result Functional Result Result Scale
Satisfactory: Satisfactory: Good Successful
• No facial asymmetry or facial asymmetry • Loss of rotation of the head ≤30 degrees
noted only by the examiner • Loss of lateral bending ≤20 degrees
• No head tilt • Unsatisfactory Fair Successful
• No lateral band • Loss of rotation of the head >30 degrees
• No scar • Loss of lateral bending >20 degrees
Unsatisfactory: Satisfactory: Fair Successful
• Apparent facial asymmetry to the par- • Loss of rotation of the head ≤30 degrees
ents and/or patients • Loss of lateral bending ≤20 degrees
• Residual deformity of sternocleidomas- Unsatisfactory Poor Unsuccessful
toid muscle
• Loss of rotation of the head >30 degrees
• Loss of lateral bending >20 degrees
*Canale ST, Griffin DW, Hubbard CN. Congenital muscular torticollis: A long-term follow-up. J Bone Joint Surg Am. 1982;64:810–816.

Table 4.  Ling’s Scoring System* for Assessment of Surgical Outcome of Congenital Muscular Torticollis Used in
This Review
Limitation of Lateral Bands/
Facial Neck Rotation Loss of Recurrent Dichotomous
Asymmetry (degrees) Head Tilt Scar Column Torticollis Result Scale
No None No Fine No, or not easily No, or only inconspicuous Good Successful
detectable lateral bands are present
Mild <25 Mild Moderate Obvious but Obvious lateral bands but Fair
cosmetically not impairing appearance
acceptable
Moderate to >25 Moderate to Wide and Cosmetically Muscles rejoined with Poor Unsuccessful
severe severe disfiguring unacceptable recurrent torticollis
*Ling CM. The influence of age on the results of open sternomastoid tenotomy in muscular torticollis. Clin Orthop Relat Res. 1976;116:142–148.

smaller deficit indicates an improvement in the
range of motion of the neck. Cobb angle, which
evaluates scoliosis of the spine, was measured
on plane radiography of the spine before and
after surgery, and a smaller Cobb angle indicates
improvement of scoliosis. The cervicomandibu-
lar angle was defined as the angle between a line
along the upper border of the C7 vertebral body
and a line connecting the lower margins of the
mandibular angle, and was measured from the
anteroposterior radiograph of the cervical spine
to quantify the degree of head tilt.16,17
Assessment of Methodologic Quality
Assessment of Risk of Bias
Two reviewers independently assessed the meth-
odologic qualities of each study using the risk of
bias for interrupted time series studies method sug-
gested by the Cochrane Effective Practice and the
Organisation of Care Group.18 During the assess-
ment of risk of bias, six standard criteria should be
considered when evaluating validity and bias in stud-
ies: (1) Was the intervention independent of other
changes? (2) Was the shape of the intervention effect
prespecified? (3) Was the intervention unlikely to
affect data collection? (4) Was the knowledge of the
allocated interventions adequately prevented dur-
ing the study? (5) Were incomplete outcome data
adequately addressed? (6) Was the study free from
selective outcome reporting?
Assessment of Publication Bias
We assessed possible publication bias using the
symmetry/asymmetry of funnel plots. The funnel
plot was drawn by plotting the logit event rates by
their precision, using Duval and Tweedie’s trim and
fill method,19 which imputes the results that are
hypothetically missing because of publication bias.
Statistical Analysis
For the five outcomes of surgical treatment
for neglected congenital muscular torticollis, a
meta-analysis was performed using one of two pro-
grams: (1) Review Manager software (RevMan 5.2,
2012), which was supplied by the Cochrane Col-
laboration; or (2) Comprehensive Meta Analysis
(version 2.2.064, July 27, 2011; Biostat, Inc., Engle-
wood, N.J.). We used the inverse variance method
to estimate the weighted mean difference. For

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 Volume 136, Number 1 • Neglected Congenital Muscular Torticollis
Fig. 1. Flow diagram of study selection.
heterogeneity, we calculated I 2 statistics by estimat-
ing the proportion of variance unexplained. Values
above 50 percent were considered significantly het-
erogeneous, but values for all reports were calcu-
lated using the random effect model because of the
inherent limitations of noncontrolled studies.
RESULTS
Identification of Studies
The database searches produced 1131 articles
(Fig. 1), and 95 duplicated articles were excluded.
Of the remaining 1036 articles, 998 publications
were excluded because it was clear from the title
and abstract that they did not meet the inclusion
criteria. We obtained full manuscripts for the
remaining 38 articles, and following scrutiny of
these, we identified 12 potentially relevant studies.
Twenty-five publications were excluded because
they did not meet the inclusion criteria: 13 stud-
ies did not meet the age criterion,14,20–31 eight were
case reports,32–39 three did not provide quantitative
outcomes before and after surgery,40–42 and one was
a review article.43 The patient sample of one study16
Copyright © 2015 American Society of Plastic Surgeons. Unauthorized reproduction of this article is prohibited.
partially overlapped with that of the other study by
Shim et al.17 Therefore, the total number of stud-
ies included in the review was 12 (Fig. 1).8,9,13,17,44–51
Study characteristics and patient populations
are listed in Table 5. All 12 studies analyzed in
this review were interrupted time series studies.
Eleven studies were retrospective,8,9,13,17,44,45,47–51
and only one46 was a prospective study. A total of
220 patients with neglected congenital muscular
torticollis were included, with 206 patients from
the 11 retrospective interrupted time series stud-
ies and 14 from the one prospective study.46 The
range of the mean age at the time of surgery for
neglected congenital muscular torticollis was 8.8
to 27.4 years and the range of the mean follow-
up period after surgery was 2.2 to 9.5 years. The
mean of the mean age at the time of surgery for
neglected congenital muscular torticollis was
15.09 years. Therefore, we used age 15 years as a
cutting point for subgroup analysis. In terms of
type of surgical intervention, six studies (50 per-
cent)8,9,17,48,50,51 used one of either unipolar or
bipolar release. In the remaining six studies, four
studies (33 percent)44–47 used only bipolar release
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 Plastic and Reconstructive Surgery • July 2015

Table 5.  Study Characteristics and Patient Populations of 12 Interrupted Time Series Studies Analyzed in This
Review
No. of Age at the Time Follow-Up Type of
Reference Study Design Patients of Surgery (yr) Period (yr) Surgical Intervention
Lim et al., 20148 Retrospective 37 27.4 2.4 Unipolar or bipolar release
Patwardhan et al., 201144 Retrospective 12 24.0 2.96 Bipolar release and Z-plasty
Sudesh et al., 201045 Retrospective 14 13.4 3 Bipolar release
Omidi-Kashani et al., 200846 Prospective 14 21.9 2.5 Bipolar release
Shim et al., 200417 Retrospective 32 14 3.25 Unipolar or bipolar release
Arslan et al., 200247 Retrospective 12 11.75 3.5 Bipolar release
Chen and Ko, 200048 Retrospective 18 11 5 Unipolar or bipolar releases
and/or Z-plasty
Akazawa et al., 199349 Retrospective 4 8.8 9.5 Unipolar releases
Minamitani et al., 199013 Retrospective 19 11.2 2.2 Unipolar releases
Itoi et al., 199050 Retrospective 15 10.6 7.7 Unipolar or bipolar release
Tse et al., 198751 Retrospective 14 11.89 3.63 Unipolar or bipolar release
Ling, 19769 Retrospective 29 ≥9 No mention Unipolar or bipolar release
Total 1 prospective
11 retrospective 220 — —

and two studies (17 percent)13,49 used only unipo-
lar release. Various postoperative programs were
used in these studies. Five studies8,9,45,47,51 of 12 (42
percent) used postoperative halter traction, and
two studies47,51 reported the duration of halter
traction. Postoperative braces were used in nine
of 12 studies8,9,13,17,45,46,48,50,51 (75 percent), in which
cast fixation,13 a neck splint resembling the halo
vest,51 and a cervical collar were used for varying
durations. Postoperative neck exercises were used
in nine of 12 studies8,9,13,17,45–48,51 for varying dura-
tions. Two studies44,49 did not use any of the above
postoperative programs.
Assessment of Risk of Bias
The assessment of the risk of bias for the 12
studies is shown in Figure 2. The risk of bias of
all 12 studies was scored as high risk for the stan-
dard criterion “Was the intervention unlikely
to affect data collection?” Ten of 12 studies (83
percent) were scored as high risk for the stan-
dard criterion “Were incomplete outcome data
adequately addressed?” Six of 12 studies (50 per-
cent) were scored as high risk for the criterion
“Was the knowledge of the allocated interven-
tions adequately prevented during the study?”
Meta-Analysis of the Outcomes of Surgical
Treatment for Neglected Congenital Muscular
Torticollis
The point estimate of the successful result of
surgical treatment for neglected congenital mus-
cular torticollis was 0.81 [11 studies, 201 subjects;
0.81 (95 percent CI, 0.73 to 0.87); I 2 = 46.20 per-
cent] (Table 6).
Among the four-level scoring systems, Cheng
and Tang’s scoring system reported the highest
point estimate of the successful result of surgical
treatment for neglected congenital muscular tor-
ticollis [three studies, 81 subjects; 0.98 (95 per-
cent CI, 0.91 to 1.00); I 2 = 0.00 percent]. Ling’s
scoring system showed the lowest point estimate
of the successful result of surgical treatment for
neglected congenital muscular torticollis [one
study, 29 subjects; 0.62 (95 percent CI, 0.44 to
0.78); I 2 = 0.00 percent] (Table 6). The point
estimates of the successful surgical treatment for
neglected congenital muscular torticollis were
0.86 in the studies using Lee and Kang’s scoring
system and 0.81 in the studies using Canale’s scor-
ing system (Table 6).
Subgroup analysis was performed to compare
surgical outcome between individuals who had
surgery before and after 15 years of age for both
Cheng and Tang’s scoring system and Lee and
Kang’s scoring system. Because all 33 patients in
the three studies using Canale’s scoring system49–51
underwent surgery at the age of 15 years or
younger, they were not included in this subgroup
analysis. Surgical results were not significantly dif-
ferent between patients who underwent surgery
after 15 years of age and those who underwent
surgery at or before the age of 15 years (Table 7).
Postoperative complications were reported in four
of 46 patients (8.70 percent) from two studies17,46:
two recurrences, one superficial hematoma, and
two hypertrophic scars.17
Meta-analysis showed that surgical treat-
ment for neglected congenital muscular torticol-
lis was associated with an increase in the range
of motion of the neck (Table 8), measured by
deficits of rotation [three studies,8,13,44 68 sub-
jects; −11.07 degrees (95 percent CI, −17.46 to
−4.68 degrees); I 2 = 87 percent] and deficits of
lateral flexion (three studies,8,13,44 68 subjects;
−20.36 degrees (95 percent CI, −28.63 to −12.09

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 Volume 136, Number 1 • Neglected Congenital Muscular Torticollis
Fig. 2. Assessment of risk of bias.
degrees); I 2 = 90 percent], where a negative value
indicates improvement in the range of motion of
the neck after surgery.
Copyright © 2015 American Society of Plastic Surgeons. Unauthorized reproduction of this article is prohibited.
Meta-analysis showed that surgical treatment
for neglected congenital muscular torticollis was
associated with an improvement in skeletal defor-
mities (Table 8), in terms of scoliosis measured by
Cobb angle [three studies,8,13,46 63 subjects; −5.54
degrees (95 percent CI, −9.31 to −1.78 degrees);
I 2 = 61 percent] and cervicomandibular angle
[one study,8 37 subjects; −11.22 degrees (95 per-
cent CI, −14.91 to −7.53 degrees)], where nega-
tive value of Cobb angle and cervicomandibular
angles indicate improvement in skeletal deformi-
ties after surgery.
Assessment of Publication Bias
The funnel plot analysis, as an exploratory
tool for assessing publication bias, is shown in
Figure 3. For the 11 studies included in the meta-
analysis of surgical treatment outcomes measured
by the scoring systems (open circles), the effect
size of a successful result was plotted against the
standard error. The summary effect size (0.81; 95
percent CI, 0.73 to 0.87) is represented by the
open diamond at the bottom of the plot. Duval
and Tweedie’s trim and fill imputed six hypo-
thetically missing studies on the left side of the
mean that are represented by the filled circles, to
make this plot symmetrical. The filled diamond
represents the adjusted estimate of the success-
ful result of surgical treatment for neglected con-
genital muscular torticollis by taking into account
the role of unpublished studies. The adjusted
value was 0.76 (95 percent CI, 0.61 to 0.86; Egger
regression, p = 0.00002), resulting in a minor
decline in the adjusted point estimate, from 0.81
to 0.76. The open diamond and the filled dia-
mond at the bottom of the plot show 1.45 [logit
(0.81) = 1.45] and 1.15 [logit (0.76) = 1.15],
respectively.
DISCUSSION
This systematic review and meta-analysis pres-
ents evidence to suggest that surgical treatment
for patients with neglected congenital muscular
torticollis is associated with an 81 percent prob-
ability of success, along with improvement in both
range of motion of the neck and secondary skel-
etal deformities. The point estimates of a success-
ful result were not significantly different between
patients who underwent surgery before 15 years
of age and those who underwent surgery after 15
years of age. These findings indicate that surgical
treatment for neglected congenital muscular tor-
ticollis is effective and the effect sizes are equiva-
lent for patients older than 15 years.
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Table 6.  Meta-Analysis of the Successful Result of Surgical Treatment for the Neglected Congenital Muscular
Torticollis
Point Estimates of
­ uccessful Event Rates
S
No. of No. of (Random Effects Model)
Stud- Sample Successful Heterogeneity
Scoring System References ies Size Events Point Estimate 95% CI I  2 (p)
Cheng and Tang’s scoring system8,17,44 3 81 81 0.98 0.91–1.00 0.00 0.84
Lee and Kang’s scoring system45–48 4 58 50 0.86 0.75–0.93 0.00 0.98
Canale’s scoring system49–51 3 33 28 0.81 0.63–0.92 0.00 0.37
Ling’s scoring system9 1 29 18 0.62 0.44–0.78 0.00 1.00
Overall 11 201 177 0.81 0.73–0.87 46.20 0.05
Test for subgroup differences 0.00

Table 7.  Subgroup Meta-Analysis of the Successful Result of Surgical Treatment for the Neglected Congenital
Muscular Torticollis between Surgery before and after 15 Years of Age
Point Estimates of
Successful Event Rates
(Random
No. of Effects Model)
No. of Sample Successful Heterogeneity
Scoring System References Studies Size Events Point Estimate 95% CI I  2 (p)
Cheng and Tang’s scoring system8,17,44 3 81 81 0.98 0.91–1.00 0.00 0.84
 Surgery ≤15 yr old17 1 32 32 0.98 0.80–1.00 0.00 1.00
 Surgery >15 yr old8,44 2 49 49 0.98 0.86–1.00 0.00 0.59
Lee and Kang’s scoring system45–48 4 58 50 0.86 0.75–0.93 0.00 0.98
 Surgery ≤15 yr old45,47,48 3 44 38 0.86 0.72–0.94 0.00 0.91
 Surgery >15 yr old46 1 14 12 0.86 0.57–0.96 0.00 1.00

Table 8.  Meta-Analysis of the Outcomes of Surgical Treatment for Neglected Congenital Muscular Torticollis
Mean Difference
(degrees)*
Outcomes of Surgical No. of Sample Heterogeneity
Treatment References Studies Size Mean Difference 95% CI I  2 (p)
Deficit of rotation of the neck8,13,44 3 68 −11.07 −17.46 to −4.68 87 0.0004
Deficit of lateral flexion of the
neck8,13,44 3 68 −20.36 −28.63 to −12.09 90 <0.0001
Cobb angle8,13,46 3 63 −5.54 −9.31 to −1.78 61 0.08
Cervicomandibular angle8 1 37 −11.22 −14.91 to −7.53 — —
*Postoperatively − preoperatively (random effects model).

Meta-analyses for deficits of rotation, deficits
of lateral flexion, and Cobb angle before and
after surgery showed significantly heterogeneous
results, with I2 values of 87, 90, and 61 percent,
respectively. Substantial variation across studies
was present in the surgical treatment method,
severity of congenital muscular torticollis, age at
the time of surgery, sample size, follow-up period,
and postoperative program. However, although
these variations could be potential sources for
the observed heterogeneity of surgical outcome
among the studies, it was not possible to deter-
mine a clear association of the characteristics of
the studies with the observed heterogeneity.
There are several surgical options for
neglected congenital muscular torticollis, such
as unipolar or bipolar release of the sternoclei-
domastoid muscle; release of one or both heads
of the sternocleidomastoid muscle, in the case of
distal release of the sternocleidomastoid muscle;
partial or complete release; and tendon or mus-
cle release. Therefore, surgical intervention is
the combination of the above options. Although
the type of surgical intervention would be one of
the most influential factors on surgical outcome,
details were not given in most studies. Ling’s study9
reported the lowest surgical success rates among
the 12 studies, but the reason for this is not clear.
Several study limitations should be noted.
There was no randomized controlled study; thus,
the level of evidence of this systematic review and
meta-analysis is Level IV according to the Ameri-
can Society of Plastic Surgeons scales for evidence
rating for therapeutic studies. However, random-
ization of intervention for neglected congeni-
tal muscular torticollis, such as randomizing to

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 Volume 136, Number 1 • Neglected Congenital Muscular Torticollis

Fig. 3. Funnel plot analysis for assessing publication bias. For 11 studies
included in the meta-analysis of surgical outcome measured by the scoring
systems (open circles), the effect size of a successful result was plotted against
the standard error. The summary effect size of a successful result of surgical
treatment (0.81; 95 percent CI, 0.73 to 0.87) is plotted against the standard
error represented by the open diamond at the bottom of the plot. Duval and
Tweedie’s trim and fill imputed six hypothetically missing studies on the left
side of the mean (filled circles), to make this plot symmetrical. The filled dia-
mond represents the adjusted estimate of the successful result of surgical
treatment for neglected congenital muscular torticollis by taking into account
the role of unpublished studies. The adjusted value was 0.76 (95 percent CI,
0.61 to 0.86; Egger regression, p = 0.00002), resulting in a minor decline in the
adjusted point estimate, from 0.81 to 0.76. The open diamond and the filled
diamond at the bottom of the plot show 1.45 [logit (0.81) = 1.45] and 1.15
[logit (0.76) = 1.15], respectively.

surgery or conservative treatment, is not practical.
All 12 studies were interrupted time series studies,
and we must be cautious of the pitfalls in interpret-
ing interrupted time series studies. The presence
of a surgical scar could prevent the blind evalua-
tion of range of motion of the neck after surgery,
which could be a source of risk of bias. However,
the radiographic evaluation of skeletal deformity,
in terms of Cobb angle and the cervicomandibu-
lar angle, could have less risk of bias, because this
evaluation can be performed in a blind manner.
The small number of studies has prevented forma-
tion of a strong body of evidence for the effective-
ness of surgical treatment for neglected congenital
muscular torticollis. However, recruitment of a
large number of patients with neglected congeni-
tal muscular torticollis is difficult because it is not
a common condition. Alternative explanations for
the observed results should be considered. Preop-
erative differences in the patients’ characteristics
in each study could produce the observed results.
There would be some selection bias, such that
the patients with severe clinical characteristics,
patients who preferred surgery, and patients with a
high intention-to-treat would be more likely to be
reported. Moreover, there was no study reporting
the number of patients lost to follow-up after sur-
gery. The funnel plot analysis showed the presence
of publication bias. Missing unfavorable or nonsig-
nificant trials with smaller effects contribute to the
asymmetry of the plot by failing to evenly distrib-
ute the underlying true mean effect size. Even with
limitations, to the best of our knowledge, this is
the first systematic review and meta-analysis of the
effectiveness of surgical treatment for neglected
congenital muscular torticollis.
CONCLUSIONS
We provide evidence to show that surgical
treatment of the sternocleidomastoid muscle
for neglected congenital muscular torticollis
had satisfactory successful results, which are
associated with improvement in both range
of motion of the neck and secondary skeletal
deformities. A standardized scoring system for
before and after surgery for congenital muscu-
lar torticollis should be developed for future
research.

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Shin-Young Yim, M.D., Ph.D.
The Center for Torticollis
Department of Physical Medicine and Rehabilitation
Ajou University School of Medicine
Worldcup-ro 164
Yeongtong-gu, Suwon 443-380, Republic of Korea
syyim@ajou.ac.kr
REFERENCES
1. Chen MM, Chang HC, Hsieh CF, Yen MF, Chen TH.
Predictive model for congenital muscular torticollis: Analysis
of 1021 infants with sonography. Arch Phys Med Rehabil.
2005;86:2199–2203.
2. Hsu TC, Wang CL, Wong MK, Hsu KH, Tang FT, Chen
HT. Correlation of clinical and ultrasonographic features
in congenital muscular torticollis. Arch Phys Med Rehabil.
1999;80:637–641.
3. Yim SY, Yoon D, Park MC, et al. Integrative analysis of con-
genital muscular torticollis: From gene expression to clinical
significance. BMC Med Genomics 2013;6(Suppl 2):S10.
4. Cheng JC, Wong MW, Tang SP, Chen TM, Shum SL, Wong
EM. Clinical determinants of the outcome of manual stretch-
ing in the treatment of congenital muscular torticollis in
infants: A prospective study of eight hundred and twenty-one
cases. J Bone Joint Surg Am. 2001;83:679–687.
5. Hwang JH, Lee HB, Kim JH, et al. Magnetic resonance
imaging as a determinant for surgical release of congenital
muscular torticollis: Correlation with the histopathologic
findings. Ann Rehabil Med. 2012;36:320–327.
6. Canale ST, Griffin DW, Hubbard CN. Congenital muscu-
lar torticollis: A long-term follow-up. J Bone Joint Surg Am.
1982;64:810–816.
7. Hollier L, Kim J, Grayson BH, McCarthy JG. Congenital mus-
cular torticollis and the associated craniofacial changes. Plast
Reconstr Surg. 2000;105:827–835.
8. Lim KS, Shim JS, Lee YS. Is sternocleidomastoid muscle
release effective in adults with neglected congenital muscu-
lar torticollis? Clin Orthop Relat Res. 2014;472:1271–1278.
9. Ling CM. The influence of age on the results of open ster-
nomastoid tenotomy in muscular torticollis. Clin Orthop Relat
Res. 1976;116:142–148.
10. Oh I, Nowacek CJ. Surgical release of congenital torticollis in
adults. Clin Orthop Relat Res. 1978;131:141–145.
11. Khoury J. CORR Insights: Is sternocleidomastoid muscle
release effective in adults with neglected congenital muscu-
lar torticollis? Clin Orthop Relat Res. 2014;472:1279–1280.
12. Stroup DF, Berlin JA, Morton SC, et al. Meta-analysis of
observational studies in epidemiology: A proposal for report-
ing. Meta-analysis Of Observational Studies in Epidemiology
(MOOSE) group. JAMA 2000;283:2008–2012.
13. Minamitani K, Inoue A, Okuno T. Results of surgical treat-
ment of muscular torticollis for patients greater than 6 years
of age. J Pediatr Orthop. 1990;10:754–759.
14. Cheng JC, Tang SP. Outcome of surgical treatment of
congenital muscular torticollis. Clin Orthop Relat Res.
1999;362:190–200.
15. Lee EH, Kang YK, Bose K. Surgical correction of mus-
cular torticollis in the older child. J Pediatr Orthop.
1986;6:585–589.
16. Shim JS, Jang HP. Operative treatment of congenital torticol-
lis. J Bone Joint Surg Br. 2008;90:934–939.
17. Shim JS, Noh KC, Park SJ. Treatment of congenital muscu-
lar torticollis in patients older than 8 years. J Pediatr Orthop.
2004;24:683–688.
76e
Copyright © 2015 American Society of Plastic Surgeons. Unauthorized reproduction of this article is prohibited.
Plastic and Reconstructive Surgery • July 2015
18. Cochrane Effective Practice and Organisation of Care Group.
Suggested risk of bias criteria for EPOC reviews. Available at:
http://epoc.cochrane.org/epoc-specific-resources-review-
authors. Accessed September 15, 2014.
19. Duval S, Tweedie R. Trim and fill: A simple funnel-plot-based
method of testing and adjusting for publication bias in meta-
analysis. Biometrics 2000;56:455–463.
20. Brückner L, Dietrich L. Long-term results of the surgical
treatment of muscular torticollis (in German). Z Orthop Ihre
Grenzgeb. 1990;128:652–656.
21. Burstein FD. Long-term experience with endoscopic surgi-
cal treatment for congenital muscular torticollis in infants
and children: A review of 85 cases. Plast Reconstr Surg.
2004;114:491–493.
22. Cai B, Chen BC, Wang ZG, et al. Surgical correction of con-
genital muscular torticollis in children over 4 years old with
bipolar release. Chin J Clin Rehabil. 2004;8:436–437.
23. Chang PY, Tan CK, Huang YF, et al. Torticollis: A long-term
follow-up study. Zhonghua Min Guo Xiao Er Ke Yi Xue Hui Za
Zhi 1996;37:173–177.
24. Daentzer D, Stüder D, Wirth CJ. Muscular torticollis (in
German). Oper Orthop Traumatol. 2010;22:177–187.
25. Dennhardt D, Daum R. Muscular torticollis: Results of
follow-up examinations after tenotomy (in German). Arch
Orthop Unfallchir. 1970;67:367–371.
26. Dong X, Yu D. Application of cicatricial contracture release
principles in muscular torticollis treatment. Aesthetic Plast
Surg. 2013;37:950–955.
27. Ferkel RD, Westin GW, Dawson EG, Oppenheim WL.
Muscular torticollis: A modified surgical approach. J Bone
Joint Surg Am. 1983;65:894–900.
28. Kraft A, Lack W, Lukeschitsch G. Subcutaneous tenotomy of
the sternocleidomastoid muscle as therapy in muscular torti-
collis (in German). Z Orthop Ihre Grenzgeb. 1985;123:35–37.
29. Öhman AM, Perbeck Klackenberg EB, Beckung ERE,
Haglund-Åkerlind Y. Functional and cosmetic status after
surgery in congenital muscular torticollis. Adv Physiother.
2006;8:182–187.
30. Lee IJ, Lim SY, Song HS, Park MC. Complete tight fibrous
band release and resection in congenital muscular torticol-
lis. J Plast Reconstr Aesthet Surg. 2010;63:947–953.
31. Park MC, Song HS, Kim CS, et al. Treatment of congenital
muscular torticollis with unipolar release. J Korean Soc Plast
Reconstr Surg. 2009;36:38–45.
32. Babu MK, Lee P, Mahadev A, Lee EH. Congenital bilateral
sternocleidomastoid contracture: A case report. J Pediatr
Orthop B 2009;18:145–147.
33. Brans J, Aramideh M, Bosch A, Speelman H. Late presenta-
tion of congenital muscular torticollis: A non-dystonic cause
of torticollis. J Neurol. 1996;243:354–356.
34. Chang SC, Cheng HT, Lin FY, Wu CI. Aesthetic correc-
tion of adult unoperated torticollis. J Craniofac Surg.
2010;21:913–915.
35. Chang SH, Ohtori S, Okawa A, et al. A surgical treat-
ment for adult muscular torticollis. Case Rep Orthop.
2013;2013:965693.
36. Chate RA. Facial scoliosis from sternocleidomastoid torticol-
lis: Long-term postoperative evaluation. Br J Oral Maxillofac
Surg. 2005;43:428–434.
37. Jones CD, Nakhdjevani A, Lidder S. Surgical management of
idiopathic torticollis secondary to a fibrotic band. Orthop Rev
(Pavia) 2012;4:e27.
38. Mohan M, Bhat S, Prasad R, Sharma SM, Jain TL. Congenital
muscular torticollis: Case report and an effective treatment
plan. J Maxillofac Oral Surg. 2012;11:364–367.
 Volume 136, Number 1 • Neglected Congenital Muscular Torticollis
39. Pombo Castro M, Luaces Rey R, Vázquez Mahía I, López-
Cedrún Cembranos JL. Congenital muscular torticollis in
adult patients: Literature review and a case report using a
harmonic scalpel. J Oral Maxillofac Surg. 2014;72:396–401.
40. Ippolito E, Tudisco C. Idiopathic muscular torticollis in
adults: Results of open sternocleidomastoid tenotomy. Arch
Orthop Trauma Surg. 1986;105:49–54.
41. Meshkini A, Pezeshkan R. Neglected congenital muscular
torticollis: A series of 5 patients treated with surgical divi-
sion of both sternal and clavicular head of SCM. Mov Disord.
2012;27(Suppl 1):S322.
42. Seyhan N, Jasharllari L, Keskin M, Savacı N. Efficacy of
bipolar release in neglected congenital muscular torticollis
patients. Musculoskelet Surg. 2012;96:55–57.
43. Balkany TJ, Mischke RE. Mastoid release for congenital torti-
collis. Laryngoscope 1980;90:337–338.
44. Patwardhan S, Shyam AK, Sancheti P, Arora P, Nagda T,
Naik P. Adult presentation of congenital muscular torticol-
lis: A series of 12 patients treated with a bipolar release of
sternocleidomastoid and Z-lengthening. J Bone Joint Surg Br.
2011;93:828–832.
45. Sudesh P, Bali K, Mootha AK, Dhillon MS. Results of
bipolar release in the treatment of congenital muscular
Copyright © 2015 American Society of Plastic Surgeons. Unauthorized reproduction of this article is prohibited.
torticollis in patients older than 10 years of age. J Child Orthop.
2010;4:227–232.
46. Omidi-Kashani F, Hasankhani EG, Sharifi R, Mazlumi M. Is
surgery recommended in adults with neglected congenital
muscular torticollis? A prospective study. BMC Musculoskelet
Disord. 2008;9:158.
47. Arslan H, Gündüz S, Subaşi M, Kesemenli C, Necmioğlu S,
Subaşý M. Frontal cephalometric analysis in the evaluation
of facial asymmetry in torticollis, and outcomes of bipolar
release in patients over 6 years of age. Arch Orthop Trauma
Surg. 2002;122:489–493.
48. Chen CE, Ko JY. Surgical treatment of muscular torticollis
for patients above 6 years of age. Arch Orthop Trauma Surg.
2000;120:149–151.
49. Akazawa H, Nakatsuka Y, Miyake Y, Takahashi Y. Congenital
muscular torticollis: Long-term follow-up of thirty-eight
partial resections of the sternocleidomastoid muscle. Arch
Orthop Trauma Surg. 1993;112:205–209.
50. Itoi E, Funayama K, Suzuki T, Kamio K, Sakurai M. Tenotomy
and postoperative brace treatment for muscular torticollis.
Contemp Orthop. 1990;20:515–523.
51. Tse P, Cheng J, Chow Y, Leung PC. Surgery for neglected
congenital torticollis. Acta Orthop Scand. 1987;58:270–272.
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