Craniofacial Deformity in Patients with
Uncorrected Congenital Muscular Torticollis:
An Assessment from Three-Dimensional
Computed Tomography Imaging
Chung-Chih Yu, M.D., Fen-Hwa Wong, Ph.D., Lun-Jou Lo, M.D., and Yu-Ray Chen, M.D.
Taipei, Taiwan
Congenital muscular torticollis is caused by idiopathic
fibrosis of the sternocleidomastoid muscle that restricts
movement and pulls the head toward the involved side.
Deformation of the craniofacial skeleton will develop if
the restriction is not released and result in aesthetic and
functional problems. The purpose of this study was to use
three-dimensional computed tomography imaging for
qualitative and quantitative evaluation of the craniofacial
deformity in a series of patients with uncorrected con-
genital muscular torticollis, and to assess age as a precip-
itating factor for severity of the deformity. A total of 14
patients from 1 month to 24 years of age were included.
The skull images were rotated into standard orientation
and reconfigured for evaluation of the cranium, endocra-
nial base, and facial skeletal structures. The midlines of
cranial base and facial bone, angle of midline deviation,
width of each hemicranium and hemiface, and the orbital
index were defined and measured. The results showed
that the cranium and cranial base deformation took place
as early as in infant stage, with the most prominent change
occurring in the posterior cranial fossa. Facial bone asym-
metry started to appear after 5 years of age, at which time
the mandibular and occlusal abnormalities were observed.
The deformity of the orbits and maxilla occurred at an
older age, characterized by the deviation and decreased
vertical height on the affected side. The severity of the
observed deformities increased with age. The angle of
midline deviation was 2.48 ⫾ 1.68 degrees in the cranial
base and 3.26 ⫾ 3.28 degrees on the facial bone. Both of
the midline deviations were significantly correlated with
age. Compared with the contralateral side, the width of
the ipsilateral posterior hemicranium was longer (54.36 ⫾
6.72 mm versus 50.81 ⫾ 6.55 mm), and the width of the
ipsilateral lower hemiface was shorter (35.30 ⫾ 7.27 mm
versus 43.49 ⫾ 11.34 mm). Both differences were statis-
tically significant. Measurement of the orbital index dem-
onstrated a significantly flatter orbit on the ipsilateral side
(89.48 ⫾ 0.11 versus 92.74 ⫾ 0.08). This study showed that
the cranium and cranial base deformity occurred early in
From the Department of Plastic and Reconstructive Surgery, Chang Gung Memorial Hospital, and the Institute of Public Health, National
Yang Ming University. Received for publication December 30, 2002; revised March 17, 2003.
DOI: 10.1097/01.PRS.0000096703.91122.69
24
patients with uncorrected torticollis, while the facial bone
deformity occurred in childhood stage. The cranial and
facial deformity became more severe with age. Early re-
lease of the muscle restriction is advised to prevent cranio-
facial deformation. (Plast. Reconstr. Surg. 113: 24, 2004.)
Torticollis, a twisting of the neck to one side,
is one of the most common congenital anom-
alies. Patients with congenital muscular torti-
collis present with tilting of the head toward
the affected side with a fibrotic and shortened
sternocleidomastoid muscle. Skull and facial
asymmetry or plagiocephaly may occur in the
presence of prolonged uncorrected head tilt
(Fig. 1). The characteristic appearance associ-
ated with torticollis includes recessed eyebrow
and zygoma, deviation of the chin point and
nasal tip, inferior orbital dystopia on the af-
fected side, commissural canting toward the
affected side, inferiorly and posteriorly posi-
tioned ipsilateral ear, and distorted craniofa-
cial skeletal structures.1– 4 Comprehensive eval-
uation and quantitative measurement of the
craniofacial skeletal deformity are difficult us-
ing conventional methods such as cephalome-
try because of the complexity in identifying
anatomical landmarks and overlapping struc-
tures in two-dimensional medical imaging.3 In
this study, three-dimensional computed to-
mography imaging was applied for qualitative
and quantitative morphological assessment of
the craniofacial osseous structures, including
Vol. 113, No. 1 / CRANIOFACIAL DEFORMITY IN TORTICOLLIS
FIG. 1. A 24-year old female patient with left torticollis. Three-dimensional computed
tomography images demonstrate dysmorphology of the soft tissue (rendered transpar-
ent) and the underlying skeletal structures.
the endocranial base, in a group of patients
with uncorrected torticollis. The extent of the
craniofacial deformation and the effect of age
upon the magnitude of the deformity were
evaluated.
PATIENTS AND METHODS
Selection of the Patients
Patients with untreated congenital muscular
torticollis who presented to the Plastic and Re-
constructive Surgery Clinic at Chang Gung Me-
morial Hospital for evaluation and manage-
ment were included in the study. Careful
history and physical examination were per-
formed. Presence of a shortened and fibrotic
sternocleidomastoid muscle was confirmed. In-
formed consent was obtained for computed
tomography scans to evaluate the craniofacial
deformity. Patients with craniofacial synostosis
or previous history of facial trauma were ex-
cluded from this study.
25
Computed Tomography Data Acquisition and
Processing
Spiral computed tomography scans were
performed according to a standard craniofa-
cial protocol.5 An oral sedative was given if the
patient was too young to cooperate during the
procedure. The computed tomography data
were reconstructed to create continuous slices
at 1.5 mm thickness and transferred to a med-
ical imaging laboratory. A Windows 2000 per-
sonal computer running Analyze software (ver-
sion 4.0; Biomedical Imaging Resource, Mayo
Foundation, Rochester, Minn.) was used to
process the data and reconstruct three-
dimensional images.6 – 8 The size of the voxel in
the reformatted images was set at 0.6 ⫻ 0.6 ⫻
0.6 mm for all scans. For convenience in ob-
servation and measurement, the torticollis side
was set to the right side; i. e., for those with left
torticollis, the images were reformatted by flip-
ping the x coordinate. Patients’ heads were
scanned with heads tilted in their resting state.
26
To facilitate assessment, measurement, and
comparisons, the heads were rotated into a
standard orientation using the Frankfurt hori-
zontal and bilateral orbital landmarks (Fig. 2,
left).5 The image rotation was carefully per-
formed to set the Frankfurt horizontal from
both lateral views, the nasion landmark in the
median sagittal plane, and the inferior orbital
rims at the same horizontal level from the an-
terior view. When the best position was ob-
tained, the data were then reformatted, re-
aligning the x, y, and z axes to this standard
orientation, using the Analyze matrix transfor-
mation function. Grayscale thresholding was
used to remove soft tissue, leaving the osseous
structures for evaluation.
Evaluation and Measurement
After standard orientation, evaluation and
measurement were performed on three-
dimensional volume-rendered osseous images.
The skull was rotated for inspection, landmark
identification, definition of midlines, and mea-
surement. The calvarial vault was clipped for
assessment of the endocranial base from the
standard top view (Fig. 2, right).
The cranium was observed for shape and
PLASTIC AND RECONSTRUCTIVE SURGERY, January 2004
symmetry. From the skull base view, the crista
galli, foramen magnum, lesser wing of sphe-
noid, petrous process, and the anterior, mid-
dle, and posterior cranial fossae were observed
(Fig. 2, right). The midline of the anterior cra-
nial fossa was defined by the crista galli, and
the midline of the posterior cranial fossa was
defined by the opisthion and basion points.9
The angle formed by intersection of these two
midlines was defined as the cranial base mid-
line deviation. For a symmetrical skull base, the
midline deviation angle should be 0 degrees.
The relationship between the midline devia-
tion angle and patient’s age was tested by re-
gression analysis. From the endocranial view, a
point LWO was defined at the junction of a
curve continuous from the ridge of lesser wing
of sphenoid bone to the inner surface of pari-
etal bone, and another point PPO was defined
at the junction of the superior ridge of petrous
process to the inner surface of the occipital
bone.10,11 The distance from point LWO to the
midline of anterior cranial fossa was defined as
the width of anterior hemicranium. The dis-
tance from point PPO to the midline of poste-
rior cranial fossa was defined as the width of
posterior hemicranium. The widths were mea-

FIG. 2. Reorientation of the three-dimensional skull image for standard evaluation and measurement. The
landmark points, midlines, and the widths were marked as described in the text. (Left) Frontal view and (right)
endocranial base view. Za, zygomatic arch; Go, gonion; N, nasion; ANS, anterior nasal spine; Ms, mandible symphysis;
Cg, crista galli; Ba, basion; Op, opisthion; LWO, point at the junction of a curve continuous from the ridge of lesser
wing of sphenoid bone to the inner surface of parietal bone; PPO, point at the junction of the superior ridge of
petrous process to the inner surface of the occipital bone.
Vol. 113, No. 1 / CRANIOFACIAL DEFORMITY IN TORTICOLLIS 27
sured using a two-dimensional distance tech- side in eight patients and on the left side in six
nique from the standard cranial base view and patients. The patients ranged in age from 39
compared using paired t test between the ipsi- days to 24 years (Table I). Thoracolumbar
lateral and contralateral sides. spine scoliosis was noted in one patient (case
Morphological assessment was also per- no. 14).
formed for the shape and symmetry of the
facial skeletal structures (Fig. 2, left). Land- Skull and Cranial Base Dysmorphology
marks were identified at nasion, anterior nasal
spine, lowest point of mandible symphysis, wid- In this group of patients, the cranial sutures
est point of zygomatic arch, and gonion (Fig. 2, were normal in appearance and timing of clo-
left). The middle facial midline was defined by sure (Figs. 3 and 4). There was deformational
a line connecting nasion and anterior nasal plagiocephaly with posterior displacement of
spine points. The lower facial midline was de- the frontal and occipital bones on the ipsilat-
fined by a line connecting anterior nasal spine eral side. Asymmetry of the skull base could be
and mandible symphysis points. The angle observed in patients as early as 1 month of age
formed by the intersection of these two lines (case 1). The deformity was more apparent on
was defined as facial midline deviation. For a the posterior cranial fossa. The ipsilateral pos-
symmetric face, the midlines should be vertical terior cranial fossa was wider and longer on the
with a deviation angle of 0 degrees. The rela- cranial base view. The petrous process of tem-
tionship of age to facial midline deviation an- poral bone was longer on the lesion side, and
gle was tested by regression analysis. The width the ipsilateral middle cranial fossa appeared
of the anterior hemiface was defined. The dis- more distorted correspondingly. In older pa-
tance from the zygomatic arch to the middle tients, the lateral boundary of the middle cra-
facial midline was defined as the width of mid- nial fossa protruded less on the lesion side.
dle hemiface. The distance from the gonion to The anterior cranial fossa was relatively sym-
the midline of the lower facial midline was metrical bilaterally. The deformity in the ante-
defined as the width of lower hemiface. The rior cranial fossa was not apparent in young
widths were measured by two-dimensional dis- patients but was observed in older patients.
tance on standard frontal view and compared The deformity was characterized by ipsilateral
using a paired t test. The height and width of recession and contralateral protrusion of the
the orbit were likewise measured, and the or- anterior boundary, creating shorter anteropos-
bital index was calculated (100 ⫻ height/width of terior dimension on the ipsilateral anterior cra-
orbit).12 The difference between both sides was nial fossa. The extent of deformation on the
also compared by paired t test. anterior cranial fossa was less prominent than
In the study, the authors carefully evaluated that on the posterior cranial fossa. The overall
the three-dimensional osseous images of all skull and cranial base deformities were more
patients for the dysmorphology and landmark apparent in older patients.
identification. A single investigator (Yu) who is
a craniofacial surgeon identified the land- TABLE I
marks and took the measurements. The land- Patients with Untreated Congenital Muscular Torticollis
marks were identified and marked before the
three-dimensional osseous image was rotated
Side of
to standard frontal or cranial basal view for Patient Sex Age at CT Scanning Torticollis
measurement. A second test revealed that the 1 Female 1 month Right
differences were within 5 percent. 2 Female 2 months Left
3 Female 5 months Left
RESULTS 4 Female 7 months Right
5 Male 8 months Right
A total of 15 patients with uncorrected con- 6 Female 2 years, 10 months Right
7 Male 5 years, 1 months Left
genital muscular torticollis underwent three- 8 Female 9 years Right
dimensional computed tomography scanning. 9 Female 10 years, 8 months Left
There was no craniosynostosis in this series. 10 Male 10 years, 10 months Right
11 Male 16 years, 9 months Right
One patient was excluded due to previous zy- 12 Male 19 years, 7 months Left
goma fracture, leaving 14 patients for the 13 Male 19 years, 8 months Right
study. There were six male patients and eight 14 Female 24 years, 7 months Left
female patients. The lesion was on the right CT, computed tomography.
28 PLASTIC AND RECONSTRUCTIVE SURGERY, January 2004

FIG. 3. Standard orientation of the cranial bases of the entire patient population. The numbers indicate
the patients listed in Table I.

FIG. 4. Standard orientation of the frontal views of entire patient population. The numbers indicate
the patients listed in Table I.
Vol. 113, No. 1 / CRANIOFACIAL DEFORMITY IN TORTICOLLIS
FIG. 5. Case 6. Female patient at 2 years of age with right-
sided torticollis.
Facial Deformity
Gross facial deformity was not observed in
patients until age 5 years (Figs. 5 to 8), at which
time jaw dysmorphology and occlusal tilting
became apparent and worsened with age. The
ipsilateral orbit appeared slightly smaller with
decreased vertical height in patients 19 years of
age and older (Fig. 8). The contralateral orbit
appeared round in shape. The maxilla was
retruded and vertically short on the lesion side.
In relation to the level of the two orbits, the
FIG. 6. Case 7. Male patient at 5 years of age with left-sided torticollis.
29
dental occlusal plane was tilted due to con-
stricted growth of the ipsilateral facial bones.
Mandibular asymmetry was most consistently
present and the most significant of all the facial
bones, with the chin point shifting toward the
lesion side. The ipsilateral hemimandible was
retrusive. The facial deformities were more
prominent in older patients.
Quantitative Measurement and Comparisons
The angle of cranial base midline deviation
was 2.48 ⫾ 1.68 degrees (range, 0.62 to 6.04
degrees). The angle showed a close correlation
with age. The regression line was Y ⫽ 0.0129␹
⫹1.299, r ⫽ 0.696 (Fig. 9, above, p ⫽ 0.006).
The posterior hemicranium was wider on the
lesion side than on the contralateral side
(54.36 ⫾ 6.72 mm versus 50.81 ⫾ 6.55 mm;
Table II, p ⫽ 0.05).
The facial midline deviation angle was 3.26
⫾ 3.28 degrees (range, 0.00 to 9.14 degrees).
The angle was similarly tested with age, and a
high correlation was obtained. The regression
line was Y ⫽ 0.0243␹⫹0.73, r ⫽ 0.765 (Fig. 9,
below, p ⫽ 0.001). Comparing the width of the
hemiface between the two sides using the
paired
t test showed no difference on the middle
hemiface but significantly narrower lower
hemiface on the lesion side (35.30 ⫾ 1.94 mm
versus 43.49 ⫾ 3.03 mm; Table II, p ⫽ 0.016).
The orbital index was significantly smaller
on the ipsilateral than on the contralateral or-
30 PLASTIC AND RECONSTRUCTIVE SURGERY, January 2004

FIG. 7. Case 11. Male patient at 16 years of age with right-sided torticollis.

bits (Table II, p ⫽ 0.025), indicating a flatter
orbital shape on the side with the restricting
sternocleidomastoid muscle.
DISCUSSION
Three-dimensional computed tomography
imaging is an accurate method for comprehen-
sive in vivo assessment and measurement of the
craniofacial structures.9,12–14 Such evaluation is
informative and may affect treatment strategy.
When considering a group of patients with the
head tilt characteristic of this deformity, de-
scription and measurement are greatly facili-
tated by orienting the images in a similar fash-
ion before analysis.
Torticollis is a common congenital anomaly

FIG. 8. Case 12. Male patient at 19 years of age with left-sided torticollis.
Vol. 113, No. 1 / CRANIOFACIAL DEFORMITY IN TORTICOLLIS
with varied incidence, ranging from 0.3 per-
cent to 1.3 percent of new births.15,16 The inci-
dence has been reported to be higher in the
Chinese population, with male dominance.16
Patients usually come to the clinic with com-
plaints of restricted neck mobility and head
tilting. Clinical evaluation should be carefully
performed to rule out ophthalmologic or neu-
rological conditions, cervical spine problems,
muscular imbalance, and posterior fossa tu-
mor.4,16,17 A shortened and fibrotic sternoclei-
domastoid muscle is the most common clinico-
pathological finding in torticollis. The
pathology is confirmed by physical examina-
tion and sonography. The exact pathogenesis
is not clear but could be associated with breech
presentation, birth trauma, or forceps use dur-
ing delivery. Treatment for congenital muscu-
lar torticollis includes stretching exercises for
FIG. 9. (Above) The cranial base midline deviation angle
and correlation with patient age. A significant correlation was
found with a regression line: Y ⫽ 0.0129␹⫹1.29, r ⫽ 0.696,
and p ⫽ 0.006. (Below) The facial midline deviation angle and
correlation with patient age. The correlation was significant
with a regression line: Y ⫽ 0.0243␹⫹0.73, r ⫽ 0.765, and p ⫽
0.001.
31
TABLE II
Measurement of the Width of Hemicranium and Hemiface
and Comparisons between the Ipsilateral and Contralateral
Sides*
Mean p
Width of the anterior hemicranium
Ipsilateral 51.08 ⫾ 5.29 0.180
Contralateral 49.25 ⫾ 6.42
Width of the posterior hemicranium
Ipsilateral 54.36 ⫾ 6.72 0.05
Contralateral 50.81 ⫾ 6.55
Width of the middle hemiface
Ipsilateral 53.63 ⫾ 11.09 0.712
Contralateral 54.07 ⫾ 12.05
Width of the lower hemiface
Ipsilateral 35.30 ⫾ 7.27 0.016
Contralateral 43.49 ⫾ 11.34
Orbital index
Ipsilateral 89.48 ⫾ 0.11 0.025
Contralateral 92.74 ⫾ 0.08
* The p value was determined from the paired t test; p ⱕ 0.05 indicates a
significant difference. The mean width is in millimeters, and the orbital index
is a ratio.
release of the muscle and a helmet orthosis for
abnormal head shape.15,18 Surgical division or
resection of the affected sternocleidomastoid
muscle is considered only if rehabilitative man-
agement fails. Careful monitoring and aggres-
sive treatment are necessary to prevent skull
and facial deformity.19,20
The deformities observed in this study in-
volved both the skull contour and cranial base
and could be observed as early as the neonatal
stage. The phenotypic clinical presentation of
synostotic plagiocephaly is not always clearly
different from that of deformational plagio-
cephaly.21 Differentiation can be achieved with
the help of computed tomography images.9
Unicoronal or unilambdoid synostosis pro-
duces overt localized distortion and increased
cranial base midline deviation angle. By con-
trast, patients in this study had a rhomboid
cranial shape and a smaller midline deviation
angle. The calvarial and endocranial base mor-
phologies are similar in deformational plagio-
cephaly (i.e., nonsynostotic plagiocephaly) for
patients with and without associated torticollis.
Deformational plagiocephaly may be caused by
fetal intrauterine head constraint, static or un-
changing neonatal or infant head position, su-
pine sleep position, or torticollis.18,22–25
The facial deformity developed later than
the cranium/cranial base deformity. The facial
deformity in torticollis has been reported to
include deviation and flattening of the face on
the affected side with recessed eyebrow and
zygoma, inferior displacement of the orbit and
32
ear on the affected side, and deviation of the
chin point and nasal tip.1– 4 Ferguson described
it as “subcranial torsional rotation of face to-
ward the affected site.”2,3 However, after stan-
dard reorientation of the three-dimensional
images and careful observation (Fig. 4), our
study did not demonstrate gross facial asymme-
try and deformity until 5 years of age. The
lower face deformity (mandible) appeared
first, followed by the maxilla, and finally the
orbits. In older patients, the restriction of the
ipsilateral facial bone development was obvi-
ous, with decreased maxilla and orbit heights
as well as significant narrowing of the lower
hemiface on the lesion side. The restriction of
the ipsilateral facial bone development was
likely caused by limited mobility from the fi-
brotic and shortened sternocleidomastoid
muscle. The contralateral deformity was influ-
enced by a compensatory growth, creating a
curvilinear facial appearance from the frontal
view. The dental occlusal plane was canted due
to the asymmetric development of the facial
bones. The occlusal plane and orbital plane
were not parallel (Fig. 4), which came closer
on the affected side of the face.
A treatment plan for patients with congenital
muscular torticollis should include early phys-
ical therapy with stretching of the restricting
muscle, molding helmet therapy for the plagio-
cephaly, and surgical release of the involved
muscle if rehabilitation fails.15,16,18,20 Early ag-
gressive management to free the head tilt is
recommended to terminate the deformational
process and, it is hoped, to reverse the defor-
mity by neurocranial growth. Nonoperative in-
tervention has been reported to be highly suc-
cessful in patients younger than 1 year of age.27
Surgical intervention should be performed be-
fore the craniofacial deformity becomes signif-
icant and/or permanent, which can be difficult
to determine. Wolfort et al.27 recommended
that the operation be delayed until the patient
is 1 year old, but it should probably be com-
pleted before the patient is of school age. How-
ever, reversal of the craniofacial asymmetry
should best be achieved at an earlier age, when
there is high growth and remodeling potential.
Cheng and Au16 reviewed a large group of
patients with infantile torticollis and found
that 97 percent of all infantile torticollis cases
resolved with conservative treatment, active
stimulation, and a passive stretching program,
and a mean treatment period of less than 6
months was needed for those patients who re-
PLASTIC AND RECONSTRUCTIVE SURGERY, January 2004
sponded to treatment. They also observed that
patients with cord-like muscular torticollis and
severe head rotation were more likely to need
an operation. Orthotic and nonorthotic treat-
ment has been reported to be effective in pa-
tients younger than 1 year of age with the
cranial deformity.15,18,22,28,29 On the basis of
these reasons, we recommend early physical
therapy and orthotic treatment for torticollis
infants and consider surgical release of the
restricting muscle before 1 year of age for pa-
tients with poor response to conservative man-
agement, severe torticollis, or a prominent re-
stricting fibrotic band. The purpose is to
restore free neck movement as early as possible
to stop and reverse the skull base deformity
and to prevent facial asymmetry. For patients
with established craniofacial deformities, surgi-
cal corrections, which may include craniotomy,
orbital osteotomy, and orthognathic surgery,
are complicated and cumbersome and have
less predictable results.1–3,22,23,26,30
CONCLUSIONS
This study examined the craniofacial defor-
mity in a series of 14 patients with untreated
congenital muscular torticollis. The results
showed that the deformity appeared on the
cranium and cranial base at an early period of
life, and the facial deformity occurred at a later
stage. Although this was not a longitudinal
study, it was found that the severity of cranial
and facial deformity was correlated with age.
The correlation was also demonstrated from
measurement of endocranial and facial mid-
line deviation angles. Early correction of torti-
collis is mandatory, and if conservative man-
agement fails, we recommend that surgical
release be performed before 1 year of age to
prevent and reverse the craniofacial deformity.
Lun-Jou Lo, M.D.
Department of Plastic and Reconstructive Surgery
Chang Gung Memorial Hospital
199 Tun Hwa North Road
Taipei, Taiwan 105
lunjoulo@ms1.hinet.net
ACKNOWLEDGMENTS
The study was supported by a grant from the National
Science Council, NSC 90-2314-B-182A-142. The authors
thank Richard A. Robb, Ph.D., Biomedical Imaging Resource,
Mayo Foundation, Rochester, Minnesota, for collaboration
and for providing the Analyze program; Dr. Alex A. Kane for
manuscript revision and comment; and Miss Meng-Chen Wu
for technical support in the medical imaging laboratory.
Vol. 113, No. 1 / CRANIOFACIAL DEFORMITY IN TORTICOLLIS
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