Craniofacial Deformity in Patients with Uncorrected ......Skull and Cranial Base Dysmorphology In...

Craniofacial Deformity in Patients withUncorrected Congenital Muscular Torticollis:An Assessment from Three-DimensionalComputed Tomography ImagingChung-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 idiopathicfibrosis of the sternocleidomastoid muscle that restrictsmovement and pulls the head toward the involved side.Deformation of the craniofacial skeleton will develop ifthe restriction is not released and result in aesthetic andfunctional problems. The purpose of this study was to usethree-dimensional computed tomography imaging forqualitative and quantitative evaluation of the craniofacialdeformity 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 14patients from 1 month to 24 years of age were included.The skull images were rotated into standard orientationand reconfigured for evaluation of the cranium, endocra-nial base, and facial skeletal structures. The midlines ofcranial base and facial bone, angle of midline deviation,width of each hemicranium and hemiface, and the orbitalindex were defined and measured. The results showedthat the cranium and cranial base deformation took placeas early as in infant stage, with the most prominent changeoccurring in the posterior cranial fossa. Facial bone asym-metry started to appear after 5 years of age, at which timethe mandibular and occlusal abnormalities were observed.The deformity of the orbits and maxilla occurred at anolder age, characterized by the deviation and decreasedvertical height on the affected side. The severity of theobserved deformities increased with age. The angle ofmidline deviation was 2.48 � 1.68 degrees in the cranialbase and 3.26 � 3.28 degrees on the facial bone. Both ofthe midline deviations were significantly correlated withage. Compared with the contralateral side, the width ofthe ipsilateral posterior hemicranium was longer (54.36 �6.72 mm versus 50.81 � 6.55 mm), and the width of theipsilateral lower hemiface was shorter (35.30 � 7.27 mmversus 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 thatthe cranium and cranial base deformity occurred early in

patients with uncorrected torticollis, while the facial bonedeformity occurred in childhood stage. The cranial andfacial 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 towardthe affected side with a fibrotic and shortenedsternocleidomastoid muscle. Skull and facialasymmetry or plagiocephaly may occur in thepresence of prolonged uncorrected head tilt(Fig. 1). The characteristic appearance associ-ated with torticollis includes recessed eyebrowand zygoma, deviation of the chin point andnasal tip, inferior orbital dystopia on the af-fected side, commissural canting toward theaffected side, inferiorly and posteriorly posi-tioned ipsilateral ear, and distorted craniofa-cial skeletal structures.1–4 Comprehensive eval-uation and quantitative measurement of thecraniofacial skeletal deformity are difficult us-ing conventional methods such as cephalome-try because of the complexity in identifyinganatomical landmarks and overlapping struc-tures in two-dimensional medical imaging.3 Inthis study, three-dimensional computed to-mography imaging was applied for qualitativeand quantitative morphological assessment ofthe craniofacial osseous structures, including

From the Department of Plastic and Reconstructive Surgery, Chang Gung Memorial Hospital, and the Institute of Public Health, NationalYang Ming University. Received for publication December 30, 2002; revised March 17, 2003.

DOI: 10.1097/01.PRS.0000096703.91122.69

24

the endocranial base, in a group of patientswith uncorrected torticollis. The extent of thecraniofacial deformation and the effect of ageupon the magnitude of the deformity wereevaluated.

PATIENTS AND METHODS

Selection of the Patients

Patients with untreated congenital musculartorticollis 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. Carefulhistory and physical examination were per-formed. Presence of a shortened and fibroticsternocleidomastoid muscle was confirmed. In-formed consent was obtained for computedtomography scans to evaluate the craniofacialdeformity. Patients with craniofacial synostosisor previous history of facial trauma were ex-cluded from this study.

Computed Tomography Data Acquisition andProcessing

Spiral computed tomography scans wereperformed according to a standard craniofa-cial protocol.5 An oral sedative was given if thepatient was too young to cooperate during theprocedure. The computed tomography datawere reconstructed to create continuous slicesat 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, MayoFoundation, Rochester, Minn.) was used toprocess the data and reconstruct three-dimensional images.6–8 The size of the voxel inthe reformatted images was set at 0.6 � 0.6 �0.6 mm for all scans. For convenience in ob-servation and measurement, the torticollis sidewas set to the right side; i. e., for those with lefttorticollis, the images were reformatted by flip-ping the x coordinate. Patients’ heads werescanned with heads tilted in their resting state.

FIG. 1. A 24-year old female patient with left torticollis. Three-dimensional computedtomography images demonstrate dysmorphology of the soft tissue (rendered transpar-ent) and the underlying skeletal structures.

Vol. 113, No. 1 / CRANIOFACIAL DEFORMITY IN TORTICOLLIS 25

To facilitate assessment, measurement, andcomparisons, the heads were rotated into astandard 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 fromboth lateral views, the nasion landmark in themedian sagittal plane, and the inferior orbitalrims 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 standardorientation, using the Analyze matrix transfor-mation function. Grayscale thresholding wasused to remove soft tissue, leaving the osseousstructures for evaluation.

Evaluation and Measurement

After standard orientation, evaluation andmeasurement were performed on three-dimensional volume-rendered osseous images.The skull was rotated for inspection, landmarkidentification, definition of midlines, and mea-surement. The calvarial vault was clipped forassessment of the endocranial base from thestandard top view (Fig. 2, right).

The cranium was observed for shape and

symmetry. From the skull base view, the cristagalli, 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, andthe midline of the posterior cranial fossa wasdefined by the opisthion and basion points.9The angle formed by intersection of these twomidlines was defined as the cranial base mid-line deviation. For a symmetrical skull base, themidline 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, apoint LWO was defined at the junction of acurve continuous from the ridge of lesser wingof sphenoid bone to the inner surface of pari-etal bone, and another point PPO was definedat the junction of the superior ridge of petrousprocess to the inner surface of the occipitalbone.10,11 The distance from point LWO to themidline of anterior cranial fossa was defined asthe width of anterior hemicranium. The dis-tance from point PPO to the midline of poste-rior cranial fossa was defined as the width ofposterior hemicranium. The widths were mea-

FIG. 2. Reorientation of the three-dimensional skull image for standard evaluation and measurement. Thelandmark 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 lesserwing of sphenoid bone to the inner surface of parietal bone; PPO, point at the junction of the superior ridge ofpetrous process to the inner surface of the occipital bone.

26 PLASTIC AND RECONSTRUCTIVE SURGERY, January 2004

sured using a two-dimensional distance tech-nique from the standard cranial base view andcompared using paired t test between the ipsi-lateral and contralateral sides.

Morphological assessment was also per-formed for the shape and symmetry of thefacial skeletal structures (Fig. 2, left). Land-marks were identified at nasion, anterior nasalspine, lowest point of mandible symphysis, wid-est point of zygomatic arch, and gonion (Fig. 2,left). The middle facial midline was defined bya line connecting nasion and anterior nasalspine points. The lower facial midline was de-fined by a line connecting anterior nasal spineand mandible symphysis points. The angleformed by the intersection of these two lineswas defined as facial midline deviation. For asymmetric face, the midlines should be verticalwith a deviation angle of 0 degrees. The rela-tionship of age to facial midline deviation an-gle was tested by regression analysis. The widthof the anterior hemiface was defined. The dis-tance from the zygomatic arch to the middlefacial midline was defined as the width of mid-dle hemiface. The distance from the gonion tothe midline of the lower facial midline wasdefined as the width of lower hemiface. Thewidths were measured by two-dimensional dis-tance on standard frontal view and comparedusing a paired t test. The height and width ofthe orbit were likewise measured, and the or-bital index was calculated (100 � height/width oforbit).12 The difference between both sides wasalso compared by paired t test.

In the study, the authors carefully evaluatedthe three-dimensional osseous images of allpatients for the dysmorphology and landmarkidentification. A single investigator (Yu) who isa craniofacial surgeon identified the land-marks and took the measurements. The land-marks were identified and marked before thethree-dimensional osseous image was rotatedto standard frontal or cranial basal view formeasurement. A second test revealed that thedifferences were within 5 percent.

RESULTS

A total of 15 patients with uncorrected con-genital muscular torticollis underwent three-dimensional computed tomography scanning.There was no craniosynostosis in this series.One patient was excluded due to previous zy-goma fracture, leaving 14 patients for thestudy. There were six male patients and eightfemale patients. The lesion was on the right

side in eight patients and on the left side in sixpatients. The patients ranged in age from 39days to 24 years (Table I). Thoracolumbarspine scoliosis was noted in one patient (caseno. 14).

Skull and Cranial Base Dysmorphology

In this group of patients, the cranial sutureswere normal in appearance and timing of clo-sure (Figs. 3 and 4). There was deformationalplagiocephaly with posterior displacement ofthe frontal and occipital bones on the ipsilat-eral side. Asymmetry of the skull base could beobserved in patients as early as 1 month of age(case 1). The deformity was more apparent onthe posterior cranial fossa. The ipsilateral pos-terior cranial fossa was wider and longer on thecranial base view. The petrous process of tem-poral bone was longer on the lesion side, andthe ipsilateral middle cranial fossa appearedmore distorted correspondingly. In older pa-tients, the lateral boundary of the middle cra-nial fossa protruded less on the lesion side.The anterior cranial fossa was relatively sym-metrical bilaterally. The deformity in the ante-rior cranial fossa was not apparent in youngpatients but was observed in older patients.The deformity was characterized by ipsilateralrecession and contralateral protrusion of theanterior boundary, creating shorter anteropos-terior dimension on the ipsilateral anterior cra-nial fossa. The extent of deformation on theanterior cranial fossa was less prominent thanthat on the posterior cranial fossa. The overallskull and cranial base deformities were moreapparent in older patients.

TABLE IPatients with Untreated Congenital Muscular Torticollis

Patient Sex Age at CT ScanningSide of

Torticollis

1 Female 1 month Right2 Female 2 months Left3 Female 5 months Left4 Female 7 months Right5 Male 8 months Right6 Female 2 years, 10 months Right7 Male 5 years, 1 months Left8 Female 9 years Right9 Female 10 years, 8 months Left

10 Male 10 years, 10 months Right11 Male 16 years, 9 months Right12 Male 19 years, 7 months Left13 Male 19 years, 8 months Right14 Female 24 years, 7 months Left

CT, computed tomography.


FIG. 4. Standard orientation of the frontal views of entire patient population. The numbers indicatethe patients listed in Table I.

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


Facial Deformity

Gross facial deformity was not observed inpatients until age 5 years (Figs. 5 to 8), at whichtime jaw dysmorphology and occlusal tiltingbecame apparent and worsened with age. Theipsilateral orbit appeared slightly smaller withdecreased vertical height in patients 19 years ofage and older (Fig. 8). The contralateral orbitappeared round in shape. The maxilla wasretruded and vertically short on the lesion side.In relation to the level of the two orbits, the

dental occlusal plane was tilted due to con-stricted growth of the ipsilateral facial bones.Mandibular asymmetry was most consistentlypresent and the most significant of all the facialbones, with the chin point shifting toward thelesion side. The ipsilateral hemimandible wasretrusive. The facial deformities were moreprominent in older patients.

Quantitative Measurement and Comparisons

The angle of cranial base midline deviationwas 2.48 � 1.68 degrees (range, 0.62 to 6.04degrees). The angle showed a close correlationwith 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 thelesion 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 ahigh correlation was obtained. The regressionline was Y � 0.0243��0.73, r � 0.765 (Fig. 9,below, p � 0.001). Comparing the width of thehemiface between the two sides using thepairedt test showed no difference on the middlehemiface but significantly narrower lowerhemiface on the lesion side (35.30 � 1.94 mmversus 43.49 � 3.03 mm; Table II, p � 0.016).

The orbital index was significantly smalleron the ipsilateral than on the contralateral or-

FIG. 5. Case 6. Female patient at 2 years of age with right-sided torticollis.

FIG. 6. Case 7. Male patient at 5 years of age with left-sided torticollis.


bits (Table II, p � 0.025), indicating a flatterorbital shape on the side with the restrictingsternocleidomastoid muscle.

DISCUSSION

Three-dimensional computed tomographyimaging is an accurate method for comprehen-sive in vivo assessment and measurement of the

craniofacial structures.9,12–14 Such evaluation isinformative and may affect treatment strategy.When considering a group of patients with thehead 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. 7. Case 11. Male patient at 16 years of age with right-sided torticollis.

FIG. 8. Case 12. Male patient at 19 years of age with left-sided 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 theChinese population, with male dominance.16

Patients usually come to the clinic with com-plaints of restricted neck mobility and headtilting. Clinical evaluation should be carefullyperformed 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. Thepathology is confirmed by physical examina-tion and sonography. The exact pathogenesisis not clear but could be associated with breechpresentation, birth trauma, or forceps use dur-ing delivery. Treatment for congenital muscu-lar torticollis includes stretching exercises for

release of the muscle and a helmet orthosis forabnormal head shape.15,18 Surgical division orresection of the affected sternocleidomastoidmuscle is considered only if rehabilitative man-agement fails. Careful monitoring and aggres-sive treatment are necessary to prevent skulland facial deformity.19,20

The deformities observed in this study in-volved both the skull contour and cranial baseand could be observed as early as the neonatalstage. The phenotypic clinical presentation ofsynostotic plagiocephaly is not always clearlydifferent from that of deformational plagio-cephaly.21 Differentiation can be achieved withthe help of computed tomography images.9Unicoronal or unilambdoid synostosis pro-duces overt localized distortion and increasedcranial base midline deviation angle. By con-trast, patients in this study had a rhomboidcranial shape and a smaller midline deviationangle. The calvarial and endocranial base mor-phologies are similar in deformational plagio-cephaly (i.e., nonsynostotic plagiocephaly) forpatients with and without associated torticollis.Deformational plagiocephaly may be caused byfetal 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 thanthe cranium/cranial base deformity. The facialdeformity in torticollis has been reported toinclude deviation and flattening of the face onthe affected side with recessed eyebrow andzygoma, inferior displacement of the orbit and

FIG. 9. (Above) The cranial base midline deviation angleand correlation with patient age. A significant correlation wasfound with a regression line: Y � 0.0129��1.29, r � 0.696,and p � 0.006. (Below) The facial midline deviation angle andcorrelation with patient age. The correlation was significantwith a regression line: Y � 0.0243��0.73, r � 0.765, and p �0.001.

TABLE IIMeasurement of the Width of Hemicranium and Hemifaceand Comparisons between the Ipsilateral and Contralateral

Sides*

Mean p

Width of the anterior hemicraniumIpsilateral 51.08 � 5.29 0.180Contralateral 49.25 � 6.42

Width of the posterior hemicraniumIpsilateral 54.36 � 6.72 0.05Contralateral 50.81 � 6.55

Width of the middle hemifaceIpsilateral 53.63 � 11.09 0.712Contralateral 54.07 � 12.05

Width of the lower hemifaceIpsilateral 35.30 � 7.27 0.016Contralateral 43.49 � 11.34

Orbital indexIpsilateral 89.48 � 0.11 0.025Contralateral 92.74 � 0.08

* The p value was determined from the paired t test; p � 0.05 indicates asignificant difference. The mean width is in millimeters, and the orbital indexis a ratio.


ear on the affected side, and deviation of thechin point and nasal tip.1–4 Ferguson describedit as “subcranial torsional rotation of face to-ward the affected site.”2,3 However, after stan-dard reorientation of the three-dimensionalimages and careful observation (Fig. 4), ourstudy did not demonstrate gross facial asymme-try and deformity until 5 years of age. Thelower face deformity (mandible) appearedfirst, followed by the maxilla, and finally theorbits. In older patients, the restriction of theipsilateral facial bone development was obvi-ous, with decreased maxilla and orbit heightsas well as significant narrowing of the lowerhemiface on the lesion side. The restriction ofthe ipsilateral facial bone development waslikely caused by limited mobility from the fi-brotic and shortened sternocleidomastoidmuscle. The contralateral deformity was influ-enced by a compensatory growth, creating acurvilinear facial appearance from the frontalview. The dental occlusal plane was canted dueto the asymmetric development of the facialbones. The occlusal plane and orbital planewere not parallel (Fig. 4), which came closeron the affected side of the face.

A treatment plan for patients with congenitalmuscular torticollis should include early phys-ical therapy with stretching of the restrictingmuscle, molding helmet therapy for the plagio-cephaly, and surgical release of the involvedmuscle if rehabilitation fails.15,16,18,20 Early ag-gressive management to free the head tilt isrecommended to terminate the deformationalprocess 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 difficultto determine. Wolfort et al.27 recommendedthat the operation be delayed until the patientis 1 year old, but it should probably be com-pleted before the patient is of school age. How-ever, reversal of the craniofacial asymmetryshould best be achieved at an earlier age, whenthere is high growth and remodeling potential.Cheng and Au16 reviewed a large group ofpatients with infantile torticollis and foundthat 97 percent of all infantile torticollis casesresolved with conservative treatment, activestimulation, and a passive stretching program,and a mean treatment period of less than 6months was needed for those patients who re-

sponded to treatment. They also observed thatpatients with cord-like muscular torticollis andsevere head rotation were more likely to needan operation. Orthotic and nonorthotic treat-ment has been reported to be effective in pa-tients younger than 1 year of age with thecranial deformity.15,18,22,28,29 On the basis ofthese reasons, we recommend early physicaltherapy and orthotic treatment for torticollisinfants and consider surgical release of therestricting 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 torestore free neck movement as early as possibleto stop and reverse the skull base deformityand to prevent facial asymmetry. For patientswith established craniofacial deformities, surgi-cal corrections, which may include craniotomy,orbital osteotomy, and orthognathic surgery,are complicated and cumbersome and haveless predictable results.1–3,22,23,26,30

CONCLUSIONS

This study examined the craniofacial defor-mity in a series of 14 patients with untreatedcongenital muscular torticollis. The resultsshowed that the deformity appeared on thecranium and cranial base at an early period oflife, and the facial deformity occurred at a laterstage. Although this was not a longitudinalstudy, it was found that the severity of cranialand facial deformity was correlated with age.The correlation was also demonstrated frommeasurement of endocranial and facial mid-line deviation angles. Early correction of torti-collis is mandatory, and if conservative man-agement fails, we recommend that surgicalrelease be performed before 1 year of age toprevent and reverse the craniofacial deformity.

Lun-Jou Lo, M.D.Department of Plastic and Reconstructive SurgeryChang Gung Memorial Hospital199 Tun Hwa North RoadTaipei, Taiwan [email protected]

ACKNOWLEDGMENTS

The study was supported by a grant from the NationalScience Council, NSC 90-2314-B-182A-142. The authorsthank Richard A. Robb, Ph.D., Biomedical Imaging Resource,Mayo Foundation, Rochester, Minnesota, for collaborationand for providing the Analyze program; Dr. Alex A. Kane formanuscript revision and comment; and Miss Meng-Chen Wufor technical support in the medical imaging laboratory.


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