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Helmet Treatment of Infants With Deformational Brachycephaly

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Deformation of the cranium in infancy represents a spectrum of deformity, ranging from severe asymmetric yet proportional distortion of the skull in plagiocephaly, to nearly symmetric yet disproportional distortion in brachycephaly. As such, the condition is best described as deformational plagiocephaly-brachycephaly with isolated plagiocephaly and/or isolated brachycephaly being at either ends of the spectrum. Due to its symmetric appearance, deformational brachycephaly is often incorrectly dismissed as being less concerning, and it has sometimes erroneously been reported that brachycephaly cannot be treated successfully with a cranial orthosis. We prospectively report on 4205 infants with isolated deformational brachycephaly treated with a cranial orthosis from 2013 to 2017. These results demonstrate that the orthosis is successful in the treatment of deformational brachycephaly with an 81.4% improvement toward normal (95.0 to 89.4) in cephalic index. We furthermore demonstrate that entrance age influences treatment results, with younger infants demonstrating both improved outcomes and shorter treatment times.
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https://doi.org/10.1177/2333794X18805618
Global Pediatric Health
Volume 5: 1 –11
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Original Article
Introduction
Brachycephaly is characterized by symmetric, bilateral
flattening of the occiput resulting in a head shape that
becomes disproportionately short and wide. While the
product of the same external forces that cause deforma-
tional plagiocephaly, deformational brachycephaly is
often dismissed as less urgent or significant.1 In particu-
lar, the lack of asymmetry often leads to the incorrect
assumption that brachycephaly is somehow more “cos-
metic” in nature. However, both plagiocephaly and
brachycephaly have been shown to deform the skull
base, affecting the position and orientation of the tem-
poromandibular joints, and affect occlusal function.2-7
Specifically, a brachycephalic deformation of the cranial
vault results in a posterior tipping of the mid cranial
fossa (central skull base) changing the angular orienta-
tion of the temporomandibular joints, and potentially
resulting in Class III malocclusion (underbite).8-13
Anterior displacement of the mandible may also affect
the soft tissue of the upper airway leading to airway
restrictions and obstructive sleep apnea.14-18
Moreover, when the back of the head is flattened, the
center of mass of the head is displaced anteriorly and
superiorly, which, in severe cases, may affect an infant’s
postural control and postural alignment.19,20 While the
muscular imbalance and restricted range of motion (ie,
torticollis, or lateral/rotational imbalance) frequently
associated with plagiocephaly is commonly discussed,
the muscular imbalance of brachycephaly (what we’ll
call AP imbalance) has largely gone unrecognized. As
the center of mass shifts, the anterior neck muscles
become shortened while the extensor neck muscles get
lengthened leading to an imbalance of the flexor/exten-
sor muscle groups. This imbalance leads to poor
805618GPHXXX10.1177/2333794X18805618Global Pediatric HealthKelly et al
research-article2018
1University of Iowa, Iowa City, IA, USA
2Barrow Cleft and Craniofacial Center, Phoenix, AZ, USA
3Southwest Craniofacial Center, Phoenix, AZ, USA
4Cranial Technologies, Tempe, AZ, USA
Corresponding Author:
Timothy R. Littlefield, Cranial Technologies, Inc, 1395 West Auto
Drive, Tempe, AZ 85248, USA.
Email: tlittlefield@cranialtech.com
Helmet Treatment of Infants With
Deformational Brachycephaly
Kevin M. Kelly, PhD1, Edward F. Joganic, MD, FACS2,
Stephen P. Beals, MD, FACS, FAAP3, Jeff A. Riggs, MA4,
Mary Kay McGuire, OTR/L4, and Timothy R. Littlefield, MS4
Abstract
Deformation of the cranium in infancy represents a spectrum of deformity, ranging from severe asymmetric
yet proportional distortion of the skull in plagiocephaly, to nearly symmetric yet disproportional distortion in
brachycephaly. As such, the condition is best described as deformational plagiocephaly-brachycephaly with isolated
plagiocephaly and/or isolated brachycephaly being at either ends of the spectrum. Due to its symmetric appearance,
deformational brachycephaly is often incorrectly dismissed as being less concerning, and it has sometimes erroneously
been reported that brachycephaly cannot be treated successfully with a cranial orthosis. We prospectively report
on 4205 infants with isolated deformational brachycephaly treated with a cranial orthosis from 2013 to 2017. These
results demonstrate that the orthosis is successful in the treatment of deformational brachycephaly with an 81.4%
improvement toward normal (95.0 to 89.4) in cephalic index. We furthermore demonstrate that entrance age
influences treatment results, with younger infants demonstrating both improved outcomes and shorter treatment
times.
Keywords
deformational brachycephaly, cranial orthosis, flat head syndrome, cephalic index
Received July 2, 2018. Received revised August 9, 2018. Accepted for publication August 28, 2018.
2 Global Pediatric Health
postural stability, with the shorter anterior muscles able
to react faster than the lengthened extensor groups. This
poor postural stability affects the efficiency of move-
ment, and infants will tend to either posture with the
chin flexed and trunk rounded or hyperextend the neck
and elevate the shoulders (ie, park the head) in order to
stabilize and maintain balance.19-23 These postures cause
less variability in the infant’s movement, which limits
their interaction with their environment.
Recently, the potential relationship between severe
deformational brachycephaly and hindbrain herniation,
due to the reduction in posterior cranial fossa volume,
has been discussed,24,25 although this has not yet been
investigated extensively. Additionally, one of the imme-
diate concerns of deformational brachycephaly is its
impact on the fit of protective equipment. In brachy-
cephaly, the head is disproportionally wider, shorter, and
often taller than the average head for that age. It is not
uncommon for parents to report having to purchase
adult-sized helmets in an attempt to accommodate for
the increased width of their child’s or adolescent’s head,
but then discovering that the helmet tends to tip into the
child’s face. When considering the number of athletic
and recreational activities that now require the use of
protective helmets, this is no small consideration and
affects not only the child’s participation, but also how
well they are protected during the activity.
While the treatment of plagiocephaly has received
considerable attention (see Flannery et al26 for a recent
review), only 2 studies27,28 have specifically addressed the
treatment of brachycephaly. This study was undertaken to
prospectively examine the effects of helmet treatment of
isolated deformational brachycephaly and to investigate
the role of 3 key treatment factors (entrance age, treat-
ment time, and initial severity) on treatment outcome.
Materials and Methods
Study subjects were identified from among a cohort of
infants who have been registered in a central clinical
research database since January 2013. Briefly, the data-
base contains information on all infants referred by their
primary physician for consultation at any of 30 clinic
locations through the Unites States. This cohort includes
infants with abnormal head shape diagnoses of all types
(synostotic and nonsynostotic), forms (plagiocephaly,
brachycephaly, dolichocephaly), and severity levels
(mild to severe). Data include demographic and assess-
ment information as well as a detailed medical history
regarding the well-established risk factors previously
reported.29-31 Quantifiable information regarding the
infant’s cranial shape is obtained using a 3-dimensional
(3D) imaging system previously documented else-
where32,33 (Figure 1). The accuracy of both the 3D image
acquisition, as well as software measurement functions,
have been previously validated to be within ±0.5
mm.32-34
Subject Identification
Patient data from the period January 2013 through
December 2017 (5 years; 128 014 patients) were evalu-
ated. The study population comprised 4205 infants (3.2%
of the total patient population) treated for isolated defor-
mational brachycephaly. Study subjects had complete
records at entry into and exit from treatment, moderate to
severe brachycephaly as previously described27,28 (ie, a
cephalic index [(Cranial Width/Cranial Length) × 100]
90), normal or minimal asymmetry (specifically, cranial
vault asymmetry, midface asymmetry, skull base asymme-
try 3 mm), and had entered into treatment between 3 and
12 months of age. All infants began treatment within 3
Figure 1. Digital Surface Imaging. Image shown in (a) photographic, (b) solid, and (c) wireframe.
Kelly et al 3
weeks of their initial treatment consultation for a cranial
remodeling orthosis described elsewhere.35-37 Patients
with confounding medical conditions ( 0.9%; eg, synos-
tosis, syndromic conditions, surgical shunt) were excluded
from the analyses. The study protocol was approved by an
external independent review board (Argus IRB, Tucson,
AZ). Informed consent was obtained for all participants.
Statistical Analysis
To more easily visualize the effects of treatment age on
treatment outcome, the study population was divided
into 3 groups based on entrance age into treatment.
Group 1 entered treatment between 3 months and <6
months of age, Group 2 entered treatment between 6
months and <9 months, and Group 3 entered treatment
between 9 months and 12 months of age. These
groups were selected based on popular thresholds
established in the literature, and for the purpose of
allowing comparison to other previously published
studies.27,28 Descriptive statistics for all treatment vari-
ables in aggregate and by treatment group are reported
in Table 1.
All statistical analyses were performed using SAS
software.38 Group differences in sex ratio were evalu-
ated using χ2 test (SAS PROC FREQ38). Analysis of
variance (SAS PROC GLM using the DUNCAN
MEANS option to assess differences among group
means38) was performed to evaluate differences among
groups with regard to parametric variables as well as to
identify how key treatment parameters (age of treat-
ment, treatment time, and initial cephalic index) contrib-
uted to treatment outcome.
Results
A total of 4205 infants were studied in this investigation
(Table 1). Mean entrance age was 5.8 months with a
mean treatment time of 13.5 (±5.7) weeks. Over the
treatment period, circumference increased an average of
18.7 mm (±7.6), from 433.6 mm to 452.3 mm. Mean
cranial width began at 130.7 mm (±6.1) and increased
marginally to 132.5 mm (±6.2), a change of only 1.8
mm (±2.7) indicating that biparietal width was held as
intended. Conversely, the cranial length increased from
137.6 mm (±6.4) to 148.2 mm (±5.9), a change of 10.5
mm (±3.5). The result was a mean overall cephalic
index reduction of 5.6% (95.0% at treatment entry to
89.4% at treatment exit, representing an 81.4% improve-
ment toward normal; Table 1).
Moreover, by cross-classifying the infants by their
initial and final severities (Table 2), we can examine
how the infants responded to treatment. Of the 4205
infants in this investigation, 2921 (69.5%) infants began
treatment initially classified as having severe brachy-
cephaly. Of those, 17.4% (509/2921) finished treatment
in the normal category; 27.3% (799/2921) finished as
mild; 39.6% (1156/2921) were moderate; with only
15.6% (457/2921) remaining in the severe category.
Another way of reporting this is that, of the 2921 infants
initially classified as having a severe deformity at the
initiation of treatment, 84.4% (2464/2921) were no
Table 1. Relevant Treatment Parameters by Age of Entry Into Treatmenta.
Parameter All, N = 4205 3 to <6, n = 2485 6 to <9, n = 1531 9 to 12, n = 189
Consult age (months)*** 5.4 (±1.5) 4.4 (±0.6) 6.5 (±0.7) 9.6 (±0.8)
Entry age (months)*** 5.8 (±1.5) 4.8 (±0.6) 6.9 (±0.7) 10.0 (±0.8)
Treatment time (weeks)*** 13.5 (±5.7) 11.9 (±5.4) 15.8 (±5.3) 17.4 (±4.3)
% Male not significant 62.8% 63.3%#62.7%#57.7%#
Initial cranial index (CI)** 95.0 (±3.2) 95.1 (±3.3) 95.0 (±3.0) 94.3 (±3.1)
Exit CI*** 89.4 (±2.8) 89.3 (±2.9) 89.6 (±2.8) 90.0 (±2.7)
Change in CI*** −5.6 (±2.3) −5.8 (±2.3) −5.4 (±2.2) −4.3 (±1.8)
Initial circumference (mm)*** 433.6 (±18.3) 426.1 (±15.3)c443.1 (±16.2)c455.7 (±17.2)
Exit circumference (mm)*** 452.3 (±17.9) 446.6 (±16.5)c459.7 (±16.2)c468.2 (±17.0)
Change circumference (mm)*** 18.7 (±7.6) 20.4 (±7.9) 16.6 (±6.4) 12.6 (±5.8)
Initial cranial width (mm)*** 130.7 (±6.1) 128.3 (±5.2) 133.7 (±5.5) 136.5 (±5.9)
Exit cranial width (mm)*** 132.5 (±6.2) 130.6 (±5.7) 134.9 (±5.7) 137.7 (±5.8)
Change cranial width (mm)*** 1.8 (±2.7) 2.2 (±2.7) 1.2 (±2.5) 0.1 (±2.3)
Initial cranial length (mm)*** 137.6 (±6.4) 135.0 (±5.2) 140.8 (±5.5) 146.0 (±5.9)
Exit cranial length (mm)*** 148.2 (±5.9) 146.3 (±5.5) 150.6 (±5.5) 153.0 (±5.7)
Change cranial length (mm)*** 10.5 (± 3.5) 11.3 (±3.5) 9.8 (±3.0) 7.0 (±2.7)
aFor overall group differences: ***P < .0001; **P < .001. For group-wise comparisons: means with “#” are not significantly different. Other
pairwise comparisons (ie, unmarked group statistics) are significant at the .05 level.
4 Global Pediatric Health
Table 2. Pretreatment Versus Posttreatment Classification of Severity.
Pretreatment Classification Posttreatment Classification
Total Normal (88) Mild (>88 to 90)
Moderate (>90
to 93) Severe (>93)
Mild (=90) 26 (0.6%) 22 (0.5%) 4 (0.1%) 0 (0.0%) 0 (0.0%)
Moderate (>90 to 93) 1258 (29.9%) 868 (20.6%) 335 (8.0%) 557 (1.3%) 0 (0.0%)
Severe (>93) 2921 (69.5%) 509 (12.1%) 799 (19.0%) 1156 (27.5%) 457 (10.9%)
Total 4205 (100.0%) 1399 (33.3%) 1138 (27.1%) 1211 (28.8%) 457 (10.9%)
Figure 2. Female infant starting treatment at 4 months of age; initial cephalic index: 98.5; exit cephalic index: 89.3; treatment
time 2¾ months (grid units 20 mm).
Kelly et al 5
longer in that category at the end of treatment, with
nearly half, 44.8% (1308/2921), having been returned to
a “normal-to-mild” classification. In totality, 60.3%
(2537/4205) ended treatment with a “normal-to-mild”
classification (Figure 2). Overall, 87.7% of the infants
(3689/4205) demonstrated improvement in cephalic
index following treatment with a cranial orthosis; 3948
infants (92.9%) having been treated with only one
Table 3. Results of Analysis of Variance for Treatment Variables Showing Differences by Treatment Groupa.
Parameter
3 to <6 Months
of Age
6 to <9 Months
of Age
9 to 12 Months
of Age F P
Initial cephalic index (CI) 95.1#95.0#94.3 5.34 .0048
Treatment time 11.9 15.8 17.4 300.34 <.0001
Change in CI −5.8 −5.4 −4.3 48.85 <.0001
aFor groupwise comparison: means with “#” are not significantly different. Other pairwise comparisons are significant at the .05 level.
Figure 3. Mean treatment time by group (with 1 standard deviation bars).
Figure 4. Mean change in cephalic index by group (with 1 standard deviation bars).
6 Global Pediatric Health
cranial orthosis. In no case did the condition worsen.
With the exception of a low incidence (0.91%) of skin
irritation (red spots, skin breakdown, heat rash), no sig-
nificant issues were reported.
To explore the effects of age on treatment, the sam-
ple was stratified into 3 equal interval treatment ranges
(3 to <6 months; 6 to <9 months; 9 to 12
months; Tables 1 and 2). Although the mean entrance
Figure 5. Male infant entering treatment at 3¾ months of age; initial cephalic index: 102.3; exit cephalic index: 90.5;
treatment time 3.25 months.
Figure 6. Male infant entering treatment at 8 months of age; initial cephalic index: 102.9; exit cephalic index: 91.1; treatment
time 9.5 months.
Kelly et al 7
cephalic index was not statistically significant different
between the groups (95.1, 95.0, 94.3; Table 3), treat-
ment time was significantly longer (11.9 weeks, 15.8
weeks, 17.4 weeks; Table 3 and Figure 3) and treat-
ment changes significantly smaller (5.8, 5.4, 4.3; Table
3 and Figure 4) moving up in age cohort. As would be
anticipated from the pediatric cranial growth charts,
mean change in circumferential growth also decreased
with entrance age (20.4 mm, 16.6 mm, 12.6 mm; Table
1), and this despite the longer treatment times docu-
mented for the older groups. Therefore, although the
initial deformity was not significantly different across
the age groups, infants treated prior to 9 months of age
received significantly greater improvements than chil-
dren treated at 9 months of age or later. Moreover, chil-
dren treated at earlier ages had significantly shorter
treatment times than those in subsequent treatment
groups (Figures 5 and 6).
Analysis of variance was also used to partition the
variation observed among set observations into portions
associated with certain factors. For example, variation in
improvement in cephalic index can be partitioned into
factors associated with “initial cephalic index,” “treat-
ment time,” and “age at entry into treatment” (Table 4).
The 2 intuitively obvious findings were that greatest
change in cephalic index could be achieved (a) in those
infants who initially presented with the most severe defor-
mities (ie, had the largest initial cephalic index), and (b)
by treating any infant (regardless of severity) for a longer
period of time. However, the more interesting and clini-
cally meaningful findings were that the younger the infant
entered treatment (c) the shorter their treatment duration,
and (d) the greater their reduction in cephalic index.
Discussion
Although the American Academy of Pediatrics’ “Back
to Sleep (BTS)” campaign is frequently cited as the
reason for the recent increase in cranial deformities,
other factors—most notably, devices of convenience
also contribute. Today infants spend extended periods
of time in devices including infant swings, bouncy
seats, carriers, and car seats.39 Although not always
appreciated, these devices result in cranial deforma-
tion that are similar to those produced by the cradle
boards used by several Native American Indian
tribes.40,41 In fact, Davis et al42 have documented that
infants from the ages of 0 to 3 months (a critical age in
the development of plagiocephaly/brachycephaly)
spent ~23 hours/day in a supine-like position. Today
the American Academy of Pediatrics, as well as many
other organizations, now advise parents to limit the
time infants spend in car seats and other devices of
convenience.43
Correction of Deformational Brachycephaly
While the treatment of deformation brachycephaly has
received limited attention, both previously published
studies27,28 reported significant correction of the defor-
mation (Table 5). In particular, in the only other treat-
ment study to have focused exclusively on deformational
brachycephaly, Graham and colleagues28 report signifi-
cant correction of deformational brachycephaly. Among
a subgroup of infants who—in common with the current
study—initiated treatment with a cephalic index 90%
(n = 92), Graham and associates observed a mean
reduction in cephalic index of 4.2% (from 96.1% to
91.9%).
Table 4. Results of Analysis of Variance for the Effects of Treatment Variables on “Change in Cephalic Index” Using the Type
III Sum of Squares to Partition Their Contribution.
Parameter Type III Sum of Squares F P
Initial cephalic index 3729.31 1038.54 <.0001
Treatment time 747.90 208.28 <.0001
Entry age 886.27 246.81 <.0001
Table 5. Deformational Brachycephaly Study Comparisons.
Parameter Teichgraeber (2004)27 Graham (2005)28 Kelly (Current)
Sample size (n) 64 92 4205
Mean initial cephalic index 93.7% 96.1% 95.0%
Mean end cephalic index 90.9% 91.9% 89.4%
Mean change in cephalic index 2.8% 4.2% 5.6%
Treatment time 4.5 months 3.7 months 3.4 months
8 Global Pediatric Health
Teichgraeber et al27 reported that helmet treatment
produced favorable outcomes for infants with both
deformational brachycephaly as well as deformational
plagiocephaly; however, they noted that “the head
shapes of the children with positional brachycephaly did
not normalize despite statistically significant improve-
ment in their cephalic index . . . ,” concluding that “. . .
helmet therapy is more effective in children with poste-
rior positional plagiocephaly than in children with posi-
tional brachycephaly.”
However, it should be noted that the challenge of
returning a brachycephalic head to within normal limits
lies in the observation that once an infant’s head has
obtained a certain width, there is no way to reduce this
dimension. By design, cranial orthotic devices do not
compress the head and therefore cannot make a head any
narrower; all that can be achieved is to redirect future
growth in the anteroposterior dimension. Additionally,
in severe cases, where the occipital bone has been
allowed to become nearly perfectly flat, it is difficult to
restore the natural occipital curve. Instead, increased
posterior growth will often result in lengthening to the
cranium and improved cephalic index, yet from a lateral
perspective, the occipital profile may still appear flat.
Hence, an argument could be made that intervention
prior to this level of deformity is warranted, both from a
treatment outcome as well as a preservation of posterior
cranial volume perspective.
Influence of Entrance Age on Outcome
Although Teichgraeber et al27 found that “the age at
which therapy was begun did not have an impact on the
final results,” they further note that “these results do not
correlate with what is seen clinically . . . ,” suspecting
that the “discrepancy between the data and the authors’
clinical experience may be a result of having arbitrarily
divided the children into 2 subgroups and the small
numbers of children in both of these subgroups.”27
Consistent with our findings, Graham et al28 found an
inverse correlation between entrance age and outcome.
For infants beginning treatment between 3.0 and 4.5
months of age, reduction in cephalic index was 5.1%;
for infants 4.5 to 6.0 months of age, it was 3.2%; and for
infants entering treatment later than 6 months, reduction
in cephalic index was 2.9%. These results mirror the
findings of other investigators who have previously
reported on the positive impact of early entrance age on
the effectiveness of the cranial orthosis37,44-52 (Table 6).
The key message from these observations is that
brachycephaly, just like plagiocephaly, should not be
allowed to progress to a severe classification before
intervention is started. Conservative efforts such as
supervised tummy time, repositioning, and limiting time
in devices of convenience should be initiated as soon as
a widening of the head is observed. If after 6 to 8 weeks
of these efforts the head is continuing to become more
brachycephalic, use of a cranial orthosis may be war-
ranted in order to leverage—as we have demonstrated
here—the benefits of early intervention that include
improved outcomes and shorter treatment times.
As with all studies, it is important to acknowledge the
limitations of the work presented so that future investi-
gators may be aware, and critically review the content in
light of these weaknesses. The most immediate limita-
tion is that this is not, nor was it designed to be, a ran-
domized control trial. While many authors have
previously discussed why execution of a randomized
control trial may be difficult or even ethically question-
able,52-54 it was simply not within our scope to be able to
perform. As a medical treatment provider, patients are
sent by physicians who have diagnosed and monitored
their patients and have prescribed treatment based on
their dissatisfaction with the progression of head shape.
The use of linear anthropometric measurements to
describe changes in a complex 3D head shape is also a
limitation of this study. Although there is a high degree
of confidence in the repeatability and reliability of
these measurements, linear measurements can only
convey so much information.55 This was why we chose
to provide so many figures illustrating the correction
that may be achieved, as these figures demonstrate the
clinically significant change in curvature, volume, and
shape that are sometimes difficult to appreciate with
just a few percentage point changes in cephalic index.
Several studies have now reported on the use of 3D
data in the form of root mean square calculations, and
we applaud the authors in those efforts and feel this is
the direction that future investigations must go.56,57
However, in the case of studies on proportionality,
Table 6. Deformational Brachycephaly Study Change Comparisons by Age Group.
Age at Treatment
Initiation Teichgraeber (2004)27 Graham (2005)28 Kelly (Current)
3.0 to 4.5 months 2.8% 5.1% 5.8% 5.8%
4.5 to 6.0 months 3.2% 5.8%
6.0 to <9.0 2.6% 2.9 % 5.2% 5.4%
Kelly et al 9
such as in deformational brachycephaly, the root mean
square measure is a less useful measurement.
Furthermore, the cephalic index is a well-established,
reproducible value that is well understood by the medi-
cal community, and in using this measure, it allowed us
to make direct comparisons to other previously pub-
lished studies on this subject.
Conclusions
As discussed, deformation of the cranium in infancy
represents a spectrum of deformity, ranging from severe
asymmetric yet proportional distortion of the skull in
plagiocephaly, to nearly symmetric yet disproportional
distortion in brachycephaly. As such, the condition is
best described as deformational plagiocephaly-brachy-
cephaly with isolated plagiocephaly and/or isolated
brachycephaly being at either ends of the spectrum.58,59
This investigation demonstrates that the cranial ortho-
sis is successful in the treatment of deformational brachy-
cephaly. These findings are consistent with the only other
2 published studies specifically looking at deformational
brachycephaly as a separate entity from deformational
plagiocephaly. It has also been demonstrated that
entrance age is a critical variable in the overall effective-
ness of treatment with younger infants demonstrating
both improved outcomes and shorter treatment times,
regardless of the severity of the presenting deformity.
When considering the mechanics of how a cranial
orthosis works (ie, holding the prominences and redi-
recting brain growth into the adjacent flattened areas), as
well as a basic understanding of normal craniofacial
growth patterns of the infants from birth to 12 months of
age, it may be recognized that the treatment of deforma-
tional brachycephaly in many ways is no different than
the treatment of deformational plagiocephaly. All that
has changed is the direction in which the corrective
forces are applied, from a contralateral pattern in plagio-
cephaly to a lateral pattern in brachycephaly. The rest is
accomplished by growth of the brain and proper adjust-
ment of the product by the treating clinician.
Authors’ Note
A preliminary portion of these data were included in the book
Smith’s Recognizable Patterns of Human Deformation, 4th
edition, by Graham JM and Sanchez-Lara P, and cited as
unpublished data (eBook ISBN: 9780323295383; Hardcover
ISBN: 9780323294942).
Acknowledgments
The authors would like to thank the members of the Image
Processing Department for their dedication and countless
hours of assistance and for whom without which this study
would not have been possible, as well as Brody Kilgore,
engineering intern with Arizona State University, for his
assistance in programing and querying the SQL database
containing the patient records utilized in this study. We
would also like to thank Canfield Scientific for their assis-
tance in developing the standardization and automation of
the measurement functions, and for providing study results
validating the accuracy of the software applications.
Author Contributions
KMK participated in the conceptualization and design of the
study, performed all statistical analysis, and reviewed, revised
and approved the final manuscript as submitted.
JAR developed the DSi Analysis reports; developed, pro-
grammed and maintained the Excel/SQL databases; and
reviewed and approved the final manuscript as submitted.
EFJ reviewed the design of the study and, reviewed and
approved the final manuscript as submitted.
SPB reviewed the design of the study and, reviewed and
approved the final manuscript as submitted.
MKM provided insight into the developmental impact of
brachycephaly and concerns being raised in the physical and
occupational therapy communities, and reviewed and approved
the final manuscript as submitted.
TRL conceptualized and designed the study, submitted the
IRB approval, trained the individuals and verified the accuracy
of the anthropometric measurements, and approved the final
manuscript as submitted.
Ethical Approval
The study protocol was approved by an external independent
review board (Argus IRB, Tucson, AZ; CF#02_01).
Declaration of Conflicting Interests
The author(s) declared the following potential conflicts of
interest with respect to the research, authorship, and/or publi-
cation of this article: Mr Littlefield, Mr Riggs, and Ms McGuire
are employees of Cranial Technologies, Inc. The remaining
authors have no financial or commercial interest.
Funding
The author(s) received no financial support for the research,
authorship, and/or publication of this article.
Informed Consent
Informed consent was obtained for all participants.
ORCID iDs
Kevin M. Kelly https://orcid.org/0000-0002-9177-1454
Timothy R. Littlefield https://orcid.org/0000-0002-8262
-5519
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... [9][10][11][12][13] Children with deformed skulls may be unable to properly wear safety equipment such as helmets or eyeglasses 8 and there can be an effect on muscle balance or postural alignment. 14 Skull deformation can also lead to difficulty styling hair or bullying by peers due to differences in appearance (such as auricular asymmetry). The child's inability to wear glasses, helmets, or the like can also contribute to a low self-image. ...
... 16 More modern studies have stretched the definition of a normal head shape, categorizing brachycephaly as CIs greater than 90%, 17,18 92%, 19 93%, 16 94%, 20 and 95%. 21 Several sources agree that the upper bound of normal head shapes post-"Back to Sleep" campaign is somewhere around 90%. 14,17,18 In various studies, CROs have been shown to reduce the appearance of deformation. However, there exists a dearth of studies with the statistical power to provide numerical evidence as to the effectiveness of CROs, and very few studies examine isolated brachycephaly. ...
... Among published studies, a few trends have emerged: younger infants achieve more correction than older infants and infants with a higher initial CI achieve more correction. 4,7,14,18,19,[22][23][24] The lack of published studies with large cohorts is detrimental to patient care because many insurance providers cite the lack of studies to deny coverage of CROs, especially for the brachycephalic population. Furthermore, current literature frequently groups brachycephaly with other deformational head shapes. ...
... Brachycephaly, defined as the shortened anteroposterior length of the head compared to the width [1], can cause several deleterious effects as well as cosmetic issues. For example, brachycephaly can change the angular orientation of the temporomandibular joint, leading to malocclusion [2,3]. Furthermore, it can cause the anterior displacement of the mandible, resulting in obstructive sleep apnea [4]. ...
... Furthermore, it can cause the anterior displacement of the mandible, resulting in obstructive sleep apnea [4]. Brachycephaly can also change the center of mass of the head, causing an imbalance in the neck flexor and extensor muscles and poor postural stability [3]. One recent report described a case in which brachycephaly caused a reduction in the cranial fossa volume, resulting in the herniation of the hindbrain [5]. ...
... The children were divided into five groups according to age. The age standard was set in consideration of other papers and the slope of head circumference [3,20,24,25]. The slope of the head circumference growth is very steep in the early infant period. ...
Full-text available
Article
Brachycephaly has several potential deleterious effects, including malocclusion, sleep apnea, and abnormal posture. Nevertheless, the research regarding helmet therapy as a treatment strategy for brachycephaly is limited. Herein, we aimed to analyze the factors influencing the effects of helmet therapy in infants with brachycephaly. We retrospectively reviewed the records of 207 infants aged 3–14 months with a cranial index (CI) >90% who received helmet therapy between May 2016 and October 2019 and complied with the treatment protocol well. We used a multiple regression analysis to determine which factors affected the duration of therapy and a Jonckheere–Terpstra test to establish differences in the duration of helmet therapy according to age and severity. We identified brachycephaly severity (p < 0.001), asymmetry (p < 0.001), and age (p < 0.001) as factors affecting the duration of therapy. Helmet therapy might be effective for infants with moderate to severe brachycephaly, assuming good protocol compliance. In addition, younger treatment initiation age and less severe and less asymmetric brachycephaly significantly shorten the treatment duration.
... Therefore, asymmetrical brachycephaly is characterized by a posterior flattening of the occiput as well as an asymmetry with compensatory posterior bossing contralaterally. These deformities are caused by external pressures on the baby's skull when the skull is in the same position for extended periods of time [1][2][3][4][5]. This relatively common occurrence is often attributed to the American Academy of Pediatrics "Back to Sleep" campaign, which recommended infants be placed supine to sleep. ...
... If repositioning therapy is attempted and is not successful, or if the skull deformity is very severe to begin with, a CRO is then considered [6]. Severity has also been shown to play a role in the outcome of the final head shape in brachycephalic and plagiocephalic head shapes [3,8]. ...
... Some studies have shown relationships between children with plagiocephaly and brachycephaly having additional health issues, with asymmetrical brachycephaly having the highest risk of complications [2]. Some of the issues associated with skull deformations include: increased need for special services at school age [1], delays in psychomotor development [18], abnormal speech development [1], jaw asymmetry [3], temporomandibular joint dysfunction [3,4,[19][20][21][22][23], abnormal muscle tone [3], ear misalignment [24], and middle ear abnormalities [24]. Additionally, abnormal head and face shapes can be very noticeable on children and adults. ...
Full-text available
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This retrospective chart review focuses on determining the most effective time to begin cranial remolding orthosis (CRO) treatment for infants with asymmetrical brachycephaly. Subjects with asymmetrical brachycephaly started CRO treatment between 3 and 18 months of age. These infants had a cranial vault asymmetry index (CVAI) ≥ 3.5 and a cranial index (CI) ≥ 90. Subjects were excluded if they had any comorbidities affecting growth, dropped out of treatment, were lost to follow-up, or were noncompliant. Factors which were found to statistically influence treatment outcomes were subject initial age, initial CVAI, and initial CI. Overall, younger subjects were more likely to achieve a corrected head shape. The presence of prematurity or torticollis had statistically nonsignificant effects on the success of treatment. Initial CI was found to be a stronger predictor than initial CVAI as to which subjects achieved correction. The less severe the starting CI, the more likely the subject was to achieve full correction. The clinical understanding is that it requires more cranial growth to “round out” a full posterior skull flattening than an asymmetry. Based on the study results, infants with asymmetrical brachycephaly should be treated as early as possible to increase chances of achieving full correction of the deformity.
... 2,6 There is evidence that if left untreated, deformation of the cranium may be permanent. 7 Some issues associated with DP include: increased need for special services at school age, 1 delays in psychomotor development, 8 abnormal speech development, 1 jaw asymmetry, 9 temporomandibular joint dysfunction, [9][10] abnormal muscle tone, 9 ear misalignment, 11 and middle ear abnormalities. 11 According to the Congress of Neurological Surgeons, infants with DP should first be treated by either repositioning strategies or physical therapy; those with persistent moderate to severe deformities should be treated with a cranial remolding orthosis (CRO). ...
... 2,6 There is evidence that if left untreated, deformation of the cranium may be permanent. 7 Some issues associated with DP include: increased need for special services at school age, 1 delays in psychomotor development, 8 abnormal speech development, 1 jaw asymmetry, 9 temporomandibular joint dysfunction, [9][10] abnormal muscle tone, 9 ear misalignment, 11 and middle ear abnormalities. 11 According to the Congress of Neurological Surgeons, infants with DP should first be treated by either repositioning strategies or physical therapy; those with persistent moderate to severe deformities should be treated with a cranial remolding orthosis (CRO). ...
Article
Infants with deformational head shapes may be treated with a Cranial Remolding Orthosis (CRO). Risks of CROs include skin irritations and other minor side effects. It is important to examine the incidence of complications with CRO treatment to determine if the potential benefits outweigh the risks.This study retrospectively examined surveys provided by both practitioners and caregivers regarding orthotic treatment side effects and wear time at four clinics in Canada. Statistical analysis including the Chi-square test and Fisher's exact tests were performed to examine how the side effects varied by treatment site and by reported orthotic wear time.In total, 5,025 surveys about the CRO treatment of 453 patients were reviewed. The most common side effects were pressure spots, redness lasting for more than 1 hour, excessive sweat, and foul odor; however, the vast majority reported no significant issues (87.08%). Only 2.81% of surveys reported multiple side effects, which was about three times more like to happen when the CRO was reported to have fit problems. Wear time is correlated with the report of side effects; infants who wore the CRO 18 to 23.5 hours had statistically fewer side effects than those who wore the CRO for less time. The reported incidence of side effects may be artificially inflated because infants experiencing side effects are more likely to have more frequent follow-up.Overall, the side effects of CRO treatment reported in this cohort were relatively rare and minimal, indicating CRO treatment is a safe way to treat deformational head shapes.
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Plagiosefali ve kask tedavisi: olgu sunumu Plagiosefali, kafatasının çeşitli nedenlerle asimetrik deformasyonunu ifade eder
Article
Background: Positional skull deformity usually manifests during first six months of life due to various factors like premature births or multiple births, improper positioning of infant’s head as the head of an infant is softer than the older children’s head, thus leading to either positional brachycephaly or positional plagiocephaly. Early helmet therapy intervention may improve the shape of the skull by reducing the risk of secondary cosmetic and nervous system complications. Aim: To study the effectiveness of helmet therapy in infants with positional skull deformity. Methods: The data source for this literature review is done by studying and reviewing articles through various data like Pub Med, Google Scholar, science direct, Elsevier and medicine Cochrane library. Conclusion: Helmet therapy is contemplated to be effective in the treatment of mild-moderate-severe positional skull deformity than repositioning therapy by improving the structure of the misshaped skull, as well as the use of helmet therapy is reviewed not to hinder the head circumference growth in infants.
Full-text available
Article
Purpose: The aim of this study was to investigate the optimal age for starting cranial-remolding-orthosis therapy in children with deformational plagiocephaly. Methods: Medical records of 310 patients with deformational plagiocephaly were retrospectively reviewed and the initial and final cranial vault asymmetry index (CVAI), age when starting therapy, duration of therapy, mean change of CVAI, improvement rate, and treatment success were analyzed. We compared outcomes according to the groups divided by ages starting therapy. Results: There were no significant differences in improvement rate and duration of cranial-remolding-orthosis therapy among patients starting therapy at the age of 3, 4, and 5 months. However, when starting therapy after the age of 6 months, the rates of CVAI improvement were significantly lower and the duration of therapy was significantly increased. Conclusion: Considering the spontaneous resolution effect according to the head growth nature, the age 5 month is the optimal period to start cranial-remolding-orthosis therapy for deformational plagiocephaly.
Full-text available
Article
OBJECTIVE Following institution of the Back to Sleep Campaign, the incidence of sudden infant death syndrome decreased while the prevalence of positional skull deformation increased dramatically. The management of positional deformity is controversial, and treatment recommendations and outcomes reporting are variable. The authors reviewed their institutional experience (2008–2014) with the treatment of positional plagiocephaly to explore factors associated with measured improvement. METHODS A retrospective chart review was conducted with risk factors and treatment for positional head shape deformity recorded. Univariate and multivariate analyses were used to assess the impact of these variables on the change in measured oblique diagonal difference (ODD) on head shape surface scanning pre- and posttreatment. RESULTS A total of 991 infants aged less than 1 year were evaluated for cranial positional deformity in a dedicated clinical program. The most common deformity was occipital plagiocephaly (69.5%), followed by occipital brachycephaly (18.4%) or a combination of both deformities (12.1%). Recommended treatment included repositioning (RP), physical therapy (PT) if indicated, or orthotic treatment with a customized cranial orthosis (CO) according to an age- and risk factor–dependent algorithm that the authors developed for this clinic. Of the 991 eligible patients, 884 returned for at least 1 follow-up appointment. A total of 552 patients were followed to completion of their treatment and had a full set of records for analysis: these patients had pre- and posttreatment 2D surface scanner evaluations. The average presenting age was 6.2 months (corrected for prematurity for treatment considerations). Of the 991 patients, 543 (54.8%) had RP or PT as first recommended treatment. Of these 543 patients, 137 (25.2%) transitioned to helmet therapy after the condition did not improve over 4–8 weeks. In the remaining cases, RP/PT had already failed before the patients were seen in this program, and the starting treatment recommendation was CO. At the end of treatment, the measured improvements in ODD were 36.7%, 33.5%, and 15.1% for patients receiving CO, RP/PT/CO, and RP/PT, respectively. Univariate analysis showed that sex, race, insurance, diagnosis, sleep position preference, torticollis history, and multiple gestation were not significantly associated with magnitude of ODD change during treatment. On multivariate analysis, corrected age at presentation and type of treatment received were significantly associated with magnitude of ODD change. Orthotic treatment corresponded with the largest ODD change, while the RP/PT group had the least change in ODD. Earlier age at presentation corresponded with larger ODD change. CONCLUSIONS Earlier age at presentation and type of treatment impact the degree of measured deformational head shape correction in positional plagiocephaly. This retrospective study suggests that treatment with a custom CO can result in more improvement in objective measurements of head shape.
Full-text available
Article
Purpose: To determine if infants with positional plagiocephaly have limitations of active and passive cervical range of motion measured with simple and reliable methods. Methods: The examiners assessed bilateral active and passive cervical rotations and passive cervical lateral flexion. Cervical assessment was performed twice by 2 different physicians to assess intertester reliability. To assess intratester reliability the first investigator performed a second examination 48 hours after the first one. Results: One-hundred nine subjects were analyzed; 70.7% of the sample had head positional preference on the right, while 29.3% had head positional preference on the left (χ 35.52, P <0.001). Cervical rotations and lateral flexion showed reliable levels of agreement for intra and intertester reliability. Conclusions: The most limited range of motion in infants with positional plagiocephaly was cervical active rotation which affected more than 90% of patients. Passive cervical rotations and lateral flexion were limited in more than 60% of patients.
Full-text available
Article
Objectives Asymmetries of the jaw and orthodontic abnormalities are suspected as long-term consequences of positional cranial deformity. But only few data exist on this issue. As plagiocephaly is a common problem in infancy, potential functional impairments should be investigated to initiate appropriate measures if necessary. The aim of our study was to compare the orthodontic situation in primary dentition of children with positional plagiocephaly and children without cranial deformities. Material and methods Fifty children treated by helmet therapy for plagiocephaly and 50 non-affected children (age 1.98–5.69 years) were examined in a cross-sectional study. Orthodontic parameters of all dimensions were assessed and analyzed. Results Children of the plagiocephalic group showed more often orthodontic alterations compared to the others. Especially the frequencies of a class II malocclusion (36 vs. 14 %), an edge-to edge bite (28 vs. 12 %), and deviations of the midline (38 vs. 16 %) were conspicuous. However, none of the differences was significant (p > 0.003). Of all observed mandibular asymmetries, 69 % appeared as a shift to the contralateral side of the former flattened occipital region. Conclusion Positional head deformity might be associated in some cases with a higher prevalence of occlusal abnormalities in primary dentition. Clinical relevance Positional plagiocephaly interfaces medicine and dentistry. As it is a common disorder, this etiology has to be considered in the prevention and therapy of malocclusion.
Article
Objectives Infants with positional plagiocephaly often exhibit complex multistructuraI asymmetries that affect the face and skull base as well as the cranial vault. Dynamic Orthotic Cranioplasty (DOC) was developed as a nonsurgical alternative for the treatment of positional plagiocephaly. The effectiveness of DOC has been discussed elsewhere. The purpose of this study was to assess the influence of factors such as entrance age, treatment time, and initial severity on the effectiveness of correction. Methods The study sample consisted of 258 children with cranial vault asymmetry (CVA) treated prior to 1 year of age. In addition, 246 patients (92%) exhibited concurrent skull base (SBA) and orbitotragial depth (OTDA) asymmetries. All patients had been diagnosed with nonsynostotic plagiocephaly, did not have other contributing medical conditions, were compliant with DOC protocol, and had complete anthropometric measurements at entrance and exit from treatment. Results Mean age at start of treatment was 6.5 (±1.9) months (range, 2.8 to 11.0 months), with an average treatment time of 4.1 (±2.2) months. The effects of the treatment variables were analyzed using three-way analysis of variance. As expected, initial severity was significantly associated with the amount of correction (p = .0001). However, treatment time was not significant (p > .05). Most importantly, the analysis revealed that, having accounted for initial severity, entrance age had a statistically significant effect [F[1,254) = 8.36, p = .0042] on the correction of CVA. Similar results were Identified for both the SBA [F[1,254) = 5.53, p = .0195] and the OTDA [F[1,254) = 5.22, p = .0231] asymmetries. Conclusions These findings support clinical observations that earlier intervention results in significantly improved treatment of plagiocephaly, independent of the severity of the presenting asymmetries.
Article
Purpose: The recommendation issued by the American Academy of Pediatrics in the early 1990s to position infants on their back during sleep to prevent sudden infant death syndrome (SIDS) has dramatically reduced the number of deaths due to SIDS but has also markedly increased the prevalence of positional skull deformation in infants. Deformation of the base of the skull occurs predominantly in very severe deformational plagiocephaly and is accompanied by facial asymmetry, as well as an altered ear position, called ear shift. Moulded helmet therapy has become an accepted treatment strategy for infants with deformational plagiocephaly. The aim of this study was to determine whether facial asymmetry could be corrected by moulded helmet therapy. Materials and methods: In this retrospective, single-centre study, we analysed facial asymmetry of 71 infants with severe deformational plagiocephaly with or without deformational brachycephaly who were undergoing moulded helmet therapy between 2009 and 2013. Computer-assisted, three-dimensional, soft-tissue photographic scanning was used to record the head shape before and after moulded helmet therapy. The distance between two landmarks in the midline of the face (i.e., root of the nose and nasal septum) and the right and left tragus were measured on computer-generated indirect and objective 3D photogrammetry images. A quotient was calculated between the two right- and left-sided distances to the midline. Quotients were compared before and after moulded helmet therapy. Infants without any therapy served as a control group. Results: The median age of the infants before onset of moulded helmet therapy was 5 months (range 3-16 months). The median duration of moulded helmet therapy was 5 months (range 1-16 months). Comparison of the pre- and post-treatment quotients of the left vs. right distances measured between the tragus and root of the nose (n = 71) and nasal septum (n = 71) revealed a significant reduction of the asymmetry (Tragus-Nasion-Line Quotient: 0.045-0.022; p < 0.0001; Tragus-Subnasale-Line Quotient: 0.045-0.021; p < 0.0001). The control group without treatment showed no significant change in the quotient (Tragus-Nasion-Line Quotient no helmet: 0.049-0.055/Tragus-Subnasale-Line Quotient no helmet: 0.039-0.055). Conclusion: Moulded helmet therapy can correct facial symmetry in infants with deformational plagiocephaly and associated facial and basal skull asymmetry.
Article
Background: Only a few studies investigating the optimal time point at which to start orthotic treatment for deformational plagiocephaly take into account the severity of skull asymmetry. The present study performs a three-dimensional analysis of the effects of age and severity of asymmetry on the final outcome. Methods: A total of 144 patients with deformational plagiocephaly treated by molding orthosis were examined and divided into three age groups (group I, <24 weeks; group II, ≥24 to <32 weeks; and group III, ≥32 weeks) and two severity levels (mild to moderate, 30-degree cranial vault asymmetry ≥3 mm to ≤12 mm; and moderate to severe, 30-degree cranial vault asymmetry >12 mm). The extent of the reduction of asymmetry was analyzed using three-dimensional stereophotogrammetry. Results: Therapy with molding orthosis led to a significant reduction in asymmetry in all defined age groups. Efficacy of reduction decreased with increasing age. Successful treatment (cranial vault asymmetry index <3.5 percent) was achieved in 83, 69, and 40 percent of patients with mild to moderate asymmetry in groups I, II, and III, respectively; and in 50, 30, and 7 percent of patients with moderate to severe asymmetry in groups I, II, and III, respectively. The average duration of treatment increased from 18.6 weeks to 25.3 weeks (age groups I and III). Conclusion: Age at the beginning of treatment and severity of asymmetry have a definite impact on the duration and effectiveness of molding orthosis therapy. Clinical question/level of evidence: Risk, II.
Article
Background: Positional plagiocephaly is the most common cause of cranial asymmetry. The underlying cause of Chiari-1 malformation has many possible theories, and anecdotally some pediatric neurosurgeons have had experience of severe cases of positional brachycephaly with Chiari-1. However, to date there have been no published cases linking non-synostotic plagiocephaly with Chiari-1 malformation. Case description: An 18-month-old boy presented with a head injury. On examination he had a Glasgow Coma score of 15 with no focal neurological deficits, but was noted to have marked posterior brachycephaly. A computed tomography scan showed a slim left-sided hemispheric acute subdural haematoma with no mass effect, which was treated conservatively. Of note, all his cranial vault sutures were open and a diagnosis of incidental positional plagiocephaly was made. Subsequent magnetic resonance imaging as part of a work up to exclude non-accidental injury showed a small posterior fossa with a steep tentorium and herniation of the cerebellar tonsils to the level of the body of the second cervical vertebra. Conclusions: Chronic hindbrain herniation is well reported in cases of craniosynostosis, but to our knowledge this is the first published case associated with non-synostotic deformational plagiocephaly. We hypothesise that severe posterior plagiocephaly can cause disproportion of the posterior fossa: hindbrain volume ratio and acquired chronic cerebellar herniation. Nevertheless, positional plagiocephaly and Chiari-1 are common entities and it is possible that the dual diagnoses were coincidental in this case. This report serves to raise awareness of the putative causal relationship between positional plagiocephaly, reduced posterior fossa volume and hindbrain herniation.
Article
AimThe objective of the study was to assess posture, muscle flexibility and balance in children aged 3-5years old with a history of nonsynostotic plagiocephaly. Methods Fifty-two children with previous history of plagiocephaly were evaluated, along with 52 control subjects matched for age, sex, height, weight and physical activity. The outcome measures included static posture, assessed through the measurement of angles and distances between anatomical landmarks; muscle flexibility, evaluated with the Stibor, Shober and finger-to-floor distance tests and balance, assessed by the Pediatric Balance Scale. ResultsOne-way analysis of variance afforded statistically significant differences (P<0.05) in head position, muscle flexibility (thoracic mobility and trunk and lower limbs muscle shortening) and balance. Conclusion Children with previous history of non-synostotic plagiocephaly present changes in head position, muscle shortening and a poor balance when compared to control children at 3-5years old.