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A Three-Dimensional Analysis
WHAT’S KNOWN ON THIS SUBJECT: Orthotic helmets and active AUTHORS: Angelo B. Lipira, MD,a Shayna Gordon, BS,a
repositioning are the most common treatments for Tron A. Darvann, PhD,b,c Nuno V. Hermann, DDS, PhD,b,c,d
deformational plagiocephaly. Existing evidence is not sufficient to Andrea E. Van Pelt, MD,a Sybill D. Naidoo, RN, CPNP,a
inform decisions between these options. Daniel Govier,a and Alex A. Kane, MDa
aDivision of Plastic and Reconstructive Surgery, Washington
WHAT THIS STUDY ADDS: A 3D, whole-head asymmetry analysis University School of Medicine, St Louis, Missouri; b3D
Craniofacial Image Research Laboratory and dPediatric
was used to compare treatment outcomes. This is the most Dentistry and Clinical Genetics, School of Dentistry, Faculty of
rigorous and comprehensive outcomes study to date on the Health Sciences, University of Copenhagen, Copenhagen,
subject. Denmark; and cIMM Informatics, Technical University of
Denmark, Copenhagen, Denmark
KEY WORDS
deformational, positional, nonsynostotic, plagiocephaly, orthotic,
helmet, active, repositioning, outcomes, asymmetry
abstract three-dimensional
ABBREVIATIONS
BACKGROUND AND PURPOSE: Orthotic helmets and active reposition- DP—deformational plagiocephaly
ing are the most common treatments for deformational plagiocephaly 2D—two-dimensional
(DP). Existing evidence is not sufficient to objectively inform decisions 3D—three-dimensional
between these options. A three-dimensional (3D), whole-head asymme- CVA—cranial vault asymmetry
try analysis was used to rigorously compare outcomes of these 2 treat- AP—asymmetry of a point P
ment methods. www.pediatrics.org/cgi/doi/10.1542/peds.2009-1249
PATIENTS AND METHODS: Whole-head 3D surface scans of 70 infants doi:10.1542/peds.2009-1249
with DP were captured before and after treatment by using stereopho- Accepted for publication Jun 9, 2010
togrammetric imaging technology. Helmeted (n ⫽ 35) and nonhel- Address correspondence to Angelo B. Lipira, BS, Division of
meted/actively repositioned (n ⫽ 35) infants were matched for sever- Plastic and Reconstructive Surgery, Washington University
ity of initial deformity. Surfaces were spatially registered to a School of Medicine, 660 S Euclid Ave, Box 8238, St Louis, MO
symmetric template, which was deformed to achieve detailed right-to- 63110. E-mail: lipiraa@wusm.wustl.edu
left point correspondence for every point on the head surface. A ratio- PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, 1098-4275).
metric asymmetry value was calculated for each point relative to its
contralateral counterpart. Maximum and mean asymmetry values Copyright © 2010 by the American Academy of Pediatrics
were determined. Change in mean and maximum asymmetry with FINANCIAL DISCLOSURE: The authors have indicated they have
treatment was the basis for group comparison. no financial relationships relevant to this article to disclose.
RESULTS: The helmeted group had a larger reduction than the reposi-
tioned group in both maximum (4.0% vs 2.5%; P ⫽ .02) and mean
asymmetry (0.9% vs 0.5%; P ⫽ .02). The greatest difference was local-
ized to the occipital region.
CONCLUSIONS: Whole-head 3D asymmetry analysis is capable of rigor-
ously quantifying the relative efficacy of the 2 common treatments of
DP. Orthotic helmets provide statistically superior improvement in
head symmetry compared with active repositioning immediately after
therapy. Additional studies are needed to (1) establish the clinical
significance of these quantitative differences in outcome, (2) define
what constitutes pathologic head asymmetry, and (3) determine
whether superiority of orthotic treatment lasts as the child matures.
Pediatrics 2010;126:e936–e945
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ARTICLE
Deformational plagiocephaly (DP) is The current standard of care for DP reserved for the most severe or refrac-
the most common head shape abnor- consists of either active reposition- tory cases.
mality in infants.1 Also known as ing or cranial molding with a custom
positional plagiocephaly or plagio- orthotic helmet. The aim of active re- Multiple studies have compared the ef-
cephaly without synostosis, DP pre- positioning is to prevent continued, fectiveness of orthotic helmets and ac-
sents as unilateral occipital flatten- prolonged pressure on the flattened tive repositioning,2,13,17,18,20–23 but cur-
ing, often with ipsilateral frontal side, whereas helmet therapy ap- rent evidence is not sufficient to
bulging and ear displacement that plies gentle external pressure to objectively guide treatment decisions.
results from repetitive, prolonged achieve a symmetric head shape. A 2005 Cochrane systematic review24
external pressure. Etiologies include Both methods rely on cranial mallea- stated that existing evidence is of mod-
sleep position, myoneural dysfunc- bility and rapid brain growth in the erate to poor methodologic quality,
tion, and intrauterine constraint, first year of life.13 Either method may and an improved measurement bat-
and risk factors have been identified also include stretching and physical tery is needed to better study treat-
that include birth injury, preterm ment effects. Previous studies have
therapy if torticollis (tilting of the
birth, male gender, and many oth- relied on two-dimensional (2D) anthro-
head to 1 side caused by contracted
ers.2–5 When viewed from the vertex, pometric data, which is prone to error
neck muscles) is present.
the resulting asymmetric head shape and does not adequately encompass
is a parallelogram.5 The choice of treatment modality is an
the three-dimensional (3D) nature of
active area of research. Helmet ther- head shape (Fig 1). Quantifying the
The incidence of DP has increased
apy is believed to be safe, with no subtle and complex elements that
dramatically since 1992, when the
American Academy of Pediatrics rec- reported detrimental effect on cranial comprise head shape requires sophis-
ommended that infants sleep supine growth,14 and has become widespread ticated whole-shape techniques. By ap-
to reduce the risk of sudden infant since first documented in 1979 by Clar- plying a more comprehensive, 3D anal-
death syndrome.6 Broad adherence ren et al.15 However, orthotic helmets ysis of head asymmetry, this study
to this recommendation has resulted are expensive ($1500 –$2500), and aims to thoroughly assess and com-
in a substantial decrease in the inci- third-party insurance payers typically pare the relative efficacies of active re-
dence of sudden infant death syn- refuse coverage, citing a lack of com- positioning and orthotic helmets in the
drome (from 0.26% in 1986 to 0.1% in pelling evidence for their superior treatment of DP.
1998),7 but DP has increased at least benefit to active repositioning.16 Active
10-fold.1,8–10 Before 1992, DP incidence repositioning is inexpensive but re- PATIENTS AND METHODS
was estimated at 0.3%,11whereas re- quires strict compliance, and some ev-
cent estimates range from 3% to 48%, idence suggests it is less effective than Whole-head, 3D surface scans of 70 in-
varying widely depending on sensitiv- helmet therapy.17,18 Although surgical fants (35 helmeted, 35 actively reposi-
ity of diagnostic criteria.12 treatment has been documented,19 it is tioned) with DP were captured before
FIGURE 1
A, Direct caliper measurement on a child. B, Illustration of potential error of 2D plane selection. Red and blue outlines illustrate the different shape of
cross-sections obtained from different plane selections. C, Head surface color-coded according to pointwise amount of asymmetry. Red and yellow indicate
flattened area.
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ARTICLE
FIGURE 3
Helmet region (red) shown on the symmetric head template.
RESULTS
Groups were similar at baseline (Table
1). Fig 6 A and B depict the spatial dis-
tribution of change in asymmetry for
the helmeted and repositioned groups,
respectively. Fig 6C shows the differ-
ence between the change for the 2
groups, thus providing the spatial dis-
tribution of amount of superiority of
either treatment protocol. A binarized
version of Fig 6C is shown in 6D, iden-
FIGURE 4
Schematic illustration of asymmetry calculation based on the ratio between distances d and d= from
tifying regions in which 1 treatment
the origin to the points P and P=, respectively (see text). The template deformation process ensures has a better effect than the other.
that P and P= are anatomically corresponding points on the left and right side of the midsagittal plane
(MSP), respectively. At the time of initiation of therapy, there
was no statistically significant difference
in average mean AP between the hel-
meted (3.5%) and actively repositioned
(3.5%) groups (P ⫽ .27). Comparison of
change in mean AP (over the entire hel-
met region) between groups revealed a
statistically significant greater reduc-
tion in the helmeted group (0.9%) versus
the repositioned group (0.5%) (P ⫽ .02)
(Fig 7). A pointwise t test of all points in
the helmet region localized the area of
statistically significant difference to a
region on the posterior head surface
(Fig 8). Within the statistically signifi-
cant region (red and green; 5% level of
FIGURE 5 significance) the helmeted group
Example of the result of asymmetry calculation in a subject, shown as color-coding on the symmetric
template surface. Red (positive) indicates the flattened area, and blue (negative) indicates asymmetry showed a 2.0% reduction of mean AP
in the contralateral area. Green dots mark the location of maximum asymmetry (max AP). Left, Before versus 1.1% in the repositioned group
treatment; right: after treatment.
(P ⫽ .001).
There was also no statistically signi-
average pretreatment to posttreat- cm. One percent of 7.5 cm is 0.75 mm. ficant difference in average initial max
ment change in mean AP and max AP, Therefore, an 8% reduction in max AP, AP between the helmeted (12.9%) and
expressed as a percentage, was the for example, corresponds to a change actively repositioned (13.0%) groups
basis of comparison between the 2 of ⬃6 mm. (P ⫽ .31). With treatment, helmeted in-
treatment groups. To better under- fants had a 4.0% mean reduction in
stand the meaning of these indices, Change in asymmetry also was as- max AP, versus 2.5% for repositioned
consider that the average cranial di- sessed as the difference between pre- infants (P ⫽ .02) (Fig 9). To visually
ameter for infants of this age group is treatment and posttreatment CVA. compare difference in outcomes, con-
⬃15 cm.29 Halving this value to esti- Head growth during treatment was structed head models with max AP that
mate the distance from the origin to a calculated by dividing total change in correspond to these values are over-
surface point on the head yields 7.5 head circumference by treatment du- laid in Fig 10.
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ARTICLE
FIGURE 9
Comparison of change in max AP between the
helmeted and repositioned groups.
DISCUSSION
FIGURE 6 This study, which used a novel 3D anal-
Group means and differences. A, Mean change in asymmetry for the helmeted group. B, Mean change ysis, reveals that orthotic helmets
in asymmetry for the repositioned (REPOS) group. C, Difference between mean asymmetry for the 2
groups. D, Binary map of the relative efficacies for each point on the head surface. Red indicates areas yield statistically superior reduction in
of the head on which the helmeted group had superior reduction in asymmetry, and blue indicates overall head asymmetry compared
areas on which the actively repositioned group had superior reduction. with active repositioning in the treat-
ment of DP. The rise in DP incidence
Mean duration of treatment was signif- since the 1992 start of the Back to
icantly shorter for helmeted infants Sleep campaign6 has intensified ef-
(3.1 months) versus repositioned (5.2 forts to determine the relative efficacy
months) (P ⬍ .001). Helmeted infants of orthotic helmet therapy and active
exhibited a mean reduction in CVA of repositioning. In 1997, experts con-
4.5 mm (from 8.4 to 3.9 mm) versus 3.4 vened at the Skull Molding Sympo-
mm for the repositioned group (8.1 to sium30 to attempt to reach a consen-
4.7 mm) (P ⫽ .14). There was no statis- sus, but concluded that there was
tically significant difference in average insufficient evidence to establish deci-
FIGURE 7
sive recommendations. A 2005 Co-
Comparison of change in mean AP (over the en- head growth during treatment
tire helmet region) between the helmeted and chrane systematic review24 similarly
repositioned groups.
(change in head circumference di-
concluded that the relative efficacy of
vided by treatment duration) between
conservative DP treatments could not
be determined because of a lack of
methodologic rigor in previous stud-
ies, and recommended that an im-
proved outcome measurement battery
be developed to better represent pa-
tient outcomes. We have developed a
methodology that eliminates much of
the error associated with 2D assess-
ments of head shape and more appro-
priately describes the 3D problem of
calvarial asymmetry.
Previous studies relied on 2D, direct
FIGURE 8 anthropometric measures to quantify
A pointwise t test assessed the statistical significance of the difference in improvement in mean
asymmetry at every point in the analyzed surface. Colors indicate the significance level (P) of superi- outcomes.2,13,17,18,20–23,31,32 Direct mea-
ority of helmet treatment. Red: P ⬍ .01; green: P ⬍ .05; blue: P ⬍ .1. surements are subject to error be-
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ARTICLE
compared with active repositioning ment goal in DP is satisfactory appear- sual responses to measured differ-
(P ⬍ .001). A retrospective study of 298 ance. However, qualitative information ences in shape, and overall change in
infants by Graham et al17 also reported is difficult to compare and vulnerable quality of life are needed to improve
a statistically significant greater im- to recall-bias. An ideal quantitative our understanding of the meaning of
provement in the helmeted group (7.1 method for studying outcomes should quantitative outcomes such as those
mm reduction in CVA versus 5.5 mm; describe shape in a complex, holistic reported in the present study.
P ⬍ .001). Rogers et al22 prospectively way such as by the human eye, in a Long-term outcomes of DP, which are
compared a custom-made orthotic de- mathematically rigorous way. Out- perhaps of greatest importance, are
vice against active repositioning in in- comes such as patient and parent not completely known. A large study by
fants younger than 4 months. Hel- satisfaction should also be studied Boere-Boonekamp et al41 revealed that
meted infants’ average CVA decreased and correlated to the quantitative 9.9% of 7609 Dutch infants had occipi-
from 11.0 mm to 3.5 mm, versus 9.0 outcomes. tal asymmetry on physical examina-
mm to 8.0 mm for repositioned infants Contemporary 3D imaging modalities, tion, and 45% of these had persistent
(P ⬍ .001). in combination with sophisticated shape asymmetry at a 2-year follow-up. Addi-
Other anthropometric studies have re- analysis techniques, are bridging the tional evidence of a lasting effect of in-
ported no statistically significant dif- gap between aesthetics and mathemat- fantile cranial molding is found in
ference between therapies. Loveday ics. The craniofacial community has rap- human remains from cultures in
and de Chalain20 prospectively studied idly adopted digital surface stereophoto- which intentional head molding took
74 infants and found no statistically grammetry, which rapidly acquires place.42– 45 This emphasizes the impor-
significant difference in CVA change detailed, high-resolution 3D studies and tance of achieving the best possible
between groups. The authors noted employs no harmful radiation. The data result during infancy because therapy
significant technical difficulties with has accuracy and precision equal to or is generally not efficacious past 12
the fit and function of the helmets in exceeding that of direct anthropome- months of age.13,14,18,31
this study that may have compromised try,37– 40 but avoids the difficulty and er- Treatment selection for individual
their results. Moss21 treated 66 infants ror associated with taking direct mea- patients should take into account
with mild to moderate plagiocephaly surements. The range of analytical more than efficacy. These treatments
(CVA of ⱕ1.2 cm) with repositioning techniques that can be applied to 3D present different demands to patients.
and physical therapy only. Moss re- data is vast, from simple methods Although active repositioning requires
ported an average CVA improvement such as linear distance calculation to daily, consistent involvement, and in
from 9.2 to 4.7 mm, which Moss stated 3D methods that analyze landmark- our series a longer treatment dura-
is equivalent to historical outcomes configurations or the entire surface. tion, helmets are costly and also in-
for helmeted infants. Analytical methods that use more sur- volve some inconvenience.
The variability in results of these stud- face points provide a richer model that As indicated by average pretreatment
ies suggests that the difference in out- encompasses more of the subtle con- cephalic indices, the infants in our
comes between orthotic helmets and tributions that details provide to study did tend to be somewhat
active repositioning is small, but it may shape. Complex shape characteristics brachycephalic, consistent with stud-
also reflect the difficulty in standardiz- such as contour, which are easily per- ies published previously.17 Our method
ing anthropometric head measure- ceived by the human eye, require so- focused on asymmetry in patients with
ment techniques. Both treatment mo- phisticated mathematical techniques DP and did not take brachycephalic
dalities are reasonably effective, as to quantify. To answer questions of deformity into account. This is a limita-
evidenced by the aforementioned stud- form in a rigorous, quantitative man- tion of our technique because posi-
ies as well as observational studies on ner, the craniofacial community must tional head shape deformity is multi-
the efficacy of helmet therapy14,31,32 continue to explore whole-shape anal- faceted and cannot be completely
and active repositioning,10,35,36 which ysis and its application to clinical prob- described by our measure. However,
judged efficacy by traditional anthro- lems. Currently, there exists no well es- asymmetry is a very important compo-
pometric measures31,32,36 and subjec- tablished framework for interpreting nent in the majority of cases of DP and
tive methods that include parent satis- mathematical values directly as clini- remains an important indicator of se-
faction with the aesthetic result.35,36 cal success or improvement in quality verity and outcomes.
Understanding aesthetic outcomes is of life. Additional studies that incorpo- Another limitation of this study is that
critical because the ultimate treat- rate parent and patient satisfaction, vi- the treatment group was dictated by
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