Infant Orthopedics and Facial Growth in Complete Unilateral Cleft Lip and

Palate Until Six Years of Age (Dutchcleft)

Catharina A.M. Bongaarts, D.D.S., Birte Prahl-Andersen, D.D.S., Ph.D., Ewald M. Bronkhorst, Ph.D., C. Prahl,
D.D.S., Edwin M. Ongkosuwito, D.D.S., Wilfred A. Borstlap, M.D., D.D.S., Ph.D., Anne M. Kuijpers-Jagtman, D.D.S.,
Ph.D.
Objective: To evaluate longitudinally the effect of infant orthopedics (IO) on
dentofacial cephalometric variables in unilateral cleft lip and palate (UCLP)
patients from 4 to 6 years of age.
Design: Prospective two-arm randomized controlled clinical trial in three cleft
palate centers in The Netherlands (Dutchcleft trial).
Patients: Fifty-four children with complete UCLP.
Interventions: Patients were divided randomly into two groups. Half of the
patients (IO+) had IO until surgical closure of the soft palate at the age of 652
weeks; the other half (IO2) received no intervention.
Mean Outcome Measures: Cephalometric values representing soft tissue,
hard tissue, and dental structures, measured on lateral headfilms made at 4 and
6 years of age.
Results: In the IO+ group, 21 patients were analyzed; in the IO2 group, 20
patients were analyzed at age 4 and 22 at age 6. No differences were found
between IO+ and IO2, except for two measurements: The interincisal angle was
larger and the mentolabial angle was smaller in the IO+ group.
Conclusions: For infants with UCLP whose surgical management included
soft palate repair at 12 months and delayed hard palate closure, cephalometric
outcomes at ages 4 and 6 provide no indication for the type of IO used in this
study.
KEY WORDS:

cephalometry, cleft palate, facial growth, infant orthopedics, multicenter, orthodontics, randomized clinical trial, treatment outcome

The characteristic face of an individual treated for a

complete unilateral cleft lip and palate was described by
Dahl in 1970 (aged 18 to 33 years). He found that the upper
face height was smaller compared with the control group;
the maxilla was short, and this was accentuated in the
dentoalveolar area as the result of retroclined incisors, and
greater height development was noted in the lower face.
More recently, Nollet et al. (2008) described the Nijmegen
unilateral cleft lip and palate (UCLP) sample born between
1976 and 1986 (aged 8 to 18 years). Cephalometrically,
both the maxilla and the mandible showed a retrusive facial
pattern, along with a rather hyperdivergent facial growth
pattern. The interincisal angle was obtuse, as was the
nasolabial angle (Nollet et al., 2008).
Besides the intrinsic deficiency, many iatrogenic factors
can influence facial growth. Repair of the lip, alveolar
process, soft palate, and hard palate may affect facial
growth, as may the surgeon himself, the timing of the
repair, patient-related factors such as scarring, or treatment
such as infant orthopedics (IO), which might affect facial
growth (Ross, 1987; Kuijpers-Jagtman and Long, 2000).
Many different types of infant orthopedic appliances
have been described. Some centers use active appliances,
others passive appliances. Active appliances are designed
with springs or screws to move the maxillary segments in

Dr. Bongaarts is Orthodontist, Department of Orthodontics and Oral

Biology, Radboud University Nijmegen Medical Center, The Netherlands. Dr. Prahl-Andersen is Emeritus Professor in Orthodontics,
Academic Center of Dentistry Amsterdam, and Former Head of the
Department of Orthodontics, Erasmus MC University Medical Center
Rotterdam, The Netherlands. Dr. Bronkhorst is Biostatistician, Department of Preventive and Curative Dentistry, Radboud University Nijmegen Medical Center, The Netherlands. Dr. Prahl is Orthodontist,
Department of Orthodontics, Academic Center for Dentistry Amsterdam,
Amsterdam, The Netherlands. Dr. Ongkosuwito is Orthodontist, Craniofacial Center Rotterdam, Erasmus Medical Center, Rotterdam, The
Netherlands. Dr. Borstlap is Maxillofacial Surgeon, Department of Oral
and Maxillofacial Surgery, Radboud University Nijmegen Medical
Centre, Nijmegen, The Netherlands. Dr. Kuijpers-Jagtman is Professor
and Chairperson, Department of Orthodontics and Oral Biology, and
Head of the Cleft Palate Craniofacial Unit, Radboud University Nijmegen
Medical Center, The Netherlands.
This research is part of the Dutch intercenter study into the effects of
infant orthopedic treatment in complete UCLP (Dutchcleft), carried out in
a collaboration between the Cleft Palate Centers of the Radboud University
Nijmegen Medical Center, Academic Center of Dentistry in Amsterdam,
and Erasmus MC University Medical Center in Rotterdam (coordinating
orthodontists, A.M. Kuijpers-Jagtman and B. Prahl-Andersen).
Submitted September 2008; Accepted March 2009.
Address correspondence to: Dr. Anne Kuijpers-Jagtman, Radboud
University Nijmegen Medical Center, Department of Orthodontics and
Oral Biology, 309 Tandheelkunde, P.O. Box 9101, 6500 HB Nijmegen,
The Netherlands. E-mail orthodontics@dent.umcn.nl.
DOI: 10.1597/08-034.1
654

Bongaarts et al., EFFECT OF INFANT ORTHOPEDICS IN UCLP 655

the desired direction. Passive appliances induce arch

alignment during growth by grinding away material of
the plate. Also described is a plate made on a reconstructed
cast to move maxillary segments in a predetermined
position. Beside these appliances, external strapping across
the cleft can be part of the treatment protocol. Also,
nasoalveolar molding is described. It is said to reshape and
reposition anatomic structures to achieve more symmetric
relationships between nasal cartilages, columella, and
alveolar segments (Grayson and Cutting, 2001; Da Silveira,
et al., 2003; Kozelj, 2007).
Because a lot of different techniques for IO have been
described, it is difficult to compare treatment results. More
important, treatment for a cleft patient consists of more than
IO alone; all steps in treatment may have an influence on facial
morphology. It is impossible to separate these steps in
treatment when comparing results in retrospective research
(e.g., Ross, 1987; Mlsted et al., 1992). Because these studies
and many others are retrospective and show conflicting results,
Dutchcleft was started; this was a prospective, randomized
clinical trial that was performed in three cleft palate centers in
The Netherlands (Kuijpers-Jagtman and Prahl-Andersen,
2006). In this project, passive appliances were used.
The purpose of the portion of the trial presented here was
to evaluate longitudinally the effects of IO on dentofacial
cephalometric variables in UCLP patients from 4 to 6 years
of age. The hypothesis is that the cephalometric outcome of
the IO+ group is better than that in the IO2 group.
PATIENTS AND METHODS
This study was designed as a prospective, two-arm,
randomized, controlled clinical trial in the cleft palate
centers in Nijmegen, Amsterdam, and Rotterdam, in The
Netherlands. Local ethical committees approved the study
protocol. Inclusion criteria included the following: complete UCLP, infants born at term, both parents Caucasian
and fluent in the Dutch language, and trial entrance within
2 weeks after birth. Exclusion criteria consisted of soft
tissue bands and other congenital malformations. Figure 1
shows the sample until the age of 6, along with the reasons
for exclusion from evaluation. When parents agreed to
participate in the study, they were asked to sign an
informed consent. Between 3 and 6 months of age, all
included children were confirmed by the geneticist on their
own CLP team as being nonsyndromic.
In a previous publication, a detailed description was given
with respect to experimental design, treatment assignment,
treatment protocol, and operators (Prahl et al., 2001). A
summary of the most important issues was provided.
Treatment
Half of the patients were treated with infant orthopedics
by means of passive plates until surgical soft palate closure
(n 5 27), and half did not get a plate (n 5 27). The plates

FIGURE 1 Flow diagram of trial children with reasons for exclusion

of evaluation.

were made on a plaster cast using compound soft and hard

acrylic. The IO+ children had their plates adjusted every 3
weeks to guide the maxillary segments, by grinding at the
cleft margins; maxillary growth and emergence of deciduous teeth indicated the necessity for a new plate. After
surgical lip closure, the plate was replaced the same day.
Checkups were planned every 4 to 6 weeks following lip
surgery. The plate was maintained until soft palate closure
was achieved. The IO2 group visited the clinic at 6 weeks,
and before and after lip surgery and soft palate closure. In
both groups, lip surgery was performed at the age of 18
weeks by the Millard technique. At lip surgery, the cleft
teams of Amsterdam and Nijmegen used McCombs
technique for the nose; the Rotterdam cleft team preferred
their own method, which combined McCombs with
Pigotts technique. Soft palate surgery was performed at
the age of about 52 weeks according to a modified Von
Langenbeck method that included levator muscle repositioning. In the studied age period (until 6 years of age),
other interventions were performed if indicated: pharyngoplasty (n 5 22), lip revision (n 5 13; in all cases, performed
before the age of 4 years), facial mask treatment (n 5 1),
plate to facilitate speech (n 5 15), and closure of the
anterior palate (n 5 6). These interventions were distributed equally over the IO+ and IO2 groups.

656 Cleft PalateCraniofacial Journal, November 2009, Vol. 46 No. 6

TABLE 1

Descriptions of Landmarks and Measurements

Landmark

Name

Description

N
S
Ar

Nasion
Sella
Articulare

Ptm

Pterygomaxillary fissure

Pg
Me
Gn
Pr
A

Pogonion
Menton
Gnathion
Prosthion
Point A

ANS
PNS

Anterior nasal spine

Posterior nasal spine

B
Go

Point B
Gonion

As
Ai
Is
Ii
ID
m2s
m2i
N9
G
Sn

Apex superior
Apex inferior
Incision superior
Incision inferior
Infradentale
Maxillary second deciduous molar
Mandibular second deciduous molar
Soft tissue nasion
Glabella
Subnasale

B9
Ls
Li
Pg9
PRN

Soft tissue point B

Labrale superior
Labrale inferior
Soft tissue pogonion
Pronasale

The most anterior limit of the frontonasal suture

The geometric center of the sella turcica
The point of intersection of the projection of the dorsal contours of the processus articularis
mandibulae and the pharyngeal part of the clivus
The lowest point on the pterygomaxillary fissure between the anterior margin of the pterygoid process
and the posterior margin of the maxillary tuberosity
The most anterior point on the chin of the mandible
The most inferior point on the symphysis of the mandible, relative to the mandibular border
A point midway between Pg and Me on the outline of the symphysis
The point of the maxillary alveolar process in the midline that projects most anteriorly
The deepest point on the anterior contour of the upper alveolar process above the tooth germs of the
permanent incisors
The tip of the bony anterior nasal spine
The posterior end of the hard palate, if visible. Otherwise at the point of intersection of the dorsal
maxillary contour and the soft palate
The deepest point on the contour of the mandible between infradentale (ID) and pogonion (Pg)
The intersection point of the outer contour of the mandible with the bisectrice of the angle formed by
the mandibular border and the tangent to the ramus from the articulare projected on the
mandibular border
The apex of the root of the most prominent maxillary central incisor
The apex of the root of the most prominent mandibular incisor
The incisal point of the most prominent maxillary central incisor
The incisal point of the most prominent mandibular incisor
The most anterior-superior point on the mandibular alveolar process
The mesiobuccal cusp of the maxillary second deciduous molar
The mesiobuccal cusp of the mandibular second deciduous molar
The deepest point of the soft tissue contour in the region of the nasofrontal suture
The most prominent point in the midsagittal plane of the forehead
The deepest point of the subnasal curvature relative to a line from the nose tip (PRN) to the upper lip
(Ls)
The point of the greatest concavity at the midline of the lower lip between Li and Pg9
The most prominent point of the vermilion border of the upper lip
The most prominent point of the vermilion border of the lower lip
The most anterior point of the soft tissue of the chin in the midsagittal plane
The most anterior point of the tip of the nose

Variables

SNA angle
SNB angle
ANB angle
Mentolabial angle
Upper and lower lip thickness
Nose angle
Facial convexity angle
Nasolabial angle
Upper and lower lip to E-plane
Upper and lower lip protrusion
Interincisal angle
Lower incisorGo-Me angle
Upper and lower incisor to APg
Upper incisorANS-PNS angle
N-ANS-Pg angle
Facial height index
ANS-PNS/Go-Me index
ANS-PNS
ANS-Me/N-Me index
ANS-Me
SNGo-Me angle
Occlusal planeSN angle
ANS-PNSSN angle

Description

Anterior-posterior position of point A in relation to the cranial base

Anterior-posterior position of point B in relation to the cranial base
The relative position of point A and B to each other
Deepness of the mentolabial fold (Pg9-B9-Li)
Thickness in mm (Pr-Ls and ID-Li)
Protrusion of the nose (PRN-N9-Sn)
Soft tissue convexity (G-Sn-Pg9)
Upper lip protrusion relative to the columella line (PRN-Sn-Ls)
Soft tissue balance between the lip and the E-line in mm
Lip protrusion in relation to Sn-Pg9 in mm
Inclination of upper and lower incisors relative to each other
Inclination of the lower incisor relative to the mandibular plane
Position of the incisors to A-Pg in mm
Inclination of the upper incisor relative to the palatal plane
Convexity of the face according to Harvold
The ratio of the posterior face height relative to the anterior face height (%)
The ratio of the maxillary length relative to the mandibular length (%)
Distance from ANS to PNS in mm
The ratio of the lower anterior face height relative to the total anterior face height (%)
Distance from ANS to Me
Inclination of the mandibular plane relative to the cranial base
Inclination of the occlusal plane relative to the cranial base
Inclination of the palatal plane relative to the cranial base

Data Acquisition
Lateral head films were obtained with the patient
positioned in a cephalostat and oriented toward the
Frankfort horizontal plane. Patients were instructed to
have the lips in a relaxed closed position when the x-ray

was taken. All lateral head films were traced by hand by

one observer; 18 lateral head films were traced twice, with
an approximate time interval of 2 months. A second
observer traced 18 lateral head films that were also traced
by the first observer. The 18 head films were randomly
selected from the 4- and 6-year samples. Both observers

Bongaarts et al., EFFECT OF INFANT ORTHOPEDICS IN UCLP 657

TABLE 2

Sample Characteristics*
IO+ (n 5 27)

Gender: male/female, n
Side of cleft: left/right, n
Patients per treatment
center: 1/2/3, n
Age 4-year cephs, yr
Age 6-year cephs, yr

IO2 (n 5 27)

20/7
17/10
7/11/9

21/6
18/9
7/10/10

Mean: 4.0
Range: 3.84.2
Mean: 6.1
Range: 6.06.3

Mean: 4.0
Range: 3.94.4
Mean: 6.0
Range: 5.96.5

P10
Age at trial entrance, days
Birth weight, g
Cleft width at birth, mm
Age lip repair, days
Age soft palate closure, days

P50 P90 P10 P50

P90
0
3
7
1
6
13
2660 3350 4020 2920
3600
4280
9.5 12.5 14.4
8.6
12.4
16.4
117 127 142 117
125
138
355 375 438 301
367
389

* Some variables are presented in percentiles because of skewness (P10, P50, and P90).

marked the landmarks on their own tracings, independently

of each other.
The landmarks and definitions used are listed in Table 1
and Figures 2A and 2B. Tracings were scanned on a flatbed
scanner (Linotype-Hell AG, type H391, Eschborn, Germany). The landmarks were digitized on scanned images
using Viewbox, version 3.1.0.5 (dHal Orthodontic Software, Athens, Greece), and angular, linear, and ratio
variables, as listed in Table 1, were calculated. All
measurements were recalculated to life-size measurements:
the magnification factor was 3.93% for Nijmegen and
3.83% for Amsterdam and Rotterdam. Direct scanning of
the cephalograms led to more errors because of the dark
area that often occurred near the cleft. Landmarks were
better identifiable on the original radiograph and tracing.
In Dutchcleft, occlusion and esthetic results were also
evaluated at age 6. The occlusion was studied on study
models with use of the 5-year-old index. The index is a 5point scale that uses 1 for excellent cases (no open bite or
crossbite; a positive overjet) and 5 for very poor cases
(crossbite on both sides, reversed overjet and poor arch
form). The esthetics was scored with a visual analogue scale
(VAS) on facial photographs. Both full faces and photographs showing the nasolabial part only were scored by
professionals and laymen. The findings are described in
Bongaarts et al. (2004 and 2008b). In the present study,
occlusal scores and facial esthetic scores were combined
with cephalometric measurements to explain possible
effects.
Statistical Analyses

FIGURE 2 A: Tracing of the hard tissue points. B: Tracing of the soft

tissue points.

Intraobserver and interobserver duplicate measurement

errors were calculated for all cephalometric measurements.
Paired t-tests showed the systematic errors. The reliability
coefficients were calculated as Pearson correlation coefficients, and duplicate measurement errors were calculated
meaning standard deviation/! 2 (in millimeters,
by St:pDev:
2
degrees, and %).

658 Cleft PalateCraniofacial Journal, November 2009, Vol. 46 No. 6

TABLE 3

Intraobserver and Interobserver Duplicate Measurement Errors Were Calculated for All Cephalometric Measurements
Interexaminer

Variable

p (mean)

SNA angle
SNB angle
ANB angle
Mentolabial angle
Upper lip thickness
Lower lip thickness
Nose angle
Facial convexity angle
Nasolabial angle
Upper lip to E-plane
Upper lip protrusion
Lower lip to E-plane
Lower lip protrusion
Interincisal angle
Lower incGoMe angle
Lower inc to APg
Upper inc to APg
Upper incANS-PNS angle
N-ANS-Pg angle
Facial height index
ANS-PNS/GoMe index
ANS-PNS
ANS-Me/N-Me index
ANS-Me
SNGoMe angle
Occl planeSN angle
ANS-PNSSN angle

.002**
.087
.005**
.512
.758
.406
.301
.559
.094
.061
.384
.293
.965
.180
.070
.003**
.005**
.151
.124
.003**
.462
.078
.171
.284
.044*
.024*
.694

(1.43)
(0.41)
(0.72)
(1.36)
(2.07)
(2.12)
(2.48)
(2.17)
(3.04)
(2.26)
(2.13)
(2.13)
(0.01)
(22.53)
(21.31)
(2.59)
(2.61)
(2.65)
(0.62)
(21.07)
(2.97)
(0.86)
(2.63)
(2.42)
(0.55)
(1.89)
(0.33)

Intraexaminer

Error

1.15
0.62
0.58
6.09
0.64
0.41
1.36
0.84
5.14
0.38
0.45
0.37
0.37
4.90
2.03
0.44
0.51
5.28
1.04
0.81
3.53
1.38
1.20
1.03
0.68
2.05
2.50

.913
.958
.955
.804
.880
.974
.734
.979
.830
.983
.963
.984
.977
.799
.887
.892
.954
.653
.888
.908
.665
.609
.655
.900
.972
.827
.403

P (mean)

.982
.604
.625
.200
.549
.777
.452
.851
.007**
.547
.191
.920
.736
.293
.441
.107
.733
.534
.219
.158
.021*
.000***
.558
.792
.441
.701
.271

(2.01)
(0.07)
(2.10)
(2.13)
(0.11)
(0.04)
(0.24)
(.053)
(4.03)
(2.07)
(2.19)
(2.01)
(2.04)
(22.73)
(0.51)
(2.19)
(2.08)
(1.49)
(0.33)
(2.37)
(2.02)
(1.41)
(0.17)
(0.07)
(0.14)
(2.19)
(2.57)

Error

0.70
0.43
0.62
4.92
0.56
0.45
0.95
0.85
4.12
0.37
0.44
0.32
0.33
7.75
2.00
0.34
0.70
7.26
0.81
0.78
2.46
0.98
0.87
0.84
0.56
1.54
1.56

.965
.986
.963
.855
.952
.974
.862
.978
.940
.984
.971
.989
.985
.683
.914
.947
.932
.497
.943
.930
.840
.804
.815
.925
.987
.888
.789

Paired t-tests showed the systematic errors. The reliability coefficients were calculated as Pearson correlation coefficients, and duplicate measurement errors were calculated by
millimeters, degrees, and %).
p values: * .05 $ p . .01; ** .01 $ p . .001; *** p $ .001.

Also, the effect of IO was tested at 4 and 6 years of age

with two tailed t tests. The significance is shown by the p
value. The increment shows longitudinal results.
Finally, regression analyses were done to test the influence
of cephalometric values, gender, and occlusion at 6 years of
age on the overall esthetic result at age 6. p values and their
effects are given to demonstrate any influence, and the R
square is given to show how much variance in the esthetic
result can be explained by each of these items.

St:pDev:

(in

largest errors are found in measurements involving point A

or ANS, or the soft tissues. The reliabilities were good to
acceptable, except for two measurements: upper incisor to
ANS-PNS angle (r 5 .497 for intraobserver agreement, and r
5 .653 for interobserver agreement) and ANS-PNS SN
angle (r 5 .789 for intraobserver agreement, and r 5 .403 for
interobserver agreement). These two measurements were
excluded from further analysis.
Treatment Effect

General

RESULTS

At intake, 54 patients participated in the study. An

overview of the sample characteristics is given in Table 2.
Two IO+ children hardly used the plate; in one case, the
plate was worn by mistake until 78 weeks. These children
remained in the IO+ group according to the intention-totreat principle: Patients are analyzed according to the
treatment group to which they were randomized, whether
they received the treatment or not. The mean duration of
IO was 50 weeks; SD was 16 weeks. The flow diagram in
Figure 1 shows the reasons for nonevaluation.
Reliability of Measurements
Table 3 shows the reliability coefficients and the measurement errors in millimeters, degrees, or percentage. The

In Table 4, the effect of IO is shown. Only two significant

differences were found between IO+ and IO2: At the age of
4 years, the interincisal angle was about 9 degrees larger in
the IO+ group. At the age of 6 years, no significant
difference could be measured any longer. The other
significant difference occurred at the age of 6: The
mentolabial angle was almost 9 degrees smaller in the
IO+ group.
For all cephalometric variables and the 5-year index,
regression analyses were done to assess the relation between
these variables and the esthetic score at 6 years. To show
the extent of the effect that a certain factor has, the esthetic
results and the 5-year index scores are given in Table 5
(Bongaarts et al., 2004 and 2008b). Regression analyses
were repeated with gender as the co-variable, and also with
pharyngoplasty and gender as co-variables. However,
because a significant influence of gender or pharyngoplasty

Bongaarts et al., EFFECT OF INFANT ORTHOPEDICS IN UCLP 659

TABLE 4

Number (n), Mean, and SD of Measurements Given for IO+, IO2 at the Age of 4 and 6 Years*
IO+

Variable

SNA angle

SNB angle

ANB angle

Mentolabial angle

Upper lip thickness

Lower lip thickness

Nose angle

Facial convexity angle

Nasolabial angle

Upper lip to E-plane

Upper lip protrusion

Lower lip to E-plane

Lower lip protrusion

Interincisal angle

Lower incGoMe angle

Lower inc to APg

Upper inc to APg

N-ANS-Pg angle

Facial height index

ANS-PNS/GoMe index

ANS-PNS

ANS-Me/N-Me index

ANS-Me

4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc
4y
6y
inc

IO2

Mean

SD

Mean

21
21
19
15
15
9
15
15
9
21
21
19
19
18
16
19
18
16
21
21
19
21
21
19
21
21
19
21
21
19
21
21
19
21
21
19
21
21
19
15
15
9
21
21
19
15
15
9
15
15
9
15
15
9
15
15
9
15
15
9
21
21
19
14
12
8
14
12
8

84.33
82.14
21.42
75.61
75.22
0.12
9.27
7.65
21.64
55.39
49.42
26.63
211.03
210.69
2.13
213.15
212.59
.08
19.72
19.87
.68
11.03
10.33
21.30
109.99
114.38
3.11
20.15
21.66
21.67
4.07
3.06
21.00
1.45
0.43
2.95
3.82
3.17
2.43
170.42
164.50
25.16
81.96
81.71
.43
22.35
22.07
2.21
20.72
20.93
2.08
11.24
10.28
21.36
61.61
60.44
2.08
82.93
82.03
21.53
41.38
44.43
2.62
56.43
56.42
2.06
48.77
53.34
3.85

4.16
4.14
1.61
3.46
3.68
1.83
2.27
2.25
1.47
14.27
16.54
21.81
1.94
2.07
1.63
2.13
2.92
2.38
2.52
2.39
2.07
4.91
5.86
2.07
9.25
12.42
11.27
1.53
2.58
1.66
1.29
2.13
1.54
2.21
3.16
2.36
2.18
2.86
2.04
9.97
11.10
14.28
4.99
5.64
3.63
1.72
1.94
1.07
1.71
1.58
.75
3.13
2.80
1.93
3.42
2.92
3.15
5.44
6.49
6.07
1.78
2.00
2.59
2.55
3.13
2.02
3.52
4.78
1.94

20
20
18
17
16
12
17
16
12
19
22
17
18
21
17
18
21
17
19
22
17
19
22
17
19
22
17
19
22
17
19
22
17
19
22
17
19
22
17
18
16
12
20
22
18
17
16
12
17
16
12
17
16
12
17
16
12
17
16
12
20
21
17
17
15
12
17
15
12

83.31
83.43
2.86
74.05
74.43
2.13
8.88
8.65
21.18
62.47
58.27
26.35
210.61
210.85
2.47
212.42
213.11
2.66
18.42
19.42
1.03
9.85
10.10
.05
115.10
116.77
.77
20.34
21.33
21.01
3.50
3.19
2.20
1.85
1.06
2.77
3.91
3.59
2.16
161.48
163.09
2.02
85.03
83.86
21.17
21.34
22.47
2.53
0.13
20.41
2.83
10.68
10.43
21.46
61.60
62.35
.30
84.89
86.33
.76
40.26
44.41
4.03
56.27
56.67
2.21
47.69
52.26
4.04

SD

p Value

3.40
3.98
2.53
2.46
3.64
1.60
3.83
3.55
2.06
17.51
11.59
14.85
1.97
1.56
1.76
2.68
2.12
2.20
2.44
2.54
2.68
5.39
5.72
2.49
10.45
10.76
7.57
2.69
2.68
1.46
1.93
1.99
1.27
2.02
2.43
2.24
1.85
2.03
1.85
12.33
11.71
16.41
6.27
5.01
5.47
2.04
2.05
.78
1.65
2.05
1.60
3.98
3.34
2.67
1.41
2.71
1.88
7.75
7.32
6.25
2.63
2.94
1.94
1.74
2.91
2.21
2.42
4.33
2.80

.397
.306
.421
.149
.550
.745
.733
.362
.576
.168
.048*
.964
.513
.780
.563
.367
.524
.353
.108
.554
.666
.473
.898
.086
.110
.503
.474
.777
.683
.219
.278
.833
.104
.550
.463
.814
.894
.585
.685
.012*
.432
.462
.090
.196
.298
.144
.597
.438
.163
.438
.208
.663
.893
.923
.994
.070
.736
.420
.095
.411
.118
.976
.075
.839
.827
.883
.320
.543
.869

660 Cleft PalateCraniofacial Journal, November 2009, Vol. 46 No. 6

TABLE 4

Continued
IO+

Variable

SN-GoMe angle

4y
6y
inc
4y
6y
inc

Occl planeSN angle

IO2

Mean

SD

Mean

15
15
9
15
15
9

37.82
39.29
2.13
2159.41
2161.07
2.72

3.40
3.71
2.31
4.57
5.21
3.31

17
16
12
17
16
12

38.53
37.65
2.25
2161.04
2163.18
2.10

SD

p Value

2.22
3.44
1.78
5.04
5.85
4.01

.565
.211
.897
.350
.299
.709

{ Differences between IO+ and IO2 were tested with t-tests. The level of significance is indicated by p values. Also, the increment (inc) is given.
* .05 $ p . .01.
** .01 $ p . .001.
*** p $ .001.

was never found (all ps are .17 or higher), only the results
for the univariate regression analyses are shown in Table 6.
Because the highest R square is .083, these measurements
explain the esthetic result only to a minimal extent (not
more than 8.3%). For the 5-year index for occlusion, the R
square is .043, which means that it explains not more than
4.3% of the esthetic result. One point difference in score in
the 5-year index means a 2.66-point reduction in esthetic
results. The number of children is not big enough to allow
for multiple regression using all variables. No clear-cut rule
can be used to decide which variables are most likely to
influence the esthetic score. Therefore, from all univariate
regression models, seven variables with the highest R2 were
included in a backward regression to look for combinations
of variables with better potential for explaining the value of
the esthetic score. This backward regression model
eliminated all but one of the variables, leaving only the
Facial Height Index. This indicates that a combination of
variables does not improve the potential for explaining the
value of the esthetic score at the age of 6.

of the landmark identification, and the observer are

important factors in the uncertainty within landmark
positioning (Baumrind and Frantz, 1971; Houston, 1983;
Marci and Athanasiou, 1995; Trpkova et al., 1997).
Differences in the magnitude of the measurement error
are caused by the precision of landmark identification and
the amount of noise in adjacent structures. Also, the
nonerupted and often rotated incisors and the displaced
and reduced size of the premaxilla can be mentioned as
causes of measurement errors in young cleft patients. As
was described by Atherton in 1967, a marked shift of the
premaxillary region occurs away from the centerline. The
premaxilla of the cleft side is reduced in size and displaced
forward. In a recent study (Bongaarts et al., 2008a),
alternatives for point A, ANS, and PNS were evaluated
in toddlers, but these alternatives were not better than the
traditional landmarks. Therefore, the traditional landmarks were used in the present study.
The errors in the present study were acceptable, given the
age of the investigated group in which shedding of the
incisors is taking place, and the difficulties associated with
locating the essential points A and ANS. Although an error
was present, this error was never as big as, or larger than,
the standard deviation (SD) of the measurements. By first
tracing and identifying the landmarks and than scanning

DISCUSSION
The error in landmark identification is the major source
of cephalometric error. The type of landmark, the precision

TABLE 5 Number (n), Mean, and SD of Esthetic Scores Given for Full Face Photographs, Nasolabial Photographs for IO+ (bold italics) and IO2 at Ages 4
and 6 Years{
4 yr
Variable

Full face

Professional
Layman

Nasolabial

Professional
Layman

5-y-index

6 yr

Mean

(SD)

p*

Mean

(SD)

p*

21
24
21
24
21
24
21
24
22
21

94.18
105.27
89.75
99.10
93.06
95.98
91.20
95.16
1.98
2.01

(12.01)
(13.94)
(11.65)
(14.22)
(13.50)
(13.09)
(12.50)
(10.98)
(0.81)
(0.73)

.006**

24
22
24
22
24
22
24
22
21
20

95.21
100.63
96.19
100.71
96.85
105.41
96.13
103.05
2.16
2.23

(11.04)
(9.47)
(9.86)
(11.19)
(11.78)
(14.57)
(13.35)
(14.25)
(0.85)
(0.84)

.08

.02*
.47
.27
.89

.15
.04*
.10
.80

{ Differences between IO+ and IO2 were tested with t-tests. Level of significance is indicated by p values ( p*). The occlusion scored with the 5-year index is also provided. n may vary because
of incidental missing values.
* .05 $ p . .01.
** .01 $ p . .001.
*** p $ .001.
The information given in this table is described in Bongaarts et al. (2004) and Bongaarts et al. (in press).

Bongaarts et al., EFFECT OF INFANT ORTHOPEDICS IN UCLP 661

TABLE 6 Relation Between Occlusion at 6 Years of Age and Cephalometric Values at Age 6 (Independent Variables) With Overall Esthetics of
the Patient (Dependent Variable){
Overall Esthetics
P Value

(95% CI)

R Square

.860
.388
.304
.718
.692
.171
.847
.582
.314
.826
.655
.370
.383
.430
.182
.331
.379
.361
.117
.615
.379
.321
.881
.142
.510
.178
.607

2.072
2.512
.749
2.042
2.371
.952
2.131
2.164
2.140
.141
.361
2.530
2.589
.151
2.427
21.047
21.174
.662
21.142
.148
.522
2.808
2.082
.889
.253
22.660
1.929

(2.898, .753)
(21.708, .684)
(2.714, 2.212)
(2.275, .191)
(22.249, 1.507)
(2.429, 2.333)
(21.488, 1.227)
(2.761, .433)
(2.417, .137)
(21.152, 1.435)
(21.256, 1.978)
(21.712, .651)
(21.935, .758)
(2.235, .537)
(21.061, .206)
(23.215, 1.121)
(23.861, 1.513)
(2.798, 2.123)
(22.587, .304)
(2.448, .744)
(2.644, 1.709)
(22.451, .834)
(21.202, 1.038)
(2.315, 2.093)
(2.523, 1.030)
(26.575, 1.255)
(25.590, 9.449)

.001
.026
.036
.003
.004
.049
.001
.007
.024
.001
.005
.019
.018
.022
.042
.033
.027
.029
.083
.009
.019
.039
.001
.073
.015
.043
.006

SNA angle
SNB angle
ANB angle
Mentolabial angle
Upper lip thickness
Lower lip thickness
Nose angle
Facial convexity
Nasolabial angle
Upper lip to E-plane
Upper lip protrusion
Lower lip to E-plane
Lower lip protrusion
Interincisal angle
Lower incGoMe angle
Lower inc to APg
Upper inc to APg
N-ANS-Pg
Facial height index
ANS-PNS/GoMe index
ANS-PNS
ANS-Me/N-Me index
ANS-Me
SN-GoMe angle
Occl planeSN angle
5-year index
Gender

{ Results of regression analysis: p values and the effects (B and the 95% confidence interval) are given. Also, the R square is given to show how much of the esthetic result can be explained by
each of these items.
* .05 $ p . .01.
** .01 $ p . .001.
*** p $ .001.

the tracing, an extra error was added. Direct scanning of

the cephalograms led to additional errors because of the
dark area that often occurred near the cleft. Landmarks
were better identifiable on the original radiograph and
tracing. The extra error that was added by scanning and
digitalizing landmarks is minimal. In the study of
Bongaarts et al. from 2008a, the error for digitalization
was 0.25 for SNA compared with a tracing error of 0.73.
In the Dutchcleft study, the sample size calculation was
based on a detectable IO effect of 3 degrees for the SNA
angle at the age of 4 years. An assumption was made for the
SD: 3.5 degrees. The minimum number of children was
found to be 23 in each group. The study started with 27
patients in each group. The number of patients involved in
the study decreased as the result of Simonarts bands and
missing records or records not taken within 6 months
before or after the birthday of the child (Fig. 1). With the
TABLE 7

results that we found, the power was recalculated for a few

variables to check whether patient groups were large
enough to reveal a possible effect of IO (Table 7). Power
was set at 80%, and the level of significance was .05, as was
done for the initial calculations. The table shows that the
IO+ and IO2 groups were large enough to show significant
differences, if any were present.
In a prospective trial in which CLP patients are followed
over a long period, it is unavoidable that different people
are involved and additional interventions are performed.
None of these subgroups was segregated out because extra
interventions were equally distributed over the IO+ and
IO2 groups. Because the interventions were equally spread
over the two groups, they did not interfere with the
objective of the study; although, they are a source of
variability. Because of this, one should be cautious when
interpreting the results.

New Power Calculations Based on Results

Original analysis
New calculation
New calculation
New calculation
New calculation

Measurement

Age, yr

Expected Size
if Effect on IO

From Results of
Research: Found SD

Number of Children Necessary

in Each Group

SNA
SNA
ANB
5-year index
Esthetic score

4
6
6
6
6

3
2
2
0.9
10

Assumption: 3.5
2
1.75
1
10

23
17
13
14
17

662 Cleft PalateCraniofacial Journal, November 2009, Vol. 46 No. 6

No clinically relevant effect of IO on facial growth was

found. These findings contradict those of other studies, in
which a positive influence of IO was described (Hotz and
Gnoinski, 1976; Gnoinski, 1990). In Eurocleft (Mlsted et
al., 1992) and in the studies of Ross (1987), no significant
effects of IO were found, but all were nonrandomized
retrospective studies. Also, the results of this randomized
clinical trial are valid only for the passive type of appliance;
it is impossible to draw conclusions about active plates or
appliances with extensions for nasal molding. For conclusiveness regarding these appliances, another clinical trial
should be set up.
Because the regression analyses in this study and those in
the study about the esthetic result of the Dutchcleft trial
(Bongaarts et al., 2008b) show almost no significant
findings until 6 years of age, the facial esthetics of young
patients with CLP is probably influenced by factors other
than treatment or growth variables. Possibly, facial
expression, texture of the skin, and color of eyes or hair
exert a larger influence than would be expected. Of course it
is possible that at a later age, a relation can be found
between esthetics and other measured factors because the
small, insignificant variations found now might become
more pronounced after the pubertal growth.
The first results of Dutchcleft showed that IO had a
temporary effect on maxillary arch dimensions, which did
not last beyond surgical soft palate closure (Prahl et al.,
2001; Bongaarts et al., 2006). Also, IO could not prevent
collapse of the maxillary arch (Prahl et al., 2003; Bongaarts et
al., 2006). In the occlusion, measured with the 5-year index at
the ages of 4 and 6 years, no differences between IO+ and
IO2 could be shown (Bongaarts et al., 2004). Feeding and
the nutritional status of the infants were not improved by IO
(Prahl et al., 2005). Data published in 2004 show the costeffectiveness of the speech outcome at the age of 2.5 years:
Listeners (speech therapists) were asked to rate speech quality
on a 10-point scale of 10 IO+ children and 10 IO2 children.
The IO+ group had a significantly better rating for speech.
The resulting cost-effectiveness ratio was 1041 euro for
1.34 point of speech improvement (Konst et al., 2003a;
Konst et al., 2004). More detailed speech findings have been
published elsewhere (Konst et al., 1999; Konst et al., 2000;
Konst et al., 2003b; Konst et al., 2003c). An evaluation of
speech data at the age of 6 still has to be performed. Finally,
the results of the esthetic scores at ages 1K and 4 to 6 years
showed no relevant effect of IO on facial appearance (Prahl et
al., 2006; Bongaarts et al., 2008b).
CONCLUSION
Given all results of Dutchcleft up to now, there is no
indication for the type of IO used in this study for infants
with UCLP, whose surgical management included soft
palate repair at 12 months and delayed hard palate closure.
Those who are promoting different methods of IO
including nasoalveolar molding should consider the long-

term benefits of their interventions by using the same

rigorous methodology as was applied in Dutchcleft.
Acknowledgment. The authors would like to thank Prof. H. Boersma for
being the second tracer and I.V. Dirks for her help in scanning all
cephalograms.

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