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doi:10.1093/ejo/cjt009
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SystematicReview
Indices to assess malocclusions in patients with cleft lip and palate
MostafaAltalibi, HumamSaltaji, RyanEdwards, Paul W.Major and CarlosFlores-Mir
School of Dentistry, Faculty of Medicine and Dentistry, University of Alberta, Edmonton, Canada
Corespondence to: Mostafa Altalibi, Orthodontic Graduate Program, University of Alberta, 5528 Edmonton Clinic
Health Academy, Edmonton, Alberta, Canada, T6G 1C9. E-mail: mostafa.altalibi@gmail.com
Introduction
Cleft lip and/or palate (CLP) is the most common congenital malformation in the head and neck and represents
a group of heterogeneous disorders that affect between one
and seven out of 1000 newborns (Vanderas, 1987). These
disorders can affect the face and the oral cavity. Moreover,
patients with this condition may exhibit an array of functional and aesthetic co-malformations, in addition to those
malformations specific to the CLP by definition, ranging
from a mid-face deficiency and a constricted maxillary
dental arch to congenitally missing and malformed teeth
(Vargervik, 1981; Mossey et al., 2009). The sum of these
malformations gives rise to a malocclusion that can be
extremely difficult to treat at times; thus, treatment is largely
through a multidisciplinary approach (Evans, 2004).
This comprehensive treatment plan can prove to be a
large burden on the public health sector, and since CLP
manifests as a spectrum of severities (Gorlin etal., 2001), it
was deemed by the government-funding agencies that there
needs to be a system to judge the severity of the malocclusions. Certain indices, having already been used for
other purposes, were applied to attempt to give an objective
measure of the severity of the malocclusion in CLP patients
(Parker, 1998; Hong etal., 2001).
Many indices are now available, each with different
criteria and uses, and although it has been quite some time
since the introduction of these indices, there is no consensus
as to which index should be used for CLP populations
(Parker, 1998; Hong et al., 2001). Furthermore, because
each index arose due to specific related circumstances,
not all the indices should be used for patients with CLP.
This ambiguity makes the task of choosing an appropriate
index to report CLP malocclusions daunting, be it to assess
the severity of malocclusions for research, governmentfunding purposes, or prognosis of surgical and orthodontic
treatment in patients with CLP. According to the World
Health Organization (WHO), the ideal index should excel
in the following criteria: reliability, validity and acceptance
Background: Several indices are now available to assess the severity of the malocclusion in cleft lip
and/or palate (CLP) patients; and although it has been quite some time since the introduction of these
indices, there is no consensus as to which index should be used for CLP populations.
Objective:To systematically review the available literature on the indices used to assess the occlusal
schemes in dental models of CLP patients, with respect to the most commonly used index and the index
that most fulfils the World Health Organization (WHO) criteria.
Search methods:Ten electronic databases, grey literature, and reference list searches were conducted.
Selection criteria:The inclusion criteria consisted of studies that aimed to assess a particular malocclusion index on study models of patients with CLP.
Data collection and analysis: Full articles were retrieved from abstracts/titles that appeared to have
met the inclusion -exclusion criteria which were subsequently reviewed using more detailed criteria for a
final selection decision. The Quality Assessment of Diagnostic Accuracy Studies tool was used to appraise
the methodological quality of the finally included studies. Due to the heterogeneity of the data, only a
qualitative analysis was performed.
Results:A total of 13 studies met the inclusion -exclusion criteria. These studies revealed seven utilized indices, namely the GOSLON Yardstick, Five-Year-Old, Bauru-Bilateral Cleft Lip and Palate Yardstick,
Huddart -Bodenham, Modified Huddart -Bodenham, EUROCRAN Yardstick, and GOAL Yardstick. The
GOSLON Yardstick was the most commonly used index, and the Modified Huddart -Bodenham performed
the best according to the WHO criteria.
Conclusions: Current evidence suggests that the Modified Huddart -Bodenham Index equalled or outperformed the rest of the indices on all the WHO criteria and that the GOSLON Yardstick was the most
commonly used index, possibly due to a longer time in use. Therefore, the Modified Huddart -Bodenham
could be considered as the standard to measure outcomes of patients with CLP.
773
774
Table1 QUADAS (Whiting etal., 2003) criteria for evaluating
diagnostic accuracy studies.
I. Study design (7 )
Results
Study characteristics
Among the included studies were eight cross-sectional
studies (Huddart and Bodenham, 1972; Mars etal., 1987;
Hathorn et al., 1996; Gray and Mossey, 2005; Tothill and
Mossey, 2007; Manosudprasit et al., 2011; Patel, 2011;
Dobbyn etal., 2012) and five longitudinal studies, (Mossey
etal., 2003, Atack etal., 1997b, Atack etal., 1997a, Bartzela
etal., 2011, Mars etal., 2006), and all of them were retrospective (Table2).
Synthesis of results
The selected articles revealed seven indices: the GOSLON
Yardstick (Mars etal., 1987; Hathorn etal., 1996; Mars etal.,
2006; Manosudprasit etal., 2011), the Five-Year-Old Index
(Atack etal., 1997a; Atack etal., 1997b; Mars etal., 2006),
the Bauru-Bilateral Cleft Lip and Palate (BCLP) Yardstick
(Bartzela etal., 2011), the HuddartBodenham (HB) Index
(Huddart and Bodenham, 1972; Bartzela et al., 2011), the
Modified HuddartBodenham (MHB) Index (Mossey etal.,
2003; Gray and Mossey, 2005; Tothill and Mossey, 2007;
Manosudprasit etal., 2011; Patel, 2011; Dobbyn etal., 2012),
the EUROCRAN Yardstick (Patel, 2011), and the GOAL
Yardstick (Friede etal., 1991). Summaries of the articles are
presented in Table2. Furthermore, the prevalence and a brief
description of these indices are presented in Tables 3 and 4.
Within the included studies, zero assessed the reliability of the GOAL Yardstick, one assessed the EUROCRAN
Yardstick (Patel, 2011), one assessed the BCLP Yardstick
(Bartzela et al., 2011), two assessed the reliability of the
Five-Year-Old Index (Atack et al., 1997a; Atack et al.,
1997b), two assessed the reliability of the HB Index
Study selection
The devised search strategy yielded 832 articles from
the 10 electronic databases, which are listed in detail
in online Supplementary Table 1 (Appendix). Of these
articles, 20 met the criteria of phase 1 selection (Mars
et al., 1987; Tobiasen et al., 1991; Mars et al., 1992;
Morris et al., 1994; Hathorn et al., 1996; Atack et al.,
1997a; Atack etal., 1997b; Parker, 1999; DiBiase etal.,
2002; Johnson and Sandy, 2003; Gray and Mossey, 2005;
Nollet et al., 2005; Mars et al., 2006; Deacon et al.,
2007; Suzuki etal., 2007; Tothill and Mossey, 2007; Liao
et al., 2010; Bartzela et al., 2011; Patel, 2011; Dobbyn
et al., 2012), and of the 20 atricles, 10 met the phase
2 selection criteria (Mars et al., 1987; Hathorn et al.,
1996; Atack etal., 1997a; Atack etal., 1997b; Gray and
Mossey, 2005; Mars et al., 2006; Tothill and Mossey,
2007; Bartzela et al., 2011; Patel, 2011; Dobbyn et al.,
2012) with an additional three added through the reference search (Huddart and Bodenham, 1972, Mossey
etal., 2003; Manosudprasit etal., 2011), yielding a total
of 13 articles. Accounts of the literature search results
are presented in Figure1.
M. ALTALIBI ET AL.
Index evaluated
Five-Year-Old Index
Five-Year-Old Index
HB + Bauru-BCLP
Yardstick
MHB
MHB
GOSLON Yardstick
Articles
32 subjects at 10 y
50 subjects at 5 y
and 50 subjects
at 10 y
184 subjects at 5 y
and 96 subjects at
10 y
27 subjects at 5 and
10 y (different sample
than above)
43 subjects at 6,9
and 12 y
54 subjects at (56)
and (1618 y)
Sample size
Reliability
Reliability
Reliability
Reliability and
validity
Reliability and
validity
Reliability and
validity
Criteria evaluated
IAER: 0.890.95***
IEER: 0.880.95***
5y
IAER: 0.8710.912*
IEER: 0.8530.887*
10 y
IAER: 0.7830.897*
IEER: 0.7460.865*
MHB and the GOSLON Yardstick/
Five-Year-Old Index have a high
degree of correlation
Comments
Results
775
GOSLON and
Five-Year-Old Index
MHB and
EUROCRAN
MHB
Patel (2011)
Reliability and
simplicity
Reliability and
Validity
Reliability
Reliability
Criteria evaluated
19 subjects with
Reliability
BCLP + 55 subjects
with isolated cleft palate
at 5 and 10 y
30 subjects at 5 y
94 subjects at 5
and 10 y
55 subjects at 12 y
GOSLON
Sample size
60 subject at 810 y
Index evaluated
Articles
MHB
IAER: 0.98*****
Inter-examiner reliability:
0.950.97*****
GOSLON
IAER: 0.95* (very good)**
IEER: 0.86* (very good)**
IAER: 0.890.95***
IEER: 0.840.95***
GOSLON
IAER: 1.00* (very good)**
IEER: 0.800.89* (very good)**
PV
Five-Year-Old Index at 5 y vs
GOSLON at 10 y: 0.090* (poor)**
GOSLON at 5 y vs at 10 y: 0.579*
(moderate)**
Reliability
MHB
IAER: 0.850.94*****
IEER: 0.740.94*****
EUROCRAN
IAER: 0.150.60******
IEER: 0.300.95***
Simplicity
Time needed to learn the index:
(no statistical difference P<0.05)
MHB: 49 and 39min
EUROCRAN: 97 + 50min
Time needed to score the models:
(statistical difference P<0.05)
MHB: 270 and 216min
EUROCRAN: 182 and 126min
Friendliness (Likert scale)
MHB: Average of 2.2 and 2.1
EUROCRAN: 3.4 and 2.6
BCLP
At 5 y
IAER: 0.310.78* (fair-good)**
IEER: 0.570.74* (moderate-good)**
At 10 y
IAER: 0.580.70* (moderate-good)**
IEER: 0.480.69* (moderate-good)**
ICP
At 5 y
IAER: 0.800.84* (very good)**
IEER: 0.640.81* (good-very good)**
At 10 y:
IAER: 0.770.86* (good-very good**
IEER: 0.660.74* (good)**
MHB is reliable in all types of oral clefts
Comments
Results
Table2Continued
776
M. ALTALIBI ET AL.
777
IAER, Intra-examiner reliability; IEER, Inter-examiner reliability; PV, predictive validity; UCLP, unilateral cleft lip and/or palate; BCLP, bilateral cleft lip and/or palate; HB, HuddartBodenham; MHB,
Modified HuddartBodenham.
*Weighted kappa () values.
**As described by Altman (1991).
***Spearman correlation coefficient.
****Regression model.
*****Intra-classcorrelation coefficient.
******Unweighted kappa () values.
MHB
IAER: 0.7390.952*
IEER: 0.8320.921*
PV: comparing the score of the MHB
with the Five-Year Index and GOSLON
showed statistically significant correlation
in the results 0.830.92***(P<0.01)
Criteria evaluated
Sample size
Index evaluated
Articles
Table2Continued
Prevalence
Bauru-Bilateral Cleft
Lip And Palate Yardstick
(Ozawa etal., 2011)
EUROCRAN Yardstick
(Fudalej etal., 2011,
Patel, 2011)
Five-Year-Old Index
(Atack etal., 1997b)
GOAL Yardstick (Friede
etal., 1991)
GOSLON Yardstick
(Mars etal., 1987)
HuddartBodenham
(Huddart and
Bodenham, 1972)
Modified Huddart/
Bodenham (Mossey
etal., 2003, Patel, 2011)
2011
2011
1997
53
43
1991
1987
286
273
1972
27
2003
24
24
Results
Comments
Ordinal
Ordinal
Ordinal
Ordinal
Ordinal
Continuous
Scoring scale
Index
Target population
An adaptation of the GOSLON Yardstick for bilateral cleft lip and palate rates the dentition
on a scale of 15 (bestworst prognosis, respectively), divided into 6-, 9- and 12-year-old
yardsticks
The 6- and 9-year-old yardsticks evaluate
the apical base relationships
the incisor relationships
the presence of a cross-bite (except for the permanent and deciduous lateral incisors,
deciduous canines and edge to edge buccal segments)
the arch form
The 12-year-old yardstick evaluates
the apical base relationship
the incisor relationships
The 12-year-old yardstick adds 1 point for a moderate-to-severe anterior open bite
A modification of the GOSLON Yardstick to remove the extremes and incorporate the
palatal morphology rates two components separately:
Dental arch relationship:
ranked from 1 to 4 (best to worst prognosis, respectively)
Palatal morphology:
ranked from 1 to 3 (best to worst prognosis, respectively)
An adaptation of the GOSLON Yardstick for deciduous dentition rates the dentition on a
scale of 15 (bestworst prognosis, respectively) based on
overjet and overbite
incisor inclinations
presence of cross-bites
maxillary arch shape and palatal vault
A modification of the GOSLON Yardstick rates the dentition on a scale of 15 (bestworst
prognosis, respectively) based on
presence of cross-bites
the apical base relationships
Rates the dentition on a scale of 15 (bestworst prognosis, respectively) based on
overjet and overbite
incisor inclinations
the apical base relationships
Rates the dentition on a scale of +2 to 18 (bestworst prognosis respectively) based on
presence and severity of cross-bites in the anterior segment and the two buccal segments
Rates the dentition on a scale of +10 to 30 (bestworst prognosis respectively) based on
presence and severity of cross-bites in the anterior segment and the two buccal segments
Use
778
M. ALTALIBI ET AL.
779
Study design
Measurements
Statistical
analysis
x
x
Total
9.5
9.5
12.5
10.5
12
9.5
11.5
11
13
13
9.5
11
10.5
Total %
67.9
67.9
89.3
75
85.7
67.9
82.1
78.6
92.9
92.9
67.9
78.6
75
AN, methodologic criteria in Table1: () Fulfilled satisfactorily the methodologic criteria (1 check point); () unclear or fulfilled partially the
methodologic criteria (0.5 check point); and (x) did not fulfil the methodologic criteria (0 check point).
The variability in the reliability scores within different studies of the same index may be due to the variance in the sample sizes among the studies, as well as the level of training
of the examiners. The EUROCRAN Yardstick had a very
irregular reliability score (Fudalej etal., 2011; Patel, 2011),
while the GOAL Yardsticks reliability was not tested in any
of the chosen studies.
In addition, with regards to using a 40-point scale, such
as the MHBs, as opposed to a 5-point scale, such as the
GOSLONs, it is much more difficult to achieve a good reliability score. This should be taken into account when evaluating the reliability of the MHB and the HB.
Validity
Within the field of CLP, true validation of an index is difficult because it would require a cohort of patients with
CLP with only the primary surgeries and who did not
receive any orthodontic treatment or restorative treatment
until adulthood. Without this cohort, validation tests will
inevitably be biased. Furthermore, there is no gold standard for prognosis in assessing the severity of malocclusions within cleft patients. Therefore, all the studies thus
far rely on face validity, which relies on what the investigator deems important and is more subjective (Atack
etal., 1997a).
The GOSLON Yardstick, HB, MHB, and BCLP showed
acceptable validity. While the studies evaluating the FiveYear-Old Index did not show a good ability to predict outcome, the EUROCRAN and GOAL Yardsticks did not have
any studies within those selected in this systematic review
that examined predictive validity.
Prevalence
780
Judgment required
The HB and MHB are the only indices that do not require
calibration and show similar rates of reliability among
trained and non-trained individuals. That is because the
point scale that the HB indices use rely on dental measurements solely, thus making it simple, objective, and eliminates the need for calibration. The disadvantage of such
a system is that it does not assess the skeletal component
of the malocclusion, not differentiating dental cross-bites
from skeletal discrepancies leading to cross-bites. Also,
it cannot differentiate between a generalized mild malocclusion and a localized severe malocclusion and cannot
assess the vertical discrepancies of the malocclusions.
Conversely, the GOSLON Yardstick uses clinical measurements, evaluates the ease of treating a particular malocclusion, and takes that into account in its score (Mossey
et al., 2003), but that requires a calculated judgment by
the evaluator, which adds to its subjectivity and need
for calibration. However, despite the fundamental difference between the MHB and the GOLSON Yardstick, the
results have been shown to be highly correlated (Gray and
Mossey, 2005).
M. ALTALIBI ET AL.
781
Conclusion
References
Love R, Walters M, Southall P, Singer S, Gillett D 2012 Dental arch relationship outcomes in children with complete unilateral cleft lip and
palate treated at princess margaret hospital for children, perth, Western
australia. Cleft Palate-Craniofacial Journal 49: 456462
782
M. ALTALIBI ET AL.
Parker W S 1998 The HLD (CalMod) index and the index question. American
Journal of Orthodontics and Dentofacial Orthopedics 114: 134141
1: five-year-olds index for dental arch relationships. Cleft PalateCraniofacial Journal 44: 434443
Patel D 2011 Evaluation of the use of the Modified Huddart Bodenham &
Eurocran Yardstick for the assessment of surgical outcome for unilateral
cleft lip and palate. Thesis, University of Dundee
Pruzansky S, Aduss H 1964 Arch form and the deciduous occlusion in
complete unilateral clefts. The Cleft Palate Journal 30: 411418
Ren S, Ma L, Sun Z, Qian J 2012 Relationship between Palate-Vomer
development and maxillary growth in submucous cleft palate patients.
Cleft Palate-Craniofacial Journal published online 20 September 2012,
doi: 10.1597/12-051
Summers C J 1971 A system for identifying and scoring occlusal disorders. American Journal of Orthodontics and Dentofacial Orthopedics
59: 552567
Suzuki A et al. 2007 Retrospective evaluation of treatment outcome in
Japanese children with complete unilateral cleft lip and palate. Part