Fig. 47.1
Goslon individual patient scores at age 9 by center. A Goslon score of 1 represents excellent maxillary prominence and a score of 5, severe maxillary retrusion. One way to consider this outcome variable is the likely future need for subsequent maxillary osteotomy, and cases falling below 3.5 at this age are likely candidates for osteotomy in the late teens
47.2.2 Follow-Up
The aims of the follow-up were: to quantify the burden of care imposed by respective protocols, to see whether the ranking of centers for different outcomes at age 9 was predictive for equivalent outcomes at age 17, to assess patient/parent satisfaction with care, and to explore interrelationships with outcome and burden (Brattström et al. 2005; Mølsted and Brattström 2005; Semb et al. 2005a, b; Shaw et al. 2005). A separate comparison of speech outcomes was carried out at age 11–14 (Grunwell et al. 2000).
47.2.3 Survey of Treatment Experience
The amount of treatment provided by the five different teams in 1976–1979 was remarkably different (Table 47.1). Most notable was the lengthy hospital stay associated with presurgical orthopedics at that time in centers D and F. The subjects in center D also had more orthodontic visits for treatment and review and for the overall number of surgeries compared to the other centers. From discussion with these centers, it would seem that the reason for the large differences in the intensity of treatment was not primarily related to clinical need but rather to differing beliefs and historical practices that had shaped the clinical protocols of the period.
Table 47.1
Amount of treatment provided by five different teams from birth to 17 years of age
|
A
|
B
|
D
|
E
|
F
|
||
|---|---|---|---|---|---|---|
|
Surgery
|
||||||
|
Mean number of surgeries
|
4.8
|
3.3
|
6.0
|
4.4
|
3.5
|
|
|
Mean days in hospital
|
33
|
31
|
60
|
24
|
26
|
|
|
Presurgical orthopedics
|
||||||
|
Months of treatment
|
13
|
0
|
15
|
0
|
5
|
|
|
Number of visits
|
11
|
0
|
8
|
0
|
17
|
|
|
Days in hospital
|
0
|
0
|
60
|
0
|
146
|
|
|
Orthodontic treatment
|
||||||
|
Treatment length (years)
|
5.6
|
3.3
|
8.5
|
3.5
|
4.0
|
|
|
Number of visits
|
Treatment
|
52
|
41
|
54
|
33
|
47
|
|
Follow-up
|
11
|
23
|
42
|
16
|
25
|
|
|
Total
|
63
|
64
|
94
|
49
|
72
|
|
47.2.4 Consistency of Outcomes over Time
The statistical analysis used to compare the five centers was a general linear mixed model applied to longitudinal data (Diggle et al. 1994). Variance terms were included in the model to account for between subject variation in the intercept as well as fixed factor for assessment point (9, 12, 17 years) and center. Full details have been reported elsewhere (Shaw et al. 2005).
As Fig. 47.2 indicates, the scores for dental arch relationship tended to improve in centers A, B, and E, but not in D and F. There was a consistent relationship over time for most cephalometric variables, e.g., soft tissue profile (Fig. 47.3), and for nasolabial appearance.
Fig. 47.2
Mean dental arch relationship scores at ages 9, 12, and 17 years for participating centers
Fig. 47.3
Mean soft tissue profile (angle SSS-NS-SMS) at ages 9, 12, and 17 years for participating centers
47.2.5 Lack of Association Between Outcome and the Amount of Treatment
Not surprisingly, follow-up of these five cohorts of patients from age 9 to age 17 confirmed the main finding of the first report, with some centers continuing to achieve considerably better outcome than others, at all age points. Perhaps, more surprising is the lack of association between amount of treatment and final outcome (Tables 47.2, 47.3, and 47.4). Especially ironic is the finding that the two centers with the highest intensity of early treatment (hospitalization in order to perform presurgical orthopedics) achieved the lowest rankings for eventual outcome (Figs. 47.2 and 47.3). Thus, the poorest ratings for nasal appearance were associated with the lengthy use of a presurgical device called T-traction designed not only to reduce the alveolar gap but also to straighten the nasal septum (Nordin et al. 1983). Patients in the center with the least favorable dentofacial outcomes (center D) also experienced the longest orthodontic treatment duration and the highest number of orthodontic visits. It appears that this was partly due to the complexity of center D’s orthodontic treatment protocols with almost continuous treatment from the eruption of the primary dentition and partly to the unfavorable dentofacial outcomes of primary surgery.
Table 47.2
The relationship between outcome assessment (dental arch relationship at 17 years) and amount of infant orthopedic treatment in the different centers
|
Objective ranking
|
Center
|
Months of treatment
|
No. of visits
|
Days in hospital
|
|---|---|---|---|---|
|
Best
|
E
|
0
|
0
|
0
|
 |
A
|
13
|
11
|
0
|
|
B
|
0
|
0
|
0
|
|
|
F
|
5
|
17
|
146
|
|
|
Worst
|
D
|
15
|
8
|
60
|
Table 47.3
The relationship between outcome assessment (dental arch relationship at 17 years) and amount of orthodontic treatment in the different centers
|
Objective ranking
|
Center
|
Treatmentlength (years)
|
No. of visits
|
|
|---|---|---|---|---|
|
Treatment
|
Checkup
|
|||
|
Best
|
E
|
3.5
|
33
|
16
|
 |
A
|
5.6
|
52
|
11
|
|
B
|
3.3
|
41
|
23
|
|
|
F
|
4.0
|
47
|
25
|
|
|
Worst
|
(D)
|
8.5
|
54
|
42
|
Table 47.4
The relationship between outcome assessment (dental arch relationship at 17 years) and the mean number of surgeries per patient in the different centers
|
Objective ranking
|
Center
|
Number of surgeries
|
|---|---|---|
|
Best
|
E
|
4.4
|
 |
A
|
4.8
|
|
B
|
3.8
|
|
|
F
|
3.5
|
|
|
Worst
|
D
|
6.0
|
This lack of association between treatment outcome and intensity may represent a key lesson for the development of future protocols. It justifies an emphasis on simplicity, economy, and minimized burden for the patient, rather than adherence to demanding protocols with unsubstantiated promise.
47.2.6 Lack of Association Between Outcome and Satisfaction
Perhaps, the most perplexing finding of the Eurocleft series is the inconsistency between objectively rated outcomes and patient/parent satisfaction. There were instances where the highest levels of dissatisfaction with treatment outcome were reported by subjects attending the centers with the best objective ratings (Table 47.5). The possible reasons for this disparity have been discussed elsewhere (Semb et al. 2005b), and it highlights the need for concerted work on the understanding and measurement of patient/parent satisfaction and the provision of more holistic models of cleft care.
Table 47.5
The relationship between objective ranking of nasolabial outcome and patient dissatisfaction
|
Objective ranking
|
Percentage of respondents dissatisfied with nasal appearance
|
Objective ranking
|
Percentage of respondents dissatisfied with lip appearance
|
||
|---|---|---|---|---|---|
|
Best
|
A
|
64
|
Best
|
B
|
14
|
 |
E
|
32
|
 |
A
|
41
|
|
B
|
14
|
F
|
6
|
||
|
D
|
45
|
E
|
42
|
||
|
Worst
|
F
|
33
|
Worst
|
D
|
16
|
47.3 Wider Networks
Following initial reports of the Eurocleft cohort study, numerous teams from Europe and elsewhere approached members of Eurocleft and arranged to undertake blinded comparisons of their records with the Eurocleft material or subsections of it, some of which were published (Flinn et al. 2006; Fudalej et al. 2009; Gaukroger et al. 2002; MacKay et al. 1994; Meazzini et al. 2008, 2010; Nollet et al. 2005; Roberts-Harry et al. 1996). Also, during the Eurocran project that was subsequently funded by the European Union from 2000 to 2005, further centers from Europe had the opportunity to undertake similar comparisons. More recently, multicenter comparisons were completed in India and Turkey, and new initiatives are underway in Thailand and South Africa (Susami et al. 2006; Alex 2011; Bellardie 2011; Dogan 2011). The most extensive initiative was carried out in North America and is described below.
47.4 Americleft
Before 2006, centers in the USA and Canada had not been as successful as those in Europe in establishing interest and commitment to intercenter collaborative outcome studies. In the 2002 WHO report “Addressing the Global Challenges of Craniofacial Anomalies,” it was noted that in the USA and elsewhere in North America, there had been little significant momentum in the area of intercenter, collaborative, clinical research, especially compared to the more successful efforts of Eurocleft and Eurocran. As a result, there was little useful information being generated from the ongoing research which would contribute to the establishment of sound evidence-based decision making in clinical care.
47.4.1 The Challenge
The reasons for this failure were complicated. While the large number of centers and individuals providing treatment for CFA in North America improved patients’ geographical accessibility to care, it simultaneously created a fractionation of the study population, thereby reducing the probability of developing patient samples of adequate size to enable valid research. The entire landscape was further complicated by noncomparable patient populations, noncomparable treatment records, unquantifiable differences in operator skills, and difficulties in letting go of biases. Also, while collaborative research could be structured without violating patient privacy laws, the rigors of doing so were sufficient discouragement for many clinicians to participate. Finally, there remained a general lack of agreement between centers on minimal standards for reporting and recording outcomes, as well as cost and ethical concerns over taking records which cannot be clearly identified as essential for diagnosis and treatment purposes.
In summary, although the desire, research talent, and patient samples were all readily available in the North America, before 2006, the failure to get centers to agree on something as basic as standardization of recording and reporting outcomes, as well as governmental hurdles and a serious lack of funding, all resulted in a huge and ongoing missed opportunity. The WHO (2002) concluded by stating that … “it seems most likely, that the most promising avenue to break out of this inertia, may still lie in the original Eurocleft approach. With a core of interested and experienced clinicians, operating at high volume centers, and willing to agree on records, outcome measures of significance, and research protocols, and additionally with the possible guidance from those involved in the successful Eurocleft, Scandcleft and Eurocran programs, it might still be possible to initiate a major inter-center collaborative research effort.”
47.4.2 Initiation
Based on this report, in 2006, the American Cleft Palate-Craniofacial Association (ACPA) and the Cleft Palate Foundation (CPF) approved funds to organize a pilot project which has become the “Americleft Project” and ACPA’s Americleft Task Force. From ACPA’s strategic plan, the purpose of this initiative is “….to continue to develop strategies and execute intercenter collaborative outcomes studies in multiple disciplines for the purpose of documenting the outcomes and assessing best practices of team care.”
Five centers were identified to participate in the pilot project which was modeled exactly after the Eurocleft study and resulted in completion of initial comparisons of dental arch relationship outcomes, cephalometric skeletodental morphology outcomes, and nasolabial appearance outcomes. The results of these, to be discussed below, confirmed the value and benefits of well-controlled and well-designed intercenter outcome comparisons. Most importantly, however, was the experience and insight that was gained in understanding the requirements, demands, and possible obstacles that must be overcome in order to participate successfully in such collaborative studies.
47.4.3 Participants
The success of Americleft has always been based on the integrity and intellectual honesty of those choosing to participate and several key requirements for participation. Participating centers’ team members had to be experienced and focally interested in CLP and with an interest in seeking knowledge about the relative merits of various treatment protocols rather than having an unquestioning loyalty to particular procedures. While we as care providers all believe that the procedures we are doing are the best possible for our patients, involvement in collaborative outcome studies implies a degree of uncertainty about the true effectiveness of our individual protocols, the ability to question our own beliefs and to accept the possibility that there may be other equally good or better outcomes with protocols different from the one(s) used by our own team. In addition, the Americleft centers had to have a high volume of patients and well-defined protocols for management of their patient population. Centers also had to have the resources to support team representatives in dedicating the time and absorbing the costs of the effort as well as the availability of the necessary records (privacy protected) and ability to secure IRB approval from the parent institution.
47.4.4 The Original Foundation Americleft Cohort Studies
The initial studies conducted as part of the Americleft Project were identical to those carried out in the original Eurocleft study (Long et al. 2011): 9-year-old, mixed dentition comparison of dental arch relationship (Hathaway et al. 2011), craniofacial form (Daskalogiannakis et al. 2011), and nasolabial appearance (Mercado et al. 2011 b). The patients were consecutively treated by high-volume surgeons in their respective centers. The outcome measures were also identical to those used in the Eurocleft study, including dental arch relationship ratings using dental casts and the Goslon yardstick (Mars et al. 1987
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