Search strategy

Main search

The search of PubMed was conducted using the following medical subject heading (MeSH) terms: [(‘Orthognathic Surgery’ OR ‘Orthognathic Surgery’ OR ‘Orthognathic Surgeries’ OR ‘Surgeries, Orthognathic’ OR ‘Surgery, Orthognathic’ OR ‘Maxillofacial Orthognathic Surgery’ OR ‘Maxillofacial Orthognathic Surgeries’ OR ‘Orthognathic Surgeries, Maxillofacial’ OR ‘Orthognathic Surgery, Maxillofacial’ OR ‘Surgeries, Maxillofacial Orthognathic’ OR ‘Surgery, Maxillofacial Orthognathic’ OR ‘Orthognathic Surgical Procedures’ OR ‘Orthognathic Surgical Procedure’ OR ‘Procedure, Orthognathic Surgical’ OR ‘Procedures, Orthognathic Surgical’ OR ‘Surgical Procedure, Orthognathic’ OR ‘Surgical Procedures, Orthognathic’) AND (‘Surgery, Computer-Assisted’ OR ‘Surgery, Computer-Assisted’ OR ‘Computer-Assisted Surgeries’ OR ‘Surgeries, Computer-Assisted’ OR ‘Surgery, Computer Assisted’ OR ‘Computer-Assisted Surgery’ OR ‘Computer Assisted Surgery’ OR ‘Computer-Aided Surgery’ OR ‘Computer Aided Surgery’ OR ‘Computer-Aided Surgeries’ OR ‘Surgeries, Computer-Aided’ OR ‘Surgery, Computer-Aided’ OR ‘Surgery, Image-Guided’ OR ‘Image-Guided Surgeries’ OR ‘Surgeries, Image-Guided’ OR ‘Surgery, Image Guided’ OR ‘Image-Guided Surgery’ OR ‘Image Guided Surgery’ OR ‘Computer-Aided Design’ OR ‘Computer Aided Design’ OR ‘Computer-Aided Designs’ OR ‘Design, Computer-Aided’ OR ‘Designs, Computer-Aided’ OR ‘Computer-Assisted Design’ OR ‘Computer Assisted Design’ OR ‘Computer-Assisted Designs’ OR ‘Design, Computer-Assisted’ OR ‘Designs, Computer-Assisted’ OR ‘Computer-Aided Manufacturing’ OR ‘Computer Aided Manufacturing’ OR ‘Manufacturing, Computer-Aided’ OR ‘Computer-Assisted Manufacturing’ OR ‘Computer Assisted Manufacturing’ OR ‘Manufacturing, Computer-Assisted’ OR ‘CAD-CAM’)].

The same search strategy was applied to the Cochrane Library, since this also uses MeSH terms.

For the search of EMBASE, the Emtree terms ‘orthognathic surgery’, ‘computer assisted surgery’ and ‘computer aided design’ were used to carry out a specific search: ‘orthognathic surgery’/syn AND (‘computer assisted surgery’/syn OR ‘computer aided design’/syn).

Health sciences descriptors (Descritores em Ciências da Saúde—DeCS) in the English language were used to search the LILACS and SciELO databases; the following searches were performed: LILACS (tw:(‘orthognathic surgery’)) AND (tw:(‘computer-aided design’)) and SciELO ‘orthognathic surgery’ AND ‘computer-aided design’.

Grey literature

A search for unpublished studies and research published in journals not indexed in major databases was conducted: (1) annals of the last three editions of the International Conference on Oral and Maxillofacial Surgery (ICOMS), promoted by the International Association of Oral and Maxillofacial Surgery, and the last three annual meetings of the American Association of Oral and Maxillofacial Surgery (AAOMS Annual Meeting). (2) Google Scholar, using MeSH terms (‘orthognathic surgery’, ‘computer aided design’, ‘computer assisted surgery’) and Boolean operators (‘AND’ and ‘OR’): ‘orthognathic surgery’ AND (‘computer aided design’ OR ‘computer assisted surgery’).

The authors of the abstracts found in the annals were contacted by e-mail for information on the results or to determine whether the study had been published.

Manual search

The references of papers identified were analyzed for further studies not located in the above-mentioned searches.

Study selection

The systematic search was conducted by one of the authors (O.L.H.J.), and articles were selected independently by two authors (O.L.H.J., O.E.B.) based on the title and abstract. Studies with the following characteristics were chosen for full-text reading: (1) intervention studies; (2) creation of a virtual surgical splint; (3) investigations assessing the accuracy of orthognathic surgery with a virtual surgical splint. Papers that did not meet these criteria were excluded from the analysis. When the authors disagreed on the selection of a paper, the study was read in full.

Studies for which the titles and abstracts were evaluated and that were accepted in the first selection process were submitted to an eligibility assessment.

The level of agreement between the authors was tested using Cohen’s kappa coefficient ( κ ).

Eligibility of the studies

The eligibility of the studies was checked by two authors (O.L.H.J., O.E.B.) who were blinded to the title, abstract, authorship, and origin of the papers. The following eligibility criteria were used: (1) the main theme of the paper had to centre on computer-aided planning for orthognathic surgery; (2) the trial had to be original and an intervention study; (3) the surgical procedure had to be computer-aided with virtual planning and with the design of a virtual surgical splint; (4) accuracy measures had to be presented for the surgical procedure. In the event of disagreement between the two authors, the study in question was discussed with the third, more experienced author (R.B.O.).

Investigations that did not meet the eligibility criteria were excluded from the analysis and the reason for their exclusion was reported.

When questions arose regarding the methodology or results of a paper, the author was contacted by e-mail to obtain the necessary answers.

The level of agreement between the authors was tested using Cohen’s kappa coefficient ( κ ).

Data extraction

Demographic and methodological data, as well as the accuracy results, were extracted from the studies that met the eligibility requirements by the two blinded authors (O.L.H.J., O.E.B). In the event of disagreement between the two authors, the study was discussed with the third author (R.B.O.). When doubts persisted, the author of the study in question was contacted by

Analysis of the methodological quality of the studies included

The quality of the papers was assessed using an adaptation of the bias analysis proposed by Clementini et al. The criteria used by these authors are related to the randomization of the sample, validation of measurements, statistical analysis, the definition of inclusion and exclusion criteria, and whether sample loss was reported in the postoperative period. In addition to these items, analysis of comparison data between interventions and blinding of the rater were included as criteria.

With respect to the risk of bias for each study analyzed, papers containing all the above-mentioned items were considered low risk, those for which one or two items were missing were deemed medium risk, and investigations that did not include three or more items were considered high risk.

Search strategy

The strategies used for the main search and grey literature search were applied for the last time on 13 January 2014. The manual search was conducted after papers had been included, with no need for updating. A flowchart showing the studies included at each stage of the systematic review is given in Fig. 1 .

Systematic review flowchart. GS, Google Scholar; ICOMS, International Conference on Oral and Maxillofacial Surgery; AAOMS M, American Association of Oral and Maxillofacial Surgery Annual Meeting; κ , measure of inter-rater agreement according to the kappa coefficient. *Abstracts found in the annals of the last three conferences; the authors were contacted by email, but no response was received and these were excluded.

Main search

Three hundred and fifty-seven articles were found in PubMed, 93 in EMBASE, and 28 in LILACS; no studies were found in the Cochrane Library or SciELO. Duplicate papers were removed, leaving a total of 375 possible studies. The oldest of these papers was published in 1984, with studies on computer-aided planning in orthognathic surgery increasing in subsequent years to a total of 157 publications in the last 3 years (2011–2013) ( Fig. 2 ).

Numbers of papers published through the years, 1984–2013.

Grey literature

With regard to the grey literature search, four studies were selected from Google Scholar, one from the ICOMS annals, and four from the annals of the AAOMS Annual Meeting. The authors of the studies found in these annals were contacted by e-mail regarding the results and to determine whether the research had been published in the form of a scientific paper. Those studies for which an author reply was not received were excluded. As a result, only the four studies found in the Google Scholar search were included in the sample submitted for eligibility assessment.

Manual search

One study was found and included in the final sample for systematic review.

Study selection

The titles and abstracts of the 375 selected studies were read and 34 of these were chosen for full-text reading. Whenever the two authors differed as to the selection of a paper, disagreement was resolved by choosing the broadest possible study. The level of agreement between the two authors in selecting studies to be read in full was measured at κ = 0.767.

Eligibility of the studies

As part of the eligibility assessment, 38 studies were read in full, 34 from the main search and four from the grey literature search. At the end of this analysis, eight papers were included in the sample for systematic review. The other 30 studies were excluded for the following reasons: computer-aided planning for orthognathic surgery was not the main theme of the paper ( n = 7 studies ), the paper was not an intervention study ( n = 14 studies ) or was not original ( n = 1 ), the surgical procedure did not involve a computer-assisted virtual surgical splint ( n = 3 ), and accuracy measures for the surgical procedure were not provided ( n = 5 ).

The level of agreement between the two authors for the eligibility assessment was measured at κ = 0.863.

Data extraction

The sample used in the systematic review consisted of nine studies, of which seven were found in the main search, one in the grey literature, and one in the manual search.

The research groups on computer-aided planning in orthognathic surgery were from different countries; however, two papers by a group from the USA and two by a research group in Spain were included in the systematic review. All of the studies were prospective and only one paper was a randomized controlled clinical trial. Six were published in 2013, and the largest sample (65 patients) was part of a multi-centre trial. The total sample from the studies included, in which CAD/CAM was used in orthognathic surgery, comprised 137 individuals; their mean age was 20–30 years and the gender (male/female) and facial deformity (class II/class III) proportions were approximately 50% ( Table 1 ).

Table 1

Demographic data for the studies included.

Author, year, and country of origin	Type of study	Sample	Age, years, mean ± SD (variation)	Gender	Type of facial deformity
Xia et al., 2007 USA	Prospective case series/pilot study	n = 5	NA	2 M, 3 F a	3 class II, 2 class III, all asymmetrical a
Centenero and Hernández-Alfaro, 2012 Spain	Prospective case series	n = 16	NA	NA	9 class II, 7 class III
Sun et al., 2013 Belgium	Prospective case series	n = 15	NA	NA	NA
Li et al., 2013 China	Prospective case series	n = 6	(19–30)	4 M, 2 F	NA
Hsu et al., 2013 USA	Prospective case series/multi-centre	n = 65 Houston: 41 Portland: 11 New York: 13	25 (15–51) Houston: 26.7 (15–51) Portland: 26.7 (16–46) New York: 21.7 (16–51)	31 M, 34 F Houston: 23 M, 18 F Portland: 3 M, 8 F New York: 5 M, 8 F	NA
Hernández-Alfaro and Guijarro-Martínez, 2013 Spain	Prospective case series/proof of concept study	n = 6	23.7 (19–37)	3 M, 3 F	5 class II, 1 class III
Shehab et al., 2013 Egypt	Prospective case series/pilot study	n = 6	23.5 (18–30)	6 F	6 class II with vertical maxillary excess
Zinser et al., 2013 Germany	Non-randomized clinical trial	n = 28 Virtual splint: 8 Surgical navigation: 10 Classic splint: 10	20.8 ± 4.9 (18–35) Virtual splint: 21.6 ± 5.45 (19–35) Surgical navigation: 20.5 ± 4.1 (18–32) Classic splint: 20.6 ± 2.6 (18–26)	15 M, 13 F Virtual splint: 4 M, 4 F Surgical navigation: 5 M, 5 F Classic splint: 6 M, 4 F	5 class II, 23 class III Virtual splint: 8 class III Surgical navigation: 1 class II, 9 class III Classic splint: 4 class II, 6 class III
De Riu et al., 2014 Italy	Randomized controlled clinical trial	n = 20 Virtual splint: 10 Classic splint: 10	Virtual splint: (21–54) Classic splint: (24–47)	10 M, 10 F Virtual splint: 3 M, 7 F Classic splint: 7 M, 3 F	Class II/class III: NA All asymmetrical

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