Three-dimensional Imaging Methods for Quantitative Analysis of Facial Soft Tissues and Skeletal Morphology in Patients with Orofacial Clefts: A Systematic Review

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Three-dimensional Imaging Methods for Quantitative Analysis of Facial Soft Tissues and Skeletal Morphology in Patients with Orofacial Clefts: A Systematic Review
  Three-dimensional Imaging Methods for QuantitativeAnalysis of Facial Soft Tissues and Skeletal Morphologyin Patients with Orofacial Clefts: A Systematic Review Mette A. R. Kuijpers 1,2 * , Yu-Ting Chiu 3 , Rania M. Nada 1 , Carine E. L. Carels 1,2 , Piotr S. Fudalej 1,4,5 1 Department of Orthodontics and Craniofacial Biology, Radboud University Medical Centre, Nijmegen, the Netherlands,  2 Cleft Palate Craniofacial Unit, RadboudUniversity Medical Centre, Nijmegen, The Netherlands,  3 Department of Dentistry and Craniofacial Center, Chang Gung Memorial Hospital, Taipei, Taiwan,  4 Departmentof Orthodontics and Dentofacial Orthopedics, University of Bern, Bern Switzerland,  5 Department of Orthodontics, Palacky University Olomouc, Olomouc, Czech Republic Abstract Background:   Current guidelines for evaluating cleft palate treatments are mostly based on two-dimensional (2D)evaluation, but three-dimensional (3D) imaging methods to assess treatment outcome are steadily rising. Objective:   To identify 3D imaging methods for quantitative assessment of soft tissue and skeletal morphology in patientswith cleft lip and palate. Data sources:   Literature was searched using PubMed (1948–2012), EMBASE (1980–2012), Scopus (2004–2012), Web of Science (1945–2012), and the Cochrane Library. The last search was performed September 30, 2012. Reference lists werehand searched for potentially eligible studies. There was no language restriction. Study selection:   We included publications using 3D imaging techniques to assess facial soft tissue or skeletal morphologyin patients older than 5 years with a cleft lip with/or without cleft palate. We reviewed studies involving the facial regionwhen at least 10 subjects in the sample size had at least one cleft type. Only primary publications were included. Data extraction:   Independent extraction of data and quality assessments were performed by two observers. Results:   Five hundred full text publications were retrieved, 144 met the inclusion criteria, with 63 high quality studies. Therewere differences in study designs, topics studied, patient characteristics, and success measurements; therefore, only asystematic review could be conducted. Main 3D-techniques that are used in cleft lip and palate patients are CT, CBCT, MRI,stereophotogrammetry, and laser surface scanning. These techniques are mainly used for soft tissue analysis, evaluation of bone grafting, and changes in the craniofacial skeleton. Digital dental casts are used to evaluate treatment and changesover time. Conclusion:   Available evidence implies that 3D imaging methods can be used for documentation of CLP patients. No dataare available yet showing that 3D methods are more informative than conventional 2D methods. Further research iswarranted to elucidate it. Systematic review registration:   International Prospective Register of Systematic Reviews, PROSPERO CRD42012002041 Citation:  Kuijpers MAR, Chiu Y-T, Nada RM, Carels CEL, Fudalej PS (2014) Three-dimensional Imaging Methods for Quantitative Analysis of Facial Soft Tissues andSkeletal Morphology in Patients with Orofacial Clefts: A Systematic Review. PLoS ONE 9(4): e93442. doi:10.1371/journal.pone.0093442 Editor:  Irina Kerkis, Instituto Butantan, Brazil Received  September 24, 2013;  Accepted  March 4, 2014;  Published  April 7, 2014 Copyright:    2014 Kuijpers et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permitsunrestricted use, distribution, and reproduction in any medium, provided the srcinal author and source are credited. Funding:  These authors have no support or funding to report. Competing Interests:  The authors have declared that no competing interests exist.* E-mail: Introduction Patients with cleft lip and palate (CLP) are treated for anextended period of time. They often undergo several types of surgery as well as other treatment procedures by specialistscollaborating with interdisciplinary teams from infancy untiladulthood. The surgical procedures are necessary to reconstructthe anatomy of the alveolar arch and the face, and to restore thefunctions of the palate, lip muscles, and nose. Although treatmentimproves function and esthetics, it potentially can lead to tissuedistortion and have a negative effect on craniofacial growth [1].This may lead to less optimal facial esthetics along with negativepsychosocial effects on a patient’s well-being [2,3].Many treatment protocols exist for the management of patientswith CLP. Therefore, evaluating the results of treatment becomesmore and more important. The Eurocleft study [4] evaluatedtreatment outcomes in Europe in the 1990s and recently the Americleft study [5,6–9] examined treatment outcome in the US.Both studies proposed documentation and record taking forevaluation of treatment outcomes at certain time points, while they PLOS ONE | 1 April 2014 | Volume 9 | Issue 4 | e93442  leave liberty for records at other time points. For record taking itappears that the first most complete data records are generally notdocumented earlier than age 5 [4,5]. At this age, some records,especially dental casts, have a predictive value for growth andfurther treatment [10,11].It is expected that the majority of cleft palate treatment teamswill use newly introduced three dimensional (3D) imaging technology to assess their treatment results. An increasing numberof papers have been published regarding 3D evaluation of facialmorphology and treatment outcomes in patients with clefts.Pharyngeal space is assessed with magnetic resonance imaging (MRI), computed tomography (CT), or cone beam computedtomography (CBCT). Results of bone grafting are evaluated withCT or CBCT. The jaw relationship, dental and alveolar arch, andthe effects of surgery are examined with digital models and CBCT.The guidelines derived from Eurocleft, and later from Americleft,are still based on two-dimensional (2D) evaluation, except fordental casts, which are 3D by nature. Further evaluation may beneeded to determine whether guidelines are necessary for thenewer craniofacial imaging technologies. A recent systematic review [12] about methods to quantify soft-tissue based facial growth and treatment outcomes in children younger than 6 years of age concluded that stereophotogrammetryseems to be the best method to longitudinally assess facial growthin these children. Studies on infants with CLP using 3D imaging techniques have been performed mainly to evaluate lip changesafter surgery [13–15] and the effect of nasoalveolar molding [16]. A systematic review of existing 3D technologies for assessing treatment outcome in patients with CLP would provide clues forevaluating treatment effects and planning, as well as a comparisonof treatment possibilities. Therefore, the objective of thissystematic review was to identify 3D imaging methods that enablea quantitative analysis of facial soft tissues, velopharyngealfunction and airway, skeletal morphology, and dentition inpatients with cleft lip and palate. Methods Protocol and Registration Inclusion criteria and methods of analysis were specified inadvance and registered as a protocol in the InternationalProspective Register of Systematic Reviews, PROSPERO( ). The registration numberis: CRD42012002041. The protocol for this systematic review andsupporting PRISMA checklist are available as supporting infor-mation; see Checklist S1 and Protocol S1. Eligibility Criteria Primary publications eligible for inclusion were those using 3Dimaging techniques for assessing facial soft tissue or skeletalmorphology in CLP patients. Further inclusion criteria were 1)cleft lip with or without cleft palate; 2) sample size larger than 10for at least one cleft type; 3) patients 5 years of age or older; and 4)publications with quantitative assessment. Patients 5 years andolder were included, because it appears that the first mostcomplete data records are generally not documented earlier thanage 5 [4,5]. Exclusion criteria were: 1) craniofacial syndromes; 2)imaging only of neurocranium; 3) injury and trauma; 4) use of only2D imaging techniques; and 5) reviews, expert opinions, letters,and case reports.No restrictions were made for language, publication date, andpublication status. Information Resources To identify publications, literature was searched until Septem-ber 2012 using PubMed (1948–2012), EMBASE (1980–2012),Scopus (2004–2012), Web of Science (1945–2012), and the Table 1.  PubMed search strategy. (((((((((4D[tiab] OR 4-dimensional[tiab])) OR (Four Dimensional Computed Tomography[tiab]))) OR (((((Tomography, X-Ray Computed[Mesh] OR Tomography, X-RayComputed[tiab])) OR (Computed Tomographic[tiab] OR CT[tiab] OR volumetric CT[tiab])) OR (Cone Beam Computed Tomography[tiab] OR CBCT[tiab] OR Spiral ConeBeam Computed Tomography[tiab])) OR (Four Dimensional Computed Tomography[tiab]))) OR (((Photogrammetry[Mesh] OR Photogrammetry[tiab])) OR(stereophotogrammetr*[tiab]))) OR (((((computed tomography[tiab])) OR (computer assisted tomography[tiab]))) OR (((((Tomography, X-Ray Computed[Mesh] ORTomography, X-Ray Computed[tiab])) OR (Computed Tomographic[tiab] OR CT[tiab] OR volumetric CT[tiab])) OR (Cone Beam Computed Tomography[tiab] ORCBCT[tiab] OR Spiral Cone Beam Computed Tomography[tiab])) OR (Four Dimensional Computed Tomography[tiab])))) OR (((Magnetic Resonance Imaging[Mesh] ORMagnetic Resonance Imaging[tiab] OR Magnetic Resonance Image[tiab] OR Magnetic Resonance Images[tiab])) OR (MRI[tiab]))) OR (((((Imaging, Three-Dimensional[Mesh] OR Imaging, Three-Dimensional[tiab])) OR (3D[tiab] OR three dimensional[tiab])) OR (3D[tiab] AND (image[tiab] OR images[tiab] OR imaging[tiab])))OR (3D image[tiab] OR 3D images[tiab] OR 3D imaging[tiab]))) AND ((((cleft lip[Mesh] OR cleft lip[tiab])) OR (cleft palate[Mesh] OR cleft palate[tiab])) OR ((((CLP[tiab])) OR(UCLP[tiab])) OR (BCLP[tiab])))doi:10.1371/journal.pone.0093442.t001 Table 2.  Quality assessment instrument. I. Study design (7 ) A. Objective–objective clearly formulated ( )B. Sample size–considered adequate ( )C. Sample size–estimated before collection of data ( )D. Selection criteria–clearly described ( )E. Baseline characteristics–similar baseline characteristics ( )F. Timing–prospective ( )G. Randomization–stated ( ) II. Study measurements (3 ) H. Measurement method–appropriate to the objective ( )I. Blind measurement–blinding ( )J. Reliability–adequate level of agreement ( ) III. Statistical analysis (5 ) K. Dropouts–dropouts included in data analysis ( )L. Statistical analysis–appropriate for data ( )M. Confounders–confounders included in analysis ( )N. Statistical significance level– P   value stated ( )O. Confidence intervals provided ( )Maximum number of s=15(Gordon JM, Rosenblatt M, Witmans M, Carey JP, Heo G, Major PW, et al. Rapidpalatal expansion effects on nasal airway dimensions as measured by acousticrhinometry. A systematic review.  Angle Orthod  . 2009;79(5): 1000–1007.).doi:10.1371/journal.pone.0093442.t002 Imaging Methods for CLPPLOS ONE | 2 April 2014 | Volume 9 | Issue 4 | e93442  Cochrane Library. The last search was performed September30th, 2012. Reference lists of identified manuscripts were thenhand searched for potentially eligible studies. Digital full textpublications were retrieved from licensed digital publishers andpaper publications were retrieved from the university library. Authors were contacted when publications could not be retrieved.Gray literature was not searched. Search Strategy  A search strategy and list of terms were developed anddatabases were selected with the help of a senior librarianspecialized in health sciences. Medical subject headings and textwords in the title and abstract were used for the search strategy inPubMed (Table 1) and search strategies for other databases werederived from this approach.The terms used in the search strategy were: N  1-  Concerning cleft lip and palate  : Cleft lip, cleft palate, CLP,UCLP, BCLP N  2-  Three dimensional  : Imaging three-dimensional, 3D, threedimensional, image, images, imaging, 3D image, 3D images,3D imaging  N  3-  CT  : Tomography, X-ray computed, computed tomograph-ic, CT, volumetric CT, computed tomography, computerassisted tomography N  4-  CBCT  : Cone beam computed tomography, CBCT, spiralcone beam computed tomography N  5-  Photos  : Photogrammetry, stereophotogrammetr* N  6-  MRI:   Magnetic resonance imaging, magnetic resonanceimage*, MRI N  7-  4D:   4D, 4-dimensional, four dimensional computedtomography N  8-  Ultrasound:   Ultrasonography, echography Figure 1. PRISMA flow chart of the study selection process. doi:10.1371/journal.pone.0093442.g001Imaging Methods for CLPPLOS ONE | 3 April 2014 | Volume 9 | Issue 4 | e93442      T   a    b    l   e    3 .      M    e    t     h    o     d    o     l    o    g     i    c    a     l    q    u    a     l     i    t    y    s    c    o    r    e    s    o     f     C     T    s    t    u     d     i    e    s    w     i    t     h    a    n    o    v    e    r    a     l     l    q    u    a     l     i    t    y    s    c    o    r    e    o     f      $      6     0     % .     F    i   r   s    t   a   u    t    h   o   r    Y   e   a   r    T   o   p    i   c    S    t   u    d   y    d   e   s    i   g   n    M   e   a   s   u   r   e    S    t   a    t    i   s    t    i   c   s    S   c   o   r   e    A    B    C    D    E    F    G    H    I    J    K    L    M    N    O      R    a    s     1     9     9     7    s    a    g     i    t    t    a     l    p    o    s     i    t     i    o    n    m    a    x     i     l     l    a    o    o . . .    o     7     5     %     V    a    n     d    e    r     M    e     i     j     2     0     0     1     b    o    n    e    g    r    a     f    t    q    u    a    n    t     i    t    y    o    o . .    o    o     6     9     %     K    a    w    a     k    a    m     i     2     0     0     3     b    o    n    e    g    r    a     f    t     h    e     i    g     h    t ,     d    e    n    s     i    t    y    o    o    o . .    o     6     9     %     V    a    n     d    e    r     M    e     i     j     2     0     0     3     b    o    n    e    g    r    a     f    t    o    o . .    o     7     7     %     K     i    t    a     2     0     0     4    n    e    e     d     b    o    n    e    g    r    a     f    t    o    o . .    o     7     7     %     S    c     h     l     i    e    p     h    a     k    e     2     0     0     6    m    a    x     i     l     l    a    r    y    a    r    c     h    w     i     d    t     h    o    o . .    o    o    o     6     2     %     K     i    m     2     0     0     8     b    o    n    e    g    r    a     f    t    s     i    z    e ,    v    o     l    u    m    e    o    o . .    o     7     7     %     S    u    r     i     2     0     0     8    m     i     d     f    a    c    e ,    m    a    x     i     l     l    a    o    o    o . . .    o     6     6     %     A     l    o    n    s    o     2     0     1     0     b    o    n    e    g    r    a     f    t    s     i    z    e ,    v    o     l    u    m    e    o    o    o    o     7     3     %     S    a     i     j    o     2     0     1     0    o    s    s     i     f     i    c    a    t     i    o    n    p    a     l    s    u    t    u    r    e    o    o . .    o . . .     7     0     %     L    e    e     2     0     1     1    p    t    e    r    y    g    o    m    a    x     i     l     l    a    r    y    r    e    g     i    o    n    o    o    o .    o .    o     6     2     %     L     i     2     0     1     1    m    a    x     i     l     l    a    o    o . . .    o     7     5     %     T    u     l    u    n    o    g     l    u     2     0     1     1    c    e    p     h    a     l    o    m    e    t    r    y     2     d    v    s     3     D    o    o    o . . . .    o     6     3     %     C     h    o     i     2     0     1     2    o    s    s     i     f     i    c    a    t     i    o    n    p    a     l    s    u    t    u    r    e    o    o . .    o .    o     6     7     %     S    e     i     k    e     2     0     1     2     b    o    n    e    g    r    a     f    t    s     i    z    e ,     d    e    n    s     i    t    y    o .    o . .    o . .    o     6     0     %     Y    e     2     0     1     2    p    a     l    a    t    a     l    s     h    e     l     f    e     l    e    v    a    t     i    o    n    o    o . .    o    o     6     9     %   =     F    u     l     f     i     l     l    e     d    s    a    t     i    s     f    a    c    t    o    r     i     l    y    t     h    e    m    e    t     h    o     d    o     l    o    g     i    c    a     l    c    r     i    t    e    r     i    a   ;    o   =     D     i     d    n    o    t     f    u     l     f     i     l     l    t     h    e    m    e    t     h    o     d    o     l    o    g     i    c    a     l    c    r     i    t    e    r     i    a   ; .   =     N    o    t    a    p    p     l     i    c    a     b     l    e .     d    o     i   :     1     0 .     1     3     7     1     /     j    o    u    r    n    a     l .    p    o    n    e .     0     0     9     3     4     4     2 .    t     0     0     3 Imaging Methods for CLPPLOS ONE | 4 April 2014 | Volume 9 | Issue 4 | e93442  The title and abstract of studies were first independentlyscreened by two reviewers (YC and MK). The reviewers werechosen based on their experience of 3D-techniques and cleft lipand palate treatment. Disagreements were resolved by discussionand consensus. After review of only the abstracts, they were scoredas’’ included’’, ‘‘excluded’’, or ‘‘unclear’’. Then, the full text wasretrieved for included articles and articles with unclear abstracts.Full text assessments were performed independently by the sametwo reviewers. Disagreements were resolved by discussion andconsensus. All studies were categorized by the method of imaging used. Quality Assessment The included studies were evaluated according to the qualityassessment instrument used by Gordon et al [17]. This instrumentincludes an assessment of study bias and criteria, as shown inTable 2. Two reviewers utilized the quality assessment instrument(QAI) independent of each other (MK and YC). After that,disagreements were resolved by discussion and consensus. Whenno consensus could be reached, a senior researcher (PF)experienced with this QAI and also familiar with cleft lift andpalate treatment made the final decision. A checkmark was scored when a criterion was fulfilled.Depending on the study design, a maximum of 15 criteria couldbe scored. When certain criteria were not applicable for the studydesign, less than 15 criteria were scored and the non-applicablecriteria were not used for assessing the overall study quality. Studyquality was expressed as the number of criteria fulfilled divided bythe total number of applicable criteria multiplied by 100. Thestudies were grouped according to the method of imaging. In caseswhere criteria were not applicable to the study design, the scoring was marked with a dot. Arbitrarily, a cut-off of 60% or higher wasgraded (after evaluation of the data) as good quality and below60% was graded as poor quality [18]. Statistics Cohens’s kappa statistics were used to assess the inter-observerreliability of the selection of articles based on the full text. Theinter-rater reliability of the quality assessment was calculated using kappa statistics on 23 randomly selected articles scored by tworeviewers (MK and YC). The strength of agreement was definedaccording to Landis and Koch [19]: poor (kappa  , 0.20), fair(kappa=0.21–0.40), moderate (kappa=0.41–0.60), good (kap-pa=0.61–0.80), and very good (kappa=0.81–1.00). Fisher’s exacttest was performed to test for differences in quality between groupsof methods with a cut-off score of 60% for the QAI. SPSS version19.0 was used. Results Study Selection The inter-observer kappa for the reliability of study selectionbased on the full text was 0.76, which qualified as good [19]. Thesearches in PubMed, EMBASE, Cochrane Library, Web of Science, and Scopus yielded a total of 4727 citations and the handsearch provided no additional publications. After adjusting forduplicates, the title and abstract of 2297 citations were screened. After this screening, 1797 articles were excluded because they didnot meet the inclusion criteria. The full text was assessed for the500 remaining articles. All of these articles were retrieved. All,except 2, articles were available in e-journals. Two articles wereretrieved by contacting the author. Reasons for excluding studiesafter full text assessment were: different anatomical region; articleswere letters, opinions, or reviews; and the studies applying finite     T   a    b    l   e    4 .      M    e    t     h    o     d    o     l    o    g     i    c    a     l    q    u    a     l     i    t    y    s    c    o    r    e    s    o     f     C     B     C     T    s    t    u     d     i    e    s    w     i    t     h    a    n    o    v    e    r    a     l     l    q    u    a     l     i    t    y    s    c    o    r    e    o     f      $      6     0     % .     F    i   r   s    t   a   u    t    h   o   r    Y   e   a   r    T   o   p    i   c    S    t   u    d   y    d   e   s    i   g   n    M   e   a   s   u   r   e    S    t   a    t    i   s    t    i   c   s    S   c   o   r   e    A    B    C    D    E    F    G    H    I    J    K    L    M    N    O      D     i    c     k     i    n    s    o    n     2     0     0     8     b    o    n    e    g    r    a     f    t    o    o .    o     7     9     %     N    a    g    a    s    a    o     2     0     0     8    n    a    s    a     l    s    e    p    t    u    m    o    o . . .    o     7     5     %     O     b    e    r    o     i     2     0     0     9     b    o    n    e    g    r    a     f    t    v    o     l    u    m    e    o    o . .     8     5     %     O     b    e    r    o     i     2     0     1     0    c    a    n     i    n    e    o    o    o . . .     7     5     %     S     h     i    r    o    t    a     2     0     1     0     b    o    n    e    g    r    a     f    t    v    o     l    u    m    e    o    o .    o . .    o     6     7     %     L     i ,     F .     2     0     1     1    m    a    x     i     l     l    a    o    o    o . .    o     6     9     %     V    e     l     i     2     0     1     1     M    a    n     d     i     b     l    e    o    o    o .    o . .     6     7     %     L    e    e    n    a    r    t    s     2     0     1     2     d    e    n    t    a     l    a    r    c     h    e    s     (     G    o    s     l    o    n     )    o    o    o . . . .     7     3     %     L     i     2     0     1     2     N    o    s    e    o    o    o . . . .    o     6     3     %     T    r     i    n     d    a     d    e  -     S    u    e     d    a    m     2     0     1     2     b    o    n    e    g    r    a     f    t    o . . .    o     8     3     %     Z     h    o    u     2     0     1     3    t    o    o    t     h     l    e    n    g    t     h    o    o .    o .    o     6     9     %   =     F    u     l     f     i     l     l    e     d    s    a    t     i    s     f    a    c    t    o    r     i     l    y    t     h    e    m    e    t     h    o     d    o     l    o    g     i    c    a     l    c    r     i    t    e    r     i    a   ;    o   =     D     i     d    n    o    t     f    u     l     f     i     l     l    t     h    e    m    e    t     h    o     d    o     l    o    g     i    c    a     l    c    r     i    t    e    r     i    a   ; .   =     N    o    t    a    p    p     l     i    c    a     b     l    e .     d    o     i   :     1     0 .     1     3     7     1     /     j    o    u    r    n    a     l .    p    o    n    e .     0     0     9     3     4     4     2 .    t     0     0     4 Imaging Methods for CLPPLOS ONE | 5 April 2014 | Volume 9 | Issue 4 | e93442
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