Extension and validation of the protocol of orofacial myofunctional assessment for individuals with cleft lip and palate

Purpose: To perform the expansion, validation of the content, criterion and construct of an orofacial myofunctional assessment protocol for individuals with cleft lip and palate; and to define evaluation parameters for the instrument use. Methods: The expansion of the instrument was performed based on the MBGR protocol; the items, sub items and possibilities of answers of the amplified instrument were analyzed for their clarity by seven examiners for the content validation.. Assessment parameters were developed for the use of the instrument in order to minimize the subjectivity. Four examiners compared the aspects of the protocol to those of another instrument for the criterion validation, and the construct validation was performed comparing the results of pre and post orthognathic surgery treatment. The Content Validation Index and the Kappa, Spearman and Wilcoxon Correlation tests were applied for the validations. Results: The mobility, tonicity and sensitivity aspects of the evaluation were added to the protocol and at content validation, 72% of the items were classified as very clear and 28% as clear. A manual with assessment parameters for all items and sub items of the protocol was constructed. The inter-examiner agreement was moderate. In the comparison of the protocols, for the validation of the criterion, a good relation among them was observed. For the validation of the construct the protocol was able to identify outcome after the treatment. Conclusion: The protocol was expanded and considered validated in its entirety: content, criterion and construct, and evaluation parameters for its use were established.


Extension and validation of the protocol of orofacial myofunctional assessment for individuals with cleft lip and palate
Ampliação e validação do protocolo de avaliação miofuncional orofacial para indivíduos com fissura labiopalatina

INTRODUCTION
Cleft lip and palate is one of the most common malformations, which needs interdisciplinary treatment, among them Speech, Language and Hearing Sciences, due to the impairments that this condition causes: esthetic, anatomic and functional alterations (1) .
The speech language and hearing evaluation in these cases should be performed along the different life phases, until the end of the craniofacial development, mainly by surgical procedures to which the individuals are submitted (2) .Among these procedures, there are: primary and secondary surgeries for the correction of the lips and palate, pharyngeal flap surgery for the correction of the velopharyngeal insufficiency, nasal surgeries, alveolar bone grafting, orthognathic surgery for the correction of dentofacial deformity, among others.Many of these procedures cause morphological and functional modifications in the oral cavity that will affect the stomatognathic system balance generating dysfunctions.One of them is orofacial myofunctional, characterized by the alteration of the normal position at rest of the lips and tongue; also of the sensibility, tonicity and mobility of the structures; of the respiratory mode, of the chewing function and the normal physiological standards of the deglutition oral phase; speech production implication, also involving velopharyngeal function and its impact on speech.
This way, standard evaluation protocols are essential due to the scientific evidence that they present and the possibility of following up the cases.A standard protocol of orofacial myofunctional evaluation turns it possible to make the orofacial myofunction dysfunction diagnosis, as well as to establish prognostic and comparison of treatment results.Specific protocols have unique characteristics for certain populations, such as the case of cleft lip and palate, that allows more accurate evaluations (3) for including features related to this condition.For a protocol to be considered a reliable measure, it is necessary the validation of this instrument, whose complete form involves three steps: content validation, related to the construct and drafting of the items that comprise the instrument, analyzed by specialists; criterion validation, that relates the items of the protocol to similar items of another instrument, considered gold standard; and validation of the construct, that verifies if the instrument represents the concepts for which it was created (4) .The literature suggests that, besides standardized and validated protocols, it is necessary previous and systematic training for the examiners regarding their application, in order to minimize the differences among the examiners with and without experience (5) .
Thus, the objective of this study was to perform the extension and validation of the content, criterion and construct of the orofacial myofunctional evaluation protocol for individuals with cleft lip and palate suggested by Graziani et al. (6) .The idea is to make this instrument more complete and present higher methodological strictness, as well as to define evaluation parameters for its use to contribute with clinical practice and scientific community.

METHODS
It is about an observational, transversal and prospective study that spared the use of the Informed Consent Form for using secondary sources, and approved by the Research Ethics Committee on Human Beings under n° 1.651.873.For sample calculation, it was based on the validation of competitor criterion considering the correlation coefficient of r=0.5 and the estimated sample was of 30 individuals.

Protocol expansion
For the expansion of the protocol suggested by Graziani et al. (6) , were added the aspects relevant to the mobility and tonicity evaluation of the orofacial structures, based on the proposition "Orofacial Myofunctional Evaluation-protocol MBGR" (3) , as well as of sensitivity.Besides, items of some aspects were modified in order to turn the instrument more complete, and called PROTIFI the shortening for "Protocol of Orofacial Myofunctional Evaluation for Individuals with Cleft Lip and Palate".The evaluation of the sensibility involved pain to the touch validation based on the MBGR (3) protocol, as well as lips, incisive papilla, tongue, cheeks and mentual tactile sensitivity.The latest one is tested by means of an aesthesiometer, a group of six nylon monofilaments (Semmes-Weintein) that preseny different diameters, which touch the tested region (7) .

Content validation
The items of the extended protocol were analyzed by seven examiners with expertise in the Orofacial Motricity area and at assessing individuals with cleft lip and palate.According to Alexandre and Coluci (8) proposition, it was applied the formula of the Content Validation Index (CVI) to measure the percentage of agreement among the examiners.Each one of the items were analyzed regarding the clarity from a Likert scale of four points: 1 = very clear, 2 = clear, 3 = little clear and 4 = without clarity.It was considered valid the item that reached agreement above 80% among the examiners and, in case this percentage was not reached, the item would be reformulated or excluded.

Definition of the evaluation parameters
For each item of the instrument were established parameters for its analysis, as well as it was attributed a punctuation for the possibilities of answer in each item.This process was performed based on the literature, from the consultation to the database: Medline, Scielo and Lilacs, in the period from 2012 to 2017, by means of the keywords in Portuguese: fissura palatina + fenda labial + avaliação + fonoaudiologia + sistema estomatognático + fala + mastigação + deglutição + respiração + estudos de validação, and in English: cleft palate + cleft lip + assessment + stomatognatic system + speech + chewing + swallowing + breathing + validation studies.It was also considered the experience of professionals and researchers that perform the orofacial myofunctional assessment of individuals with cleft lip palate, with the objective of unifying the parameters among the examiners.

Criterion validation
Were selected photos/recordings of 30 young adults (average = 23.8 years old) with operated cleft lip and palate, enrolled in a specialized institution for these cases.The selected individuals belonged to both genders and did not present other associated impairment, such as hearing loss, neurological or syndrome problem, according to the notes on the institution record.
Four speech-language pathologists, among them an experienced examiner (E4) and three examiners without experience at assessing orofacial myofunctional of individuals with cleft lip and palate (E1, E2, E3), were invited to analyze the images.Previously they received live training for the application of the proposed instrument, according to the assessment parameters established, as well as guidelines on the application of the protocol of Myofunctional Assessment with Scores -AMIOFE (9) , selected as criterion.
The examiners had title of Master and/or PhD and experience from 10 to 20 years helping cases of cleft lip and palate.The experienced examiner was selected for having experience at assessing orofacial myofunctional cases with cleft lip and palate and systematic training for the application of the protocol and performed his analysis individually.The other examiners were considered without experience for not performing the full orofacial myofunctional assessment of these individuals, that is, they prioritize the assessment of the velopharyngeal and speaking function without describing the other morphological and functional aspects proposed in the protocol of study, and proceeded the analyses in consensus.All of them filled out the assessment protocols according to the corresponding items (Chart 1).

Construct validation
From the selected patients, 19 were submitted to orthognathic surgery for maxillary advancement, in order to correct maxillomandibular discrepancies.At this stage, were compared the oromiofunctional assessments pre-surgical (1 to 3 days before the surgery) and post-surgical (3 to 6 months after the surgery), to verify the protocol ability to identify treatment results.

Data analysis
The results were presented in tables.At the content validation analysis, it was applied the equation of the Content Validation Index -CVI (8) ; at the criterion validation, it was used the Kappa Test to verify the agreement among the examiners (experienced examiner x examiners without experience).Data interpretation was based on the classification: almost perfect (0.80-1.00), substantial (0.60-0.79), moderate (0.40-0.59), regular (0.20-0.39), poor (0.00-0.19) and without agreement (<0).The correlation coefficient of Spearman was used to analyze the correlation among protocols; and for the validation of the construct in order to verify the result of treatment at the comparison between pre and post orthognathic surgery, the Wilcoxon Test was applied.

RESULTS
The extension of the items of the protocol was based on MBGR protocol, with the addition of the items: mobility, tonicity and sensibility, that are of general nature in other populations.The terminologies of some answers of the protocol were modified/added and others excluded, in order to turn them clearer (Chart 2).
At Content Validation, the items, sub items and answer possibilities for the extended protocol were analyzed regarding their clarity and, after this analysis, three sub items were excluded relating to the morphological aspects of the items.They are: Lips, it was excluded the sub item upper oral vestibule; Tongue, it was excluded the sub item limitation of the tongue frenulum function; and Soft Palate, it was excluded the sub item levator muscle insertion.These items were excluded for being classified by most of the examiners as "little clear" and "without clarity" and not reaching the agreement percentage of 80% among them.The results of the Content Validation Index application are presented on Table 1.
The orofacial myofunctional assessment protocol for individuals with cleft lip and palate (PROTIFI) has got morphological and functional aspects of the stomatognathic system, that allow a detailed evaluation (Appendix A).With the objective of making it easier the application of PROTIFI, it was elaborated a manual which contains assessment parameters in order to reduce subjectivity, which presents the characterization of each answer possibility of the evaluated items, as visualized in Appendix B.
For the Criterion Validation, it was verified substantial agreement among the examiners for the occlusion (overbite) and almost perfect for the other aspects (Table 2).
Regarding the results of the items corresponding to the protocols, it was verified good correlation among them (r >0.7), reminding that the scores among the protocols are inversely proportional (Table 3).It was considered valid the sample calculation (n=30), that was based on the competitor criterion validation considering a correlation coefficient of r=0.5.
Chart 2. Results of the protocol aspects that suffered modification/addition/exclusion ASPECTS ITEM MODIFICATION/EXTENSION/EXCLUSION Tongue mucosa replacement of the "marked" word by the word mark and addition of the word "orthodontic" Palatine Tonsils presence replacement of the word "present" by the word "yes" Teeth n° of teeth Written in full the "right" and "left" side of the quadrant oral health (teeth/gingiva) replacement of the word "good" for "adequate" and the word "bad" for "inadequate" use of braces addition of the word "orthodontic" Use of prosthesis addition of sub items "partial" and "total" Hard Palate fistula addition of sub items "present" and "absent"; replacement of the word "vestibular" for the word "oral vestibule" size exclusion of the option: "small", "medium" and "big", and addition of the item for taking note of the measurement in millimeters Soft Palate and Uvula fistula addition of sub items "present" and "absent"; replacement of the word "vestibular" for the word "oral vestibule" size exclusion of the option: "small", "medium" and "big", and addition of the item for taking note of the measurement in millimeters Soft palate and uvula mobility transfer of this item to mobility evidence Pharynx sidewalls transfer of this item to mobility evidence; replacement of the options "good", "regular", "little" and "unobservable" for "adequate", "altered" and "without movement" rear wall exclusion of the option "attempt" Breathing nasal flow replacement of the terms "when arriving" and "after cleaning" for "precleaning" and "post-cleaning" Speech resonance addition of this item that involved the options "hypernasality" and "hyponasality", displaced from other sub item, and addition of the option "balanced" speech language disorder replacement of the words "speech language disorder" for the words "speech language alteration" Acoustic distortion and mouth opening Exclusion of these items Functional adaptation replacement of the word "interposition" for the words "lingual interdentalization" mandibular movement replacement of the options "deviation to the right" and "deviation to the left" for the option "deviation" and of option "anteriorization" for "projection" saliva addition of sub item "on the commissures" Mobility lips addition of this item that involved the options: bulging, retract and snap tongue addition of this item that involved the options: palate sucking, snap, vibrate, touch lips and commissures, in the incisive papilla, on the right and left cheek soft palate addition of this item pharynx addition of this item Tonicity lips addition of this item cheeks addition of this item tongue addition of this item mentual addition of this item Sensibility pain to the touch addition of this item that involved the options: anterior temporal, superficial masseter, trapezium, sternocleidomastoid and ATM tactile sensitivity addition of this item that involved the options: mentual, lips, incisive papilla tongue and cheeks At the Construct Validation, the results of the evaluations of 19 patients submitted to orthognathic surgery were compared in order to observe the treatment results.The return of these patients for assessment was from 3 to 6 months after the surgical procedure.It could be observed modification of some of the analyzed aspects, such as: cheeks, tongue, occlusion, breathing and speech/voice analysis involving articulation, intelligibility and resonance, besides pitch loudness and type of voice (Table 4).

DISCUSSION
This study was developed with the purpose of extending and performing the total validity of an orofacial myofunctional assessment protocol for individuals with cleft lip and palate, previously developed (6) for which it was performed the content validity.In this regard, the literature has referred to the importance of standardized and validated tools for use at clinical practice (10)(11)(12) .The validation is a continuous process, based in a greater oy minor level of evidences: content validation, criterion validity, construct validity (13) , and refers to an investigation to determine the accuracy of a prediction or inference performed from scores of a test (4) .
Besides, with the application, at clinical routine, of the protocol previously proposed (6) , it was perceived the need to add the items mobility, tonicity and sensibility of the orofacial structures, because such aspects contribute to the understanding of the dysfunctions and the establishment of the treatment planning.It should be highlighted that these added aspects comprise some protocols and propositions of general orofacial myofunctional assessment, applied to several populations (3,9) .
Once new items have been added and there were terminology modifications, in order to clarify the wording, new content validation is necessary.Some authors refer that at each instrument modification there is the need to investigate the clarity and usefulness of the changes (8,14) .This is because the content validity investigates the representativeness of the items that comprise the instrument and the objectives to be measured, analyzed by specialists (8,15) .
It was observed, from the opinion of seven selected experts, who contributed to this validation phase, that the extended content allows to assess what is proposed, once the application of IVC (8) reached percentage above 80% for the majority of the items and sub items analyzed.This way, according to the values established by the literature (14,16) , the results obtained in this study show that the protocol can be considered valid regarding its content.
At clinical practice, it is observed that even skillful examiners can adopt different reference parameters at orofacial myofunctional assessment to analyze morphological and functional aspects.Thus, to mitigate the subjectivity at the assessment process, this study has established analysis parameters for the application of the assessment protocol from the characterization of each possibility of answer of the sub items of the protocol.It is believed that the use of this material can also turn it possible the training of students for the assessment process of these patients, as well as it will help less experienced professionals to use it, and will also permit the standardization of inter center results.However, it is worth highlighting that there is the need of systematic training of the assessors for the application of the elaborated protocol, as suggested by some authors (5,17) .
The validity of the competitor criterion was used in this study and had as objective to correlate the results of the studied protocol, simultaneously to the other validated instrument and with the same destination, classified as (AMIOFE) criterion, which presented similar measures.In this type of validation, the more the protocol results are related to the standard (criterion) bigger will be the criterion validity (18) .It can be observed in the results of this study good agreement among the examiners and coherence at their results reproducibility with agreement above 90% among the instruments, as well as strong correlation among them (r >0.7).This way, such results did not differ from those found in an instrument already validated and, therefore, the result reached with this tool is considered reliable.
The construct validity refers to a specific concept that was built for a scientific purpose.In this study, the aim was to verify the protocol ability of identifying orofacial myofunctional disorders, mainly after interventions performed, in this case, orthognathic surgery.The surgery objective is to heal maxillomandibular discrepancies once the orthodontic treatment was not enough to correct skeletal disharmony and of the soft tissue (19) .This kind of surgery intervention turns it possible the facial harmony, balanced occlusion and recovery of functional standards (20,21) .The comparison of the obtained results of the pre and post-surgery assessment for the construct validation, showed differences for the following aspects: occlusion, cheeks, tongue, breathing and speech/voice analysis.The ability of the protocol in differentiating pre and post-surgery alterations assures the construct validity of this instrument.
The study sample was formed by individuals that presented dentofacial deformities, related to alterations at horizontal, vertical and transversal relations.Thus, as expected and observed, the occlusion aspect suffered modification in relation to the initial condition, after performing the orthognathic surgery.
In literature it is not common reporting on alterations in the cheek mucous in these cases but, it is believed that orthodontic intervention at long term, due to existing malocclusion, simultaneous to the functional adaptation for the performance of orofacial functions, mainly chewing and speaking, can lead to the presence of dental marks, wounds and linea alba.In the other hand, after the dentofacial deformity correction, some muscle and bone balance could justify the reduction of occurrence of these alterations.Some authors (22,23) also refer that there is some increase in the cheeks internal angle after the repositioning of the bone bases, what would involve less friction between occlusion and musculature.
In the cases of maxillomandibular discrepancies, it is common the habitual tongue posture be altered in the mouth floor (24,25) , accommodated close to the mandible.From the analyzed cases, it could be observed a spontaneous adaptation of this structure after the surgical procedure; the tongue adapted to the palate and its posture was adequate, however this modification not always happen for all the cases (26) .Such adaptation might have happened because it is a flexible and adaptable structure regarding its format, posture and mobility to perform the orofacial functions (27) .
For the adequate speech production there is the need of integrity of the structures that comprise the stomatognathic system, and the tongue is an important structure at the performance of speech motor control.From the analyzed cases, it could be observed that before the surgery, the speech was affected, with phonetic distortions, mainly related to the change of articulatory place, once the altered morphology impairs the correct articulatory performance.However, for these cases, it was observed that, after the anatomic change, there was the adaptation to these deviations (28) .Particularly for the sample analyzed, it was not observed any change on the vocal condition after the surgical procedure.
The cleft lip and palate can take to the reduction of the nasal cavity dimensions, with great probability of occurring oral breathing, fact observed in the studied sample, once approximately 80% of the individuals presented oral breathing.It is worth to highlight that the maxillary advancement can also have effect on breathing, because there is an increase in the dimension of the middle third of the face, together with the volume increase of the pharyngeal airway space and, consequently, breathing improve (29) .Such findings were the same found in this sample once for the majority of the cases, the breathing was presented nasal after the surgery, as expected.
This way, it could be observed that the analyzed individuals presented orofacial myofunctional disorders before the surgical intervention, and that in some cases, it did not occur adaptation after the surgical procedure, what justifies the action of the speech-language pathologist in the team (30) .

CONCLUSION
The assessment protocol of orofacial myofunctional for individuals with cleft lip and palate had its items extended to become more complete and, with the obtained results, it showed evidence of the validity of content, criterion and construct to be used at the diagnosis of orofacial myofunctional disorders in individuals with cleft lip and palate.Saúde oral dos dentes: observar as arcadas superior e inferior adequada ausência de resíduo alimentar/placa bacteriana/ cárie e/ou fratura regular presença de resíduo alimentar/placa bacteriana inadequada presença de resíduo alimentar/placa bacteriana/cárie e/ou fratura Saúde oral da gengiva: observar a gengiva entre as arcadas superior e inferior adequada ausência de hiperemia/edema/ferida e/ou sangramento regular presença de hiperemia/edema inadequada presença de hiperemia/edema/ferida e/ou sangramento Uso de aparelho ortodôntico: observar as arcadas superior e inferior ausente ausência de aparelho ortodôntico presente:

Table 2 .
Inter Examiners Agreement (experienced x non-experienced) for the validation of the Criterion by Kappa Test

Table 3 .
Analysis of Criterion validity among the aspects corresponding to the PROTIFI and AMIOFE Protocols by Sperman Correlation (n=30) *p≤0.005