Proposal and content validation of an orofacial myofunctional assessment protocol for individuals with cleft lip and palate

Purpose: To create and validate the content of an orofacial myofunctional assessment protocol for individuals with cleft lip and palate. Methods: The first version of an orofacial myofunctional assessment protocol for individuals with cleft lip and palate was created by two speech-language pathologists, who contemplated the structural and functional aspects of the stomatognathic system. This version was analyzed by other two speech-language pathologists experienced in cleft lip and palate assessment, who suggested changes that led to the second version of the protocol. Dynamic and static images necessary for performing the orofacial myofunctional examination were recorded from three individuals with cleft lip and palate, who represented three life stages: childhood, adolescence, and adulthood. Five examiners evaluated the images, applied the proposed protocol, and judged each item regarding its clarity to validate the content, from Content Validity Index. Results: The assessment protocol was finalized with 13 items, ten related to structural aspects and three related to functional aspects, with their corresponding sub-items. The general agreement in the validation of its content was 100%, so that only one stage was required. Conclusion: A protocol to evaluate the orofacial myofunctional aspects of individuals with cleft lip and palate was created with 13 items, as well as their corresponding sub-items, and its content was validated.


INTRODUCTION
Cleft lip and palate can affect the lip, the palate, or both, and can be associated with other more complex malformations (1) .In such cases, a number of stomatognathic system structures are affected, which requires us to understand the alterations presented.Thus, the use of a protocol to perform the orofacial myofunctional assessment facilitates establishing the diagnosis, defining the conduct and treatment planning, and carrying out all relevant referrals.
It is recommended (2) that the evaluation be performed at least twice in the first year and once in a year until adolescence, the period when the pharyngeal tonsil undergoes the process of involution; and after this phase, it should be conducted every 2 years until the completion of dental-skeletal development.Furthermore, it should also be performed before and after the interventions.This assessment shall include aspects such as anatomy and physiology, language, speech, and voice, as well as investigate the velopharyngeal function by instrumental methods (3) .
The use of a standardized assessment tool facilitates the comparison of pre-and post-treatment results and leads to the discussion among professionals from different study fields (4)(5)(6) .In several areas of health, the validation of assessment tools has been performed to obtain more accurate and reliable results (7)(8)(9) .In Speech-Language Pathology and Audiology, some studies validated assessment protocols for diverse populations (10)(11)(12)(13)(14) .
Specifically for individuals with cleft lip and palate, the literature presents some validated instruments (14,15) , which cover aspects related to speech.In Brazil, professionals who assist individuals with cleft lip and palate use their own forms of evaluation, which require standardization and validation to facilitate the comparison of results and the development of research.This paper aims to contribute to the development and validation of the contents of a specific orofacial myofunctional assessment tool for individuals with cleft lip and palate, which favors the scientific improvement (16) in this field and supports the proposal of the Orofacial Motricity Committee of the Brazilian Society of Speech-Language Pathology and Audiology.

METHODS
The study was approved by the Research Ethics Committee of the Hospital for Rehabilitation of Craniofacial Anomalies of University of São Paulo (USP), under protocol no.200.397, and all participants signed an informed consent.
For the development of the first version of the orofacial myofunctional assessment protocol, literature was consulted and, based on the clinical experience of two experts in orofacial motricity, particularly in cleft lip and palate patients, items and subitems were proposed on aspects related to the stomatognathic system and the performance of orofacial functions, in addition to possible answers for each item.
This version was presented to two other guest speechlanguage pathologists, with broad experience in the assessment of individuals with cleft lip and palate, who analyzed the protocol with respect to the items, subitems, and possible responses, and thus the second version was obtained after adjustments.
Static and dynamic images of three operated unilateral cleft lip and palate individuals at ages 7, 14, and 20 years, representing three stages of life: childhood, adolescence, and adulthood, were selected.Such individuals, randomly selected, were taken from a sample of 75 individuals from another study, and individuals with neurological or motor problems, hearing loss, syndrome or other associated malformations were not included.
For image capturing, subjects sat on a chair with a back support and feet flat on the floor.The images obtained with a digital camera (Sony DSC-HX1 model) helped in the assessment of each item and subitem of the proposed protocol, and an endoscopic camera (CCC Waterproof USB Endoscope, 10 mm), specifically for capturing oropharyngeal images, was used as well.The camera was attached to a tripod and positioned in front of the participants.The lenses were 1 m away from them, to frame the shoulders, neck and face.To have a better visualization of lips, tongue, hard palate, and soft palate, the camera was approximated.A single professional was responsible for capturing the images, which were stored in a computer and transferred to a mobile device (flash drive) to be subsequently analyzed.
Five new examiners with clinical experience ranging from 6 to 20 years in the care of individuals with cleft lip and palate analyzed the images from the proposed protocol.They received previous verbal and written guidance as to the completion of the protocol.After assessment, the examiners rated each item on its clarity in a four-point scale: 1 = no clarity; 2 = unclear; 3 = clear and 4 = very clear, to perform content validation by applying the equation of the Content Validation Index (CVI) (17) .If the examiners marked options 1 or 2, the items had to be reformulated (18) .

RESULTS
The orofacial myofunctional assessment protocol specific for individuals with cleft lip and palate was elaborated after analysis by four speech-language pathologists.It contains 13 items, 10 related to structural aspects and 3 related to functional aspects, with their corresponding subitems (Appendix 1).The items included referenes to the lips, tongue, cheeks, teeth and occlusion, palatine tonsils, hard palate, soft palate, and uvula and pharyngeal walls, as well as breathing, speech, and velopharyngeal functions.
In content validation, the investigators examined the items on the basis of their clarity for the calculation of the CVI (Tables 1 and 3); 75% of the examiners classified the items as "very clear" and 25% as "clear", with 100% agreement.

DISCUSSION
The purpose of this study was to develop and validate the content of a specific instrument for orofacial myofunctional assessment for individuals with cleft lip and palate.Some Orofacial assessment in cleft lip and palate CoDAS 2015;27(2):193-200 aspects concerning the general orofacial myofunctional assessment, such as mobility, muscular tonus, chewing and swallowing, were not anticipated, as it is believed that such assessments do not differ from those applied in other cases and thus other available assessment tools (5,6) can be used.
In the first version proposed, the examiners who analyzed it suggested some adjustments related to possible answers, which helped clarify the proposal.According to some authors, the assessment of the instrument by experienced and competent examiners in the specific area to be tested is essential and should be considered in the content validation process (8,13,29) .The content validation refers to the judgment from different examiners of an instrument, who must consider the items regarding content and the relevance of objectives to be measured, as well as make suggestions on how to remove, add, or modify items (7) .On the basis of evaluation conducte by a group of experts, some authors performed content validation only by means of qualitative analysis (18,29) , whereas other authors considered it highly relevant to perform a quantitative analysis (8,13) .
In this study, for the content validation, performed through analysis from the examiners, images from individuals with complete unilateral cleft lip and palate were selected, due to its incidence and also because this type of cleft affects many aspects of the stomatognathic system.Thus, all items proposed in the protocol could be included.In addition, one individual at every stage of life (childhood, adolescence, and adulthood) was selected so that the instrument could be applied to different age groups.
In the content validation, the CVI was used to measure the percentage of agreement between the five examiners who assessed the second version.The 100% agreement was obtained, in which 75% examiners classified the items as "very clear" and 25% as "clear."According to some authors, as the examiners did not mark the "no clarity" and "unclear" options, there was no need to exclude or reformulate any item (18) .
The proposal was adequate and the content of the instrument was validated in a single step, with a percentage of agreement above that established in the literature to be considered valid (13,17,18,29) .Thus, the content of the instrument proposed in this study was considered to be a valid and accurate measure for the 13 items evaluated, as well as their subitems.
This study did not aim to establish assessment criteria for the judgment of certain items, which will be conducted in a new study, as well as the continuity of the validation process of the instrument.In the course of this study, it was verified that the quality of images requires suitable equipment and techniques to facilitate the visualization of detailed structures for assessment.

CONCLUSION
A proposed protocol for the orofacial myofunctional assessment of individuals with cleft lip and palate, consisting of 13 items covering both structural and functional aspects, was developed and its content was validated.
*AFG participated in the study idealization, data collection, analysis and interpretation, and drafting of the article; APF participated in the idealization of the study, data analysis, and interpretation; KFG participated in the idealization of the study, data analysis and interpretation, and drafting of the article.

Table 1 .
Distribution of the frequency of the content validity index regarding the assessment of aspects of the lips, tongue, cheeks, palatine tonsils, teeth, occlusion, and hard palate

Table 2 .
Distribution of the frequency of the content validity index regarding the assessment of aspects of the soft palate, uvula, pharynx, and mirror test

Table 3 .
Distribution of the frequency of the content validity index regarding the assessment of aspects of speech, voice, and breathing