Instrument
Participants
Reporter
Instrument development
Pediatric Voice Handicap Index (pVHI) [39]
Children aged 4–21 years old with laryngeal airway concerns and healthy children
Caregiver (parent)
23 items adapted from an adult instrument, the Voice Handicap Index [40]
Children aged 2–18 with tracheostomies or decannulation; 385 children with ORL conditions not limited to voice
Caregiver (parent)
4 items adapted from an adult instrument, Voice Outcomes Survey [43]
Pediatric Voice-Related Quality of Life (pVRQOL) [44]
Children aged 2–18 who visited ORL office with all conditions, not limited to voice
Caregiver (parent)
10 items adapted from an adult instrument, Voice-Related Quality of Life (VRQOL) [45]
Pediatric Voice-Related Quality of Life (pVRQOL) [46]
Children aged 3–15 who visited a pediatric voice clinic
Child and caregiver (parent)
10 items adapted from an adult instrument; children completed a child-adapted version of the pVRQOL
Children aged 6–12 with dysphonia due to vocal fold nodules, edema, paralysis
Child
30 items used directly from an adult instrument; VHI [40]. Some items are not appropriate for children
Pediatric Voice Symptom Questionnaire (PVSQ) [48]
Children aged 9–13 (French)
Child and caregiver (parent)
29 items developed from interview study [49] and further work to assess item comprehension. Separate parent and child versions
Children’s Voice Handicap Index (CVHI) and CVHI-P for parent report [50, 51]
Children aged 8–14 (Italian; English translation)
Child and caregiver (parent)
10 items from adult measure, VHI-10; then modified based on child interviews [52]
As described in a systematic review on the topic of voice-related quality of life and its measurement [38], instrument development must start by talking with patients. To develop an instrument concerning children’s voices, initial input must come from children. As a first step, interview or focus group information must be obtained and incorporated into instrument design and content. Rigorous qualitative research methods, using grounded theory analysis and coding, should be used to discover themes that apply to children who have life experiences with a voice disorder. These interviews or focus groups must be performed by examiners trained in these methodologies. In contrast to trained interviewers and focus group facilitators, healthcare providers are trained to sift, combine, remove irrelevant information, and summarize from a patient’s clinical and oral histories to form a diagnosis and treatment plan. While this approach works very well in the clinic, the risk in using these clinical history-taking methods in qualitative research is one of filtering a patient’s experience through the lens of the practitioner and not forming a true picture of the information as conveyed by the patient. Thus, it is rarely acceptable to replace a rigorous interview or focus group process with retrospective review of medical histories or chart notes in this initial process of instrument development. It is even worse to start with an instrument developed for a different population and apply it elsewhere.
When themes are discovered in patient interview transcripts through systematic coding, instrument items can be developed that target those themes [39–52]. Appropriate triangulation testing can be used to determine if items are comprehensible to children and written in ways that define those initial themes. The second and third stages of instrument development involve field testing the instrument and psychometric analyses [38]. Unfortunately, most of the PROMs used in the area of voice disorders did not follow these standardly accepted instrument development methodologies [38].
When direct input from children with voice disorders is not part of the development process, the result is a violation in the first step of instrument validation. With the exception of the PVSQ (French) [48], all pediatric voice-related PROMs are derived from adult instruments. That is, development of these instruments began with adult questionnaires that were then revised to reference a child’s voice rather than an adult’s voice. For example, the pVOS [41, 42], pVHI [39], and pVRQOL [44] were developed by changing item referents from an adult patient’s voice (e.g., “your voice”) to that of a child (e.g., “your child’s voice”). The CVHI [50, 51] used an adult instrument as a starting point, then interviewed children, and revised items. As such, while these PROMs had the goal of assessing the child’s perspective, they were unfortunately not developed using methods that allowed realization of this goal.
As shown in Table 13.1, most currently available pediatric voice PROMs use proxy administration. As discussed earlier in this chapter, parent or caregiver proxy assumes that a more accurate report may be obtained from a parent or caregiver than from the child. Other assumptions specific to voice are that children may have a limited awareness of their voice disorders, that parents are knowledgeable about the entirety of their child’s perceptions regarding the voice disorder, and that children and adults share a similar framework for the concept of quality of life and the manner in which a voice disorder contributes to quality of life. We know that these things are not true [12, 49, 52]. As such, proxy administration may be biased toward the views of the respondent and can be problematic, especially when items reflect internal states, such as thoughts, emotions, and experiences that occur independently from the parent/caregiver. It is not surprising that in the area of pediatric voice disorders, research has shown that parents and children are often not in congruence regarding the child’s voice-related quality of life [49, 52]. Thus, asking children to provide responses about their own voices is paramount. Children as young as 6 years old are aware of their voice disorder and capable of discussing their life experiences regarding their voices [49, 52]. Therefore, the use of parent/caregiver proxy responding for children 6 years old or older is poorly justified. As stated by Branski et al. (2010) [38], “Manipulation of instruments to apply to other populations for proxy application violates the fundamental tenets of instrument development.” The need for more instruments that emphasize child reporting is clear.
Currently available pediatric PROM instruments in common use assert that they are reliable and valid [38, 39, 41, 42, 44]. Reliability reflects the degree to which a measure, items within a measure, instrument, or tool provides a stable or repeatable value [53]. For instance, a questionnaire administered twice should have values that are relatively similar to be considered reliable. Additionally, items within a questionnaire that measure similar constructs should be correlated (internal consistency). Validity can be defined as the degree to which an entity, in this case a pediatric voice PROM, accurately reflects an underlying truth. Therefore, a valid pediatric voice PROM would faithfully reflect the viewpoints of children with voice disorders. There are multiple types of validity; a complete list and definitions of different components of validity are beyond the scope of this chapter (see Kimberlin, 2009, for a review) [54]. Validity can be complex to determine [55] and cannot be claimed solely with statistical testing in the absence of appropriate instrument development. In this vein, it has been stated that validity and reliability best refer to data, and not to measures [55]. An example given by Sechrest [55] is a tape measure – this is a valid and reliable instrument for measuring the length of a piece of lumber only inasmuch as its use conforms to generally accepted principles of how to use a tape measure. As mentioned previously, validity of an instrument depends upon how it was designed, how it is administered, and whether the population tested is reflective of the target population for use of the instrument, in addition to statistical analyses and psychometrics. Several of the pediatric voice PROMs in current clinical use were validated, using parent proxy, with children who had complex airway disturbances and tracheostomies. While these children may also have had voice disorders, the voice-related quality of life concerns may not be typical of children with more commonly occurring benign vocal fold lesions. Further, voice-related quality of life concerns may have been part of a larger constellation of illness that arguably could have more grossly affected parent perceptions of quality of life. Thus, while statistical tests may reassure us that a pediatric voice-related PROM available for use is valid, and claims of validity are made in published papers, inadequate development protocols, proxy administrations, and other practices may not be consistent with this notion.
The limitations in current approaches for measuring pediatric voice-related quality of life described in this chapter are echoed in the larger context of health-related quality of life assessment in children [26]. In summary, the major limitations include faulty development protocols that do not include qualitative analysis of direct interviews or focus groups with children, use of an adult instrument revised for children, and proxy administration. It must also be acknowledged that children can view the concept of health-related quality of life differently throughout the rapidly changing childhood years. Therefore, thought should be given to the reasonableness of collapsing data from the wide childhood age range into a single version of an instrument [26]. This issue has not been considered in any of the currently available pediatric voice PROMs shown in Table 13.1. Research has shown that children’s perceptions of their voice disorders differ across the age range, where younger children are primarily focused on physical variables and adolescents add concerns around emotional factors to the previously established physical concerns [52]. These flaws in instrument development result not only in faulty assumptions regarding pediatric PROMs and inadequate tracking of treatment effects, but do not easily allow study of other interesting aspects of voice-related quality of life in children. With a properly designed and validated pediatric voice PROM instrument, it is possible to study how a voice disorder affects children’s function in educational settings and within a family context. In addition, it is possible to examine how developmental changes/maturation affect the manner in which a voice disorder is perceived and how particular dynamic variables, such as emerging independence and social relationships, influence voice outcomes. As pediatric voice PROMs are refined and developed, answers to these important questions about the lives of children will be answered.
Conclusion
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