Phase 1 consisted of pre-procedure otoscopy, a routine ear examination with FLACC evaluated from 30 s prior to the speculum entering the ear canal until 60 s after the ear exam. Phase 2 included earset installation and filling comprised of insertion of a soft earplug and filling the ear canal with otic anesthetic solution which can yield a surprising and ticklish sensation with FLACC evaluated from 30 s prior to the iontophoresis earset first touching the child's head until 60 s after otic anesthetic solution filling of the external ear canal was complete. Phase 3 consisted of iontophoresis during which electrical current is delivered to the ear in the presence of the anesthetic solution to numb the ear drum with FLACC evaluated from the initiation of the iontophoresis process until 60 s after the otic anesthetic solution was removed from the external ear canal. Phase 4 included tympanic membrane anesthesia assessment, in which the ear drum is lightly touched with a dull otologic instrument to determine if the TM is insensate or not, and tube placement during which a myringotomy incision is made and the tube placed across the ear drum. Phase 4 FLACC evaluation was initiated from the moment the speculum enters the ear until 60 s after the automated tympanostomy device is withdrawn from the external ear canal. Phase 5 consisted of the 3-min period post-procedure, from the end of the prior phase until 180 s later. In addition, a sixth FLACC score was assigned as an overall score for the entire procedure. A mean FLACC score for each procedural phase and overall was calculated from the average FLACC scores of the study participant population.
Results
Most patients (91.4%, 203/222) were medically indicated to receive bilateral tubes, and tubes were successfully placed in the majority of the sample. Successful tube placement was achieved for 86% (175/203) of bilaterally indicated participants, and 100% (19/19) unilateral patients (Lustig et al., 2020). As per the FDA-agreed upon protocol, patients were excluded from FPS-R or FLACC analyses if they did not successfully complete the procedure for all indicated ears. Reasons for incomplete procedures included excessive movement or other behavior that interfered with completion (n = 11), inadequate anesthesia as determined by the tympanic membrane anesthesia assessment (n = 7), discomfort/anxiety (n = 4), anatomic challenges (n = 3), intolerance of iontophoresis (n = 2), and partial tube medialization (n = 1). The following results reflect the 194 children with successful tube placements. Per protocol and so that reports reflected the entire procedure, no FPS-R scores were collected from the 11 children for whom tube placement was incomplete. Similarly, FLACC analyses included only patients with successful procedures given that unsuccessful procedures would be dissimilar (e.g., truncated intervention, shortened procedure). Post hoc analyses revealed no differences in FLACC scores between these 194 successful cases and the full sample. FPS-R scores are reported from 5- to 12-year-old patients except two children that were incapable of self-reporting FPS-R scores; one child had autism, one had spinal muscular atrophy.
FPS-R scores ranged from 0–10 for all phases, and FPS-R baseline pain score mean was 0.59 (n = 88, SD = 1.46), post-tube placement mean score was 3.30 (n = 89, SD = 3.39), and 5-min post-procedure mean score was 1.69 (n = 89, SD = 2.43) (Table 1). There were no significant differences in males' and females' tube placement scores, p > 0.05. In addition, age was not correlated with tube placement FPS-R score, p > 0.05 (Fig. 1).
FLACC was scored for all patients except 5 due to technical issues resulting in no video recording. FLACC scores ranged from 0–10 for all phases except the pre-procedure otoscopy, which ranged from 0–8. Across all 5 procedural phases and the overall impression, the average FLACC behavioral distress score ranged from 0.1 to 4.0 (Table 2). FLACC pre-procedure otoscopy mean score for all subjects was 0.4 (n = 179, SD = 1.5), earset install and filling mean score was 0.8 (n = 187, SD = 2.2), iontophoresis mean score was 0.5 (n = 188, SD = 1.6), TM tap and tube placement mean score was 2.4 (n = 188, SD = 3.3), post-procedure mean score was 0.8 (n = 188, SD = 1.7), and overall procedure mean score was 1.3 (n = 189, SD = 2.6). Breakdown of FLACC scores for each age group is further described in Table 2. Given the overall low phase scores, statistical analyses were only conducted for the tube placement phase which exhibited the highest FLACC scores. There were no significant differences in tube placement FLACC scores between boys (n = 110, M = 2.14, SD = 3.16) and girls (n = 78, M = 2.71, SD = 3.48), p > 0.05. There was a significant correlation between FLACC and age for the tube placement phase, p < 0.01, with younger participants having higher FLACC scores than older participants (Fig. 2).
Given the differences regarding the timing of data collection for FPS-R and FLACC scores, comparison of FPS-R and FLACC scores could only be assessed for the tube placement phase. At tube placement, there was not a significant correlation between FPS-R and FLACC scores, p > 0.05, for children ages 5–12.
Discussion
Results were generally consistent with expectations. Although there were no gender differences, older children displayed lower behavioral distress. In terms of primary aims, distress was overall low as hypothesized, but there was variability in outcomes. For example, children under 12 months old had relatively higher distress, especially at the tube deployment phase. This was expected given that we were requiring that these infants stay in an unfamiliar environment (e.g., relatively cold room, bright lights), remain supine for extended periods of time, and healthcare professionals were touching and looking in the child's sensitive ears. In addition, at tube deployment there is a loud clicking sound that can be startling and frightening; this auditory event alone could explain elevated distress behavior. Anecdotal reports from the coders corroborated the impressions that distress in young patients was related to these environmental factors rather than the myringotomy and tube deployment procedure.
As expected, older children had minimal to any reported pain and displayed little distress. Specifically, the self-reported pain score means fell in the "mild" pain range based on research aiming to contextualize 0–10 pain scores (e.g., Tsze et al., 2018). Acknowledging important differences based on setting, population, and procedure, the FPS-R pain scores collected in this study do not appear to be clinically meaningfully different than mean scores reported from similarly aged patients following surgery (2.93; von Baeyer et al., 2009), receiving immunizations (range 3.0–6.6; Berberich & Landman, 2009; Boivin et al., 2008), dental injections (range 3.0–6.3; Asvanund et al., 2015; Deepak et al., 2017; Kamath, 2013), IV cannulation (3.9; Taddio et al., 2005), venipuncture (range 3.3–6.5; Inal & Kelleci, 2012; Karakaya & Gozen, 2016), and ear piercing (3.9; Hicks et al., 2001). The mean FPS-R score reported five minutes after tube placement was 1.69, suggesting that distress was transient.
Overall, the mean FLACC scores in this young sample were generally in a range that was comparable to mean FLACC scores reported for 4- to 6-year-olds receiving immunization injections (Berberich & Landman, 2009; Franck et al., 2015), 4- to 13-year-olds receiving dental injections (Asvanund et al., 2015; Pala et al., 2016; Thoppe-Dhamadharan et al., 2015), 1- to 10-year-olds undergoing venipuncture (Gupta et al., 2014; Minute et al., 2012), and 2- to 7-year-olds undergoing allergy skin prick test (Goldberg et al., 2014). A review of the literature indicates that younger children generally display higher behavioral distress than older children, consistent with the findings in this study (von Baeyer & Spagrud, 2007; Young, 2005).
The in-office tympanostomy procedure FLACC can be compared to post-operative distress after OR-placed tympanostomy tubes. Mean FLACC scores in the recovery room after OR-placed tympanostomy tubes from two separate studies ranged from 2.0 to 4.8, varying with use of pre-operative and intra-operative medications such as midazolam, dexmedetomidine, acetaminophen, fentanyl, and morphine (Hippard et al., 2012; Dewhirst et al., 2014). In a large study of 3669 children, 21% of patients in the recovery after OR-placed tubes had FLACC scores of 7–10, despite pre-operative midazolam and intra-operative fentanyl or ketorolac (Stricker et al., 2017). It is not known if this post-operative distress is due to excessive pain from the tube placement or related to the after-effects of the anesthetic (i.e., emergence agitation). In contrast, this office tube placement study using local anesthesia had low mean FLACC scores 3-min post-procedure consistent with low post-operative distress.
The lack of correlation between FPS-R and FLACC scores is notable. Data within a single study and across studies suggest that associations between FPS-R and FLACC range from no significant relationship to strong correlations (da Silva et al., 2011; Emmott et al., 2017). Consistent with the perspective that self-report and observer-report scales might be reflecting different—and valuable—aspects of the same event (Cohen et al., 2008), our findings underscore that we assessed different outcomes in line with the long-standing recommendation that pediatric procedural pain assessment be conducted in a multimethod manner (American Academy of Pediatrics Committee on Psychosocial Aspects of Child and Family Health; Task Force on Pain in Infants, Children and Adolescents; 2001; Cohen et al., 2020; McGrath & Gillespie, 2001).
It is important to note that whereas the majority of children reported and displayed minimal distress, a subset of patients reported or exhibited high distress. This distress was transitory as shown by low scores within 3-min post-procedure; peaked during tube delivery; and while discomfort may have been associated with tube insertion, anecdotally the distress often occurred when a child was encouraged to lay in a supine position, when the head was stabilized, or when the click of the device surprised the child. The broader pediatric procedural literature indicates that children exhibit higher distress when laying on their back during medical procedures (Bice & Wyatt, 2017; Cohen, 2008; Sri Rahyanti et al., 2017).
Particularly in pediatric patients, variability in pain and distress is anticipated. This is believed to be due, in part, to the fact that children have challenges in distinguishing pain from emotional aspects of distress. FLACC scores have been shown to range from 0 to 10, associated with simple palpation of a vein in children 1- to 6-year-olds (Lunoe et al., 2015), and Koc and Gozen (2015) showed that 83% of FLACC scores for infants (1 to 12 months old) were greater than 4 after simple physical measurements (height, weight, head circumference, and oxygen saturation). Tsze et al. (2018) demonstrated that 32% of children (mean age of 8.6 years old) who were not in any pain reported an FPS-R score of 2 and over 11% of children who were not in any pain reported FPS-R scores of 4, 6, 8 or 10. These reports suggest that FLACC and FPS-R evaluation cannot distinguish between pain and the emotional aspects of distress, as noted by Blount and Loiselle (2009).
We wish to highlight limitations and future directions. First, the lack of a control condition prohibits attributions of low distress to qualities of the device or behavioral program. However, given the sensitivity of the eardrum, it is challenging if not unethical to conduct tympanostomy absent anesthesia and behavioral support for pediatric patients. However, dismantling studies might help identify key intervention ingredients. Second, generalizability of findings is limited given our inclusion and exclusion criteria which restricted the patient population to children with anatomy compatible with safe use of the devices and anesthetic, and with compliant behavior for the office procedure. Future research might evaluate whether behavioral strategies might be effective for more challenging patients. Third, two of the authors were funded for work on the project, which could have introduced investigator bias. That said, all data coding was conducted by researchers who were blind to study hypotheses. Given that the behavioral program appeared to be helpful to some but not all children, we encourage researchers in this area to assess additional unique characteristics (e.g., coping styles, temperament) to advance the field in matching intervention components and strategies to individuals.
Myringotomy with tympanostomy tube placement is the most common pediatric surgery, and it is a fairly simple and brief procedure. Unfortunately, the current norm is to use general anesthesia, which invites a host of potential behavioral and other problems for young patients (e.g., Kain et al., 1999; Zhang et al., 2015). The current findings in conjunction with Lustig et al. (2020) indicate that the Tula System and behavioral program allow pediatric patients to receive in-office tympanostomy tube placement without general anesthesia, without sedation, and without mechanical restraints, and with minimal distress.