Rabino, Sheri Rarback MA; Peretz, Shirley Rokach BA; Kastel-Deutch, Tsophie MD; Tirosh, Emanuel MD
INTRODUCTION AND PURPOSE
Congenital muscular torticollis is the third most common pediatric diagnosis in infancy, with a prevalence as high as 16% of neonates who are normal.1 Previous reports2,3 have claimed that these infants are at risk for delayed motor milestones. Early physical therapy is usually an effective treatment, especially when initiated within the first year.4 Since parents are responsible for implementing the treatment program at home, identifying factors positively influencing parental adherence is of central importance. This study defined adherence as visits attended, exercises performed, and premature termination of services. Previous research suggests that attendance, adherence to the program, and premature termination were predicted by parental expectations and belief in psychological treatment and behavioral training for children and adolescents.5,6 Studies of parental adherence to pediatric treatments for cystic fibrosis, rheumatoid arthritis, asthma, and diabetes cited effective health care professional and patient communication and trust as central to fostering adherence, but adherence was not consistently defined.7,8 Researchers9 have suggested that patients have a preferred type of involvement in health care, that is, self-initiated autonomous behavioral involvement, including self-care, versus a passive, trusting role, receiving information and following doctor's instructions. Attitudes toward these 2 different types of involvement in treatment can be measured reliably, and preferences for 1 of the 2 types can be predicted.9 Research in the 1990s10 emphasized the importance of the patients' perspectives on health beliefs in general and their illness in particular. Social psychological theorists proposed that patients' perceptions of “disease threat,” that is, vulnerability to or actually having a disease with serious consequences, was an important causal determinant of behavior.11 The belief that by following a set of recommendations, the severity will be reduced or the disorder abolished has been suggested as a necessary prerequisite for adherence.11(p337) A meta-analysis of the relationship between adherence and the patient's perception of “disease threat” revealed an odds ratio (OR) of 2.25 (95% confidence interval [CI]: 1.38-3.60).12 Parents' beliefs in the seriousness of their children's condition or in the severity of the complications their children might suffer if they fail to adhere related positively to adherence in celiac disease and asthma.8 Physical therapy treatment of congenital torticollis has been studied, but there is no prospective study of parental adherence to an intervention program for this condition. This research differs from previous adherence studies in 2 ways. Both our population, mothers of infants, and the condition treated, a congenital, resolvable developmental condition, were different from all previous research. We chose 4 possible predictors of parental adherence on the basis of a literature review and empirical studies: (1) communication, satisfaction with, and trust in the therapist13; (2) expectations and belief in treatment14; (3) perceived “disease threat”12; and (4) preference for an active role versus a passive role in health care.9 The first 3 predictors had been studied on parents of children in treatment programs and the last on adults in treatment programs. We hypothesized that the mothers' perceptions of the severity of the condition and the role of treatment in improving their children's motor function would provide the strongest motivation to adhere to the treatment program. We further hypothesized from our clinical experience that mothers would be more highly motivated to adhere to the treatment program by the fear of the effect of the condition on their infants' function than by their relationship with the therapist, their expectations, and their belief in the treatment or preferred type of involvement in health care. The second hypothesis was supported by the meta-analysis of the relationship between adherence and “disease threat.”12
Sixty-five mothers of infants (31 females) referred by their pediatrician to a pediatric physical therapy clinic with the diagnosis of congenital torticollis, who met the inclusion criteria, were sequentially recruited into the study. The inclusion criteria were male or female, single birth, minimum of 37-weeks gestational age, and with or without plagiocephaly. Infants with sensory-motor or other chronic neurological or physiological impairment and non-Hebrew speaking mothers were excluded. Four additional families refused to participate because they thought it would be too time consuming. This article used the word adherence instead of compliance to incorporate broader notions of cooperation and partnership between therapist and client. This study was approved by Helsinki Ethics Committee of the Maccabi Health Services. The mothers signed an informed consent for the research and publication of the results.
Two experienced developmental physical therapists (PTs) participated in the study. The 2 therapists established reliability before the study. The interview process was simulated by the 2 researchers who standardized their approach and wording. A closed questionnaire was used to minimize possible responder's errors. A third researcher (E.T.), not involved in the intervention, participated in establishing interresearcher reliability in the interview process, introduction of questionnaires, and treatment protocol. The intervention program was simulated and practiced until full agreement between therapists was reached. Mothers provided demographic and health-related information.
An infant's head control was examined for asymmetry between active range of head rotation and lateral flexion of the neck to the right and to the left in the supine position, the prone position, in rolling facilitated by the PT, and when held by the parent. Resolution of the torticollis was determined by the PT when the infant's active head control was symmetrical in these same positions and activities. No standardized test was used. The infant's asymmetrical head control and significance in development was explained to the parents.
Physical Therapy Program
The physical therapy program included elongation (stretching), positioning, and handling to facilitate active neck rotation to the affected side, prevention of side flexion to the affected side, and strengthening of side flexion to the contralateral side15 (see Appendix 1, Supplemental Digital Content 1, http://links.lww.com/PPT/A44). The mothers learned that attending all the scheduled sessions and performing all the recommended exercises would result in resolution of the torticollis. They learned how to record the daily exercise logs (Appendix 2, Supplemental Digital Content 2, http://links.lww.com/PPT/A45). They were informed that the PT's role in the treatment sessions was to teach them to correctly perform the exercises at home and to discuss any problems that might arise. The program commenced when the exercises were performed as agreed upon by the 2 PTs during prestudy protocol reliability ascertainment. Treatment sessions were scheduled weekly for the first month, followed by twice-monthly sessions in the second month and once-monthly sessions in the third month and thereafter until the clinician determined treatment termination. Since no standard schedule for visits was found in the literature, this frequency was based on clinical experience and common practice in our center. The duration of the initial treatment was 1 hour and each subsequent session was 40 minutes. The parents' role was to demonstrate how they performed the exercises at home; their handling was corrected if necessary.
Dependent and Independent Variables
Adherence was determined by the following: (1) the proportion of prescribed exercises performed, that is, the number of exercises performed of the number recommended; (2) the proportion of visits attended, that is, the number of visits attended of the number of visits scheduled; and (3) termination of treatment with the resolution of the torticollis determined by the PT or premature termination. Premature termination was not based on the therapist's evaluation of an infant's status, rather, premature termination was based on the parent's decision to terminate treatment before clinician-determined resolution had occurred (yes or no).
The 4 instruments used to evaluate the independent variables were the Health Opinion Survey (HOS), the Parent Medical Interview Satisfaction Scale (PMISS), perceived “disease threat,” and the Credibility/Expectancies Questionnaire–Parent Version (CEQ-P).
The HOS is a 16-item (scored as agree = 1, disagree = 0) questionnaire in Hebrew, which measured preference for different roles in treatment, that is, an active self-care approach versus a passive trusting approach.9(p977) The HOS score was the average of 16 dichotomous items (scored 0 or 1). The validity and reliability of the HOS had been originally established on college students. Eighty college students were tested and retested over a 7-week period. The measure yielded a total score and 2 relatively independent subscales that measured preferences for information versus behavioral involvement (ie, self-care and active participation) in medical care, respectively. A test-retest reliability of 0.77 for the total score and 0.74 and 0.76 for the 2 subscores were obtained. This scale classified the mothers into 2 groups, related to their preference for the type of involvement in the treatment process. Group A preferred self-initiated autonomous behavioral involvement in treatment, including self-care. Group B preferred a more passive role, receiving information and following medical recommendations.
The PMISS13 is a 16-item questionnaire that measures parents' satisfaction with and trust in their children's medical care on the basis of communication with the therapist. The questionnaire has a high Cronbach α reliability (0.95). The word therapist was substituted for the word physician, and the questionnaire was translated into Hebrew and back into English, and the translation was reviewed by 2 bilingual PTs. A 7-point Likert scale was used. The questionnaires were answered in full, with a score of 1 to 7 for each item. A mean score of the 16 items was calculated.
Perceived “disease threat” included 2 items that specifically assessed maternal perception of the severity of the torticollis and maternal perception of treatment effect on the infant's function. These items were as follows: (1) “My child's torticollis seriously interferes with his regular activities,” and (2) “I believe that my child's condition will deteriorate if I do not carry out the home program.” The items were scored in the same manner as the PMISS, with a low of 1 and a high of 7 for each of the 2 items. The mean score of the 2 items was calculated.
The CEQ-P14 is a 6-item questionnaire composed of 2 components: (1) parental expectations of the treatment and (2) their belief in the credibility of the treatment. This scale demonstrated high internal consistency within each factor, with a standardized Cronbach α of between 0.79 and 0.90 for the expectancy factor and between 0.84 and 0.85 for the whole scale. It was translated into Hebrew and back into English, and the translation was reviewed by 2 bilingual PTs. The questionnaire consisted of 6 items, 3 assessed expectations, and 3 assessed beliefs in the credibility of the treatment. Items 1, 2, 3, and 5 were scored on a 9-point scale. Items 4 and 6, originally coded on a percentage scale, were recoded to correspond to the 1- to 9-point scale used for the other items. A mean score of the items was calculated. The correlation between the 2 subscales (r = 0.58) suggests that treatment credibility and expectancies are related but do not overlap. The PMISS and the CEQ-P were validated on parents of children with diagnoses other than torticollis. The validity of these instruments as related to torticollis had not been reported. No other instrument specifically related to parental perceptions and torticollis had been published.
Means and standard deviations were calculated for demographic factors, birth weight, gestational age, and age at initial meeting. Spearman correlation was used to analyze relationships between the 3 measures of adherence, that is, visits attended, exercises performed, and premature termination, and the scores from the HOS, the PMISS, perceived “disease threat,” and the CEQ-P. The mean scores of the latter 4 instruments were derived from the average rating of each participant's total score.
Logistic regression was performed by using the scores of the HOS, the PMISS, perceived “disease threat,” and the CEQ-P to represent the independent variables and the 3 measures of adherence as dependent variables. The dependent variables of visits attended and exercises performed were each expressed as a proportion. The third dependent variable of premature termination was binary, and therefore all 3 required logistic regression. Regression with proportions as the dependent variable is usually modeled, using logistic regression. A particular case is when the observed proportion can be either 0 or 1.16
At the initial meeting, the mean age of the children was 11 weeks and 2 days (SD = 5 weeks and 4 days). The mean birth weight was 2445 g (SD = 367 g). The mean gestational age was 39 weeks (SD = 7 days). Table 1 depicts infants' characteristics. The distribution of the dependent variables is depicted in Table 2. Seventy-eight percent of the scheduled visits were attended. Treatment was terminated with the resolution of the torticollis in 69.2% of the cases. Treatments were terminated prematurely by the mothers in 30.8% of the cases. Of the prematurely terminated group, 2 children were transferred to a child development center (10%) and 3 (15%) moved or changed therapists. Spearman correlation revealed statistically significant relationships between the proportion of clinic attendance and the proportion of exercises performed (r = 0.69, P < .0001), with fewer premature terminations (r = −0.43, P = .0003). This indicated that mothers who attended more treatment sessions also tended to perform a higher proportion of the exercises and not to terminate prematurely. The range of the data for the measures used to evaluate the independent variables is included in Table 3. Maternal perception of the seriousness of the condition and the treatment effect on future function were evaluated by the questions labeled disease threat. Disease threat predicted the dependent measures of proportion of exercises performed and proportion of visits attended, with an OR of 1.42 (95% CI: 1.113-1.843) and an OR of 1.464 (95% CI: 1.121-1.913), respectively. The logistic regression for exercises performed as the dependent variable is presented in Table 4, and visits attended as the dependent variable in Table 5. Accordingly, the odds of exercises performed increased by 42% with an increase of 1 point in the disease threat score. Similarly, the odds of visits attended increased by 46% with an increase of 1 point in the disease threat score (P = .005 for both outcomes). The outcome of premature termination was not predicted by any of the independent variables Table 6).
Treatment adherence refers to active, voluntary, collaboration to produce a therapeutic result. Treatment attendance and adherence are related but distinct constructs, since one must attend treatments to be able to adhere to the treatment regime but one can attend without adhering to the program. The choice of factors influencing parental adherence to developmental physical therapy intervention was derived from previously reported factors influencing adult adherence to treatment, parental adherence to treatment programs for their children's chronic illnesses, and parental adherence to child and adolescent psychotherapy. As we hypothesized, maternal perception of the severity of the torticollis and the effect on the infant's daily activities, measured by “disease threat,” predicted the dependent measures of proportion of exercises performed and proportion of visits attended.
Our results agreed with those reported concerning adult compliance with treatment for chronic disease, that is, that psychosocial factors such as attitudes toward disease, the personal meaning, and significance of the illness and the perceived importance of the treatment were significantly related to compliance.7(p903)
A meta-analysis of the adherence literature found a positive significant correlation between patients' adherence and their perceptions of disease severity threat (P < .001).12(p525) A meta-analysis of the relationship between parental adherence and their perceptions of “disease threat” revealed an OR of 2.25 (95% CI: 1.38-3.60).12(p524) In the present study, we found that maternal perceptions of the gravity of the condition, including their perceptions of the effect on their child's daily activities and importance of the treatment on their future function, were significantly related to 2 variables of adherence, the proportion of clinic visits attended and the proportion of exercises performed. Mothers with higher scores on disease threat had higher attendance and performed more of the prescribed exercises. These maternal perceptions were not related to the reason for termination.
Dimatteo,8(p339) in reviewing both empirical studies and theories, related adherence to communication and trust between health care providers and the parents of children and adolescent patients with chronic illnesses. In the present study, the PMISS was used to evaluate parent-therapist communication and satisfaction with and trust in the medical care. This instrument was validated on parents of children with diagnoses other than torticollis. We did not find a relationship between communication and trust in the therapist and adherence. The different results in the present study compared with Dimatteo's8 findings of adherence in chronic illness may possibly be attributed to the characteristic of torticollis, a congenital nonchronic developmental condition. Kazdin et al,17 studying parental adherence in the context of child and adolescent psychotherapy, reported that barriers to family's adherence—poor relationship with the therapist and perception that treatment is not relevant or too demanding—were associated with premature termination. Our findings do not support their findings. Perhaps the difference is a result of the different populations studied. Our study supports the findings of Tirosh et al,18 who reported a statistically significant relationship between low attendance and premature termination of treatment. Our study evaluated maternal expectations and belief in the treatment program, using the CEQ-P. This instrument was validated on parents of children with diagnoses other than torticollis. Unlike our study, 2 previous studies5(p155),19 reported statistically significant relationships between parental expectations and belief in treatment and attendance and premature termination. Nock et al19(p27) evaluated adherence only in the initial 8 treatment sessions. It is possible that parents' expectations, beliefs, and adherence and the relations among these constructs change later in treatment.19 In their respective studies, Nock et al5,19 reported similar findings. Both of these studies found that parental adherence correlated significantly with expectations and belief in child psychotherapy treatment. Nock et al19 used the CEQ-P as we did. We have no reason to believe that the psychometric properties of this instrument would be different in torticollis. The variable of the mothers' preferred role in health care procedures was evaluated using the HOS.9 The HOS was validated on adults, not parents. This is the first time to our knowledge that this instrument has been used for parents; therefore, no data related to validity are available. This instrument was used to classify the mothers into 2 groups dependent on their preference for a type of involvement in the treatment process: (a) autonomous behavioral involvement, including self-care, or (b) a passive role of following instructions. Krantz et al9 proposed that when the client's preference for a certain role in the health care procedure is synchronous with the treatment style of the therapist, the best outcomes are reached. The rational for using this instrument was to discover whether adherence was related to the mothers' preferred role in the treatment program. No relationship was found between the mothers' preferences and their adherence with the program. The logistic regression analysis using the results of the 4 instruments that evaluated the 4 independent variables—communication with and trust in the therapist, parental expectations and belief in the program, preference for active versus passive type of involvement in medical care, and “disease threat”—resulted in only 1 strongly positive and significant variable, “disease threat.” This is in line with our second hypothesis. Once “disease threat” was introduced as a predictive factor, all the other independent variables related to adherence became insignificant. Therefore, the effect of maternal perceptions of the possible effect the condition might have on their infants' future function appears to be significantly more dominant than any other perceptions evaluated in the present study. Since this article relates specifically to congenital torticollis, a resolvable impairment requiring developmental intervention, conclusions cannot be generalized to chronic neurodevelopment disorders. Our findings imply that “disease threat” appears to be the predictive variable for parental adherence. The 2 statements representing “disease threat” can be included in the parental interview. On the basis of the parents' responses (agree or disagree) to both statements, future adherence can be predicted.
In this study, the possibility of a type 2 error should be considered. Unlike previous findings suggesting the importance of communication with and trust in the therapist, expectations and belief in the program, and preference for active versus passive role in medical treatment, this was not the finding in this study.
The goal of this study was to prospectively identify and describe the significant factors influencing maternal adherence to a treatment program for congenital torticollis. The only variable found to be strongly positive and significant in predicting adherence as measured by clinic attendance and the proportion of home exercises performed was “disease threat.” “Disease threat” measured maternal perceptions of the severity of the torticollis and the importance of the treatment in improving future function. The factors potentially affecting adherence, derived from studies related to chronic pediatric medical problems and adolescents in psychotherapy, appear to have no relevance in the case of an intervention program involving mothers and their infants with a treatable congenital condition. Our results can be generalized only to transient developmental disorders. It appears that adherence to physical therapy for this condition in early infancy depends on the maternal perceptions of the severity and the importance of carrying out the treatment to resolve the impairment. Future studies addressing these questions in infants and children with chronic neurodevelopment impairments would be highly contributory.
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