Design
The study was a randomized controlled trial (RCT) with two groups of parents of children with SCD (Figure 5). One group was randomly assigned to the PEIP group, and the other was the control (SEP) group. The Parent Educational Intervention Program (PEIP)consisted of two video clips; the first video addressed the physical aspects -- basic information about SCD, signs and symptoms, complications, potential triggering factors for sickle cell crisis, and the management of SCD (7 minutes). The second video (6 minutes) addressed 1) emotional aspects -- fear, anxiety, worry, sleep disruptions; 2) social -- relationship with peers, siblings, ability to enjoy leisurely activities; and 3) cognitive aspects -- the child’s ability to do well in school, ability to communicate, solve problems, make decisions, resolve conflicts. The PEIP was delivered via smartphone with reinforcement phone conversation facilitated by the PI once per week, over a 4-week period. Measurement of outcomes (parents’ knowledge, parents’ self-efficacy, and parent’s perceptions of HRQOL as well as the children’s perception of their HRQOL were done at baseline, and at the end of the intervention, which was 4 weeks after enrollment.
Setting
The parents (either father or mother) of children with SCD were recruited from the Royal Hospital (RH) and Sultan Qaboos University Hospital (SQUH), both tertiary hospitals in the Sultanate of Oman. All parents of children with SCD who were followed in the hematology clinics in these hospitals were invited to participate. Approximately, 300 children with SCD were seen annually in the hematology clinics at both hospitals.
Sample
A convenience sampling approach was used to recruit parents of children with SCD. First, parents were eligible if the age of their child with SCD is between 8 to 12 years. In Oman, children until 12 years of age were seen in the pediatric clinics, and children ages of 13 and older were seen in the adult clinics. The rationale for selecting this age group was because school age children were able to report pain, and other symptoms related to the disease. Also, the information on the emotional, social, and cognitive health were more important for children in this age group. An assumption of the study was that parents, as the child’s primary caregiver, would be able to apply the knowledge as they provide care for their child.
Second, the parents were eligible if they were Omani citizens because the PEIP was specifically designed so that it was culturally appropriate for Omanis. Therefore, parents from other nationalities, who did not speak Arabic and who came from different cultures were excluded. Parents were also excluded if they: 1) were not available for consenting procedures; 2) were not willing to participate in completing the study procedures; and 3) had physical, cognitive, and/or neurological impairments that prevented them from consenting and/or completing study procedures. The nurse screened the eligible participants for any neurological impairment using the Decision-Making Capacity Assessment tool. This was done to assess the ability of parents who may have or may be experiencing cognitive impairments to make an informed decision about being part of the study. In the event that the Omani parents were not able to read, a nurse not involved in the study procedures would read the study materials.
The PEIP was delivered via a smartphone. Parents who did not have smartphones were included, and were provided with a smartphone for study use. A total of nine parents were excluded from the study as four of them were not Omanis and the other five were not willing to participate in completing the study procedures.
Sample Size determination
The total number of parents that were enrolled were 74 (37 in each group). Using G-power, the sample size of 37 in each group was able to detect a 0.71 effect size increase in knowledge51 0.59 increase in parent’s self-efficacy 23 and 0.5 increase in HRQOL 41 compared to control groups.
A trained nurse coordinator worked with the hematology team at the Royal Hospital identify the eligible parents, screened the prospective participants provided information about the study for eligible participants, and created the list of children’s names and their parents, who were interested; the list was provided to the principal investigator (PI) for more detailed explanation of the study and consenting procedures. In the event that a parent was not able to read, the nurse coordinator explained in terms the parent is able to understand about the study.
The PI met parents assigned to the intervention group in a private room in the clinic for 15 minutes to explain the details of the study procedure to the parents and their children using the study information sheet. They were allowed to ask questions, and talk with family members and care providers as needed. Parents signed the consent, and the child signed the assent form prior to enrollment and study procedures.
Randomization Procedures: To minimize the risk of contamination, a cluster randomization approach was used, with enrollment occurring on two separate days for the intervention (PEIP) and the control groups (SEP). To minimize potential temporal bias, a coin flipping was used every week to decide on the days for the cluster placement to the PEIP or the SEP group. Participants enrolled during the first day were assigned to the PEIP group and those attending the clinic on the subsequent day were assigned to SEP; this procedure was continued alternatingly.
Data Collection Procedures. All parents, regardless of group assignment were asked to complete the following pre-and post-intervention assessments using paper and pen at time of enrollment: 1) Parents Knowledge Questionnaire (PKQ) (10 minutes), 2) PedsQL (Generic) (5 minutes), 3), PedsQL SCD module (10 minutes), 4) Self-Efficacy Scale (SES) (5 minutes), 5) Demographics Questionnaire (2 minutes). The children were asked to complete the following age appropriate PedsQL: 1) PedsQL (Generic) (5 minutes), 2) PedsQL SCD module (5 minutes). Completion of the assessments took approximately 30 minutes at the beginning and at the end of the intervention period.
Parental Knowledge Questionnaire (PKQ). The SCD Parental Knowledge Questionnaire (PKQ) has 25 items; 16 items were True/False statementsand 9 items were multiple-choice (A, B, C, D) type questions. The items measured the knowledge about SCD, signs and symptoms, complications, treatment and triggering factors (16 items). The other nine items were psychological (4 items), social (3 items), and the cognitive problems (2 items) (derived from NHLBI, 2014). . Each item on the PKQ was scored as 1 to yield a maximum total score of 25 points; , range 0 to 25, with higher scores indicating higher knowledge. The English to Arabic translation was available, and the Arabic back to English translation procedure as well as the content validation were done by 3 experts (2 hematologists, one nurse in Oman) was done prior to implementation. The readability index for PKQ is appropriate for the 7th grade level.. The Arabic PKQ was then piloted on 5 parents who were excluded from the study. The internal consistency reliability using Cronbach alpha was (0.80) prior to commencement of the study and (0.87) 4 weeks post intervention. .
Pediatric Quality of Life Inventory (PedsQL-GENERIC). The Pediatric Quality of Life Inventory (PedsQL-GENERIC) developed by Varni and colleagues was a generic core scale that had 23 items ara designed to measure four dimensions of HRQOL: 1) physical functioning (8 items), 2) emotional functioning (5 items), 3) social functioning (5 items) and 4) school functioning (5 items). The parent proxy report format and the corresponding child version for 8-12 years was used. 56 The English to Arabic and Arabic back to English translation, as well as the content validation procedures done by 3 experts (2 hematologists, one nurse in Oman) prior to implementation. Both parent and child versions of the PEdsQL-GENERIC was then piloted on 5 parents and children who were excluded from the study. The internal consistency reliability using Cronbach alpha was (0.8) prior the commencement of the study and (0.85) 4 weeks post intervention. The internal consistency reliability of the Arabic version is (0.80).
Scoring & Interpretation of PedsQL-GENERIC. The items on the PedsQL-GENERIC were scored from 0-4 scale, and rated as 0 is “never a problem”; 1 is “almost never a problem”; 2 is “sometimes a problem”, 3 is “often a problem”; and 4 is “almost always a problem”. Following the instructions for scoring, the 0 to 4 scores were reversely converted to the 0 to 100 scores for standardized interpretation, so that 0 was scored as 100; 1 was scored as 75; 2 was scored as 50; 3 was scored as 25 and 4 was scored as 0. The items were averaged so that the total scores ranged from 0 to 100 and the t the Physical and Psychosocial Summary Scores were computed. Higher scores indicated higher HRQOL. 56
Pediatric Quality of Life Inventory SCD (PedsQL-SCD)
The HRQOL-SCD module had 43 items, and nine scales: Pain & hurts (9 items), pain impact (10 items), pain management & control (2 items), worry I (5 items), worry II (2 items), emotion (2 items), treatment (7 items), communication I (3 items), and communication II (3 items). 48 Similar to the parent version, it has a 5-point Likert type response scale . The English to Arabic and Arabic back to English translation, as well as validation procedures of both parent and child versions were done by 3 experts (2 hematologists, one nurse in Oman) prior to implementation. Both parent and child versions of the PedsQL-SCD were then piloted on 5 parents and children who were excluded from the study. The parent proxy report format and the child version for 8-12 years was used. Both parent and child versions of the PedsQL-SCD was then piloted on 5 parents and children who were excluded from the study. The internal consistency reliability showed Cronbach’s coefficient alpha=0.70 for children, which was considered acceptable. Internal consistency reliability coefficients for the PedsQL-SCD Module showed reliable with Cronbach’s alpha (0.93) for children with SCD. The PedsQL was piloted, and the internal consistency reliability was (0.85) prior conducting the study and (0.89) 4 weeks post intervention. Also, test-retest reliability, and estimating interclass correlation (ICC) for the tool was done. The period between the test-retest was 2 days. Two days was found appropriate because children with SCD were prone for any complication at any time; therefore, waiting for a longer period to conduct the retest may result in scores variation. The internal consistency reliability was (0.9).
Scoring & Interpretation of PedsQL. Similar to the HRQOL-GENERIC, the items on the HRQOL-SCD Module was scored from 0-4 scale, 0 was “never a problem”; 2 was “almost never a problem”; 3 was “often a problem”; and 4 was ”almost always a problem”, and were converted to the 0 to 100 scores for standardized interpretation as described above. The items were averaged and the Physical and Psychosocial Summary Score were computed as described above (Panepinto et al., 2013).
Parent SCD Self-Efficacy
The self-efficacy scale for parents (SES) was adopted and modified from self-efficacy instrument specific to sickle cell disease (SCSES) that was developed by Edwards and colleagues for use in adults with sickle cell disease. The original questionnaire has 9 items that measured disease specific perception of self-efficacy regarding the patients’ ability to function on a day-to-day basis and to manage their child’s symptoms and pain related to SCD. 24 The items in the parent self-efficacy tool remained the same. However, wording was changed so the items reflect that it was the parent reporting (rather that child reporting) of their ability to manage the symptoms, and pain of their children on a day-to day basis. The questionnaire was administered at the time of enrollment and 4 weeks post intervention. The internal consistency reliability showed a Cronbach’s alpha of (r=0.89). In the study, experts assessed the modified tool for content; construct validity before and after translation to Arabic languae. In addition, the SES was piloted on five participants who were excluded from the study, and the internal consistency reliability indicated the Cronbach’s α =0.9 prior conducting the study, and α=0.954 weeks post intervention.
Scoring & Interpretation. Response choices for each item on the SES were 0=”Not at all sure”, 1=”Not sure”, 2= “Neither“, 3= “Sure” , or 4= “Very sure”. The total scores were obtained by summing responses for all nine items; the range of scores were 0 to 36, with higher scores indicating greater self-efficacy.
Demographics & Medical Information Sheet
Parent demographics, which included age, gender, educational level, socio-economic status, and marital status were collected by the nurse coordinator in the haematology clinic at the time of enrolment. In addition, demographics and medical information related to the children (age, gender, use of hydroxyurea, SCD diagnosis, history of SCD-related complications, including number of pain crisis requiring hospitalization the previous year) were also collected.
Procedures for Delivery of Parent Educational Intervention Program (PEIP). Following the pre-intervention assessments, parents assigned to the PEIP group were oriented by the PI and requested to navigate the educational materials on their smartphones. The orientation session was 20 minutes. The two video educational materials were downloaded to the parents’ own smartphone. If parents did not have one, parents were provided with one; however, all parents had smartphones and were not required to get another one. Downloading of the two videos into the smartphone took approximately 2-3 minutes depending on the network. The parents were allowed to practice accessing the materials and opening the videos.
After the orientation session, the PI informed the parents to watch both videos on their free time when they reached home and that the PI would contact them by a phone call once per week (every Wednesday), focusing on one section per week (physical, emotional, social, cognitive health) for four consecutive weeks, to remind parents to watch the videos, read the materials, and refer to the material about pain control. Parents were encouraged to refer to videos whenever needed.
Call duration was approximately 10- 12 minutes per participant. Most of the time, the parents picked up the phone call from the first time; however, some parents were approached after the second call. In the first 2 minutes of the call, the parents were asked to brief about the section they saw that week. This was done to ensure that parents have actually seen the assigned section and understood the information provided for them. Generally, all parents were able to provide information about the section they saw.
For the physical aspect, parents were asked about pain management for their child “what was done or what would be done” to minimize the child’s pain and they were instructed to refer to the video for pain management. Also, they were asked to list the precautions they have taken to prevent future pain for their children, and they were referred to watch the video for VOC triggering factors. If the child was on hydroxyurea, the parents were encouraged to give it on time and as prescribed. For the emotional aspect, after 2 minutes briefing about the section, the parents were asked if they have noticed any emotional changes in their children and how they responded to them. The PI requested the parents to refer to the information on the role parents on improving the emotional health for their children. Similar to the emotional aspect, for the social aspect, the parents were asked about the social problems or changes observed in their children and how they responded to them. Parents were reminded to refer to the material on the parents’ role in enhancing the child’s social health. For the cognitive aspect, parents were asked about the child’s current school performance, any cognitive issues, and what was done to solve those issues. The PI emphasized on the importance to collaborate with the school for better cognitive outcomes and refer to the material on the cognitive health.
In addition, all participants’ questions and concerns were addressed during the call. Any problem experienced by the parents in regards to the video were also addressed. Almost all the parents watched the videos 4-5 times throughout the intervention. A unique feature of the PEIP was the ability to allow parents the opportunity to openly talk about the child’s health that were not routinely part of clinic visits, and reinforced different aspects of the HRQOL-- physical, emotional, social, cognitive health respectively during the four weeks.
Procedures for Delivery of Standard Education Program (SEP).
To avoid contamination, the parents in the SEP were oriented separately in the Pediatric hematology clinic. The parents assigned to the SEP met with the PI for an orientation session at a private room in the hematology clinic. The PI explained the details of the study. Parents were allowed to ask questions, and talk with family members and care providers as needed. They were given time to think about participation as needed. The participants were informed that care would continue regardless of whether they participate in the study or not. They were also informed that the information would be kept confidential so that no one would know the individual responses to the questionnaires. Moreover, the parents were informed that the participation was voluntary and they had the right to withdraw anytime from the study. Parents received a $30 “Lulu” a shopping voucher for their participation at the end of the study. Parents willing to participate were then asked to sign the consent form when they were ready. When consenting process finished, the parents were instructed to complete the study questionnaires, using paper and pen.
The control group received the standard education program (SEP) that was given by the staff nurses in the clinic. The SEP consisted of verbal information about the follow up appointments. For the study, a booklet containing questions and answers related to typical questions and answers that parents receive about SCD were distributed to the control group. Examples of the questions were: “What diet should sickle cell patients follow? “What is the effect of cold temperature on people with SCD?” “How does warm temperature affect people with SCD?” “ Why should I take the vitamin folate?” There were no videos accessible by smartphone, and no follow-up phone calls about their understanding of health education or allowing them to talk about the physical, emotional, social, and cognitive aspects of the child’s health. To minimize the risk of contamination through information sharing between parents in the PEIP and the SEP groups, the researcher coordinated with the registration department and the assigned physicians to schedule separate dates for the parents in the PEIP and the SEP group for the subsequent appointments during the 4-week period of participation in the study.