What is the state of children's participation in qualitative research on health interventions?: a scoping study.

BACKGROUND: Children are the focus of numerous health interventions throughout the world, yet the extent of children's meaningful participation in research that informs the adaptation, implementation, and evaluation of health interventions is not known. We examine the type, extent, and meaningfulness of children's participation in research in qualitative health intervention research. METHOD: A scoping study was conducted of qualitative published research with children (ages 6-11 years) carried out as part of health intervention research. Following Arksey and O'Malley's scoping study methodology and aligned with the PRISMA-ScR guidelines on the reporting of scoping reviews, the authors searched, charted, collated, and summarized the data, and used descriptive and content analysis techniques. Ovid MEDLINE was searched from 1 January 2007 to 2 July 2018 using the keywords children, health intervention, participation, and qualitative research. Study selection and data extraction were carried out by two reviewers independently. RESULTS: Of 14,799 articles screened, 114 met inclusion criteria and were included. The study identified trends in when children were engaged in research (e.g., post-implementation rather than pre-implementation), in topical (e.g., focus on lifestyle interventions to prevent adult disease) and geographical (e.g., high-income countries) focuses, and in qualitative methods used (e.g., focus group). While 78 studies demonstrated meaningful engagement of children according to our criteria, there were substantial reporting gaps and there was an emphasis on older age (rather than experience) as a marker of capability and expertise. CONCLUSIONS: Despite evidence of children's meaningful participation, topical, geographical, and methodological gaps were identified, as was the need to strengthen researchers' skills in interpreting and representing children's perspectives and experiences. Based on these findings, the authors present a summary reflective guide to support researchers toward more meaningful child participation in intervention research.

Parental understanding of newborn screening for cystic fibrosis after a negative sweat-test.

BACKGROUND: Newborn screening for cystic fibrosis (CF) in Illinois uses an immunoreactive trypsinogen/DNA methodology; most false-positive results identify unaffected carriers. METHODS: Parents whose child received a negative result from the sweat test after a positive newborn screening for CF were surveyed ≥ 6 weeks later by telephone. All parents received genetic counseling while waiting for the sweat-test results. RESULTS: A total of 90 parents participated. Overall knowledge of CF was high (78%), but the ability to understand the CF screening results was mixed. Although 94% of the parents understood that their child did not have CF, only 79% (62 of 78) of participants whose child had a mutation knew their child was definitely a carrier, and only 1 of 12 parents whose child had no mutation understood that the child may be a carrier. Respondents stated that most relatives were not interested in genetic testing. Both parents had been tested in only 13 couples. Fewer than half (36 of 77 [47%]) of the untested couples expressed interest in genetic testing. Although most participants were satisfied with the process, parents expressed frustration because of the lack of prospective newborn screening discussions by prenatal and pediatric providers and lack of knowledge and sensitivity by those who initially notified them of the abnormal newborn screening results. Speaking to a genetic counselor when scheduling the sweat test decreased anxiety for many parents (53 of 73 [73%] were "very worried" at notification versus 18 of 73 [25%] after scheduling; P < .001). CONCLUSIONS: Parental knowledge about CF is high, but confusion about the child's carrier status and the concept of residual risk persist despite genetic counseling. Relatives express low interest in carrier testing.

A pilot study to evaluate knowledge and attitudes of Illinois pediatricians toward newborn screening for sickle cell disease and cystic fibrosis.

In Illinois, newborn screening (NBS) for sickle cell disease (SCD) began in 1989 and for cystic fibrosis (CF) in 2008. We measured pediatricians' knowledge and attitudes regarding CF and SCD, the significance of carrier status, and NBS methodologies. Of 730 eligible Illinois pediatricians randomly selected from the American Academy of Pediatrics Web-based directory, 391 (54%) fully or partially completed the survey. Approximately three-fifths were women, two-thirds were Caucasians, and one-quarter had specialty training. Ninety-seven percent (377/390) and 93% (364/391) of respondents have at one point cared for a patient with SCD and CF, respectively, and virtually all support NBS for SCD (389/391, 99.5%) and CF (382/389, 98%). Overall mean knowledge of SCD (81.2%) and CF (84.5%) was high but did not correlate with self-reported familiarity. Questions regarding the interpretation of NBS results were less well understood, with 37% of our respondents unaware that Illinois NBS identifies all infants with sickle cell trait, and 28% unaware that Illinois NBS does NOT identify all infants who are CF carriers. Pediatricians support NBS but need additional education about the meaning of a positive and negative screen with respect to carrier detection to effectively counsel or appropriately refer.

Maternal attitudes about sickle cell trait identification in themselves and their infants.

INTRODUCTION: We examined knowledge and attitudes about sickle cell trait (SCT) and sickle cell disease (SCD) in women whose infants were identified with SCT through newborn screening. METHODS: Mothers of infants identified with SCT by newborn screening were contacted 8 to 52 weeks post partum. Mothers were recruited if: (1) they participated in a postpartum newborn screening survey and agreed to be recontacted if their infant had a positive newborn screening result (n = 27); (2) they revealed that their child had SCT during participation in a similar study regarding newborn screening for cystic fibrosis (n = 2); or (3) their child attended a community health center and they did not refuse to be contacted for research (n = 71). RESULTS: Ninety-five of 100 participants self-identified as black or African American. The average age was 26 +/- 6 years. Eight participants self-reported having SCD and 52 SCT, yet only 34 of the 60 (57%) had received professional hemoglobinopathy counseling. Fewer than half of respondents (45 of 100) knew their SCT status prior to their first pregnancy and less than one-fourth (23/100) reported prenatal discussions of newborn screening. Forty-one respondents were unaware that their child had SCT until notified by a study recruiter (mean age = 116 +/- 64 days). Overall mean knowledge score was high (71%; 95% CI, 69%-73%). Virtually all (> 93%) disclosed their child's SCT status to first-degree relatives and pediatricians but were more discriminating about disclosure to others. CONCLUSIONS: Women are knowledgeable about SCT despite inadequate professional education and counseling. Research is necessary to reduce gaps in reporting maternal and infant SCT results and to understand and promote informed disclosure decisions.

Reactions of pediatricians to refusals of medical treatment for minors.

PURPOSE: Treatment refusals in pediatrics must balance parental decision-making authority and best interest. General pediatricians and subspecialists were surveyed to understand the factors that influence their responses to refusals including (1) prognosis, (2) concordance of parent-minor decision, and (3) minor autonomy. METHODS: Of 1,120 eligible pediatricians, 421 (37.6%) randomly selected from the American Academy of Pediatrics Web-based Directory completed a survey about their reactions to refusals of treatment by parents, minors, or both in cancer scenarios with a 5-year expected overall survival of 80% or 15% for both an 11-year-old and a 16-year-old minor. Statistical analyses compared pediatrician willingness to respect a refusal and the relative importance of various factors to explain physician reasoning. RESULTS: Pediatricians were less likely to respect refusals when prognosis was good. Pediatricians were most likely to respect a refusal when prognosis was poor and when parent and minor concurred in their decision (93%, n = 360/385 for the 16-year-old vs. 89%, n = 345/386 for the 11-year-old, p < .05). When parent-minor dyad disagreed, pediatricians were more likely to accept a refusal by a 16-year-old minor as compared with an 11-year-old (28%, n = 111/393 vs. 4%, n = 18/405 in good prognosis, p < .001; and 65%, n = 251/384 vs. 20%, n = 79/389 in poor prognosis, p < .05). CONCLUSIONS: Pediatricians' decisions whether to respect treatment refusals for minor patients are multifactorial. When prognosis is good, best interest dominates. When prognosis is poor, parental authority is more important in younger minors, and minor autonomy is more important in older minors.

A pilot study to explore knowledge, attitudes, and beliefs about sickle cell trait and disease.

INTRODUCTION: In the United States, newborn screening programs universally identify newborns with sickle cell disease (SCD) and heterozygote carriers (sickle cell trait [SCT]). Although there is a consensus to disclose SCT to parents, there are limited empirical data about whether and how this information is transmitted to the carrier children. METHODS: In-person questionnaires were administered to parents with SCT and parents of a child with either SCD or SCT to examine the knowledge, attitudes, beliefs, and disclosure patterns about SCT of parents. RESULTS: Fifty-three adults were interviewed, half (27) of whom had a child with SCD. There was significant misunderstanding about sickle cell inheritance (mean score, 68%), but parents who have a child with SCD have better knowledge compared to those without a child with SCD (78% vs 58%, p = .002). Respondents perceive minimal stigma associated with SCT. Unless there is an affected proband, individuals with SCT rarely receive counseling or education outside of the family. CONCLUSIONS: There is significant misinformation about what it means to be a carrier and its health and reproductive implications. Formal professional counseling is rare, especially for those families without an affected proband. Strategies to increase the utilization of counseling and improve genetic literacy are necessary.

Maternal knowledge and attitudes about newborn screening for sickle cell disease and cystic fibrosis.

Illinois introduced mandatory newborn screening (NBS) for sickle cell disease (SCD) in 1989 and for cystic fibrosis (CF) in 2008. We examined maternal understanding of NBS for SCD and CF, and their knowledge of the genetics, symptoms, and treatments of both conditions. Our methods consisted of conducting interviews of inpatient post-partum women (>18 years and English speaking). Our results showed that of the 388 eligible participants, 34 self-identified as sickle cell carriers, 1 with SCD and 1 as a CF carrier. Almost 3/4 were African American (282/387). Although all but 5 women had prenatal care, only 35% (133/378) recalled their prenatal care provider mentioning NBS, and only 56% (217/388) of participants recalled nursery staff mentioning NBS. There was more self-reported familiarity with SCD (3.32/5) than CF (1.97/5, P < 0.001). Over 2/3 (260/388) of participants could not answer CF knowledge questions because they had never heard of CF. Among those who had heard of the conditions, mean knowledge scores were 66% for SCD (n = 372) and 63% for CF (n = 128). Bivariate analysis identified education, age, race, marital status, and insurance status as statistically significant. After linear regression education remained significant for both conditions. We conclude that in a sample of predominantly African American post-partum women, we found poor understanding of NBS, greater familiarity with SCD, and significant knowledge gaps for both SCD and CF. There are many missed educational opportunities for educating parents about NBS and specific conditions included in NBS panels in both the obstetric clinics and the nursery.

Ethics and professionalism in the pediatric curriculum: a survey of pediatric program directors.

OBJECTIVE: Since 1982, pediatric residency programs have been asked to evaluate trainees for ethical behavior. In 2007, the Accreditation Council for Graduate Medical Education required documenting teaching and evaluation of professionalism. Pediatric residency program directors were surveyed to ascertain what they know about the content and process of their ethics and professionalism curricula. METHODS: From February to May 2008, 394 program directors from the Association of Pediatric Program Directors were surveyed. RESULTS: Of 386 eligible survey respondents, 233 (60%) returned partial or complete surveys. Programs were evenly divided on whether ethics was taught as an organized curriculum or integrated. Professionalism was combined with the ethics curriculum in 27% of programs and taught independently in 38% of programs, but 35% had no professionalism curriculum. More than one third of the respondents did not answer each content and structure question. Approximately two thirds of those who responded stated that their program dedicated <10 hours per year to ethics and professionalism, respectively. Nearly three fourth of programs identified crowding of the curriculum and one third identified lack of faculty expertise as curricular constraints. Respondents expressed interest in more curricular materials from the American Board of Pediatrics or Association of Pediatric Program Directors. CONCLUSIONS: Despite requirements to train and evaluate residents in ethics and professionalism, there is a lack of structured curriculum, faculty expertise, and evaluation methodology. Effectiveness of training curricula and evaluation tools need to be assessed if the Accreditation Council for Graduate Medical Education requirements for competencies in these areas are to be meaningfully realized.