Communication about Children's Clinical Trials as Observed and Experienced: Qualitative Study of Parents and Practitioners

Background

Recruiting children to clinical trials is perceived to be challenging. To identify ways to optimise recruitment and its conduct, we compared how parents and practitioners described their experiences of recruitment to clinical trials.

Methods and Findings

This qualitative study ran alongside four children''s clinical trials in 11 UK research sites. It compared analyses of semi-structured interviews with analyses of audio-recordings of practitioner-family dialogue during trial recruitment discussions. Parents from 59 families were interviewed; 41 had participated in audio-recorded recruitment discussions. 31 practitioners were interviewed. Parents said little in the recruitment discussions contributing a median 16% of the total dialogue and asking a median of one question. Despite this, parents reported a positive experience of the trial approach describing a sense of comfort and safety. Even if they declined or if the discussion took place at a difficult time, parents understood the need to approach them and spoke of the value of research. Some parents viewed participation as an ‘exciting’ opportunity. By contrast, practitioners often worried that approaching families about research burdened families. Some practitioners implied that recruiting to clinical trials was something which they found aversive. Many were also concerned about the amount of information they had to provide and believed this overwhelmed families. Whilst some practitioners thought the trial information leaflets were of little use to families, parents reported that they used and valued the leaflets. However, both parties agreed that the leaflets were too long and wanted them to be more reader-friendly.

Conclusions

Parents were more positive about being approached to enter their child into a clinical trial than practitioners anticipated. The concerns of some practitioners, that parents would be overburdened, were unfounded. Educating practitioners about how families perceive clinical trials and providing them with ‘moral’ support in approaching families may benefit paediatric research and, ultimately, patients.     

Qualitative interview study of communication between parents and children about maternal breast cancer

Objective To examine parents'' communication with their children about the diagnosis and initial treatment of breast cancer in the mother. Design Qualitative interview study within a cross-sectional cohort. Setting Two breast cancer treatment centers. Participants 32 women with stage I or stage II breast cancer with 56 school-aged children. Main outcome measures Semistructured interview regarding timing and extent of communication with children about the diagnosis and initial treatment of the mother''s illness, reasons for talking to children or withholding information, and help available and requested from health professionals. Results Women were most likely to begin talking to their children after their diagnosis had been confirmed by biopsy, but a few waited until after surgery or said nothing at all. Family discussion did not necessarily include mention of cancer. There was considerable consistency in the reasons given for either discussing or not discussing the diagnosis. The most common reason for not communicating was to avoid children''s questions, particularly those about death. Although most women had helpful discussion with a physician concerning their illness, few were offered help with talking to their children; many would have liked help, particularly the opportunity for both parents to talk to a health professional with experience in understanding and talking to children. Conclusion Parents diagnosed with cancer or other serious illnesses should be offered help to think about whether, what, and how to tell their children and about what children can understand, especially as they may well be struggling themselves to come to terms with their illness.     

Qualitative interview study of communication between parents and children about maternal breast cancer

ObjectiveTo examine parents'' communication with their children about the diagnosis and initial treatment of breast cancer in the mother.DesignQualitative interview study within cross sectional cohort.SettingTwo breast cancer treatment centres.Participants32 women with stage I or stage II breast cancer with a total of 56 school aged children.ResultsWomen were most likely to begin talking to their children after their diagnosis had been confirmed by biopsy, but a minority waited until after surgery or said nothing at all. Family discussion did not necessarily include mention of cancer. There was considerable consistency in the reasons given for either discussing or not discussing the diagnosis. The most common reason for not communicating was avoidance of children''s questions and particularly those about death. While most mothers experienced helpful discussion with a doctor concerning their illness, few were offered help with talking to children; many would have liked help, particularly the opportunity for both parents to talk to a health professional with experience in understanding and talking to children.ConclusionsParents diagnosed with cancer or other serious illnesses should be offered help to think about whether, what, and how to tell their children and about what children can understand, especially as they may well be struggling themselves to come to terms with their illness.     

Notifying patients exposed to blood products associated with Creutzfeldt-Jakob disease: theoretical risk for real people

BACKGROUND: In July 1995 the Canadian Red Cross Society recalled blood products because of the hypothetical risk of transmission of Creutzfeldt-Jakob disease (CJD) through those blood products. The authors undertook a survey to determine the views of patients and parents of patients about being notified that they or their child had received such blood products. METHODS: The study population consisted of 528 transfusion recipients, of whom 453 (85.8%) were under 16 years of age, notified by the Hospital for Sick Children, Toronto, of the CJD recalls in 1995 and 1996. Families attending an information session were asked to complete a self-administered questionnaire (85 cases). Ninety-seven families randomly selected from those who did not attend the session were interviewed by telephone. The questionnaire was adapted from a questionnaire used to evaluate families'' responses to notification of transfusion and risk of HIV infection. RESULTS: More than 80% of the respondents said they wanted to be notified and would want to be notified if there were another recall. On initial receipt of the notification about two-thirds of the respondents had been anxious, fearful or angry. There was no one method of conveying the information that suited all, but a personalized letter was seen as the most acceptable method. INTERPRETATION: Most parents of children who have received blood products are in favour of being informed about the risk of CJD, despite the uncertainty of the information on risk and the anxiety that such information causes.     

‘THEY SAY ISLAM HAS A SOLUTION FOR EVERYTHING,SO WHY ARE THERE NO GUIDELINES FOR THIS?’ ETHICAL DILEMMAS ASSOCIATED WITH THE BIRTHS AND DEATHS OF INFANTS WITH FATAL ABNORMALITIES FROM A SMALL SAMPLE OF PAKISTANI MUSLIM COUPLES IN BRITAIN

This paper presents ethical dilemmas concerning the termination of pregnancy, the management of childbirth, and the withdrawal of life-support from infants in special care, for a small sample of British Pakistani Muslim parents of babies diagnosed with fatal abnormalities. Case studies illustrating these dilemmas are taken from a qualitative study of 66 families of Pakistani origin referred to a genetics clinic in Southern England. The paper shows how parents negotiated between the authoritative knowledge of their doctors, religious experts, and senior family members in response to the ethical dilemmas they faced. There was little knowledge or open discussion of the view that Islam permits the termination of pregnancy for serious or fatal abnormality within 120 days and there was considerable disquiet over the idea of ending a pregnancy. For some parents, whether their newborn baby would draw breath was a main worry, with implications for the baby's Muslim identity and for the recognition of loss the parents would receive from family and community. This concern sometimes conflicted with doctors' concerns to minimize risk to future pregnancies by not performing a Caesarean delivery if a baby is sure to die. The paper also identifies parents' concerns and feelings of wrong-doing regarding the withdrawal of artificial life-support from infants with multiple abnormalities. The conclusion considers some of the implications of these observations for the counselling and support of Muslim parents following the pre- or neo-natal diagnosis of fatal abnormalities in their children.     

Expectation versus Reality: The Impact of Utility on Emotional Outcomes after Returning Individualized Genetic Research Results in Pediatric Rare Disease Research,a Qualitative Interview Study

PurposeMuch information on parental perspectives on the return of individual research results (IRR) in pediatric genomic research is based on hypothetical rather than actual IRR. Our aim was to understand how the expected utility to parents who received IRR on their child from a genetic research study compared to the actual utility of the IRR received.MethodsWe conducted individual telephone interviews with parents who received IRR on their child through participation in the Manton Center for Orphan Disease Research Gene Discovery Core (GDC) at Boston Children’s Hospital (BCH).ResultsFive themes emerged around the utility that parents expected and actually received from IRR: predictability, management, family planning, finding answers, and helping science and/or families. Parents expressing negative or mixed emotions after IRR return were those who did not receive the utility they expected from the IRR. Conversely, parents who expressed positive emotions were those who received as much or greater utility than expected.ConclusionsDiscrepancies between expected and actual utility of IRR affect the experiences of parents and families enrolled in genetic research studies. An informed consent process that fosters realistic expectations between researchers and participants may help to minimize any negative impact on parents and families.     

Carrier testing of children for two X-linked diseases: a retrospective evaluation of experience and satisfaction of subjects and their mothers

Carrier testing of children for inherited disease that will not affect the health of the children themselves but of their future children is generally regarded as problematic. In this retrospective study, we determined how young women had experienced genetic carrier testing when they were children. The families of 66 young females who had been tested for carriership during childhood between 1984 and 1988, were approached. Of them, 23 young females in families affected by Duchenne muscular dystrophy, and 23 young females in families affected by hemophilia A, and their mothers, participated in our study. We used a questionnaire including multiple-choice and open-ended questions. We recorded general attitudes to testing, satisfaction with testing, degree of trust in test results, making decisions regarding testing, privacy, and opinions about age at testing. Thirty-five out of 46 of the young women tested (76%) were satisfied with carrier testing in childhood. However, the young women in whom the test results had been uncertain were statistically more often unsatisfied with the testing than those who had been found or not found to be carriers (p = 0.002). In each group, the opinions of mothers were parallel to those of their daughters. Seventy-eight percent of daughters regarded carrier testing as a family matter in which parents can make a decision. About half of those tested recalled that they had been allowed to participate in decision-making in a satisfying way. Thirty-nine out of 46 (85%) of the young women tested, and 33/46 (72%) of the mothers, suggested that carrier testing should be performed in childhood or during teenage years.     

Prenatal hemoglobinopathy screening. IV. Follow-up of women at risk for a child with a clinically significant hemoglobinopathy.

To determine the benefits and burdens of prenatal hemoglobinopathy carrier identification and genetic counseling and its impact on subsequent reproductive behavior, we recontacted women whom we had previously identified as at risk for having a child with a clinically significant hemoglobinopathy, regardless of whether they had accepted the offer of prenatal diagnosis. Of the 46 such women, 31 were available for interview. These 31 women had received offers of prenatal diagnosis in 47 pregnancies. Seventeen had been accepted, and 30 had been declined. The proportion of patients accepting the offer of prenatal diagnosis was higher for the index pregnancy (50%) than for subsequent pregnancies (22%). The mean interval between the initial counseling of the patient and the follow-up interview was 43 mo (standard error +/- 2.7 mo). Ninety-four percent of those interviewed recalled having received information from the screening program; 74% recalled the name of their condition; 90% knew that trait did not affect their health; 84% recalled the name of the condition for which their fetus had been at risk; and 77% could state at least one symptom of the disease. Of the 29 women asked whether they intended to use prenatal diagnosis in future pregnancies, 13 said yes and 16 said no. Of the 26 patients asked about satisfaction with their previous decision about prenatal diagnosis, all were satisfied with their decision. Eighteen said they would make the same decision in their next pregnancy, but seven patients said they would not, and one was undecided.(ABSTRACT TRUNCATED AT 250 WORDS)     

Androgen insensitivity syndrome (AIS): emotional reactions of parents and adult patients to the clinical diagnosis of AIS and its confirmation by androgen receptor gene mutation analysis

The emotional reactions of parents and adult patients on disclosure of the clinical diagnosis of androgen insensitivity syndrome (AIS) and its later confirmation by gene mutation analysis were assessed. A semistructured interview and three questionnaires were used. Parents came from 18 different families with a total of 20 children (15 complete AIS, 5 partial AIS), 19 raised as girls, 1 as a boy. Ten adult women with complete AIS came from six families. The short-term reaction upon the clinical diagnosis was in the majority of both parents and adult patients associated with shock, grief, anger, and shame and in the mothers and adult patients with guilt. Emotional reactions were more long-lasting in mothers and adult patients than in fathers. The confirmation by DNA analysis did not alter the actual feelings of both parents. Adolescents with AIS should be informed completely - but in a step-by-step way - about their condition, since adult patients indicated that they had suffered from being not at all or misinformed about AIS in their adolescence.     

Early detection of visual defects in infancy

To determine the part played by screening in detecting visual defects questionnaires were sent to 240 families with blind or partially sighted children identified from the Family Fund''s database. Questions were asked on social and family background, the visual disorder and its severity, any other disability, and how and when the disabilities were discovered and subsequently managed. Data from 189 families were analysed, constituting all those with children with major visual defects from the 219 families who replied. The visual defect was first discovered in 111 children by parents, friends, and neighbours, and in 36 by a doctor at the neonatal examination. In only three children who did not have a family history of visual impairment was the defect discovered during a formal screening examination at a child health clinic. Dissatisfaction about medical services was expressed by about a third of the parents, particularly a lack of provision of information and consideration of their worries and a failure to refer the child promptly to educational and treatment services.Visual defects in children under 5 are generally detected by family and friends, not by screening, but detection by the medical profession could be improved by increased awareness and observation and quick referral.     

Fostering caring relationships: Suggestions to rethink liberal perspectives on the ethics of newborn screening

Newborn screening (NBS) involves the collection of blood from the heel of a newborn baby and testing it for a list of rare and inheritable disorders. New biochemical screening technologies led to expansions of NBS programs in the first decade of the 21st century. It is expected that they will in time be replaced by genetic sequencing technologies. These developments have raised a lot of ethical debate. We reviewed the ethical literature on NBS, analyzed the issues and values that emerged, and paid particular interest to the type of impacts authors think NBS should have on the lives of children and their families. Our review shows that most authors keep their ethical reflection confined to policy decisions, about for instance (a) the purpose of the program, and (b) its voluntary or mandatory nature. While some authors show appreciation of how NBS information empowers parents to care for their (diseased) children, most authors consider these aspects to be ‘private’ and leave their evaluation up to parents themselves. While this division of moral labor fits with the liberal conviction to leave individuals free to decide how they want to live their private lives, it also silences the ethical debate about these issues. Given the present and future capacity of NBS to offer an abundance of health‐related information, we argue that there is good reason to develop a more substantive perspective to whether and how NBS can contribute to parents’ good care for children.     

Patients’ Perceptions of Information and Education for Renal Replacement Therapy: An Independent Survey by the European Kidney Patients' Federation on Information and Support on Renal Replacement Therapy

Background

Selection of an appropriate renal replacement modality is of utmost importance for patients with end stage renal disease. Previous studies showed provision of information to and free modality choice by patients to be suboptimal. Therefore, the European Kidney Patients’ Federation (CEAPIR) explored European patients’ perceptions regarding information, education and involvement on the modality selection process.

Methods

CEAPIR developed a survey, which was disseminated by the national kidney patient organisations in Europe.

Results

In total, 3867 patients from 36 countries completed the survey. Respondents were either on in-centre haemodialysis (53%) or had a functioning graft (38%) at the time of survey. The majority (78%) evaluated the general information about kidney disease and treatment as helpful, but 39% did not recall being told about alternative treatment options than their current one. Respondents were more often satisfied with information provided on in-centre haemodialysis (90%) and transplantation (87%) than with information provided on peritoneal dialysis (79%) or home haemodialysis (61%), and were more satisfied with information from health care professionals vs other sources such as social media. Most (75%) felt they had been involved in treatment selection, 29% perceived they had no free choice. Involvement in modality selection was associated with enhanced satisfaction with treatment (OR 3.13; 95% CI 2.72–3.60). Many respondents (64%) could not remember receiving education on how to manage their kidney disease in daily life. Perceptions on information seem to differ between countries.

Conclusions

Kidney patients reported to be overall satisfied with the information they received on their disease and treatment, although information seemed mostly to have been focused on one modality. Patients involved in modality selection were more satisfied with their treatment. However, in the perception of the patients, the freedom to choose an alternative modality showed room for improvement.     

Frequency of somatic and germ-line mosaicism in retinoblastoma: implications for genetic counseling.

Although mosaicism can have important implications for genetic counseling of families with hereditary disorders, information regarding the incidence of mosaicism is available for only a few genetic diseases. Here we describe an evaluation of 156 families with retinoblastoma; the initial oncogenic mutation in the retinoblastoma gene had been identified in these families. In 15 ( approximately 10%) families, we were able to document mosaicism for the initial mutation in the retinoblastoma gene, either in the proband or in one of the proband's parents. The true incidence of mosaicism in this group of 156 families is probably higher than our findings indicate; in some additional families beyond the 15 we identified, mosaicism was likely but could not be proven, because somatic or germ-line DNA from key family members was unavailable. Germ-line DNA from two mosaic fathers was analyzed: in one of these, the mutation was detected in both sperm and leukocyte DNA; in the other, the mutation was detected only in sperm DNA. Our data suggest that mosaicism is more common than is generally appreciated, especially in disorders such as retinoblastoma, in which a high proportion of cases represent new mutations. The possibility of mosaicism should always be considered during the genetic counseling of newly identified families with retinoblastoma. As demonstrated here, genetic tests of germ-line DNA can provide valuable information that is not available through analysis of somatic (leukocyte) DNA.     

Couples facing death. II--Unsatisfactory communication.

Forty-one couples facing the prospect of separation by death were interviewed about how the nature and prognosis of their illness had been discussed with them by their general practitioner hospital staff, and the staff of the continuing care unit to which they were admitted. Communication between husband and wife and how they coped with telling their parents and their children was also assessed. Treatment was given when they were anxious or dissatisfied about the quality of communication in any of these areas. The couples found hospital doctors least successful at communication. Almost all who were dissatisfied wanted more information, not less. The wish to protect dependent relatives conflicted with the wish to be open, making decisions very difficult. Considerable suffering is caused by poor communication, and much of this is avoidable.     

“Those Comments Last Forever”: Parents and Grandparents of Preschoolers Recount How They Became Aware of Their Own Body Weights as Children

Background

Parents'' and grandparents'' willingness to talk about children''s body weights may be influenced by their own childhood experiences of body weight awareness and ‘weight talk’ in the family; however, little is known about how adults describe their recollected weight-related childhood experiences.

Aims

This paper examines how parents and grandparents of preschoolers describe the emergence of their own body weight awareness in childhood or adolescence. The analysis highlights the sources that participants identify as having instigated their body weight awareness, the feelings and experiences participants associate with the experience of becoming aware of their body weights, and their framings of potential links between childhood experiences and attitudes and practices in adulthood.

Methods

49 participants (22 parents, 27 grandparents, 70% women, 60% with overweight/obesity) from sixteen low-income families of children aged 3–5 years (50% girls, 56% with overweight/obesity) in the Pacific Northwest were interviewed. The interviews were videotaped, transcribed, and analyzed qualitatively.

Results

Twenty-five participants (51%) said they became aware of their body weights in childhood or adolescence. Fourteen participants said their body weight awareness emerged through comments made by others, with the majority citing parents or peers. No participant described the emergence of body weight awareness in positive terms. Four participants directly linked their own negative experiences to the decision not to discuss body weight with their preschoolers. All four cited critical comments from their parents as instigating their own body weight awareness in childhood.

Conclusions

In most cases, participants associated their emergent awareness of body weight with overtly negative feelings or consequences; some participants said these negative experiences continued to affect them as adults. Since family-based childhood obesity interventions involve open discussion of children''s body sizes, the results suggest that clinicians should reframe the discussion to deconstruct obesity stigma and emphasize inclusive, affirmative, and health-focused messages.     

Further experiments on resting site selection by the typical and melanic forms of the moth, Allophyes oxyacanthae (Caradrinidae)

The melanic and typical morphs from 13 families of the moth, Allophyes oxyacanthae (Linnaeus) were tested for evidence of resting site selection in a large box lined with oak bark of three reflectances. For 12 of the families, one or both of the parents originated from two families, 0/1 and 0/3, which had been tested in a similar manner in the previous year. In all of these 12 families typicals showed a preference for the palest background available, "light grey". The melanic results for the same families were significantly heterogeneous but could be partitioned on the basis of parentage, into six families in which melanics preferred light grey, and six families in which melanics preferred the background on which they are most cryptic, dark grey. In the only family in which neither of the parents originated from either 0/1 or 0/3, both typical and melanics showed a preference for the background on which typicals are most cryptic, bare bark. The results of the two years' experiments indicate that it is necessary to postulate that, in addition to the pleiotropic effects of alleles at the melanic locus, another locus (or loci) is involved in the control of resting site selection.     

Children in wheelchairs.

Three hundred and seventy-five families replied to a questionnaire about the use of their disabled children''s wheelchairs. Many problems were found, such as difficulty in folding the chair and placing it in the car boot and difficulty in using pulbic transport. These families need adequate guidance to anticipate and deal with the problems. Provision of a wheelchair does not solve the mobility problems. The parents are faced with the task of transporting both the disabled child and the wheelchair whenever they want to use their own car or public transport. Ease of folding and lightness are important criteria for wheelchairs carried by cars. Parents need careful guidance about selecting suitable cars and also need to be shown how to lift the chairs without endangering their backs. Such advice should be provided by every wheelchair clinic and assessment and rehabilitation certre.     

Sex roles in nest keeping – how information asymmetry contributes to parent‐offspring co‐adaptation

Parental food provisioning and offspring begging influence each other reciprocally. This makes both traits agents and targets of selection, which may ultimately lead to co‐adaptation. The latter may reflect co‐adapted parent and offspring genotypes or could be due to maternal effects. Maternal effects are in turn likely to facilitate in particular mother‐offspring co‐adaptation, further emphasized by the possibility that mothers are sometimes found to be more responsive to offspring need. However, parents may not only differ in their sensitivity, but often play different roles in postnatal care. This potentially impinges on the access to information about offspring need. We here manipulated the information on offspring need as perceived by parents by playing back begging calls at a constant frequency in the nest‐box of blue tits (Cyanistes caeruleus). We measured the parental response in provisioning to our treatment, paying particular attention to sex differences in parental roles and whether such differences alter the perception of the intensity of our manipulation. This enabled us to investigate whether an information asymmetry about offspring need exists between parents and how such an asymmetry relates to co‐adaptation between parental provisioning and offspring begging. Our results show that parents indeed differed in the frequency how often they perceived the playback due to the fact that females spent more time with their offspring in the nest box. Correcting for the effective exposure of an adult to the playback, the parental response in provisioning covaried more strongly (positive) with offspring begging intensity, independent of the parental sex, indicating coadaptation on the phenotypic level. Females were not more sensitive to experimentally increased offspring need than males, but they were exposed to more broadcasted begging calls. Therefore, sex differences in access to information about offspring need, due to different parental roles, have the potential to impinge on family conflicts and their resolution.     

Loss in higher multiple pregnancy and multifetal pregnancy reduction.

From early pregnancy into childhood, higher multiples have much higher rates of mortality, whether from spontaneous abortion, the 'vanishing twin' syndrome, fetal or infant death. Many parents must cope with the death of one baby whilst the siblings remain critically ill or later become disabled and yet there grief is often underestimated. Little is known about the long term feelings of parents who choose to have a multifetal pregnancy reduction (MFPR). Most say they made the right decision but also that there was insufficient respect for their loss. They are often anxious about what, if anything, to tell the survivors and how they might react. Long term follow-up studies of the children as well as the parents are needed. Meanwhile parents who chose to have a MFPR must be given more information and ongoing support.