MANUSCRIPT CITATION 

Dorner RA, Boss RD, Burton VJ, Raja K, Lemmon ME. Parent preferences for neurodevelopmental screening in the neonatal intensive care unit. Dev Med Child Neurol 2020; 62:500-505. PMID 31909496

REVIEWED BY

Amanda Kwong, Dr
Victorian Infant Brain Studies (VIBeS), Murdoch Children’s Research Institute, VIC, Australia
Newborn Research, Royal Women’s Hospital
Department of Physiotherapy, University of Melbourne

Abbey Eeles, Dr
Victorian Infant Brain Studies (VIBeS), Murdoch Children’s Research Institute, VIC, Australia
Department of Paediatrics, Monash University
Newborn Research, Royal Women’s Hospital
Department of Physiotherapy, University of Melbourne

Alicia Spittle, Professor
Department of Physiotherapy, University of Melbourne
Victorian Infant Brain Studies (VIBeS), Murdoch Children’s Research Institute, VIC, Australia
Newborn Research, Royal Women’s Hospital

TYPE OF INVESTIGATION 

Diagnosis, qualitative study

QUESTION

What are the views of parents of preterm infants born ≤ 34 weeks’ gestation with regards to early screening for early neurodevelopmental screening during their time within the Neonatal Intensive Care Unit, and how do they want to receive information about neurodevelopmental screening?

METHODS

• Design: Exploratory, qualitative study
• Allocation: Not applicable
• Blinding: Not applicable
• Follow-up period: Not applicable
• Setting: USA; urban level IV NICU and associated level III NICU
• Patients/Participants: Parents of preterm infants born ≤ 34 weeks’ GA.  At time of data collection, infants needed to be between 28 and 34 weeks’ of age.
• Intervention: Early neurodevelopmental screening using a hypothetical ”non-invasive, non-painful” test
• Outcomes:  Idenfication of common themes and views of parents of infants born preterm about their preferences for early neurodevelopmental screening for risk of disability, including cerebral palsy.
• Analysis and Sample Size: Directed content analysis approach was used by three authors to analyse transcribed interviews. The interviews were coded independently and then collated to create themes and categories. A broader research team covering areas of neurology, neonatology, neurodevelopmental disabilities were consulted to resolve discrepancies. The frequency of key themes were noted from the interviews. Nineteen interviews were conducted (15 mothers, four fathers) for 15 infants by which point thematic saturation was achieved.

MAIN RESULTS: 

The median interview length: 25 minutes (interquartile range [IQR] 18–34min), maximum interview time, 68 minutes.

Median infant gestational age at birth: 29.6 weeks (IQR 25.7–31.2wks).

Median corrected gestational age at the time of interview of 32.2 weeks’ gestational age (IQR 30.8–34.2wks). One interview was conducted before 28 weeks’ gestational age at the request of the child’s parents.

Four themes and subsequent sub-themes were identified that represented parent perceptions of early screening for neurodevelopmental problems:

1. Acceptability (parent perspective on theoretical NICU screening)
   • Perceived benefits of early neurodevelopmental screening
     • Parents placed value on having additional information and thought that early identification of risk of neurodevelopmental disability would allow time to emotionally adjust, engage in early intervention, and allow the family to adjust to any additional needs of their child, particularly in the face of severe disability.
   • Perceived negatives of early neurodevelopmental screening
     • Parents noted that anxiety could be associated with neurodevelopmental screening in addition to others stressors within the NICU, particularly if the test produced a false-positive result. Neurodevelopmental screening focused on the negative possibilities of the child.
   • Parent autonomy versus child’s interests
     • Most parents (14/16) supported other parents’ rights to refuse neurodevelopmental screening. However, parents saw the benefit in the ability to engage in early intervention because of neurodevelopmental screening. As such, two parents felt that refusing screening was unethical.
2. Disclosure of results
   • Approach to testing and disclosing results
     • Parents wanted to be advised of potential harm within the neurodevelopmental screening process and the accuracy of testing. Emotional preparation for the results was important for parents, as was receiving results from experts within the setting of NICU to allow the opportunity to ask questions.
   • Impact of false-positive results
     • While parents acknowledged emotional burden between the time of positive screening result and definitive diagnosis, the ability to commence early intervention outweighed stress associated with a positive neurodevelopmental screening result.
3. Emotional burden of uncertainty
   • Taking things day by day
     • Parents coped with emotional burden by taking things “day by day” and focused on daily concerns such as respiratory or feeding issues, rather than future development.
   • Looking for information
     • Parents were uncertain about future neurodevelopmental prognosis and had minimal information about their child’s future. However, the broad spectrum of outcomes made expectations difficult, and parents changed between wanting to know more and feeling overwhelmed. Other parents sought information from other sources such as nurses, physical therapists, friends or Facebook.
4. Disability – parent experience and expectations for their infant’s future
   • Prior experience
     • Parents who had work exposure to disability or family members with disability helped them conceptualise the broad spectrum of disability, whereas those who did not have prior experience held misconceptions about cerebral palsy.
   • Value placed on outcome
     • Parents held low importance for their child’s neurodevelopmental outcome, and felt that they would adapt their parenting to the child’s needs regardless of their outcome. Parents placed higher value on early screening for conditions that also held high concern based on their own personal experiences with disability, for example cognitive, future independence, blindness and quality of life.

CONCLUSION

The authors conclude that parents are interested in early neurodevelopmental screening and are shaped by their previous experiences with disability. It was important for parents to have autonomy and access to pretest counselling and emotional support for the discussion of results.

COMMENTARY 

Infants born preterm are at higher risk of neurodevelopmental problems, including cerebral palsy (CP) compared with infants born at term. Early screening for neurodevelopmental conditions such as CP is crucial to commence early intervention, which can improve motor and cognitive outcomes, and reduce parents stress and anxiety.(1,2) The diagnostic category of ‘high-risk of CP’ is now operational,(3) with evidence suggesting that triangulating brain neuroimaging, General Movements Assessment and Hammersmith Infant Neurological Examination results may reduce the age of diagnosis for CP amongst high-risk infants.(4) The possibility of earlier diagnoses of CP is encouraged by families with children with established CP.(5) However, clinicians should acknowledge that parent mental health can be at-risk in the Neonatal Intensive Care Unit (NICU) and support should be provided accordingly;(6) this extends to early neurodevelopmental screening with a family-centered approach. Dorner et al.’s qualitative study provides insight from interviews with 19 parents within a USA urban Level III and Level IV NICUs about their perceptions and preferences for early neurodevelopmental screening.

Dorner et al. found that parents were mostly accepting of early neurodevelopmental screening, but required the right supports (e.g. emotional preparation, medical expert consultation, social work presence) and information to be able to make an informed decision and cope with the potential state of their child’s neurodevelopmental status. While most parents supported early neurodevelopmental screening for themselves, parents felt that other parents should have the right to refuse screening. Generally, parents felt that potential long term gains from accessing early intervention as a result of early neurodevelopmental screening outweighed initial anxiety and stress during the screening process. Counselling support with respect to existing individual parent/family knowledge and experiences of disability significantly shaped participants expectations and concept of future disability.

This study interviewed a sample of parents who were well-resourced, had high levels of education, were fluent in English, and only one infant in the sample had a major brain injury. While the study’s transferability is limited, the findings provide important insight into the individual needs of parents of infants within the NICU who require neurodevelopmental screening. The nature of the questions avoided the specifics involved with the screening process, only identifying it as a hypothetical “non-invasive, non-painful test”. Identifying the specific process of screening to participants may have altered participant responses. Readers are encouraged to consult the interview questions (supplementary files) to gain the context of the responses analysed within Dorner et al.’s study. Parent experiences of early neurodevelopmental screening in different geographical locations, cultural groups, sociodemographic status, infant risk groups, as well as other time points within and beyond the journey through NICU are needed to extend Dorner et al.’s findings.

Dorner et al.’s study highlights the sensitive nature of early neurodevelopmental screening and the depth of information and counselling required to individualise and adequately support and empower parents through the screening process. Finally, the study also emphasises the need to understand parents’ existing knowledge of and experience with disability and acknowledge parents’ day-to-day changes in emotions within the NICU setting.

REFERENCES:

1. Spittle AJ, Orton J, Anderson PJ, Boyd R, Doyle LW. Early developmental intervention programmes provided post hospital discharge to prevent motor and cognitive impairment in preterm infants. Cochrane Database of Systematic Reviews. 2015(11).
2. Morgan C, Darrah J, Gordon AM, Harbourne R, Spittle A, Johnson R, et al. Effectiveness of motor interventions in infants with cerebral palsy: a systematic review. Dev Med Child Neurol. 2016;58(9):900-9.
3. Novak I, Morgan C, Adde L, Blackman J, Boyd RN, Brunstrom-Hernandez J, et al. Early, Accurate Diagnosis and Early Intervention in Cerebral Palsy: Advances in Diagnosis and Treatment. JAMA Pediatr. 2017;171(9):897-907.
4. Maitre NL, Burton VJ, Duncan AF, Iyer S, Ostrander B, Winter S, et al. Network Implementation of Guideline for Early Detection Decreases Age at Cerebral Palsy Diagnosis. Pediatrics. 2020;145:5-15.
5. Guttmann K, Flibotte J, DeMauro SB. Parental Perspectives on Diagnosis and Prognosis of Neonatal Intensive Care Unit Graduates with Cerebral Palsy. J Pediatr. 2018;203:156-62.
6. Treyvaud K, Spittle A, Anderson PJ, O’Brien K. A multilayered approach is needed in the NICU to support parents after the preterm birth of their infant. Early Hum Dev. 2019;139:104838.