“he had his hearing tested twice before lockdown before the March lockdown and then all the appointments got postponed. So, what they said is that he’s got a glue ear and he needs to be monitored closely but has not had any tests ever since” (mother 15)
Discussion
To the best of our knowledge, this is the first study exploring parental experiences of caring for pre-school children with PCD. It provides valuable insights into parental adjustment and adaptation to the diagnosis and management of their child’s PCD. Diagnosis of this rare and persistent condition was a disruptive life event. Adjustment to a new normality was a gradual process, facilitated by getting a diagnosis and an explanation for the child’s symptoms. Some families lived in countries where PCD health care was coordinated through a multidisciplinary team, whilst other families received fragmented care and were personally responsible for coordinating appointments with different specialties. As the COVID-19 pandemic started part way through the study, the information collected also covers PCD care during this unpredictable stressful life event.
As there is no cure for PCD, the health status of the child is dependent on PCD management. Parents found the additional effort to keep their young child healthy and happy was not only physically but also emotionally taxing. Although parents recognized the benefits of a PCD treatment regimen, similar to the reports of parents with other chronic conditions (Hawkins et al, 2020), the commitment provided challenges to the fulfilment of their social, vocational, and parenting roles. Parents were not able to attend social activities to a level previously enjoyed. The parent had to be more cautious in their interaction with others and PCD caring instructions prevented participation in social activities.
Consistent with the experiences of parents caring for children with other lifetime chronic conditions (Hatzmann et al, 2014) the parent’s vocational role and functioning was often affected. As reported by other studies (DeRigne & Porterfield, 2017) it was usually the mother who experienced changes in work status. In this study the parents often reported a need for more flexible, less demanding vocational roles. Families were often financially affected by combined reduction in working hours, as well as efforts to save for future healthcare procedures (e.g. fertility treatments).
Most parents felt a constant worry for their child’s wellbeing. About 50 percent of the parents reported recurrent feelings of anxiety and depression and 20 percent of the parents disclosed formal mental health diagnosis or treatment. Although it seems that a lot of parents responded emotionally to the caregiving responsibility, the assumed prevalence of mental health problems from this informal source of reporting is similar to the existing lifetime mental health prevalence in the general population (Schaefer et al., 2017). More formal psychiatric epidemiological testing among caregivers of individuals living with PCD would be appropriate to determine a more accurate prevalence of mental health problems among this population.
Limitations
Although we recruited an international, diverse sample of parents caring for young children diagnosed with PCD, this group of parents might not be representative of the population of parents caring for young PCD children as 11 of the 24 participants were recruited from Southampton Children’s Hospital UK, more mothers than fathers participated, and most participants had a white ethnic background while the prevalence of PCD is higher in other ethnic groups (O’Callaghan et al, 2010). In addition, due to the partially retrospective aspect of the project, we cannot exclude the possibility of recall bias when the parents commented on historic events.
Practical implications
The findings of this study reveal that lack of awareness of PCD by clinicians sometimes limits disease management as it impedes GP care, emergency care, and pharmaceutical care. Due to the rarity of PCD the symptom pattern is not always recognized early in life, thereby delaying diagnosis. Over the past decade, diagnostic awareness campaigns have been able to lower the age of diagnosis of this rare hereditary chronic condition in some countries. It would be prudent to extend these awareness campaigns and improve the knowledge of health care providers about PCD and other rare diseases.
The impact of PCD care on the parent can be overwhelming at times. It induces time constraints and requires not only physical but also emotional efforts. The mental health and resilience of caregivers is important to the quality of care provided. Parents of children living with PCD are often unable to get a regular break from their caring responsibilities. Caregiver burden might be eased and more personalized care provided if PCD health care was integrated with social care, coordinating health care services with support for tasks of daily living and engagement with local community (Stein, 2001). Currently PCD services are geared towards providing appropriate services to cover the child’s PCD medical needs, but some parents reported a need for respite care; support with practical implementation of PCD management at home; caregiver appreciation; financial advice and support; awareness of community resources. A more holistic PCD care is responsive to meet not only the health care needs but also cover the social care needs of the family living with PCD.
Conclusion
This is the first international study’ examining the experiences of parents whose young child has been diagnosed with PCD. The findings from this study can be used to advise clinical practice in the holistic management of the child’s care. To end with these words from one of the parents: