“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: