key: cord-0693362-hzx4dtdq authors: Oude Lansink, Irene L. B.; van Stam, P. C. Carolien; Schafrat, Eline C. W. M.; Mocking, Manouk; Prins, S. Dido; Beelen, Anita; Cuppen, Inge; van der Pol, W. Ludo; Gorter, Jan Willem; Ketelaar, Marjolijn title: ‘This battle, between your gut feeling and your mind. Try to find the right balance’: Parental experiences of children with spinal muscular atrophy during COVID‐19 pandemic date: 2022-04-26 journal: Child Care Health Dev DOI: 10.1111/cch.13014 sha: e2acbdaaf2bd30a4fb331a134a023add58f73dfb doc_id: 693362 cord_uid: hzx4dtdq AIMS: Parents of children with spinal muscular atrophy (SMA) often struggle with the all‐consuming nature of the demands of caring for a child with substantial physical needs. Our aim was to explore experiences, challenges and needs of parents of a child with SMA in a COVID‐19 pandemic situation. METHOD: Nineteen parents of 21 children (15 months to 13 years of age) with SMA types 1–3 participated in semi‐structured interviews in June to July 2020. The interviews were analysed using inductive thematic analysis. RESULTS: Parents mentioned the protection of the health and well‐being of the child as the central perspective and driving force during the COVID‐19 pandemic. Three subthemes were identified: (1) responsibility, (2) balancing vulnerability and resilience and (3) (in)security. Some parents focused on the positive aspects during the lockdown, such as continuation of nusinersen treatment and family life. Some parents described helpful and positive cognitions to cope with the situation. In general, parents described a need for information with regard to COVID‐19 and their child with SMA and a need for discussing their dilemmas and insecurities with a healthcare professional. INTERPRETATION: Parents put the health and well‐being of their children first during the pandemic. From this study, we learned that parents of children with SMA need information and value direct contact with a healthcare professional to share their dilemmas and insecurities. The dialogue can help to empower parents in the conflicts and decisions they have to make during a pandemic. Hereditary proximal spinal muscular atrophy (SMA) is a severe neuromuscular disease that causes progressive muscle weakness and impairment. Due to the weakness, children are in need of supportive (para)medical care, and there is a high caregiving demand from the parents. Recently, three genetic therapies have been introduced (i.e. nusinersen, risdiplam and onasemnogene abeparvovec) that improve motor function in a substantial percentage of patients. Before the introduction of genetic therapies, parents already struggled with the all-consuming demands of caring for a child with substantial physical needs and the ways in which this affects the emotional and social lives of the entire family (Chan et al., 2020; Halanski et al., 2014; Higgs et al., 2016; Qian et al., 2015; Rempel et al., 2004) . A qualitative study of parents' experiences of taking care of their children with SMA in the context of new treatments (van Kruijsbergen et al., 2021) showed that autonomy and a feeling of being in control are important factors for parents to be able to carry out their task of caregiver, especially when they are confronted with additional factors. On 27 February 2020, the first case of COVID-19 was confirmed in the Netherlands (RIVM, n.d.) . The COVID-19 pandemic resulted in a general 'lockdown' in the Netherlands in the spring and summer of 2020 with movement restrictions and social distancing of people other than your own household members. These restrictions especially affected patients with a chronic disease and their family members (Arim et al., 2020) . People had to work from home and school and day care centres were closed. Regular outpatient hospital visits were postponed, but urgent and necessary care was continued if possible. In the beginning of the COVID-19 pandemic, nothing was clear about the transmissibility of COVD-19 and the risk for children with neuromuscular diseases. SMA is characterized by respiratory muscle weakness and increased vulnerability to viral infections (Wijngaarde et al., 2020) . From the start of the COVID-19 pandemic, parents with a child with SMA were confronted with new levels of challenges and uncertainty, including whether intrathecal nusinersen treatment could be continued, extra demands in caregiving, increasing stress and need for additional decision making about going to school and therapies. Parents had to make many difficult decisions, often lacking sufficient and specific information (Arim et al., 2020; Cacioppo et al., 2020) . Since the outbreak of the COVID-19 pandemic, several articles have been published about the difficulties parents of children with chronic disabilities experienced due to the COVID-19 pandemic (Arim et al., 2020; Cacioppo et al., 2020; Darlington et al., 2021; Dhiman et al., 2020; Disabled Children's Partnership, 2020; EACD COVID-19 Surveys Initial Report, 2020; Grumi et al., 2020; Majnemer et al., 2020) . Families with children with disabilities or chronic diseases experienced difficulties due to social distancing, reduction in services (like rehabilitation), and the mental load (anxiety and depression) in combination with the lack of help/support and other restrictions to prevent the spreading of the virus. All of the above-mentioned studies were online surveys (Arim et al., 2020; Cacioppo et al., 2020; Darlington et al., 2021; Dhiman et al., 2020 ; Disabled Children's Partnership, 2020; EACD COVID-19 Surveys Initial Report, 2020; Grumi et al., 2020) , providing quantitative data. Only in one study (Darlington et al., 2021) added open text box questions qualitative data, providing some insight in the experiences of parents of children with cancer, in relation to the virus, lockdown and isolation in children with cancer. The results globally demonstrated views and experiences of this population, and the authors emphasize the importance of future studies in order to understand the ongoing experiences of families. In general, we feel it is important to get insight into the perspectives and experiences of parents, including the reasoning behind the choices parents made. The aim of this study was to explore experiences, challenges and needs of parents of a child with SMA in a COVID-19 pandemic situation. We conducted an exploratory qualitative interview study with parents of children with SMA. This study is reported in accordance with the guidelines of the Standards for Reporting Qualitative Research (SRQR) checklist (O'Brien et al., 2014) . We invited parents of a child with SMA Types 1, 2 and 3, aged <16 years to participate in this study. Treatment of SMA with genetic therapies in the Netherlands is centralized in the UMC Utrecht (UMCU). Not being able to communicate in Dutch or English was reason for exclusion from the study. We recruited parents through an invitation in the widely read newsletter of the SMA Center at the UMCU that is being sent to all families of children with SMA. Parents of children with SMA Type 1 who were in regular contact with the social worker of our team were reminded of the invitation at the end of the conversation. Parents who were willing to participate received an information letter. Two weeks after this first announcement, all parents of children with SMA Type 1, 2 or 3, <16 years who had not responded to the invitation also received an information letter. • Protecting the health and well-being of the child with SMA is the central perspective and driving force mentioned by parents, during the COVID-19 pandemic. • During the COVID-19 pandemic, parents focused on keeping balance between the risk of getting affected by COVID-19 and the mental and physical well-being of their child with SMA, siblings and family. • Parents appreciate information and conversation with healthcare professionals to reflect on the insecurities/ decision-making process of parents. Parents were contacted to schedule the interview after their consent. We ensured anonymity of participants by changing names in the transcripts before the start of the coding process. We assigned a research number to all participants and the key to the corresponding personal data could only be accessed by researchers planning and conducting the interviews (ES, MM) and by the study coordinator (CvS). The Medical Ethics Committee of the University Medical Center exempted this study from Medical Research Involving Human Subjects Act (17-904); all participants gave informed consent. We collected data by means of semi-structured interviews using a topic list with relevant issues of the COVID-19 pandemic, including the impact of the COVID-19 pandemic on well-being of the family, resilience and expectations parents have from the hospital and clinicians (see topic list in Appendix A). The content of the topic list was based on expert clinician input and parent's questions and input in the first months of the pandemic. Two trained psychological assistants (ES, MM) performed these semi-structured interviews via telephone calls between June 3 and July 30, 2020. In this time frame in the Netherlands, we faced the original COVID strain. The interviews were audio-recorded and transcribed verbatim. We collected and analysed data simultaneously in an iterative process. A thematic analysis was conducted as described by Braun and Clarke (2019) . We sent the invitation for participation to 120 families. Parents from 24 families gave permission to be contacted (20%), with a withdrawal of five parents after initial permission. We included parents of 19 families with 21 children with SMA (Table 1) . After 16 interviews, we approached code saturation, and after three additional interviews, we confirmed code saturation. Duration of the interviews varied from 40 to 90 min, with an average of 55 min. In two interviews, both parents were present. We interviewed 16 mothers without the father present and one father without the mother. Parents' and children's characteristics are summarized in Table 1 . Qualitative analysis of the interviews showed that the essence of the experiences of the respondents fell under four main themes, with one overarching theme and three subthemes. Protecting the health and well-being of the child was found as the central perspective and driving force mentioned by parents, with three subthemes (1) Min-max 1-13 Median 7 Interquartile range (IQR) 5 Male 10 Female 11 No 7 Families with two children with SMA 2 Treatment for SMA Nusinersen (Spinraza ® ) 1 7 Risdiplam 3 responsibility, (in)security and the balance of vulnerability and resilience, which in turn in the interviews often were interrelated. This battle, between your gut feeling and your mind. Try to find the right balance. This quote summarizes the predominant feeling of parents with a child with SMA during the COVID-19 pandemic. The priority of all participating parents was to protect the health and well-being of their child. In an attempt to prevent any family member getting infected with the virus, there was no other choice than to withdraw from public life during the lockdown at the peak of the pandemic for most of the families. I was so worried, that I decided that nobody could enter or leave the house, no caregivers. Our bubble reduces my worries. We completely isolated ourselves, at least the first eight weeks … we did not do anything, no therapy, no caregivers at our place, nothing: just the five of us. Parents experienced various difficulties in decision making after some of the restrictions in the Netherlands were lifted (because of reduction of COVID-19 infections in the country). … you have many things to consider, you have to overthink a lot and make a lot of decisions … Parents struggled with the necessity to keep their children inside in order to reduce the risk of a COVID-19 infection. For some children, it was difficult not to have social contacts, and parents described the struggle of their children because they missed their family members, caregivers, school and friends. Yesterday, she went back to school again for the first time, which she found amazing. She had missed her friends at school a lot, she is really keen on social contacts, wants to play with other kids and is really nice to other children. Yes … she missed that a lot. Parents struggled how to make the best decision for the wellbeing of their child, balancing between the risk of an infection and the social-emotional well-being of their child by letting them go to school and play with friends. My daughter also sees other children going outside. Yes, yes, we will manage to find the discipline to deal with the situation, because it is for the best of your child. We will do anything for our child. Parents felt great responsibility to protect the health and wellbeing of their child with SMA. Most parents wanted to continue intrathecal treatment with nusinersen for their child to ensure continued effectiveness. Most parents considered treatment for SMA more important than doing everything to prevent infection with COVID-19. Sometimes, parents described the feeling that they did not even think about having a choice; delaying treatment was not an option. Also, some parents said they were lucky to have had an appointment for nusinersen injection prior to the lockdown, so there was no need to visit the hospital again soon. … well you want your child to get the nusinersen treatment and therefore you need to go to the hospital, that's how it is. Almost all supportive treatments, such as physical therapy, hydrotherapy and occupational therapy, were cancelled because of the lockdown. Parents were afraid that not having therapy for a long time would negatively affect their child's well-being and felt responsible to take over these therapies as much as they could, mostly using video consultation with a therapist. Sometimes parents allowed therapists to come over, but with restrictions and personal protective equipment. Staying at home was still favoured in comparison to going to a physical therapist or rehabilitation centre. When therapies restarted in the Netherlands (due to limiting of COVID-19 restrictions), parents often decided to slowly resume therapies, sometimes starting in their home environment. Parents felt a challenging balance in taking responsibility to make the right decision for their child with SMA: Physical therapy is medically necessary but with additional risk of getting infected with the COVID-19 virus. So in this case the mind beats the gut feeling, because it is important to take care of the child's teeth and eyes, so the doctors' appointments had to continue. … we really tried to keep our child active and putting him in the stander every day. We did go to really important appointments, because there are also different welfare interests than the prevention of a COVID-19 infection. Also, some parents described their child with SMA being more sad than usual because they missed their peers. Parents felt responsible for the mental well-being of the child with SMA and the (mental) well-being of their siblings. I want for her to be able to participate in society, despite the fact that we from our point of view are afraid that she will be infected. Parents and siblings also missed their social contacts. Parents expressed the dilemma to balance between the best practice for their child and relaxing/social time for themselves or siblings. The interviews showed that some situations created a feeling of insecurity in parents while other situations did not. You have to make a lot more decisions, you have to think about a lot of things and make new choices, like could my child be in contact with other children? Exactly, people ask whether she is going to school again or when we want her to go back to school. I do not know! Almost all parents experienced a lack of information. During the COVID-19 pandemic, the frequency of the contacts between all parents and healthcare professionals was decreased. And when they did speak to each other, the health care professional often did not have the answers parents were yearning for. Many parents were disappointed with the scarcity and ambiguity of information they received. Parents often did not feel sufficiently in control to be able to fulfil the caregiving demands. Many parents described a feeling of insecurity to go to the hospital for an appointment. Sometimes this feeling disappeared after the hospital visit. One parent described to have been reassured after the hospital visit. That first fear of going to the hospital. Is it safe to go to the hospital, do professionals maintain the 1.5 meter social distance, is everyone protected? But everyone was really relaxed in the hospital, after seeing that, I was more relaxed myself. Parents felt more secure when it seemed that children were not severely affected, that children with neuromuscular disorders did not seem to have a higher infection rate nor being severely affected and that general infection rates decreased. Several parents decided to wait a couple of weeks (or during the summer holiday) and finally they felt safe to let their child go back to school. … when it was clearly stated in the news that COVID-19 is not really dangerous for children, I became less anxious …. We looked at the infection numbers, and technically it was going better. You should take the step, because you cannot keep them at home until December. Going to school was a physical risk, but parents were tired from all weeks at home, working, teaching and caring for their children. Children were yearning to go back to school and see their friends. For most parents, these feelings supported the decision to overcome the insecurity of letting them go to school. But I dared it anyway, although I still have doubts because I still find it scary, but yes I did it. It is important for her well-being and her 'mood', so to speak. Because sitting at home is quite difficult for them too. While protecting the health and well-being of their child with SMA was a general theme of this study, we also found variation between individual experiences. This variation, between parents and within parents over time, was most clear in the subthemes: balancing between vulnerability and resilience, balancing between feelings of security and insecurity and the feeling of being responsible for your child while risking to sacrifice their own needs. For some parents, it was a tough time, and for others, it felt like they finally found some time to slow down, normalize and experience some relaxation. During the analysis, the research group identified self-sacrifice to be an important theme. However, this was not mentioned by the parents themselves, nor did they reflect on their experiences in this way. This is an interesting interpretation of the research group, which underscribes the importance of listening to parents to understand their individual perspectives and needs and preventing assumptions from a professional's perspective. The needs of a parent can change over time, but typically are observed and compared between periods before and after a major change. The rapidly evolving pandemic forced parents to adjust to new information almost every day. As different parents have different needs at different times, it is important for healthcare professionals to tune in on parents' specific questions and (information) needs and to understand why they are important to them at that time. Obviously, parents described a need for information in regard to COVID-19 and their child with SMA. Clinicians can do well by having individual conversations with parents to reflect on their insecurities and challenges. Even if no clear answers could be given, parents often felt supported when they had the opportunity to discuss their uncertainties with a healthcare professional. It may be helpful for parents to know that they are not the only ones who are struggling and to normalize the challenges they face and the impact on their lives. This study obviously has limitations. We cannot exclude selection bias, because parents experiencing huge challenges and stress may have declined participation in this study. Although we found three fathers willing to participate (of which two couples were included), most of the participants were mothers. It The authors would like to thank all parents who contributed their time and experiences to this study and E. Veldhoen, F.L. Asselman and J. Panka for their help in starting the study, in the procedural preparations and in inviting the families. The authors declare that there is no conflict of interest. Effect of the COVID-19 pandemic on parental well-being The following questions are about the consequences of the COVID- -To what extent do you manage to combine your work with caring for your children? -Are there financial consequences for your family? -How do you assess your mood in general over the past 2 weeks? -Is your mood different than before the COVID-19 pandemic? -How do you assess your night's sleep? -What was this like before the COVID-19 pandemic? -What do you miss the most? -What is the reason that it is not possible now: COVID-19 restrictions, extra care for the children, financially, otherwise …. Effect of the COVID-19 pandemic on your child's well-being The following questions are about the consequences of the COVID-19 pandemic on the well-being of your child -How do you assess your child's mood in general over the past 2 weeks? -What was this like before the COVID-19 pandemic? -How do you assess your child's night's sleep -What was this like before the COVID-19 pandemic? -Do you notice a difference in how your child with SMA experiences the COVID-19 pandemic and how his/her brothers and/or sisters experience this? (difference in fear of infection, difference in dealing with the restrictions to prevent infection) Some parents feel that, no matter how difficult it is, they can handle the situation. Other parents have the feeling that they are completely overwhelmed. The impact of the COVID-19 pandemic on Canadian families of children with disabilities. Data to Insights for a Better Canada Thematic analysis revised Since January 2020 Elsevier has created a COVID-19 resource centre with free information in English and Mandarin on the novel coronavirus COVID-19. The COVID-19 resource centre is hosted on Elsevier Connect, the company' s public news and information Care in COVID: A Qualitative analysis of the impact of COVID-19 on the health and care of children and young people with severe physical neurodisability and their families Informal home care providers: The forgotten health-care workers during the COVID-19 pandemic Stress, resilience, and well-being in Italian children and their parents during the COVID-19 pandemic COVID-19 and children with cancer: Parents' experiences, anxieties and support needs Since January 2020 Elsevier has created a COVID-19 resource centre with free information in English and Mandarin on the novel coronavirus COVID-19. The COVID-19 resource centre is hosted on Elsevier Connect, the company's public news and information #LeftInLockdown-Parent carers' experiences of lockdown Rehabilitation services lockdown during the COVID-19 emergency: The mental health response of caregivers of children with neurodevelopmental disabilities Assessing the needs of the SMA population: Survey results of health care providers and families A short time but a lovely little short time": Bereaved parents' experiences of having a child with spinal muscular atrophy type 1 Knowledge empowers: Responding to the knowledge needs of youth with disabilities and families during the pandemic Standards for reporting qualitative research: A synthesis of recommendations Understanding the experiences and needs of individuals with spinal muscular atrophy and their parents: A qualitative study Parents' perspectives on decision making after antenatal diagnosis of congenital heart disease Parents' perspectives on nusinersen treatment for children with spinal muscular atrophy Natural history of lung function in spinal muscular atrophy