Caregiving is a committed role that focuses on providing the required support and care to a care receiver who is either ill, disabled, or dependent to the extent that they are incapable of independent functioning. The topic of caregiving has been extensively studied worldwide, mainly focused on the negative aspects of caregiving, like caregiver stress, burden, role conflicts, and burnout among caregivers. However, limited efforts have been made to understand the positive aspects of caregiving among informal caregivers who spend most of their time in this unpaid role. The present study addresses this concern by exploring the positive aspects of caregiving among family caregivers of older persons in India.This is a qualitative study, which was conducted, using the existential-phenomenological approach. In this study, a purposive sampling technique was used, and a total of a 100 family caregivers participated from four cities in India, namely Prayagraj, Pune, Visakhapatnam, and Guwahati. Twenty-five family caregivers between the age of 30–50  years participated from each of these four cities. The data was collected using six semi-structured interview questions on positive aspects of caregiving based on the lived experiences of caregivers. The interview schedule was developed based on the PERMA model and finalized after the pilot study. Each interview took 30–50 min and was recorded and transcribed.The data was analyzed using thematic analysis. Some commonalities and differences were observed in the responses given by family caregivers from the four cities. Four major themes that emerged for the “Nature of positive caregiving” among participants from all four cities are “Caregiver’s attitude,” “Care and compassion,” “Roles and responsibilities,” and “Beliefs and values.” Four significant themes for the “Factors contributing to positive caregiving” are “Meaningfulness in life,” “Sense of belongingness,” “Personal growth,” and “Empathetic understanding.”Based on the study’s findings, it can be said that the themes were associated with the PERMA model. Positive caregiving is an important indicator of caregivers’ and care receivers’ well-being. The findings have implications for planning some action research, training, and counseling programs for promoting positive aspects of caregiving among informal caregivers. India is the second most older adults populated country globally, with 103.8 million senior citizens . The neeInformal caregiving is a dynamic, evolving, and intersubjective life experience in which connections are co-created consciously or unconsciously and manifested in acts and behaviors of care, responsibility, and attention . CaregivLooking into the literature, it is observed that many researchers have focused on the negative side of caregiving and have highlighted factors such as burden, stress, clinical depression, cognitive impairment, physical health problems, and burnout commonly observed in formal and informal caregivers . HoweverPerceptions of caregiving and the factors influencing caregivers differ across cultures . A studyIn the Indian context, caregiving is usually provided by family members. Additionally, extended family, friends, and neighbors feel responsible for caring for older people . HoweverSome of the studies also point out that individual factors also determine the caregiver’s attitude, along with cultural factors. A systematic review  carried The literature shows that the researchers’ focus over the last decade has shifted to looking into caregiving outcomes by considering different internal and external factors. Irrespective of the type of care, i.e., providing care to the person with or without physical or psychological or multiple impairments , caregivers of older adults experience moderate to higher levels of burden due to factors like characteristics of the care-receivers and their illness, socioeconomic status, caregivers’ physical health, psychopathological symptoms, and disruption of the family routine , 24, 25.Studies have also pointed out that caregivers of patients with psychiatric and chronic medical illnesses risk experiencing burnout, dysphoria, emotional distress, stress, anxiety, and depression, even though the burnout levels are moderate , 25–28. From the above discussion, it can be inferred that individual differences have an essential role in the caregiving outcome, and caregiving generally depends on how well the caregivers adjust to the demands of caregiving. The government and policymakers must consider these cultural and individual differences while framing any policies and schemes. The government must support and facilitate the informal caregivers by teaching them problem-focused coping strategies, providing home health visit services, and identifying different types of needs  so that these facilities mitigate the possible adverse effects associated with caregiving , 14, 29.Although most studies have highlighted the problematic side of caregiving, some researchers have also investigated the positive outcomes associated with caregiving. When the caregivers have a good relationship with the care-receiver, they choose to provide care out of their own will, allocate some time for self-care, vent out emotions occasionally, and are not working out of the home; they have been shown to exhibit satisfaction in their caregiving , 16. HavFrom the above studies, it is evident that a limited number of studies have been done to explore the positive aspects of caregiving. Given this gap, the researchers must explore the flourishing aspects of caregiving and come to a consensus on the domain of factors associated with it so that they can be nurtured and act as preventative factors against any potential negative consequences of caregiving. Given the need, the current study aims to understand the nature of positive caregiving and explore the factors associated with such experiences among informal caregivers using the PERMA model.PERMA model is a multi-dimensional approach to defining what it means to flourish in life. It was developed by Martin Seligman  to definAccording to the PERMA model, Positive emotions (P) comprise hope, interest, joy, love, compassion, pride, amusement, and gratitude. They are essential indicators of flourishing and well-being . StudiesIn the PERMA model, Meaning (M) is associated with having a purpose in life and is guided by personal values. Studies have highlighted that having a purpose in participants’ lived experiences reduces health problems . It has To date, limited studies have been conducted by researchers who looked at the caregiving of the older adults using the PERMA model. Also, the essence of flourishing in caregivers can be understood well qualitatively based on their lived experiences; however, only a few studies have focused on it in this way. The current study incorporated the PERMA Model as it is an evidence-based approach giving a scientific base to exploring flourishing among caregivers based on their positive caregiving lived experiences. Using the PERMA model, the present study aims to understand the nature of positive caregiving and explore the factors contributing to positive caregiving experiences among family caregivers of older adults in India based on the existential-phenomenological approach.2.This study is a qualitative study that was conducted using the existential-phenomenological approach. The approach views people as being in a constant process of interpretation as they journey from one situation to another. Each situation has meaning only through people’s interpretation and definition of it. According to this approach, one must understand a person’s experience from their lifeworld. It is the world as lived by the person every day in direct and immediate experience . In the 2.1.In this study, a purposive sampling technique was used, and a total of 100 family caregivers from four cities in India, namely Prayagraj, Pune, Visakhapatnam, and Guwahati participated in this study.These cities were selected as they have a sizeable population of older adults and because they have an increased number of informal caregivers due to the limited availability of paid caregiving services. Prayagraj from Uttar Pradesh represented North India, Visakhapatnam from Andhra Pradesh represented South India, Pune from Maharashtra is in Western India, and Guwahati is in the Eastern part of India. Twenty-five middle-income family caregivers aged 30–50 years, participated from each of these four cities. From Prayagraj, 19 female caregivers and 6 male caregivers; from Visakhapatnam, 21 female caregivers, and 4 male caregivers; from Pune, 16 female caregivers, and 9 male caregivers; and from Guwahati, 18 female caregivers and 7 male caregivers participated in this study. Across the four cities, the female caregivers were mostly daughters-in-law and daughters, and the male caregivers were sons taking care of their older adults parents.2.2.Discuss how your role of caregiving has impacted your life.Share some of your positive emotional experiences associated with your role as a caregiver.How engaged and involved do you find yourself in your caregiver’s role?What type of relationship do you share with your care receiver?How and in what ways has your role as a caregiver defined the meaning of your life?What type of satisfaction did you get in this role of caregiver? Share some of your accomplishments.The data was collected using the interview technique. Six semi-structured interview questions on positive aspects of caregiving based on participants’ lived experiences were asked from each participant. The interview schedule was developed based on the PERMA model. The six questions were focused on exploring positive emotions, engagement, relationships, meaning, and accomplishment of caregivers in their caregiving roles. They are as follows:The interview schedule was tested and finalized after the pilot study, which was done in all four cities.The data collection was done in four phases. Each phase took a month. In phase one, data was collected in Prayagraj; in phase two, data was collected in Visakhapatnam; in phase three, data was collected in Pune; in phase four, data was collected in Guwahati. Each interview in the final study took 30–50 min and was recorded and transcribed. Interviews were primarily conducted in Hindi and English in all four cities. In Visakhapatnam, some interviews were conducted in Telugu, and in Guwahati, some were conducted in Assamese.To maintain this study’s reliability, before data collection, the researchers practiced bracketing by first writing their preconceptions about family caregiving. This was done to go for data collection with an open mind for accepting participants’ experiences without any preconceived notions. In practice, bracketing is not so easy and difficult to follow; however, the researchers tried to become aware of their own ideas regarding informal caregiving and not to let those ideas interfere in data collection and analysis.To establish the validity in the present research study, the following strategies were applied:Prolonged engagement in the field: the researcher’s prolonged engagement in the field is an important strategy for establishing validity. According to experts , during Theoretical sampling: theoretical sampling refers to the process of selecting informants who represent the community, not statistically, but theoretically, in the sense of having more knowledge about the community for which the study is designed or any specific aspect of the community life . This stExtensive quotations: extensive quotations from field notes and transcripts of the interviews are also an essential way of establishing credibility in qualitative research . This stMember checking: member checking has been described as the most crucial technique for establishing credibility . It consMultiple researchers checking the data: it involves more than one researcher reviewing the data and its interpretation . This isAfter the checking of transcripts by researchers and participants the transcribed responses were treated as final data for the analysis.2.3.The data was analyzed using thematic analysis.Step 1: familiarizing oneself with the data—this was the first step of data analysis. In this step, the researchers transcribed all 25 interviews collected in the case of each city. After that, all 25 transcriptions were read and re-read by the researchers one by one, specifically for each of the six semi-structured questions asked in case of each interview. Based on the readings of each transcription, some ideas related to each question were identified.Step 2: generating initial codes—in this step, based on the ideas noted in the earlier step, codes were generated. Codes identify a feature of the data that appears significant to the analyst and refer to the distinctive features of the raw data that can be analyzed in a meaningful way regarding the phenomenon . The codSeparate coding for each of the six questions in each participant’s case from four cities was done. Though coding was done based on the questions asked, if any participant gave any response which was not directly related to the question but the participant confirmed it to be the answer for the same question, then it was also noted because being a phenomenological study any response from the participant was considered significant.Step 3: searching for themes—in this step, two separate lists of codes, one for the nature of positive caregiving and another one for factors related to positive caregiving, were prepared, which were extracted under the respective six questions in the earlier step from each of the 25 interviews. After this, based on those codes, themes were identified for nature and factors related to positive caregiving.Step 4: reviewing themes—in this step, the themes were refined, which were prepared in the earlier step. During this review phase, the researchers combined some sub-themes and made them into one theme.Step 5: defining and naming themes—in this step based on the ‘essence’ of what each theme is about and what aspect of the data each theme captures, all the themes generated were defined and named.Step 6: producing the report—after the final themes were named and defined, a write-up containing the description of each theme (with responses) was prepared, which is a part of this study’s result and discussion chapter.The thematic analysis of the responses of participants from each of the four cities was done separately but simultaneously. In the case of each city’s data, thematic analysis was done flexibly following six steps stated by Braun and Clarke . These sThough these steps are mentioned separately, in practice, the researchers often worked simultaneously on reading transcripts, preparing codes, making themes based on codes, and reviewing themes. The same procedure was followed for the data in all four cities.3.Based on the thematic analysis of the responses given by the 100 family caregivers in this study, four major themes that emerged for the “Nature of positive caregiving” are “Caregiver’s attitude,” “Care and compassion,” “Roles and responsibilities,” and “Beliefs and values.”3.1.3.1.1.Caregiver’s attitude was the dominant theme among participants from all four cities. The respondents emphasized that since caregiving has brought meaningfulness to their life, they want to give their best in this role by serving the care receivers with the utmost respect. Most of the participants from Pune and Guwahati mentioned that taking care of older adults family members is like expressing one’s gratitude for what they did for these caregivers when they were young. Like a caregiver  from Pune said, “This caregiving role gives me lots of satisfaction as I am serving my father who fulfilled all my wishes when I was a kid.” Another caregiver  from Guwahati mentioned, “When I was a child, my mother took good care of me, and now it is my responsibility to give back through my caregiving role.” Most caregivers from Prayagraj and Visakhapatnam emphasized fulfilling their caregiving role with utmost seriousness to inculcate positive values in their younger generation. A caregiver from Prayagraj  said, “I am doing my caregiving duty for my mother-in-law with all my heart so that my children should learn such good values now and perform such roles when required.” Similarly, a caregiver  from Visakhapatnam said, “I am glad that I am able to fulfill my caregiving role with dedication which I am sure will inspire my children to learn good values like taking care of older adults parents and grandparents.”3.1.2.Care and compassion was the second dominant theme among respondents from all four cities. The respondents emphasized that caregiving is a pious job as the caregiver not only takes care of the care receiver but also motivates them when they feel low due to illness and disability. In this way, they raise the care receiver’s self-worth through their caring behavior and compassionate service. Like a caregiver from Prayagraj  said, “Whenever my mother feels low because of her illness, I try to cheer her up. It makes me feel good.” Another caregiver from Visakhapatnam  said, “My father is 80, and sometimes he feels low because of his mobility issues; during that time as a son and caregiver, I encourage him to think positively.” Similarly, a caregiver from Guwahati  said, “I think caregiver’s role is a very holy job as in this way we help someone in need.” Some caregivers from Pune further shared their experiences of raising the care receiver’s confidence level through their care and determination. Like a caregiver  mentioned, “Whenever my parents want to go for long walks, it is hard for them to do that; in that situation, I join them and boost their self-confidence.”3.1.3.Most participants across the four cities emphasized that the caregiving role and responsibility gives a purpose to their life through engagement and involvement. According to caregivers, this brings satisfaction in their life and makes the family bond stronger. Like a respondent from Pune  said, “Serving my parents-in-law is my main responsibility; this work gives me satisfaction and keeps me busy.” Another caregiver from Prayagraj  said, “This caregiving responsibility adds more importance to my life. I will fulfill my responsibility with utmost sincerity.” Some respondents from Guwahati and Visakhapatnam emphasized that this role has strengthened their family bonding. Like a caregiver from Guwahati  said, “I am satisfied that I am in a position to take care of my mother; this is strengthening our bonding.” Another one from Visakhapatnam  said, “I am glad that I am taking care of my parents in their old age, it is making our relationship stronger.”3.1.4.This theme revolves around caregivers’ belief that their caregiving role contributes to their spiritual development and family welfare. It also highlights the importance or value they give to their caregiving role, which is bringing good deeds (Karma). Here most participants from Prayagraj and Pune mentioned that their caregiving role is helping them to attain their spiritual development. Like a caregiver from Prayagraj  said, “Taking care of elders is a holy job which is helping me in my spiritual growth.” Another caregiver from Pune  said, “This caregiving is a pious work and a means to spiritual attainment.” Some participants from Guwahati believed it led to the building of intergenerational bonding in the family. Like a participant  said, “Due to this role, I have become closer to my parents, and my children have become closer to me. We all respect and love each other.” Some others in Visakhapatnam related it to good karma. Like a participant  said, “I am sure these deeds of mine in the form of caregiving will bring good karma for my family and me.”3.2.Based on the thematic analysis, the four significant themes that emerged as factors contributing to positive caregiving are “Meaningfulness in life,” “Sense of belongingness,” “Personal growth,” and “Empathetic understanding.” Here also, some commonalities and differences were observed in the responses given by participants across four cities.3.2.1.In the case of the theme “Meaningfulness in life,” participants from Prayagraj, Visakhapatnam, and Guwahati derived meaningfulness in their work and life because of the sense of satisfaction and fulfillment they experienced in this role. Thus, highlighting meaningfulness and purposefulness is an essential factor in caregivers’ positive caregiving experiences. Like a participant from Prayagraj said , “This duty of mine towards my parents gives a purpose to my life.” Another caregiver from Visakhapatnam  said that “I am satisfied that I am taking care of my parents in their old age. It adds meaning to my life.” Similarly a caregiver from Guwahati  said, “This caregiving role is tough, but I am glad that my life is useful for someone.” Most participants from Pune equated it more with positive self-worth. Like a caregiver  said, “My caregiving role adds more value to my life and makes my life meaningful.”3.2.2.Most participants from all four cities reported a sense of belongingness between caregiver and care receiver as a significant contributing factor to positive caregiving. Here many participants emphasized the strengthening of family bonding and the development of a friendly relationship between caregiver and care receivers. Like a participant from Pune  said, “I always share all my information with my mother, and she shares everything with me. We are emotionally connected to each other,” Another participant from Prayagraj  said, “Through this caregiving role I have come closer to my mother-in-law. This has strengthened our relationship. She admires me for my hard work.” Under this theme, some caregivers also emphasized on building friendly relationships with their care receivers. Like a caregiver from Visakhapatnam  said, “While taking care of my parents-in-law, we have become good friends.” Similarly, a caregiver from Guwahati  said, “Me and my parents have become very good friends, we share every aspect of our lives with each other.”3.2.3.In the context of this theme, all caregivers from four cities shared that their caregiving role has made them more self-regulated, disciplined, flourished, and organized which in turn has contributed to their positive caregiving experiences. Like a participant from Pune  said, “Due to caregiving role, I learned how to manage my time in between my work and family.” Similarly, another caregiver from Guwahati  said, “This caregiving role is tough, but it also teaches lots of self-discipline, which I am learning while caring for my father.” Regarding personal growth, some participants have also reported that their caregiving role has made them more patient and self-regulated. Like a participant from Prayagraj  mentioned, “I do have a lot of stress issues, but when I think for a while and interact with my mother-in-law, I feel relaxed.” Similarly, another caregiver from Visakhapatnam  said, “My caregiving role for my parents has taught me to be patient and manage things well.”3.2.4.Regarding the theme “Empathetic understanding,” most participants from Prayagraj and Visakhapatnam reported that due to their caregiving role, they have become more empathetic and can recognize care receiver’s emotions well and thus feel optimistic about it. Due to this, they can provide need-based emotional support to care receivers. Like participants from Prayagraj  said, “While taking care of my parents, I have become more understanding towards their needs. I am glad I am able to fulfill my responsibility as a daughter.” Similarly, another participant from Visakhapatnam  said, “People often talk about tussles between daughter-in-law and mother-in-law; this happens because of lack of understanding. In our case, I make special efforts to understand her situation and provide emotional support to my mother-in-law. She also appreciates this.”Compared to the above, most participants from Pune and Guwahati emphasized that they can empathetically understand the challenges faced by their care receivers, work on resolving those challenges, and thus experience positive caregiving experiences. Like a caregiver from Pune  said, “My mother-in-law is very old, and she cannot walk on her own, so I have made it a habit that in the morning every day, I assist her in her walks. In this way, she meets other elders and interacts with them. She feels good about it, which gives me satisfaction.” Similarly, another participant from Guwahati  said, “As my parents are mostly staying at home, I help them connect with their friends and extended family via WhatsApp and Zoom. I know they like doing that. It makes me feel happy.”4.The findings of this study clearly indicate that informal caregiving, which is often considered burdensome, can be a flourishing experience for a family or informal caregiver, which not only contributes to their personal and spiritual growth but also strengthens their family bonding.Based on the study’s findings, it can be said that the themes have been found to be associated with the PERMA model. All four themes for the Nature of Caregiving, namely Caregiver’s attitude, Care and compassion, Roles and responsibilities, and Beliefs and values, are related to the domains of the PERMA model like Positive Emotion (P), Engagement (E), Relationships (R), Meaning (M), and Accomplishment (A). In the case of the first theme, Caregiver’s attitude, many caregivers across four cities reported that their caregiving experiences had helped them develop positive attitudes, making life more meaningful and satisfying. This is strongly related to the “meaning” domain of the PERMA model and justifies that getting involved in a cause that matters can bring more meaning to life . SimilarIn the case of the second theme, Care and compassion, most caregivers shared the positive feelings they experienced while providing care to their older adults parents or in-laws. The positive emotions experienced by most of these caregivers are hope, interest, joy, love, compassion, pride, amusement, and gratitude, which are very similar to the ones mentioned in the “positive emotion” domain of the PERMA model .Similarly, through the third theme, roles and responsibilities, caregivers reported that their caregiving roles have added purpose to their life through engagement and involvement in the care receiver’s life. This is related to the “engagement” domain of the PERMA model, which emphasizes on being mindful of the present moment and focusing on the current endeavors . In otheRegarding the fourth theme, beliefs and values, most participants shared their beliefs and values about their caregiving roles and emphasized how it contributes to their well-being. Based on their lived experiences, most of them considered caregiving a pious and holy duty that would bring them good karma and help them attain their spiritual growth. This is very similar to the “meaning” domain, which is associated with having a purpose in life and guided by personal values. Some association can also be established with the domain of “accomplishment,” which means achievement, mastery, or competence, as many caregivers reported their caregiving role as a valued accomplishment in their life.Thus, based on the findings and the above discussion, it can be concluded that caregiving can be a flourishing and positive experience among informal caregivers The attributes of positive caregiving will mainly comprise of caregiver’s positive attitude towards their life and caregiving role, care and compassion towards their care receivers, commitment towards their caregiving roles and responsibilities, and their profound beliefs and values which they associate with their caregiving duty.After discussing the nature of positive caregiving, the next important step is to explore the determinants or contributing factors of positive caregiving among informal caregivers. The four significant themes that emerged as factors contributing to positive caregiving are “Meaningfulness in life,” “Sense of belongingness,” “Personal growth,” and “Empathetic understanding.” All these themes have a strong association with the PERMA model.Regarding meaningfulness in life, most caregivers reported that they found their caregiving role significant and worthwhile. Thus, it has added more meaningfulness to their life. This is very similar to the “meaning” domain of the PERMA model, which is associated with doing things that give individuals a sense of purpose and worth. This sense of meaning can be different for everyone and is guided by personal values .The second contributing factor to positive caregiving is sense of belongingness. In this regard, most caregivers mentioned that caregiving leads to the strengthening of family bonding and the building of friendly relationships. This highlights the “relationship” domain of the PERMA model, which relates to how people connect with others . ResearcThe theme of personal growth emphasizes the growth and gains that caregivers have reported due to the caregiving role. Many shared that due to their caregiving role, they are leading a self-regulated and disciplined life and are experiencing various signs of personal growth like spiritual development, a sense of personal control, control of anger, and high self-esteem. These experiences are related to the “accomplishment” domain of the PERMA model, which emphasizes competence and personal growth .Another important contributing factor is empathetic understanding. Most caregivers reported that as they spend maximum of their time with their care receivers, they have become empathetic towards them. This empathy is not only helping the care receivers but also serving the caregivers in experiencing positive emotions, building positive relationships, and finding meaningfulness in their roles. All these are essential determinants of flourishing .Thus, based on the findings and the above discussion, we can say that positive caregiving is an important indicator of caregivers’ and care receivers’ well-being. The results suggest that when the caregivers looked at caregiving with a positive attitude by considering their caregiving role as pious and assigning meaningfulness to their act of caregiving, it not only made them connect with the care receivers on an emotional level but also contributed to their family bonding and personal growth. Findings also highlight that, in the context of older adults care receivers, positive caregiving in the family not only promotes healthy aging and reduces the percentage of older adults seeking institutional support but also contributes to building intergenerational bonding and making societies elder-friendly. The findings have implications for planning some action research, training, and counseling programs for promoting positive aspects of caregiving among informal caregivers.5.The study has certain limitations that need to be considered by researchers who wish to explore this area further. The study utilized a purposive sampling technique, which may not represent the entire population of caregivers of the older adults in India. The study was limited to only four cities in India, so the results may not be generalized to other regions. This study relied on self-reported data from caregivers, which may have been subject to social desirability or recall bias.6.Future studies could use a larger sample size and employ a mixed methods approach to triangulate qualitative findings with quantitative data results and provide a more comprehensive understanding of the positive aspects of caregiving. Future studies could also investigate the effectiveness of intervention programs that are designed to promote positive caregiving. This could include exploring the impact of training, support groups, or counseling programs on the well-being of caregivers and care receivers. It is also necessary to plan longitudinal studies to investigate how the dimensions of positive caregiving might change over time and to explore the factors that contribute to sustained positive caregiving practices among caregivers.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.Ethical review and approval was not required for the study on human participants in accordance with the local legislation and institutional requirements. The patients/participants provided their written informed consent to participate in this study.NM conceptualized and designed the study, reviewed, and analyzed the data. RD reviewed the data and checked the methodological quality. AT reviewed the data and edited the manuscript. MS reviewed the data and guided the field investigators along with NM to conduct interviews. All the authors contributed to data collection, interpretation of the data, manuscript writing and review of the manuscript.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Classic Galactosemia (CG) is a rare, autosomal recessive condition. Newborn screening and a timely galactose-restricted diet can resolve acute symptoms and decrease fatalities, but significant chronic, progressive morbidities remain and significantly impact daily life. The objective of this study was to better understand the burden of disease in children and adults with CGs and describe how morbidities evolve over time.A total of 49 individuals with CG from the United States (US) were included in the qualitative surveys (13 adults [9 self-reported] and 36 pediatric patients). Fifteen follow-up interviews were conducted with 5 adults and 10 caregivers, discussing 17 individuals with CG overall (2 caregivers each discussed 2 children).Qualitative survey and interview data demonstrated the substantial burden of CG. Difficulties in a wide range of functions were experienced, which included: speech articulation; language and communication; cognition, memory and learning; emotions; and social interactions. Most difficulties appeared in childhood and persisted or worsened with age. Most adults did not live independently. Others lived semi-independently and experienced many daily challenges and required support. Caregivers also described the burden of caring for someone with CG and spoke about the impact this has on their day-to-day life, work, and relationships.These findings demonstrate the pronounced and persistent burden of disease encountered by individuals with CG, and that the condition has a significant impact on the quality of life of caregivers. GALT) gene mutation that occurs in 1/16,000 to 1/60,000 births worldwide  has gotten better. Some thing[s] are hard for her to process though” . Some participants also described worsening of language and communication difficulties, “As she’s gotten older it has been more noticeable and significant” .Language and communication difficulties were frequently described by survey participants (n = 22) and were also discussed during interviews (n = 8). These difficulties typically began in early childhood (average age of onset: 3.5 years), they were experienced by children and adults, and they remained present into adulthood. Difficulties with expressive language (communicating what they are thinking and feeling) were most frequently reported by survey participants  such as, “I process information slower than average. Um, yeah for sure. I- I do- I can definitely have a foggy brain for sure. Um, just processing information not necessarily learning information just processing it, like hearing it, seeing it. Um, and I- I need like more time to- like I need to like gather my thoughts to answer questions. I need more time with that” . Adults typically experienced trouble with information processing while children generally experienced difficulties paying attention. An adult with CG explained how services in school had helped, “Learning difficulties have improved with accommodations in school. Been followed (in school) pretty closely. I still have problems processing information, but when it is shown and written out, information can be better understood” . Even participants who reported some improvements stated their difficulties persisted.Difficulties with thinking, memory, and learning were reported to have started (or were first noticed) in early childhood (average age of onset: 5 years) and persisted into adulthood. Thinking, learning, and memory difficulties were more frequently reported for adolescents and adults rather than younger children with CG, although initial indications appeared earlier, and typically worsened or showed no signs of improvement as the child aged. A range of difficulties were reported during the survey (n = 23) and interviews (n = 14), with difficulties related to decision making being the most frequent. Other difficulties experienced related to short- and long-term memory, information processing, learning, cognitive development, ability to follow instructions, and focusing and attention, “Um, I take medication now for anxiety, um, and I used to worry a lot as a kid” . Feelings of low mood/depression were typically reported for older children and adults with CG, further indicating these difficulties worsen with time.Emotional difficulties were noticed at a variety of ages (ranging from 1 to 23 years), but once noticed remained a challenge for the rest of their life and in some instances became worse with time. Emotional difficulties were reported in surveys (experienced by n = 20) and interviews (n = 16) including, anxiety, worry, low mood and depression, anger, and defiant behavior. Feelings of anxiety or worry were the most frequently reported emotional impacts for all participants . Among children with CG, issues with anger or getting upset easily were also frequently reported by caregivers. Adolescents and adults were reported to frequently experience low mood or depression. Some individuals received services such as therapy  or medications  to manage these difficulties, I just don't know when people, um, are being, say, funny or serious, sometimes... I guess that would be it, just normal social cues, I guess” . Adults with CG and their caregivers described increased social isolation with age as individuals avoided social situations where they might experience difficulties; “I just try to avoid social situations. I try to avoid talking to people as I know I can't explain things. I know people have been offended by things I have said and I don't want to offend people but other people think I am a snob because I don't talk to them so it's difficult to even be in a social situation” .Social difficulties were typically first noticed in later childhood (average age of onset: 7 years) and remained or were reported to worsen into adulthood. Survey data for all age groups who experienced social difficulties (n = 17) highlighted difficulties with social interactions , and trouble making friends  being the most frequently experienced. These were also reflected in the interviews (n = 16). However, for adults, being shy around new people was also frequently reported , which was described during interviews as being related to communication difficulties, ““She tends to be a little clumsy and may bump into things” , “Her coordination and balance are not real good. Walking, if it's an uneven path, she would have trouble with that…I think even as a child, she was not able to do all the physical things that peers of her same age would be able to do” . Some individuals received supportive services such as leg braces to try and assist with these difficulties, but not all.Difficulties with movement were reported across age groups in surveys (n = 11) and interviews (n = 12), and were generally first noticed in childhood (average age 6.5 years) and worsened into adulthood. Movement difficulties became more apparent with age, with adults most frequently reporting that they experienced tremors. The most frequently experienced movement difficulties were trouble with balance and coordination which were experienced by older children, adolescents and adults, I had to start on birth control when I was, I think, 13 just to get my period. Um, so I, I couldn't, like, have it by myself” .Based on the surveys, all 6 adults with CG who identified as female and 1 who preferred not to report their gender experienced ovarian failure as did all female-identified people aged 13 to 17 years (n = 6/6). In addition, caregivers of 2 children aged 2 to 6 years discussed concerns over ovarian failure and infertility in the future. Ovarian failure and related issues were noticed during adolescence (average age 15 years) around the time of puberty. Infertility was the most common difficulty reported for adults, while delayed puberty was most common for children with CG. Most survey participants who experienced ovarian failure had received estrogen replacement therapy , with other participants with ovarian failure receiving either hormone replacement therapy or other treatment, “They're stable and at this point, she sees the eye doctor every six months and as long as things aren't getting worse, we've elected not to have surgery so far. But that may be something that she'll need in the future” .In qualitative surveys, participants who reported vision problems related to cataracts (n = 4) typically first noticed these in adulthood (average age of 23 years for adults), which was consistent with the fact that vision problems were only present for 1 child with CG for whom it started at 1 year old. For survey participants, once vision problems due to cataracts were experienced, they remained either with no change  or became worse with time . A caregiver described the current state of an adult’s cataracts as, ““The reality of galactosemia and the cognitive issues it has presented in [name] impacts my life in that he needs assistance and guidance day to day by me. I am his go to when he needs help ” . Caregivers of both children and adults with CG had to make sacrifices in other areas of their lives in order to provide the needed time and resources to provide care. Caregivers also discussed feelings of guilt, stress, and frustration. As one parent caregiver stated, “we feel a lot of, like, the guilt and just kind of like, ‘Oh, we did this to you’ but we didn’t know, obviously” . Caregivers also reported negative impacts on family dynamics and activities, limitations on time and resources, and financial burden. Further, caregivers reported their ability to work, pursue a career, and make future plans were significantly impaired. Caregivers of children with CG explained that they were unable to work because of the time required to provide care. For example, “It affected me working. I ended up going part-time at work. Um, my parents were taking care of my kids because I didn't trust other people” . Caregivers were not only impacted currently, but also worried about their plans and goals for the future. For caregivers of children with CG, this was focused on the unknown as to whether they would need to provide care for the rest of their lives “We have no idea what your plans are gonna be. Whether we’re gonna have to be caretakers, caretakers for the rest of our lives, or if we’re gonna be able to retire, you know, and enjoy being old” .Caregivers’ responses in both open-ended survey questions and interviews also highlighted the impact of caring for someone with CG. Caregivers reported damaging social, emotional, and financial impacts because of their caregiver role. Caregivers’ day-to-day lives were impacted by the need to provide constant care and support to the patient and that they were the “go-to” for the person they care for, In qualitative surveys, participants were asked to rank the top 5 difficulties they/the person they care for experienced from Most Challenging (5 points) to Least Challenging (1 point) , including a family member or caregiver (46%), or partner/roommate (23%). Survey and interview participants described how adults require varying degrees of assistance in their day-to-day lives from caregivers, parents, or roommates. The degree of daily assistance results in most adults not living independently with others living only semi-independently. As part of the qualitative survey, caregivers reported whether they felt that the child they care for would be able to live independently in the future. Although many survey respondents indicated that they believed the child they care for would be able to live independently on their own , during interviews when discussing difficulties in-depth, caregivers expressed concerns over potential future challenges to live independently, with some caregivers explicitly stating they thought the child would not be able to live independently. These concerns included the child’s ability to look after themself but also to form relationships with others outside of the caregiver relationship.In this study we extended our investigations into the burden of disease of CG by exploring the experience of CG in children. Our findings are consistent with the assortment of difficulties reported by other investigators and our earlier work –7, 15–18Our findings show that most difficulties appear in childhood and either persist or are reported to worsen through adulthood. This is consistent with our earlier study which revealed that adults with CG face substantial HRQoL challenges. While individuals with CG inevitably experience worsening difficulties, a noteworthy finding of this study was that although some individuals reported improvement with current interventions and services, the benefits were limited, difficulties remained, and individuals with CG typically functioned at a level lower than their peers. In addition, some functional areas reported a clear worsening with age. Current interventions and therapies may be inadequate and, in some cases, ineffectual to address the difficulties associated with CG. As such, a treatment, intervention or therapy that has superior and more universal benefits will contribute not only to improving quality of life, but that of caregivers as well.Further, this study provided insight on what individuals with CG and their caregivers felt were the most challenging difficulties, these were in the cognitive, communication, and social areas, indicating that the diminished ability to have normal social and intellectual functioning weighed heavily on the individuals with CG as well as their caregivers. Thus, future directions for this research include exploring how new treatments can meet the needs of individuals with CG as well as their caregivers in these areas identified as most challenging. In addition, future research should consider tracking changes in the burden associated with CG over time in longitudinal investigations to better understand how treatments can address the changing developmental needs of individuals with CG.This study has some limitations that should be considered when evaluating the findings. The participants were all from the US, mostly White, and mostly non-Hispanic; thus, there was a lack of geographic, racial, and ethnic diversity. Although there is no evidence these demographic factors can significantly affect experience related to CG, it would have been desirable to have participants of a more diverse population to ensure that the findings are representative of the broader population. In future research, a study with targeted recruitment to include a more diverse sample of individuals with CG and their caregivers can help to further understanding of the ways in which individuals historically excluded, particularly those with less access to specialty care and supportive services, experience the burden associated with this condition. Participants were recruited via the Galactosemia Foundation, which can introduce bias regarding those who respond to advertisements. However, this is a commonly used recruitment method for qualitative burden of illness research, particularly for rare diseases . Also, mThis study extended understanding of the experience of CG for children and adults (from 1 to 60 years old) and their caregivers. Specifically, the study addressed the 4 aims: to increase understanding of the burden of disease, how difficulties evolve over time, what participants perceive as the most challenging difficulties, and the ability of individuals with CG to live independent lives. CG imposes a substantial disease burden on individuals, which mostly appears in childhood and persists or worsens into adulthood making it difficult for adults with CG to function independently or at least requiring some day-to-day support. These challenges impose difficulties for caregivers as well, and especially for those caregivers of children with CG whose fears of future worsening weighed heavily on them. Some but not all benefit from current supportive services and even those benefits are limited. These findings demonstrate an unmet medical need for CG patients for treatments that would more consistently and durably improve their symptoms and by extension the quality of life of their caregivers.


Frequency showed that mainly “Defense in Line of progression” and “Proximal contact” were the most activated variables, followed by “Harassment” and “Deterrence” for all constraints. However, only constraint 2 highly activated two collective motor behaviors, while the rest only did it with individual motor behaviors. It is concluded that the constraints used in training seem to be effective in activating a group of defensive handball motor behaviors, specifically those that are basic for female school handball players.Traditional models to train and teach young players in team sports assume that athletes learn as linear systems. However, an actual methodology called Non-Linear Pedagogy (NLP) accounts for the fact that the players and the team are complex dynamic systems. Experiences in handball under this methodology are scarce; due to this, an observational study has been conducted with a follow-up, idiographic and multidimensional design, in which 14 female school handball players belonging to four different local teams in Santiago, Chile (age = 15.55 + 0.51) agreed to participate in three special handball training sessions with the use of the NLP methodology where three different constraints were used. Descriptive analysis with the Chi-squared test showed a total of 252 observations where most of the variables were dependent on the constraints ( Handball, as a team sport, requires the domain of tactical knowledge, coordinative skills, physical fitness, mental strength, motor skills, social relationships, leadership, communicative skills, power, endurance, speed and decision-making abilities, among several other variables ,2. All oHowever, traditional teaching models for team sports have only focused on the development of technique, followed by tactical skills and the introduction of the rules of the game, using an isolated context complemented with strength and conditioning training . Once thIn this sense, traditional training models have pushed the nature of teams sports as a non-cyclical game to a standardized one, where the elements of uncertainty have decreased, improving the dependence on strength and conditioning work with a lack of creativity from the players, except for those exceptional highly talented ones .The aforementioned is possible to see in handball, as it has been stated that performance indicators in the World Handball Championship for men and women are exclusively the “Anthropometric Indicators” (only for men), “Age index,” and “Scoring index,” while tactical behaviors, decision-making, and team players interaction are not taken into account . Also, iIn response to these limitations, an alternative teaching model has been developed based on the complex dynamic system theory called Non-Liner Pedagogy (NLP), which involves the interaction between the players and the environment through the implementation of adapted games , comprehThe NLP allows players to elaborate paths of verbal and non-verbal communication, organize strategical responses, reproduce emergent tactical motor behaviors, analyze the environment to make decisions, and especially adapt their individual resources to improve the team’s performance . The NLPThe NLP to teach team sports was first described by the study of Chow et al.  as an arWith regards to the CMs and their effects on motor behavior, one of the first studies that have described an experimental design is the study of Praxedes et al. . Their rLater, specifically in handball, only a few studies were conducted using the NLP ,17,18,19Limited evidence has been reported according to handball’s defensive motor behaviors developed by NLP. At the time this article was made, only the study of Flores–Rodríguez and Ramírez–Macías  has giveAs the evidence has demonstrated, the effectiveness of NLP on handball´s attacking motor behaviosrs is still limited, and due to the lack of more evidence regarding handball’s defensive motor behavior and the absence of this kind of study in female players, researchers have hypothesized that the use of NLP in handball training might activate specific handball’s defensive behaviors. Therefore, is the aim of this research to describe the effects of the use of NLP methodology in the development of defensive motor behaviors in young female handball players.This is a descriptive, quantitative, observational study planned to obtain relevant information regarding our investigation objective. Particularly, this corresponds to a follow-up, idiographic and multidimensional design, as there was an inter and intra-sessional analysis which provided data about the frequency of the performed actions and about some behaviors of the players independently as part of a system (team) in both individual and collective responses . The samBefore the study started, both the parents/guardians and the participants were notified and informed about the procedures, making it clear that their participation was totally voluntary. Also, informed consent was signed by the parents/guardians as well as by the players. It should be noted that this research was approved by the Research Ethics Committee of the University of Granada under the registration number: 2000/CEIH/2021, responsibly ascribing to the standards agreed in the Helsinki agreement [(A)Warm-up: Where all players were asked to jog for 5 min, then to practice freely and individually, for 5 more minutes, all the different abilities they knew, and later in couples, to jog freely, passing the ball between them;(B)Individual defensive technical training: Consisted in different exercises related to defensive movements, specifically to move oriented according to their opponent, to maintain distance, or to apply pressure depending on the attacker’s intention;(C)NLP exercises: A specific exercise is applied in every session, however, with different constraints in each 1;(D)Offensive training: Comprised of specific exercises to improve mobility and dynamism during the ball circulation performed by the attackers. The goal was always to keep the speed of the ball, passing it among players while they were moving side to side by performing different combinations of movements with no opponents;(E)Cool-down: In all sessions, players were asked to apply self-massage and stretching.All the selected players participated in 3 special handball training sessions that comprised the same structure described next:All participants were asked not to eat food and not to ingest stimulant beverages such as coffee or energy drinks at least an hour before training sessions. Also, players were asked to maintain the same type of diet that they present daily. For hydration, the players did its ad-libitum. At the time players participated in the study training sessions, no training on their clubs was performed, thus not competing either.As seen in previous studies regarding NLP methodology ,23,24, a-Defenders must prevent attackers from moving forward to the goal area;-Defenders have individual responsibilities, although they can help their teammates without losing their individual opponents;-Defenders must prevent attackers from penetrating the space between and behind the defenders.In each session, before the exercise started, the coach explained out loud to the defenders the next: These 3 indications correspond to what Anton (2003) has described as the fundamental pillars of defensive tasks.-Session 1: Each attack of the 6 had to be done with the 6 balls at the same time, and no circulation of an attacker could be done through the central circle.-Session 2: Each attack of the 6 had to be done with only one ball at a time, and no circulation of an attacker could be done through the central circle.-Session 3: Each attack of the 6 had to be done with only one ball at a time, and the circulation of an attacker can be done through the central circle as a pivot position for only 3 s.For each session, a constraint was given to conditioning the motor behavior of defenders. These constraints were the next:The coach remembered out loud the constraints for the DT players, and no other information was given before or during the exercise, as well as no feedback after it. It is important to highlight that none of the players had experience with collective defensive tactics before this study.® (USA) added to a tripod which was located on the side of the court 4 m high [® (USA), in which the NLP exercise of every session was saved independently of the rest of the session. Every NLP video was at the time edited to highlight every time the exercise started or finished and also when the role of the teams changed; this was to facilitate posterior analysis using the Kinovea® software (Open Source). For the observation, an ad hoc observation instrument described by Anguera et al. [As this is an observational study, every session was video recorded using the main integrated camera of an iPhone XR 64GB4 m high . Every ra et al.  was adapa et al.  : Every time the player should perform the action, but she didn´t; EP (Effectively Performed): Every time the player performed a correct action as the situation need it; NN (Non-Necessary): Every time a defensive behavior was not necessary to be performed. ® spreadsheet using a numeric code for each qualification  to determine the final amount of observation correctly performed.Finally, all the results were arranged in an Excel (USA)® v28  was used for the data analysis. As this is an ideo-graphic, follow-up, and multidimensional observational study, descriptive statistics are presented as frequency. To compare the frequency of the emergent or non-emergent handball´s defensive behaviors regarding the applied constraints, a contingency table was used. To determine if emergent or non-emergent handball motor behaviors are dependent on the applied constraints, the chi-square test was used.The statistical software SPSSp-value < 0.001 was obtained except for “Proximal Contact,” in which the results showed a p-value of <0.005, and for “Defensive Adjustment Through the Back” where the values remain all the same for each observation, so this variable was taken as a constant, so the frequency was not given. In this sense, it is evident that almost all variables are effectively dependent on the constraints. The descriptive analysis  followedWhen constraint 1 was applied , all the handball’s specific defensive motor behaviors got activated. However, not all of them were at the same frequency. Mainly, two individual behaviors, which are “Defense in line of progression” and “Proximal contact,” were activated the most, with an average of 57 observed actions. For the rest of the handball’s specific defensive motor behaviors, an average of 10 observed actions were obtained .Regarding constraint 2 , “Defense in line of progression” and the “Proximal contact” were highly activated again with the same average of 57 observed actions. Nonetheless, group defensive motor behaviors were also activated in a higher frequency, with 57 and 52 observed actions for “Timely Helps” and Defensive Titl” correspondingly. All the remaining handball’s specific defensive motor behaviors were activated as well but at a lower frequency. However, compared to constraint 1, a higher number of actions were seen for “Harassment”, with 39 more actions in constraint 2 compared with constraint 1. In the same way for “Deterrence”, 24 actions were detected above the four seen in constraint one. A similar phenomenon occurred for “Defensive Adjustment”, “Ball Interception”, and “Change of Opposition”, where an average of 14 observed actions got activated above the seven actions seen during the first constraint .On the other hand, the third constraint  also activated the “Defense in Line of Progression” and “Proximal Contact” in this opportunity with a higher number of observed actions (79 and 74 correspondingly). Differences are possible to observe when comparing constraint three with constraints one and two, as the group defensive motor behaviors reduced their frequency to 16 for “Timely Helps” and 0 for “Defensive Tilt”. Another difference is that “Harassment”, “Deterrence”, and “Defensive Adjustment” were activated more during the third constraint compared with the second and the first. These handball’s defensive motor behaviors presented an average frequency of 23 observed actions above the same behaviors during constraint 2 and an average of 46 above constraint one .Finally, constraint 2 was the one which has activated more collective actions such as “Timely helps”, “Defensive Tilt”, “Change of Oppositions”, “Deterrence”, and “Defensive Adjustment” compared with constraints 1 and 3. However, constraint 3 has activated in a higher frequency two of the aforementioned behaviors while constraint 1 only activated a few behaviors in a lower amount.The aim of the present study was to describe the effects of the use of NLP methodology in the development of defensive motor behaviors in young female handball players. For this, three constraints were introduced to a specific exercise which was repeated in three training sessions (one constraint per session) following the examples of the studies of Flores-Rodríguez & Ramírez-Macías .It has been stated that it is essential for handball coaches to be aware of the influence that constraints exert on the players’ behaviors . As mentIn handball, to defend against any offensive action, there are several technics and tactical skills which respond to three basic pillars that always have to be accomplished during the whole game: to avoid attackers from getting near their zone of effectiveness, to avoid attackers from shooting at the goal, and to recover the ball at any given opportunity ,31,32. TRegarding the motor behaviors of “Harassment” and “Deterrence,” these were slightly activated during constraint one. This may have happened due to the lack of receivers of a pass, as all attackers had a ball in their possession at the beginning of the exercise. In addition, the limitations that attackers in possession of the ball have in terms of mobility throughout the court compared with defenders due to the rules of the game  probablyParticularly constraint two was the only one that activated in a high frequency of actions the two collective motor behaviors, “Timely helps” and “Defensive Tilt”. As mentioned before, this constraint allowed more attackers to move unlimitedly through the court, with the exception of the central zone, prioritizing movements in width over those in-depth toward it. In general, the attackers tend to seek free space to penetrate into the central zone, as often is done in the real game in young female teams ,38. The In addition, the study of Musa & Menezes  about coThe handball’s motor behavior of “Ball Interception”, “Change of Oppositions”, and “Ball Interception during Dribbling” were all slightly activated in all three constraints. The explanation for this is that in most of the attacks in every session, dribbling was barely used, passes were often given far away from the defense, and crosses of the attacker were not given regularly, except for the constrain two that activate “Change of Opposition” 21 times. However, these crosses were not significant in reaching the goal.One of the main limitations of our research has been the difficulty of limiting the order effect that sessions may have had on the results. However, as this is an observational study, the results are a description of what has been observed. Also, a second difficulty was finding data regarding technical and tactical skills in young female handball players, as most of the studies refer to strength and conditioning training or anthropometric profiles. However, there was enough literature from well-known international handball coaches that helped the present discussion.After the analysis of the results and the discussion of them, the authors agree that the constraints used in the proposed exercise give the impression of being effective in activating a group of specific defensive handball motor behaviors for young female handball players. It is important to highlight that also, by analyzing the three constraints separately, these activate differently, so the exercise used in this research with the correspondent constraint could also be used independently to improve a specific group of defensive motor behaviors.


In “Author’s Responses to Peer Reviews of ‘Forecasting the COVID-19 Pandemic in Saudi Arabia Using a Modified Singular Spectrum Analysis Approach: Model Development and Data Analysis’” :e28742) one error was noted.The reviewer IDs of two anonymous reviewers have been removed from the published article to preserve anonymity.The correction will appear in the online version of the paper on the JMIR Publications website on April 26, 2021, together with the publication of this correction notice. Because this was made after submission to PubMed, PubMed Central, and other full-text repositories, the corrected article has also been resubmitted to those repositories.


F = 13.74, p < 0.001, one of which was a significant predictor of MPA, t(99) = 3.06, p = 0.003. This factor comprised themes of failure, catastrophising, and incompetence/dependence. Study 2 qualitative analysis revealed various key parenting themes experienced in childhood that differentiated low- and high-MPA scorers in adulthood. Findings from both studies are discussed in light of clinical applications and interventions, and implications for both parents and music educators.Music Performance Anxiety (MPA) is a common challenge for classical musicians, however its etiology has received minimal research, particularly in regards to caregiver experiences during childhood and adolescence. The aim of this research was to explore the impact of childhood experiences with parents along with patterns of dysfunctional cognitive schemas that develop through childhood  on the manifestation and severity of MPA in adulthood. Study 1 employed 100 adult professional, amateur, and tertiary student classical musicians from across Australia. Participants completed the Young Schema Questionnaire (YSQ) and the Kenny Music Performance Anxiety Inventory (K-MPAI). Study 2 included eight participants from Study 1, five of whom scored 1.5 standard deviations or more above the mean K-MPAI score and three of whom scored 1.5 standard deviations or more below the mean K-MPAI score. Participants were interviewed about experiences of parenting during childhood and adolescence, along with their experiences of MPA and musical training. Interpretative phenomenological analysis was used to explore themes in the interview data. Study 1 factor analysis revealed four higher-order EMS factors,  Research into the phenomenology of music performance anxiety (MPA) has received significant empirical attention, though to-date it has predominantly focused on factors such as symptomatology, demographic and contextual factors, and comorbid psychopathology , a generalized psychological vulnerability , and a more specific psychological vulnerability . Barlow's model therefore acknowledges the interplay between biological predisposition, early social experiences, and learned response patterns to repeated life experiences that induce anxiety (such as past anxiety-inducing performing experiences). In relation to MPA, Barlow's model provides a foundation to explore the impact of early social learning experiences on the development, experience, and perpetuation of MPA in adult musicians. In the context of MPA research, two prongs of Barlow's model are illustrated by Osborne and Kenny  predicted MPA in adolescents, and that this prediction was strengthened by the presence of negative appraisals by young musicians in relation to their perceived “worst” performance experiences .In order to provide a robust theoretical framework for a developmental investigation of MPA, Barlow's  model ofnd Kenny , who fougeneralized psychological vulnerability that may precipitate MPA in classically-trained musicians. The research comprised both quantitative and qualitative studies, and investigated the impact of early experiences with primary carers on the development of cognitive schemas (or cognitive organizing frameworks) that are relevant to a musician's perception of their social world and their sense of self. Through this research, we hoped to illuminate core social and emotional processing schemas that might predispose the development of MPA.This paper outlines two studies that explored the role of early interpersonal experiences with primary caregivers in the development of MPA, and examined how the development of MPA may be shaped by an individual's developing self-concept, beliefs, and perceptions of the social world through their formative years. In order to address the shortfall in research exploring early parenting experiences in the development of MPA, we applied Young et al.  Early MaYoung's Early Maladaptive Schema model .EMSs therefore serve as templates for the processing of later experiences Young, . They ardysfunctional childhood experiences. For example, an individual may develop an EMS relating to perceived incompetence and dependence, which may develop in the context of parental overprotection or over-sheltering during childhood, leading to destructive beliefs around lack of self-efficacy and autonomy as the child matures.EMSs do not always develop as the consequence of early trauma; rather, Young et al.  refers tcore emotional needs that children require for healthy psychological development. These include: (1) Secure attachments to others ; (2) Autonomy, competence, and a sense of identity; (3) Freedom for the child to express valid needs and emotions; (4) Spontaneity and play; and (5) Realistic limits and self-control. Young et al.  were found to mediate the relationship between childhood adversity and anhedonic symptoms in adolescents  mediated the relationship between childhood adversity and anxious symptoms. Similar results have been found for the mediating role of various EMSs  in the relationship between poor parenting in childhood (low care and high overprotection) and the development of depression in adulthood   would demonstrate a significant positive association with MPA scores. Given that previous research has identified a broad range of EMSs associated with relevant constructs such as Social Phobia and anxiety more generally, further exploration of specific groups of EMSs that may serve EMSs as predictors of MPA was exploratory in nature.In Study 2 we hypothesized that particular aversive experiences of parenting during childhood (such as overcontrolling or neglectful parenting experiences) would be more likely to be reported in adults who experienced high levels of MPA.n = 100) were recruited via social media, email, and advertisements placed within The University of Melbourne, and were required to be 18 years and over, classically-trained, and have five or more years of experience on their instrument. The sample comprised 25 males, 74 females, and one participant who preferred not to provide gender information. The age range of participants varied from 18 to 24 years (13%) through to 65 years or older (1%). A majority of participants were in the 25–34 years (29%) and 35–44 years (30%) age groups, with 17% of participants in the 45–54 years and 10% in the 55–65 years age groups. The sample contained a mixture of full-time (25%) and part-time (24%) professional performers, undergraduate and postgraduate students (14%), amateur musicians (32%), and five participants who had ceased playing due to their MPA (5%). Principal instruments were varied, but were biased toward string players, with 60% of the sample playing a stringed instrument, and the remainder being spread across woodwind (17%), keyboard (8%), brass (4%), percussion (2%), and voice (9%).Participants , primary musical instrument, years of experience on instrument, experience of treatment, and use of beta blockers.K-MPAI; Kenny, r = −0.53 with Core Self-Evaluations (measuring self-esteem and self-efficacy). A cut-off score of 105 on the measure was indicated for clinically significant levels of distress  was demonstrated for the previous 75-item version of the YSQ-S3 (measuring 15 rather than 18 EMSs) in an Australian undergraduate sample . As all alpha coefficients were above the cut-off point of 0.70  and for all YSQ-S3 EMSs  was significantly higher than in a previously published sample of tertiary flute players  = 8.51, p < 0.001; Kenny et al., M = 83.73, SD = 40.72; t(471) = 9.00, p < 0.001; Kenny et al., Scores for the K-MPAI in the current study ranged from 61 to 186. Interestingly, the mean K-MPAI score  = 1173.40, p < 0.001, indicated that the correlations between items were significantly large to support a principal axis factor analysis. These four factors were labeled according to descriptions of the EMSs provided by Young et al.  and z = 1.18 (Factor 2), indicating that they all adhered to assumptions of normality and were appropriate for further parametric testing. Variable numbers were within those recommended by Cohen  = 13.74, p < 0.001, R2 = 36.70% revealed that Inadequacy/Impaired Autonomy was the only significant predictor of MPA, t(99) = 3.06, p = 0.003  as defined by Young et al.  was identified as a significant predictor of MPA in adult musicians. This group of EMSs included g et al.  entitledImpaired Autonomy and Performance also includes the Enmeshment/Underdeveloped Self EMS, which did not emerge within Factor 3 of the current model. Indeed, in the population of musicians investigated in the study, Enmeshment/Underdeveloped Self appeared as a stand-alone EMS with its own independent significant factor loading (Factor 4), with minimal loadings onto other factors.In addition to the EMSs within Factor 3 of the current study, Young et al.  domain oImpaired Autonomy and Performance domain and Factor 3 of the current model it is not unexpected. Young's EMS domains were developed from a theoretical model rather than statistical analyses, and previous factor analytic investigations have revealed mixed support for their robustness . The combination of Vulnerability to Harm or Illness and Dependence/Incompetence suggests a particularly debilitating mix of cognitions and beliefs in which an individual may harbor a deep dependency on others due to their poor self-efficacy, coupled with a pervasive belief that others  will not be safe and will not support them. In the context of musical performance, this may manifest as anxiety regarding potential negative repercussions of performance and audience feedback (expectations of harm and catastrophe) coupled with a lack of sense of control or capacity to manage the pressures of the performance adequately (a sense of dependence and incompetence). This has been demonstrated in previous studies in which musicians' “catastrophic” fears were identified as a fear of negative evaluation  typically develop from a family background characterized by enmeshment, undermining of a child's confidence, overprotection, or failing to reinforce the child for performing competently outside the family; a proposition that has received empirical support in non-musician populations . Collectively these EMSs, which are characterized by expectations of failure, a sense of incompetence and dependence, and an expectation of catastrophe and concurrent sense of vulnerability in adulthood, are a significant predictor of MPA in adulthood.By exploring the relationship between EMSs and MPA in adulthood, Study 1 identified typical maladaptive cognitive schema patterns experienced by musicians with MPA. Through this investigation, likely childhood environments associated with those particular clusters of EMSs could be posited based on Young's theory and existing empirical research. Study 1 findings suggest that if core emotional needs of children are not met by primary carers, particularly through overcontrolling or overprotective parenting styles, this may contribute to the development of a combination of EMSs . This cut-off was chosen to gain an adequate sample size for comparative interviews and meaningful qualitative data analysis, based on willingness and responsiveness of participants from Study 1. The final sample comprised three low scorers (K-MPAI range = 87–91), and five high scorers (K-MPAI range = 148–179), capturing the range of MPA.Study 2 participants were selected from participants who scored +/- 1.5 standard deviations from the mean K-MPAI score of 122.53 , reflecting strongly conflicting and unstable messages of love and affection provided by her.However, this participant also described violent, unpredictable episodes of rage by his mother at other times  along with involvement in the child's learning, both in school and extracurricular activities.Several participants with high MPA recalled their parents not providing for them or supporting them adequately in practical day-to-day needs:At home, when we were playing, our Mum never played with us at all. Didn't use to read us stories at night. [HighMPA-6]I just don't remember that [my parents] were around that much…I remember Dad doing a lot of the cooking, but it was very basic ‘soup a la Dad': Brown some mince, put a couple of cans of soup in and a chunk of frozen mixed vegetables. Ready in 20 minutes, and then they'd both be off…They would have been able to put us to bed I guess. We would come home from school and there would be babysitters…Mostly we'd just sit there and watch telly or whatever. [HighMPA-5]This was coupled with recollections by some high MPA participants that they were not taught basic daily living skills that would have assisted with increased autonomy and confidence as they entered adulthood:[My mother] just did stuff. She certainly didn't delegate. Even when we got older, she just did it all. I never learned how to cook until I moved out of home. Even the washing. We would say “Mum, why don't you get us to put on the washing?” and she just never did…A bit of a martyr syndrome thing going on…I felt angry because it did impact on us…because in a way, she projected her feeling of martyrdom and we had to carry the burden of being the burden for her. And I felt angry about that. [HighMPA-6]We had a cleaning lady who would come, but I didn't really learn how to…I had no idea when I left home how to operate a washing machine…I was pretty useless as a human being other than I could play the piano reasonably well. [HighMPA-5]Conversely, the strong focus on music practice was a theme in the high MPA participants:[Mum] tells me now that ‘I was expecting you to practice every day, so I felt like that was enough'. That she didn't expect us to tidy our rooms as well. Personally, I think that's a mistake. I think she's got it topsy turvy. [HighMPA-5]Parents of high MPA participants were in some cases remembered as having critical and/or personal involvement in their child's music practice and achievements, or in other cases being completely disengaged and disinterested:I know at one point [my mother] had 100 students [as an instrumental music teacher]. On top of that was this eisteddfod and running her violin ensembles…maybe she just couldn't say no. If someone wanted to learn violin, she felt obliged to teach them because she felt if she didn't do it, there was no-one else. But her loyalty was there…One thing I have wondered is, on some level I was made to feel I was somehow special by my parents, by my Mum. I think by not having to do chores around the house or contribute to the household but also being always made to practice…because of this musical training…I felt like I was – it sounds really embarrassing and juvenile – that I was special in some way. I had this underneath idea that I was better than other people because I played an instrument…But at the same time, I had this feeling of total incompetence or worthlessness. [HighMPA-5][Mum] never came to any lessons, ever [and] Dad had very little part in my life. He was like a background shadow…[but] the classic thing was you'd be doing your piano practice and [Mum would] be in the kitchen doing something like the dishes or making the dinner or whatever, and then she'd just go insane! She'd go “No! No! That's wrong! That's wrong!!” and she'd come running into the room and say “That's wrong! You have to do this!” And you'd have to do that! [HighMPA-7]In contrast, all the participants with low MPA recalled involvement by their parents in daily activities and daily life, describing encouraging and safe environments, actively supporting intrinsic motivation and valuing family connection:My poor father was a taxi driver [driving me to rehearsals and concerts throughout the week]… We [also] played sport together as a family every week. From my early teens until I left home, we played badminton and table tennis every Saturday night at the local sports center. [LowMPA-2]This key parenting theme relates to the demonstrations of non-judgemental encouragement, emotional support, and unconditional praise provided by parents. Participants with high MPA frequently described only conditional praise when reflecting on their parents' provision of encouragement and support:It seemed like you'd only get attention if you did something good…You'd get told off enough, but you wouldn't get positive affirmations. It felt like the only time that you'd get positive affirmations was when you do something that my Mum could brag about. I think both my Mum and Dad were insecure and felt inferior, so the fact that she'd have something so she could say “My daughter's better than you”. [HighMPA-7]Other high MPA participants reported of a lack of encouragement expressed by parents entirely:[During music practice] I remember some pretty awful times actually. I remember us both screaming at each other. [Mum] crying but still sitting next to me, making me do it. I don't remember any nice things about that. [HighMPA-5]Of the low MPA participants, experiences were described as consistently supportive, free from specific expectations by parents, and encouraging of freedom and autonomy:They expected us to be quite independent. But they were very supportive of things that we wanted to do. I never felt like it was their expectation for doing things that was driving any of the things that we did at school. There was very much a feeling of what we did was up to us. [LowMPA-1]My parents] always encouraged me and my brother as well, to do whatever we liked, and whatever interested us to follow up to the nth degree. Very supportive. Although they weren't from a musical family themselves. [LowMPA-3]This key parenting theme relates to parenting styles that focus on particular vested interests and outcomes rather than a child's intrinsic motivation. It was often implemented through overcontrol and criticism, and frequently incorporated comparisons with other children.Of the high MPA participants, expectations regarding music training were high, though they were associated with differing parental agendas:Mum was the president of the [local] eisteddfod, so she ran this big music competition every year…and so I was made to go in it every single year. Most of the time I wouldn't even get a place. And I just remember, I would just be devastated and howling on the bed, and it was just awful…I think there was this pretty strong message there with how I performed was linked to my value as a person, to be honest. Because not very much time was spent with my [parents] other than being concerned with whether or not I had practiced. [HighMPA-5]I think my Mum had this idea that we had to be ‘perfect ladies'. She used to say that to me and my sisters and had this idea of what perfect ladies should be. We had to have gloves, fancy dresses, a handbag. I never really knew what the handbag was about. I'd say to her “What's a handbag for?” and she'd say “You have to keep your hanky and your bottle of perfume”. This was in primary school, from probably around eight or nine…Part of ‘being a lady' was that you had to be able to play the piano.” [HighMPA-7][I remember one time] where I couldn't find my school shoes and I hadn't practiced so I wasn't allowed to go to school, and by the time I'd got to school, I remember having to be taken out of the classroom very quickly because I must have been hysterical. I was about seven. And I remember telling the teacher that Mum had told me that if I didn't do my practice and didn't do things properly I wouldn't get to go to university. [HighMPA-4]In contrast, all participants with low MPA uniformly recalled their parents' unconditional support of their interests and autonomy:It never felt like I should have done better in [Mum's] eyes. She was pretty unconditional about that. If this is what you want to do, then we'll support it. I never felt like I wasn't good enough…I never felt like I was letting my mother down with anything that I did. As long as I tried, I think she was really fine with that. [LowMPA-1]My parents didn't push me to do stuff…I really loved school and the learning experience, and the group dynamics of playing in ensembles and the solo instrument as well. That appealed to me as well and I got good at it quite quick. [LowMPA-2]This key parenting style relates to experiences of unpredictable and/or explosive anger outbursts by parents, which were described by several participants with high MPA:We were really scared of our Mum. My parents had their own mental health problems, so I can remember coming home and my older sisters would say “Mum's in a bad mood!” and we'd go somewhere and hide because we didn't want to get near her. Her rage was just really, really scary. Yeah, she would hit us. But was mostly her rage that you were most afraid of…Basically I've grown up feeling that I'm a bad person and I find it really, really hard to throw that off. That I'm always worrying that I'm going to be criticized and what I've done wrong now…I think I just go about with the expectation that people aren't going to accept me for what I am. [HighMPA-7]My Mum is quite a highly anxious person. Very, very stressed, and has depression. I think that was all undiagnosed when we were small and that's come to light. Lots of conversations about that stuff now and how it has affected us. Very, very angry. Lots of anger…I think that was her way of expressing things. Her struggles came out in quite an angry way and often directed at us. I remember being in trouble a lot. I would get smacked a lot. I was quite scared of her because of that side of her personality. I remember being very stressed and very upset as a child. I remember a lot of crying and a lot of sick in your stomach feeling because of the tension in our house. There was a lot of fighting…I think as a child I just would have felt a lot of the time that I was bad and naughty, and I don't know if that's actually true, but I do remember feeling guilty that I was bad as a child. I think I have the capacity to take things on and feel responsible for them. I feel guilty and like ‘Could I have done that better? Is it my fault?'. [HighMPA-4]Mum is terrifying and wonderful at the same time…So, things were sometimes quite frightening when I was a kid…Mum was violent. She suffered quite badly from rages [HighMPA-8]When [Mum] was angry, it was never outwardly expressed. She was a passive aggressive angry person. She'd slam cupboard doors and bang pot lids and you'd stay out of her way until she went and got more normal again. We didn't deal with emotional stuff outwardly at all when I was growing up. [HighMPA-6]Broadly, the developmental etiology suggested by the current findings is consistent with prior research highlighting the role of overcontrolling, neglectful, and critical parenting in the development of EMSs  on the ongoing engagement and enthusiasm for music training in children is well established  Secure attachments to others; (2) Autonomy, competence, and a sense of identity; and (3) Freedom for the child to express valid needs and emotions. These core emotional needs align closely with the content of the five key parenting themes, and further support the impact of a generalized psychological vulnerability as proposed by Barlow  which may assist in elucidating/differentiating more generalized risk factors from early childhood parenting experiences and those that are more specifically associated with the development of MPA.mosborne@unimelb.edu.au.The datasets presented in this article are not readily available because participants of this study were not asked to consent for their data to be shared publicly. Requests to access the datasets should be directed to The studies involving human participants were reviewed and approved by the University of Melbourne Human Ethics Research Committee. The patients/participants provided their written informed consent to participate in this study.JK designed and executed the study, performed the analysis, and wrote the manuscript. SW and MO collaborated in the study design, methodology, analysis, and writing of the paper. All authors contributed to the article and approved the submitted version.