Kendra Joy Massie
BSc., University of Lethbridge, 2002 A Thesis Submitted in Partial Fulfillment of the
Requirements for the Degree of MASTER OF ARTS
in the Devartment of Educational Psychology and Leadership Studies
O Kendra Joy Massie, 2004 University of Victoria
All rights reserved. This thesis may not be reproduced in whole or in part, by photocopy or other means, without the permission of the author.
Supervisor: Dr. Jillian J. Roberts
Abstract
Pectus excavatum (PE) is a congenital deformity that is characterized by a sunken chest. Children with PE often experience embarrassment and shame over their physical
appearance and can undergo the Nuss procedure, a minimally invasive surgery, to correct their deformities. Because a child's characteristics can affect the well-being and
experiences of hisiher parents, one might expect that children with PE would impact the lives and behaviors of their parents. The present study employed phenomenology to investigate the experiences, perceptions, and recommendations of three fathers and six mothers whose children have undergone the Nuss procedure. Interviewers were
conducted, transcribed verbatim, and thematically analyzed. Results indicate parents were most concerned about their child's well-being when hisher pectus became severe and symptomatic, and when helshe recovered fiom surgery. Moreover, many of the children's pediatricians were unaware of the Nuss procedure; as such, parents suggested that
Table of Contents
. .
Abstract
...
ii...
...
Table of Contents ill List of Tables...
vi . . Acknowledgements...
vii...
...
Dedication viii Chapter 1 : Introduction...
1Statement of the Problem
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3Research Purpose
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4Definition of Terms
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4. . . Delimitations
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5Chapter 2: Literature Review
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7Body Image and Well-Being
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7Impact of Disfigurement
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8Child-Parent Influence
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11Impact of Physical Disfigurement on Parents
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12...
Pectus Excavatum 17...
Need for Research 18 Chapter 3: Methodology...
20...
General ApproachIParadigm 20...
Research Design 21 Entering Assumptions...
23 Reflexivity...
24...
Participants 25
Data Collection Methods
...
25Data Collection
...
27...
Data Analysis 28 Rigor...
29Ethical Considerations
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30Chapter Four: Results
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32...
Participant Profile 32 Essential Experience...
33Categorical. Cluster. and Thematic Structures
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36Category One: Pectus and Pre-Surgery Issues
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37Category Two: Surgery. Recovery. and Outcome
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45Category Three: Parental Behavior and Impact
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52Category Four: Healthcare
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58Participant Recommendations
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64...
Research Reflection 65 Chapter 5: Discussion...
68 Summary...
68 Research Contributions...
68Purpose One: Examine Parental Experiences
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69Purpose Two: Examine Parental Perceptions
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73...
Purpose Three: Identify Recommendations 79
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Implications
...
81...
Direction for Research 83 Final Summary...
84References
...
86Appendix A: Interview Guide
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94Appendix B: Informed Consent Form
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95List of Tables
Table 1 Participant Names
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32 Table 2 Categories and Their Respective Clusters...
37 Table 3 Clusters and Supporting Themes for the Category, Pectus, and Pre-Surgery IssuesTable 4 Clusters and Supporting Themes for the Category, Surgery, Recovery, and
Outcome
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46 Table 5 Clusters and Supporting Themes for the Category, Parental Behavior and ImpactAcknowledgements
I must first recognize the mothers and fathers who participated in this study. Your stories and willingness to participate provided me purpose and strength, and I thank you for your encouragement and interest in the study.
I would also like to acknowledge my supervisor, Dr. Jillian Roberts. I am grateful for the direction, support, and encouragement you have provided. Thank you for
demonstrating that one can be a caring and successful academic, psychologist, and mom. Thank you Ryan for your early mentorship; you assisted me in developing research skills that have contributed to my academic success.
I also want to thank Lia, my grad school companion. Your encouragement and guidance helped me develop the vision I needed to complete this degree and thesis.
Dedication
I dedicate this thesis to my parents and sisters. Thank you Mom, Dad, Ranelle, and Crystal your love, support, and interest in my personal and academic endeavors. This thesis was possible because I modeled the hard work and devotion that each of you exhibit daily.
I also dedicate this thesis to my Grandpa Massie. You continue to demonstrate the importance of commitment and family, and I am thankful for your influence and role in my life.
Examining the impact that physical disfigurement has on the lives of children and families has become an area of active research. In particular, researchers have begun to examine the effect that physical deformities have on quality of life, family functioning, adjustment, and psychosocial well-being. Although there are numerous forms and severities of physical disfigurements, research has typically examined the effects of craniofacial deformities on affected children and their families. Few researchers have investigated the effects that less common or visibly apparent conditions such as pectus excavatum (PE), a congenital chest-wall deformity, have on individual and familial well- being and adjustment.
Individuals with visible deformities have appearances that are at odds with
America's social standard of beauty and they often face environments of prejudice and/or rejection (Hill-Beuf & Porter, 1984). Consequently, these individuals often have negative body concepts (MacGregor, Abel, Bryt, Laver, & Weissman, 1953) and they fi-equently experience anxiety, depression, and low self-esteem (Landsdown, Rumsey, Bradbury, Carr, & Partidge, 1997; Thompson & Kent, 2001). Furthermore, these individuals can have high degrees of self observation, motivational problems, and anxiety during social contact (Einsiedel & Clausner, 1999).
Many researchers suggest that factors such as interpersonal relationships and parental attitudes and acceptance affect an individual's self-perception, ability to cope, and psychological and social development (Emery, 200 1 ; Rutter & Rutter, 1992; Werner & Smith, 1982). That is, children use the attitudes and behaviors of others to determine who they are and how to perceive themselves. Furthermore, Belsky (1 984) indicates that
a child's characteristics can make himher more or less difficult to care for and thus, a child's characteristics can affect the quality of hisher parent's care and the amount of care that helshe receives. As such, children with physical deformities who exhibit psychological and social difficulties have unique characteristics and care requirements that can impact parental adjustment and thus, parental well-being. It is important, therefore, that parents are well-adjusted to their role of raising a child with a physical deformity (Rutter & Rutter, 1992).
Researchers such as Barden, Ford, Jensen, Rogers-Salyer, and Salyer (1 989), Bradbury and Hewison (1 994), and Miller, Gordon, Daniele, and Diller (1 992) indicate children with physical deformities can impact the well-being and functioning of their parents. For instance, psychological distresses such as worrying and wondering are two to three times more prevalent in mothers of disabled children than in mothers of non-
disabled children (Miller et al., 1992). Furthermore, parent-child relationships can be negatively affected when the child has a deformity (Barden et al., 1989; Bradbury & Hewison, 1994). Although many researchers have written about the psychological and social consequences that visible deformities (Bradbury & Hewison, 1994) such as cleft lip have on the lives of affected children and their parents, little research has examined the effects of less visibly apparent deformities. In particular, few researchers have examined the effects that PE has on the lives of affected children and their families.
Pectus excavatum is a congenital deformity that is present in one to eight of every 1000 children born and is characterized by a caved-in or sunken chest (Emery, 2001). Children with PE often experience embarrassment and shame over their physical
confidence (Emery, 2001). Thus, children with PE have psychological and social difficulties that present their parents with unique demands and care requirements. Although one might expect that children with PE would drastically affect the lives of their parents, no known researcher has investigated the experiences of such parents. A need exists, therefore, for research that will inquire into the experiences of parents whose children have PE to develop an understanding that will benefit other families who live with PE and guide professionals who work with and support these families. Because children with PE can undergo the Nuss procedure to correct their pectus deformities, research should also investigate what parents experience when determining whether the Nuss procedure will be performed.
Statement of the Problem
Based upon the aforementioned research need, the problem of the study,
expressed as a question, is: What are the experiences, perceptions, and recommendations of parents whose adolescent children have undergone the Nuss procedure to correct their pectus deformities?
Addressing the research problem will develop a comprehensive understanding of the experiences and recommendations of parents whose children have PE and enhance knowledge of how these parents function and perceive and use social support. This enhanced knowledge will contribute valuable information to families affected by PE and the medical and educational communities who support and care for them. That is, by developing a better understanding of the experiences of, and challenges faced by, parents of children with PE, professionals will enhance their ability to develop and provide the support and resources required by these individuals. Furthermore, by acquiring
recommendations and knowledge about what parents experience and consider when they make a decision regarding corrective surgery, medical and helping professionals will be better able to assist and support parents who are in the process of making similar
decisions.
Research Purpose
Given the lack of knowledge regarding the experiences and needs of parents whose children have PE and the need to provide information to guide healthcare
professionals in supporting such individuals, it is necessary to examine and disseminate what parents encounter when they raise a child with PE. The purpose of the present study, therefore, is to develop a comprehe&ive understanding of the experiences, perceptions, and recommendations of parents whose children have corrected pectus deformities. The study will explore what impact a child with PE has on parental
functioning and relationships, and how parents view the availability and effectiveness of social support. The study will also examine what parents experience and consider when deciding whether their child will undergo the Nuss procedure, and inquire into parental recommendations for healthcare professionals and resources.
DeJinition of Terms
Several specialized and uncommon terms will be used in this study. To ensure readers correctly interpret these terms, their working definitions are provided.
Pectus excavatum: a congenital deformity characterized by a depressed sternum that results in a caved-in or sunken chest (Emery, 2001).
Nuss procedure: a minimally invasive surgical technique that places a curved steel bar behind the sternum to force it into shape and correct a pectus excavatum deformity (Emery, 2001).
Ravitch repair: the original and most common procedure used to correct PE that involves removing the ribs that attach to the sternum at the point of depression, breaking and repositioning the sternum, and using a metal strut to fasten the sternum in a correct and outward position (Hu, Feng, Lui, Jiang, Wei, Tang, et al., 2000; Tjan, Semik, Rotering, Rolf, & Scheld, 1999; Williams & Crabbe, 2003).
Delimitations
Like all research, the present study has methodological restrictions that influence its design and outcome. To ensure readers of the research have a framework to
comprehend and appreciate its results, these delimitations must be acknowledged and explicated. The following delimitations will be imposed by the researcher:
1. The study will be limited to nine parents whose children have undergone the Nuss procedure to correct their PE deformities;
2. The study will be limited to parents who live in a Western Canadian city; 3. The study will be limited to parents who are able to participate in a one-on-one
interview;
4. The study will be limited to data gathered from January 2004 to February 2004; 5. The study will be limited to the topics of family functioning, relationships,
perceptions and use of social support, decisions regarding corrective surgery, and healthcare recommendations; and,
6. All variables, conditions, or populations not specified are beyond the scope of the study.
Summary
Chapter one has identified a need for research that will examine the experiences and recommendations of parents whose children have corrected pectus deformities to enhance understanding of what parents of children with correctable physical deformities experience and guide healthcare professionals in supporting these families. This chapter has also identified the study's statement of the problem and purpose, defined relevant terms, and presented the study's delimitations.
Chapter two reviews literature regarding the well-being, functioning, and adjustment of children with physical deformities, and the impact these children have on their parents. Emphasis is placed on the lives of children with PE and the lack of research regarding the experiences of these children's parents.
Chapter 2: Literature Review
Chapter two examines literature pertaining to physical deformity and the impact physical disfigurement has on the lives of children. The influence that children with physical deformities have on their parents is highlighted, as is the well-being of children who have PE and the potential for these children to have a multifaceted effect on their parents.
Body Image and Well-Being
Although body image can be defined as the perception of body appearance (Auchus, Kose, & Allen, 1993) and how one thinks, feels, and acts about hisher physical attributes (Muth & Cash, 1997), Hubert Lacey and Birtchnell(1986) suggest body image is a complex concept that operates on many levels. These researchers elaborate further and suggest neurological disorders, acute dismemberment, and the presence of actual physical deformities can disturb one's body image. Because the appearances of people with visible deformities are at odds with North America's social standard of beauty (Hill- Beuf & Porter, 1984), and society typically rejects those with physical disfigurements (MacGregor et al., 1953), individuals with physical handicaps or visible deformities often face environments of prejudice andlor rejection (Hill-Beuf & Porter, 1984). In fact, Clifford and Walster (1973) have found that attractive children are more likely than unattractive children to be perceived as having positive personality traits and high
scholastic achievement. Furthermore, Cash (1 990) indicates attractive people may receive more help from others, have an advantage in job interviews, and elicit greater social efforts to please. As such, individuals with physical deformities often receive differential treatment and they can be perceived less positively than those without deformities.
Impact of Disfigurement
Cash (1 990) indicates there are two manners in which researchers examine the difficulties faced by individuals with deformities. The first perspective, "view from the outside," is social and cultural in nature and examines how appearance influences social perceptions and interactions. The second perspective, "view from the inside," investigates the impact of appearance on perceptions of self-concept, emotional well-being, and
quality of life.
In an attempt to conceal their deformities, individuals with disfigurements often avoid social settings and interaction (Thompson & Kent, 2001). Elliott, Ziegler, Altman, and Scott (1982) expand on this finding and suggest there are three additional manners in which individuals can react to their handicaps. These reactions include deflecting
attention towards a less discrediting attribute, emphasizing an interest or ability unrelated to the handicap, and remaining apathetic in social situations. Although the personalities of individuals with physical deformities are typically well adjusted, research suggests these individuals often have negative body concepts (MacGregor et al., 1953).
Researchers have also found that individuals with physical anomalies frequently experience psychological distresses including anxiety, depression, and low self-esteem (Landsdown et al., 1997; Thompson & Kent, 2001). Additionally, Einsiedel and Clausner (1 999) found that individuals with deformities have high degrees of self observation, motivational problems, and anxiety during social contact.
Researchers such Liskey-Fitzwater, Moore, and Gurel(1993) and Sarimski (2001) have empirically examined the well-being of individuals who have both visible and less visibly apparent deformities. Sarimski examined the problem behavior of children with
Apert syndrome, a condition characterized by severe craniofacial anomalies. The mothers and/or fathers of 25 children with Apert syndrome assessed their children's behavior for such things as social withdrawal, anxiety, social problems, thought problems, and aggression. Sarimski found that although children with Apert syndrome do not present with severe psychological adjustment problems, they are at risk for developing social problems, social withdrawal, and attention difficulties.
Liskey-Fitzwater and colleagues (1 993) investigated the self-perceptions of individuals with scoliosis, a physical condition in which the spine is curved. Compared to craniofacial deformities, scoliosis is a less visibly apparent deformity because it is
typically covered by clothing. Liskey-Fitzwater and colleagues had female adolescents with and without scoliosis assess numerous issues related to self-perception. These researchers' results indicate that, compared to age-matched controls, adolescents with scoliosis have lower perceived levels of social acceptance, athletic competence, physical appearance, romantic appearance, and self-worth. As such, Liskey-Fitzwater and
colleagues conclude that physical handicaps can affect an individual's psychological and physical well-being.
Although not researched for all types of physical deformities, researchers have found that the visibility or severity of disfigurement and the degree of distress
experienced by those with the disfigurement is not equivocal (Thompson & Kent, 2001). Whereas Knudson-Cooper (1 98 1) found that visibility was not an accurate predictor of psychological morbidity, Williams and Griffiths (1 99 1) report that the visibility of a bum injury was the best predictor of psychological outcome. Furthermore, early studies indicate that the severity of a deformity is not a good predictor of emotional response
(MacGregor et al., 1953; Porter, Beuf, Nordlund, & Lerner, 1979; Wright, 1960). One can deduce, therefore, that individuals with physical deformities are affected by and experience many multifaceted issues.
As children with congenital defects age and enter adolescence, their psychological problems may increase (Einsiedel & Clausner, 1999). Adolescence is a vulnerable period of time for the development of self-image and self-esteem (Forstenzer & Roye, 1988). Crain (2000) indicates adolescence is associated with a period of rapid physical
development and that for many, this growth can cause a sense of identity confusion. As such, adolescents can be extremely concerned about and aware of their appearance, and they may develop identity problems because they are worried that they are not attractive to others. Additionally, Thompson (1 999) reports that adolescents may experience considerable self-consciousness because they assume others scrutinize their appearance to the extent that they do. In particular, female adolescents frequently focus emotional and mental energy on their weight, shape, hair, and skin because they believe they are under increased scrutiny by peers (Forstenzer & Roye, 1988). In examining the development of body dissatisfaction during adolescence, Hargreaves and Tiggemann (2002) found that female body satisfaction worsened over time compared to adolescent males. These researchers conclude that for females, adolescence can be a period of increasing body image disturbance. As such, if adolescents, and particularly female adolescents, have physical defects, they may be less accepted by social networks and may be viewed as, and criticized for being, "visually different." These possibilities enhance the likelihood that adolescents with disfigurements may suffer psychological, social, and behavioral problems.
Many researchers suggest that factors, such as interpersonal relationships and parental attitudes and acceptance, affect an individual's self-perception, ability to cope, and psychological and social development (Emery, 2001; Rutter & Rutter, 1992; Werner
& Smith, 1982). That is, children look to others to determine who they are and how to
perceive themselves. As Forstenzer and Roye (1988) suggest therefore, the degree to which physical perfection is valued by an individual with a physical anomaly, and therefore, hisiher well-being, depends not only on the images of society, but also on parental values. For instance, these authors suggest that female adolescents who feel valued, appreciated, and "pretty" in the eyes of their parents will adjust more easily to a physical defect than children who are frequently criticized about their appearances. It is important, therefore, that parents are well-adjusted to their role of raising a child with a physical deformity (Rutter & Rutter, 1992). Belsky (1984) indicates, however, that a child's characteristics can make himher more or less difficult to care for, and thus, that child characteristics can affect parental care quantity and quality. For instance, a child's temperament andlor behavior can influence the quality and amount of care that helshe receives. As such, children with physical deformities who experience psychological and social difficulties present their parents with unique situations and care requirements that can impact parental adjustment, and thus, parental well-being.
Child-Parent Influence
Research that has investigated the impact a child and hislher deformity has on parents is based on the belief that parenting is a two-way process or a mutual interaction between parent and child whereby parents and children influence the well-being and functioning of each other (Reder & Lucey, 2000). Carter and McGoldrick (1989) describe
this phenomenon as a family life cycle transition in which children, through an evolving process, are brought up in family units where they exhibit behaviors that are influenced by others and impact the behaviors of others. Reder and Lucey further describe the family life cycle transition model and indicate families exist in the context of the external world as they interact with social agencies and structures such as schools, churches, and the law. Consequently, it is through continuous interaction with family members and the external world that parents and children develop throughout their lives.
Based upon the family life cycle transition model, Reder and Lucey (2000) indicate certain events, such as the birth or death of a family member, marriage, and the onset of illness, can affect all family members and require them to adjust their
relationships and how they view themselves. Although some families can manage such events without much difficulty, the ability of others to adjust can be a difficult and stressful process (Reder & Lucey, 2000). Based upon the family life cycle transition model, therefore, the birth of a child who has a birth defect and may develop
psychosocial and behavioral difficulties creates a situation in which the well-being and functioning of parents, and thus children, can be affected in a multitude of ways.
Impact of Physical DisJgurement on Parents
Although parents must adjust when they give birth to a baby with visible disfigurements (Bradbury & Hewison, 1994), Mintzer, Als, Tronick, and Brazelton (1 985) suggest the responsibilities of parenting a child with a defect are similar to those of parenting a typical child. That is, parental responsibilities include exhibiting empathy, learning the child's cues, organizing and expanding alert periods, and supporting the infant. Freud (1 960), however, suggests mothers should not be expected to have the
intuition required to effectively raise children with handicaps, and that without guidance, mothers may feel helpless in raising such children.
Many researchers have explored and reported on the experiences of parents who give birth to a child with a physical defect. Although numerous responses to the arrival of a baby with a physical anomaly have been observed, Bradbury and Hewison (1 994) indicate early responses are similar to the grief response as parents initially experience denial, anger, distress, and potentially, rejection or over-protection of the child. Mintzer et al. (1985) suggest reactions to a child born with a defect are influenced by seven intricate factors:
1) The severity and correctability of the defect;
2) Prenatal expectations for the child and their perceptions of themselves as parents; 3) Prior means of coping with stress;
4) The marital relationship and their own experiences in the parent-child relationship;
5) The infant's temperament;
6) The short- and long-range implications of the defect and its impact on the child and family; and,
7) The role of environmental supports.
This information suggests responses to the birth of a child with a deformity are dependent upon numerous factors, and therefore, that parental responses may differ.
As indicated, parents who give birth to a child with a physical defect go through a series of reactions that are similar to the grief process. Solnit and Stark (1 96 1) report that grief is the response to the loss of a valued person, possession, status, ideal, part of the
body, and so on that follows a consistent course and is modified mainly by the abruptness of the loss, the preparation for the event, and the significance of the lost object. In their review, Solnit and Stark describe the grief or mourning reaction that parents experience after giving birth to a child with a deformity. The first reaction parents experience is numbness and disbelief. These reactions occur when the parents realize their wishes and expectations for their desired and perfect baby did not occur, and the anxious fears of having a damaged child are realized. This mourning process leads to the awareness of feelings of loss and disappointment and finally, to the last phase of the grief process where the intense memories and expectations for an idealized child begin to dissipate.
According to Mintzer and colleagues (1 985), the reaction and adaptation to the birth of a child with a physical anomaly consists of three stages. In the first stage, parents often experience a state of shock and disappointed. The second stage can be a period of lowered self-esteem, painful feelings, inadequate defenses, and at times, inappropriate handling of the infant. The third stage includes renewed self-esteem, increased awareness of existing feelings, and more appropriate meeting of the infant's and their own needs. The process of adaptation, however, is not a smooth process and in the first two years of a child's life, parents experience concerns regarding the outcome of surgical procedures, the consequences of their child's defect, and how they will assist their child in
understanding and adjusting to hisher defect (Mintzer et al., 1 985).
Numerous researchers have examined the impact children with physical defects have on their parents. In investigating the influence children with anomalies have on parenting practices and attachment, Clements and Barnett (2002) found that the visibility of the child's defect was the most important variable in the prediction of parental quality.
That is, parental quality was higher when the children's physical anomalies were more apparent. These authors propose that when disfigurements are visible, parents exhibit more sensitivity, awareness, and caregiving behaviors because they believe their behavior will facilitate their child's well-being. Weiss (1998) also found that the visibility of a defect affected parental characteristics. Specifically, this researcher found parents were more bothered by more external and openly visible impairments than those that were internal and disguised.
Barden (1 990) indicates children with physical anomalies can elicit stress within a family. Although parents of children with deformities must deal with problems faced by all parents, they must also deal with problems that are specific to the child's physical deformity (Harvey & Greenway, 1982). These problems include their child being the object of ridicule, taking their child to the hospital, and monitoring their child's physical, mental, and social development. Furthermore, as a child ages, parents are often required to confront issues related to surgical correction and problems or complications that may result from the defect (Barden, 1990; Lynch, 1989).
Miller and colleagues (1 992) report that distress is two to three times more prevalent in mothers of disabled children than in mothers of non-disabled children. For instance, these mothers may be more concerned about their child's well-being than typical parents. Additionally, many researchers have demonstrated that parent-child relationships are negatively affected when a child has a deformity (Barden et al., 1989; Bradbury & Hewison, 1994). Barden and colleagues found that, compared to mothers of children without deformities, mothers of infants with facial deformities display less nurturant behavior, engage in less tactile-kinesthetic stimulation, touch their infants
affectionately less often, and spend less time demonstrating a toy. Concurrently, these results suggest infant attractiveness is an important predictor of the quality of mother- infant interactions (Barden et al., 1989).
Although research has examined how children with defects can affect the well- being and functioning of parents, research has also investigated how parents can impact the lives of their children. For instance, Fishman and Fishman (1 97 1) examined how maternal attitudes and behaviors impact the self-esteem and adjustment of children with physical defects. The participants in their study were mothers and their children who were between eight and 14 years old and had birth defects such as spina bifida, cleR lip and palate, and hydrocephalus. By employing semi-structured interviews and
questionnaires, Fishman and Fishman found that several maternal variables were
positively correlated with the children's self-esteem, likeability, and involvement in, and enthusiasm regarding, day-to-day activities. These variables included the degree to which the mother was concerned with, and actively sought information about, the child's
condition, the quality of the mother's understanding of her child's condition, the
frequency with which the mother and child communicated about the child's thoughts and feelings, and the extent to which the mother was sensitive and responsive to these
communications. Additional variables were the extent to which the mother appeared to like, value, and respect her child, and the extent to which the mother had developed a positive and accepting attitude towards the child's future. Although these results are not current, they clearly indicate that the behavior, beliefs, and attitudes of parents can affect the well-being of their children.
Despite the numerous stressors, challenges, and obstacles faced by parents whose children have physical defects, most families adapt to their situations (Barden, 1990). Trout (1 983), however, suggests researchers must study families over time to understand if and how a child with a physical handicap is integrated into a family. Furthermore, although many researchers have written about the psychological and social consequences of visible deformities (Bradbury & Hewison, 1994) on the lives of affected children and their parents, few researchers have examined the effects of deformities that are not always visible. In particular, few researchers have examined the effect that PE has on the lives of affected children and their families.
Pectus Excavatum
Pectus excavatum is the most common congenital chest-wall deformity as it is present in one to eight of every 1000 children born (Emery, 2001). The sternum of individuals with PE is depressed, resulting in a caved-in or sunken chest (Emery, 2001) and occasionally, cardiopulmonary dysfunction (Haller & Loughlin, 2000), limited endurance, and heart murmurs (Comarow, 2002). Researchers indicate many children with pectus deformities experience embarrassment and shame over their physical appearances (Roberts et al., 2003) and have negative self-images and little self- confidence (Emery, 2001). Furthermore, Einsiedel and Clausner (1 999) found that individuals with PE have high degrees of self observation, motivational problems, anxiety regarding social contact, and depression. Research clearly indicates, therefore, that PE can impact all areas of an affected child's life (Emery, 2001) and like those with other physical impairments, children with PE can have psychological and social
Currently, there are two surgical procedures that individuals with PE can undergo to correct their deformities. The original and most common procedure is the Ravitch Repair. This surgery includes removing the ribs that attach to the sternum at the point of depression, breaking and repositioning the sternum, and using a metal strut to fasten the sternum in a correct and outward position (Hu et al., 2000; Tjan et al., 1999; Williams & Crabbe, 2003). The Ravitch Repair is frequently associated with significant blood loss, a substantial incision that results in an obvious scar, and significant postoperative pain (Wheeler & Foote, 2000).
The Nuss procedure was introduced in 1987 as an alternative corrective procedure. The Nuss procedure is a minimally invasive procedure in which 2.5cm incisions are made on each side of the chest cavity, a curved steel bar is inserted through the thoracic cavity, and the bar is rotated to force out the concavity (Nuss, Kelly,
Croitoru, Katz, 1998; Wheeler & Foote, 2000). After approximately two years, the bar is removed from the chest. Although postoperative pain is significant, there is substantially less scarring (Coln, Gunning, Ramsay, Swygert, & Vera, 2002; Leonard, n.d.; Nuss et al., 1998; Wheeler & Foote, 2000; Williams & Crabbe, 2003). Moreover, researchers such as Lawson, Cash, Akers, Vasser, Burke, Tabangin, et al. (2003) and Roberts and colleagues (2003) have found that individuals who undergo the Nuss procedure experience enhanced self-confidence and psychosocial well-being.
Need for Research
An extensive literature review indicates that although researchers have initiated study into the psychological and social well-being of children who have PE, no located studies examine how parents of children with PE are affected by their child's deformity
and psychosocial well-being. A strong need exists, therefore, for a foundational study that will examine how the lives of parents are impacted when their child has PE. Because children often exhibit improved physical and psychosocial well-being when their pectus deformities are corrected by the Nuss procedure (Lawson et al., 2003; Roberts et al., 2003), research must also investigate how parents, together with their children, decide whether the procedure is performed. Furthermore, because social support is related to parental functioning (Belsky, 1984) and a lack of social support has been shown to be a stressful factor associated with caring for a disabled child (Bristol, 1979), research must also examine parental perceptions regarding the availability and effectiveness of social support. Such research will develop a comprehensive understanding of what parents of children with PE experience, and contribute valuable information to families who live with PE and the medical and educational communities who encounter and work with them.
Summary
Chapter two has examined literature concerning physical deformity, the impact children with physical deformities have on parents, and the well-being of children with PE. The chapter emphasized the need to investigate how children with PE influence the lives of their parents to enhance understanding in the areas of physical disfigurement and parent and child well-being and to provide information that will guide professionals who work with and support these families.
Chapter three will provide an overview of the study's methodology, highlighting the research approach and design, participants, data collection methods, and procedures for data collection and analysis.
Chapter 3: Methodology
Chapter three outlines the methodology of the study that seeks to answer the question, what are the experiences, perceptions, and recommendations of parents whose adolescent children have undergone the Nuss procedure to correct their pectus
deformities? An overview of the research approach and design, participants, data collection methods, and procedures for data collection and analysis is provided.
General Approach/Paradigm
The purpose of the study is to develop a comprehensive understanding of the experiences of parents whose children have PE. A qualitative approach will be employed as qualitative research seeks to "interpret, understand, explain, and bring meaning" (Anderson & Arsenault, 1998, p. 1 19) to experiences or phenomena. In qualitative investigations, researchers are key data collectors who assume "that a profound
understanding of the world can be gained through conversation and observation in natural settings rather than through experimental manipulation under artificial conditions"
(Anderson & Arsenault, 1998, p. 1 19). Thus, qualitative researchers use multiple perspectives or participants to examine phenomena in natural settings and they employ methods of analysis to bring meaning to these experiences (Anderson & Arsenault,
1998).
Although the quality of qualitative research is dependent on such things as the participant's ability to articulate their experiences and the skills of the researcher, qualitative research can be highly rigorous. That is, qualitative researchers can use inductive inquiry and their understanding of the research environment to produce rich, useful, and valid research findings (Anderson & Arsenault, 1998).
Research Design
To develop a comprehensive understanding of the experiences of parents whose children have corrected pectus deformities, phenomenology was employed.
Phenomenology is a qualitative method that focuses on the meaning of specific human experiences (Polkinghorne, 1989). Phenomenological researchers assume perceptions provide evidence of the world as it has been lived, and they believe they are responsible for understanding the essence of how people attend to the world (Morse & Richards, 2002). To understand the meaning or essence of an experience, phenomenologists collect nake descriptions of the experiences they are investigating (Valle & Halling, 1989). Unlike other qualitative researchers who may provide descriptions of actions or behaviors, phenomenologists develop clear, precise, and systematic descriptions of specific human experiences (Polkinghorne, 1989).
There are numerous phenomenological orientations that are based on specific themes and characterized by certain ways of thinking. van Manen (2002) indicates that phenomenological research can be performed by professional philosophers and
professional practitioners. Professional philosophers are interested in the philosophy of phenomenological method and in philosophical topics, themes, and issues that stem from the works of leading phenomenologists (van Manen, 2002). Such researchers may
assume orientations such as existential and hermeneutical phenomenology. Unlike philosophers who conduct phenomenological research, professional practitioners are more interested in the practice and application of phenomenology (van Manen, 2002). That is, such researchers employ phenomenology in applied or professional settings (van Manen, 2002). Researchers who are concerned with the practice and application of
phenomenological methods assume a phenomenology of practice or applied
phenomenology orientation (van Manen, 2002). Because the purpose of the present study is to provide a description of parental experiences and recommendations for healthcare professionals and to do so, the researcher used phenomenological methods in a context of practical concerns of everyday living, the researcher has assumed an applied
phenomenology orientation.
Research participants in phenomenological studies are chosen individuals who have had the experience under investigation and are able to provide rich descriptions of the experience (Polkinghorne, 1989). To gather descriptions from their participants, phenomenological researchers utilize semi-structured interviews. According to
Moustakas (1 994), phenomenological interviews are informal and interactive processes that employ open-ended comments and questions. Furthermore, Polkinghorne reports interviews are theme-oriented conversations in which interviewers seek descriptions of an experience by encouraging participants to share details of the experience under
examination. Phenomenologists use the descriptions provided in an interview to identify and develop a description of an experience's essential features (Polkinghorne, 1989). That is, phenomenological researchers analyze their participants' descriptions to identify the constituents or common elements that make the experience what it is (Polkinghorne,
1989). Using the essential features, phenomenologists produce a description of the structural relationship that exists between the elements that constitute the experience under investigation (Polkinghorne, 1989). More simply, these researchers produce a report that provides clear, precise, and systematic descriptions of the examined experience (Polkinghorne, 1989).
Entering Assumptions
Colaizzi (1978) indicates that to completely understand a phenomenon or participant's account, researchers must suspend the presuppositions and preconceptions they have about the phenomenon they are investigating. Polkinghorne (1 989) indicates self-reflection is a preparatory step to data collection in which a researcher identifies and records hisher presuppositions and biases. Furthermore, Moustakas (1 994) proposes that bracketing encourages researchers to approach their research with a naivetk and openness that is free of their own beliefs, knowledge, and prior experiences. Researchers, therefore, engage in bracketing to identify their preconceived thoughts and expectations so that they (the thoughts and expectations) will not influence data collection and analysis
(Polkinghorne, 1989). Additionally, when a researcher brackets or suspends their beliefs, they are able to listen openly to their participants' experiences. As such, by identifying and suspending their beliefs, knowledge, and expectations, researchers enhance their ability to obtain and present valid accounts of their participants' experiences. To add rigor to the study, the researcher has considered her assumptions and they are:
1. The participants will be honest in their accounts;
2. The participants will carry out the instructions provided by the researcher;
3. I believe the experiences of parents whose children have PE must be examined so the lives and experiences of similar parents can be improved;
4. A review of the literature indicates parents whose children have physical deformities experience a variety of stressors, demands, and responsibilities that they are able to effectively or ineffectively manage. I am prepared to conduct a
thorough examination of the lived experiences of parents whose children have PE with an open mind; and,
5. The perspectives of the participants will be seen as equal in value to other experts. To identifj those thoughts or expectations that may impose into data collection and analysis, the researcher has also considered her reasons for conducting the present investigation. These considerations are detailed below.
In the past several years, I have become aware of and concerned about the number of individuals that live with congenital and/or chronic illness and/or disability. Although I do not want to assume a medical role to treat these individual's diseases or disabilities, I have developed a desire to support their psychological well-being and the well-being of their family members. Although numerous researchers are actively investigating how illness or disability affects children, the parents and siblings of these individuals are frequently disregarded or forgotten about. For instance, researchers have initiated research into how children are affected by PE, but failed to examine how these children's parents may also be affected. As such, I conducted the present study to provide the parents of children with PE a voice that will be heard by researchers and medical professionals. It is my hope that by conducting the present investigation, the lives of families that live or will live with PE will be optimized.
Rejlexivily
Horsburgh (2003) reports researchers have the ability to influence the research process and their data, and by engaging in reflexivity or self-reflection, the researcher admits that neutrality and detachment from data collection and analysis is impossible. For
instance, Rossman and Rallis (2003) indicate that a participant's words and actions trigger a researcher's thoughts, hypotheses, and understandings of the setting and participants. Researchers such as Horsburgh suggest engaging in reflexivity or self- reflection adds rigor to qualitative research. That is, researchers who are reflexive add thoroughness and validity to their research. Furthermore, by engaging in reflexivity, researchers acknowledge that their actions and decisions can impact the meaning and context of an investigated experience (Horsburgh, 2003). A section of chapter four is devoted to the researcher's reflection of her research, participants, and findings.
Participants
To ensure a variety of familial characteristics and experiences were represented by the participants, parents were purposefully selected from families who volunteered to participate in a larger, longitudinal study. Thus, criterion sampling was employed. The mothers andlor fathers of six adolescent children who had undergone the Nuss procedure participated in the study. Specifically, three Caucasian fathers and six Caucasian mothers of four boys and two girls participated. Each parent came from two parent families and each family determined which parent(s) would participate. The parents' children were between the ages of 12 and 17 at the time of the interviews. All participant families resided in a Western Canadian city.
Data Collection Methods
To gain descriptions of the experience under investigation, each parent
participated in an in-depth and semi-structured interview. The interviews ranged in time from approximately 25 to 70 minutes. Interviews that consist of open-ended questions and comments enable researchers to develop comprehensive understandings of their
participants' experiences (Roberts & Cairns, 1999). During the interviews, therefore, the researcher asked a variety of open-ended questions to evoke comprehensive accounts of the participants' experiences. Each interview began with a brief description of the study's purpose and the interview question: What is it like to be the parent(s) of a child who has PE? Each interview proceeded with follow-up questions that were informed by Cash's (1 990) description of how individuals with disfigurements have typically been examined. The follow-up questions, however, also addressed parental functioning, family life, and the decision to pursue the Nuss Procedure. Follow-up questions were posed to ensure that a comprehensive description of the experiences was obtained and that a free flow of ideas occurred. If an interview question was unclear to a parent, the question was asked in a scaled-down or simplified manner.
As indicated, several of the interview's follow-up questions were based on Cash's (1 990) description of how individuals with physical deformities have frequently been investigated. Cash indicates that researchers typically examine how appearance
influences social perceptions and interactions ("view from the outside,") and the impact that appearance has on self-concept, emotional well-being, and quality of life ("view from the inside.") Although researchers have examined these factors in individuals with physical deformities, research indicates that when parents have children with physical deformities, their social perceptions and interactions, self-concept, and quality of life can be impacted. Consequently, Cash's "views" can guide an examination of the experiences of parents whose children have PE. As such, several of the interview's follow-up
questions inquired about perceptions of social support and the impact a child and hisher deformity has on parental functioning and relationships. To conclude each interview,
parents were asked to provide recommendations for healthcare and helping professionals. Open questions were also posed to enable the parents to communicate anything about their experiences (see Appendix A for the interview guide).
Data Collection
As previously indicated, the study's participants were selected from families who volunteered to participate in a longitudinal investigation. These families, therefore, were expecting periodic contact from the research team. Potential participants received a telephone call from the researcher. During the conversation, the researcher introduced herself, explained her reason for calling, and invited each parent to participate in her study. If the parent(s) agreed to participate, a face-to-face meeting in either the family's home or a hospital conference room was scheduled.
At the start of each meeting, the researcher outlined and had each parent sign the informed consent form (Appendix B). Each participant family received a signed copy of the consent form. Interviews were tape-recorded and they proceeded as outlined by Appendix A. After each interview, the parents were thanked and told that an interview transcript would be mailed to them for their verification. The participants were also informed that a second telephone interview would take place approximately one week after they received the transcript. The purpose for the second interview was described. Each interview was transcribed verbatim and thematically analyzed (stages one through three as described below). A copy of each interview transcript and the initial analysis was be returned to the participant(s). Approximately 10 days after a participant family received their transcript, the researcher called the participant(s) to conduct a second interview. During this interview, the researcher asked if there was anything that
the participant(s) wanted to add to or change in the transcript. Moreover, the
participant(s) were asked to validate the accuracy of the transcript and initial analysis. Following participant verification and researcher modification of the transcript, data analysis was completed. The results of data analysis (Chapter 4) were e-mailed to the participants for final validation.
Data Analysis
Each interview was transcribed verbatim and thematically analyzed using Colaizzi's (1 978) method that has been modified and informed by Roberts and Cairns (1999). That is, each interview transcript was analyzed in five different stages. The first stage involved listening to and reading each interview to acquire a sense of the meaning of each participant's experience. In the second stage, the researcher identified phrases that highlighted important details and pertained to a participant's experience. The third stage involved assigning theme words to the extracted phrases or sentences. That is, the researcher assigned words that reflected the essential point or meaning of the
participant's words. To ensure the transcripts and thematic representations reflected the participants' experiences and descriptions, they were returned to their respective participants.
During a second interview that was conducted over the telephone, each participant validated the accuracy of the transcript and thematic representations. No participant requested that changes be made to their transcript or analysis; as such, no modifications were made. Subsequent to participant validation of the transcripts and thematic
representations, the researcher proceeded to stage four. This stage included grouping the themes into theme clusters. Each theme cluster encompassed, reflected, and categorized
similar themes. Theme clusters were only formulated from themes that were common to four, five, or all of the six participant families. The final stage of data analysis entailed grouping similar clusters to form categories that encompassed, reflected, and categorized these clusters.
Once the data were analyzed, the thematic, cluster, and categorical representations were reviewed by the researcher's advisor. This step enabled the researcher to attain a second perspective on the validity of the analysis of the data. The categories and their respective clusters and themes were then presented in tables. The researcher used the thematic, cluster, and categorical representations to reveal the structure and logic of the experience under investigation (Polkinghorne, 1989). The researcher also developed a clear and systematic description of the examined experience (Polkinghorne, 1989) by detailing the participants' essential experience.
Rigor
Establishing and maintaining rigor is a crucial element of high quality research (Mill & Ogilvie, 2003), and Richards (2002) suggests a rigorous study is thorough and purposively complete. Mays and Pope (1 995) propose that rigorous qualitative studies have systematic research designs, data collections, data interpretations, and data presentations. Moreover, to perform rigorous research, qualitative researchers should strive to accomplish two tasks: 1. to create an account of method and data that can stand independently such that other researchers can analyze the data the same way and draw essentially the same conclusions and, 2. to develop a plausible and coherent explanation of the examined phenomenon. Researchers ( e g Barbour, 2000; Mays & Pope, 1995; Richards, 2002) present many methods and strategies qualitative researchers can
implement to conduct a rigorous investigation. For instance, to enhance reliability, researchers can, prior to analysis, select the interpretative procedures that will be employed. Moreover, researchers can engage in multiple coding whereby multiple researchers code or analyze data and the resulting analyses are compared for similarity. To enhance validity, researchers can ask participants to validate that the analyzed data or research findings provide a reasonable account of their experiences.
Based upon the aforementioned information, the researcher has designed and conducted the present examination to be a rigorous investigation. Specifically, the researcher selected and implemented a method of data analysis prior to data collection, the researcher's academic supervisor reviewed the results of data analysis, and the researcher's participants verified that the thematic representations and study's results reflected their experiences. Moreover, the researcher selected a group of participants that possessed characteristics and lived in circumstances that were relevant to the
phenomenon under investigation (Mays & Pope, 1995). Lastly, the researcher minimized her bias by ensuring there was a rigid distinction between the results and discussion of her study, and she supplemented the results of the qualitative analysis with a quantitative summary of the results (Mays & Pope, 1 995).
Ethical Considerations
As indicated, the present study is part of a larger, longitudinal study that is being conducted at the University of Victoria. Ethical approval for the longitudinal study and thus, the present study, was obtained from the University of Victoria (Appendix C).
All participants were informed that their participation was voluntary and that they could withdraw from the study at any time. The participants were also advised that their
anonymity would be ensured as no names would be attached to their data. All records were contained in a locked cabinet throughout the duration of the study and they will be destroyed after five years.
Summary
Chapter three has provided an overview of the qualitative approach and
phenomenological design that was employed to answer the research question, what are the experiences, perceptions, and recommendations of parents whose adolescent children have undergone the Nuss Procedure to correct their pectus deformities? This chapter also outlined the participants, data collection methods, and procedures for data collection and analysis.
Chapter four outlines the results of data analysis. Particularly, the essential structure of the experience under investigation is detailed, and an overview of the thematic, cluster, and categorical representations is provided. Moreover, participant quotations are provided to convey the meaning of the participants' experience, and the parents7 recommendations are presented.
Chapter Four: Results
Chapter four presents the results of data analysis. Specifically, this chapter offers a brief description of the participants, an outline of their essential experience, a
description of the thematic, cluster, and categorical structures that compose this experience, and an overview of the participants' recommendations. The chapter concludes with the researcher's reflection of her research. As such, the chapter is organized into five parts: participant profile, essential structure, an overview of the categories and their respective clusters and themes, participant recommendations, and research reflection. Direct participant quotes are presented in the third section to convey the meaning of the examined experience.
Participant Profile
The names of the participants and the names and ages of their children are presented in Table 1. Pseudonyms are used to protect the identity of each participant. Dr. Alton is a pseudonym for the participants7 surgeon.
Table 1 Participant Names
Participant Child's Child's Age
Family Name (years)
Ann and John Marie 15
Leanne Dawn 17
Lee Dave 14
Gail and Paul Ray 17 Debby and Ken William 13
Essential Experience
The present investigation focused on the experiences and perceptions of six participant families. Interviews were conducted with nine parents whose children had undergone the Nuss procedure to correct their pectus deformities to identify how these parents were affected by their child's experiences and how they perceive their child's encounters, behaviors, and characteristics. One purpose of the present study, therefore, was to inquire into and describe the participants' fundamental experience of parenting a child who underwent the Nuss procedure to correct hisher PE.
The essential experience of parents whose children have undergone the Nuss procedure to surgically correct their PE is characterized by events that occur at three time periods: life before surgery, life during recovery, and life after surgery. By detailing the experiences that occur during each time period, an accurate description of the parental experience results.
Parents frequently spoke of how their child's pectus progressed. In the time period before surgery, pectus was both a non-issue and a major concern. Many parents indicated that when their child was young, hislher pectus was not a source of concern and that it did not affect himher. Furthermore, numerous parents were unaware that their child's pectus could increase in severity and impact hisher physical well-being. Many parents, however, said that throughout their child's life, they asked their general
practitioners and pediatricians about whether pectus could be corrected. Whereas many parents were told that no surgery was available, others were told about the Ravitch repair. The majority of parents spoke of how the Ravitch repair was not an option for their child.
The children's pectus deformities became more severe when they entered puberty. Parents frequently detailed how their child began to complain of pain and discomfort and how their child became more concerned about hislher appearance. Moreover, the parents often described how these situations caused them to be concerned about their child's well-being. Once it was apparent that their child's pectus was affecting hisker well-being and they became worried about the implications of hisher pectus, parents began to more actively inquire into whether pectus could be surgically corrected. Whereas some parents learned of the Nuss procedure through medical professionals, others were informed by fi-iends and family. Each parent described how they actively sought information to learn about the Nuss procedure. Whereas many parents consulted with other people, some parents enhanced their knowledge by accessing resources such as those on the internet.
Many parents detailed how they included their child when they considered whether they would pursue the Nuss procedure. Several parents indicated that the decision to undergo surgery was made with ease and that once the decision was made, progression towards surgery was quick and uneventful. Furthermore, parents spoke of the surgeon's caring, approachable, and honest nature and how these attributes made the decision to pursue surgery much easier.
Throughout the interviews, the parents often focused on what life was like for them and their child when helshe was in the hospital and recovering from surgery. Each participant family detailed difficulties such as pain and nausea that their child
encountered following surgery. Parents also described how their child managed hisker encounters in the hospital. Moreover, each participant family spoke of the care their child received in the hospital. Specifically, parents indicated that their child's nurses and
doctors were attentive and willing to answer questions and do everything possible to ensure their child's pain was managed effectively.
When detailing their child's experiences, parents discussed what they encountered and how their child's experiences impacted them. Many parents described how they ensured one parent was always with their child and how they were involved in hisker medical care. Additionally, the parents frequently spoke of how it was stressful to watch their child in pain as helshe recovered from surgery. Moreover, many talked about how their child's condition and experience following surgery was unexpected and that they had an unanticipated reaction to their child's situation.
Each of the participant families spoke of the support they did or did not receive in the hospital. Whereas some families spoke of receiving beneficial and much needed support, others indicated the support they needed was not provided. Still other parents conveyed that they did not require support, but believed the support was there if they needed it.
Participants also discussed what they and their child experienced after returning home from the hospital. Particularly, parents spoke of the difficulties and limitations their child experienced in the weeks following surgery, and how their child's pain and '
discomfort often limited hisher activities. Several parents detailed how they were more protective of, and concerned about, their child and hisher well-being. Parents also focused discussion on their child's current thoughts, appearance, and overall well-being. Many parents indicated that although their child's chest is not perfect, they and their child are happy with the outcome of surgery. Whereas many parents have seen positive
surgery, some parents are discouraged by the pain and fatigue their child continues to experience.
Despite discrepancies in the details provided by each participant, the participants' descriptions conveyed information that constituted the essential parental experience. This experience is defined by three life periods and consists of various issues related to pectus, surgical correction, recovery, and how parents perceive and are affected by their child's experiences and attributes.
Categorical, Cluster, and Thematic Structures
Data analysis revealed four categories: 1) pectus and pre-surgery issues, 2) surgery, recovery, and outcome, 3) parental behavior and impact, and 4) healthcare (Table 2). Whereas the first three categories are composed of three clusters, the last category is composed of one theme and one cluster. Tables 3,4,5, and 6 present the themes that compose each category's clusters and the percentage of participant families that described each theme. Each category and its respective clusters are described below.
Table 2 Categories and Their Respective Clusters Categories
Pectus and Pre- Surgery, Recovery, Parental Behavior Healthcare Surgery Issues and Outcome and Impact
Clusters Within Each Caterrorv
Pectus Child's Surgical Parental Support (Theme)
Experience Involvement
Pre-Surgery Recovery Hospital Attributes and
Experience and Behavior Impact
Surgery Child Post-Surgery Post-Surgery
Progression Actions and
Thoughts
Category One: Pectus and Pre-Surgery Issues
Pectus excavatum is a deformity that was present in each participant's child at birth. This deformity however, did not noticeably affect the lives of the parents and their child until the child entered puberty and the deformity became more severe and
symptomatic. The category pectus and pre-surgery issues is comprised of three clusters: 1) pectus, 2) pre-surgery, and 3) surgery progression (Table 3). Although the clusters are distinct and describe issues pertinent to specific times and events of the participant families7 lives, when mutually considered, they portray what life was like prior to surgical correction.
Table 3 Clusters and Supporting Themes for the Category, Pectus and Pre-Surgery Issues (% = percentage of participant families who addressed each theme)
Pectus and Pre-Surrrerv Issues
Pectus Pre-Surgery Surgery Progression
Themes Within Each Cluster Pectus Discovery (83%) Pectus a Non-Issue (67%) Pectus Progression (67%) Severity of Pectus (83%) Appearance Concern (83%) Physical and Developmental Concern (83%) Previous Medical Conditions (67%) Child's Behavior Before Surgery (67%) Discovery of Surgical Correction (67%) Surgical Options (1 00%) Education About Surgical Correction (1 00%) Surgical Decision (67%) Moving Towards Surgery (1 00%)
Cluster One: Pectus. Throughout their interviews, the participants described the pectus issues that they and their children encountered and managed prior to surgical inquisition. These issues centered around four themes: 1) pectus discovery, 2) pectus a non-issue, 3) pectus progression, and 4) severity of pectus (Table 3). Each theme addresses issues specific to PE and how this deformity affected or did not impact the lives of the participants and their children.
The participants' children were born with a visible yet non-severe chest deformity. Although all of the participants were aware that their child looked different because helshe had an indent in hisiher chest, some of the participants were unaware of
the deformity's name and what its implications could be. As Lee describes, many participants were unconcerned about their child's deformity as they believed the deformity would not negatively impact hisher well-being:
When he was born, we thought it was just a cute little divot in his chest which we used to call his little soup bowl. And [as he grew]
. . .
we were told that [pectus] was the name of it, and ah, and that really um, nothing would ever, um come of it it probably wouldn't get worse...
Many of the parents also indicated their young child adapted well to hisher deformity and that the deformity did not affect himiher. In the early years, therefore, pectus was a not major issue or source of concern for the parents or children.
Participants indicated that as their children approached and began puberty, their pectus deformities became more severe and they began to develop symptoms including pain and discomfort. As represented by Ann's comment, many participants were shocked to discover how severe their child's pectus had become:
When she hit the growth spurt she said "mom, my ribcage is sticking out." And our course at that age I'm not looking at her, on a regular basis as when she was a child. You know? And, I I of course didn't notice the change and then, she
showed me and I my eyes nearly popped..
.
This comment conveys that pectus was often an invisible deformity that frequently progressed and increased in severity unexpectedly. The progression of the pectus to a severe and symptomatic deformity led many parents to inquire into surgical correction.
Cluster Two: Pre-surgery. Four themes comprise the cluster, pre-surgery: 1) appearance concern, 2) physical and developmental concern, 3) previous medical
