Cultural Considerations for Autonomy and Medical Adherence in a Young Palestinian American Muslim Female With Spina Bifida: A Longitudinal Case Study in a Research Context

Abstract

This case study examines the unique presentation of a young Palestinian-American Muslim female, who is a part of an ongoing longitudinal study examining family and peer relationships, psychological adjustment, and neuropsychological functioning in youth with spina bifida. Throughout ten years of data collection, Palestinian-Arab culture and Islamic faith have consistently emerged as important factors that can be considered when interpreting this participant’s general level of autonomy, medical autonomy, medical adherence, and psychological adjustment. This case study examines important aspects of adaptive and independent functioning for youth with spina bifida and how this family’s culture interacts with these different domains of functioning. Moreover, it explores potential conflicts between an Arab collectivist family structure and Western biomedical ethics and suggests how clinicians might navigate these conflicts and bolster culturally-rooted strengths of families and patients from non-Western backgrounds.

Implications for Impact Statement

This case study examines the unique presentation of a young Palestinian-American Muslim female with spina bifida myelomeningocele using data gathered from a longitudinal research study. Specifically, this case study explores how the participant’s faith and culture interact with different areas of adaptive functioning. This case study hopes to highlight potential conflicts that could arise between clinicians and patients due to differences between an Arab collectivist family structure and Western biomedical ethics.

When asked how her family has managed her daughter’s spina bifida, Amineh Fadel^{1 1}responded: “We handle it all with faith. It is a faith-based home that handles all the good, the bad, the ugly, with saying ‘thank you God, we believe in you and please help us to cope.’ That strong faith takes us from day one to day two, [receiving] that much love helps us to continue. If you know the history of what she has been through and to see her the way she is today—there is God who helped her. [And] we all took part, my family, my mom, my sister, my husband; we all took care of the family, to protect it. That’s what others lack.” Farah Fadel is a 19-year-old Palestinian-American female with myelomeningocele spina bifida (SB). Farah lives with her mother, father, younger brother (age 17), and older sister (age 22) in a middle class suburb of a Midwestern city. Farah is a first generation American and a practicing Muslim. Her parents, both Palestinian, emigrated from two Middle Eastern countries in their youth (Amineh, her mother, from Jordan at five years old, and Mahlik, her father, from Kuwait at 17 years old). Both parents received college degrees, and her father is a store manager.

Farah has demonstrated extraordinary resilience in response to her chronic condition. Indeed, when speaking with this young adult, one might not expect to hear that, before the age of five, Farah underwent eight surgeries: five shunt revisions, two foot surgeries, and a tethered cord release. During multiple research-related home visits, culture emerged as an important factor when attempting to understand the resilient nature of this young adult. Moreover, culture-related themes also emerged as important factors to consider with regard to understanding her general level of autonomy and medical autonomy, critical issues for an individual with SB during emerging adulthood and during the transition from pediatric to adult healthcare. The most prominent themes included collectivist care and family structure, Arab parenting styles, and the differential attainment of autonomy. Some of the family and medical autonomy patterns presented in this case are common in families of children with SB across cultural contexts; others, however, are probably best understood within the frame of Islamic and Palestinian-Arab culture. This study presents data from both the Fadel family and a larger SB research sample described below (Devine et al., 2012; Holbein et al., 2015). Using quantitative data, this study will draw comparisons between Farah and her family and the larger research sample. This study will also demonstrate how quantitative data cannot always fully capture the true experience of an individual or family. Therefore, this study also presents qualitative data to enrich our understanding of this young adult and aid our efforts to understand how her presentation compares to that of the larger research sample. By weaving these two sources of data together the following case study will examine how culture—specifically faith and ethnicity—have influenced Farah’s general autonomy, medical autonomy, medical adherence, and psychological adjustment.

Spina Bifida and Study Background

Spina bifida (SB) is a relatively common congenital birth defect that occurs in the first month of pregnancy when the embryonic neural tube fails to close completely (Copp et al., 2015). SB is associated with a number of complications, including bowel and bladder incontinence, varying degrees of paralysis of the lower extremities, clubfoot and other orthopedic conditions, hydrocephalus, increased risk of neurocognitive issues and learning disabilities, as well as psychological, academic, and social difficulties (Holmbeck et al., 2003; 2010). Therefore, individuals with SB are often required to follow a demanding medical regimen, including catheterization, bowel programs, medications, skin checks, and shunt monitoring (Zukerman, Devine, & Holmbeck, 2011). The presentation and severity of SB varies from person to person; Farah has a sacral level lesion, Chiari II malformation, neurogenic bladder and bowel, ventriculoperitoneal shunt, intelligence in the average range (WASI FSIQ at age 15 was 90; Wechsler, 1999), ambulates by walking with leg braces, and was mainstreamed in school. In sum, Farah has the most severe type of SB (myelomeningocele) but has a more moderate symptom profile than most youth with this type of SB.

This case study was drawn from a larger, ongoing longitudinal study that examines family and peer relationships, psychological adjustment, and neuropsychological functioning in youth with SB (Devine et al., 2012; Holbein et al., 2015). While currently in its fifth and sixth waves of data collection, inclusion criteria at baseline were: (1) a diagnosis of SB; (2) age 8–15 years; (3) proficiency in English or Spanish; (4) involvement of at least one primary caregiver; and (5) residence within 300 miles of the laboratory (to allow for data collection in participants’ homes). Data were collected during three-hour long home visits and included questionnaires, interviews (Parent Audiotape Questionnaire [PAQ]; Kelly, Holmbeck, & O’Mahar, 2010), semi-structured videotaped family and peer interaction tasks (Family Interaction Macro-Coding System [FIMS]; Kaugers et al., 2011; Peer Interaction Macro-Coding System [PIMS]; Holbein et al., 2014), neuropsychological testing, and consent to also collect data from teachers, medical charts, and health professionals.

The Fadel family has participated in all six waves of this study and were re-consented for the present case study. However, there are some instances of missing data for this family. First, unlike the rest of the sample, Farah’s mother and father filled out the parent questionnaires together. Therefore, there is only one parent questionnaire available across time points. Second, this study is missing questionnaire data from Time 1 (age 8) for Farah, Time 2 (age 10) for Farah and her parents, and Time 5 (age 16) for her parents.

This case study will describe the Fadel family using the available data. To contextualize this data, clinical cut off scores will be provided when possible (e.g. Child Behavior Checklist; Achenbach & Rescorla, 2001; Children’s Depression Inventory; Kovacs, 1992; Parent Protection Scale [for Time 1, at other time points, she is too old to use the clinical cut off scores]; Thomasgard, Metz, Edelbrock, & Shonkoff, 1995; Vulnerable Child Scale; Perrin, West, & Culley, 1989). However, due to the unique nature of this sample, some questionnaires have been adapted in such a way that prevents the use of clinical cut off scores (Family Environment Scale; Moos & Moos, 1994; Spina Bifida Independence Survey; Wysocki et al., 1996; Sharing of Spina Bifida Management Responsibilities; Anderson, Auslander, Jung, Miller, & Santiago, 1990). Finally, some measures do not provide clinical cut off scores (e.g. Family Stress Scale; Quittner, Glueckauf, & Jackson, 1990; Network of Relationships Inventory). For such measures, the case data will be compared to the mean of the research sample. This study will consider one standard deviation (1 SD) above or below the mean of the research sample to constitute a meaningful difference. Data from Farah’s parents will be compared to data from mother report from the larger research sample because the sample size is larger for mother report and more consistently available across time. It should be noted that the scores for Farah and the Fadel family often do not differ significantly from the research sample, as the research sample is often elevated on the presented measures. This case study will also supplement the aforementioned quantitative data with available qualitative data (Adolescent/Young Adult Self-Management and Independence Scale [AMIS]; Buran, Brei, Sawin, Stevens, & Neufeld, 2006; Parent Audiotape Questionnaire; Family Interaction Macro-Coding System [FIMS]; Kaugers et al., 2011; Questions about the Future-P). Therefore, this case study will demonstrate how this family differs from the larger research sample by presenting differences between Farah and the larger sample based on the quantitative data as well as differences that are evident when examining the family context, which will be done by presenting relevant qualitative data.

Family Environment

According to Amineh, the Fadel family operates in a collectivist fashion, with multiple family members being responsible for the care of the children. Amineh explained that the family consistently works as a unit; individuals in the family are expected to depend on each other and consistently consult each other on matters of significance (Adolescent/Young Adult Self-Management and Independence Scale [AMIS]; Buran, Brei, Sawin, Stevens, & Neufeld, 2006.) Owing to this more collectivist structure, all parent questionnaires were completed by Farah’s parents as a parenting dyad, rather than completing questionnaires separately. This family structure is common in Palestinian culture (El-Islam, 2008). Similar to other Arab cultures, Palestinian families typically have a patriarchal structure. In addition to paternal influence, the contributions of extended family members are also seen as important for healthy family functioning. Collectivist care is both an Arab value and an essential tenet of the Islamic faith (El-Islam, 2008; Aitcheson et al., 2016). Not surprisingly, perceived family support has been found to be significantly more important than perceived peer support for positive psychological adjustment in young adults from collectivist cultures (Pearson & Child, 2007; Jibeen, 2016). Indeed, it appears that this family structure has produced a positive environment for Farah. Parent report on a family environment measure administered at three time points (at ages 8, 12, and 15 for Farah) indicates that the Fadel family was highly organized and exhibited low levels of conflict (Organization scores ranged 2–3 SD above the sample mean across time points; level of conflict scores ranged 1–2 SD below the sample mean across time points). Moreover, family cohesiveness and expressiveness scores fell just below 1 SD above the sample mean. Further, Farah’s parents reported that this positive family environment remained stable over time (i.e. ages 8, 12, 15; Table 1; Family Environment Scale [FES]; Moos & Moos, 1994) and reported lower levels of family stress compared to the general sample over time (i.e. ages 8, 12, 15; Table 1; Family Stress Scale [FSS]; Quittner, Glueckauf, & Jackson, 1990). Finally, at age 19, Farah reported experiencing high levels of social support and low levels of negative interactions from her siblings, Mother, and Father on a questionnaire evaluating positive and negative features of potential peer and family relationships (Possible range: 1–5; Negative interactions with Mother: 1.10, Father: 1.45, Brother: 1.15; Social support from Mother: 3.55, Father: 3.50, Brother: 3.65; Network of Relationships Inventory [NRI]; Buhrmester & Furman, 2008).

Table 1.

Parent, Youth, and Larger Sample Report of Psychological and Family Functioning and Spina Bifida Management, M (SD)

Parent and Larger Sample Report	Time 1		Time 3		Time 4
	Farah	Sample	Farah	Sample	Farah	Sample
	(Age 8)	(Age 8–15)	(Age 12)	(Age 12–19)	(Age 15)	(Age 14–21)
CBCL Internalizing Problems	50	55.68 (10.52)	63	53.20 (10.50)	43	52.31 (11.13)
CBCL Externalizing Problems	54	49.38 (10.52)	40	48.00 (9.59)	40	46.85 (9.30)
CBCL Total Problems	51	54.61 (10.01)	54	51.74 (10.53)	45	51.35 (9.98)
FES
Cohesion	3.00	2.81 (.28)	3.78	3.14 (.38)	3.22	2.97 (.37)
Conflict	2.44	2.15 (.27)	1.00	1.94 (.41)	1.00	2.04 (.42)
Expressiveness	2.67	2.59 (.25)	3.00	2.74 (.28)	3.11	2.71 (.29)
Independence	3.00	1.42 (.28)	3.44	2.93 (.31)	3.67	2.63 (.29)
Achievement Orientation	3.33	2.50 (.30)	3.11	2.71 (.32)	2.89	2.90 (.29)
Organization	3.11	2.53 (.26)	3.56	2.92 (.35)	3.78	2.77 (.37)
Control	3.67	2.51 (.27)	2.33	2.61 (.22)	2.33	2.53 (.20)
FSS Total Score	16.00	34.50 (13.00)	16.00	34.23 (11.47)	17.00	34.50 (13.00)
PPS Total Score	37.00	29.47 (7.03)	46.00	26.56 (6.91)	40.00	24.58 (7.02)
SBIS Total “No” Responses	13	9.98 (10.69)	4	3.56 (7.57)	4	.55 (1.40)
SBIS Ratio of “Yes” Responses to Total	.55	.68 (.25)	.80	.80 (.22)	.79	0.90 (.18)
Responses
SOSBMR Total Score	1.13	1.76 (.41)	1.12	1.96 (.44)	1.50	2.17 (.38)
VCS Total Score	30.00	28.43 (6.88)	34.00	26.53 (6.24)	33.00	(7.10)
Youth and Larger Sample Report M (SD)
	Time 3		Time 4		Time 5	Time 6
	Farah	Sample	Farah	Sample	Farah	Farah
	(Age 12)	(Age 12–19)	(Age15)	(Age 14–21)	(Age 16)	(Age 19)
CDI	30.00	33.29 (5.43)	27.00	35.23 (6.81)	27.00	---
SOSBMR	1.67	2.27 (.34)	1.89	2.42 (.29)	2.19	2.74

Note. M = Mean; SD (Standard Deviation); CBCL = Child Behavior Checklist; CDI = Child Depression Inventory; FES = Family Environment Scale; FSS = Family Stress Scale; PPS = Parent Protection Scale; SBIS = Spina Bifida Independence Scale; SOSBMR = Sharing of Spina Bifida Management Responsibilities; VCS = Vulnerable Child Scale. Missing data from the parent report occurred for all questionnaires at Times 2, 5, and 6 (planned missing at Time 6). Clinical Cutoffs: CBCL (<67 = Normal Range, 67–70 = Borderline Clinical, >70 = Clinical Range); PPS (For Farah’s relevant age range: 97–108 months- 32; clinical cut off scores end at 132 months); VCS (above 10 is considered to categorize subjects as “perceived vulnerable”). Missing data from the youth report occurred for the CDI at Time 1, Time 2, and Time 6, and for the SOSBMR at Time 1 and Time 2. The larger research sample means and standard deviations are provided as a comparison to Farah’s scores at Times 3 and 4. Clinical cut offs: CDI (<30 = Very much below average, 30–34 = Much below average, 35–39 = Below average, 40–44 = Slightly below average, 45–55 = Average, 56–60 = Slightly above average, 61–65 = Above average, 66–70 = Much above average, >70 = Very much above average).

These self-report measures are corroborated by examining observational data from family interaction tasks completed throughout this study (FIMS; Kaugers et al., 2011). The Fadel family exhibited a great deal of cohesiveness in these tasks across time (as seen through observed behaviors such as: verbal statements, nonverbal cues, and collaborative problem solving; FIMS; Kaugers et al., 2011). Further, Farah’s mother also expressed notably positive views of Farah’s condition during these tasks. When discussing a vignette of a female adolescent with SB who is concerned about telling a potential date that she has SB, Amineh stated: “the way they look at it—if they think about it as complicated, it’s complicated; if they think about it as a blessing, it’s a blessing; it’s a reality and she shouldn’t be ashamed or worried, it’s something she was blessed with.” According to the Islamic tradition, sickness is viewed as a type of blessing. This concept is illustrated in the Hadith, a religious resource for Muslims and a record of sayings from the Prophet, Mohammad: “For when a Muslim becomes sick, Allah (God) takes away his sins just as fire takes away impurities in gold and silver” (Sunan Abu Dawood 20:3086). Moreover, caring for the sick is also considered a divine act: “If a man visits a patient in the evening, seventy thousand angels come along with him seeking forgiveness from Allah (God) for him till the morning, and he will have a garden in the Paradise” (Sunan Abu Dawood 21:3092). Research has demonstrated an association between positive parent beliefs about their child’s illness and reduced distress, as well as the promotion of adaptive functioning and positive family functioning (Kazak et al., 2004). Therefore, it appears that the Fadel family is demonstrating a coherent integration of condition beliefs, religious beliefs, and family functioning.

General Autonomy

Farah is currently enrolled in a certificate program to become an early childhood educator. With this educational trajectory, Farah may be able to contribute financially to the household in the future; an opportunity that is not possible for all youth with SB. This educational trajectory differs greatly from what individuals with SB often experience. Indeed, there are significantly lower rates of college attendance and attainment of full or part time employment in this population (Bowman et al., 2001). Farah’s parents’ positive attitude regarding their daughter’s illness may have motivated Farrah’s achievement-oriented behavior. However, it should also be noted that education is valued a great deal in Middle Eastern culture. Moreover, education is also held in high esteem in immigrant families (Greenman, 2012). Therefore, Farah’s parents’ achievement-oriented behavior may also be related to these cultural norms regarding education. Across time, Farah’s parents reported that they had confidence in their daughter’s ability to achieve her goals, including reaching milestones of adulthood (e.g. living independently, getting married, having children). The aforementioned results are in line with previously identified associations between greater parental aspirations for their child and greater educational attainment for youth with SB (Holmbeck et al., 2003; Holbein et al., 2017).

Nevertheless, there are functioning areas in which Farah is not autonomous. In a structured interview regarding her current level of autonomy (AMIS; Buran et al., 2006), Farah explained that her mother and siblings take care of several daily living tasks, such as household chores like laundry and meal preparation. Moreover, Farah is currently dependent on her family financially, as well as for transportation. However, Farah’s current level of autonomy is not unusual for a young adult with SB. Youth with SB have been found to be more dependent on adults relative to their peers, and have demonstrated delays in reaching developmental milestones (Devine et al., 2011; Lennon, Murray, Bechtel, & Holmbeck, 2015).

Parenting practices are critical for fostering autonomy and related self-management behaviors for youth with SB (Zukerman et al., 2011; Devine et al., 2011). The current literature demonstrates that adaptive parenting is positively associated with self-management behaviors (Sawin, 2009), autonomous decision making (Holmbeck, Johnson, et al., 2002), and achieving milestones of emerging adulthood (Zukerman et al., 2011). Specifically, low levels of parental psychological control and higher levels of parental behavioral control and acceptance, have been found to be associated with increased autonomy and positive psychological adjustment (Murray et al., 2015). In contrast, parental overprotectiveness or intrusiveness were found to be associated with lower levels of preadolescent decision making autonomy and parents’ unwillingness/hesitance to grant autonomy to their child in the future (Holmbeck et al., 2003). Other factors that have been found to have positive associations with autonomy include higher socioeconomic status, stronger neuropsychological performance, supportive family environment, and positive psychological adjustment (Murray et al., 2015; Tuminello, Holmbeck & Olson, 2012; Sawin, 2009).

Using this literature to interpret the parent reported data, it would appear that the Fadels are exhibiting protective parenting behaviors, which may undercut Farah’s autonomy development. Further, this protective parenting has remained elevated over time (at Time 1 [age 8] the score was above the clinical cut off [a score of 32 or higher]; at Times 3 and 4 [ages 12 and 15] scores fell between 2–3 SD above the mean; Table 1; Parent Protection Scale [PPS]; Thomasgard, Metz, Edelbrock, & Shonkoff, 1995). Despite Farah’s parents’ expressed confidence in their daughter, the parent report also indicated that they have consistently viewed Farah as highly vulnerable over time (1 SD above sample mean at Time 3 and just under 1 SD above the sample mean at Time 4; Table 1; Vulnerable Child Scale [VCS]; Perrin, West, & Culley, 1989). These data are consistent with literature on parents of children with a chronic illness (Houtzager et al., 2015). This trajectory might be the result of Farah’s parents’ protective nature. However, it may also be related to a cultural norm, in which the heads of the household traditionally make most decisions for family members (Hopkins, 2003).

During the study home visit when Farah was a young adult (19), Amineh interrupted the administration of a structured self-management interview to provide context to her daughter’s responses regarding her current level of autonomy: “There is a certain thing you need to understand about the way that we live our life; we don’t subject her to do anything. It’s not like the typical [household] where when you are 18 you are on your own. She is never on her own, never. We take care of each other in a way that—not that she can’t—she can do more than me. But I have to do everything for her because that is how I’m culturized.”

“Arab parenting styles” have been characterized as both authoritarian and collective (Dor & Fridel, 2010). Traditional Arab parenting style appears to function on a continuum rather than falling into discrete categories (e.g. it can involve equal parts authoritarianism and permissiveness; Dwairy et al., 2006). Moreover, unlike what has been found among families from Western cultures, authoritarian parenting in Arab families has not been found to be related to poor psychological adjustment for children. Indeed, when parents exert a great deal of control over their children, this parenting strategy is thought to be natural and even an expression of love (Ahmad, Smetana, & Klimstra, 2014; Dor & Fridel, 2010). Therefore, Farah’s lack of autonomy was likely considered normative in this culture and may not necessarily be harmful to her adjustment.

Examining Culture-Specific Views on Medical Autonomy

To understand medical autonomy in this case, it is important to review how perspectives on medical autonomy differ globally. Based on a Western view of autonomy, self-reliance and self-determination are held in high esteem (Rathor et al., 2016). With regard to medicine, Beauchamp and Childress (2009) characterized patient autonomy as “the benchmark for moral adequacy of institutional rules.” In the past few decades, Western medicine has placed even more importance on the individual patient’s right to self-determination (Cherry, 2015). This development is part of a modern effort to curtail paternalistic relationships between medical providers and patients. However, this focus on autonomy has also led providers to view families as potential purveyors of undue pressure (Ho, 2008). Therefore, family-driven medical decisions may be met with suspicion or disregarded entirely.

Nevertheless, self-determining patients still function in relation to others, and this reality is especially important for Muslims (Ho, 2008). In Islam, human freedom and religious morality are not incompatible. Autonomy is considered very important, but it is not absolute, and ultimately, the collective takes precedence over the individual (Rathor et al., 2016). One has a responsibility to not only themselves, but also to God and family (Westra et al., 2009). For Muslims, maintaining Beauchamp and Childress’ pillars of nonmaleficence and beneficence is considered far more important than maintaining the patient’s autonomy (Westra, Willems, & Smit, 2009). Other medical systems, such as the Chinese medical system, have incorporated collectivist elements related to Confucian ideology into patient care. There is a growing trend in China of doctor-patient-family-relationships (DPFR; Cong, 2004). However, in the Western world, the burden is placed on the individual to consistently affirm the family’s authority to take part in medical decisions. Recent changes in HIPPA laws have further restricted information that families can receive without explicit authorization from the patient (Cherry, 2015). This is further proof of how individual self-determination is viewed as one of, if not the most important human rights in Western society. However, recent research has encouraged clinicians to respect the family in making medical decisions, as that may ultimately support their patient’s self-determination (Cherry, 2015; Rosenberg et al., 2017; Rathor et al., 2016). It is our hope that presenting this case, which highlights positive aspects of collectivist care, will encourage this trend in clinical patient care.

Medical Autonomy

Qualitative and quantitative measures of self-management responsibility collected over the course of the research study indicated that Farah gradually gained more responsibility over her self-management as she grew older. However, she did so at a slower pace than the research sample, indicating an overall delayed trajectory (i.e. ages 12, 15, 16; [PAQ]; Kelly et al., 2010; Table 1; SOSBMR; Anderson, Auslander, Jung, Miller, & Santiago, 1990). Nevertheless, despite reported gains in medical autonomy, Farah’s responses on the AMIS (Buran et al., 2006), a structured interview about her current level of autonomy, indicated that she relies on her family to manage her condition.

In terms of health literacy, Farah (age 19) reported that she “does not know much” about her condition, needing help from her mother to explain SB about 50% of the time. Moreover, she also reported that she would need help from her mother to explain her “health history” to someone else about 50% of the time. It also appears that Farah has incomplete knowledge regarding complications common to SB. Specifically, at age 19 she was unaware that individuals with SB often experience skin breakdowns, or skin tissue damage/death due to prolonged pressure (AMIS; Buran et al., 2006). This gap in knowledge is problematic, as reports from her medical charts indicated that she has experienced skin breakdowns in the past. However, an interview with Amineh when Farah was 15 years old provides some context for this information: “Well, so far, she takes care of everything to do with herself, aside from changing her diaper, because I am extremely conscious about her and wanting her to be in good condition and never to have a skin infection. And so I go out of my way to take care of her and never allow her to have any issues regarding that” (PAQ; Kelly et al., 2010). During the year of this statement, Farah’s medical chart indicated that she had a skin breakdown in the “diaper area.” Therefore, Amineh may have restricted her daughter’s knowledge of her condition as well as her motivation to take responsibility for her own self-management by maintaining control over changing her diaper. Indeed, Farah’s parents reported that their daughter’s SB knowledge and skills did not change between age 12 and age 15 (Table 1; Spina Bifida Independence Survey [SBIS]; Wysocki et al., 1996). Although Farah did take responsibility for changing her diaper by age 19, a gap in knowledge is still present, likely hindering her ability to take full responsibility for all areas of self-management.

Medical Adherence

Issues noted above indicate that Farah and the Fadel family have not always been adherent with recommendations from their health care providers for managing her SB. Specifically, for the past ten years, Farah’s health providers have reported nonadherence with recommendations for catheterization and bowel management. This is notable, as nonadherence to these recommendations can result in urinary tract infections, kidney damage, and pressure sores. Moreover, in addition to potential medical complications, nonadherence to catheterization and proper bowel management can undermine one’s social and vocational autonomy. Remaining in diapers can result in offensive smells and uncomfortable leeks that could limit the participant’s ability to engage with her peers and participate in daily activities. When asked why Farah decided not to follow the recommendations of health professionals, Amineh explained that Farah was not comfortable with catheterization and that, while participating in a SB support group, Amineh had heard that children who “cath” report a high rate of infections. Therefore, Farah’s noncompliance appears to be due to personal refusal/anxiety regarding catheterization, parental avoidance/reinforcement, and/or issues of condition literacy. Recently, Bruni, Lancaster, and Kullgren (2017) published a case study on an adolescent female with SB that echoes some of these trends; the patient’s nonadherence to self-catheterization was in part a result of her parents’ reinforcement of her refusal behavior and over involvement in the patient’s condition management.

Reviewing the Value of Medical Autonomy

While the example above could represent a potentially dangerous outcome of minimal or delayed autonomy (due to the ever present possibility of infection), other areas of self-management viewed from a collectivist perspective do not necessarily result in poor health consequences. For example, Farah stated that her mother is in charge of scheduling doctor’s appointments, fixing orthopedic braces, and ordering supplies for managing her condition. Due to their existing family structure, if Amineh is no longer be able to take care of these tasks, Amineh reported that this responsibility would fall to Farah’s elder sister. If Farah gets married, then the responsibility will be transferred to her husband. As a young adult, Farah reported that the vast majority of her self-management tasks are managed by her parents. When asked how she felt about that ratio, she reported that she was “very satisfied.” As previously stated, while medical autonomy is considered a pillar of Western biomedical ethics (Beauchamp & Childress, 1979), it is not always considered a critical component of patient care in non-Western societies (Rosenberg et al., 2017). Moreover, regarding adherence, the previously described arrangement could be beneficial, as research has shown that increased medical responsibility in children, adolescents and young adults is associated with poorer adherence (Pai & Ostendorf, 2011; Psihogios, Kolbuck, & Holmbeck, 2015; Wiebe et al., 2014). Therefore, it is necessary for clinicians trained in Western countries to scrutinize the idealization of autonomy while at the same time respecting collectivist approaches to medical care (Ho, 2008; Rosenberg et al., 2017; Westra et al., 2009).

Psychological Adjustment

Youth with SB are at risk for poorer psychological adjustment compared to their TD peers (Ammerman et al., 1998; Holmbeck et al., 2003, 2010). Specifically, rates of internalizing symptoms are elevated in this population (Ammerman et al., 1998; Bellin et al., 2010) and youth with SB are also at increased risk for developing externalizing symptoms (Ammerman et al., 1998). Throughout the course of this study, Farah’s parents’ ratings of Farah’s internalizing and externalizing symptoms were always below the clinical range (Table 1; Child Behavior Checklist [CBCL]; Achenbach & Rescorla, 2001). Moreover, Farah reported little to no depressive symptoms, with scores that would constitute either “very much below average” or “much below average,” according to this measure’s clinical cut off criteria (Table 1; Child Depression Inventory [CDI]; Kovacs, 1992). However, at two time points (i.e., ages 12 and 16), Farah’s teachers rated her internalizing symptoms in the clinical range and her externalizing symptoms in the sub-clinical range (Time 3 [age 12] Internalizing Problems: 75, Externalizing Problems: 69; Time 5 [age 16] Internalizing Problems: 72, Externalizing Problems: 69; Teacher Report Form [TRF]; Achenbach & Rescorla, 2001). This discrepancy could reflect multiple issues. First, mental illness is stigmatized in Arab and Muslim communities (Ciftci, 2012); therefore, Farah’s parents and Farah could be underreporting due to fear of stigmatization. This discrepancy could also be because Farah’s teachers, unlike her parents, can compare her presentation to same-aged peers. However, it is also possible that Farah’s teachers may have interpreted behaviors, such as being withdrawn or submissive, as evidence of internalizing symptoms rather than behaviors that are normative when examined in the context of Arab and Muslim culture. Given the discrepancies across reporters, it is difficult to interpret whether Farah’s mental health represents an area of resilience or risk. It would be important for a clinician to understand the psychological profile of this young adult, as increased depressive symptoms are associated with a number of health outcomes in this population, including poor self-management (Bellin et al., 2010) and lower levels of medical autonomy (Stern, Driscoll, Ohanian, & Holmbeck, 2018).

Limitations

While the present case study has many strengths, including the use of longitudinal data that covers a broad range of constructs pertinent to understanding individual and family functioning, the study also has several limitations. First, this case has missing data and did not have two individual parent reporters, which limits some of our conclusions. Data are still being collected for the larger sample at Times 5 and 6 of this study; therefore, we could not present the mean of the research sample for comparison at these time points. We also do not collect data from parents once the participant is 18. This is a particularly important limitation as these missing data reduce our ability to fully explore the collectivist structure of this family as Farah moves into adulthood. Moreover, clinical cut off scores were not available for some measures, which limits our ability to draw conclusions. Further, it is unclear whether many of our measures have been validated with Arab participants; therefore, it is possible that some of the data are not capturing the full breadth of this family’s functioning. Moreover, the CBCL is often elevated for youth with chronic conditions, thereby limiting our ability to interpret the aforementioned data. Finally, it should be noted that this family may not adhere to all of the traditional beliefs or practices of Palestinian or Muslim culture, and while it is important to be cognizant of potential areas in which culture and treatment might interact, clinicians should not make assumptions about individuals or families solely based on ethnic or religious background.

Clinical Implications

The current case study has several clinical implications. First, it encourages clinicians to critically evaluate what level of autonomy is necessary and adaptive for each individual patient at different points across development given one’s functioning level and cultural background. Second, this case study furthers the current literature on the importance of the family environment and parenting for autonomy, self-management, and psychological adjustment across nonwestern cultures. Parenting and family environment did not have a monolithic impact in this case, further encouraging clinicians to attend to multiple aspects of their patients’ growth and adjustment. In this study, a positive family environment appears to have affected the participant’s mental health favorably and encouraged her to pursue her professional and personal goals in spite of limitations associated with SB. However, this same family environment also has discouraged the participant from gaining full autonomy, both medically and otherwise. By recognizing these different aspects of patient growth and adjustment, clinicians will be able to not only address areas in which their patients are delayed or deficient, but also bolster areas in which they are thriving. For example, a clinician might use cognitive-behavioral techniques to both support this participant’s adaptive belief system regarding her condition, as well as encourage positive behaviors that would benefit her within her collectivist family structure. This could be done through problem solving and behavioral activation experiments, which might test fears about catheterization and demonstrate the potential freedom she could gain from engaging in clean intermittent catheterization. However, these efforts must occur in a culturally sensitive way. Clinicians must work to promote adaptive culturally-rooted family elements, while respectfully addressing elements which may conflict with the best interest of the patient within the framework of the family’s culture and religion.