Matern Child Health J DOI 10.1007/s10995-013-1427-2

Understanding Utilization of Outpatient Clinics for Children with Special Health Care Needs in Southern Israel Hagit Peres • Yael Glazer • Daniella Landau • Kyla Marks • Hana’a Abokaf • Ilana Belmaker Arnon Cohen • Ilana Shoham-Vardi

•

 Springer Science+Business Media New York 2014

Abstract To understand the pattern of utilization of ambulatory care by parents of children with special health care needs (CSHCN) and to explore parental challenges in coping with health maintenance of their infants after discharge from a neonatal intensive care unit (NICU). CSHCN require frequent utilization of outpatient ambulatory clinics especially in their first years of life. Multiple barriers are faced by families in disadvantaged populations which might affect adherence to medical referrals. Our study attempts to go beyond quantitative assessment of adherence rates, and capture the influence of parental agency as a critical factor H. Peres Department of Anthropology and Sociology, Ashkelon Academic College, 12 Ben-Zvi Ave, 78211 Ashkelon, Israel H. Peres (&)
Y. Glazer
D. Landau
H. Abokaf
I. Shoham-Vardi Department of Public Health, Faculty of Health Sciences, BenGurion University of the Negev, POB 563, 84100 Be’er Sheva, Israel e-mail: [email protected]; [email protected] D. Landau
K. Marks Neonatal Intensive Care Unit (NICU), Soroka University Medical Centre, Be’er Sheva, Israel K. Marks
I. Belmaker Faculty of Health Sciences, Ben-Gurion University of the Negev, Be’er Sheva, Israel A. Cohen Department of Quality Measures and Research, Chief Physician Office, General Management, Clalit Health Services, Tel Aviv, Israel A. Cohen Siaal Research Center for Family Medicine and Primary Care, Faculty of Health Sciences, Ben-Gurion University of the Negev, Be’er Sheva, Israel

ensuring optimal utilization of healthcare for CSHCN. A prospective, mixed-methods, cohort study followed 158 Jewish and Bedouin-Arab infants in the first year post discharge from NICU in southern Israel. Rates of utilization of ambulatory clinics were obtained from medical records, and quantitative assessment of factors affecting it was based on structured interviews with parents at baseline. Qualitative analysis was based on home visits or telephone in-depth interviews conducted about 1 year post-discharge, to obtain a rich, multilayered, experiential perspectives and explained perceptions by parents. Adherence to post-discharge referrals was generally good, but environmental, cultural, and financial obstacles to healthcare, magnified by communication barriers, forced parents with limited resources to make difficult choices affecting utilization of healthcare services. Improving concordance between primary caregivers and health care providers is crucial, and further development of supportive healthcare for CSHCN in concordance with parental limitations and preferences is needed. Keywords Children with special health care needs
Adherence
Concordance
Barriers to health services

Introduction In OECD countries the prevalence of vulnerable infants discharged to the community after lengthy neonatal stays is steadily growing due to increase in incidence of low and very low birth weight infant and their improved survival [1–3]. Ensuring development and quality of life for children with special health care needs (CSHCN) often requires extensive utilization of health care services [4–7], presenting a challenge for health care systems. Substantial research evidence exists for the association of adverse birth

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outcomes (i.e. low birth weight and prematurity) and poor developmental achievements, with low socioeconomic position (SEP) and impoverished environments [8–13]. Rural residency is a notable factor that decreases ambulatory access to healthcare [14, 15]. Low SEP has been associated with poor adherence of CSHCN to the recommended schedule of follow up visits [11, 16–18], even under universal health insurance [19], and with higher risk for hospital readmissions in comparison with CSHCN of higher SEP [20, 21]. With the aim of ensuring provision of optimal health care to CSHCN, the American Academy of Pediatrics established the concept of Medical Home, calling upon health care providers to establish alliances with families, recognizing them as the primary source for infant support [18, 22]. It is therefore of importance to understand the obstacles to optimal utilization of healthcare services by families of CSHCN. Israel is an OECD country with National Health Insurance. An ethnically mixed population of Bedouin-Arab and Jewish infants in the southern region of the country allowed us to examine the utilization of health care services of CSHCN in a population with varied SEP and cultural backgrounds. In the present mixed methods study we examine parental responses, to post-discharge from Neonatal Intensive Care Unit (NICU) referrals to ambulatory clinics. In line with strong critic on the term ‘compliance’ as a term implying a general sense of blame for those who do not follow medical recommendations [23, 24], we use the term ‘adherence’ to acknowledge families’ right to choose whether or not to follow medical advice, as it encompasses the complexity of their attitudes, beliefs and life circumstances [25, 26].

Objectives 1.

2.

To examine among Jewish and Bedouin parents of CSHCN adherence rates to referrals to outpatient clinics during the first year post-discharge from the (NICU) and to analyze the role of socio-demographic and medical characteristics associated with adherence. To understand how parents explain their perspective, their considerations, modes of coping and choices regarding clinical follow ups.

Setting The study was conducted in southern Israel (Negev), where the population at the time included a Jewish majority

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(n * 392,000) and a Bedouin-Arab-Moslem minority (n * 188,100) [27]. The Jewish population in the area is mostly urban, low and middle class SEP. Originally a nomadic tribal society; the Bedouins of the Negev have been undergoing a drastic and rapid transition from nomadism to a sedentary and urban lifestyle [28–30]. This transition has improved the health of Bedouin children, but adverse health outcomes are still markedly prevalent especially among women and children [31]. Today, about 60 % of the Bedouin population reside in towns established by the government while the rest live in rural villages, which due to an unsettled dispute over land ownership, are not legally recognized by the government and lack basic municipal infrastructure such as electricity, running water, sewage system, paved roads, garbage disposal, and organized public transportation. The Bedouin towns are among the poorest in Israel with high unemployment rates and poor public services [29, 30, 32]. At the time of the study the total fertility rate of the Muslim population in Be’er Sheva area was 6.91 in comparison with 2.96 in Jews [27]. Consanguinity is the preferred type of marriage among Bedouins (about 60 % of marital unions) [33], and had been associated with higher prevalence of birth defects [34–37]. Polygamy is practiced by an estimated 20 % of the Bedouin population [38], with no official data, as polygamy is prohibited by law in Israel. Practically all births in the area take place in the regional hospital—Soroka University Medical Center (SUMC)— located in Be’er Sheva. SUMC provides birth facilities, NICU, as well as other tertiary services including specialist ambulatory clinics. Since 1995 all Israeli citizens are covered by a National Health Insurance which pays for most healthcare services for infants. Except for Bedouin women, originally from surrounding areas, who are married to Israeli Bedouins (about 13 %), all Bedouins are Israeli citizens and all children are insured by the National Health Insurance Law.

Methods Study Design A prospective cohort, mixed-methods, study was conducted to follow for 1 year infants discharged between July 2007 and June 2008 from NICU at SUMC. The study was approved by the SUMC IRB committee [approval no. 10100]. All parents signed informed consent forms. Information regarding health services utilization was obtained from computerized SUMC databases. About 1 year after discharge, home visits or telephone interviews were conducted with parents about their experiences coping with healthcare needs of their babies. The mixed-

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method approach was quantitatively driven with a simultaneous qualitative component [39, 40] and enabled us to quantitatively assess utilization patterns and simultaneously understand the rich, multilayered, experiential perspectives and explained perceptions by parents. Study Population Parents of infants born at SUMC with severe medical conditions (see ‘‘Appendix’’) as evaluated by a specialist in Neonatal Medicine, and/or with very low birth weight (\1,500 g); were recruited during the hospitalization at the NICU. Parents of infants who were judged by the medical staff as having very poor survival prognosis were not approached. Of the 211 eligible families, 41 were discharged or transferred to other hospitals before contact was made. Of the remaining 170 families 141 were interviewed. Of these, one family asked to discontinue participation, four families’ infants died before discharge, and four other families’ infants died after discharge during the first year of life and were excluded from the present analysis. Of the remaining 131 families comprising 161 infants, data on referrals to ambulatory outpatient clinics in SUMC were available for 158 infants. Data Collection 1.

2.

3.

Intake interview: Parents were interviewed at the NICU using a comprehensive questionnaire which included structured as well as open ended questions. Follow-up interviews: Home visits were conducted around 1 year following discharge from NICU in 50 Bedouin families, sampled by geographic area and type of settlement. Follow-up phone interviews were conducted with the rest of the families. Data on utilization of out-patient clinics were extracted from the SUMC outpatient and hospitalization database.

Independent Variables Familial and Socio-Demographic Background The structured interviews with the parents in the NICU were used to gather information regarding the family structure, home residence, environmental conditions, distance from the main road and from health services, travel costs, economic conditions, main income sources, as well as parents’ characteristics such as age, education and working status. Infant’s Medical Condition The NICU computerized medical records were used to obtain basic medical data including birth weight, gender, gestational age, dates and length of neonatal hospitalization. Type and level of health risk at discharge were determined by two neonatologists (DL and KM, see ‘‘Appendix’’). Data Analysis Statistical Analysis The analysis is based on comparison of the two ethnic groups (Jews and Bedouins) and a comparison between the two groups of Bedouin infants, those living in towns and those living in unrecognized villages. Descriptive statistics were analyzed using Chi square tests or t tests corresponding to the type of the analyzed variables. As adherence rates were not normally distributed, non-parametric tests were used at the univariate level; and for multivariate analysis we used generalized linear models (GLM) for Poisson regression models with the number of referrals as the offset variable. Results were considered significant if P \ 0.05. SPSS version 18.0 was used to conduct all analyses. Qualitative Data

Main Outcome Variables Adherence to referrals at discharge, as well as subsequent referrals made by the out-patients clinics (OPC) in SUMC during the first 365 days after discharge, was computed as the proportion of referrals given to an infant followed by a visit to the clinic. Referrals scheduled for a date when the infant was hospitalized were excluded from the analysis. When, for administrative reasons, there was more than one referral to the same clinic at the same time and date, we counted them as a single referral, and any visit to the specific clinic on that date was recorded as adherence to that referral.

Qualitative data were gathered at follow up interviews (home visits or phone interviews) and contained accounts of the experienced challenges faced by parents coping with health care needs. An explicit question posed to parents was: ‘‘During the last year, have you ever missed a scheduled clinical appointment for your infant at any OPC?’’ ‘‘If yes, what prevented you from keeping the appointment?’’ From each thematic (meaning unit) that was obtained from reading and re-reading of parental accounts, themes emerged that were later indexed into codes and super-codes. These codes created patterned categories from which we could learn about the assumed

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Matern Child Health J Table 1 Socio-demographic and medical characteristics of 158 infants followed for 1 year post NICU discharge from SUMC by ethnic group and residential locality Entire cohort

Bedouins: residential locality

Jews N = 49

Bedouins N = 109

P value

Recognized villages N = 47

Unrecognized villages N = 62

P value

Less than high school

4 (9.8)

59 (54.1)

\0.001

21 (44.7)

38 (61.3)

0.085

High school?

37 (90.2)

50 (45.9)

26 (55.3)

24 (38.7)

Less than high school

4 (8.7)

64 (59.3)

25 (53.2)

39 (63.9)

High school?

42 (91.3)

44 (40.7)

22 (46.8)

22 (36.1)

9 (20.0)

7 (12.5)

36 (80.0)

49 (87.5)

35 (76.1)

35 (59.3)

11 (23.9)

24 (40.7)

Socio-demographic characteristics Father’s education (years)

Mother’s education (years) \0.001

0.260

Mother-employed Yes No Father-employed

37 (80.4)

16 (15.8)

9 (19.6)

85 (84.2)

Yes

39 (92.9)

70 (66.7)

No

3 (7.1)

35 (33.3)

\0.001

0.001

0.305

0.071

Family’s economic situation as evaluated by the mother Very good/good

25 (55.6)

29 (27.4)

16 (35.6)

13 (21.3)

Reasonable/not good

20 (44.4)

77 (72.6)

0.001

29 (64.4)

48 (78.7)

0.104

Yes

6 (12.5)

59 (56.7)

\0.001

22 (47.8)

37 (63.8)

0.103

No

42 (87.5)

45 (43.3)

24 (52.2)

21 (36.2)

34 (89.5) 4 (10.5)

58 (64.4) 32 (35.6)

0.004

27 (75.0) 9 (25.0)

31 (57.4) 23 (42.6)

0.088

No

7 (43.3)

62 (56.9)

\0.001

23 (48.9)

39 (32.9)

0.145

Yes

42 (87.5)

47 (43.1)

24 (51.1)

23 (37.1)

Yes

34 (77.3)

93 (88.6)

36 (83.7)

57 (91.9)

No

10 (22.7)

12 (11.4)

7 (16.3)

5 (8.1)

Always

39 (83.0)

52 (48.6)

26 (55.3)

26 (43.3)

Not always

8 (17.0)

55 (51.4)

21 (44.7)

34 (56.7)

Reported inability to buy basic products for the child due to financial difficulties

Permanent income Parents work with and without other sources of income Other sources of income Family holds supplemental health insurance

Living in proximity to extended family 0.076

0.223

Vehicle availability \0.001

0.218

Polygamy Yes

N/A

No

12 (26.7)

24 (38.7)

33 (73.3)

38 (61.3)

0.193

Spouses are relatives Yes

N/A

No No. of dependents on the father One to seven

N/A

Eight?

26 (55.3)

32 (51.6)

21 (44.7)

30 (48.4)

32 (71.1)

30 (50.8)

13 (28.9)

29 (49.2)

0.701

0.037

Travel time to the hospital (by car and/or on foot) Less than 30 min 30? min

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N/A

33 (84.6)

30 (56.6)

6 (15.4)

23 (43.4)

0.004

Matern Child Health J Table 1 continued Entire cohort

Bedouins: residential locality

Jews N = 49

Bedouins N = 109

P value

High

6 (12.2)

40 (36.7)

0.002

Low

43 (87.8)

69 (63.3)

Male

28 (57.1)

49 (45.0)

Female

21 (42.9)

60 (55.0)

Singletons

28 (57.1)

72 (66.1)

Twins or triplets

21 (42.9)

37 (33.9)

1

30 (62.5)

33 (30.6)

2–3

15 (33.1)

29 (26.9)

4–5 6?

2 (4.2) 1 (2.1)

20 (18.5) 26 (24.1)

Recognized villages N = 47

Unrecognized villages N = 62

P value

13 (27.7)

27 (43.5)

0.088

34 (72.3)

35 (56.5)

19 (40.4)

30 (48.4)

28 (59.6)

32 (51.6)

Infant’s characteristics Health risk level at discharge

Infant’s gender 0.156

0.408

Plurality 0.282

34 (72.3)

38 (61.3)

13 (27.7)

24 (38.7)

0.228

Birth order \0.001

17 (36.2)

16 (26.2)

9 (19.1)

20 (32.8)

10 (21.3) 11 (23.4)

10 (16.4) 15 (24.6)

0.383

Birth weight (gr) Median

1,212.0

1,346.0

1,252.0

1,485.5

Mean (SD)

1,201.1 (296.6)

1,713.1 (902.1)

0.003

1,645.7 (976.6)

1,764.2 (845.9)

\1,500 gr

43 (87.8)

66 (60.6)

33 (70.2)

33 (53.2)

1,500 gr?

6 (12.2)

43 (39.4)

14 (29.8)

29 (46.8)

Median

29.0

31.4

Mean (SD)

29.5 (3.0)

32.4 (4.6)

Less than 32 weeks

41 (83.7)

54 (52.9)

32?

8 (16.3)

48 (47.1)

0.001

0.130 0.072

Gestational age (weeks) Range

Less than 37 weeks

48 (98.0)

80 (78.4)

37?

1 (2.0)

22 (21.6)

dispositions of ‘‘deep-structure’’, and ‘‘cultural axioms’’ on which the discursive practices are based [41]. Rather than reducing data to a few numerical codes, the use of qualitative analysis allows an issue to be discussed from a plurality of angles [41, 42] and observe a spectrum of responses and experiences within contextualized challenges; namely, the diverse parental reactions that were observed as strategies and tactics for coping with certain obstacles.

Findings Of the 161 infants (31 % Jewish and 69 % Bedouins) who were followed, 158 (97.6 %) had at least one referral to any

\0.001 \0.001 0.002

31.0

31.6

31.8 (4.7)

32.8 (4.4)

26 (57.8)

28 (49.1)

19 (42.2)

29 (50.9)

35 (77.8)

45 (78.9)

10 (22.2)

12 (21.1)

0.180 0.385 0.887

OPC at SUMC during the follow-up period. Of the Bedouin infants 57 % lived in unrecognized villages and 43 % in towns. Table 1 presents the socio-demographic characteristics of the study population by ethnic group (Jews compared to Bedouins) and among the Bedouins by type of residence (towns compared to unrecognized villages). The SEP of Jews was higher than that of Bedouins (higher level of education, lower rate of unemployment, and better reported financial situation). From the two Bedouin subgroups, those living in unrecognized villages were of lower SEP and lived further from the hospital than towns’ residents. However, there were no significant differences between the two Bedouin subgroups in terms of family structure (i.e. consanguineous marriage, rates of polygamy, and family size).

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Matern Child Health J Table 2 Referral days, visits and adherence to referrals to Soroka University Medical Center out-patient clinics during 1 year post birthhospitalization discharge by ethnic group All outpatient clinics N = 158 Jews N = 49

Bedouins N = 109

Total number of referral days

680

Number of visit days Number of referral days per child

521

Mean (SD)

13.9 (5.6)

12.4 (7.0)

Median

14.0

11.0

Mean (SD)

10.6 (5.0)

8.6 (6.0)

Median

10.0

9.0

Not visited at all

1 (2.0)

7 (6.4)

Visited at least once

48 (98.0)

102 (93.6)

73.8 %

65.0 %

P value

Bedouins: residential locality Towns N = 47

Unrecognized villages N = 62

1,350

555

795

940

398

542

0.049

11.8 (5.5)

12.8 (7.9)

11.0

11.0

8.5 (4.6)

8.7 (6.9)

9.0

9.0

2 (4.3)

5 (8.1)

45 (95.7)

57 (91.9)

69.3 %

61.7 %

P value

0.895

Number of visits days per child 0.019

0.878

Infant visit to any ambulatory clinic at least once during 1st year post discharge N (%)

Average proportion of visit days/referral days per child (%)

0.436 0.125

0.696 0.158

Characteristics of Infants

Bedouin Infants

In this cohort of high-risk (HR) infants, most Jewish infants were included due to very low birth weight (\1500 g) while Bedouin infants were more likely than Jewish infants to have congenital anomalies or genetic diseases. As a result more Bedouin infants are at higher risk for either chronic disease and/or neuro-developmental impairments (see ‘‘Appendix’’). Within the Bedouin CSHCN, the proportion of infants assessed at discharge as HR was higher in unrecognized villages than in towns. Table 2 presents data about the number of days on which appointments at discharge and during 1 year postdischarge were made to any OPC (referral days) and the number of days on which visits were made. Bedouin, compared to Jewish infants had significantly less referrals days and significantly less visit days. To assess the adherence to referrals we calculated the proportion of referral days on which a visit occurred. The average proportion of visit days/referral days was slightly higher among Jewish than Bedouin infants, and higher among Bedouin infants from towns compared to unrecognized villages, however, these differences were not statistically significant. As large differences were found in sociodemographic and medical characteristics of Jewish and Bedouin infants, we examined factors that might affect adherence to OPCs for each ethnic group separately. The data are presented in Table 3.

Infants classified as ‘‘HR’’ adhered significantly more to referrals to OPC’s than infants at lower risk, and this difference was more prominent among infants who lived in unrecognized villages. Lower, but borderline statistically significant, adherence rate was associated with multiple birth. Lower adherence rates were also associated with the low SEP. In Bedouin towns, but not in unrecognized villages, lower adherence rates were associated with unemployment and polygamy.

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Jewish Infants Factors associated with lower adherence rates included: transportation costs to the hospital regarded by the parent as burdensome as well as father’s low level of education. Multivariable analysis was conducted initially for the entire study population and then separately for the two groups of Bedouin infants as the size of the Jewish group was insufficient for multivariable analysis. We examined several Poisson regression models to assess the independent contribution of factors identified in the univariate analysis as associated with low adherence. The number of OPC visits was used as the outcome variable and the number of referrals as an offset variable (Table 4). The first model suggests that the difference in adherence rates between Jews and Bedouins was no longer significant when

Matern Child Health J Table 3 OPC’s adherence by socio-demographic characteristics and population group Jews N

Total

Bedouin-Arabs type of residence Mean % of appointments kept (SD)

49

73.8 (19.7)

High

6

62.7 (24.9)

Low

43

75.4 (18.7)

Singleton

30

72.5 (24.2)

Multiple

19

75.8 (9.5)

P value

Towns N

Mean % of appointments kept (SD)

47

69.3 (25.2)

13

77.9 (26.8)

34

66.0 (24.1)

34

69.9 (23.9)

13

67.7 (29.2)

Unrecognized villages P value

N

Mean % of appointments kept (SD)

P value*

62

61.7 (28.8)

0.110

27

72.7 (18.5)

0.019

35

53.2 (32.4)

39

66.6 (27.4)

23

53.3 (29.8)

Health risk at discharge 0.135

0.048

Type of birth 0.615

0.943

0.062

Birth order 1

30

74.3 (19.3)

18

72.3 (21.3)

Male

28

72.0 (19.1)

Female

21

76.3 (20.7)

2? Baby’s gender

0.717

0.627

17

63.8 (31.2)

30

72.4 (21.0)

19

73.1 (27.4)

28

66.7 (23.7)

0.478

0.216

16

61.2 (25.8)

45

62.3 (30.2)

30

62.9 (29.2)

32

60.5 (28.8)

0.594

0.746

Father’s education (years) Not completed high school High school?

4

56.2 (20.3)

37

75.6 (20.2)

4

85.0 (2.4)

42

72.6 (21.0)

0.048

21

63.8 (28.0)

26

73.7 (22.2)

25

71.1 (21.8)

22

67.2 (29.0)

0.294

38

61.0 (29.9)

24

62.8 (27.5)

39

58.5 (31.4)

22

68.2 (23.2)

0.712

Mother’s education (years) Not completed high school High school?

0.160

0.806

0.332

Father- employed Yes

39

72.7 (21.6)

No

3

76.2 (15.5)

0.769

35

72.5 (26.5)

11

58.4 (19.2)

0.032

35

64.6 (30.4)

24

62.0 (21.8)

0.327

Mother-employed Yes

37

74.5 (20.0)

9

70.3 (22.6)

31

70.0 (23.0)

7

84.0 (12.6)

Very good/good

25

80.0 (13.8)

Reasonable/not good

20

66.8 (24.9)

20

62.6 (23.7)

19

82.8 (13.3)

No

7

73.7 (23.3)

Yes

42

73.8 (19.4)

39

76.3 (15.8)

8

59.0 (31.3)

No Main source of income Parents work with/without other sources of income Other sources of income

0.698

0.059

9

82.5 (17.6)

36

66.1 (26.5)

28

72.3 (28.0)

10

63.1 (23.4)

16

71.9 (28.7)

29

67.4 (24.0)

23

63.6 (28.4)

18

76.4 (21.9)

23

65.3 (25.5)

24

73.1 (24.8)

26

72.0 (23.0)

21

65.9 (27.9)

0.078

0.174

7

80.8 (9.8)

49

63.1 (27.5)

32

63.7 (30.3)

23

56.0 (29.2)

13

81.5 (18.6)

48

56.2 (29.1)

43

64.0 (27.1)

16

51.0 (32.5)

39

59.3 (28.6)

23

65.6 (29.2)

26

64.9 (26.5)

34

58.6 (31.0)

0.118

0.212

Mother evaluation of family’s economic situation

Transportation costs to the Soroka University Medical Center Negligible Burden

0.053

0.001

0.302

0.109

0.001

0.183

Family holds supplemental health insurance 0.775

0.201

0.386

Vehicle availability Always N = not always

0.192

0.541

0.424

Travel time to the hospital (minutes) \30

33

69.8 (28.2)

30?

6

71.1 (16.8)

0.626

30

61.9 (27.9)

23

63.0 (27.3)

0.979

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Matern Child Health J Table 3 continued Jews N

Bedouin-Arabs type of residence Mean % of appointments kept (SD)

P value

Towns

Unrecognized villages

N

Mean % of appointments kept (SD)

P value

N

Mean % of appointments kept (SD)

P value*

Yes

12

56.9 (27.5)

0.056

24

64.4 (25.8)

0.817

No

33

73.7 (22.9)

38

59.9 (30.7)

Polygamy

Number of dependents on father \7

32

66.6 (27.0)

8?

13

75.0 (22.2)

0.270

30

68.6 (28.5)

29

58.3 (24.9)

0.045

Table 4 Poisson regression models for the prediction of non-adherence to referrals to OPCs B

P value

RR

95 % CI

All children (N = 158) Bedouin (vs. Jews)

0.134

0.185

1.14

0.94–1.39

Poor economic situation as perceived by parents

0.410

\0.001

1.51

1.24–1.83

Bedouins infants (N = 109) Model 1 Unrecognized village (vs. town)

0.120

0.244

1.13

0.92–1.38

Poor economic situation as perceived by parents

0.507

\0.001

1.66

1.29–2.14

Unrecognized village (vs. town)

0.148

0.146

1.16

0.95–1.42

Mother unemployed

0.512

0.003

1.67

1.19–2.33

Unrecognized village (vs. town)

0.233

0.025

1.26

1.03–1.55

Low level of risk at discharge

0.231

0.029

1.26

1.02–1.55

Model 2

Model 3

the parents’ perception of their family economic situation was included in the model. In an analysis including only Bedouin infants, low medical risk and living in an unrecognized village were the two independent predictors of poor adherence. Qualitative Data Analysis As apparent from the quantitative data, most parents kept their appointments to ambulatory follow ups, but nonetheless, did not succeed to adhere to all appointments. As Bedouin parents reported significantly more obstacles to health care access than Jewish parents, we chose to focus our qualitative analysis on Bedouin Parents. Excerpts illustrating each theme are presented in Table 5.

the clinic. Often, several obstacles were mentioned, forming a cluster of co-occurring obstacles. Excerpt #1 in Table 5 demonstrates the multiplicity of obstacles and ways in which parents had coped with them. Mothers often reported that fathers made decisions about which clinics to attend and when to go, while they had only a secondary role in decision making, as they were not allowed to travel independently, nor did they have control of necessary resources (Table 5 #2). Working fathers explained that having to accompany their wives and children, had caused them to sacrifice working days, thus jeopardizing their jobs and family’s income. Some fathers of very vulnerable infants reported they had to quit their jobs in order to find the time for indispensable medical needs of their infants. Medically Defined Versus Perceived Risk

Obstacles to Healthcare A wide range of obstacles causing difficulties to access medical care were reported. The most prevalent were: distance, lack of transportation, and inability to pay for travel to

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The quantitative models have shown that within this cohort of vulnerable Bedouin infants, those at a relatively lower risk were less likely to be brought for follow up visits at the OPCs.

Prioritizing needs

Perception of severity of health risk

‘‘Right now my child has a skin rash but I will not go to the doctor because I will not have the money to buy the medicine. …appointments in the MCH clinic are free of charge so I go. And I do not miss appointments to the hospital [outpatient clinics]; I borrow money from the neighbors if I do not have any.’’ [sno 24]

‘‘… We were happy but a bit concerned. We took home an oxygen tube … H.-Sh. Did not raise her head. At six months it was a concern. … We were referred to the CDI but got an appointment only for 1.3 years. Only after an intense struggle they agreed to anticipate the appointment to 0.6 years. I had to beg to Prof. G. for an earlier visit. Eventually they gave us an appointment and we started providing physiotherapy and hydrotherapy once every 2 weeks [sno 121]

Excerpt #9: Jewish mother to a preterm baby with severe developmental problems

Excerpt #10: Bedouin mother of an infant in a compromised health situation, who had already lost several children due to an unidentified genetic disease

‘‘We feel that she has often difficulties in breathing. At home we have two inhalation devices… we don’t miss any appointment even when we don’t have money… we take money from her brothers and give it to her’’. [sno 50]

In comparison to children of his age he is a bit slow in motor skills. Also his speech is not yet… we have started treating this matter. Overall he is a smart boy. You can tell he is smart but with some help he certainly understands [sno 3]

Excerpt #6: Jewish mother

Excerpt #8: Bedouin Parents of a baby girl with severe respiratory distress syndrome (RDS)

‘‘[she is] remarkably developed. She is growing according to her real age, and not the adjusted age. From the motor aspect, she started walking late, because she has a problem of ‘‘HIPO’’ [Hypotonia]. Today she sings, talks sentences, she is articulate, very clever. She has a charming attitude, some kind of gracefulness. She is very expressive,[thus] she falls a little…’’ [sno 58]

Excerpt #5: Jewish mother of a preterm baby-girl

‘‘… At the beginning the appointments were so many, now there are less … we try to keep every medicine M needs at home. We did not go to the Child Development Institute. I was working and it was hard for us to get there. The time was not convenient. Not that I did not want to go. … M suffered from severe seizures so we rushed to the emergency room [at SUMC] at least fifteen times—only for M, and each time he would be hospitalized for one to 3 days….’’ [sno 81a]

‘‘She was alright, only many appointments to child development clinic. For me she was a normal child. When she was little she had a bandage on her head and I had to change it twice a day, but now nothing…. Now all my worry [is] that her hair doesn’t grow there [on part of her scalp]… There were appointments for ear checkups but I didn’t go because it was too difficult, so I checked her by myself: she can hear and she can see…’’ [sno 38]

Excerpt #4: Bedouin mother of a baby girl born with a large scalp defect and other congenital problems which were (medically) defined as extremely severe and were also very visible. Despite this, at the year’s follow up, the mother insisted that her daughter was already healthy:

Excerpt #7: Bedouin father of twins, one of whom, suffered frequent grand mal seizures during his first year of life

‘‘She walks, she talks, and she can hear and see well… A clever girl… My husband did not take me to the clinics… we did not go for hearing test or for ‘‘child development’’. He would not agree to take me… They invited us to come a couple of times from the hospital but he would not agree to take me.’’ [sno 41]

‘‘[At first] we did not take them for their appointments in Soroka [SUMC] because my husband refused, and after 3 months he agreed and we took them to the appointments. We went to the cardiology institute, to the lung clinic for children, …[but] to the child development clinic we did not go because my husband did not want.’’ [sno 26]

Excerpt #2: F., a Bedouin mother in a multiple birth

Excerpt #3: Second (Bedouin) wife and mother of two children of which the youngest, was born preterm (31 weeks of gestation), and discharged from the NICU with a list of referrals to several ambulatory clinics

‘‘There are so many things we don’t have, and there’s no money. We live on some help from my parents and on the money from [governmental] child allowance. My 17 year old son had to quit school and to work for us. …To go to the hospital I walk with my little son in my arms. Public transportation does not arrive regularly to this area so I walk to the main road and sometimes wait for hours in the heat of the day. Sometimes no car arrives and I turn back home without traveling. …’’ [sno 80]

Excerpt #1: A Bedouin mother from an unrecognized village who could not attend any follow up visits at ambulatory outpatient clinics

Obstacles to healthcare: multiplicity of obstacles to healthcare

Parental perception of health status and its influence on attendance

Citation

Biographic details on parents cited

Theme

Table 5 Qualitative explanation to non-adherence

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‘‘For these children every sickness can lead to a serious problem. We are very alert and do not let her be sick. We go to the clinic for every fever, but we are hesitant to let any doctor treat her. If Dr. S. who knows her best is not in the clinic, they start treating her with the regular protocol, sending her immediately to ER and hospitalizing her. So we prefer to take the risk and leave her at home, fearing it will get worse there [in the hospital]. We have experience and know better than many doctors how to treat her. They are not as familiar with the problem as we are, and we wish they would trust us more with all the experience we have gained.’’ [sno 16] ‘‘We [also] went to the orthopedic clinic—with good results—to the neurologist, and to the eye clinic with both twins. … We went several times to appointments that we understood what they were for [in some of the visits] they repeated the same test so many times until my wife said ‘enough’. … That is why we do not come for every appointment. But we always have all the required medications at home. …. [Re] appointments for Child Development Institute, we tried but could not go. It was on my working days and my wife tried to go [and]… there were appointments to which she decided not to go when she had seen no need.’’ [sno 81b] ‘‘It’s a pity that preterm children are not well cared. The MCH clinic said they cannot do anything for preterm. They referred me to the CDI, but there were no available appointments there. Only after I talked with prof. G. I was accepted… they didn’t even matter she was a preterm. … You need a lot of power, especially in dealing with all the bureaucracy that we have in the State… I have a message to the State: they need to care more. Especially for patients in the South. Every appointment takes 3 months. Why do I have to travel so far for every hydrotherapy treatment? And I do not talk about the travel cost and … also the carrying of a sick baby…’’

Excerpt #11: Bedouin parents of an infant with a severe metabolic genetic disease (the family has already another affected child with the same disease)

Excerpt #12 [continuation of Excerpt #5]

Excerpt #13 (continuation of excerpt #9)

Exercising parental agency

Citation

Biographic details on parents cited

Theme

Table 5 continued

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Parental Perceptions of their Infants’ Health Status The quantitative models have shown that within this cohort of very vulnerable Bedouin infants, parents of lower risk infants were less likely to adhere. Qualitative analysis shows that both Jewish and Bedouin parents’ employed own assessments for their infant’s health status, and not necessarily in agreement with medical assessment, often associated with partial adherence, especially in the presence of major logistic obstacles. Bedouin mothers had utilized their own, intuitive ways of assessments of their children, emphasizing motor performance (excerpts #3 and #6), while Jewish mothers (excerpts #5 and #6), emphasized cognitive development, and compared their children with other same-age children, mentioning professional terminology such as growth graphs and developmental assessment . This finding suggests a stronger incorporation of the medico-scientific lingo in Jewish parents, and better acknowledgment of the importance of OPCs. Perception of Severity of Health Risk In both groups, acute and possibly life-threatening situations seemed to affect adherence in two opposite ways. Some families reported that emergency situations had interfered with adherence to scheduled OPC visits, while other families, reported that anxiety and concerns about the child’s health had motivated them to adhere to all referrals. Excerpts #7 and #8 show such contrasting responses among Bedouin parents. Excerpt #9 of an Orthodox Jewish mother of a daughter with severe developmental problem is an example of a persistent struggle to obtain even more than is offered by the health care system. Prioritizing Needs In the absence of sufficient financial resources, some parents were compelled to make difficult choices between diverse health care needs as well as other needs of their infants. This finding was more prominent in Bedouin families but existent in both groups. As shown excerpt #10 S, a mother of an infant in a compromised health situation, was determined to invest all her available resources, even beyond what she had, for her child’s health. To this end, she chose adherence to OPC at the expense of other immediate health problems which she probably perceived as less critical to her child’s health. Exercising Parental Agency Parents with extensive experience in caring for their vulnerable infants, sometimes explained non-adherence as a deliberate decision based on their discontent from previous encounters with medical care, for example in excerpt #11,

where care had been given by medical staff not familiar with a child’s problems. Moreover, parents who cope on a daily basis with the compromised health of their children expressed dissatisfaction and frustration caused by health care providers who had ignored their experience. Overwhelmed by the large number of referrals, some parents responded with selective adherence. The father in excerpt #12 listed three different reasons for non-adherence: not understanding the purpose of the visit, discontent from a specific medical process, and logistic obstacles. In this particular family the husband described the mother as the one deciding to skip the visit. Missed visits can be divided into two categories: visits parents would have liked to make but were unable to overcome obstacles, and visits they had deliberately decided to avoid. Obstacles emanated from poverty and harsh living conditions, and from cultural constraints. Deliberate decisions not to adhere were based on parental different perceptions of risk, choices between competing health and other demands, and previous unsatisfactory interactions with the healthcare system.

Discussion Studies have shown that the needs of CSHCN and their families are often not satisfactorily met [18, 20, 43, 44] and various models of post discharge interventions have been suggested [1, 2, 5, 17, 45, 46], and assessed [47]. The most widely accepted is that of the Medical Home [48], recognizing the fundamental role of parents in creating a health-promoting social and behavioral environment, and the need to develop partnerships with families and find ways to enhance parental resources. These crucial aspects of care provision were added as indicators of quality of healthcare [3, 22, 49, 50]. Using mixed methodology we set out to examine patterns of utilization of outpatients clinics to which a cohort of CSHCN were referred post discharge from the NICU. Emphasizing parental perspectives and modes of coping. Our quantitative analysis shows that while most parents adhere to the schedule of follow-up visits, adherence is seldom complete. The qualitative data reflect parental perspective on adherence. While obstacles are not always amenable to change, learning from parental coping patterns, either successfully or otherwise, can guide public health policy and interventions. Our study was conducted in a country with universal health insurance, where modern health care facilities are available, and offered by the same health care system to a population with marked ethnic, socio-economic and cultural differences. Thus, utilization of services is not affected by differential health services or by unavailability of health services, as is often the situation for minorities in

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other countries. Consistent with previous studies showing higher rates of infant mortality among Bedouins [51, 52] our study also demonstrates higher prevalence of complex health problems among Bedouin infants. Moreover, all 8 cases of infant deaths within our cohort were Bedouins. As shown in previous research in the same context, [53, 54] our analysis demonstrates how SEP and cultural characteristics function as social determinants of infant health. Unrecognized villages suffer from the worst living conditions and experience many difficulties in access which might affect health status [32, 55]. As evident from the qualitative data, geographical isolation is a major obstacle which turns into a barrier when complicated by financial hardship, lack of private transportation, and lack of access to public transportation. Access is also affected by cultural limitations on women travelling unaccompanied by a male family member, causing the later to miss days of work and jeopardize familial income. Lubetzky [54] observed significant differences between Jews and Bedouins ‘‘compliance’’ with referrals to Child Development Center at SUMC, a gap that diminished after an intervention which included telephone contacts, translation facilities, and detailed explanations to parents in their own language during visits [56–58]. Kushnir et al. [58] suggest that lack of adequate explanation of the purpose for the recommended appointments might create ambivalence, lack of trust and resentment toward health care providers who for lack of inter-cultural communication skills, deficiency of time, and language barriers, fail to communicate appropriately. Moreover, dissatisfaction with coordination of treatment may jeopardize the continuity of healthcare [49]. In accordance with these studies, our research demonstrates that previous bad experiences with the medical system may deter parents from significant encounters. Unlike other studies [20, 44], we found that as health risk becomes more tangible, parental adherence to routine follow-up increases. Higher severity of the infant’s health conditions is associated with higher likelihood of adherence. The qualitative data indicate that referrals to developmental follow up for premature infants with no other specific complications are often given low priority by parents. This is particularly true in the presence of access barriers and limited resources. Coreil [59] identified the imminent tension experienced by mothers who carry the primary responsibility for their children’s health, but, as seen in many traditional minority groups, lack autonomy and ability to mobilize resources to take the required action. The present study underscored such tension, where mothers were forced to make difficult choices in an attempt to balance the health needs of their vulnerable children and other demanding social, familial and domestic responsibilities, creating a substantial emotional burden. In so doing, mothers often applied their own best judgment to assess the severity of health risk in ways that might

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contradict medical recommendations. Our qualitative findings show that parents of infants that experience acute health situations often gave low priority to visits to the otolaryngology and ophthalmology clinics and especially to the Child Development Institute. Parents explained this choice by maintaining that their children’s sensory and motor development is normal, stressing a deep misunderstanding of the value of preventive developmental or psycho-motor therapy. The negation of the existence of a medical problem might also be a way to justify their inability to meet the medically recommended treatment. Another, rather implicit, explanation for choosing to give up visits for ‘‘non-acute problems’’ is that certain follow-ups could not ameliorate the health situation of their children. A further issue concerns the lack of satisfying communication regarding the purposes of referrals. While the dominant discourse of adherence to medical referrals usually demonstrates an unquestioned recognition of the importance of adherence to medical recommendation, at the margins of this narrative, a different voice asks for legitimacy to parental agency, subtly questions the above [medicalized] concept. As previously suggested by Van Herk et al. [60] accessibility to healthcare is influenced by parental feelings of confidence within and around health services. Our study suggest that parents ask that appointments should take into consideration their limited resources, their beliefs, and their life circumstances; and include a medical explanation to the importance of each treatment for their infant’s health. Moreover, parents feel that their daily struggle with the complicated health situations of their infants taught them a lot, and they deserve to be listened to more intently by health professionals. They want both empathy for their daily coping with delicate health situations and recognition in their accumulated experience. Bissell et al. [24] suggested the term ‘‘concordance’’ to describe this notion. Concordance articulates the domestic context of the infant’s family, SEP, parental experience and perspective, and treatment preferences within medical recommendations for assuring the maintenance of healthcare in general and in planning ambulatory follow-up in particular (see also [23, 25, 26]). Through the term concordance, the often unnoticed parents’ voice gains recognition for their desire to communicate more profoundly and on a more egalitarian level with healthcare providers, in order to achieve a mutual understanding and acknowledgement of a potentially different perspective without feeling devalued.

Conclusions Overcoming barriers to health care calls for building concordance between professional medical recommendations

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and parental abilities and perceptions. Providers of health care services to CSHCN should be aware not only of the barriers to care but also of the choices parents make in order to negotiate the best possible care for their vulnerable infants, especially when dealing with ethnic and rural minorities or other underserved populations. Healthcare providers and parents need to assume a mutual responsibility for treatment obtained through communication and integration of perspectives until a shared, agreed upon treatment plan can be reached. Acknowledgments The study was funded by the National Institute for Health Services and Health Policy (NIHP) of Israel [Grant Nos. R23/2005, and R9/56]. Editing was funded by Ashkelon Academic College, Ashkelon, Israel.

Appendix: Criteria for Defining Health Risk (Daniela Landau and Kyla Marks) All infants at HR of morbidity, according to definition of the cohort (C). However within the cohort we defined further risk groups according to diagnoses at discharge. The groups were defined according to: 1.

HR for severe neurodevelopmental impairment (A)

Intraventricular hemorrhage grade 3-4, Periventricular leukomalacia, asphyxia (hypoxic ischemic encephalopathy grade 2-3), congenital malformations of the nervous system, severe physical malformations (spina bifida, arthrogryphosis, congenital hypotonia), bacterial meningitis 2.

4.

5.

6.

7.

8.

9.

10.

11.

12.

HR for chronic health problems (B)

Oxygen dependency at hospital discharge (severe bronchopulmonary dysplasia - PBD), severe congenital heart disease, chronic renal failure, inborn errors of metabolism, ileostomy at discharge 3.

3.

13.

14.

Low risk within the cohort (C)

Preterm, Small for Gestational Age, Mild congenital malformations, bronchopulmonary dysplasia (BPD) without home oxygen therapy after discharge, risk of mildmoderate neurodevelopmental impairment (other abnormalities on head US, extremely low birth weight).

15.

16.

17.

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