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Social Deprivation and Congenital Upper Extremity Differences—An Assessment Using PROMIS

Published:October 20, 2020DOI:https://doi.org/10.1016/j.jhsa.2020.08.017

      Purpose

      Social deprivation, a measure of socioeconomic status, has been shown to negatively affect perceptions of orthopedic conditions and outcomes of treatment. The objective of this study was to assess whether social deprivation correlates with subjective assessment of function in pediatric patients with congenital hand differences.

      Methods

      Patients enrolled in the Congenital Upper Limb Differences (CoULD) registry were assessed using the Patient-Reported Outcomes Measurement Information System (PROMIS). The PROMIS scores for Pain Interference, Peer Relations, Anxiety, Depression, and Upper Extremity (UE) function were obtained for all patients 5 years and older at initial presentation. Social deprivation was determined by the Area Deprivation Index (ADI); the index ranges from 0 to 100 with higher scores being the most deprived. The PROMIS scores were correlated with the ADI for all patients.

      Results

      Three hundred seventy-five pediatric patients with congenital UE differences were evaluated. Average age was 11 years, 56% were female, and 55% had bilateral involvement. Overall, PROMIS scores were within 1 SD of normal for Peer Relations, Pain, Depression, and Anxiety. However, child-reported scores for UE function (39) were more than 1 SD below the national average (50). The mean ADI for the cohort was lower than the national average, indicative of less deprivation, with 14% of patients in the most deprived national quartile. Children in the highest ADI quartile reported PROMIS scores that reflected higher Pain Interference (41 vs 45), lower Peer Relations (55 vs 50), higher Anxiety (44 vs 49), and higher Depression (43 vs 47) than children in the lowest ADI quartile.

      Conclusions

      The PROMIS scores were normal for psychosocial measures in children with congenital hand differences when evaluated as an entire cohort. However, child self-reported PROMIS scores for Pain Interference, Peer Relations, Anxiety, and Depression were worse in more socially deprived areas, suggesting more psychosocial challenges in these children.

      Clinical relevance

      Pediatric patients with congenital upper extremity differences in areas of higher social deprivation report lower psychosocial well-being. The care of these individuals must be considered within the context of their environment because they may be more at risk for negative outcomes secondary to environmental and societal stressors.

      Key words

      External stress can affect an individual’s physical and emotional health. Stress may originate from sources such as malnutrition, limited education, poverty, and exposure to violence. These stresses, when considered collectively, can be termed social deprivation. Social deprivation has been shown to have an impact on health outcomes in orthopedic patients.
      • Urwin M.
      • Symmons D.
      • Allison T.
      • et al.
      Estimating the burden of musculoskeletal disorders in the community: the comparative prevalence of symptoms at different anatomical sites, and the relation to social deprivation.
      • Horton T.C.
      • Dias J.J.
      • Burke F.D.
      Social deprivation and hand injury.
      • Davis E.T.
      • Lingard E.A.
      • Schemitsch E.H.
      • Waddell J.P.
      Effects of socioeconomic status on patients’ outcome after total knee arthroplasty.
      • Court-Brown C.M.
      • Aitken S.A.
      • Duckworth A.D.
      • Clement N.D.
      • McQueen M.M.
      The relationship between social deprivation and the incidence of adult fractures.
      • Dy C.J.
      • Lane J.M.
      • Pan T.J.
      • Parks M.L.
      • Lyman S.
      Racial and socioeconomic disparities in hip fracture care.
      However, few investigations have assessed the effect of social deprivation in the pediatric orthopedic population.
      • Pillai A.
      • Atiya S.
      • Costigan P.S.
      The incidence of Perthes' disease in southwest Scotland.
      • Menon M.R.
      • Walker J.L.
      • Court-Brown C.M.
      The epidemiology of fractures in adolescents with reference to social deprivation.
      • Ramaesh R.
      • Clement N.D.
      • Rennie L.
      • Court-Brown C.
      • Gaston M.S.
      Social deprivation as a risk factor for fractures in childhood.
      • Okoroafor U.C.
      • Gerull W.
      • Wright M.
      • Guattery J.
      • Sandvall B.
      • Calfee R.P.
      The impact of social deprivation on pediatric PROMIS health scores after upper extremity fracture.
      Recently, social deprivation was evaluated in the hand surgery literature in pediatric patients receiving treatment for upper extremity fractures.
      • Okoroafor U.C.
      • Gerull W.
      • Wright M.
      • Guattery J.
      • Sandvall B.
      • Calfee R.P.
      The impact of social deprivation on pediatric PROMIS health scores after upper extremity fracture.
      This study found that pediatric patients in more deprived areas have lower outcome scores for both function and psychosocial well-being.
      The level of social deprivation can be correlated with a person’s household location and is determined by multiple factors including income level, education level, living conditions, and social/societal supports. The Area Deprivation Index (ADI) is a quantitative measure of social deprivation by geographical location that utilizes an individual’s zip code. The ADI has been shown to be a reliable measure of social deprivation.
      • Singh G.K.
      Area deprivation and widening inequalities in US mortality, 1969–1998.
      At this time, little is known about the psychosocial characteristics of the congenital hand difference population and the impact of social deprivation. It is understood that congenital differences can have an impact on an individual’s functional abilities,
      • Bae D.S.
      • Canizares M.F.
      • Miller P.E.
      • Waters P.M.
      • Goldfarb C.A.
      Functional impact of congenital hand differences: early results from the Congenital Upper Limb Differences (CoULD) Registry.
      with reported high self-concept
      • Andersson G.B.
      • Gillberg C.
      • Fernell E.
      • Johansson M.
      • Nachemson A.
      Children with surgically corrected hand deformities and upper limb deficiencies: self-concept and psychological well-being.
      and health-related quality of life.
      • Ylimainen K.
      • Nachemson A.
      • Sommerstein K.
      • Stockselius A.
      • Norling Hermansson L.
      Health-related quality of life in Swedish children and adolescents with limb reduction deficiency.
      However, the contribution of external stresses on psychological well-being in this population has not been evaluated. This study investigated whether social deprivation would correlate with a subjective assessment of function and psychosocial well-being in pediatric patients with congenital upper extremity differences. We hypothesized that patients from more socially deprived areas would have worse functional and psychosocial scores as assessed by the Patient-Reported Outcomes Measurement Information System (PROMIS).

      Methods

      Institutional review board approval was obtained prior to commencing this study. We evaluated patients in the Congenital Upper Limb Differences Registry (CoULD).
      • Bae D.S.
      • Canizares M.F.
      • Miller P.E.
      • Waters P.M.
      • Goldfarb C.A.
      Functional impact of congenital hand differences: early results from the Congenital Upper Limb Differences (CoULD) Registry.
      This registry is a multi-institutional database of congenital upper extremity difference patients that enrolls patients prior to any surgical intervention. We used demographic information and physician-provided classification of the extremity difference by the Oberg-Manske-Tonkin system.
      • Oberg K.C.
      • Feenstra J.M.
      • Manske P.R.
      • Tonkin M.A.
      Developmental biology and classification of congenital anomalies of the hand and upper extremity.
      Patients within the registry with complete demographic information were eligible for inclusion.
      The PROMIS, a series of questionnaires developed by the National Institutes of Health, was utilized to assess perceived outcomes of mental, social, and health domains in the general population
      • Broderick J.E.
      • DeWitt E.M.
      • Rothrock N.
      • Crane P.K.
      • Forrest C.B.
      Advances in patient-reported outcomes: the NIH PROMIS measures.
      and has been used in the pediatric population.
      • Quinn H.
      • Thissen D.
      • Liu Y.
      • et al.
      Using item response theory to enrich and expand the PROMIS pediatric self report banks.
      • Irwin D.E.
      • Gross H.E.
      • Stucky B.D.
      • et al.
      Development of six PROMIS pediatrics proxy-report item banks.
      • Cunningham N.R.
      • Kashikar-Zuck S.
      • Mara C.
      • et al.
      Development and validation of the self-reported PROMIS pediatric pain behavior item bank and short form scale.
      • Varni J.W.
      • Thissen D.
      • Stucky B.D.
      • et al.
      PROMIS Parent Proxy Report Scales for children ages 5–7 years: an item response theory analysis of differential item functioning across age groups.
      Scores range from 0 to 100; 50 represents the population normal score, with a standard deviation (SD) of 10.
      • Bevans M.
      • Ross A.
      • Cella D.
      Patient-Reported Outcomes Measurement Information System (PROMIS): efficient, standardized tools to measure self-reported health and quality of life.
      Minimally important difference (MID) for PROMIS scores in the pediatric population was defined as a difference of 3 points.
      • Thissen D.
      • Liu Y.
      • Magnus B.
      • et al.
      Estimating minimally important difference (MID) in PROMIS pediatric measures using the Scale-Judgement method.
      Patients with complete PROMIS scores for Pain Interference, Peer Relations, Anxiety, Depression, and Upper Extremity (UE) function were included in this study. The PROMIS scores are collected beginning at the age of 5 years. Parents or guardians completed PROMIS scores for all patients, and children/adolescents 8 years of age or older also completed PROMIS questionnaires themselves.
      The ADI was utilized to establish the level of social deprivation. The ADI utilizes the 9-digit zip codes for precise geographical definitions of residence. The index ranges from 0 to 100, with higher scores being the most deprived. The ADI calculator is made available at no cost through the Department of Medicine at the University of Wisconsin Web site.
      University of Wisconsin School of Medicine and Public Health
      2018. Area Deprivation Index.
      Patients with a usable 9-digit zip code were included in this study.
      Three hundred seventy-five pediatric upper extremity difference patients with complete demographic, 9-digit zip code and PROMIS data from initial enrollment were included from the CoULD registry. The average age of inclusion was 11 years. Fifty-six percent were female and 55% of patients had bilateral upper extremity involvement. Descriptive statistics were used for reporting of demographic information, upper extremity classification, PROMIS scores, and ADI for the entire cohort. The ADI scores were divided into quartiles, consistent with prior studies evaluating social deprivation.
      • Dy C.J.
      • Lane J.M.
      • Pan T.J.
      • Parks M.L.
      • Lyman S.
      Racial and socioeconomic disparities in hip fracture care.
      ,
      • Singh G.K.
      Area deprivation and widening inequalities in US mortality, 1969–1998.
      ,
      • Kind A.J.
      • Jencks S.
      • Brock J.
      • et al.
      Neighborhood socioeconomic disadvantage and 30-day rehospitalizations: a retrospective cohort study.

      Results

      Of the 375 patients, there were 247 extremities with a malformation of the entire upper extremity and 261 with a malformation of the hand plate. One hundred thirty-seven extremities had a dysplasia, 4 had a deformation, and 54 had an associated syndrome.
      The average PROMIS scores were within 1 SD of normal for Peer Relations, Pain, Depression, and Anxiety for both parent- and patient-reported scores. In contrast, UE function scores were decreased and averaged more than 1 SD below the mean for child-reported scores, but within the SD for parent scoring (Table 1).
      Table 1Mean and SD of PROMIS Scores for the Entire Cohort
      CohortPROMIS ScoreSD
      Parent-Proxy (5–17-Year-Olds)
       UE Function4012
       Pain Interference4610
       Peer Relations5210
       Anxiety4912
       Depression459
      Child (8–17-Year-Olds)
       UE Function395
       Pain Interference4210
       Peer Relations5311
       Anxiety4511
       Depression4310
      The mean ADI for the entire cohort was lower than the national average (38 vs 50) with a range of 1 to 100, indicating that, on average, our patient sample faced less social deprivation. Forty-two percent were in the highest quartile, 28% in the second quartile, 18% in the third, and 14% in the lowest, or most deprived, quartile.
      We evaluated each of the 5 PROMIS domains in relation to ADI. Parent-rated PROMIS scores were very similar between highest and lowest ADI quartile and did not surpass the MID value of 3.
      • Thissen D.
      • Liu Y.
      • Magnus B.
      • et al.
      Estimating minimally important difference (MID) in PROMIS pediatric measures using the Scale-Judgement method.
      In contrast, there was a difference of 4 points or greater in child-reported PROMIS scores for Pain, Peer Relations, Anxiety, and Depression between highest and lowest quartiles, which is over the MID. Children with higher social deprivation reported higher Pain Interference, lower Peer Relations, higher Anxiety, and higher levels of Depression (Table 2).
      Table 2PROMIS Score in Highest (1) and Lowest (4) Quartile of ADI
      CohortPROMIS Score

      Quartile 1 (SD)
      PROMIS Score

      Quartile 4 (SD)
      Parent-Proxy (5–17-Year-Olds)
       UE Function40 (12)40 (12)
       Pain Interference45 (9)46 (10)
       Peer Relations51 (11)52 (11)
       Anxiety48 (12)47 (11)
       Depression45 (9)45 (9)
      Child (8–17-Year-Olds)
       UE Function39 (5)38 (4)
       Pain Interference41 (8)45 (10)
       Peer Relations55 (10)50 (10)
       Anxiety44 (10)49 (12)
       Depression43 (9)47 (12)
      Bold numbers signify scores differences greater than MID of 3 points.
      • Thissen D.
      • Liu Y.
      • Magnus B.
      • et al.
      Estimating minimally important difference (MID) in PROMIS pediatric measures using the Scale-Judgement method.

      Discussion

      The impact of external environmental and social stresses, represented by social deprivation, not only affect an individual’s ability to cope with a congenital difference but may also influence health outcomes, as seen in other orthopedic conditions.
      • Urwin M.
      • Symmons D.
      • Allison T.
      • et al.
      Estimating the burden of musculoskeletal disorders in the community: the comparative prevalence of symptoms at different anatomical sites, and the relation to social deprivation.
      • Horton T.C.
      • Dias J.J.
      • Burke F.D.
      Social deprivation and hand injury.
      • Davis E.T.
      • Lingard E.A.
      • Schemitsch E.H.
      • Waddell J.P.
      Effects of socioeconomic status on patients’ outcome after total knee arthroplasty.
      • Court-Brown C.M.
      • Aitken S.A.
      • Duckworth A.D.
      • Clement N.D.
      • McQueen M.M.
      The relationship between social deprivation and the incidence of adult fractures.
      • Dy C.J.
      • Lane J.M.
      • Pan T.J.
      • Parks M.L.
      • Lyman S.
      Racial and socioeconomic disparities in hip fracture care.
      When children 8 years or older with congenital upper extremity differences live in more deprived areas, they report a higher level of Pain Interference, lower Peer Relations, higher Anxiety, and higher Depression than those in less deprived areas. Despite the small sample, it is concerning that, with the current patient cohort, children are reporting lower psychosocial scores in the more deprived environments. It is possible that these children face a more negative psychosocial impact as a result of their differences, putting them at psychological risk. This study raises the concern that children in more deprived areas may have fewer and weaker coping skills and support than those in more affluent neighborhoods.
      Previously, social deprivation has been evaluated in pediatric orthopedic conditions including upper extremity fractures.
      • Okoroafor U.C.
      • Gerull W.
      • Wright M.
      • Guattery J.
      • Sandvall B.
      • Calfee R.P.
      The impact of social deprivation on pediatric PROMIS health scores after upper extremity fracture.
      Okoroafor et al
      • Okoroafor U.C.
      • Gerull W.
      • Wright M.
      • Guattery J.
      • Sandvall B.
      • Calfee R.P.
      The impact of social deprivation on pediatric PROMIS health scores after upper extremity fracture.
      found that children with upper extremity fractures living in more deprived areas had lower PROMIS UE function, Mobility, Pain Interference, and Peer Relations than those living in less deprived areas. In contrast to Okoroafor et al,
      • Okoroafor U.C.
      • Gerull W.
      • Wright M.
      • Guattery J.
      • Sandvall B.
      • Calfee R.P.
      The impact of social deprivation on pediatric PROMIS health scores after upper extremity fracture.
      however, a majority of congenital upper extremity patients reside in areas with less deprivation, with 42% in the least deprived quartile versus 21% in that quartile in the fracture cohort. The authors found that the impact of increased social deprivation was similar between the 2 populations with both cohorts of children reporting higher Pain Interference and worse Peer Relations. The UE function score was low, with child-reporting, in all quartiles of ADI for the congenital cohort, but varied in the pediatric UE fracture patients depending on social deprivation level.
      Uppal et al
      • Uppal S.
      Impact of the timing, type and severity of disability on the subjective well-being of individuals with disabilities.
      reported on subjective well-being in a cohort of Canadians with congenital and acquired disabilities. A notable conclusion from the study was that individuals with disabilities were happiest early and later in life, with worse well-being in midlife. While Uppal et al
      • Uppal S.
      Impact of the timing, type and severity of disability on the subjective well-being of individuals with disabilities.
      identified age to be an intervening variable in patient perceptions of well-being, we identified socioeconomic status, as measured by area deprivation, to be another intervening variable. Interestingly, our results were in contrast to Uppal et al’s study,
      • Uppal S.
      Impact of the timing, type and severity of disability on the subjective well-being of individuals with disabilities.
      which also concluded that family economic status had no effect on happiness. Our results revealed that those children in the lower socioeconomic quartile had worse scores for Peer Relations and Anxiety and Depression. The basis for this discrepancy in results is unclear, but it seems logical that increased stress from higher levels of social deprivation would yield higher levels of anxiety and depression as seen in our study.
      Another study, from Sweden, of adolescents with long-term illness or disability, identified that adolescents generally had similar well-being to others without these conditions.
      • Berntsson L.
      • Berg M.
      • Brydolf M.
      • Hellstrom A.L.
      Adolescents’ experiences of well-being when living with a long-term illness or disability.
      A sense of acceptance of their disability, a feeling of support, and a feeling of growth were the themes with the most impact. Thus, efforts to improve an individual’s living environment and socioeconomic status, as represented by the ADI, and providing psychosocial support could potentially contribute to an improvement in well-being in our cohort with lower PROMIS scores.
      The limitations of this investigation include those inherent to a longitudinal cohort study in which patients are enrolled at birth. The CoULD registry is limited to inclusion of only patients who present for hand surgery care; thus, individuals in lower ADI regions may be underrepresented because of access to care and transportation barriers. However, we were able to pull out these individuals by breaking down the cohort by quartile, which gives insight into the more socially deprived group. The CoULD registry includes a wide range of congenital upper extremity differences, but only children 5 years and older have PROMIS scores. The PROMIS questionnaires have not yet been applied to patients younger than 5 years and, therefore, many younger patients were not included in this analysis. A limitation specific to this study was the lack of demographic information for patients with incomplete data; only patients with complete data were included. A previous CoULD publication reported a 10% rate of incomplete data
      • Bae D.S.
      • Canizares M.F.
      • Miller P.E.
      • Waters P.M.
      • Goldfarb C.A.
      Functional impact of congenital hand differences: early results from the Congenital Upper Limb Differences (CoULD) Registry.
      ; thus, we assume a similar rate for this study, which implies a potential bias in our data and conclusions. For example, if all 10% were potentially in the lower quartile and had better PROMIS scores, or all were in the highest quartile and had lower scores, then our conclusions would be altered. In addition, we did not conduct any subanalysis to investigate differences based on severity of the congenital difference. Lastly, a post hoc power analysis showed that the study was underpowered to explore the differences in parent-reported PROMIS scores between ADI quartiles.
      Limitations are also inherent in the use of the ADI measurement of social deprivation. although it is an accepted tool for assessing the social deprivation of a community, there should be an understanding of the continual fluidity of communities. Thus, utilizing an individual’s zip code to identify and classify an individual may be inherently biased, especially in areas of quickly changing demographics. However, at this time, the ADI remains one of the accepted tools for assessing social deprivation.
      • Dy C.J.
      • Lane J.M.
      • Pan T.J.
      • Parks M.L.
      • Lyman S.
      Racial and socioeconomic disparities in hip fracture care.
      ,
      • Singh G.K.
      Area deprivation and widening inequalities in US mortality, 1969–1998.
      ,
      • Kind A.J.
      • Jencks S.
      • Brock J.
      • et al.
      Neighborhood socioeconomic disadvantage and 30-day rehospitalizations: a retrospective cohort study.
      In conclusion, in this study of patients with congenital upper extremity differences, there was lower psychosocial well-being seen in areas of higher social deprivation. This cohort of patients may be at greater risk for stress and may benefit from physician and multidisciplinary intervention. There is evidence that individuals with congenital differences will have increasing levels of depression and anxiety over time as they age,
      • Newbronner E.
      • Glendinning C.
      • Atkin K.
      • Wadman R.
      The health and quality of Thalidomide survivors as they age—evidence from a UK survey.
      supporting a need for psychosocial support. These interventions, such as family support groups or genetic counseling, when combined with nonsurgical or surgical treatments to improve hand and extremity function, may improve the overall health status of children with congenital upper extremity differences.

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