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Imputing missing race/ethnicity in pediatric electronic health records: reducing bias with use of U.S. census location and surname data.

Comparative effectiveness research using electronic health record (EHR) data promises timely understanding of health disparities (Olsen, Aisner, and McGinnis 2007; Fiks et al. 2012) but requires accurate racial/ethnic data, which are often completely or partially missing in EHR data (Bierman et al. 2002; Kressin et al. 2003; Hasnain-Wynia, Pierce, and Pittman 2004; Fremont et al. 2005; Bilheimer and Sisk 2008; Smith et al. 2010; Wynia, Ivey, and Hasnain-Wynia 2010; Bergdall et al. 2012).

Novel analytic methods for missing EHR race/ethnicity data are needed, as many current methods have shortcomings. Complete case analysis confined to observations without missing values produces biased estimates, except when data are missing completely at random (MCAR)--unassociated with any outcomes or covariates--or when the chosen statistical model assumes only missingness at random (Molenberghs and Kenward 2007; National Research Council (United States), Panel on Handling Missing Data in Clinical Trials, National Research Council (United States), and Committee on National Statistics 2010; Graham 2012). The missing indicator method--using a missing category indicator for missing variables such as race/ethnicity--also produces biased estimates and (Greenland and Finkle 1995; Knol et al. 2010). Mean imputation can understate variance and underestimate p-values. Multiple imputation, though effective when used appropriately, is inefficient compared to an analysis based on all data if no values were missing, especially when a large fraction of the data is missing or key predictors of missing values are absent (Rubin 1987).

To address problems of missing race/ethnicity in disparities research, RAND researchers created the Bayesian Improved Surname Geocoding (BISG) method to impute race/ethnicity among adult patients using U.S. Census geospatial and Census surname data (Fiscella and Fremont 2006; Elliott et al. 2008, 2009). Subsequently, a categorized version of the 2009 algorithm was validated for classifying patients into racial/ethnic groups, with favorable results across the age spectrum (Adjaye-Gbewonyo et al. 2014), and a couples version was validated in a study of marriage licenses (Elliott et al. 2013). The Centers for Medicare & Medicaid Services and health plans have successfully used the BISG to better understand racial/ethnic disparities (Derose et al. 2013; Martino et al. 2013).

Here, we used the BISG's probability outputs in a pediatric cohort to enhance generally accepted methods of imputing missing race/ethnicity data.

METHODS

Using a retrospective cohort design, we added BISG probability outputs to pediatric data to evaluate whether these probabilities improve existing methods of imputing missing race/ethnicity. We extracted EHR data for children with known race/ethnicity and experimentally assigned race/ethnicity to "missing" for a sample of these children to measure the performance of different strategies for estimating missing race/ethnicity. We included children who received preventive health care at 30 practices in Pennsylvania and New Jersey between July 1, 2009, and June 30, 2010, before their 19th birthday. The 30 practices routinely recorded parent-reported race/ethnicity during registration. Children were identified by preventive health care evaluation and management codes in the EHR (CPT codes 99381-99385, 99391-99395).

Beginning with 220,090 children, we excluded 26,007 children whose address could not be accurately geocoded, 1,608 for whom U.S. Census race/ ethnicity data were not available for their census block group, 19,447 with unknown or low-frequency race/ethnicity values (e.g., Native American, multiracial), and 2,857 children without insurance, leaving 170, 171 children.

Study variables included demographic and clinical characteristics possibly associated with race/ethnicity. In addition to parent-reported race and ethnicity, these variables were child surname, home address, gender, age and insurance type, presence of asthma, and presence of attention-deficit hyperactivity disorder (ADHD) as identified by ICD-9 codes specific to these conditions on the problem list (codes beginning with the digits 493 and 314, respectively).

Bayesian Improved Surname Geocoding Method

The BISG method estimates a posterior probability of membership in each of six racial/ethnic categories--(1) Hispanic/Latino; or non-Hispanic (2) black/ African American; (3) white; (4) Asian/Pacific Islander; (5) Native American/ Alaska Native; or (6) multiracial--using the patient's surname and the U.S. Census block group of their home address. The BISG algorithm in this instance used 2000 U.S. Census data about the race/ethnicity of each block group and the probability of each surname belonging to each of the six racial/ ethnic categories above. Race/ethnicity information is available only for surnames that occur at least 100 times in the U.S. population (Word et al. 2008); for rare surnames, the algorithm uses aggregate race/ethnicity information for all U.S. Census respondents whose surnames occur fewer than 100 times (Elliott et al. 2009).

Geocoding Address to Census Block Group

Patient address was geocoded to the corresponding latitude, longitude, and 2000 census block group using the Geocoder/U.S. v2.0 software package (Erie n.d.), which scored the confidence of the match with a 0 to 1 score. Scores less than 1 indicated the need for corrections (e.g., misspelled street names). 194,083 (88 percent) of the children in our cohort were geocoded with a score of 0.8 or higher. To ensure accurate geocoding, we manually validated a random sample of 50 addresses with scores in the 0.8 to 0.9 range and confirmed that all were geocoded correctly.

Statistical Methods

As a first comparison, we used logistic regression to model the true racial/ethnic category as a function of the imputed value. This model produced an area under a receiver operating characteristic curve (AROC) as a measure of the predicted value's ability to discriminate between the given racial/ethnic category and other categories.

Iterative Simulations. We iteratively performed 200 simulations with the following steps: (1) generated hypothetical outcomes, both continuous and binary; (2) set known race/ethnicity to missing; (3) applied four strategies for analyzing data in the presence of missing values; and (4) used regression models to estimate the association of the outcomes with the racial/ethnic groups. We compared the true values for the hypothetical outcomes with the median values and interquartile ranges for each analytic method: complete case analysis, missing value indicator method, imputation using patient information, and imputation with BISG probabilities added to patient information. The simulation analysis was performed on a 10 percent sample of children (n = 17,017) for computational efficiency.

Generating Outcome Variables. To test our ability to derive unbiased estimates for the association of race/ethnicity with outcome, we constructed two outcome variables strongly associated with race/ethnicity: one continuous and one dichotomous (equations 1 and 2). The continuous outcome represented a normally distributed score similar to an ADHD symptom score (Wolraich et al. 1998), with mean of 100 and standard deviation of 10. The dichotomous outcome represented an endpoint such as pediatric asthma hospitalization, often strongly associated with black/African American race/ethnicity. We constructed these outcomes to have plausible associations with race/ethnicity and disease status that could result in bias if the data were not missing randomly.

Race/Ethnicity Missing Not at Random. Using the complete dataset with the added outcomes noted above, we then set race/ethnicity to missing for a fraction of the observations. For the continuous outcome, we used an equation that increased the chances of being missing for black children with an outcome score that was less than 90, and for Hispanic children whose outcome scores exceeded 105. This equation correlated the probability of missingness with true race/ethnicity and also with the value of the outcome, resulting in a nonignorable (or Missing Not At Random) missing data mechanism. Similarly, for the binary outcome simulations, we increased the chance of missingness for black children when the outcome was 0 and for Hispanic children where the outcome was 1 (Table 1).

Missing Data Methods. We used four strategies to handle missing race/ethnicity: (1) complete case analysis, using only children with nonmissing data; (2) missing indicator method; (3) traditional multiple imputation without BISG enhancement (Schafer 1999); and (4) traditional imputation enhanced by the BISG method.

For traditional multiple imputation, we used variables that might be predictive of race/ethnicity. We used the method of chained equations with race/ ethnicity as a multinomial outcome in the regression model to impute race/ ethnicity from the following patient-level covariates: gender, age in years, insurance type, diagnosis of asthma, and diagnosis of ADHD (Royston 2004; Van Buuren 2007; White, Royston, and Wood 2011). In addition, because the location of care in a highly diverse major city and its suburbs might be highly predictive of race/ethnicity, we included as a series of 29 indicator variables the primary care practice site. Finally, as recommended (Little 1992), we used the dichotomous and continuous outcome scores as predictors in the imputation model. In the enhanced imputation, we used these same predictor variables--including care location--and the probability of membership in the six race/ethnicity categories calculated with the U.S. Census geospatial and surname data (the BISG method's output). For each of the 200 iterations in this simulation, we repeated the imputation to construct 10 datasets for both the traditional imputation and the enhanced imputation.

Estimating the Association of Race and Ethnicity with Outcome

The associations between demographic characteristics (race/ethnicity, gender, age, and primary care site) and the outcome variables were calculated using multivariable linear regression for the continuous outcome and multivariable logistic regression for the dichotomous outcome. We compared the associations observed in each of the experimental datasets with the true associations observed in the gold standard dataset.

Linear and logistic regressions were performed using Stata v 73.0 (2008; Stata Corp, College Station TX, USA). The Institutional Review Board at The Children's Hospital of Philadelphia approved the study and waived the requirement for consent from individual children/families.

RESULTS

Demographic and clinical characteristics for the study cohort appear in Table 2.

Performance of BISG-Supplemented Imputation

The surname for 171,093 (88 percent) of the 194,083 geocoded patients exactly matched entries in the U.S. Census surname list, similar to the population rates of common surnames (Elliott et al. 2009). As expected from the construction of the Census surname list (Word et al. 2008), the unmatched surnames included hyphenated names, misspellings of common names, and uncommon names. All unmatched surnames were included as "rare surnames" (see BISG methods section).

Ability of BISG-Supplemented Imputation to Identify True Race/Ethnicity

For the major race/ethnicity categories, the census-based probabilities had good qualities as a test for parent-reported race and ethnicity. The AROC was high for all race and ethnicities prevalent in our cohort (Table 3).

Estimated Racial/Ethnic Proportions in the Cohort

By design, the distribution of race/ethnicity among cases with nonmissing data differed substantially from the true proportions. In all categories, the addition of the BISG probabilities to formal imputation models slightly improved the estimates for the proportion of children in each race/ethnicity category. In most categories the addition of care location also resulted in slight improvements (Table 4).

Performance of Imputation Methods

Median values from simulations for the association of race/ethnicity and outcome appear in Table 5. As expected by the design, the regression estimates obtained using all data in the gold-standard dataset matched the stipulated values for both the continuous and dichotomous outcomes. Two methods for handling missing data--complete case analysis in which only data with nonmissing values are used and the missing indicator variable method--produced highly biased results. With the binary outcome for these two methods, the direction for the odds ratio was reversed for the black subjects (an odds ratio greater than 1 was estimated when the true odds ratio was 0.5).

Standard multiple imputation reduced the bias somewhat, especially for the estimates of both the continuous and dichotomous outcome associated with black race. However, in the case of the dichotomous outcome, the odds ratio estimate using multiple imputation was only slightly less than 1 for black race/ethnicity (true value 0.5). Bias fell substantially, although it did not disappear, for BISG-supplemented-multiple imputation. In the case of the dichotomous outcome, only the BISG-supplemented approach correctly estimated the odds ratio associated with black race/ethnicity to be less than 1.

As a final check, we assessed whether the use of the BISG probabilities introduced bias in multiple imputation when race/ethnicity data were missing in an unbiased fashion. We found that no bias was introduced by the use of BISG data in this scenario (data not shown).

DISCUSSION

One promise of "big data" is that large datasets derived from EHR and administrative sources will expedite understanding of health disparities. Unfortunately, large clinical data research networks that aggregate data from multiple health systems, such as those funded by the Patient-Centered Outcomes Research Institute, will encounter randomly and nonrandomly missing race/ ethnicity data for the foreseeable future.

We investigated whether the addition of the new information derived from the BISG surname/address method to traditional methods of multiple imputation, improves the accuracy of estimates in the presence of partially missing data on race and ethnicity in a large pediatric EHR dataset. We deliberately used conventional missing data methods because the software for those methods is increasingly available and the output provides discrete categories rather than probabilities, allowing reporting both from regression analyses and contingency tables. A case of extreme bias with a calculated odds ratio in the wrong direction for a dichotomous outcome was almost completely corrected using the BISG-enhanced imputation approach. In this case, the BISG-enhanced imputation odds (0.65) were similar to the true odds (0.5). In our cohort, the enhanced imputation model improved estimates and reduced bias. An additional advantage of formal imputation of missing data, as contrasted with use of only complete data, lies in the increased statistical power of the larger usable samples.

Limitations

Although the 30 primary care sites in this study care for a racially and ethnically diverse group of children, the sample was limited to a single health system in two states. The data for this study were derived from EHRs captured as part of routine care and likely include misclassification errors in all variables. We excluded multiracial children from the dataset because there was no reliable way to enter this classification in our EHR. Multiracial children were typically described as having "other" or "unknown" race/ethnicity at the time of this study. Also, as less than 1 percent of the children in our cohort were American Indian, we were unable to evaluate performance for this subgroup. Insurance type was our only marker for socioeconomic status. More precise markers such as household income should be included in multiple imputation when available.

Race/ethnicity probabilities from the BISG algorithm do not include estimates of the uncertainty of those probabilities. However, uncertainty in BISG probabilities has been shown to have negligible effects on estimation under reasonable assumptions (McCaffrey and Elliott 2008). Finally, with increasing numbers of racially blended families, the surnames of children over time might have decreasing value in predicting racial/ethnic backgrounds.

CONCLUSIONS

When used with standard statistical tools for proper imputation of missing values, the additional data from an algorithm for identifying race/ethnicity using U.S. Census data on surnames and home address lead to reduced bias in typical analyses of associations between race/ethnicity and health outcomes. Our results suggest that, even in pediatric settings, this algorithm should be preferred over standard methods for addressing missing race/ethnicity data.

DOI: 10.1111/1475-6773.12295

ACKNOWLEDGMENTS

Joint Acknowledgment/Disclosure Statement: This research was supported by grant funding to the American Academy of Pediatrics from the Maternal & Child Health Bureau (UB5MC20286).

Disclosures: None.

Disclaimers: None.

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SUPPORTING INFORMATION

Additional supporting information may be found in the online version of this article:

Appendix SA1: Author Matrix.

Address correspondence to Robert W. Grundmeier, M.D., The Children's Hospital of Philadelphia, 3535 Market Street, Philadelphia, PA 19104; e-mail: grundmeier@email.chop.edu. Lihai Song, M.S., Mark J. Ramos B.S., and Alexander G. Fiks, M.D., M.S.C.E., are also with The Children's Hospital of Philadelphia, Philadelphia, PA. Marc N. Elliott, Ph.D., and Allen Fremont, M.D., are with the RAND Corporation, Santa Monica, CA. Wilson Pace, M.D., is with the DART-Net Institute, University of Colorado Denver, Aurora, CO. Richard C. Wasserman, M.D., M.P.H., is with the University of Vermont, Burlington, VT. Russell Localio, Ph.D., is with the Department of Biostatistics and Epidemiology, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA.
Table 1: Methods for Generating Hypothetical Outcomes (Equations 1
and 2) and Then for Setting Known Race Ethnicity Categories to Missing
(Equations 3 and 4) for Purposes of Testing the Performance of Missing
Data Imputation Methods

Equation Name                     Equation Definition

Continuous and dichotomous
outcomes

Equation 7: Continuous outcome    [Score.sub.c] = 100 = 10 *
                                  [RE.sub.black] + 10 *
                                  [RE.sub.hispanic] - 5 *
                                  [Dx.sub.asthma] + e (1)

Equation 2: Dichotomous outcome   log-odds(E(Score)) =
                                  [[mu].sub.outcome] = logit(0.2) -
                                  0.693 * [RE.sub.black] + 0.693 *
                                  [RE.sub.Hispanic] + 0.289 *
                                  [Dx.sub.asthma] (2)

Setting race and ethnicity
to missing to introduce bias

Equation 3: Bias for              [[mu].sub.missing] = logit(0.2) +
continuous outcome                2.25 * [RE.sub.black] *
                                  ([Score.sub.c] <90) + 1.25 *
                                  [RE.sub.Hispanic] * ([Score.sub.c]
                                  > 105) (3)

Equation 4: Bias for              [[mu].sub.missing] = logit(0.2) +
dichotomous outcome               2.25 * [RE.sub.black] * (Score = 0)
                                  + 1.25 * [RE.sub.Hispanic] *
                                  (Score = 1) (4)

Variable Name                     Definition

Score                             Outcome score (c = continuous)

RE                                Race/ethnicity

Dx                                Indicator variable for asthma or
                                  ADHD

e                                 Error term, normally distributed,
                                  mean = 0, standard deviation = 20

[[mu].sub.outcome]                Mean of a binary score drawn
                                  randomly from a binomial
                                  distribution

[[mu].sub.missing]                Log odds of the predicted
                                  probability of race being missing

Table 2: Characteristics of the Study Cohort

Group                             N         %

Hispanic or Latino ethnicity     8,706     5.1
Non-Hispanic
  White                        101,377    59.6
  Black or African American     55,286    32.5
  Asian or Pacific Islander      4,802     2.8
Gender
  Female                        83,700    49.2
  Male                          86,471    50.8
Age
  Birth to 4 years              54,987    32.3
  5 to 11 years                 65,950    38.8
  12 years and up               49,234    28.9
Disease
  Asthma                        22,216    13.1
  Attention-deficit disorder     7,462     4.4

Table 3: Area under the Receiver Operating Characteristic (AROC) Curve
as a Measure of the Ability of the BISG Measures of Race/Ethnicity to
Discriminate among the True Race and Ethnicity Groups

                                                        % Correctly
                                        ROC Area        Identified
Race or Ethnicity *            N       ([dagger])    ([double dagger])

Hispanic or Latino            8,706       0.88             50.2
White                       101,377       0.95             97.2
Black or African American    55,286       0.97             75.8
Asian or Pacific Islander     4,802       0.91             62.6
Total                       170,171       0.95
                                       ([section])

* American Indian/Alaska Natives and multiracial categories were
excluded.

([dagger]) ROC area compares the performance of the BISG
probability membership in a particular race or ethnic group as a
continuous score (as in a diagnostic test score) compared to the
true dichotomous value for the child's membership in that group
(i.e., 1 if the child is a member of that race/ ethnic group,
versus 0 if the child is not).

([double dagger]) Reports the percent of children in each
race/ethnic group for which the BISG algorithm assigned the highest
probability to the correct group. This metric corresponds to the
less accurate dichotimization of BISG probabilities
(Adjaye-Gbewonyo et al. 2014).

([section]) Population-weighted average of area under the receiver
operating characteristic curve. This metric corresponds to the more
accurate direct uses of BISG probabilities (Elliott et al. 2009).

Table 4: Estimated Proportion of Races by Method of Imputation
in the Sample Cohort (Excludes Multiracial Designations)

                         Before Imputation       Traditional Imputation

                       Parent-
                      Report (%)     Exclude
Groups                 ([double    Missing (%)                   With
([dagger])             dagger])    ([section])   No Site (%)   Site (%)

Hispanic                  5.1          4.9           4.9          4.8
White                    59.6         67.6          65.2         62.8
Black ([paragraph])      32.5         24.5          26.8         29.3
Asian                     2.8          4.9           3.1          3.1

                         BISG-Enhanced
                           Imputation

Groups                   No        With
([dagger])            Site (%)   Site (%)

Hispanic                 4.9        5.0
White                   61.9       61.4
Black ([paragraph])     30.4       30.7
Asian                    2.8        2.9

Notes: N = 17,105; 10% random sample of the cohort. These results
are not based on simulations.

([dagger]) American Indian/Alaska Natives and multiracial
categories were dropped.

([dagger]) Parent-report reflects the actual report of
race/ethnicity in the study practices prior to setting data to
missing.

([section]) Data were assigned to missing in a nonrandom fashion
and then imputed using standard and census enhanced imputation
methods.

([paragraph]) Black race was significantly under-represented after
data were set to missing. Adding additional predictors (primary
care practice site and BISG probabilities) to multiple imputation
partially recovered the true proportions.

Table 5: Bias of Different Methods of Estimating Racial/Ethnic
Differences: True Values and Estimates by Method for Continuous
and Binary Outcomes

Race/Ethnicity        True Value        All Data *

Continuous outcome
([dagger])
  Black                  -10       -10.0 [-10.3, -9.71]
  Hispanic               +10        9.97 [9.52, 10.6]
Binary outcome
([double dagger])
  Black ([section])      0.5        0.50 [0.48, 0.52]
  Hispanic               2.0        1.99 [1.89, 2.11]

                            Complete          Indicator Variable
Race/Ethnicity           Case Analysis              Method

Continuous outcome
([dagger])
  Black               -2.59 [-2.89, -2.23]   -0.96 [-1.26, -0.70]
  Hispanic             6.88 [6.09, 7.47]      8.21 [7.38, 8.79]
Binary outcome
([double dagger])
  Black ([section])    1.35 [1.29, 1.41]      1.54 [1.48, 1.60]
  Hispanic             1.33 [1.26, 1.42]      1.48 [1.40, 1.58]

                            Multiple            BISG-Enhanced
Race/Ethnicity             Imputation             Imputation

Continuous outcome
([dagger])
  Black               -2.96 [-3.41, -2.49]   -8.05 [-8.31, -7.74]
  Hispanic             7.62 [6.86, 8.29]      8.30 [7.61, 8.88]
Binary outcome
([double dagger])
  Black ([section])    1.30 [1.22, 1.39]      0.65 [0.62, 0.68]
  Hispanic             1.34 [1.27, 1.45]      1.55 [1.45, 1.64]

* All data in the gold-standard dataset were included to confirm
that in the ideal situation with no data missing the regression
will closely estimate the true value.

([dagger]) Median values and interquartile ranges of estimated
coefficients from linear regression in 200 simulations.

([double dagger]) Median values and interquartile ranges of
estimated odds ratios from logistic regression in 200 simulations.

([section]) In the case of the binary outcome bias was extreme for
black race (the true odds ratio was 0.5, but the estimated odds
ratio was more than 1 for complete case analysis, indicator
variable method, and multiple imputation when only patient data was
included). Adding BISG probabilities to multiple-imputation
estimated coefficients (for the continuous outcome) and odds ratios
(for the binary outcome) that were closer to the true value than
the other methods.
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Title Annotation:METHODS BRIEF
Author:Grundmeier, Robert W.; Song, Lihai; Ramos, Mark J.; Fiks, Alexander G.; Elliott, Marc N.; Fremont, A
Publication:Health Services Research
Date:Aug 1, 2015
Words:4922
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