1. Children with Orofacial Clefts in Massachusetts: Hospitalization and Associated Costs Emily Lu, MPH 12 th Annual Maternal and Child Health Epidemiology Conference December 8, 2006

2. Co-authors: Judith Weiss, ScD 1 Milton Kotelchuck, PhD, MPH 1 Marlene Anderka, ScD, MPH 2 Diego Wyszynski, MD, PhD 3 Scott Grosse, PhD 4 Raul Garcia, DMD 5 Wanda Barfield, MD, MPH 4 Howard Cabral, PhD, MPH 1 Cathleen Higgins 2 Stephen Evans, MPH 1 Susan Manning, MD, MPH 2 Jessica Taubner 1 1 BU School of Public Health, 2 MA Dept. of Public Health, 3 AMGEN, 4 CDC, 5 BU School of Dental Medicine

3. 3 Background: Orofacial Clefts Cleft lip and/or cleft palate (CL/P) Among the most common birth defects in the United States –~6,800 infants/year; 10.48 per 10,000 live births* Correctable defects that often require multiple medical treatments Data on medical costs for CL/P are sparse and outdated (Harris 1997, Waitzman 1994) *MMWR, 1/6/2006

4. 4 Objective To assess hospital utilization and associated costs among children with orofacial clefts and compare to all other children born in Massachusetts

5. 5 Data Source Population-based cohort study MA Pregnancy to Early Life Longitudinal (PELL) Data System Birth and infant death certificates* linked to infant’s hospital discharge birth record^ Post-birth hospital discharge records linked to infant’s birth record up to 2 years postpartum Birth certificates linked to MA Birth Defects Monitoring Program (MBDMP) * Vital records from MA Dept. of Public Health, Registry of Vital Records & Statistics ^ Hospital Discharge Data from Division of Health Care Finance and Policy

6. 6 Study Population All children born in MA hospitals to resident mothers during 1998*–2002 who were alive at age 2 years Cases: Infants with diagnoses of CL/P identified by MBDMP (N = 476) Comparison group: All other MA children without craniofacial malformations** (N = 377,381) * Eastern MA: 1/1/98 to 8/31/98; statewide thereafter ** Craniofacial malformations: orofacial cleft, craniosynostosis, anotia/microtia

7. 7 STUDY GROUP: BC 511 cases 1 born out of state HD Birth 503 cases (98.4% linkage rate) 27 died < 2 years old (5%) MBDMP 512 cases 8 cases not linked to HD birth record 476 Cases

8. 8 COMPARISON GROUP: HD Birth 379,729 children (97.7% linkage) Death Certificate 1,699 died < 2 years old (0.4%) BC 388,540 children 8,811 children unlinked 649 craniofacial malformation cases 377,381 children

9. 9 Case Classification Isolated: infants with CL/P as major defects +/- minor defects Non-isolated: infants with CL/P + sequences, syndromes, or multiple major defects

10. 10 Hospitalization Definitions Birth hospital stay: –Initial birth hospitalization –Transfer to another hospital ≤ 1 day of discharge –Readmission(s) to same or different hospitals ≤ 1 day of discharge from the transfer hospitalization Post-Birth (PB) hospital stay: All hospitalization(s) after the birth hospital stay up to age 2 years

11. 11 Methods Outcome variables: –# of days hospitalized and hospital costs (birth to age 2 years) Cost (Total charges) x (Hospital-specific cost-to-charge ratios*) Incremental cost (Costs for case-patients) – (Costs for comparison group) Statistics: Mean and total * included capitol costs and adjusted for inflation in 2003 dollars

12. 12 Post-Birth Hospitalizations First 2 Years of Life Cases: 78.6% (374) had at least one post- birth hospitalization Comparison group: 8.4% (31,807) had at least one post-birth hospitalization PELL, 2006

13. 13 Distribution of Children with Orofacial Clefts* by Defect Type and Case-Classification TotalIsolatedNon-Isolated NCol %NRow %N Total Cases 476100.034171.613528.4 Palate only 21244.510850.910449.1 Lip only 10421.89793.376.7 Palate and Lip 16033.613685.02415.0 * Born in MA hospitals to resident mothers, 1998-2002. PELL, 2006

14. 14 Mean # Hospital Days First 2 Years of Life 4.0 PELL, 2006 7.6 26.1 12.9 *PB = Post-Birth

15. 15 Mean # Hospital Days First 2 Years of Life Non-IsolatedIsolated PELL, 2006 *PB = Post-Birth

16. 16 Mean Cost* of Hospitalization First 2 Years of Life PELL, 2006 $11,988 $44,185 $2,503 $21,090 *Included capitol costs and adjusted for inflation in 2003 dollars **PB = Post-Birth

17. 17 Mean Cost* of Hospitalization First 2 Years of Life Non-IsolatedIsolated PELL, 2006 *Included capitol costs and adjusted for inflation in 2003 dollars

18. 18 Results Mean incremental hospital costs from birth to age 2: Total CL/PIsolatedNon-isolated $18,587$9,485$41,682 Total hospital expenditure during the first 2 years of life for CL/P children –$10 million dollars (1998-2004) PELL, 2006

19. 19 Summary Linkage of MBDMP and a population-based data system (PELL) provides hospital use and associated cost data on children with birth defects Most children with CL/P were hospitalized at least once during their first 2 years of life Longer hospital stays and higher costs for children with non-isolated CL/P (birth and post- birth hospitalization) Isolated CL/P defects also have increased costs.

20. 20 Limitations Costs understate total costs of hospitalization –Included hospital facility fees only; physician fees are not part of the hospital discharge records No data were available for other medical costs (e.g. outpatient care, prescription drug) Linked data quality

21. 21 Next Steps Evaluate health care utilization and costs by sociodemographic factors Assess Early Intervention Program use and costs Examine children with other craniofacial malformations: craniosynostosis and anotia/microtia

22. 22 Acknowledgements Massachusetts Dept. of Public Health (MDPH) Sally Fogerty Joseph Burgio Boston University School of Public Health Mark McLaughlin Email: Emily.Lu@state.ma.usEmily.Lu@state.ma.us Funding Sources: Center for Birth Defects Research and Prevention, MDPH, contract # INTF 3122 H25 W64 207007; ASPH/Centers for Disease Control cooperative agreement #S3485-23/23

23. 23 Extra Slides

24. 24 Distribution of Children with Orofacial Clefts* by Defect Type and Case- Classification TotalIsolated Only Isolated w/ MinorNon-Isolated NCol %NRow %N N Total Cases 476100.028159.06012.613528.4 Palate only 21244.58339.22511.810449.1 Lip only 10421.88884.698.776.7 Palate and Lip 16033.611068.82616.32415.0 * Born in MA hospitals to resident mothers, 1998-2002. PELL, 2006

25. 25 Results Median # of days hospitalized during first 2 years of life: –Isolated CL/P vs. comparison group3x longer –Non-isolated CL/P vs. comparison group6x longer

26. 26 Total # Hospital Days and Total Cost* First 2 Years of Life N Total # of Days (Birth + PB**)N^ Total $ (Birth +PB) Total Cases4766,125474$9,996,799 Isolated3412,606340$4,075,998 Non-Isolated1353,519134$5,920,801 Comparison Group377,3811,502,184376,646$942,641,939 * Total charge multiplied by cost-to-charge ratios including capitol costs and adjusted for inflation, in 2003 $s. ** Post-birth. ^ Children wit missing or $0 cost data for any hospitalization are excluded. PELL, 2006