California Newborn Screening Program. Long Term Follow-up Data System for Metabolic Disorders



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California Newborn Screening Program Long Term Follow-up Data System for Metabolic Disorders Prepared by Lisa B Feuchtbaum, Dr.P.H., M.P.H. Genetic Disease Screening Program State of California Health and Human Services Agency California Department of Public Health

Introduction California Long Term Follow-Up Data System Surveillance of diagnosed cases through age five using an annual survey instrument: Metabolic Center Annual Patient Summary (MCAPS) Availability of ongoing care and management Clinical outcomes and developmental assessment Impact on health care utilization

A Brief History Summer, 2005: Web-based Screening Information System (SIS) go-live MS/MS screening go-live Summer, 2007: Implementation of long term follow up data system using Metabolic Center Annual Patient Summaries (MCAPS)

The Metabolic Center Annual Patient Summary (MCAPS) Yearly assessment, or snapshot, to capture status of child at the end of each completed year of life Web-based Screening Information System (SIS) adds each child s name to the MCAPS pending case list after the child s birthday After the report is completed the name falls off the pending list and will reappear the following year - through age five.

MCAPS Data Elements Clinic follow-up status Services provided by the metabolic center in previous year Date of last visit/interaction with patient that occurred in previous year Total number of patient visits to the metabolic center in previous year Total number of hospitalizations/emergency room visits, with length of stay and reason for admission, in previous year Has patient been symptomatic in previous year? Persistent health problems/symptoms in previous year Treatments/therapies/strategies prescribed in previous year Assessment of patient s development and function (speech/cognitive/physical/fine motor/gross motor) Loss of skills that had been previously achieved Global Health Assessment (scale: 1=critical through to 6=excellent)

Count of MCAPS Entered in SIS by Disorder Type & Age of Child (8/1/2007 2/9/2009) 1 st Year 2 nd Year 3 rd Year 4 th Year 5 th Year All 3MCC 19 14 4.. 37 ARG. 1... 1 ASAL 2.... 2 BD Profound 8 8 CPT-2 3.... 3 CTD/CUD 8 8 4.. 20 Cit:type_I 3 5 2.. 10 Cit:type_lI 1... 1 GA-1 10 6 3.. 19 GA-2/MAD 3 1 1.. 5 HCY 1.... 1 IBDHD 6 1 1.. 8 IVA 6 6 3.. 15 LCHADD 1 1... 2 MAT 2 4... 6 MCADD 40 29 4.. 73 MCD 2 2

Count of MCAPS Entered in SIS by Disorder Type & Age of Child (8/1/2007 2/9/2009) 1 st Year 2 nd Year 3 rd Year 4 th Year 5th_Year All MMA:Cbl AB 1 2... 3 MMA:Cbl CDF 10 7 2.. 19 MMA:mut- 9 3 2.. 14 MMA:mut0 4 5 1.. 10 MSUD 8 3 2.. 13 MA (Malonic Aciduria) 1 1 PKU 19 24 22 26 28 119 PKU Variant 15 13 11 15 6 60 Prolinemia_Type_I. 2... 2 Prolinemia_Type_II 1 1 Propionic_acidemia 2 1... 3 SCADD 21 16 5.. 43 VLADD 11 8 2.. 21 Total (to date) * 234 175 71 42 35 557 * Note: Counts of all diagnosed disorder types are not displayed

MCAPS Summary Patient Clinic Status: All Disorders Combined (8/1/2007 2/9/2009) 1 st Year 2 nd Year 3 rd Year 4th Year 5th Year (n=234) (n=175) (n=71) (n=42) (n=35) Active Patient 84% 80% 86% 83% 86% Lost To Follow-up 4% 5% 4% 5% - Refused Treatment 3% <1% - - 3% Treatment Deemed Not Necessary 4% 7% 6% 7% 3% Patient Died* 2% <1% - - - Moved out of state 1% 5% 3% 2% 3% Transferred to another center 2% 1% 1% 2% 6%

MCAPS Summary Developmental Milestones: All Disorders Combined (8/1/2007 2/9/2009) Age Appropriate Speech Physical Mental/ Cognitive Gross Motor Fine Motor Year 1 (n=234) Year 2 (n=175) Year 3 (n=71) Year 4 (n=42) Year 5 (n=35) 81.2% 84.2% 82.5% 81.6% 80.3% 69.7% 76.0% 75.4% 76.0% 76.6% 69.0% 81.7% 76.1% 83.1% 77.5% 81.0% 85.7% 80.1% 88.1% 88.1% 85.7% 94.3% 91.4% 94.3% 97.1%

MCAPS Summary Developmental Milestones: All Disorders Combined (8/1/2007 2/9/2009) Moderate and/or Severe Delay Speech Physical Mental/ Cognitive Gross Motor Fine Motor Year 1 (n=234) Year 2 (n=175) Year 3 (n=71) Year 4 (n=42) Year 5 (n=35) 3.9% 2.6% 2.6% 5.1% 4.3% 7.4% 3.4% 4.6% 5.7% 5.2% 8.4% 4.2% 6.0% 2.8% 1.4% - - - - - 2.9% - - - -

MCAPS Summary Other Outcome Measures: All Disorders Combined (8/1/2007 2/9/2009) Symptoms associated with disorder Loss of Previous Skills No Hospitalizations Reported Year 1 (n=234) Year 2 (n=175) Year 3 (n=71) Year 4 (n=42) Year 5 (n=35) 25.2% 3.0% 70.9% 28.6% 1.8% 80.0% 19.7% - 90.1% 14.3% - 100% 14.3% - 100%

MCAPS Summary Disorders With Highest Health Services Utilization (8/1/2007 2/9/2009) Utilization Measures Year One Year Two Year Three Avg. Days Hospitalized 66 days - PA 27.2 days - MMA:mut0 12.5 days - MMA:Cbl CDF; 27.5 days - MMA:mut0 6 days - PA 12 days - MMA:mut0 Avg. # Hospitalizations 5 - Cit:type I 3 - ARG 4 - MMA:mut0; 3 - MMA:mut0 2 - PA; MSUD 3 - MMA:mut-; MSUD Avg. # ER Visits 5 - Cit:type I 13 - VLCADD 4 - MMA:mut-; MMA:mut0 2 - MMA:mut0; MSUD 2 - CIT:type I; ARG 1 VLCADD; MMA:Cbl CDF MSUD; Cit-type I Avg. # Clinic Visits 11 - MA 8 - LCHADD 5 Cit-type I 11 - MMA:mut0 6 - PA; Cit:type I 4 - MMA:mut-; MMA:Cbl CDF

MCAPS Summary Provider Health Assessment of Child s Health Status (8/1/2007 2/9/2009) Critical Poor Fair Good Very Good Excellent Year 1 (n=234) Year 2 (n=175) Year 3 (n=71) Year 4 (n=42) Year 5 (n=35) 9.0% 9.0% 6.8% 18.8% 32.5% 23.9% 9.7% 12.6% 10.3% 16.0% 26.9% 24.6% 4.2% 14.1% 8.5% 14.1% 32.4% 26.8% 11.9% 7.1% 2.4% 7.1% 19.1% 52.4% 14.3% 2.9% 2.9% 5.7% 31.4% 42.9%

Health Impacts With & Without Screening: MCADD in the absence of newborn screening for MCADD, premature death or serious disability occurs in 20% to 25% of children with the disorder. (Grosse, Khoury, Greene, Crider, & Pollitt, 2006) 33% of survivors will have irreversible neurologic damage (Iafolla, Thompson, & Roe, 1994; Pollitt & Leonard, 1998). MCADD Year One (n=40) Year Two (n=32) In active care at follow-up center 92.5% 81.3% Reported deaths* 0% 0% No disorder related symptoms 67.5% 53.1% Age appropriate cognitive function 97.5% 84.4%/3.1% mild # Clinic Visits 0 2 = 37.5% 3-5 = 45% # Hospitalizations 0-1 = 90% 2-3 = 10% 0-2 = 71.9% 3-4 = 25% 0-1 = 90.6% 2 = 9.4% * 3 additional MCADD cases died before follow-up was initiated

Conclusion This preliminary review demonstrates the feasibility and utility of the California Long Term Follow Up Data System using Metabolic Center Annual Patient Summaries The surveillance approach addresses: Availability of ongoing care and management Clinical outcomes and developmental assessment Impact on health care utilization

Acknowledgements Special thanks to staff at the following State-contracted metabolic follow-up centers who have made this project possible. Cedar-Sinai Medical Center Children s Hospital and Research Center-Oakland Children s Hospital Central California Children s Hospital Los Angeles Children s Hospital of Orange County Children s Hospital San Diego Health Center Harbor/UCLA Medical Center Kaiser Permanente-Northern California Kaiser Permanente- Southern California LAC/USC Medical Center Stanford University Medical Center Sutter Memorial Hospital, Sacramento UC Davis Medical Center UC San Francisco Medical Center UCLA Medical Center