Evidence Limited to Inform Decisions about Maternal-Fetal Surgery (Text Version)

Slide presentation from the AHRQ 2010 conference.

On September 27, 2010, Katherine E. Hartmann made this presentation at the 2010 Annual Conference. Select to access the PowerPoint® presentation (510 KB). Free PowerPoint® Viewer (Plugin Software Help).


Slide 1

Evidence Limited to Inform Decisions about Maternal-Fetal Surgery

Evidence Limited to Inform Decisions About Maternal-fetal Surgery

Katherine E. Hartmann, MD, PhD
Lucius E. Burch Chair, Obstetrics and Gynecology
Deputy Director, Institute for Medicine and Public Health
Director, Vanderbilt Evidence-based Practice Center

Slide 2

Technical Brief Intent

Technical Brief Intent

Summarizing the state of the field including:

  • Content of the research
  • Access to fetal surgery
  • Training of providers
  • Ethical aspects
  • Coverage of procedures
  • Ongoing studies

Slide 3

Selection of Target Conditions

Selection of Target Conditions

  • Cardiac malformations
  • Congenital diaphragmatic hernia
  • Myelomeningocele
  • Obstructive uropathy
  • Sacrococcygeal teratoma
  • Thoracic lesions (Congenital Pulmonary Airway Malformations)
  • Twin-twin transfusion syndrome

Slide 4

Review Team and Expert Panel Selection

Review Team & Expert Panel Selection

  • Fetal sonographers/echocardiographers
  • Maternal-fetal medicine specialists
  • Perinatal geneticists
  • Pediatric surgeons
  • "Fetal" surgeons
  • Neonatologists
  • Biomedical ethicists
  • Generalists prenatal care providers
  • Familiarity with perinatal hospice

Slide 5

State of the Evidence Methods

State of the Evidence Methods

  • PubMed®, MEDLINE® (no date limits)
  • English, ≥ 2 cases, condition of interest, fetal intervention, training information, outcomes including quality of life (for mother or fetus)
  • Included RCT, cohorts with comparison, case-control, and case series

Slide 6

State of the Evidence Review Methods

State of the Evidence Review Methods

  • Dual review of abstracts
  • Dual review of articles
  • Extraction of descriptive data into standardized forms:
    • Study design and setting
    • Fetal and maternal inclusion criteria
    • Training of providers
    • Outcomes measured
    • Length of follow up

Slide 7

Disposition of the Literature

Disposition of the Literature

  • Abstracts: 1382
  • Articles: 620
  • Relevant studies: 166
  • Study designs
    • RCTS: 3
    • Cohort studies: 47
    • Case series: 116
  • Studies from US: 74

Slide 8

Cardiac Malformations

Cardiac Malformations

Studies (n=9)

  • Pulmonary atresia with intact ventricular septum–2 case series
  • Aortic stenosis–8 case series
  • Hypoplastic left heart syndrome and intact atrial septum–3 case series

State of the Science

  • 90 pregnancies in literature
  • 1 study provided infant outcomes beyond hospital stay
  • Maternal inclusion criteria poorly reported
  • No data on long term survival or neurologic outcomes

Slide 9

Congenital Diaphragmatic Hernia

Congenital Diaphragmatic Hernia

Studies (n=21)

  • 1 RCT
  • 3 prospective cohorts
  • 5 retrospective cohorts
  • 2 prospective case series
  • 10 retrospective case series

State of the Science

  • Fetal inclusion criteria well-reported; 9 of 21 reported maternal criteria
  • Few measured outcomes beyond hospital stay (8/21)
  • Longest reported infant follow up: 24-48 months
  • Long term maternal outcomes addressed in 2 studies

Slide 10

Overview

Overview

 Cardiac (n=9)Diaphragmatic hernia (n=21)Myelomening-ocele(n=11)Obstructive uropathy (n=25)Sacrococcygeal teratoma (n=5)Thoracic lesions (n=11)Twin-twin transfusion (n=84)Total Literature (n=166)
U.S.6121114462174
European2904134968
Asian00020158
Other100501916

Slide 11

Overview

Overview

Study TypeCardiacDiaphragmatic herniaMyelomening-oceleObstructive uropathySacrococcygeal teratomaThoracic lesionsTwin-twin transfusionTotal Literature
Case series9124141373116
Retro. cohort055847636
Prospective cohort032301311
Randomized clinical trial01000023

Slide 12

Overview

Overview

Last infant assessmentCardiacDiaphragmatic herniaMyelomening-oceleObstructive uropathySacrococcygeal teratomaThoracic lesionsTwin-twin transfusionTotal Literature
Birth6319242247
≤ 6 mos1922021935
> 6 to ≤ 12 mos01410118
> 12 mos28411341951
Unknown0002002325

Slide 13

Outcomes Reported

Outcomes Reported

ConditionChild Developmental StatusAny Maternal Outcomes
Cardiac malformations0/94/9
CDH7/2111/21
Myelomeningocele3/114/11
Obstructive uropathy5/252/25
Sacrococcygeal teratoma03/5
Thoracic lesions2/111/11
Twin-twin transfusion syndrome13/844/84

Slide 14

Challenges for the field Include:

Challenges for the Field Include:

  • Rare conditions with no method to consolidate new cases
  • Cases accrue slowly—innovations change the benefit equation
  • Clinical and research classifications of fetal anomalies vary
  • Spontaneous regression/stabilization occurs
  • Prognosis cannot be determined with high accuracy
  • Changes in the spectrum of disease reduces comparability to prior natural history studies of anticipated outcomes
  • Intervention techniques change rapidly
  • Higher SES patients are over-represented
  • Regionalization of care needs consideration
  • No formal plan for certified training programs exists

Slide 15

Strategies to Reduce Evidence Gaps

Strategies to Reduce Evidence Gaps

  • Continue to develop consensus operational definitions
  • Enhance attention to common data elements
  • Use valid and reliable tools for outcomes
  • Support platforms for cooperative data sharing
  • Use analyses to help define equipoise for trials
  • Develop new methods for funding and cost sharing
  • Establish a consistent set of ethical standards for research in the field

Slide 16

 Challenges for Technical Brief Process

Challenges for Technical Brief Process

  • Content of the research: organizing classification, defining families, resisting urge to cross into outcomes
  • Access to fetal surgery: identifying pool of experts—able to establish via direct communication with those who publish
  • Training of providers: identifying sites—able to establish
  • Ethical aspects: balancing priorities for the report
  • Coverage: identifying accurate, available sources
  • Ongoing studies: documenting trials more achievable than other studies

Slide 17

 Technical Brief Conclusions

Technical Brief Conclusions

  • Limited RCT evidence
  • Literature dominated by case series (70% of studies)
  • Lack of clearly described maternal inclusion criteria and long term follow-up for infant or mother
  • Limited outcomes reported:
  • Literature not typical level of quality of studies or aggregate strength of evidence sufficient to guide care
Current as of December 2010
Internet Citation: Evidence Limited to Inform Decisions about Maternal-Fetal Surgery (Text Version). December 2010. Agency for Healthcare Research and Quality, Rockville, MD. http://www.ahrq.gov/news/events/conference/2010/hartmann/index.html