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

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

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Slide 1

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

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

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

Slide 4

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

• 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

• 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

• 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

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

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

	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.	6	12	11	14	4	6	21	74
European	2	9	0	4	1	3	49	68
Asian	0	0	0	2	0	1	5	8
Other	1	0	0	5	0	1	9	16

Slide 11

Overview

Study Type	Cardiac	Diaphragmatic hernia	Myelomening-ocele	Obstructive uropathy	Sacrococcygeal teratoma	Thoracic lesions	Twin-twin transfusion	Total Literature
Case series	9	12	4	14	1	3	73	116
Retro. cohort	0	5	5	8	4	7	6	36
Prospective cohort	0	3	2	3	0	1	3	11
Randomized clinical trial	0	1	0	0	0	0	2	3

Slide 12

Overview

Last infant assessment	Cardiac	Diaphragmatic hernia	Myelomening-ocele	Obstructive uropathy	Sacrococcygeal teratoma	Thoracic lesions	Twin-twin transfusion	Total Literature
Birth	6	3	1	9	2	4	22	47
≤ 6 mos	1	9	2	2	0	2	19	35
> 6 to ≤ 12 mos	0	1	4	1	0	1	1	8
> 12 mos	2	8	4	11	3	4	19	51
Unknown	0	0	0	2	0	0	23	25

Slide 13

Outcomes Reported

Condition	Child Developmental Status	Any Maternal Outcomes
Cardiac malformations	0/9	4/9
CDH	7/21	11/21
Myelomeningocele	3/11	4/11
Obstructive uropathy	5/25	2/25
Sacrococcygeal teratoma	0	3/5
Thoracic lesions	2/11	1/11
Twin-twin transfusion syndrome	13/84	4/84

Slide 14

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

• 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

• 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

• 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