Document Detail


In utero repair of myelomeningocele: experimental pathophysiology, initial clinical experience, and outcomes.
MedLine Citation:
PMID:  12912746     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
HYPOTHESIS: Experimental work raises the possibility that in utero repair of myelomeningocele (MMC) may improve lower extremity, bladder, and bowel function, ameliorate the Arnold-Chiari malformation, and decrease the need for postnatal shunting. DESIGN: We previously developed fetal lamb models to create and reverse lower extremity damage and the Arnold-Chiari malformation in utero. We then applied our extensive experience with fetal surgery, including fetal endoscopic (fetoscopic) surgical manipulation, to develop techniques for MMC repair. SETTING: A tertiary referral center. PATIENTS: All patients treated between 1998 and 2002 for a prenatally diagnosed MMC. INTERVENTIONS: Either fetoscopic MMC repair, fetoscopic patch repair, or limited maternal hysterotomy and microsurgical 3-layered fetal MMC repair was performed. MAIN OUTCOME MEASURES: Gestational age at delivery, survival, neurologic outcome, and need for ventricular shunting at 1 year. RESULTS: Complete fetoscopic repair was accomplished in 1 fetus. Two other fetuses underwent partial fetoscopic procedures. The remaining 10 patients underwent limited maternal hysterotomy and microsurgical 3-layered fetal MMC repair. Four of 13 patients died, and the mean gestational age at delivery of 11 fetuses born alive was 31 weeks. Five of 9 required ventricular shunting by age 1 year. In 2 patients, lower extremity function improved by more than 2 vertebral levels compared with prenatal ultrasonography. Five of 10 patients who lived longer than 3 weeks required postnatal wound revision within 7 days after birth. CONCLUSIONS: Fetoscopic repair, although feasible, does not yet yield optimal surgical results. Open surgical repair before 22 weeks' gestation is physiologically sound and technically feasible. One third of patients appear to be spared the need for a shunt at age 1 year, but improvement in distal neurologic function is less clear. Additionally, fetal mortality is associated with this procedure. Our results complement the data published by groups at Children's Hospital of Philadelphia, in Pennsylvania, and Vanderbilt University, Nashville, Tenn. A National Institutes of Health-sponsored prospective randomized trial is now underway at these 3 centers to compare fetal repair with postnatal repair.
Authors:
Diana L Farmer; Cornelia S von Koch; Warwick J Peacock; Moise Danielpour; Nalin Gupta; Hanmin Lee; Michael R Harrison
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Archives of surgery (Chicago, Ill. : 1960)     Volume:  138     ISSN:  0004-0010     ISO Abbreviation:  Arch Surg     Publication Date:  2003 Aug 
Date Detail:
Created Date:  2003-08-12     Completed Date:  2003-09-02     Revised Date:  2004-11-17    
Medline Journal Info:
Nlm Unique ID:  9716528     Medline TA:  Arch Surg     Country:  United States    
Other Details:
Languages:  eng     Pagination:  872-8     Citation Subset:  AIM; IM    
Affiliation:
Departments of Surgery and Neurosurgery and the Fetal Treatment Center, University of California, San Francisco, CA 94143-0570, USA. farmerd@surgery.ucsf.edu
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MeSH Terms
Descriptor/Qualifier:
Adult
Animals
Female
Fetal Diseases / physiopathology,  surgery*
Fetoscopy
Fetus / surgery*
Gestational Age
Humans
Hysterotomy
Meningomyelocele / physiopathology,  surgery*
Microsurgery
Pregnancy
Sheep / embryology
Treatment Outcome

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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