Inclusion Cysts after Fetal Spina Bifida Repair: A Third Hit?

Autor:	Heye P; Department of Pediatric Surgery, University Children's Hospital Zurich, Zürich, Switzerland, pascalheye@gmail.com.; Children's Research Center, University Children's Hospital Zurich, Zürich, Switzerland, pascalheye@gmail.com., Moehrlen U; Department of Pediatric Surgery, University Children's Hospital Zurich, Zürich, Switzerland.; Children's Research Center, University Children's Hospital Zurich, Zürich, Switzerland.; Division of Fetal Surgery, University Children's Hospital Zurich, Zürich, Switzerland.; The Zurich Center for Fetal Diagnosis and Therapy, University of Zurich, Zürich, Switzerland., Mazzone L; Department of Pediatric Surgery, University Children's Hospital Zurich, Zürich, Switzerland.; Children's Research Center, University Children's Hospital Zurich, Zürich, Switzerland.; Division of Fetal Surgery, University Children's Hospital Zurich, Zürich, Switzerland.; The Zurich Center for Fetal Diagnosis and Therapy, University of Zurich, Zürich, Switzerland., Weil R; Department of Pediatric Surgery, University Children's Hospital Zurich, Zürich, Switzerland.; Children's Research Center, University Children's Hospital Zurich, Zürich, Switzerland.; Division of Pediatric Neurosurgery, University Children's Hospital Zurich, Zürich, Switzerland., Altermatt S; Department of Pediatric Surgery, University Children's Hospital Zurich, Zürich, Switzerland.; Children's Research Center, University Children's Hospital Zurich, Zürich, Switzerland.; Division of Pediatric Neurosurgery, University Children's Hospital Zurich, Zürich, Switzerland., Wille DA; Children's Research Center, University Children's Hospital Zurich, Zürich, Switzerland.; Division of Pediatric Neurology, University Children's Hospital Zurich, Zürich, Switzerland., Scheer I; Children's Research Center, University Children's Hospital Zurich, Zürich, Switzerland.; Division of Diagnostic Imaging, University Children's Hospital Zurich, Zürich, Switzerland., Meuli M; Department of Pediatric Surgery, University Children's Hospital Zurich, Zürich, Switzerland.; Children's Research Center, University Children's Hospital Zurich, Zürich, Switzerland.; Division of Fetal Surgery, University Children's Hospital Zurich, Zürich, Switzerland.; The Zurich Center for Fetal Diagnosis and Therapy, University of Zurich, Zürich, Switzerland., Horst M; Department of Pediatric Surgery, University Children's Hospital Zurich, Zürich, Switzerland.; Children's Research Center, University Children's Hospital Zurich, Zürich, Switzerland.; Division of Pediatric Urology, University Children's Hospital Zurich, Zürich, Switzerland.
Jazyk:	angličtina
Zdroj:	Fetal diagnosis and therapy [Fetal Diagn Ther] 2019; Vol. 46 (1), pp. 38-44. Date of Electronic Publication: 2018 Sep 10.
DOI:	10.1159/000491877
Abstrakt:	Introduction: Fetal spina bifida repair (fSBR) has proven effective in the reversibility of hindbrain herniation, lower rate of shunt-dependent hydrocephalus, and independent ambulation. Besides distinct advantages, there are also concerns related to fSBR. One of these is the postnatal occurrence of inclusion cysts (IC). Methods: In a prospective study, 48 children who underwent fSBR were followed up. Postnatal assessment included clinical examination, cystometry, and spinal MRI. Indication for IC resection was the evidence of a spinal mass on MRI in the presence of deteriorating motor or bladder function, pain, or considerable growth of the IC. Results: Fourteen children (30%) developed IC, all within the first 2 years of life. Six children underwent IC resection; 4 children due to deteriorating function, 2 children due to doubling of the mass on MRI within 1 year. Following IC resection, 4/6 children (67%) demonstrated altered motor function and 6 children (100%) were diagnosed with neurogenic bladder dysfunction. Conclusions: Systematic follow-up of patients with a history of fSBR revealed a high incidence of IC. Whether these are of dysembryogenic or iatrogenic origin, remains unclear. Since both IC per se and IC resection may lead to loss of neurologic function, IC can be considered a "third hit". (© 2018 S. Karger AG, Basel.)
Databáze:	MEDLINE
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