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Inclusion Cysts after Fetal Spina Bifida Repair: A Third Hit?
Pascal Heye1,2, Ueli Moehrlen3,4,5,6, Luca Mazzone3,4,5,6
1Department of Pediatric Surgery, University Children's Hospital Zurich, Zürich, Switzerland, pascalheye@gmail.com.
Insights
Fetal spina bifida repair (fSBR) can lead to postnatal inclusion cysts (IC). These cysts, and their resection, may cause further neurological and bladder dysfunction in children.
Area of Science:
- Pediatric Surgery
- Neuroscience
- Developmental Biology
Background:
- Fetal spina bifida repair (fSBR) offers benefits like reversed hindbrain herniation and improved ambulation.
- Postnatal inclusion cysts (IC) are a known complication following fSBR.
- The origin of these inclusion cysts remains unclear, with possibilities including dysembryogenic or iatrogenic factors.
Purpose of the Study:
- To investigate the incidence and impact of postnatal inclusion cysts (IC) after fetal spina bifida repair (fSBR).
- To assess the outcomes of IC resection in children who underwent fSBR.
Main Methods:
- A prospective study followed 48 children post-fSBR.
- Assessments included clinical examination, cystometry, and spinal MRI.
- IC resection was indicated for spinal mass, functional decline, pain, or significant growth.
Main Results:
- Inclusion cysts (IC) developed in 30% of children within 2 years of fSBR.
- Six children underwent IC resection due to functional deterioration or cyst growth.
- Post-resection, 67% experienced altered motor function and 100% had neurogenic bladder dysfunction.
Conclusions:
- Systematic follow-up reveals a high incidence of inclusion cysts (IC) after fetal spina bifida repair (fSBR).
- Both the presence of IC and their surgical resection can lead to neurological deficits.
- Inclusion cysts are considered a significant complication, potentially a 'third hit' affecting patient outcomes.
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".
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