Fetal Operation Followed By Caesarean Section May Have A Beneficial Effect Upon Neuromuscular Function In Spina Bifida Aperta
Authors: Renate Verbeek[1], Axel Heep[2], Natasha Maurits[1], Reinhold Cremer[3], Oebele Brouwer[1], Johannes van der Hoeven[1], Deborah Sival[4]
Background
Spina bifida aperta (SBA) is associated with neurological
dysfunction cranial and caudal to the meningomyelocele (MMC). Fetal
surgery may ameliorate cranial abnormalities, but effects upon
neuromuscular function caudal to the MMC are unclear. SBA myotomes
cranial to the MMC are influenced by cerebral dysfunction, whereas
myotomes caudal to the MMC are additionally influenced by the MMC.
Increased muscle ultrasound density (MUD) reflects neuromuscular
damage. By the intra-individual difference in MUD (dMUD =
[MUDcaudal] - [MUDcranial]) the effect by the MMC upon
neuromuscular integrity is derived. In the present study, we aimed
to compare dMUD and neuromuscular function between fetally and
postnatally operated SBA patients.
Materials and Methods
We compared dMUD and neuromuscular function in 6 (age- and MMC-)
matched pairs of fetally and postnatally operated SBA patients [age
0-3.5 years; lumbar (6) and lumbar-sacral (6) MMC]. In all
patients, quadriceps muscle was innervated cranial- and calf muscle
caudal- to the MMC. Fetally operated patients were delivered by
caesarean section (CS) at Bonn; postnatally operated patients were
delivered vaginally at Groningen.
Results
dMUD was smaller in fetally than in postnatally operated SBA
patients [10 (-11 to 37) vs 28 (4 to 47), medians (ranges);
p<0.05]. Comparing SBA patients, indicated more preserved
neuromuscular function in fetally than postnatally operated
patients [median difference: 1 and 1.5 myotome (ranges 1-3); motor
and sensory function resp.; p<0.05)].
Conclusions
In SBA, dMUD provides a diagnostic tool to compare neuromuscular
integrity between treatment groups. Present data suggest that fetal
operation followed by CS has some beneficial effect upon
neuromuscular outcome caudal to the MMC. Future assessment in SBA
patients (treated by postnatal operation followed by CS) may allow
further differentiation between the effect by operation and way of
delivery upon neuromuscular outcome.
1Department of Neurology and
4Department of Paediatrics, University Medical Center
Groningen, University of Groningen, Hanzeplein 1, PO Box 30001,
9700RB Groningen, the Netherlands
2Department of Neonatology, University
of Bonn, Sigmund Freud Str. 25, 53105, Bonn, Germany
3Department of Pediatrics, Children's
Hospital Cologne, Amsterdamer Str. 59, 50735 Cologne,
Germany
E-mail: r.j.verbeek@neuro.umcg.nl
