'We did everything we could'- A qualitative study exploring the acceptability of maternal-fetal surgery for spina bifida to parents

OBJECTIVE: To explore the concepts and strategies parents employ when considering maternal-fetal surgery (MFS) as an option for the management of spina bifida (SB) in their fetus, and how this determines the acceptability of the intervention. METHODS: A two-centre interview study enrolling parents whose fetuses with SB were eligible for MFS. To assess differences in acceptability, parents opting for MFS (n=24) were interviewed at three different moments in time: prior to the intervention, directly after the intervention and 3-6 months after birth. Parents opting for termination of pregnancy (n=5) were interviewed only once. Themes were identified and organised in line with the framework of acceptability. RESULTS: To parents opting for MFS, the intervention was perceived as an opportunity that needed to be taken. Feelings of parental responsibility drove them to do anything in their power to improve their future child's situation. Expectations seemed to be realistic yet were driven by hope for the best outcome. None expressed decisional regret at any stage, despite substantial impact and, at times, disappointing outcomes. For the small group of participants, who decided to opt for termination of pregnancy (TOP), MFS was not perceived as an intervention that substantially could improve the quality of their future child's life. CONCLUSION: Prospective parents opting for MFS were driven by their feelings of parental responsibility. They recognise the fetus as their future child and value information and care focusing on optimising the child's future health. In the small group of parents opting for TOP, MFS was felt to offer insufficient certainty of substantial improvement in quality of life and the perceived severe impact of SB drove their decision to end the pregnancy This article is protected by copyright. All rights reserved

Publications applying rat stereotaxy and data derived from them

Strains: F=Fischer; L=Lewis; LE=Long-Evans; LH=Lister-hooded; nr=not reported; SD=Sprague-Dawley; W=Wistar. Sex: F=female, F+M=female and male; M=male; nr=not reported. Number of subjects: nr=not reported. Laterality: B=bilateral, L=left; nr=not reported; R=right. AP coordinates=anteroposterior coordinates ML coordinates=mediolateral coordinates AP/ML reference=stereotaxic reference used for anteroposterior and mediolateral coordinates: B=bregma; CS=calamus scriptorius; IA=interaural line; L=lambda; OC=occipital crest. DV coordinates=dorsoventral coordinates DV reference=stereotaxic reference used for dorsoventral coordinates: B=bregma; D=dura/brain surface; IA=interaural line; S=skull. Implant/procedure: BPS=biopsy; C=cannula implantation; ELE=electrode implantation; EXT=extraction; FUS=focused ultrasound; INJ=injection; MD=microdialysis catheter implantation; OCT=optical coherence tomography probe implantation; THM=thermode implantation. Accuracy verification method: EP=electrophysiology; H=histology; MR=magnetic resonance imaging; nr=not reported; *=intraventricular/intracisternal injection. Stereotaxic atlas: K=König and Klippel; nr=not reported; P=Paxinos. Grey text represents deducted data with some uncertainty due to insufficient reporting. Grey highlighted text represents data deducted from growth curves as explained in the Material and methods section

Data for: Stereotaxy in rat models: current state of the art.

Selected publications on rat stereotaxy and data obtained from them. Strains: F=Fischer; L=Lewis; LE=Long-Evans; LH=Lister-hooded; nr=not reported; SD=Sprague-Dawley; W=Wistar. Sex: F=female, F+M=female and male; M=male; nr=not reported. Number of subjects: nr=not reported. Laterality: B=bilateral, L=left; nr=not reported; R=right. AP coordinates=anteroposterior coordinates ML coordinates=mediolateral coordinates AP/ML reference=stereotaxic reference used for anteroposterior and mediolateral coordinates: B=bregma; CS=calamus scriptorius; IA=interaural line; L=lambda; OC=occipital crest. DV coordinates=dorsoventral coordinates DV reference=stereotaxic reference used for dorsoventral coordinates: B=bregma; D=dura/brain surface; IA=interaural line; S=skull. Implant/procedure: BPS=biopsy; C=cannula implantation; ELE=electrode implantation; EXT=extraction; FUS=focused ultrasound; INJ=injection; MD=microdialysis catheter implantation; OCT=optical coherence tomography probe implantation; THM=thermode implantation. Accuracy verification method: EP=electrophysiology; H=histology; MR=magnetic resonance imaging; nr=not reported; *=intraventricular/intracisternal injection. Stereotaxic atlas: K=König and Klippel; nr=not reported; P=Paxinos. Grey text represents deducted data with some uncertainty due to insufficient reporting. Grey highlighted text represents data deducted from growth curves as explained in the Material and methods section

Deep brain stimulation for pantothenate kinase-associated neurodegeneration: A meta-analysis

Background: Pantothenate kinase-associated neurodegeneration is a rare autosomal-recessive disorder, characterized by progressive neurodegeneration associated with brain iron accumulation. DBS has been trialed to treat related movement disorders, particularly dystonia. The objective of this study was to determine the outcome and safety of DBS for pantothenate kinase-associated neurodegeneration. Methods: We performed a meta-analysis using independent participant data (n = 99) from 38 articles. Primary outcome was change in movement and disability scores of the Burke-Fahn-Marsden Dystonia Rating Scale 1 year postoperatively. Secondary outcomes were response rate and complications. Results: Patients with classic-type (n = 58) and atypical-type (n = 15) pantothenate kinase-associated neurodegeneration were operated on at a median age of 11 and 31 years, respectively (P 1 year following GPi-DBS or with other DBS targets. Overall, small sample sizes limited generalizability. Conclusions: This meta-analysis provides level 4 evidence that GPi-DBS for pantothenate kinase-associated neurodegeneration may improve dystonia movement scores in classic type and atypical type and disability scores in atypical type 1 year postoperatively. © 2019 International Parkinson and Movement Disorder Society. © 2019 International Parkinson and Movement Disorder Societ

Patient empowerment improves follow‐up data collection after fetal surgery for spina bifida: institutional audit

Objectives To define and grade fetal and maternal adverse events following fetal surgery for spina bifida and to report on the impact of engaging patients in collecting follow-up data. Methods This prospective single-center audit included one hundred consecutive patients undergoing fetal surgery for spina bifida from the first onwards. In our setting, patients return to their referring unit for further pregnancy care and delivery. On discharge, referring hospitals were requested to return outcome data. For this audit, we prompted patients and referring hospitals for missing outcomes. Outcomes were categorized as missing, returned spontaneously or following additional request, and as either provided by the patients or referring center. Postoperative maternal and fetal complications until delivery were defined and graded according to the Maternal and Fetal Adverse Event Terminology (MFAET) and the Clavien-Dindo classification. Results There were no maternal deaths and seven (7%) severe maternal complications (anemia in pregnancy, postpartum hemorrhage, pulmonary edema, lung atelectasis, urinary tract obstruction, and placental abruption). No uterine ruptures were reported. Perinatal death occurred in 3% and other severe fetal complications in 15% (perioperative fetal bradycardia/cardiac dysfunction, fistula-related oligohydramnios, and preterm rupture of membranes <32 weeks). Preterm rupture of membranes occurred in 42% and overall, delivery took place at a median gestational age of 35.3 weeks [IQR 34.0-36.6]. Information following additional request, both from centers, but mainly through patients reduced missing data by 21% for the gestational age at delivery, by 56% for the uterine scar status at birth, and by 67% for the shunt insertion at 12 months. Compared to the generic Clavien-Dindo classification, the Maternal and Fetal Adverse Event Terminology ranked complications in a clinically more relevant way. Conclusions The nature and rate of severe complications were similar to those reported in other larger series. Spontaneous return of outcome data by referring centers was low, yet patient empowerment improved data collection

Patient empowerment improves follow-up data collection after fetal surgery for spina bifida: institutional audit

OBJECTIVES: To define and grade fetal and maternal adverse events following fetal surgery for spina bifida and to report on the impact of engaging patients in collecting follow-up data. METHODS: This prospective single-center audit included one hundred consecutive patients undergoing fetal surgery for spina bifida from the first onwards. In our setting, patients return to their referring unit for further pregnancy care and delivery. On discharge, referring hospitals were requested to return outcome data. For this audit, we prompted patients and referring hospitals for missing outcomes. Outcomes were categorized as missing, returned spontaneously or following additional request, and as either provided by the patients or referring center. Postoperative maternal and fetal complications until delivery were defined and graded according to the Maternal and Fetal Adverse Event Terminology (MFAET) and the Clavien-Dindo classification. RESULTS: There were no maternal deaths and seven (7%) severe maternal complications (anemia in pregnancy, postpartum hemorrhage, pulmonary edema, lung atelectasis, urinary tract obstruction, and placental abruption). No uterine ruptures were reported. Perinatal death occurred in 3% and other severe fetal complications in 15% (perioperative fetal bradycardia/cardiac dysfunction, fistula-related oligohydramnios, and preterm rupture of membranes <32 weeks). Preterm rupture of membranes occurred in 42% and overall, delivery took place at a median gestational age of 35.3 weeks [IQR 34.0-36.6]. Information following additional request, both from centers, but mainly through patients reduced missing data by 21% for the gestational age at delivery, by 56% for the uterine scar status at birth, and by 67% for the shunt insertion at 12 months. Compared to the generic Clavien-Dindo classification, the Maternal and Fetal Adverse Event Terminology ranked complications in a clinically more relevant way. CONCLUSIONS: The nature and rate of severe complications were similar to those reported in other larger series. Spontaneous return of outcome data by referring centers was low, yet patient empowerment improved data collection. This article is protected by copyright. All rights reserved

Acute low frequency dorsal subthalamic nucleus stimulation improves verbal fluency in Parkinson's disease

Background: Parkinson's disease (PD) is a common neurodegenerative disorder that results in movement-related dysfunction and has variable cognitive impairment. Deep brain stimulation (DBS) of the dorsal subthalamic nucleus (STN) has been shown to be effective in improving motor symptoms; however, cognitive impairment is often unchanged, and in some cases, worsened particularly on tasks of verbal fluency. Traditional DBS strategies use high frequency gamma stimulation for motor symptoms (∼130 Hz), but there is evidence that low frequency theta oscillations (5–12 Hz) are important in cognition. Methods: We tested the effects of stimulation frequency and location on verbal fluency among patients who underwent STN DBS implantation with externalized leads. During baseline cognitive testing, STN field potentials were recorded and the individual patients’ peak theta frequency power was identified during each cognitive task. Patients repeated cognitive testing at five different stimulation settings: no stimulation, dorsal contact gamma (130 Hz), ventral contact gamma, dorsal theta (peak baseline theta) and ventral theta (peak baseline theta) frequency stimulation. Results: Acute left dorsal peak theta frequency STN stimulation improves overall verbal fluency compared to no stimulation and to either dorsal or ventral gamma stimulation. Stratifying by type of verbal fluency probes, verbal fluency in episodic categories was improved with dorsal theta stimulation compared to all other conditions, while there were no differences between stimulation conditions in non-episodic probe conditions. Conclusion: Here, we provide evidence that dorsal STN theta stimulation may improve verbal fluency, suggesting a potential possibility of integrating theta stimulation into current DBS paradigms to improve cognitive outcomes