Fetal Endoscopic Third Ventriculostomy Is Technically Feasible in Prenatally Induced Hydrocephalus Ovine Model

Peiró, José Luis; Duru, Soner; Fernández-Tomé, Blanca; Peiro, Lucas; Encinas, José Luis; Sánchez‐Margallo, Francisco M.; Oria, Marc

doi:10.1227/neu.0000000000002361

Cited by 6 publications

(5 citation statements)

References 26 publications

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“…In-utero intervention for fetal AS by a purposedesigned ventriculoamniotic shunt and endoscopic third ventriculostomy (ETV) is being investigated in a large animal model. If a benefit is documented in the animal model, demonstrating the ability to make an accurate prenatal diagnosis of fetal AS is required before translation to humans [9,[15][16][17][18].…”

Section: Discussionmentioning

confidence: 99%

Prenatal Diagnosis of Fetal Aqueductal Stenosis: A Multicenter Prospective Observational Study through the North American Fetal Therapy Network

Emery,

Lopa,

Peterson

et al. 2024

Fetal Diagn Ther

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Introduction: A critical component of an evidence-based reassessment of in-utero intervention for fetal aqueductal stenosis (fetal AS) is determining if the prenatal diagnosis can be accurately made at a gestational age amenable to in-utero intervention. Methods: A multicenter, prospective, observational study was conducted through the North American Fetal Therapy Network (NAFTNet). Pregnancies complicated by severe CNS ventriculomegaly (lateral ventricle diameter >15 mm) not secondary to a primary diagnosis (myelomeningocele, encephalocele, etc.) were recruited at diagnosis. Imaging and laboratory findings were recorded in an online REDCap database. After evaluation, investigators were asked to render their degree of confidence in the diagnosis of fetal AS. The prenatal diagnosis was compared to the postnatal diagnosis obtained through neonatal neuroimaging. Performance characteristics of ultrasound and MRI were calculated, as was the mean gestational age at diagnosis. Results: Between April 2015 and October 2022, eleven NAFTNet centers contributed 64 subjects with severe fetal CNS ventriculomegaly. Of these, 56 had both prenatal and postnatal diagnoses recorded. Ultrasound revealed 32 fetal AS true positives, 4 false positives, 7 false negatives, and 13 true negatives rendering a sensitivity of 0.82, a specificity of 0.76, a positive predictive value of 0.89, and a negative predictive value of 0.65. The mean gestational age at diagnosis by ultrasound was 25.5 weeks (std +/- 4.7w). The proportion of agreement (true positive + true negative/n) was highest at 24 weeks gestation. For fetal MRI (n=35), the sensitivity for fetal AS was 0.95, specificity was 0.69, positive predictive value was 0.84, and negative predictive value was 0.90. MRI was performed at 25 weeks on average. Conclusion: The prenatal diagnosis of fetal aqueductal stenosis can be made with accuracy at a gestational age potentially amenable to in-utero intervention. Only 7% of subjects were incorrectly diagnosed prenatally with fetal AS by ultrasound and 11% by MRI. Diagnostic accuracy of fetal AS will likely improve with increased experience.

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Section: Discussionmentioning

confidence: 99%

Prenatal Diagnosis of Fetal Aqueductal Stenosis: A Multicenter Prospective Observational Study through the North American Fetal Therapy Network

Emery,

Lopa,

Peterson

et al. 2024

Fetal Diagn Ther

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“…Triggered by postnatal experience, ETV was preclinially attempted, yet so far it was not translated clinically. 26 In that clinical report, fetuses demonstrating a response to ETV were less likely to have abnormal scores on the Ages and Stages-neurodevelopmental score. Such early successes may reopen the debate on prenatal management of isolated severe ventriculomegaly, in view of today's potential progress in patient selection and surgical techniques.…”

mentioning

confidence: 85%

“…The rationale of in utero ventriculostomy, rather than inserting a non‐valved shunt, is that the former avoids permanent contact between the developing brain and the amniotic cavity, resulting in the dynamics of fluid production and resorption being more physiological. Triggered by postnatal experience, ETV was preclinially attempted, yet so far it was not translated clinically 26 . In that clinical report, fetuses demonstrating a response to ETV were less likely to have abnormal scores on the Ages and Stages‐neurodevelopmental score.…”

Section: Figurementioning

confidence: 99%

The new fetal therapy section awaits your research

Deprest,

Van Mieghem

2023

Prenatal Diagnosis

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The new fetal therapy section awaits your researchAlthough the journal title "Prenatal Diagnosis" may not suggest the interest of our readerships in fetal therapy, we have been satisfying their needs by publishing on medical and surgical fetal treatments since the 1980s. Our aims and scope actually call for authors to submit their "research on developing fetal therapy as well as the wider impact of fetal treatment." 1 Over the last 10 years, the number of papers published in this field across all journals has been growing steeply (Figure 1). This reflects the increasing number of treatment options but also an increased research interest, generating better evidence and stimulating its wider acceptance. Based on a PubMed search, Prenatal Diagnosis has followed that global publication trend, without diluting the quality, eventually capturing a steady share of around 3%-5% of fetal therapy publications, which we deemed relevant to our multidisciplinary readership. 14 With a growing numerator however, the denominator is also increasing. It is for that reason that the Editors have opted to formalize the interest of Prenatal Diagnosis in the field of fetal therapy, by creating a dedicated section of the journal to this growing discipline. Within this section we aim to cover the entire journey of a fetal intervention: from the concept of a new or alternative treatment to its long-term outcomes. This means that the journal is open to original research on the pathophysiology of conditions potentially amenable to fetal therapy (including preclinical models), as well as early clinical experience, including instrument and device development, drug studies, feasibility and safety studies, longer term outcome studies, research on the ethical and social issues associated with fetal therapy as well as documentation of maternal impact. (Re)consideration of (new) conditions for fetal intervention, or the introduction of novel therapies, typically renews interest in the conditions under study. It generates new research into the natural history of disorders as well as into more accurate diagnostic tools and prediction models, which in this day and age will often include the use of molecular tools, genetic profiling and artificial intelligence. Better imaging and prognostication will improve case selection-a conditio sine qua non for fetal therapy. With growing experience, the level of evidence for a given fetal intervention must eventually mature by conducting well-conceived and properly conducted clinical studies with sufficient numbers of participants. Ideally interventions are compared against the "gold-standard" in a randomized clinical trial.Despite logistic, financial and at times ethical concerns and prejudice from clinicians, [15][16][17][18] the fetal surgery community has shown it is possible to successfully complete such trials (Figure 1). These landmark papers justify the wider spread of the procedure, in turn generating new data and research opportunities. Although Prenatal

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“…New interest in this intervention has recently risen, and its feasibility has now been demonstrated in several studies 54,55 . Prenatal endoscopic ventriculostomy of the 3 rd ventricle (ETV) for congenital aqueductal stenosis has been successful in a large animal model 56 . Nonetheless, the beneficial effect of these fetal interventions on neurodevelopmental outcomes remains to be proven.…”

Section: In‐utero Treatmentmentioning

confidence: 99%

Fetal macrocephaly: Pathophysiology, prenatal diagnosis and management

Shinar,

Chitayat,

Shannon

et al. 2023

Prenatal Diagnosis

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Macrocephaly means a large head and is defined as a head circumference (HC) above the 98th percentile or greater than +2SD above the mean for gestational age. Macrocephaly can be primary and due to increased brain tissue (megalocephaly), which in most cases is familial and benign or secondary. The latter may be due to various causes, including but not limited to communicating or non‐communicating hydrocephalus, cerebral edema, focal and pericerebral increased fluid collections, thickened calvarium and brain tumors. Megalocephaly can be syndromic or non‐syndromic. In the former, gyral and structural CNS anomalies are common. It is important to exercise caution when considering a diagnosis of megalocephaly due to limitations in the accuracy of HC measurement, lack of nomograms for specific populations, inconsistencies between prenatal and postnatal HC growth curves and progression over time. The degree of macrocephaly is important, with mild macrocephaly ≤2.5SD carrying a good prognosis, especially when one of the parents has macrocephaly and normal development. Cases in which the patient history and/or physical exam are positive or when parental HC are normal are more worrisome and warrant a neurosonogram, fetal MRI and genetic testing to better delineate the underlying etiology and provide appropriate counseling.

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Fetal Endoscopic Third Ventriculostomy Is Technically Feasible in Prenatally Induced Hydrocephalus Ovine Model

Cited by 6 publications

References 26 publications

Prenatal Diagnosis of Fetal Aqueductal Stenosis: A Multicenter Prospective Observational Study through the North American Fetal Therapy Network

Prenatal Diagnosis of Fetal Aqueductal Stenosis: A Multicenter Prospective Observational Study through the North American Fetal Therapy Network

The new fetal therapy section awaits your research

Fetal macrocephaly: Pathophysiology, prenatal diagnosis and management

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