Letter to the editor: Fetal brain surgery and the historical record

Letter to the editor: © Zerbor - stock.adobe.com

To the editors:

I recently read with interest the article in Contemporary OB/GYN entitled "First successful in-utero brain surgery performed" which reported on the in-utero treatment of a vein of Galen malformation through embolization in a fetus at 34 2/7 weeks gestation, complicated by procedurally related preterm premature ruptured membranes resulting in labor induction just 2 days after the procedure.¹ Reassuringly, a report of the infant doing well at 6 weeks post-delivery was also communicated.

The purpose of my letter is primarily to assure that the historical record of in-utero fetal surgery is maintained accurately in medical reporting. It is not quite the case that this treatment of a vein of Galen malformation was the "first" successful fetal brain surgery as the article states. In fact, in 1981, Birnholz and Frigoletto performed serial in-utero cephalocenteses in a hydrocephalic fetus.² The same year, noting cephalocentesis could only intermittently relieve elevated intracranial pressure, Clewell and colleagues performed the first placement of a ventriculo-amniotic shunt in a human fetus.³ Under continuous ultrasound guidance, a 13-gauge needle was used to introduce the shunt through the fetal calvarium and into the ventricle of the brain. By the time this successful procedure was reported in 1982, Clewell had successfully performed the procedure on 2 additional fetuses affected by progressive hydrocephalus. However, over the next several years with more centers performing this surgery and more outcomes evaluated, it became widely recognized that despite the control of fetal ventricular enlargement, that an overall poor neurological prognosis was encountered, particularly given the preponderance of fetuses with multiple anomalies related to syndromic and genetic abnormalities associated with this prenatal finding. As such, the procedure was ultimately abandoned by the fetal surgical community.⁴

While one might be tempted to argue that ventriculo-amniotic shunting was unsuccessful and thus the title of the article was correct, until many fetuses are treated for vein of Galen malformation with in-utero surgery and long-term outcomes analyzed to understand the long-term outcomes, I believe it is important to put this procedure in context with the rich history of fetal brain surgery that was pioneered by Clewell, who did demonstrate the first procedural success in human fetus shunt placement for treating progressive prenatally diagnosed hydrocephalus. Perhaps in the future with further advancements in prenatal diagnosis, genetic testing and technical capabilities, a specific fetal patient population will be identified where ventriculo-amniotic shunting may once again be considered and found associated with improved perinatal outcomes.

-Jordan H. Perlow MD

Clinical Professor: University of Arizona College of Medicine – Phoenix

Faculty: Division of Maternal-Fetal MedicineBanner University Medical Center – Phoenix

Associate Director Maternal Fetal Medicine, Phoenix Perinatal Associates: 1992-2022

References:

1. Krewson C. First successful in-utero brain surgery performed. Contemp Ob Gyn. May 9, 2023. Accessed May 20, 2023. https://www.contemporaryobgyn.net/view/first-successful-in-utero-brain-surgery-performed
2. Birnholz JC, Frigoletto FD. Antenatal treatment of hydrocephalus. N Engl J Med. 1981;304(17):1021-1023. doi:10.1056/NEJM198104233041706
3. Clewell WH, Johnson ML, Meier PR, et al. A surgical approach to the treatment of fetal hydrocephalus. N Engl J Med. 1982;306(22):1320-1325. doi:10.1056/NEJM198206033062202
4. Clewell WH. Congenital hydrocephalus: treatment in utero. Fetal Ther. 1988;3(1-2):89-97. doi:10.1159/000263338