Multicenter Trial to Study Treatment for Near Universally Fatal Disease

An experimental therapy offers a life raft for the fetus enduring the congenital anomaly early pregnancy renal anhydramnios, but the treatment needs to be regulated and consistent.

Early pregnancy renal anhydramnios (EPRA), in which no amniotic fluid surrounds a fetus due to a problem with the fetus’s kidneys, is a devastating congenital anomaly. Doctors often counsel women to terminate the pregnancy because the baby will almost certainly die, either in the womb due to compression of the umbilical cord or right after birth due to respiratory failure. An experimental therapy exists — serial amnioinfusions — but how it is applied and how patients are managed varies from physician to physician.

“A huge number of parents who were seeking help for this problem got it in a very disorganized and unscientific fashion — there was no knowledge of the safety, efficacy and even feasibility of this intervention to counsel patients about what the result would be and what the postnatal outcomes would be like,” says pediatric and fetal surgeon Eric Jelin. “There’s a huge need for people to study this therapy in a prospective and systematic way so society in general can know what the right thing to do is for these patients.”

That concern prompted Jelin, who oversees the fetal program at Johns Hopkins Children’s Center, to initiate in 2017 an ethics symposium with colleagues from the Johns Hopkins Berman Institute of Bioethics to discuss how physicians should best move forward with the treatment. That led Jelin to design a multicenter trial with fetal medicine centers across the country to guide appropriate and safe use of the therapy, during which a small needle is periodically placed through the pregnant woman’s skin and into the womb.

“Because there was success in the early cases, there were people doing this without supervision, without counseling, without ethical or regulatory oversight,” says Jelin. “We decided we would stop doing it until we had a prospective trial.”

The North American Fetal Therapy Network, a consortium of over 30 fetal medicine centers in the United States, endorsed the trial, and the National Institutes of Health’s National Institute of Child Health and Human Development supported Jelin with a $3.3 million R01 grant for a multicenter trial over five years. Pregnant women with a diagnosis of EPRA can enroll in the Renal Anhydramnios Fetal Therapy (RAFT) trial at Johns Hopkins Children’s Center or one of eight other centers. Because there is a significant risk of early rupture of membranes and early delivery for patients who receive amnioinfusions, potential trial participants will be counseled about the risk before they decide whether to join the trial.

“We’re enrolling patients with very strict inclusion and exclusion criteria who have been well counseled about all the possibilities,” says Jelin. “They are going into this as well informed as we can make them.”

Participants may also choose not to receive the amniotic infusions but receive close monitoring for the remainder of their pregnancy. The study will follow babies and their families until nonsurvival or four years after kidney transplant. Treatment for lung disease secondary to abnormal lung development may also be required.

“The trial is not just the intervention — it allows us to follow those cases who don’t want to undergo the intervention, to see what their outcomes truly are like and not just use anecdotal data,” says Jelin.

Noting that EPRA occurs in more than one in 2,000 pregnancies and affects 1,500 families in the U.S annually, Jelin says many parents who want their baby to survive hear there’s no hope. “But there actually is hope,” he says. “We want to offer that hope but in a responsible way.”

For more information, contact the Johns Hopkins Children’s Center fetal program, which includes cardiologists, ethicists, geneticists, maternal fetal medicine physicians, neonatologists, nephrologists, obstetricians, pediatric surgeons, social workers and transplant surgeons. The team collaborates with the Johns Hopkins Center for Fetal Therapy, headed by gynecologist obstetrician Ahmet Baschat.