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Case Reports
. 2022 Jun 29:9:870503.
doi: 10.3389/fmed.2022.870503. eCollection 2022.

Balancing Benefits and Risks of Indomethacin in the Management of Antenatal Bartter Syndrome: A Case Report

Omar Ala' Alajjuri  1 , Mayar Essam Samaha  1 , Ulrich Honemeyer  2 , Ghada Mohammed  3 , Noha A Mousa  3
Affiliations
Case Reports

Balancing Benefits and Risks of Indomethacin in the Management of Antenatal Bartter Syndrome: A Case Report

Omar Ala' Alajjuri et al. Front Med (Lausanne). .

Abstract

Background: Bartter syndrome, a very rare inherited renal tubular disorder, characterized by urinary salt wastage, hypokalemia, polyuria, and metabolic alkalosis, may manifest antenatally as severe isolated polyhydramnios. Indomethacin is known to reduce salt wastage and subsequent polyhydramnios during pregnancy; however, it reduces the Ductus Arteriosus diameter among other potential complications, such as inhibition of gastrointestinal perfusion and increasing the risk of renal toxicity.

Case: A 36-year-old multigravida presented with severe isolated polyhydramnios at 30 weeks of gestation. Based on a history of a previous pregnancy affected with Bartter syndrome, indomethacin was initiated. Amniotic fluid volume and Ductus Arteriosus diameter were monitored. As evidence lacks on optimal dose and duration of indomethacin, multiple-dose adjustments were made to reduce the amniotic fluid volume while maintaining normal Ductus Arteriosus diameter. Progressive polyhydramnios led to Cesarean section at 34+ weeks of gestation resulting in a healthy fetus diagnosed with Bartter syndrome in the early neonatal period.

Conclusion: We share our experience in the adjustment of the dose and duration of Indomethacin therapy in the treatment of severe polyhydramnios associated with antenatal Bartter syndrome. Amniotic fluid index, Ductus Arteriosus diameter, and umbilical artery doppler work together as key indicators to guide the success and safety of the therapy.

Keywords: Bartter syndrome; Ductus Arteriosus; amniotic fluid index; indomethacin; polyhydramnios.

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Conflict of interest statement

The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

Figures

Figure 1
Figure 1
An ultrasound scan showing severe polyhydramnios with a single DVP of 13.9 cm at 30 weeks and 2 days.
Figure 2
Figure 2
An ultrasound scan showing an AFI of 24.1 cm at 31 weeks 2 days.
Figure 3
Figure 3
An ultrasound scan shows DA diameter at 31 weeks 6 days (3.57 mm, 6th centile).
Figure 4
Figure 4
An ultrasound scan showing findings at 34 weeks + 2 days (A): AFI of 45.9 cm. (B): Umbilical artery Doppler waveform with increased PI above the 99th centile.
Figure 5
Figure 5
A summary diagram to outline changes in AFI and DA diameter during the antenatal indomethacin therapy of the patient.
See this image and copyright information in PMC

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