Case Report
Copyright ©The Author(s) 2023. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Sep 16, 2023; 11(26): 6262-6267
Published online Sep 16, 2023. doi: 10.12998/wjcc.v11.i26.6262
Efficacy of tolvaptan in an infant with syndrome of inappropriate antidiuretic hormone secretion associated with holoprosencephaly: A case report
Mari Mori, Satoru Takeshita, Nami Nakamura, Yuki Mizuno, Akiko Tomita, Mineyoshi Aoyama, Hiroki Kakita, Yasumasa Yamada
Mari Mori, Satoru Takeshita, Nami Nakamura, Hiroki Kakita, Yasumasa Yamada, Department of Perinatal and Neonatal Medicine, Aichi Medical University, Nagakute 480-1195, Japan
Satoru Takeshita, Mineyoshi Aoyama, Hiroki Kakita, Department of Pathobiology, Nagoya City University Graduate School of Pharmaceutical Sciences, Nagoya 467-8603, Japan
Nami Nakamura, Department of Pediatrics, Aichi Medical University, Nagakute 480-1195, Japan
Yuki Mizuno, Akiko Tomita, Department of Pharmacy, Aichi Medical University, Nagakute 480-1195, Japan
Author contributions: Mori M wrote this manuscript under the supervision of Takeshita S, Aoyama M, Kakita H, and Yamada Y; Nakamura N was involved in the patient’s treatment planning; Mizuno Y and Tomita A confirmed the drug’s side effects and safety; and all authors have read and approved the final manuscript.
Informed consent statement: Informed written consent was obtained from the patient for the publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no competing interests.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Satoru Takeshita, MD, PhD, Lecturer, Department of Perinatal and Neonatal Medicine, Aichi Medical University, 2811-1 Yazakokarimata, Nagakute 480-1195, Japan. satorut@aichi-med-u.ac.jp
Received: June 6, 2023
Peer-review started: June 6, 2023
First decision: July 28, 2023
Revised: July 31, 2023
Accepted: August 15, 2023
Article in press: August 15, 2023
Published online: September 16, 2023
Core Tip

Core Tip: Holoprosencephaly with the syndrome of inappropriate antidiuretic hormone secretion (SIADH) is very rare. The main treatments for SIADH are restriction of water intake and sodium administration, which could inhibit the infant’s growth. Tolvaptan, a vasopressin V2 receptor antagonist, is effective in adults with SIADH but has not been reported in infants. In the present neonatal case, the patient developed SIADH, and a restriction of water intake and sodium administration caused the fluctuations in serum sodium levels. Subsequent administration of tolvaptan improved the serum sodium level. Tolvaptan could be a novel standard treatment for infants with SIADH.