Efficacy of tolvaptan in an infant with syndrome of inappropriate antidiuretic hormone secretion associated with holoprosencephaly:A case report  

作　　者：Mari Mori Satoru Takeshita Nami Nakamura Yuki Mizuno Akiko Tomita Mineyoshi Aoyama Hiroki Kakita Yasumasa Yamada 

机构地区：[1]Department of Perinatal and Neonatal Medicine,Aichi Medical University,Nagakute 480-1195,Japan [2]Department of Pathobiology,Nagoya City University Graduate School of Pharmaceutical Sciences,Nagoya 467-8603,Japan [3]Department of Pediatrics,Aichi Medical University,Nagakute 480-1195,Japan [4]Department of Pharmacy,Aichi Medical University,Nagakute 480-1195,Japan

出　　处：《World Journal of Clinical Cases》2023年第26期6262-6267,共6页世界临床病例杂志

摘　　要：BACKGROUND Holoprosencephaly(HPE)is a congenital malformation with various degrees of incomplete separation of the cerebral hemispheres due to differentiation disorders of the forebrain.Although HPE with diabetes insipidus due to associated pituitary dysfunction has been reported,HPE with the syndrome of inappropriate antidiuretic hormone secretion(SIADH)is very rare.Tolvaptan,a vasopressin V2 receptor antagonist,is effective in adults with SIADH.However,there is no report of its efficacy in infants with SIADH.The purpose of this report is to demonstrate that tolvaptan is effective for SIADH in infants and that administration of tolvaptan eliminates the need for restriction of water intake and sodium administration.CASE SUMMARY A 2414-g female infant was born at 38 wk by normal vaginal delivery.Facial anomalies and head magnetic resonance imaging indicated semilobar HPE.After birth,she had hyponatremia due to SIADH and was treated using water and sodium restriction.However,she developed an exaggerated response to the fluid restrictions,resulting in large fluctuations in serum sodium levels.Subsequent administration of tolvaptan improved the fluctuations in serum sodium levels without the need for adjustment of water or sodium administration.Serum sodium was maintained within the normal range after discontinuation of tolvaptan at 80 d of life.There were no side effects,such as hypernatremia or liver dysfunction,during the administration of tolvaptan.CONCLUSION This is the first report on the safety and efficacy of tolvaptan in an infant with SIADH associated with HPE.

关 键 词：TOLVAPTAN HOLOPROSENCEPHALY Antidiuretic hormone Syndrome of inappropriate secretion of antidiuretic hormone HYPONATREMIA Case report 

分 类 号：R969[医药卫生—药理学]