Intrahepatic cholestasis of pregnancy after ovarian hyperstimulation syndrome with wild-type ABCB4 gene: a peculiar case and literature review

Abstract Background Intrahepatic cholestasis of pregnancy (ICP) in the first trimester occurring after ovarian hyperstimulation syndrome (OHSS) is a rare condition and few cases are reported in the literature. Hyperestrogenism may explain this problem in genetically predisposed women. The objective...

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Main Authors: Loris Marin, Guido Ambrosini, Ludovica Nuzzi, Giovanni Buzzaccarini, Federica Esposito, Giampiero Capobianco, Vito Chiantera, Antonio Simone Laganà, Alessandra Andrisani
Format: Article
Language:English
Published: BMC 2023-06-01
Series:BMC Women's Health
Subjects:
Online Access:https://doi.org/10.1186/s12905-023-02471-4
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author Loris Marin
Guido Ambrosini
Ludovica Nuzzi
Giovanni Buzzaccarini
Federica Esposito
Giampiero Capobianco
Vito Chiantera
Antonio Simone Laganà
Alessandra Andrisani
author_facet Loris Marin
Guido Ambrosini
Ludovica Nuzzi
Giovanni Buzzaccarini
Federica Esposito
Giampiero Capobianco
Vito Chiantera
Antonio Simone Laganà
Alessandra Andrisani
author_sort Loris Marin
collection DOAJ
description Abstract Background Intrahepatic cholestasis of pregnancy (ICP) in the first trimester occurring after ovarian hyperstimulation syndrome (OHSS) is a rare condition and few cases are reported in the literature. Hyperestrogenism may explain this problem in genetically predisposed women. The objective of this article is to report one of these rare cases and offer an overview of the other published cases. Case presentation We report a case of severe OHSS followed by ICP in the first trimester. The patient was admitted to the intensive care unit and was treated according to the guidelines for the management of OHSS. Moreover, the patient also received ursodeoxycholic acid for ICP, which brought to an improvement of her clinical conditions. The pregnancy continued without other complications until the 36th week of gestation, when the patient developed ICP in the third trimester and underwent cesarean section for increased bile acid levels and cardiotocographic (CTG) pathologic alterations. The newborn was a healthy baby weighing 2500 gr. We also reviewed other case reports published by other authors about this clinical condition. We present what is, to our knowledge, the first case of ICP developed in the first trimester of pregnancy after OHSS in which genetic polymorphisms of ABCB4 (MDR3) have been investigated. Conclusions ICP in the first trimester might be induced by elevated serum estrogen levels after OHSS in genetically predisposed women. In these women, it might be useful to check for genetic polymorphisms to know if they have a predisposition for ICP recurrence in the third trimester of pregnancy.
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spelling doaj.art-99d1db5799874954aa054933165f50752023-06-18T11:23:54ZengBMCBMC Women's Health1472-68742023-06-012311710.1186/s12905-023-02471-4Intrahepatic cholestasis of pregnancy after ovarian hyperstimulation syndrome with wild-type ABCB4 gene: a peculiar case and literature reviewLoris Marin0Guido Ambrosini1Ludovica Nuzzi2Giovanni Buzzaccarini3Federica Esposito4Giampiero Capobianco5Vito Chiantera6Antonio Simone Laganà7Alessandra Andrisani8Department of Women’s and Children’s Health, University of PaduaDepartment of Women’s and Children’s Health, University of PaduaDepartment of Women’s and Children’s Health, University of PaduaObstetrics and Gynaecology Unit, IRCCS San Raffaele Scientific Institute, Vita-Salute San Raffaele UniversityDepartment of Women’s and Children’s Health, University of PaduaDepartment of Surgical, Microsurgical and Medical Sciences, Gynecologic and Obstetric Clinic, University of SassariUnit of Gynecologic Oncology, ARNAS “Civico – Di Cristina – Benfratelli”, Department of Health Promotion, Mother and Child Care, Internal Medicine and Medical Specialties (PROMISE), University of PalermoUnit of Gynecologic Oncology, ARNAS “Civico – Di Cristina – Benfratelli”, Department of Health Promotion, Mother and Child Care, Internal Medicine and Medical Specialties (PROMISE), University of PalermoDepartment of Women’s and Children’s Health, University of PaduaAbstract Background Intrahepatic cholestasis of pregnancy (ICP) in the first trimester occurring after ovarian hyperstimulation syndrome (OHSS) is a rare condition and few cases are reported in the literature. Hyperestrogenism may explain this problem in genetically predisposed women. The objective of this article is to report one of these rare cases and offer an overview of the other published cases. Case presentation We report a case of severe OHSS followed by ICP in the first trimester. The patient was admitted to the intensive care unit and was treated according to the guidelines for the management of OHSS. Moreover, the patient also received ursodeoxycholic acid for ICP, which brought to an improvement of her clinical conditions. The pregnancy continued without other complications until the 36th week of gestation, when the patient developed ICP in the third trimester and underwent cesarean section for increased bile acid levels and cardiotocographic (CTG) pathologic alterations. The newborn was a healthy baby weighing 2500 gr. We also reviewed other case reports published by other authors about this clinical condition. We present what is, to our knowledge, the first case of ICP developed in the first trimester of pregnancy after OHSS in which genetic polymorphisms of ABCB4 (MDR3) have been investigated. Conclusions ICP in the first trimester might be induced by elevated serum estrogen levels after OHSS in genetically predisposed women. In these women, it might be useful to check for genetic polymorphisms to know if they have a predisposition for ICP recurrence in the third trimester of pregnancy.https://doi.org/10.1186/s12905-023-02471-4Intrahepatic cholestasis of pregnancyFirst trimesterOvarian hyperstimulation syndromeIn vitro fertilizationGenetic polymorphisms
spellingShingle Loris Marin
Guido Ambrosini
Ludovica Nuzzi
Giovanni Buzzaccarini
Federica Esposito
Giampiero Capobianco
Vito Chiantera
Antonio Simone Laganà
Alessandra Andrisani
Intrahepatic cholestasis of pregnancy after ovarian hyperstimulation syndrome with wild-type ABCB4 gene: a peculiar case and literature review
BMC Women's Health
Intrahepatic cholestasis of pregnancy
First trimester
Ovarian hyperstimulation syndrome
In vitro fertilization
Genetic polymorphisms
title Intrahepatic cholestasis of pregnancy after ovarian hyperstimulation syndrome with wild-type ABCB4 gene: a peculiar case and literature review
title_full Intrahepatic cholestasis of pregnancy after ovarian hyperstimulation syndrome with wild-type ABCB4 gene: a peculiar case and literature review
title_fullStr Intrahepatic cholestasis of pregnancy after ovarian hyperstimulation syndrome with wild-type ABCB4 gene: a peculiar case and literature review
title_full_unstemmed Intrahepatic cholestasis of pregnancy after ovarian hyperstimulation syndrome with wild-type ABCB4 gene: a peculiar case and literature review
title_short Intrahepatic cholestasis of pregnancy after ovarian hyperstimulation syndrome with wild-type ABCB4 gene: a peculiar case and literature review
title_sort intrahepatic cholestasis of pregnancy after ovarian hyperstimulation syndrome with wild type abcb4 gene a peculiar case and literature review
topic Intrahepatic cholestasis of pregnancy
First trimester
Ovarian hyperstimulation syndrome
In vitro fertilization
Genetic polymorphisms
url https://doi.org/10.1186/s12905-023-02471-4
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