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Maternal liver-related symptoms during pregnancy in primary sclerosing cholangitis

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Maternal liver-related symptoms during pregnancy in primary sclerosing cholangitis. / Nayagam, Jeremy S; Weismüller, Tobias J; Milkiewicz, Piotr; Wronka, Karolina M; Bik, Emil; Schramm, Christoph; Fuessel, Katja; Zhou, Taotao; Chang, Johannes; Färkkilä, Martti; Carlsson, Ylva; Lundman, Anastasia; Cazzagon, Nora; Corrà, Giorgia; Rigopoulou, Eirini; Dalekos, George N; Båve, Aiva Lundberg; Bergquist, Annika; Ben Belkacem, Karim; Marzioni, Marco; Mancinelli, Martina; Verhelst, Xavier; Marschall, Hanns-Ulrich; Heneghan, Michael A; Joshi, Deepak; International PSC Study Group (IPSCSG).

in: JHEP REP, Jahrgang 6, Nr. 1, 01.2024, S. 100951.

Publikationen: SCORING: Beitrag in Fachzeitschrift/Zeitung › SCORING: Zeitschriftenaufsatz › Forschung › Begutachtung

Harvard

Nayagam, JS, Weismüller, TJ, Milkiewicz, P, Wronka, KM, Bik, E, Schramm, C, Fuessel, K, Zhou, T, Chang, J, Färkkilä, M, Carlsson, Y, Lundman, A, Cazzagon, N, Corrà, G, Rigopoulou, E, Dalekos, GN, Båve, AL, Bergquist, A, Ben Belkacem, K, Marzioni, M, Mancinelli, M, Verhelst, X, Marschall, H-U, Heneghan, MA, Joshi, D & International PSC Study Group (IPSCSG) 2024, 'Maternal liver-related symptoms during pregnancy in primary sclerosing cholangitis', JHEP REP, Jg. 6, Nr. 1, S. 100951. https://doi.org/10.1016/j.jhepr.2023.100951

APA

Nayagam, J. S., Weismüller, T. J., Milkiewicz, P., Wronka, K. M., Bik, E., Schramm, C., Fuessel, K., Zhou, T., Chang, J., Färkkilä, M., Carlsson, Y., Lundman, A., Cazzagon, N., Corrà, G., Rigopoulou, E., Dalekos, G. N., Båve, A. L., Bergquist, A., Ben Belkacem, K., ... International PSC Study Group (IPSCSG) (2024). Maternal liver-related symptoms during pregnancy in primary sclerosing cholangitis. JHEP REP, 6(1), 100951. https://doi.org/10.1016/j.jhepr.2023.100951

Vancouver

Nayagam JS, Weismüller TJ, Milkiewicz P, Wronka KM, Bik E, Schramm C et al. Maternal liver-related symptoms during pregnancy in primary sclerosing cholangitis. JHEP REP. 2024 Jan;6(1):100951. https://doi.org/10.1016/j.jhepr.2023.100951

Bibtex

@article{b0070d0456ff458796c820d17db6301f,
title = "Maternal liver-related symptoms during pregnancy in primary sclerosing cholangitis",
abstract = "BACKGROUND & AIMS: Although worsening liver-related symptoms during pregnancy can occur in primary sclerosing cholangitis (PSC), there are insufficient data to effectively counsel patients on their pre-conception risk and no clear recommendations on monitoring and management during pregnancy. We aimed to describe maternal liver-related symptoms in pregnancy, both before and after PSC diagnosis, and explore factors associated with worsening symptoms and liver-related outcomes.METHODS: We conducted a multicentre retrospective observational study of females with PSC and known pregnancy with live birth, via the International PSC Study Group. We included 450 patients from 12 European centres. Data included clinical variables, liver-related symptoms (pruritus and/or cholangitis) during pregnancy, and liver biochemistry. A composite primary endpoint of transplant-free survival from time of PSC diagnosis was used.RESULTS: There were 266 pregnancies in 178 patients following PSC diagnosis. Worsening liver-related symptoms were reported in 66/228 (28.9%) pregnancies; they had a reduced transplant-free survival (p = 0.03), which retained significance on multivariate analysis (hazard ratio 3.02, 95% CI 1.24-7.35; p = 0.02).Abnormal biochemistry and/or liver-related symptoms (pruritus and/or cholangitis) were noted during pregnancy before PSC diagnosis in 21/167 (12.6%) patients. They had a reduced transplant-free survival from pregnancy (p = 0.01), which did not retain significance in a multivariable model (hazard ratio 1.10, 95% CI 0.43-2.85; p = 0.84).CONCLUSIONS: Liver-related symptoms are frequently encountered during pregnancies before the diagnosis of PSC, and pregnancy may expose the pre-clinical phase of PSC in some patients. Worsening liver-related symptoms were seen in a third of our cohort with known PSC during pregnancy; and this subgroup had a poorer prognosis, which may be related to more advanced liver disease at time of pregnancy and/or a more severe disease phenotype.IMPACT AND IMPLICATIONS: Patients with PSC can develop worsening of their liver-related symptoms during pregnancy; however, risk factors for this and the long-term implications are not known. We identified that there is a significant risk of these symptoms in pregnancy, both before and after PSC has been diagnosed, particularly in patients with elevated alkaline phosphatase. Furthermore, our findings suggest that worsening symptoms during pregnancy may be associated with adverse long-term clinical outcomes of liver transplantation and death in patients with known PSC. This may be related to the presence of more advanced liver disease at time of pregnancy. This information can be used to counsel patients with PSC before conception and identify patients who need close follow-up after delivery.",
author = "Nayagam, {Jeremy S} and Weism{\"u}ller, {Tobias J} and Piotr Milkiewicz and Wronka, {Karolina M} and Emil Bik and Christoph Schramm and Katja Fuessel and Taotao Zhou and Johannes Chang and Martti F{\"a}rkkil{\"a} and Ylva Carlsson and Anastasia Lundman and Nora Cazzagon and Giorgia Corr{\`a} and Eirini Rigopoulou and Dalekos, {George N} and B{\aa}ve, {Aiva Lundberg} and Annika Bergquist and {Ben Belkacem}, Karim and Marco Marzioni and Martina Mancinelli and Xavier Verhelst and Hanns-Ulrich Marschall and Heneghan, {Michael A} and Deepak Joshi and {International PSC Study Group (IPSCSG)}",
note = "{\textcopyright} 2023 The Author(s).",
year = "2024",
month = jan,
doi = "10.1016/j.jhepr.2023.100951",
language = "English",
volume = "6",
pages = "100951",
journal = "JHEP REP",
issn = "2589-5559",
publisher = "Elsevier",
number = "1",

}

RIS

TY - JOUR

T1 - Maternal liver-related symptoms during pregnancy in primary sclerosing cholangitis

AU - Nayagam, Jeremy S

AU - Weismüller, Tobias J

AU - Milkiewicz, Piotr

AU - Wronka, Karolina M

AU - Bik, Emil

AU - Schramm, Christoph

AU - Fuessel, Katja

AU - Zhou, Taotao

AU - Chang, Johannes

AU - Färkkilä, Martti

AU - Carlsson, Ylva

AU - Lundman, Anastasia

AU - Cazzagon, Nora

AU - Corrà, Giorgia

AU - Rigopoulou, Eirini

AU - Dalekos, George N

AU - Båve, Aiva Lundberg

AU - Bergquist, Annika

AU - Ben Belkacem, Karim

AU - Marzioni, Marco

AU - Mancinelli, Martina

AU - Verhelst, Xavier

AU - Marschall, Hanns-Ulrich

AU - Heneghan, Michael A

AU - Joshi, Deepak

AU - International PSC Study Group (IPSCSG)

N1 - © 2023 The Author(s).

PY - 2024/1

Y1 - 2024/1

N2 - BACKGROUND & AIMS: Although worsening liver-related symptoms during pregnancy can occur in primary sclerosing cholangitis (PSC), there are insufficient data to effectively counsel patients on their pre-conception risk and no clear recommendations on monitoring and management during pregnancy. We aimed to describe maternal liver-related symptoms in pregnancy, both before and after PSC diagnosis, and explore factors associated with worsening symptoms and liver-related outcomes.METHODS: We conducted a multicentre retrospective observational study of females with PSC and known pregnancy with live birth, via the International PSC Study Group. We included 450 patients from 12 European centres. Data included clinical variables, liver-related symptoms (pruritus and/or cholangitis) during pregnancy, and liver biochemistry. A composite primary endpoint of transplant-free survival from time of PSC diagnosis was used.RESULTS: There were 266 pregnancies in 178 patients following PSC diagnosis. Worsening liver-related symptoms were reported in 66/228 (28.9%) pregnancies; they had a reduced transplant-free survival (p = 0.03), which retained significance on multivariate analysis (hazard ratio 3.02, 95% CI 1.24-7.35; p = 0.02).Abnormal biochemistry and/or liver-related symptoms (pruritus and/or cholangitis) were noted during pregnancy before PSC diagnosis in 21/167 (12.6%) patients. They had a reduced transplant-free survival from pregnancy (p = 0.01), which did not retain significance in a multivariable model (hazard ratio 1.10, 95% CI 0.43-2.85; p = 0.84).CONCLUSIONS: Liver-related symptoms are frequently encountered during pregnancies before the diagnosis of PSC, and pregnancy may expose the pre-clinical phase of PSC in some patients. Worsening liver-related symptoms were seen in a third of our cohort with known PSC during pregnancy; and this subgroup had a poorer prognosis, which may be related to more advanced liver disease at time of pregnancy and/or a more severe disease phenotype.IMPACT AND IMPLICATIONS: Patients with PSC can develop worsening of their liver-related symptoms during pregnancy; however, risk factors for this and the long-term implications are not known. We identified that there is a significant risk of these symptoms in pregnancy, both before and after PSC has been diagnosed, particularly in patients with elevated alkaline phosphatase. Furthermore, our findings suggest that worsening symptoms during pregnancy may be associated with adverse long-term clinical outcomes of liver transplantation and death in patients with known PSC. This may be related to the presence of more advanced liver disease at time of pregnancy. This information can be used to counsel patients with PSC before conception and identify patients who need close follow-up after delivery.

AB - BACKGROUND & AIMS: Although worsening liver-related symptoms during pregnancy can occur in primary sclerosing cholangitis (PSC), there are insufficient data to effectively counsel patients on their pre-conception risk and no clear recommendations on monitoring and management during pregnancy. We aimed to describe maternal liver-related symptoms in pregnancy, both before and after PSC diagnosis, and explore factors associated with worsening symptoms and liver-related outcomes.METHODS: We conducted a multicentre retrospective observational study of females with PSC and known pregnancy with live birth, via the International PSC Study Group. We included 450 patients from 12 European centres. Data included clinical variables, liver-related symptoms (pruritus and/or cholangitis) during pregnancy, and liver biochemistry. A composite primary endpoint of transplant-free survival from time of PSC diagnosis was used.RESULTS: There were 266 pregnancies in 178 patients following PSC diagnosis. Worsening liver-related symptoms were reported in 66/228 (28.9%) pregnancies; they had a reduced transplant-free survival (p = 0.03), which retained significance on multivariate analysis (hazard ratio 3.02, 95% CI 1.24-7.35; p = 0.02).Abnormal biochemistry and/or liver-related symptoms (pruritus and/or cholangitis) were noted during pregnancy before PSC diagnosis in 21/167 (12.6%) patients. They had a reduced transplant-free survival from pregnancy (p = 0.01), which did not retain significance in a multivariable model (hazard ratio 1.10, 95% CI 0.43-2.85; p = 0.84).CONCLUSIONS: Liver-related symptoms are frequently encountered during pregnancies before the diagnosis of PSC, and pregnancy may expose the pre-clinical phase of PSC in some patients. Worsening liver-related symptoms were seen in a third of our cohort with known PSC during pregnancy; and this subgroup had a poorer prognosis, which may be related to more advanced liver disease at time of pregnancy and/or a more severe disease phenotype.IMPACT AND IMPLICATIONS: Patients with PSC can develop worsening of their liver-related symptoms during pregnancy; however, risk factors for this and the long-term implications are not known. We identified that there is a significant risk of these symptoms in pregnancy, both before and after PSC has been diagnosed, particularly in patients with elevated alkaline phosphatase. Furthermore, our findings suggest that worsening symptoms during pregnancy may be associated with adverse long-term clinical outcomes of liver transplantation and death in patients with known PSC. This may be related to the presence of more advanced liver disease at time of pregnancy. This information can be used to counsel patients with PSC before conception and identify patients who need close follow-up after delivery.

U2 - 10.1016/j.jhepr.2023.100951

DO - 10.1016/j.jhepr.2023.100951

M3 - SCORING: Journal article

C2 - 38089547

VL - 6

SP - 100951

JO - JHEP REP

JF - JHEP REP

SN - 2589-5559

IS - 1

ER -