Severity in polycystic liver disease is associated with aetiology and female gender: Results of the International PLD Registry

Aerts, R; Kievit, Wietske; Jong, Michiel E. de; Ahn, Curie; Reiterová, Jana; Nevens, Frederik; Drenth, Joost P.H.

doi:10.1111/liv.13965

Cited by 23 publications

(32 citation statements)

References 27 publications

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“…The International PLD Registry consists of patients with PLD defined as having >10 liver cysts . For this study, patients from two referral centers (Radboud University Medical Center (Nijmegen, The Netherlands) and University Hospital Leuven (Leuven, Belgium)) were evaluated for inclusion.…”

Section: Methodsmentioning

confidence: 99%

“…Patients were informed about the intended use of the questionnaire in an accompanying information letter. All patient data were entered in the database codified . All questionnaires were coded before processing.…”

Section: Methodsmentioning

confidence: 99%

“…PLD occurs in isolated form as autosomal dominant polycystic liver disease (ADPLD) or as a secondary manifestation in patients with autosomal dominant polycystic kidney disease (ADPKD). Risk factors for disease severity are ADPKD, age, and gender . PLD is particularly penetrant in women, and >80% of patients from large cohort studies are women .…”

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confidence: 99%

“…PLD is particularly penetrant in women, and >80% of patients from large cohort studies are women . Endogenous estrogen production decreases after the menopause, which is defined as the time in a woman's life when menstruation periods cease permanently, and literature suggests that liver volume increases in premenopausal patients but stabilizes postmenopausally . These observations have led to the hypothesis that estrogen must be regarded as a trophic factor for PLD.…”

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confidence: 99%

“…These data raise the question whether the advice to discontinue estrogen‐containing oral contraceptives in premenopausal patients with PLD is valid. Through the International PLD Registry, we were able to explore this controversy in a large cohort covering all stages of PLD . The goal of this cross‐sectional study was to assess whether estrogen‐containing oral contraceptives and/or pregnancies are associated with PLD severity at first presentation in premenopausal and postmenopausal patients.…”

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confidence: 99%

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Estrogen‐Containing Oral Contraceptives Are Associated With Polycystic Liver Disease Severity in Premenopausal Patients

Aerts

Bernts

Gevers

et al. 2019

Clin Pharma and Therapeutics

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The association between estrogen‐containing oral contraceptives and history of pregnancies with disease severity in women with polycystic liver disease (PLD) is unclear. We performed a cross‐sectional cohort study to assess this association by selecting female patients with PLD of which imaging was available prior to any liver volume‐reducing therapy. Patients received a questionnaire to collect detailed information on estrogen use and pregnancies. Preplanned subgroup analyses were performed on premenopausal and postmenopausal patients. The questionnaire was returned by 287 of 360 selected patients (80%). There was no significant association between estrogen‐containing oral contraceptives and height‐adjusted total liver volume (hTLV) in the total group (P = 0.06) and postmenopausal subgroup (P = 0.7). By contrast, each year of exposure corresponds with a 1.45% higher hTLV (P = 0.02) in the premenopausal subgroup, equivalent to a 15.5% higher hTLV for every 10 years of use. Pregnancy duration was not associated with hTLV. In conclusion, patients with PLD should avoid exogenous estrogens.

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Section: Methodsmentioning

confidence: 99%

Section: Methodsmentioning

confidence: 99%

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confidence: 99%

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confidence: 99%

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confidence: 99%

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Estrogen‐Containing Oral Contraceptives Are Associated With Polycystic Liver Disease Severity in Premenopausal Patients

Aerts

Bernts

Gevers

et al. 2019

Clin Pharma and Therapeutics

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Questionnaire PLD‐complaint‐specific assessment identifies need for therapy in polycystic liver disease: A multi‐centric prospective study

et al. 2023

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Background and AimsPolycystic liver disease (PLD) can lead to extensive hepatomegaly. Symptom relief is the primary goal of the treatment. The role of the recently developed disease‐specific questionnaires for identification of the thresholds and the assessment of therapy needs further investigation.MethodsA five‐year prospective multi‐centric observational study in 21 hospitals in Belgium gathered a study population of 198 symptomatic PLD‐patients of whom the disease‐specific symptom questionnaire PLD‐complaint‐specific assessment (POLCA) scores were calculated. The thresholds of the POLCA score for the need for volume reduction therapy were analyzed.ResultsThe study group consisted of mostly (82.8%) women with baseline mean age of 54.4 years ±11.2, median liver volume expressed as height‐adjusted total liver volume(htLV) of 1994 mL (interquartile range [IQR] 1275; 3150) and median growth of the liver of +74 mL/year (IQR +3; +230). Volume reduction therapy was needed in 71 patients (35.9%). A POLCA severity score (SPI) ≥ 14 predicted the need for therapy both in the derivation (n = 63) and the validation cohort (n = 126). The thresholds to start somatostatin analogues (n = 55) or to consider liver transplantation (n = 18) were SPI scores of ≥14 and ≥ 18 and the corresponding mean htLVs were 2902 mL (IQR 1908; 3964) and 3607 mL (IQR 2901; 4337), respectively. Somatostatin analogues treatment resulted in a decrease in the SPI score −6.0 versus + 4.5 in patients without somatostatin analogues (p < 0.01). Changes in the SPI score were significantly different between the liver transplantation group and no liver transplantation group, +4.3 ± 7.1 versus −1.6 ± 4.9, respectively, (p < 0.01).ConclusionA polycystic liver disease‐specific questionnaire can be used as a guide on when to start a volume reduction therapy and to assess the effect of treatment.

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Congenital Cystic Lesions of the Biliary Tree

Lasagni

Morana²,

Strazzabosco

et al. 2021

Diseases of the Liver and Biliary Tree

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Severity in polycystic liver disease is associated with aetiology and female gender: Results of the International PLD Registry

Abstract: Aetiology is presented as a new risk factor associated with PLD severity. Young females with ADPKD represent a subgroup of PLD patients with the most severe phenotype expressed in hTLV.

Cited by 23 publications

References 27 publications

Estrogen‐Containing Oral Contraceptives Are Associated With Polycystic Liver Disease Severity in Premenopausal Patients

Estrogen‐Containing Oral Contraceptives Are Associated With Polycystic Liver Disease Severity in Premenopausal Patients

Questionnaire PLD‐complaint‐specific assessment identifies need for therapy in polycystic liver disease: A multi‐centric prospective study

Congenital Cystic Lesions of the Biliary Tree

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