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Article ; Online: Extrarenal Manifestations: Polycystic Liver Disease and Its Complications.

Bugazia, Seif / Hogan, Marie C

2023 Volume 30, Issue 5, Page(s) 440–453

Abstract: The liver is the commonest site of involvement outside of the kidney in autosomal dominant polycystic kidney disease. Most individuals with polycystic liver disease are asymptomatic and require no therapeutic interventions, but a small number of affected ...

Abstract	The liver is the commonest site of involvement outside of the kidney in autosomal dominant polycystic kidney disease. Most individuals with polycystic liver disease are asymptomatic and require no therapeutic interventions, but a small number of affected individuals who experience symptomatic polycystic liver disease develop medical complications as a result of massive enlargement of cyst number and size and hepatic parenchyma and its subsequent associated complications. This can lead to deterioration in overall health and quality of life, increasing morbidity and mortality. In this review, we will touch upon disease pathogenesis, prevalence, and complications and discuss recent advances in surgical and medical management.
MeSH term(s)	Humans ; Quality of Life ; Liver Diseases/etiology ; Cysts/etiology
Language	English
Publishing date	2023-11-08
Publishing country	United States
Document type	Journal Article ; Review
ZDB-ID	3156601-7
ISSN	2949-8139
ISSN (online)	2949-8139
DOI	10.1053/j.akdh.2023.10.004
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Zs.A 4627: Show issues

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Article ; Online: Concurrent Targeting of Vasopressin Receptor 2 and Somatostatin Receptors in Autosomal Dominant Polycystic Kidney Disease: A Promising Approach for Autosomal Dominant Polycystic Kidney Disease Treatment?

Hogan, Marie C / Masyuk, Tatyana V

Clinical journal of the American Society of Nephrology : CJASN

2023 Volume 18, Issue 2, Page(s) 154–156

MeSH term(s)	Humans ; Polycystic Kidney, Autosomal Dominant/drug therapy ; Receptors, Somatostatin ; Receptors, Vasopressin ; Kidney ; Tolvaptan/therapeutic use
Chemical Substances	Receptors, Somatostatin ; Receptors, Vasopressin ; Tolvaptan (21G72T1950)
Language	English
Publishing date	2023-01-16
Publishing country	United States
Document type	Journal Article ; Comment
ZDB-ID	2226665-3
ISSN	1555-905X ; 1555-9041
ISSN (online)	1555-905X
ISSN	1555-9041
DOI	10.2215/CJN.0000000000000055
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Zs.A 6584: Show issues

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Article ; Online: A practical guide for the management of acute abdominal pain with fever in patients with autosomal dominant polycystic kidney disease.

Jouret, François / Hogan, Marie C / Chebib, Fouad T

Nephrology, dialysis, transplantation : official publication of the European Dialysis and Transplant Association - European Renal Association

2021 Volume 37, Issue 8, Page(s) 1426–1428

MeSH term(s)	Abdominal Pain/etiology ; Humans ; Kidney ; Polycystic Kidney, Autosomal Dominant/complications
Language	English
Publishing date	2021-02-10
Publishing country	England
Document type	Journal Article
ZDB-ID	90594-x
ISSN	1460-2385 ; 0931-0509
ISSN (online)	1460-2385
ISSN	0931-0509
DOI	10.1093/ndt/gfab040
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Zs.A 2198: Show issues			Location: Je nach Verfügbarkeit (siehe Angabe bei Bestand) bis Jg. 1994: Bestellungen von Artikeln über das Online-Bestellformular Jg. 1995 - 2021: Lesesall (1.OG) ab Jg. 2022: Lesesaal (EG)
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Article: Examining the safety and effectiveness of a 4-week supervised exercise intervention in the treatment of frailty in patients with chronic kidney disease.

Lorenz, Elizabeth C / Hickson, LaTonya J / Hogan, Marie C / Kennedy, Cassie C

Clinical kidney journal

2023 Volume 16, Issue 11, Page(s) 2003–2010

Abstract: Background: The optimal duration of antifrailty interventions and how best to deliver them to patients with chronic kidney disease (CKD) is unknown. The aim of this study was to examine the safety, feasibility and preliminary efficacy of a 4-week ... ...

Abstract	Background: The optimal duration of antifrailty interventions and how best to deliver them to patients with chronic kidney disease (CKD) is unknown. The aim of this study was to examine the safety, feasibility and preliminary efficacy of a 4-week supervised exercise intervention on frailty in patients with CKD. Methods: We conducted a prospective feasibility study involving patients with ≥stage 3 CKD (1 patient with stage 3 CKD, 7 patients with stage 4 CKD and 17 patients with stage 5 CKD) who were either frail or prefrail according to the physical frailty phenotype and/or had a Short Physical Performance Battery (SPPB) score ≤10. The exercise intervention consisted of two supervised outpatient sessions per week for 4 weeks (eight total sessions). Frailty and other study measures were assessed at baseline and after 4 weeks of exercise. Results: Of the 34 participants who completed the baseline assessment and were included in the analyses, 25 (73.5%) completed the 4-week assessment. Overall, 64.0% of patients were on dialysis and 64.0% had diabetes mellitus. After 4 weeks of exercise, frailty prevalence, total SPPB scores and energy/fatigue scores improved. No adverse study-related outcomes were reported. Conclusions: The 4 weeks of supervised exercise was safe, was associated with an excellent completion rate and improved frailty parameters in CKD patients with CKD. This study provides important preliminary data for a future larger prospective randomized study. Clinical trialgov: registration
Language	English
Publishing date	2023-08-09
Publishing country	England
Document type	Journal Article
ZDB-ID	2655800-2
ISSN	2048-8513 ; 2048-8505
ISSN (online)	2048-8513
ISSN	2048-8505
DOI	10.1093/ckj/sfad192
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Zs.A 6587: Show issues

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Article ; Online: Expanding the clinical application of the polycystic liver disease questionnaire: determination of a clinical threshold to select patients for therapy.

Barten, Thijs R M / Staring, Christian B / Hogan, Marie C / Gevers, Tom J G / Drenth, Joost P H

HPB : the official journal of the International Hepato Pancreato Biliary Association

2023 Volume 25, Issue 8, Page(s) 890–897

Abstract: Background: Polycystic liver disease (PLD) causes symptoms resulting from cystic volume expansion. The PLD-specific questionnaire (PLD-Q) captures symptom burden. This study aims to develop a threshold to identify patients with symptoms requiring ... ...

Abstract	Background: Polycystic liver disease (PLD) causes symptoms resulting from cystic volume expansion. The PLD-specific questionnaire (PLD-Q) captures symptom burden. This study aims to develop a threshold to identify patients with symptoms requiring further exploration and possibly intervention. Methods: We recruited PLD patients with completed PLD-Qs during their patient journey. We evaluated baseline PLD-Q scores in (un)treated PLD patients to determine a threshold of clinical importance. We assessed our threshold's discriminative ability with receiver operator characteristic statistics, Youden Index, sensitivity, specificity, positive and negative predictive value parameters. Results: We included 198 patients with a balanced proportion of treated (n=100) and untreated patients (n=98, PLD-Q scores 49 vs 19, p<0.001; median total liver volume 5827 vs 2185 ml, p<0.001). We established the PLD-Q threshold at 32 points. A score of ≥32 differentiates treated from untreated patients with an area under the ROC of 0.856, Youden Index 0.564, sensitivity of 85.0%, specificity of 71.4%, positive predictive value of 75.2%, and negative predictive value of 82.4%. Similar metrics were observed in predefined subgroups and an external cohort. Conclusion: We established the PLD-Q threshold at 32 points with high discriminative ability to identify symptomatic patients. Patients with a score ≥32 should be eligible for treatment or inclusion in trials.
MeSH term(s)	Humans ; Liver Diseases/diagnosis ; Liver Diseases/therapy ; Cysts/diagnosis ; Cysts/therapy ; Surveys and Questionnaires
Language	English
Publishing date	2023-04-11
Publishing country	England
Document type	Journal Article
ZDB-ID	2131251-5
ISSN	1477-2574 ; 1365-182X
ISSN (online)	1477-2574
ISSN	1365-182X
DOI	10.1016/j.hpb.2023.04.004
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Zs.A 6326: Show issues

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Article ; Online: Beyond Loss of Kidney Function: Patient Care in Autosomal Dominant Polycystic Kidney Disease.

Hogan, Marie C / Simmons, Kathryn / Ullman, Lawrence / Gondal, Maryam / Dahl, Neera K

Kidney360

2023 Volume 4, Issue 12, Page(s) 1806–1815

Abstract: Patients with autosomal dominant polycystic kidney disease benefit from specialized care over their lifetimes, starting with diagnosis of the condition with ongoing discussion of both the renal course and extra-renal issues. Both renal and extra-renal ... ...

Abstract	Patients with autosomal dominant polycystic kidney disease benefit from specialized care over their lifetimes, starting with diagnosis of the condition with ongoing discussion of both the renal course and extra-renal issues. Both renal and extra-renal issues may continue to cause major morbidity even after successful kidney transplant or initiation of RRT, and extra-renal disease aspects should always be considered as part of routine management. In this review, we will focus on updates in pain/depression screening, cardiac manifestations, liver and pancreatic cysts, kidney stone management, and genetic counseling. In some instances, we have shared our current clinical practice rather than an evidence-based guideline. We anticipate more standardization of care after the release of the Kidney Disease Improving Global Outcomes guidelines for management in autosomal dominant polycystic kidney disease later this year.
MeSH term(s)	Humans ; Polycystic Kidney, Autosomal Dominant/diagnosis ; Polycystic Kidney, Autosomal Dominant/therapy ; Kidney ; Kidney Transplantation ; Kidney Calculi ; Patient Care
Language	English
Publishing date	2023-11-27
Publishing country	United States
Document type	Review ; Journal Article
ISSN	2641-7650
ISSN (online)	2641-7650
DOI	10.34067/KID.0000000000000296
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Article ; Online: Somatostatin analog therapy effectiveness on the progression of polycystic kidney and liver disease: A systematic review and meta-analysis of randomized clinical trials.

Suwabe, Tatsuya / Barrera, Francisco J / Rodriguez-Gutierrez, Rene / Ubara, Yoshifumi / Hogan, Marie C

PloS one

2021 Volume 16, Issue 9, Page(s) e0257606

Abstract: Background: Uncertainty underlies the effectiveness of somatostatin analogues for slowing the progression of polycystic kidney or liver disease.: Methods: Eligible studies included randomized controlled trials (RCTs) evaluating somatostatin analog as ...

Abstract	Background: Uncertainty underlies the effectiveness of somatostatin analogues for slowing the progression of polycystic kidney or liver disease. Methods: Eligible studies included randomized controlled trials (RCTs) evaluating somatostatin analog as therapy for patients with polycystic kidney disease (PKD) or polycystic liver disease (PLD) compared to placebo or standard therapy. Two reviewers independently screened studies identified from databases (MEDLINE, EMBASE, Cochrane Database), clinical trial registries, and references from pertinent articles and clinical practice guidelines. Outcome measurements were changes in total liver volume (TLV), total kidney volume (TKV), and estimated glomerular filtration rate (eGFR). Results: Of 264 nonduplicate studies screened, 10 RCTs met the inclusion criteria. The body of evidence provided estimates warranting moderate confidence. Meta-analysis of 7 RCTs including a total of 652 patients showed that somatostatin analogs are associated with a lower %TLV growth rate compared to control (mean difference, -6.37%; 95% CI -7.90 to -4.84, p<0.00001), and with a lower %TKV growth rate compared to control (mean difference, -3.66%; 95% CI -5.35 to -1.97, p<0.0001). However, it was not associated with a difference in eGFR decline (mean difference, -0.96 mL/min./1.73m2; 95% CI -2.38 to 0.46, p = 0.19). Conclusions: Current body of evidence suggests that somatostatin analogs therapy slows the increase rate of TLV and TKV in patients with PKD or PLD compared to control within a 3-year follow-up period. It does not seem to have an effect on the change in eGFR. Somatostatin analogs therapy can be a promising treatment for ADPKD or ADPLD, and we need to continue to research its effectiveness for ADPKD or ADPLD.
MeSH term(s)	Humans ; Polycystic Kidney, Autosomal Dominant ; Randomized Controlled Trials as Topic
Language	English
Publishing date	2021-09-24
Publishing country	United States
Document type	Journal Article ; Meta-Analysis ; Research Support, Non-U.S. Gov't ; Systematic Review
ISSN	1932-6203
ISSN (online)	1932-6203
DOI	10.1371/journal.pone.0257606
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Article ; Online: Establishing a nephrology genetic clinic.

Pinto E Vairo, Filippo / Kemppainen, Jennifer L / Lieske, John C / Harris, Peter C / Hogan, Marie C

Kidney international

2021 Volume 100, Issue 2, Page(s) 254–259

MeSH term(s)	Ambulatory Care Facilities ; Nephrology
Language	English
Publishing date	2021-07-22
Publishing country	United States
Document type	Editorial
ZDB-ID	120573-0
ISSN	1523-1755 ; 0085-2538
ISSN (online)	1523-1755
ISSN	0085-2538
DOI	10.1016/j.kint.2021.05.008
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Zs.A 797: Show issues

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Article ; Online: Incorporation of Genetic Studies in the Kidney Transplant Evaluation Clinic: The Value of a Multidisciplinary Approach.

El Ters, Mireille / Pinto E Vairo, Filippo / Prochnow, Carri / Schinstock, Carrie / Dean, Patrick / Kemppainen, Jennifer / Lazaridis, Konstantinos / Cosio, Fernando / Fervenza, Fernando C / Cornell, Lynn / Amer, Hatem / Hogan, Marie C

Transplantation

2023 Volume 107, Issue 4, Page(s) 952–960

Abstract: Background: Recent studies identified underlying genetic causes in a proportion of patients with various forms of kidney disease. In particular, genetic testing reclassified some focal segmental glomerulosclerosis (FSGS) cases into collagen type 4 (COL4) ...

Abstract	Background: Recent studies identified underlying genetic causes in a proportion of patients with various forms of kidney disease. In particular, genetic testing reclassified some focal segmental glomerulosclerosis (FSGS) cases into collagen type 4 (COL4)-related nephropathy. This knowledge has major implications for counseling prospective transplant recipients about recurrence risk and screening biologically related donors. We describe our experience incorporating genetic testing in our kidney transplant multidisciplinary practice. Methods: Patients' DNA was analyzed using whole exome sequencing for a comprehensive kidney gene panel encompassing 344 genes associated with kidney diseases and candidate genes highly expressed in the kidney. Results were correlated with phenotype by a multidisciplinary committee of nephrologists, renal pathologists, geneticists, and genetic counselors. Between October 2018 and July 2020, 30 recipient and 5 donor candidates completed testing. Results: Among recipient candidates, 24 (80%) carried the diagnosis of FSGS, 2 (6.7%) tubulointerstitial nephritis, and 1 (3.3%) nephrolithiasis, and 3 (10%) had an unknown cause of kidney disease. The yield for pathogenic/likely pathogenic variants was 43.3%, with majority being COL4 variants (53.8%). Among those with FSGS diagnosis, the yield was 10 of 24 (41.6%), with 29% reclassified into a COL4-related nephropathy. Family history of kidney disease was the only clinical characteristic difference between recipients with positive and negative results (76.9 versus 29.4%; P = 0.025). One of 5 donors tested positive for a pathogenic/likely pathogenic variant and was excluded from donation. Conclusions: We conclude that thoughtful use of genetic testing can be valuable for kidney donor selection and transplant recipient management.
MeSH term(s)	Humans ; Kidney Transplantation/adverse effects ; Glomerulosclerosis, Focal Segmental/diagnosis ; Glomerulosclerosis, Focal Segmental/genetics ; Glomerulosclerosis, Focal Segmental/complications ; Prospective Studies ; Kidney/pathology ; Kidney Diseases
Language	English
Publishing date	2023-03-31
Publishing country	United States
Document type	Journal Article
ZDB-ID	208424-7
ISSN	1534-6080 ; 0041-1337
ISSN (online)	1534-6080
ISSN	0041-1337
DOI	10.1097/TP.0000000000004363
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Zs.A 488: Show issues			Location: Je nach Verfügbarkeit (siehe Angabe bei Bestand) bis Jg. 1994: Bestellungen von Artikeln über das Online-Bestellformular Jg. 1995 - 2021: Lesesall (1.OG) ab Jg. 2022: Lesesaal (EG)
Zs.MO 509: Show issues

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Article ; Online: Comparison of treatment options in adults with frequently relapsing or steroid-dependent minimal change disease.

Heybeli, Cihan / Erickson, Stephen B / Fervenza, Fernando C / Hogan, Marie C / Zand, Ladan / Leung, Nelson

Nephrology, dialysis, transplantation : official publication of the European Dialysis and Transplant Association - European Renal Association

2020 Volume 36, Issue 10, Page(s) 1821–1827

Abstract: Background: Studies comparing all treatment options for frequently-relapsing/steroid-dependent (FR/SD) minimal change disease (MCD) in adults are lacking.: Methods: Medical records of 76 adults with FR/SD MCD who were treated with corticosteroids as ... ...

Abstract	Background: Studies comparing all treatment options for frequently-relapsing/steroid-dependent (FR/SD) minimal change disease (MCD) in adults are lacking. Methods: Medical records of 76 adults with FR/SD MCD who were treated with corticosteroids as the first-line therapy were reviewed. Treatment options were compared for the time to relapse, change of therapy and progression (relapse on full-dose treatment). Results: Second-line treatments included rituximab (RTX; n = 13), mycophenolate mofetil (MMF; n = 12), calcineurin inhibitors (CNI; n = 26) and cyclophosphamide (CTX; n = 16). During the second-line treatments, 48 (71.6%) patients relapsed at median 17 (range 2-100) months. The majority of relapses occurred during dose tapering or off drug. Twenty of 65 (30.8%) changed therapy after the first relapse. The median time to relapse after the second line was 66 versus 28 months in RTX versus non-RTX groups (P = 0.170). The median time to change of treatment was 66 and 44 months, respectively (P = 0.060). Last-line treatment options included RTX (n = 8), MMF (n = 4), CNI (n = 3) and CTX (n = 2). Seven (41.2%) patients had a relapse during the last-line treatment at median 39 (range 5-112) months. The median time to relapse was 48 versus 34 months in the RTX versus non-RTX groups (P = 0.727). One patient in the RTX group died presumably of heart failure. No major adverse event was observed. During the median follow-up of 81 (range 9-355) months, no patients developed end-stage renal disease. Conclusions: Relapse is frequent in MCD in adults. Patients treated with RTX may be less likely to require a change of therapy and more likely to come off immunosuppressive drugs.
MeSH term(s)	Adult ; Humans ; Immunosuppressive Agents/therapeutic use ; Mycophenolic Acid/therapeutic use ; Nephrosis, Lipoid/drug therapy ; Recurrence ; Rituximab ; Steroids ; Treatment Outcome
Chemical Substances	Immunosuppressive Agents ; Steroids ; Rituximab (4F4X42SYQ6) ; Mycophenolic Acid (HU9DX48N0T)
Language	English
Publishing date	2020-09-12
Publishing country	England
Document type	Journal Article
ZDB-ID	90594-x
ISSN	1460-2385 ; 0931-0509
ISSN (online)	1460-2385
ISSN	0931-0509
DOI	10.1093/ndt/gfaa133
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