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Article ; Online: False-positive anti-PLA2R ELISA testing in patients with diabetes mellitus.

Caza, Tiffany N / Larsen, Christopher P

2023 Volume 103, Issue 2, Page(s) 425

MeSH term(s)	Humans ; Enzyme-Linked Immunosorbent Assay ; Glomerulonephritis, Membranous ; Receptors, Phospholipase A2 ; Diabetes Mellitus/diagnosis ; Autoantibodies
Chemical Substances	Receptors, Phospholipase A2 ; Autoantibodies
Language	English
Publishing date	2023-01-20
Publishing country	United States
Document type	Letter ; Comment
ZDB-ID	120573-0
ISSN	1523-1755 ; 0085-2538
ISSN (online)	1523-1755
ISSN	0085-2538
DOI	10.1016/j.kint.2022.11.004
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Zs.A 797: Show issues

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Article ; Online: Lipoic acid in neural epidermal growth factor-like 1-associated membranous nephropathy: more than a coincidence?

Caza, Tiffany N / Larsen, Christopher P

Kidney international

2022 Volume 101, Issue 2, Page(s) 418–419

MeSH term(s)	EGF Family of Proteins ; Glomerulonephritis, Membranous/diagnosis ; Glomerulonephritis, Membranous/drug therapy ; Humans ; Kidney ; Receptors, Phospholipase A2 ; Thioctic Acid/adverse effects
Chemical Substances	EGF Family of Proteins ; Receptors, Phospholipase A2 ; Thioctic Acid (73Y7P0K73Y)
Language	English
Publishing date	2022-01-22
Publishing country	United States
Document type	Letter ; Comment
ZDB-ID	120573-0
ISSN	1523-1755 ; 0085-2538
ISSN (online)	1523-1755
ISSN	0085-2538
DOI	10.1016/j.kint.2021.12.001
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Zs.A 797: Show issues

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Article ; Online: Serologic Studies in Membranous Nephropathy: Novel Strategies and Strengthened Associations.

Caza, Tiffany N / Larsen, Christopher P

Kidney360

2022 Volume 4, Issue 2, Page(s) 128–130

MeSH term(s)	Humans ; Glomerulonephritis, Membranous ; Serologic Tests
Language	English
Publishing date	2022-04-12
Publishing country	United States
Document type	Editorial ; Comment
ISSN	2641-7650
ISSN (online)	2641-7650
DOI	10.34067/KID.0000000000000065
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Article ; Online: Nephronophthisis: a pathological and genetic perspective.

Wolf, Matthias T F / Bonsib, Stephen M / Larsen, Christopher P / Hildebrandt, Friedhelm

Pediatric nephrology (Berlin, Germany)

2023

Abstract: Nephronophthisis (NPHP) is an autosomal recessive cystic kidney disease and is one of the most frequent genetic causes for kidney failure (KF) in children and adolescents. Over 20 genes cause NPHP and over 90 genes contribute to renal ciliopathies often ... ...

Abstract	Nephronophthisis (NPHP) is an autosomal recessive cystic kidney disease and is one of the most frequent genetic causes for kidney failure (KF) in children and adolescents. Over 20 genes cause NPHP and over 90 genes contribute to renal ciliopathies often involving multiple organs. About 15-20% of NPHP patients have additional extrarenal symptoms affecting other organs than the kidneys. The involvement of additional organ systems in syndromic forms of NPHP is explained by shared expression of most NPHP gene products in centrosomes and primary cilia, a sensory organelle present in most mammalian cells. This finding resulted in the classification of NPHP as a ciliopathy. If extrarenal symptoms are present in addition to NPHP, these disorders are defined as NPHP-related ciliopathies (NPHP-RC) and can involve the retina (e.g., with Senior-Løken syndrome), CNS (central nervous system) (e.g., with Joubert syndrome), liver (e.g., Boichis and Arima syndromes), or bone (e.g., Mainzer-Saldino and Sensenbrenner syndromes). This review focuses on the pathological findings and the recent genetic advances in NPHP and NPHP-RC. Different mechanisms and signaling pathways are involved in NPHP ranging from planar cell polarity, sonic hedgehog signaling (Shh), DNA damage response pathway, Hippo, mTOR, and cAMP signaling. A number of therapeutic interventions appear to be promising, ranging from vasopressin receptor 2 antagonists such as tolvaptan, cyclin-dependent kinase inhibitors such as roscovitine, Hh agonists such as purmorphamine, and mTOR inhibitors such as rapamycin.
Language	English
Publishing date	2023-11-06
Publishing country	Germany
Document type	Journal Article ; Review
ZDB-ID	631932-4
ISSN	1432-198X ; 0931-041X
ISSN (online)	1432-198X
ISSN	0931-041X
DOI	10.1007/s00467-023-06174-8
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Zs.A 2196: Show issues

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Article ; Online: EXT1 and NCAM1-associated membranous lupus nephritis in a cohort of patients undergoing repeat kidney biopsies.

Miller, Paul P / Caza, Tiffany / Larsen, Christopher P / Charu, Vivek

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

2022 Volume 38, Issue 2, Page(s) 396–404

Abstract: Background: Exostosin 1/2 (EXT1/2) and neural cell adhesion molecule 1 (NCAM1) associated membranous lupus nephritis (MLN) may represent distinct disease phenotypes with prognostic significance.: Methods: We searched our archives for patients with ... ...

Abstract	Background: Exostosin 1/2 (EXT1/2) and neural cell adhesion molecule 1 (NCAM1) associated membranous lupus nephritis (MLN) may represent distinct disease phenotypes with prognostic significance. Methods: We searched our archives for patients with systemic lupus erythematous (SLE) and at least two kidney biopsies demonstrating MLN. Each biopsy was stained for EXT1 and NCAM1 and scored as positive or negative. Histopathologic and clinical data were reviewed. Results: We identified 31 patients with a clinical diagnosis of SLE and at least two kidney biopsies with MLN. A total of 28 patients (90%) showed concordant staining for EXT1 and NCAM1 in both biopsies; 8 patients (26%) were EXT1 positive and NCAM1 negative, 18 patients (58%) were EXT1 negative and NCAM1 negative and 2 patients (7%) were EXT1 negative and NCAM1 positive. A total of three patients (10%) had discordant EXT1 staining between their first and last biopsies; two patients (7%) were EXT1 positive in their first biopsy and EXT1 negative in the last biopsy and one patient (3%) was EXT1 negative in his first biopsy and EXT1 positive in the last biopsy. Compared with the EXT1-negative cohort at the time of the first biopsy, the EXT1-positive cohort had a higher average estimated glomerular filtration rate (eGFR; 141 versus 108 mL/min/1.73 m2; P = 0.04), lower average percent global glomerulosclerosis (0.5 versus 12%; P = 0.05), lower average interstitial fibrosis and tubular atrophy (2.5 versus 11.7%; P = 0.06) and lower average total National Institutes of Health (NH) chronicity scores (0.75 versus 2.33; P = 0.05). On long-term follow-up, the rate of change in eGFR did not significantly differ between the two groups (P = 0.24). One EXT1-positive patient (12.5%) developed stage 4 chronic kidney disease (CKD) or end-stage kidney disease (ESKD) compared with four patients (20%) in the EXT-negative group and two of the three EXT1-discordant patients (P = 0.38). Conclusions: We performed the largest retrospective repeat-biopsy study to evaluate EXT1 and NCAM1 autoantigens in MLN. Our data demonstrate that EXT1 positivity is associated with better kidney function at the time of diagnosis and raises the possibility that EXT1 status may change throughout the disease course of MLN.
MeSH term(s)	Humans ; Lupus Nephritis/pathology ; Retrospective Studies ; Glomerulonephritis, Membranous/pathology ; Lupus Erythematosus, Systemic/pathology ; Biopsy ; Kidney/pathology ; Neural Cell Adhesion Molecules ; CD56 Antigen
Chemical Substances	Neural Cell Adhesion Molecules ; NCAM1 protein, human ; CD56 Antigen
Language	English
Publishing date	2022-01-24
Publishing country	England
Document type	Journal Article ; Research Support, N.I.H., Extramural
ZDB-ID	90594-x
ISSN	1460-2385 ; 0931-0509
ISSN (online)	1460-2385
ISSN	0931-0509
DOI	10.1093/ndt/gfac058
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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)
Zs.MO 329: Show issues

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Article ; Online: Renal Manifestations of Inflammatory Bowel Disease.

Ambruzs, Josephine M / Larsen, Christopher P

Rheumatic diseases clinics of North America

2018 Volume 44, Issue 4, Page(s) 699–714

Abstract: Renal and urinary involvement has been reported to occur in 4% to 23% of inflammatory bowel disease (IBD) patients. Parenchymal renal disease is rare and most commonly affects glomerular and tubulointerstitial compartments. The most common findings on ... ...

Abstract	Renal and urinary involvement has been reported to occur in 4% to 23% of inflammatory bowel disease (IBD) patients. Parenchymal renal disease is rare and most commonly affects glomerular and tubulointerstitial compartments. The most common findings on renal biopsy of IBD patients are IgA nephropathy and tubulointerstitial nephritis. Overall morbidity of IBD-related renal manifestations is significant, and there is often only a short window of injury reversibility. This, along with subtle clinical presentation, requires a high index of suspicion and routine monitoring of renal function. There are no established guidelines for the optimal screening and monitoring of renal function in IBD patients.
MeSH term(s)	Humans ; Inflammatory Bowel Diseases/physiopathology ; Pain/diagnosis ; Pain/etiology ; Pain Management
Language	English
Publishing date	2018-09-07
Publishing country	United States
Document type	Journal Article ; Review
ZDB-ID	92118-x
ISSN	1558-3163 ; 0889-857X
ISSN (online)	1558-3163
ISSN	0889-857X
DOI	10.1016/j.rdc.2018.06.007
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Article ; Online: Renal Manifestations of Common Variable Immunodeficiency.

Caza, Tiffany N / Hassen, Samar I / Larsen, Christopher P

Kidney360

2020 Volume 1, Issue 6, Page(s) 491–500

Abstract: Background: Common variable immunodeficiency (CVID) is one of the most common primary immunodeficiency syndromes, affecting one in 25,000-50,000 people. Renal insufficiency occurs in approximately 2% of patients with CVID. To date, there are no case ... ...

Abstract	Background: Common variable immunodeficiency (CVID) is one of the most common primary immunodeficiency syndromes, affecting one in 25,000-50,000 people. Renal insufficiency occurs in approximately 2% of patients with CVID. To date, there are no case series of renal biopsies from patients with CVID, making it difficult to determine whether individual cases of renal disease in CVID represent sporadic events or are related to the underlying pathophysiology. We performed a retrospective analysis of renal biopsy specimens in our database from patients with a clinical history of CVID ( Methods: Light, immunofluorescence, and electron microscopy were reviewed. IgG subclasses, PLA2R immunohistochemistry, and THSD7A, EXT1, and NELL1 immunofluorescence were performed on all membranous glomerulopathy cases. CD3, CD4, CD8, and CD20 immunohistochemistry was performed on cases of tubulointerstitial nephritis. Results: AKI and proteinuria were the leading indications for renal biopsy in patients with CVID. Immune-complex glomerulopathy was present in 12 of 22 (54.5%) cases, including nine cases with membranous glomerulopathy, one case with a C3 glomerulopathy, and one case with membranoproliferative GN with IgG3 Conclusions: Membranous glomerulopathy and tubulointerstitial nephritis were the predominant pathologic findings in patients with CVID. Membranous glomerulopathy cases in patients with CVID were IgG1 subclass dominant and showed mesangial immune deposits. Four of the membranous glomerulopathy cases had associated proliferation, with mesangial and/or endocapillary hypercellularity, with or without crescent formation. CVID should be considered as a potential cause when membranous glomerulopathy or chronic tubulointerstitial nephritis is seen in a young patient with a history of recurrent infections.
MeSH term(s)	Common Variable Immunodeficiency/complications ; Glomerulonephritis, Membranous/diagnosis ; Humans ; Kidney/pathology ; Nephritis, Interstitial/pathology ; Retrospective Studies
Language	English
Publishing date	2020-04-21
Publishing country	United States
Document type	Journal Article
ISSN	2641-7650
ISSN (online)	2641-7650
DOI	10.34067/KID.0000432020
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Article: A practical approach to the pathology of renal intratubular casts.

Dvanajscak, Zeljko / Cossey, L Nicholas / Larsen, Christopher P

Seminars in diagnostic pathology

2020 Volume 37, Issue 3, Page(s) 127–134

Abstract: The identification and proper characterization of pathologic renal intratubular casts can be an arduous task, especially since they often admixed with non-pathologic casts, obfuscating debris and inflammation. The list of pathologic intratubular casts is ...

Abstract	The identification and proper characterization of pathologic renal intratubular casts can be an arduous task, especially since they often admixed with non-pathologic casts, obfuscating debris and inflammation. The list of pathologic intratubular casts is long, and they can be easily missed or misdiagnosed without a thorough understanding of their pathophysiology and morphologic variety. Correct characterization of tubular casts is important since each cast type has a unique pathogenic mechanism, with specific treatment and prognostic implications. This review discusses the clinicopathologic characteristics of the six most common pathologic casts: light chain, hemoglobin, myoglobin, red cell, neutrophilic and bile casts. We also discuss hyaline and uromodulin casts, the commonly encountered "benign" cast types that share certain histologic features with pathologic casts. We limit the discussion to proteinaceous and cellular intratubular casts, with crystalline casts discussed in a separate review within the same journal issue. While not exhaustive, this review covers pathogenesis, clinical and prognostic significance, and a practical discussion of the histomorphologic spectrum of each cast type, along with commonly encountered pitfalls.
MeSH term(s)	Humans ; Kidney Diseases/pathology ; Kidney Tubules, Proximal/pathology
Language	English
Publishing date	2020-02-14
Publishing country	United States
Document type	Journal Article ; Review
ZDB-ID	605834-6
ISSN	1930-1111 ; 0740-2570
ISSN (online)	1930-1111
ISSN	0740-2570
DOI	10.1053/j.semdp.2020.02.001
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Zs.A 1988: Show issues

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Article: A diagnostician's field guide to crystalline nephropathies.

Nicholas Cossey, L / Dvanajscak, Zeljko / Larsen, Christopher P

Seminars in diagnostic pathology

2020 Volume 37, Issue 3, Page(s) 135–142

Abstract: The kidney's role in filtration of blood and production of urine occurs via a combination of size and charge filtration at the glomerular basement membrane and resorption and excretion of molecules through a complex tubular system embedded within an ion ... ...

Abstract	The kidney's role in filtration of blood and production of urine occurs via a combination of size and charge filtration at the glomerular basement membrane and resorption and excretion of molecules through a complex tubular system embedded within an ion gradient. This delicate system provides the kidney with a unique propensity for substrate saturation and crystal nucleation within the nephron. While crystalline nephropathies may seem exotic to the uninitiated, they are comprised of easily recognizable morphologies and generally lack complicated classification schemas. Additionally, unlike many intrinsic kidney diseases, crystalline nephropathies are often associated with systemic conditions that, upon further investigation, may elucidate critically important information. This review focuses on practical, diagnostically relevant and high yield information that can be utilized by diagnosticians. Our hope is to equip the reader who reviews renal tissue with a practical toolkit that they feel empowered to use when faced with crystal formation in a kidney biopsy, pre-implantation biopsy, or nephrectomy specimen. Short Abstract The kidney's role in filtration of blood and production of urine provides a unique propensity for substrate saturation and crystal nucleation within the nephron. While crystalline nephropathies may seem exotic to the uninitiated, they are comprised of easily recognizable morphologies and generally lack complicated classification. Additionally, crystalline nephropathies are often associated with systemic conditions that, upon further investigation, may elucidate critically important information.
MeSH term(s)	Crystallization ; Humans ; Kidney Diseases/pathology
Language	English
Publishing date	2020-02-21
Publishing country	United States
Document type	Journal Article ; Review
ZDB-ID	605834-6
ISSN	1930-1111 ; 0740-2570
ISSN (online)	1930-1111
ISSN	0740-2570
DOI	10.1053/j.semdp.2020.02.002
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Zs.A 1988: Show issues

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Article ; Online: COVAN is the new HIVAN: the re-emergence of collapsing glomerulopathy with COVID-19.

Velez, Juan Carlos Q / Caza, Tiffany / Larsen, Christopher P

Nature reviews. Nephrology

2020 Volume 16, Issue 10, Page(s) 565–567

MeSH term(s)	AIDS-Associated Nephropathy/epidemiology ; AIDS-Associated Nephropathy/etiology ; AIDS-Associated Nephropathy/genetics ; Apolipoprotein L1/genetics ; Apolipoprotein L1/metabolism ; Betacoronavirus ; COVID-19 ; Coronavirus Infections/complications ; Coronavirus Infections/epidemiology ; DNA/genetics ; Global Health ; Humans ; Incidence ; Pandemics ; Pneumonia, Viral/complications ; Pneumonia, Viral/epidemiology ; Polymorphism, Single Nucleotide ; SARS-CoV-2
Chemical Substances	APOL1 protein, human ; Apolipoprotein L1 ; DNA (9007-49-2)
Keywords	covid19
Language	English
Publishing date	2020-08-05
Publishing country	England
Document type	Journal Article ; Review
ZDB-ID	2490366-8
ISSN	1759-507X ; 1759-5061
ISSN (online)	1759-507X
ISSN	1759-5061
DOI	10.1038/s41581-020-0332-3
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Zs.A 6334: Show issues			Location: Je nach Verfügbarkeit (siehe Angabe bei Bestand) bis Jg. 2021: Bestellungen von Artikeln über das Online-Bestellformular ab Jg. 2022: Lesesaal (EG)
Zs.MG 107: Show issues

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