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Nephrotic syndrome, type 22(NPHS22)

MedGen UID:
1745920
Concept ID:
C5436909
Disease or Syndrome
Synonyms: NEPHROTIC SYNDROME, TYPE 22; NPHS22
 
Gene (location): NOS1AP (1q23.3)
 
Monarch Initiative: MONDO:0030895
OMIM®: 619155

Definition

Nephrotic syndrome type 22 (NPHS22) is an autosomal recessive renal disease characterized by onset of progressive kidney dysfunction in infancy. Affected individuals usually present with edema associated with hypoproteinemia, proteinuria, and microscopic hematuria. Renal biopsy shows effacement of the podocyte foot processes, glomerulosclerosis, and thickening of the glomerular basement membrane. The disease is steroid-resistant and progressive, resulting in end-stage renal disease usually necessitating kidney transplant (Majmundar et al., 2021). For a general phenotypic description and a discussion of genetic heterogeneity of nephrotic syndrome, see NPHS1 (256300). [from OMIM]

Clinical features

From HPO
Nephrotic syndrome
MedGen UID:
10308
Concept ID:
C0027726
Disease or Syndrome
Nephrotic syndrome is a collection of findings resulting from glomerular dysfunction with an increase in glomerular capillary wall permeability associated with pronounced proteinuria. Nephrotic syndrome refers to the constellation of clinical findings that result from severe renal loss of protein, with Proteinuria and hypoalbuminemia, edema, and hyperlipidemia.
Glomerular sclerosis
MedGen UID:
61248
Concept ID:
C0178664
Disease or Syndrome
Accumulation of scar tissue within the glomerulus.
Microscopic hematuria
MedGen UID:
65997
Concept ID:
C0239937
Finding
Microscopic hematuria detected by dipstick or microscopic examination of the urine.
Nephrotic range proteinuria
MedGen UID:
619713
Concept ID:
C0445118
Disease or Syndrome
Severely increased amount of excretion of protein in the urine, defined as 3.5 grams per day or more in adults and 40 mg per meter-squared body surface area per hour in children.
Thickened glomerular basement membrane
MedGen UID:
488906
Concept ID:
C0445347
Finding
Prominent glomerular basement membrane (GBM), reflecting an increase in thickness (subjective estimate) of the basal lamina of the glomerulus of the kidney.
Stage 5 chronic kidney disease
MedGen UID:
384526
Concept ID:
C2316810
Disease or Syndrome
A degree of kidney failure severe enough to require dialysis or kidney transplantation for survival characterized by a severe reduction in glomerular filtration rate (less than 15 ml/min/1.73 m2) and other manifestations including increased serum creatinine.
Podocyte foot process effacement
MedGen UID:
481733
Concept ID:
C3280103
Finding
An anomaly of podocyte morphology characterized by the loss of the interdigitating foot process pattern (generally called foot process effacement; FPE). The term FPE designates the loss of the usual interdigitating pattern of foot processes of neighboring podocytes, leading to relatively broad expanses of podocyte processes covering the glomerular basement membrane (GBM). It is widely viewed as a pathological derangement that is associated with leakage of macromolecules such as albumin through the glomerular filtration barrier.
Hypoproteinemia
MedGen UID:
581229
Concept ID:
C0392692
Finding
A decreased concentration of protein in the blood.
Generalized edema
MedGen UID:
376817
Concept ID:
C1850534
Pathologic Function
Generalized abnormal accumulation of fluid beneath the skin, or in one or more cavities of the body.

Professional guidelines

PubMed

Morioka F, Nakatani S, Nishide K, Machiba Y, Uedono H, Tsuda A, Ishimura E, Mori K, Emoto M
Clin Exp Nephrol 2022 Oct;26(10):955-962. Epub 2022 May 19 doi: 10.1007/s10157-022-02235-8. PMID: 35587298
Taşdemir M, Canpolat N, Yıldız N, Özçelik G, Benzer M, Saygılı SK, Özkayin EN, Türkkan ÖN, Balat A, Candan C, Çelakıl M, Yavuz S, Akıncı N, Göknar N, Akgün C, Tülpar S, Alpay H, Sever FL, Bilge İ
Turk J Med Sci 2021 Aug 30;51(4):1781-1790. doi: 10.3906/sag-2012-297. PMID: 33581711Free PMC Article
Esfahani ST, Afsharipour M, Abedzadeh AA, Fahimi D, Moghtaderi M, Bazargani B, Abbasi A
Iran J Kidney Dis 2020 Dec;14(6):478-481. PMID: 33277452

Recent clinical studies

Etiology

Soares LR, Pantoja Junior JMS, Jorge LB, Yu L, Cavalcante LB, Malheiros DMAC, Woronik V, Dias CB
Braz J Med Biol Res 2022;55:e11861. Epub 2022 Feb 28 doi: 10.1590/1414-431X2022e11861. PMID: 35239780Free PMC Article
Hodson EM, Sinha A, Cooper TE
Cochrane Database Syst Rev 2022 Feb 28;2(2):CD003233. doi: 10.1002/14651858.CD003233.pub3. PMID: 35224732Free PMC Article
Mayer U, Schmitz J, Bräsen JH, Pape L
Pediatr Nephrol 2020 May;35(5):829-842. Epub 2020 Feb 12 doi: 10.1007/s00467-019-04436-y. PMID: 32052153Free PMC Article
Kemmner S, Verbeek M, Heemann U
J Nephrol 2017 Apr;30(2):201-209. Epub 2016 Sep 2 doi: 10.1007/s40620-016-0345-y. PMID: 27589851
Gupta A, Quigg RJ
Adv Chronic Kidney Dis 2015 Sep;22(5):343-51. doi: 10.1053/j.ackd.2015.06.003. PMID: 26311595

Diagnosis

Soares LR, Pantoja Junior JMS, Jorge LB, Yu L, Cavalcante LB, Malheiros DMAC, Woronik V, Dias CB
Braz J Med Biol Res 2022;55:e11861. Epub 2022 Feb 28 doi: 10.1590/1414-431X2022e11861. PMID: 35239780Free PMC Article
Elmougy R
J Appl Biomed 2021 Dec;19(4):228-233. Epub 2021 Oct 12 doi: 10.32725/jab.2021.022. PMID: 34907742
Mayer U, Schmitz J, Bräsen JH, Pape L
Pediatr Nephrol 2020 May;35(5):829-842. Epub 2020 Feb 12 doi: 10.1007/s00467-019-04436-y. PMID: 32052153Free PMC Article
Kemmner S, Verbeek M, Heemann U
J Nephrol 2017 Apr;30(2):201-209. Epub 2016 Sep 2 doi: 10.1007/s40620-016-0345-y. PMID: 27589851
Holmberg C, Laine J, Rönnholm K, Ala-Houhala M, Jalanko H
Kidney Int Suppl 1996 Jan;53:S51-6. PMID: 8770991

Therapy

Soares LR, Pantoja Junior JMS, Jorge LB, Yu L, Cavalcante LB, Malheiros DMAC, Woronik V, Dias CB
Braz J Med Biol Res 2022;55:e11861. Epub 2022 Feb 28 doi: 10.1590/1414-431X2022e11861. PMID: 35239780Free PMC Article
Hodson EM, Sinha A, Cooper TE
Cochrane Database Syst Rev 2022 Feb 28;2(2):CD003233. doi: 10.1002/14651858.CD003233.pub3. PMID: 35224732Free PMC Article
Mayer U, Schmitz J, Bräsen JH, Pape L
Pediatr Nephrol 2020 May;35(5):829-842. Epub 2020 Feb 12 doi: 10.1007/s00467-019-04436-y. PMID: 32052153Free PMC Article
Kemmner S, Verbeek M, Heemann U
J Nephrol 2017 Apr;30(2):201-209. Epub 2016 Sep 2 doi: 10.1007/s40620-016-0345-y. PMID: 27589851
Feng M, Yuan W, Zhang R, Fu P, Wu T
Cochrane Database Syst Rev 2013 Jun 5;2013(6):CD006335. doi: 10.1002/14651858.CD006335.pub3. PMID: 23740567Free PMC Article

Prognosis

Yang L, Wang G, Ye N, Xu X, Cheng W, Sun L, Dong H, Kong L, Zhao X, Geng Y, Cheng H
Front Immunol 2023;14:1297107. Epub 2024 Jan 5 doi: 10.3389/fimmu.2023.1297107. PMID: 38250076Free PMC Article
Elmougy R
J Appl Biomed 2021 Dec;19(4):228-233. Epub 2021 Oct 12 doi: 10.32725/jab.2021.022. PMID: 34907742
Mayer U, Schmitz J, Bräsen JH, Pape L
Pediatr Nephrol 2020 May;35(5):829-842. Epub 2020 Feb 12 doi: 10.1007/s00467-019-04436-y. PMID: 32052153Free PMC Article
Kemmner S, Verbeek M, Heemann U
J Nephrol 2017 Apr;30(2):201-209. Epub 2016 Sep 2 doi: 10.1007/s40620-016-0345-y. PMID: 27589851
Gupta A, Quigg RJ
Adv Chronic Kidney Dis 2015 Sep;22(5):343-51. doi: 10.1053/j.ackd.2015.06.003. PMID: 26311595

Clinical prediction guides

Ji Y, Wang YL, Xu F, Jia XB, Mu SH, Lyu HY, Yuan XY, Na SP, Bao YS
Clin Appl Thromb Hemost 2022 Jan-Dec;28:10760296221108967. doi: 10.1177/10760296221108967. PMID: 35862263Free PMC Article
Elmougy R
J Appl Biomed 2021 Dec;19(4):228-233. Epub 2021 Oct 12 doi: 10.32725/jab.2021.022. PMID: 34907742
Ghiggeri GM, Seitz-Polski B, Justino J, Zaghrini C, Payré C, Brglez V, Dolla G, Sinico A, Scolari F, Vaglio A, Prunotto M, Candiano G, Radice A, Bruschi M, Lambeau G; Italian Study Group for Membranous Nephropathy
Clin J Am Soc Nephrol 2020 Dec 7;15(12):1762-1776. Epub 2020 Nov 30 doi: 10.2215/CJN.02500220. PMID: 33257410Free PMC Article
Kemmner S, Verbeek M, Heemann U
J Nephrol 2017 Apr;30(2):201-209. Epub 2016 Sep 2 doi: 10.1007/s40620-016-0345-y. PMID: 27589851
Suvanto M, Jahnukainen T, Kestilä M, Jalanko H
Clin Exp Nephrol 2015 Jun;19(3):481-8. Epub 2014 Aug 14 doi: 10.1007/s10157-014-1020-z. PMID: 25117488

Recent systematic reviews

Hodson EM, Sinha A, Cooper TE
Cochrane Database Syst Rev 2022 Feb 28;2(2):CD003233. doi: 10.1002/14651858.CD003233.pub3. PMID: 35224732Free PMC Article
Delplanque M, Galicier L, Oziol E, Ducharme-Bénard S, Oksenhendler E, Buob D, Grateau G, Boutboul D, Georgin-Lavialle S
J Allergy Clin Immunol Pract 2021 Feb;9(2):745-752.e1. Epub 2020 Sep 30 doi: 10.1016/j.jaip.2020.09.023. PMID: 33007500
Feng M, Yuan W, Zhang R, Fu P, Wu T
Cochrane Database Syst Rev 2013 Jun 5;2013(6):CD006335. doi: 10.1002/14651858.CD006335.pub3. PMID: 23740567Free PMC Article
Wu S, Kim C, Baer L, Zhu X
J Am Soc Nephrol 2010 Aug;21(8):1381-9. Epub 2010 Jun 10 doi: 10.1681/ASN.2010020167. PMID: 20538785Free PMC Article

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