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Benign familial hematuria(TMN; BFH; BFH1)

MedGen UID:
66039
Concept ID:
C0241908
Disease or Syndrome
Synonyms: Hematuria, Benign Familial; Thin Membrane Nephropathy; Thin-Basement-Membrane Nephropathy
SNOMED CT: Thin basement membrane nephropathy (236418003); Thin basement membrane disease (236418003); Benign familial hematuria (236418003)
 
Related genes: COL4A4, COL4A3
 
Monarch Initiative: MONDO:0957317
OMIM®: 120131; 141200
OMIM® Phenotypic series: PS141200
Orphanet: ORPHA97562

Definition

Benign familial hematuria (BFH) is an autosomal dominant condition manifest as nonprogressive isolated microscopic hematuria that does not result in renal failure. It is characterized pathologically by thinning of the glomerular basement membrane (GBM), and can be considered the mildest end of the spectrum of renal diseases due to type IV collagen defects of the basement membrane. The most severe end of the spectrum is represented by Alport syndrome (301050; 203780, 104200), which results in end-stage renal failure and may be associated with hearing loss and ocular anomalies (review by Lemmink et al. (1996)). Genetic Heterogeneity of Benign Familial Hematuria See also benign familial hematuria-2 (BFH2; 620320), caused by mutation in the COL4A3 gene (120070). [from OMIM]

Clinical features

From HPO
Hematuria
MedGen UID:
5488
Concept ID:
C0018965
Disease or Syndrome
The presence of blood in the urine. Hematuria may be gross hematuria (visible to the naked eye) or microscopic hematuria (detected by dipstick or microscopic examination of the urine).
Thin glomerular basement membrane
MedGen UID:
478451
Concept ID:
C3276821
Finding
Decreased thickness of the glomerular basement membrane (GBM), measured from endothelial to visceral epithelial plasma membrane and mainly attributable to a decrease in thickness of the lamina densa, generally to an overall thickness more than 2 standard deviations less than that of the normal mean GBM thickness for health age- and sex matched individuals. May be focal or diffuse, although the term thin GBMs generally implies thinning of over 50% of GBMs.

Professional guidelines

PubMed

Savige J, Storey H, Watson E, Hertz JM, Deltas C, Renieri A, Mari F, Hilbert P, Plevova P, Byers P, Cerkauskaite A, Gregory M, Cerkauskiene R, Ljubanovic DG, Becherucci F, Errichiello C, Massella L, Aiello V, Lennon R, Hopkinson L, Koziell A, Lungu A, Rothe HM, Hoefele J, Zacchia M, Martic TN, Gupta A, van Eerde A, Gear S, Landini S, Palazzo V, Al-Rabadi L, Claes K, Corveleyn A, Van Hoof E, van Geel M, Williams M, Ashton E, Belge H, Ars E, Bierzynska A, Gangemi C, Lipska-Ziętkiewicz BS
Eur J Hum Genet 2021 Aug;29(8):1186-1197. Epub 2021 Apr 15 doi: 10.1038/s41431-021-00858-1. PMID: 33854215Free PMC Article
Savige J, Gregory M, Gross O, Kashtan C, Ding J, Flinter F
J Am Soc Nephrol 2013 Feb;24(3):364-75. Epub 2013 Jan 24 doi: 10.1681/ASN.2012020148. PMID: 23349312

Recent clinical studies

Etiology

Puapatanakul P, Miner JH
Curr Opin Nephrol Hypertens 2024 May 1;33(3):283-290. Epub 2024 Mar 13 doi: 10.1097/MNH.0000000000000983. PMID: 38477333Free PMC Article
Deltas C, Papagregoriou G, Louka SF, Malatras A, Flinter F, Gale DP, Gear S, Gross O, Hoefele J, Lennon R, Miner JH, Renieri A, Savige J, Turner AN
Genes (Basel) 2023 Aug 25;14(9) doi: 10.3390/genes14091686. PMID: 37761826Free PMC Article
Morinière V, Dahan K, Hilbert P, Lison M, Lebbah S, Topa A, Bole-Feysot C, Pruvost S, Nitschke P, Plaisier E, Knebelmann B, Macher MA, Noel LH, Gubler MC, Antignac C, Heidet L
J Am Soc Nephrol 2014 Dec;25(12):2740-51. Epub 2014 May 22 doi: 10.1681/ASN.2013080912. PMID: 24854265Free PMC Article
Vivante A, Calderon-Margalit R, Skorecki K
Curr Opin Nephrol Hypertens 2013 May;22(3):325-30. doi: 10.1097/MNH.0b013e32835f7241. PMID: 23449218
Pescucci C, Longo I, Bruttini M, Mari F, Renieri A
J Nephrol 2003 Mar-Apr;16(2):314-6. PMID: 12768082

Diagnosis

Puapatanakul P, Miner JH
Curr Opin Nephrol Hypertens 2024 May 1;33(3):283-290. Epub 2024 Mar 13 doi: 10.1097/MNH.0000000000000983. PMID: 38477333Free PMC Article
Deltas C, Papagregoriou G, Louka SF, Malatras A, Flinter F, Gale DP, Gear S, Gross O, Hoefele J, Lennon R, Miner JH, Renieri A, Savige J, Turner AN
Genes (Basel) 2023 Aug 25;14(9) doi: 10.3390/genes14091686. PMID: 37761826Free PMC Article
Vivante A, Calderon-Margalit R, Skorecki K
Curr Opin Nephrol Hypertens 2013 May;22(3):325-30. doi: 10.1097/MNH.0b013e32835f7241. PMID: 23449218
Knier B, Büschges-Seraphin B, Hilgers KF, Amann KU, Uder M, Eckardt KU, Jacobi J
BMC Nephrol 2012 Feb 1;13:7. doi: 10.1186/1471-2369-13-7. PMID: 22296661Free PMC Article
Coates R
J Insur Med 2011;42(2-4):85-91. PMID: 21888194

Therapy

Haas M
Arch Pathol Lab Med 2006 May;130(5):699-706. doi: 10.5858/2006-130-699-TGBMNI. PMID: 16683888

Prognosis

Deltas C, Papagregoriou G, Louka SF, Malatras A, Flinter F, Gale DP, Gear S, Gross O, Hoefele J, Lennon R, Miner JH, Renieri A, Savige J, Turner AN
Genes (Basel) 2023 Aug 25;14(9) doi: 10.3390/genes14091686. PMID: 37761826Free PMC Article
Coates R
J Insur Med 2011;42(2-4):85-91. PMID: 21888194
Pescucci C, Longo I, Bruttini M, Mari F, Renieri A
J Nephrol 2003 Mar-Apr;16(2):314-6. PMID: 12768082
Yoshikawa N, Matsuyama S, Iijima K, Maehara K, Okada S, Matsuo T
Arch Pathol Lab Med 1988 Aug;112(8):794-7. PMID: 3395216
Marks MI, Drummond KN
Pediatrics 1969 Oct;44(4):590-3. PMID: 4899625

Clinical prediction guides

Horinouchi T, Yamamura T, Nagano C, Sakakibara N, Ishiko S, Aoto Y, Rossanti R, Nakanishi K, Shima Y, Morisada N, Iijima K, Nozu K
Kidney360 2020 Sep 24;1(9):936-942. Epub 2020 Jul 16 doi: 10.34067/KID.0000372019. PMID: 35369551Free PMC Article
Lenarduzzi S, Morgan A, Faletra F, Cappellani S, Morgutti M, Mezzavilla M, Peruzzi A, Ghiselli S, Ambrosetti U, Graziano C, Seri M, Gasparini P, Girotto G
Hear Res 2019 Sep 15;381:107769. Epub 2019 Jul 13 doi: 10.1016/j.heares.2019.07.006. PMID: 31387071
Vivante A, Calderon-Margalit R, Skorecki K
Curr Opin Nephrol Hypertens 2013 May;22(3):325-30. doi: 10.1097/MNH.0b013e32835f7241. PMID: 23449218
Coates R
J Insur Med 2011;42(2-4):85-91. PMID: 21888194
Yoshikawa N, Matsuyama S, Iijima K, Maehara K, Okada S, Matsuo T
Arch Pathol Lab Med 1988 Aug;112(8):794-7. PMID: 3395216

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