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Hematuria, benign familial, 2(BFH2)

MedGen UID:
1841057
Concept ID:
C5830421
Disease or Syndrome
Synonym: BFH2
 
Gene (location): COL4A3 (2q36.3)
 
Monarch Initiative: MONDO:0958186
OMIM®: 620320

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 (see 301050), which results in end-stage renal failure and may be associated with hearing loss and ocular anomalies (review by Lemmink et al. (1996)). For a discussion of genetic heterogeneity of BFH, see BFH1 (141200). [from OMIM]

Clinical features

From HPO
Proteinuria
MedGen UID:
10976
Concept ID:
C0033687
Finding
Increased levels of protein in the urine.
Microscopic hematuria
MedGen UID:
65997
Concept ID:
C0239937
Finding
Microscopic hematuria detected by dipstick or microscopic examination of the urine.
Abnormal glomerular basement membrane morphology
MedGen UID:
1786252
Concept ID:
C5539478
Anatomical Abnormality
Any abnormal sttructure of the glomerular basement membrane.
Sensorineural hearing loss disorder
MedGen UID:
9164
Concept ID:
C0018784
Disease or Syndrome
A type of hearing impairment in one or both ears related to an abnormal functionality of the cochlear nerve.

Recent clinical studies

Etiology

Togashi R, Tamura Y, Arai S, Asakawa S, Yamazaki O, Uchida S, Shibata S, Fujigaki Y
Int Urol Nephrol 2023 Mar;55(3):749-758. Epub 2022 Sep 26 doi: 10.1007/s11255-022-03373-4. PMID: 36155873
Mastrangelo A, Madeira C, Castorina P, Giani M, Montini G
Nephrol Dial Transplant 2022 Nov 23;37(12):2398-2407. doi: 10.1093/ndt/gfab334. PMID: 35090027
Voskarides K, Stefanou C, Pieri M, Demosthenous P, Felekkis K, Arsali M, Athanasiou Y, Xydakis D, Stylianou K, Daphnis E, Goulielmos G, Loizou P, Savige J, Höhne M, Völker LA, Benzing T, Maxwell PH, Gale DP, Gorski M, Böger C, Kollerits B, Kronenberg F, Paulweber B, Zavros M, Pierides A, Deltas C
PLoS One 2017;12(3):e0174274. Epub 2017 Mar 23 doi: 10.1371/journal.pone.0174274. PMID: 28334007Free PMC Article
Hoshino Y, Kaga T, Abe Y, Endo M, Wakai S, Tsuchiya K, Nitta K
Clin Exp Nephrol 2015 Oct;19(5):918-24. Epub 2015 Feb 13 doi: 10.1007/s10157-015-1090-6. PMID: 25672293Free PMC Article
Meglic A, Kuzman D, Jazbec J, Japelj-Pavesić B, Kenda RB
Pediatr Nephrol 2003 Feb;18(2):127-32. Epub 2003 Jan 17 doi: 10.1007/s00467-002-1024-x. PMID: 12579401

Diagnosis

Emami A, Nili F, Sotoudeh Anvari M, Salarvand S, Seirafi G
Arch Iran Med 2024 Jan 1;27(1):8-14. doi: 10.34172/aim.2024.02. PMID: 38431955Free PMC Article
Mastrangelo A, Madeira C, Castorina P, Giani M, Montini G
Nephrol Dial Transplant 2022 Nov 23;37(12):2398-2407. doi: 10.1093/ndt/gfab334. PMID: 35090027
Tadros NN, Conlin MJ
Can J Urol 2021 Feb;28(1):10510-10515. PMID: 33625340
Li A, Gao EZ, Cui YX, Liu JH, Lv X, Wei XX, Xia XY, Gao CL, Liu FX, Xia ZK, Asan, Liu ZH, Li XJ
Cytogenet Genome Res 2018;154(1):30-36. Epub 2018 Feb 15 doi: 10.1159/000486979. PMID: 29669314
Hoshino Y, Kaga T, Abe Y, Endo M, Wakai S, Tsuchiya K, Nitta K
Clin Exp Nephrol 2015 Oct;19(5):918-24. Epub 2015 Feb 13 doi: 10.1007/s10157-015-1090-6. PMID: 25672293Free PMC Article

Therapy

Tadros NN, Conlin MJ
Can J Urol 2021 Feb;28(1):10510-10515. PMID: 33625340

Prognosis

Mastrangelo A, Madeira C, Castorina P, Giani M, Montini G
Nephrol Dial Transplant 2022 Nov 23;37(12):2398-2407. doi: 10.1093/ndt/gfab334. PMID: 35090027
Li A, Gao EZ, Cui YX, Liu JH, Lv X, Wei XX, Xia XY, Gao CL, Liu FX, Xia ZK, Asan, Liu ZH, Li XJ
Cytogenet Genome Res 2018;154(1):30-36. Epub 2018 Feb 15 doi: 10.1159/000486979. PMID: 29669314

Clinical prediction guides

Togashi R, Tamura Y, Arai S, Asakawa S, Yamazaki O, Uchida S, Shibata S, Fujigaki Y
Int Urol Nephrol 2023 Mar;55(3):749-758. Epub 2022 Sep 26 doi: 10.1007/s11255-022-03373-4. PMID: 36155873
Mastrangelo A, Madeira C, Castorina P, Giani M, Montini G
Nephrol Dial Transplant 2022 Nov 23;37(12):2398-2407. doi: 10.1093/ndt/gfab334. PMID: 35090027

Recent systematic reviews

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