Atypical Hemolytic Uremic Syndrome (p.Gly1110Ala) with Autoimmune Disease.
Journal
The American journal of case reports
ISSN: 1941-5923
Titre abrégé: Am J Case Rep
Pays: United States
ID NLM: 101489566
Informations de publication
Date de publication:
03 May 2020
03 May 2020
Historique:
entrez:
4
5
2020
pubmed:
4
5
2020
medline:
6
1
2021
Statut:
epublish
Résumé
BACKGROUND Hemolytic uremic syndrome (HUS) can be categorized as primary (typical or atypical) or secondary (with a coexisting diseases). Typical HUS usually means shiga-toxin-medicated and thrombotic thrombocytopenic purpura. Secondary HUS is often initiated by coexisting diseases or conditions such as infections, transplantation, cancer, and autoimmune disease. Atypical HUS (aHUS) is usually induced by genetic mutations of one or several complement-regulating genes and associated with dysregulated complement activation. In the era of compliment-inhibiting therapy, early recognition of aHUS is important for patient prognosis. However, compliment-inhibiting therapy is not always beneficial in patients with secondary HUS. CASE REPORT We present a case of a 49-year-old woman with aHUS, which was caused by a novel genetic point mutation of complement factor H gene (p.Gly1110Ala) mimicking secondary HUS with scleroderma. Instead of administering eculizumab treatment for C5 polymorphism, the patient was successfully treated with mycophenolate mofetil. CONCLUSIONS HUS has complex and mixed etiologies and requires genetic testing. Attention should be paid to new point mutations in aHUS.
Identifiants
pubmed: 32361709
pii: 922567
doi: 10.12659/AJCR.922567
pmc: PMC7214013
doi:
Substances chimiques
CFH protein, human
0
Enzyme Inhibitors
0
Complement Factor H
80295-65-4
Mycophenolic Acid
HU9DX48N0T
Types de publication
Case Reports
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
e922567Références
Clin J Am Soc Nephrol. 2013 Apr;8(4):554-62
pubmed: 23307876
Clin J Am Soc Nephrol. 2018 Feb 7;13(2):300-317
pubmed: 29042465
Clin J Am Soc Nephrol. 2010 Oct;5(10):1844-59
pubmed: 20595690
Semin Arthritis Rheum. 2005 Aug;35(1):35-42
pubmed: 16084222
Biochem J. 1988 Jan 15;249(2):593-602
pubmed: 2963625
J Atheroscler Thromb. 2019 Feb 1;26(2):99-110
pubmed: 30393246
Presse Med. 2014 Oct;43(10 Pt 2):e305-14
pubmed: 25174767
Front Immunol. 2019 Aug 02;10:1750
pubmed: 31428091
Curr Opin Nephrol Hypertens. 2010 May;19(3):242-7
pubmed: 20186056
J Immunol. 2018 Apr 1;200(7):2464-2478
pubmed: 29500241
Clin Exp Immunol. 2006 May;144(2):342-52
pubmed: 16634809
Mol Immunol. 2004 Jun;41(4):355-67
pubmed: 15163532
N Engl J Med. 2002 Aug 22;347(8):589-600
pubmed: 12192020
Kidney Int. 2010 Oct;78(8):721-3
pubmed: 20877372
Kidney Int. 2019 Jun;95(6):1443-1452
pubmed: 30982675
Hum Mol Genet. 2005 Mar 1;14(5):703-12
pubmed: 15661753