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
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

e922567

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Auteurs

Sihyung Park (S)

Department of Internal Medicine, Inje University Haeundae Paik Hospital, Busan, South Korea.

Yoo Jin Lee (YJ)

Department of Internal Medicine, Inje University Haeundae Paik Hospital, Busan, South Korea.

Yang Wook Kim (YW)

Department of Internal Medicine, Inje University Haeundae Paik Hospital, Busan, South Korea.

Junghae Ko (J)

Department of Internal Medicine, Inje University Haeundae Paik Hospital, Busan, South Korea.

Jin Han Park (JH)

Department of Internal Medicine, Inje University Haeundae Paik Hospital, Busan, South Korea.

Il Hwan Kim (IH)

Department of Internal Medicine, Inje University Haeundae Paik Hospital, Busan, South Korea.

Hee-Jin Kim (HJ)

Department of Laboratory Medicine and Genetics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, South Korea.

Doyeun Oh (D)

Department of Hematology-Oncology, CHA University School of Medicine, Bundang CHA Hospital, Seongnam, South Korea.

Bong Soo Park (BS)

Department of Internal Medicine, Inje University Haeundae Paik Hospital, Busan, South Korea.

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Classifications MeSH