Adolescence-onset atypical hemolytic uremic syndrome: is it different from infant-onset?

Clin Exp Nephrol. 2024 Oct;28(10):1027-1037. doi: 10.1007/s10157-024-02505-7. Epub 2024 May 5.

Abstract

Background: Atypical hemolytic uremic syndrome (aHUS) is a rare, mostly complement-mediated thrombotic microangiopathy. The majority of patients are infants. In contrast to infantile-onset aHUS, the clinical and genetic characteristics of adolescence-onset aHUS have not been sufficiently addressed to date.

Methods: A total of 28 patients (21 girls, 7 boys) who were diagnosed as aHUS between the ages of ≥10 years and <18 years were included in this study. All available data in the Turkish Pediatric aHUS registry were collected and analyzed.

Results: The mean age at diagnosis was 12.8±2.3 years. Extra-renal involvement was noted in 13 patients (46.4%); neurological involvement was the most common (32%). A total of 21 patients (75%) required kidney replacement therapy. Five patients (17.8%) received only plasma therapy and 23 (82%) of the patients received eculizumab. Hematologic remission and renal remission were achieved in 25 (89.3%) and 17 (60.7%) of the patients, respectively. Compared with the infantile-onset aHUS patients, adolescent patients had a lower complete remission rate during the first episode (p = 0.002). Genetic analyses were performed in all and a genetic variant was detected in 39.3% of the patients. The mean follow-up duration was 4.9±2.6 years. At the last visit, adolescent patients had lower eGFR levels (p = 0.03) and higher rates of chronic kidney disease stage 5 when compared to infantile-onset aHUS patients (p = 0.04).

Conclusions: Adolescence-onset aHUS is a rare disease but tends to cause more permanent renal dysfunction than infantile-onset aHUS. These results may modify the management approaches in these patients.

Keywords: Adolescence; Atypical hemolytic uremic syndrome; Complement; Genetics; Turkish aHUS registry.

Publication types

  • Comparative Study

MeSH terms

  • Adolescent
  • Age of Onset*
  • Antibodies, Monoclonal, Humanized* / therapeutic use
  • Atypical Hemolytic Uremic Syndrome* / genetics
  • Atypical Hemolytic Uremic Syndrome* / therapy
  • Child
  • Complement Factor H* / genetics
  • Complement Factor I / genetics
  • Complement Inactivating Agents / therapeutic use
  • Diacylglycerol Kinase
  • Female
  • Humans
  • Infant
  • Male
  • Membrane Cofactor Protein / genetics
  • Mutation
  • Plasma Exchange
  • Registries
  • Remission Induction
  • Renal Replacement Therapy
  • Treatment Outcome
  • Turkey / epidemiology

Substances

  • eculizumab
  • Antibodies, Monoclonal, Humanized
  • Complement Factor H
  • Complement Factor I
  • DGKE protein, human
  • Membrane Cofactor Protein
  • CFH protein, human
  • CFI protein, human
  • Complement Inactivating Agents
  • CD46 protein, human
  • Diacylglycerol Kinase