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ORIGINAL RESEARCH article
Front. Pediatr.
Sec. Pediatric Nephrology
Volume 12 - 2024 |
doi: 10.3389/fped.2024.1378083
Clinical follow-up of 2 families with glomerulopathy caused by COQ8B gene variants and literature review
Provisionally accepted- 1 Hebei Provincial Children's Hospital, Shijiazhuang, China
- 2 Duke Clinical Research Institute, School of Medicine, Duke University, Durham, North Carolina, United States
We report 4 patients from two different families with biopsy-proven steroid-resistant nephrotic syndrome (SRNS). The patients were followed clinically for almost 3 years. Clinical genetic testing performed to evaluate for the cause of unexplained proteinuria in the patients revealed a rare homozygous missense variant of COQ8B in affected individuals from the first family and a rare compound heterozygous missense variant of COQ8B in an affected individual from the second family. An affected individual from the family bearing the homozygous COQ8B variant died at age 11 from kidney failure. The condition of her two affected siblings improved with early coenzyme Q10 treatment and their renal function remains normal. The affected patient from the family bearing the compound heterozygous COQ8B variant who received coenzyme Q10 treatment later after being unresponsive to corticosteroids and immunosuppressants, developed kidney failure.
Keywords: COQ8B variant, Glomerulopathy, steroid-resistant nephrotic syndrome, focal segmental glomerulosclerosis, autosomal recessive
Received: 04 Apr 2024; Accepted: 17 Dec 2024.
Copyright: © 2024 Zhang, Hall, Han, Li and Cui. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) or licensor are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
* Correspondence:
Lei Zhang, Hebei Provincial Children's Hospital, Shijiazhuang, China
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