ArticleJournal of Korean medical science2020
A Premature Baby with Severe Oligohydramnios and Hypotension: a Case Report of Renal Tubular Dysgenesis.
Article in Journal of Korean medical science, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.
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Who cites it
6 citing papers in PubMed, 8 citations in OpenAlex.
- ACE-related renal tubular dysgenesis (RTD-ACE) in a neonate surviving beyond the early neonatal period.Pediatric nephrology (Berlin, Germany) · 2026Article
- Biallelic PKD1 mutations causing neonatal death in an extremely preterm infant: a Korean case report of very early-onset ADPKD.BMC nephrology · 2026Article
- Long-term kidney function stabilization with fludrocortisone in autosomal recessive renal tubular dysgenesis: a case report.Pediatric nephrology (Berlin, Germany) · 2026Article
- Article
- Functional tests to guide management in an adult with loss of function of type-1 angiotensin II receptor.Pediatric nephrology (Berlin, Germany) · 2021Article
- Patterns of differentiation of renin lineage cells during nephrogenesis.American journal of physiology. Renal physiology · 2021Article
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Authors and funding
7 authors at 3 institutions in 1 country.
Funding
Abstract
Renal tubular dysgenesis (RTD) is a rare fatal disorder in which there is poor development of proximal tubules, leading to oligohydramnios and the Potter sequences. RTD occurs secondary to renin-angiotensin system (RAS) blockade during the early stages of fetal development or due to autosomal recessive mutation of genes in the RAS pathway. A boy born at 33+1 weeks due to cord prolapse was found to be anuric and hypotensive. Pregnancy was complicated by severe oligohydramnios from gestational age 28+4 weeks. Abdominal sonography revealed diffuse globular enlargement of both kidneys with increased cortical parenchymal echogenicity. Infantogram showed a narrow thoracic cage and skull X-ray showed large fontanelles and wide sutures suggestive of ossification delay. Basal plasma renin activity was markedly elevated and angiotensin-converting enzyme was undetectable. Despite adequate use of medications, peritoneal dialysis, and respiratory support, he did not recover and expired on the 23rd day of life. At first, autosomal recessive polycystic kidney disease was suspected, but severe oligohydramnios along with refractory hypotension, anuria, skull ossification delay and high renin levels made RTD suspicious.
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