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Having Multiple Renal Cysts in a Young Adult is not Always a Sign of Polycystic Kidney Disease

Multiple renal cysts in adult patients could have asymptomatic, benign and a nonprogressive course. However, these cysts could be renal features of a very rare hereditary, progressive syndrome defined as cranioectodermal dysplasia (CED or Sensenbrenner syndrome). Affected patients show dysmorphic fe...

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Autores principales: Kaynar, K, Kayıpmaz, S, Çebi, AH, Hüseynova, Ş
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Sciendo 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9524178/
https://www.ncbi.nlm.nih.gov/pubmed/36249524
http://dx.doi.org/10.2478/bjmg-2021-0016
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author Kaynar, K
Kayıpmaz, S
Çebi, AH
Hüseynova, Ş
author_facet Kaynar, K
Kayıpmaz, S
Çebi, AH
Hüseynova, Ş
author_sort Kaynar, K
collection PubMed
description Multiple renal cysts in adult patients could have asymptomatic, benign and a nonprogressive course. However, these cysts could be renal features of a very rare hereditary, progressive syndrome defined as cranioectodermal dysplasia (CED or Sensenbrenner syndrome). Affected patients show dysmorphic features such as craniosynostosis, nail dystrophy, cutaneous dyshydrosis, dry or scaly palmar skin, trichodysplasia, deafness, pectus excavatum, telecanthus, hypertelorism, low set ears, everted lower lip, anteverted nares, short neck and height, joint laxity, inguinal hernia, widely spaced teeth, microdontia, hypodontia in addition to nephronophthisis. We report a 22-year-old male hypertensive patient with multiple renal cysts and dental malformations listed as malocclusion, screwdriver shaped crowns, widely spaced front teeth, microdontia and hyperdontia. Molecular analysis reported missense p.(Ala875Thr) and p.(Lys969Asn) variants in the WDR35 gene. The 1-year follow-up of this case provided the knowledge that angiotensin II receptor blocker drug (olmesartan) reduced the microalbuminuria to normal levels and preserved the renal functions. We suggest interdisciplinary studies, especially intraoral and genetic evaluations for patients with cystic renal diseases. For the first time we report that hyperdontia could be found as a dental feature of CED.
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spelling pubmed-95241782022-10-14 Having Multiple Renal Cysts in a Young Adult is not Always a Sign of Polycystic Kidney Disease Kaynar, K Kayıpmaz, S Çebi, AH Hüseynova, Ş Balkan J Med Genet Case Report Multiple renal cysts in adult patients could have asymptomatic, benign and a nonprogressive course. However, these cysts could be renal features of a very rare hereditary, progressive syndrome defined as cranioectodermal dysplasia (CED or Sensenbrenner syndrome). Affected patients show dysmorphic features such as craniosynostosis, nail dystrophy, cutaneous dyshydrosis, dry or scaly palmar skin, trichodysplasia, deafness, pectus excavatum, telecanthus, hypertelorism, low set ears, everted lower lip, anteverted nares, short neck and height, joint laxity, inguinal hernia, widely spaced teeth, microdontia, hypodontia in addition to nephronophthisis. We report a 22-year-old male hypertensive patient with multiple renal cysts and dental malformations listed as malocclusion, screwdriver shaped crowns, widely spaced front teeth, microdontia and hyperdontia. Molecular analysis reported missense p.(Ala875Thr) and p.(Lys969Asn) variants in the WDR35 gene. The 1-year follow-up of this case provided the knowledge that angiotensin II receptor blocker drug (olmesartan) reduced the microalbuminuria to normal levels and preserved the renal functions. We suggest interdisciplinary studies, especially intraoral and genetic evaluations for patients with cystic renal diseases. For the first time we report that hyperdontia could be found as a dental feature of CED. Sciendo 2022-06-05 /pmc/articles/PMC9524178/ /pubmed/36249524 http://dx.doi.org/10.2478/bjmg-2021-0016 Text en © 2021 K Kaynar et al., published by Sciendo https://creativecommons.org/licenses/by-nc-nd/4.0/This work is licensed under the Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License.
spellingShingle Case Report
Kaynar, K
Kayıpmaz, S
Çebi, AH
Hüseynova, Ş
Having Multiple Renal Cysts in a Young Adult is not Always a Sign of Polycystic Kidney Disease
title Having Multiple Renal Cysts in a Young Adult is not Always a Sign of Polycystic Kidney Disease
title_full Having Multiple Renal Cysts in a Young Adult is not Always a Sign of Polycystic Kidney Disease
title_fullStr Having Multiple Renal Cysts in a Young Adult is not Always a Sign of Polycystic Kidney Disease
title_full_unstemmed Having Multiple Renal Cysts in a Young Adult is not Always a Sign of Polycystic Kidney Disease
title_short Having Multiple Renal Cysts in a Young Adult is not Always a Sign of Polycystic Kidney Disease
title_sort having multiple renal cysts in a young adult is not always a sign of polycystic kidney disease
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9524178/
https://www.ncbi.nlm.nih.gov/pubmed/36249524
http://dx.doi.org/10.2478/bjmg-2021-0016
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