Cornelia de Lange Syndrome Accompanied by Cholelithiasis and Nephrolithiasis: A Case Report

Cornelia de Lange syndrome (CdLS) is a rare genetic disorder characterized by a distinctive facial appearance, growth/cognitive retardation, developmental delay, skeletal malformation, hypertrichosis, and other abnormalities. Patients with mild CdLS have less severe phenotypes, while retaining repre...

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Main Authors: So Yoon Choi, Yoo-Rha Hong, Chi-Eun Oh, Jung Hyun Lee
Format: Article
Language:English
Published: MDPI AG 2024-11-01
Series:Children
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Online Access:https://www.mdpi.com/2227-9067/11/12/1433
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author So Yoon Choi
Yoo-Rha Hong
Chi-Eun Oh
Jung Hyun Lee
author_facet So Yoon Choi
Yoo-Rha Hong
Chi-Eun Oh
Jung Hyun Lee
author_sort So Yoon Choi
collection DOAJ
description Cornelia de Lange syndrome (CdLS) is a rare genetic disorder characterized by a distinctive facial appearance, growth/cognitive retardation, developmental delay, skeletal malformation, hypertrichosis, and other abnormalities. Patients with mild CdLS have less severe phenotypes, while retaining representative facial features. Mutations in the genes <i>NIPBL</i>, <i>SMC1A</i>, <i>SMC3</i>, <i>HDAC8</i>, and <i>RAD21</i> have been associated with CdLS, with mutations in <i>NIPBL</i> accounting for approximately 60% of cases. Herein, we present a case of CdLS accompanied by cholelithiasis and nephrolithiasis. A 9-year-old Korean boy presented with vomiting and abdominal pain. Abdominal ultrasonography revealed several gallstones and renal stones. Extracorporeal shock wave lithotripsy failed; therefore, cholecystectomy and nephrolithotomy were performed. Postoperative stone composition analysis revealed calcium oxalate as the primary component. CdLS was suspected based on the characteristic appearance and physical examination, with genetic testing confirming an <i>NIPBL</i> gene mutation. Simultaneous CdLS, cholelithiasis, and nephrolithiasis requires careful management and treatment tailored to each patient’s specific needs and challenges.
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spelling doaj-art-77f8b82c367049e89d6eb7b937e9e3482025-08-20T02:57:12ZengMDPI AGChildren2227-90672024-11-011112143310.3390/children11121433Cornelia de Lange Syndrome Accompanied by Cholelithiasis and Nephrolithiasis: A Case ReportSo Yoon Choi0Yoo-Rha Hong1Chi-Eun Oh2Jung Hyun Lee3Departments of Pediatrics, Kosin University Gospel Hospital, Kosin University College of Medicine, Busan 49267, Republic of KoreaDepartments of Pediatrics, Kosin University Gospel Hospital, Kosin University College of Medicine, Busan 49267, Republic of KoreaDepartments of Pediatrics, Kosin University Gospel Hospital, Kosin University College of Medicine, Busan 49267, Republic of KoreaDepartments of Pediatrics, Kosin University Gospel Hospital, Kosin University College of Medicine, Busan 49267, Republic of KoreaCornelia de Lange syndrome (CdLS) is a rare genetic disorder characterized by a distinctive facial appearance, growth/cognitive retardation, developmental delay, skeletal malformation, hypertrichosis, and other abnormalities. Patients with mild CdLS have less severe phenotypes, while retaining representative facial features. Mutations in the genes <i>NIPBL</i>, <i>SMC1A</i>, <i>SMC3</i>, <i>HDAC8</i>, and <i>RAD21</i> have been associated with CdLS, with mutations in <i>NIPBL</i> accounting for approximately 60% of cases. Herein, we present a case of CdLS accompanied by cholelithiasis and nephrolithiasis. A 9-year-old Korean boy presented with vomiting and abdominal pain. Abdominal ultrasonography revealed several gallstones and renal stones. Extracorporeal shock wave lithotripsy failed; therefore, cholecystectomy and nephrolithotomy were performed. Postoperative stone composition analysis revealed calcium oxalate as the primary component. CdLS was suspected based on the characteristic appearance and physical examination, with genetic testing confirming an <i>NIPBL</i> gene mutation. Simultaneous CdLS, cholelithiasis, and nephrolithiasis requires careful management and treatment tailored to each patient’s specific needs and challenges.https://www.mdpi.com/2227-9067/11/12/1433Cornelia de Lange syndrome<i>NIPBL</i>cholelithiasisnephrolithiasis
spellingShingle So Yoon Choi
Yoo-Rha Hong
Chi-Eun Oh
Jung Hyun Lee
Cornelia de Lange Syndrome Accompanied by Cholelithiasis and Nephrolithiasis: A Case Report
Children
Cornelia de Lange syndrome
<i>NIPBL</i>
cholelithiasis
nephrolithiasis
title Cornelia de Lange Syndrome Accompanied by Cholelithiasis and Nephrolithiasis: A Case Report
title_full Cornelia de Lange Syndrome Accompanied by Cholelithiasis and Nephrolithiasis: A Case Report
title_fullStr Cornelia de Lange Syndrome Accompanied by Cholelithiasis and Nephrolithiasis: A Case Report
title_full_unstemmed Cornelia de Lange Syndrome Accompanied by Cholelithiasis and Nephrolithiasis: A Case Report
title_short Cornelia de Lange Syndrome Accompanied by Cholelithiasis and Nephrolithiasis: A Case Report
title_sort cornelia de lange syndrome accompanied by cholelithiasis and nephrolithiasis a case report
topic Cornelia de Lange syndrome
<i>NIPBL</i>
cholelithiasis
nephrolithiasis
url https://www.mdpi.com/2227-9067/11/12/1433
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AT chieunoh corneliadelangesyndromeaccompaniedbycholelithiasisandnephrolithiasisacasereport
AT junghyunlee corneliadelangesyndromeaccompaniedbycholelithiasisandnephrolithiasisacasereport