ArticleJournal of cardiology cases2020
A boy with Alagille syndrome coexisting with mid-aortic syndrome and renovascular hypertension.
Article in Journal of cardiology cases, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 5 papers.
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Who cites it
5 citing papers in PubMed, 10 citations in OpenAlex.
- Kidney and vascular involvement in Alagille syndrome.Pediatric nephrology (Berlin, Germany) · 2025Review
- Parachute Mitral Valve and Mid-Aortic Syndrome - Unusual Associations of Alagille Syndrome.European journal of case reports in internal medicine · 2024Article
- A rare middle aortic syndrome with celiac trunk, superior mesenteric and bilateral renal artery involvement.Heliyon · 2023Article
- Long-segmental middle aortic coarctation: a rare case first diagnosed by transthoracic echocardiography.BMC cardiovascular disorders · 2022Article
- Diagnosis and Management of Genetic Causes of Middle Aortic Syndrome in Children: A Comprehensive Literature Review.Therapeutics and clinical risk management · 2022Review
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Authors and funding
6 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundAlagille syndrome (ALGS) is characterized by cholestasis due to paucity of intrahepatic bile ducts, cardiac anomalies, ophthalmologic abnormalities, skeletal abnormalities, and characteristic facies. Mid-aortic syndrome (MAS) is a rare entity characterized by segmental narrowing of the proximal abdominal aorta and ostial stenosis of its major branches. We report a case of ALGS with MAS involving severe renal artery stenosis (RAS). CASE: A four-year-old Japanese boy was referred to our hospital because of cholestatic liver dysfunction. He was diagnosed with ALGS due to having all five characteristic hallmarks. He had high blood pressure (152/84 mmHg) at his first visit. 3D-CT angiography showed coarctation of the abdominal aortic trunk, severe ostial stenosis of the celiac artery, superior mesenteric artery, and bilateral RAs. He was diagnosed with MAS, and treated with metoprolol, cilnidipine, and aspirin. DISCUSSIONS: While vascular abnormalities are reported to occur in 9% of ALGS patients, MAS with ALGS was only reported in 11 patients between 1951 and 2011. In Japan, there were no reports of ALGS coexisting with MAS with the exception of one case with RAS. In addition to the vessels of the heart, it is important to examine patients with ALGS for abnormalities of other vessels.<
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