Idiopathic infantile arterial calcification: Two case reports, a review of the literature and a role for cardiac transplantation
: Idiopathic infantile arterial calcification (IIAC) is a rare, but important, cause of rapidly progressive ischemic heart disease in children. In this paper, we report two recent cases of IIAC seen at tertiary referral hospitals. Both cases presented in infancy with signs of heart failure and, ult...
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| Veröffentlicht in: | Pediatric transplantation 2006-03, Vol.10 (2), p.225-233 |
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| container_title | Pediatric transplantation |
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| creator | Glatz, Andrew C. Pawel, Bruce R. Hsu, Daphne T. Weinberg, Paul Chrisant, Maryanne R. K. |
| description | : Idiopathic infantile arterial calcification (IIAC) is a rare, but important, cause of rapidly progressive ischemic heart disease in children. In this paper, we report two recent cases of IIAC seen at tertiary referral hospitals. Both cases presented in infancy with signs of heart failure and, ultimately, died with the diagnosis of IIAC confirmed at postmortem examination. A thorough review of the literature reveals approximately 160 reported cases of IIAC. The clinical outcomes, radiographic findings and pathologic details are summarized. Proposed etiologic mechanisms are reviewed, including promising research into the role of inorganic pyrophosphate as a regulatory factor in the development of IIAC. Because of the typically fatal outcome of IIAC and the lack of proven therapies, the potential role for cardiac transplantation is discussed. |
| doi_str_mv | 10.1111/j.1399-3046.2005.00414.x |
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Because of the typically fatal outcome of IIAC and the lack of proven therapies, the potential role for cardiac transplantation is discussed.</description><identifier>ISSN: 1397-3142</identifier><identifier>EISSN: 1399-3046</identifier><identifier>DOI: 10.1111/j.1399-3046.2005.00414.x</identifier><identifier>PMID: 16573612</identifier><language>eng</language><publisher>Oxford, UK: Munksgaard International Publishers</publisher><subject>Arteries - pathology ; Basement Membrane - pathology ; Biological and medical sciences ; Calcinosis - pathology ; Calcinosis - surgery ; Cardiomyopathies - pathology ; Coronary Vessels - pathology ; Diphosphates - therapeutic use ; diphosphonate ; Disease Progression ; Fatal Outcome ; Female ; Heart Transplantation ; Humans ; idiopathic infantile arterial calcification ; Infant ; Medical sciences ; Myocardial Ischemia - etiology ; Myocardial Ischemia - pathology ; Myocardial Ischemia - surgery ; Myocardium - pathology ; pyrophosphate ; Surgery (general aspects). 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K.</creatorcontrib><title>Idiopathic infantile arterial calcification: Two case reports, a review of the literature and a role for cardiac transplantation</title><title>Pediatric transplantation</title><addtitle>Pediatr Transplant</addtitle><description>: Idiopathic infantile arterial calcification (IIAC) is a rare, but important, cause of rapidly progressive ischemic heart disease in children. In this paper, we report two recent cases of IIAC seen at tertiary referral hospitals. Both cases presented in infancy with signs of heart failure and, ultimately, died with the diagnosis of IIAC confirmed at postmortem examination. A thorough review of the literature reveals approximately 160 reported cases of IIAC. The clinical outcomes, radiographic findings and pathologic details are summarized. Proposed etiologic mechanisms are reviewed, including promising research into the role of inorganic pyrophosphate as a regulatory factor in the development of IIAC. Because of the typically fatal outcome of IIAC and the lack of proven therapies, the potential role for cardiac transplantation is discussed.</description><subject>Arteries - pathology</subject><subject>Basement Membrane - pathology</subject><subject>Biological and medical sciences</subject><subject>Calcinosis - pathology</subject><subject>Calcinosis - surgery</subject><subject>Cardiomyopathies - pathology</subject><subject>Coronary Vessels - pathology</subject><subject>Diphosphates - therapeutic use</subject><subject>diphosphonate</subject><subject>Disease Progression</subject><subject>Fatal Outcome</subject><subject>Female</subject><subject>Heart Transplantation</subject><subject>Humans</subject><subject>idiopathic infantile arterial calcification</subject><subject>Infant</subject><subject>Medical sciences</subject><subject>Myocardial Ischemia - etiology</subject><subject>Myocardial Ischemia - pathology</subject><subject>Myocardial Ischemia - surgery</subject><subject>Myocardium - pathology</subject><subject>pyrophosphate</subject><subject>Surgery (general aspects). 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Graft diseases</subject><subject>transplantation</subject><subject>Tunica Intima - pathology</subject><subject>Vascular Diseases</subject><issn>1397-3142</issn><issn>1399-3046</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2006</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqNkUtv1DAUhS0EoqXwF5A3sCLBjh9JkFhUVWmLKl4aHjvLcWzVQyYOtoeZ7vjp3MyM2iVElnLl-53jax-EMCUlhe_1sqSsbQtGuCwrQkRJCKe83D5Ax3eNh7u6Lhjl1RF6ktKSECp5wx-jIypFzSStjtGfq96HSecbb7AfnR6zHyzWMdvo9YCNHox33ujsw_gGLzYBtpLF0U4h5vQKayh_e7vBweF8Y_HgQanzOoLJ2M_tAH4uRNDF3muDc9RjmgY4aWf6FD1yekj22eF_gr6-O1-cXRbXHy-uzk6vC8Ol5EVPmKjrSrYwdt9oR52zEq7HW93Pi1bWatN3QHddzy3jHW8b6IgOuo1hJ-jl3neK4dfapqxWPhk7wCA2rJOSdVPRioh_grTlgjdCAtjsQRNDStE6NUW_0vFWUaLmmNRSzWmoOQ01x6R2MaktSJ8fzlh3K9vfCw-5APDiAOgEGTh4M-PTPVdLUouqBe7tnttAbLf_PYD6dL74AhXoi73ep2y3d3odf8KDsFqo7x8uFOPf3svLH5_B7C8SzL9g</recordid><startdate>200603</startdate><enddate>200603</enddate><creator>Glatz, Andrew C.</creator><creator>Pawel, Bruce R.</creator><creator>Hsu, Daphne T.</creator><creator>Weinberg, Paul</creator><creator>Chrisant, Maryanne R. K.</creator><general>Munksgaard International Publishers</general><general>Blackwell</general><scope>BSCLL</scope><scope>IQODW</scope><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7QP</scope><scope>7X8</scope></search><sort><creationdate>200603</creationdate><title>Idiopathic infantile arterial calcification: Two case reports, a review of the literature and a role for cardiac transplantation</title><author>Glatz, Andrew C. ; Pawel, Bruce R. ; Hsu, Daphne T. ; Weinberg, Paul ; Chrisant, Maryanne R. K.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c4664-d03577269361d8af1ffe639749ad9ad912eeacdbc46bbd4e34b4989ad5bad98c3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2006</creationdate><topic>Arteries - pathology</topic><topic>Basement Membrane - pathology</topic><topic>Biological and medical sciences</topic><topic>Calcinosis - pathology</topic><topic>Calcinosis - surgery</topic><topic>Cardiomyopathies - pathology</topic><topic>Coronary Vessels - pathology</topic><topic>Diphosphates - therapeutic use</topic><topic>diphosphonate</topic><topic>Disease Progression</topic><topic>Fatal Outcome</topic><topic>Female</topic><topic>Heart Transplantation</topic><topic>Humans</topic><topic>idiopathic infantile arterial calcification</topic><topic>Infant</topic><topic>Medical sciences</topic><topic>Myocardial Ischemia - etiology</topic><topic>Myocardial Ischemia - pathology</topic><topic>Myocardial Ischemia - surgery</topic><topic>Myocardium - pathology</topic><topic>pyrophosphate</topic><topic>Surgery (general aspects). Transplantations, organ and tissue grafts. Graft diseases</topic><topic>transplantation</topic><topic>Tunica Intima - pathology</topic><topic>Vascular Diseases</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Glatz, Andrew C.</creatorcontrib><creatorcontrib>Pawel, Bruce R.</creatorcontrib><creatorcontrib>Hsu, Daphne T.</creatorcontrib><creatorcontrib>Weinberg, Paul</creatorcontrib><creatorcontrib>Chrisant, Maryanne R. 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K.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Idiopathic infantile arterial calcification: Two case reports, a review of the literature and a role for cardiac transplantation</atitle><jtitle>Pediatric transplantation</jtitle><addtitle>Pediatr Transplant</addtitle><date>2006-03</date><risdate>2006</risdate><volume>10</volume><issue>2</issue><spage>225</spage><epage>233</epage><pages>225-233</pages><issn>1397-3142</issn><eissn>1399-3046</eissn><abstract>: Idiopathic infantile arterial calcification (IIAC) is a rare, but important, cause of rapidly progressive ischemic heart disease in children. In this paper, we report two recent cases of IIAC seen at tertiary referral hospitals. Both cases presented in infancy with signs of heart failure and, ultimately, died with the diagnosis of IIAC confirmed at postmortem examination. A thorough review of the literature reveals approximately 160 reported cases of IIAC. 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| subjects | Arteries - pathology Basement Membrane - pathology Biological and medical sciences Calcinosis - pathology Calcinosis - surgery Cardiomyopathies - pathology Coronary Vessels - pathology Diphosphates - therapeutic use diphosphonate Disease Progression Fatal Outcome Female Heart Transplantation Humans idiopathic infantile arterial calcification Infant Medical sciences Myocardial Ischemia - etiology Myocardial Ischemia - pathology Myocardial Ischemia - surgery Myocardium - pathology pyrophosphate Surgery (general aspects). Transplantations, organ and tissue grafts. Graft diseases transplantation Tunica Intima - pathology Vascular Diseases |
| title | Idiopathic infantile arterial calcification: Two case reports, a review of the literature and a role for cardiac transplantation |
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