Idiopathic infantile arterial calcification: Two case reports, a review of the literature and a role for cardiac transplantation

Andrew C. Glatz, Bruce R. Pawel, Daphne T. Hsu, Paul Weinberg, Maryanne R K Chrisant

Research output: Contribution to journalArticle

28 Citations (Scopus)

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. 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.

Original languageEnglish (US)
Pages (from-to)225-233
Number of pages9
JournalPediatric Transplantation
Volume10
Issue number2
DOIs
StatePublished - Mar 2006
Externally publishedYes

Fingerprint

Heart Transplantation
Fatal Outcome
Tertiary Care Centers
Myocardial Ischemia
Arterial calcification of infancy
Autopsy
Heart Failure
Research

Keywords

  • Diphosphonate
  • Idiopathic infantile arterial calcification
  • Pyrophosphate
  • Transplantation

ASJC Scopus subject areas

  • Pediatrics, Perinatology, and Child Health
  • Transplantation

Cite this

Idiopathic infantile arterial calcification : Two case reports, a review of the literature and a role for cardiac transplantation. / Glatz, Andrew C.; Pawel, Bruce R.; Hsu, Daphne T.; Weinberg, Paul; Chrisant, Maryanne R K.

In: Pediatric Transplantation, Vol. 10, No. 2, 03.2006, p. 225-233.

Research output: Contribution to journalArticle

Glatz, Andrew C. ; Pawel, Bruce R. ; Hsu, Daphne T. ; Weinberg, Paul ; Chrisant, Maryanne R K. / Idiopathic infantile arterial calcification : Two case reports, a review of the literature and a role for cardiac transplantation. In: Pediatric Transplantation. 2006 ; Vol. 10, No. 2. pp. 225-233.
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