Infantile cortical hyperostosis (ICH) secondary to prostaglandin E1 infusion in a newborn with complex cyanotic heart disease: a case report

Infantile cortical hyperostosis (ICH), also known as Caffey’s disease, is a benign self-limiting condition affecting young infants. The initial symptom of the classical ICH is usually irritability, fever and soft tissue swelling affecting one or more bones. Diagnosis is usually delayed because its c...

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Main Authors:	Subramaniam, Kishan Rao, Karupiah, Rajandra Kumar, Mor Japar Khan, Ed Simor Khan, Zakaria@Mohamad, Zamzuri
Format:	Article
Language:	English
Published:	Crimson Publishers 2018
Subjects:	RD701 Orthopedics
Online Access:	http://irep.iium.edu.my/64154/ http://irep.iium.edu.my/64154/ http://irep.iium.edu.my/64154/ http://irep.iium.edu.my/64154/1/2018%20Infantile%20Cortical%20Hyperostosis%20Secondary%20to%20Prostaglandin%20E1%20Infusion%20in%20a%20Newborn%20with.pdf

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spelling	iium-641542018-11-19T08:24:40Z http://irep.iium.edu.my/64154/ Infantile cortical hyperostosis (ICH) secondary to prostaglandin E1 infusion in a newborn with complex cyanotic heart disease: a case report Subramaniam, Kishan Rao Karupiah, Rajandra Kumar Mor Japar Khan, Ed Simor Khan Zakaria@Mohamad, Zamzuri RD701 Orthopedics Infantile cortical hyperostosis (ICH), also known as Caffey’s disease, is a benign self-limiting condition affecting young infants. The initial symptom of the classical ICH is usually irritability, fever and soft tissue swelling affecting one or more bones. Diagnosis is usually delayed because its clinical presentation mimics many other clinical conditions such as osteomyelitis, scurvy, hypervitaminosis A, child abuse, bone tumor or even fracture. There are no laboratory tests to confirm the diagnosis. There are two types of ICH been described; the classical mild infantile form and the more severe prenatal form. Most of the cases of ICH are sporadic, but autosomal dominant and recessive patterns have been reported as well. It is linked with missense mutation in COL1A1, the gene encoding the α1 chain of type I collagen and has raised some doubts whether it is a type of collagen disorder, like osteogenesis imperfect. In this case, the newborn developed ICH due to prolonged infusion with prostaglandin E1 which was given in view of congenital cyanotic heart disease. Prostaglandin E1 infusion is used for maintaining the patency of ductus arteriosus in ductus dependent congenital heart defects in neonates and is usually administered before corrective heart surgery. ICH is mostly self-limiting and resolves within 12-24 months and usually does not require any treatment. Anti-inflammatory drugs such as NSAIDS(indomethacin and naproxen) and steroids has been used in symptomatic cases with success. The outcome is generally good with symptoms resolution usually before the age of one year. Relapses are uncommon. Crimson Publishers 2018 Article PeerReviewed application/pdf en http://irep.iium.edu.my/64154/1/2018%20Infantile%20Cortical%20Hyperostosis%20Secondary%20to%20Prostaglandin%20E1%20Infusion%20in%20a%20Newborn%20with.pdf Subramaniam, Kishan Rao and Karupiah, Rajandra Kumar and Mor Japar Khan, Ed Simor Khan and Zakaria@Mohamad, Zamzuri (2018) Infantile cortical hyperostosis (ICH) secondary to prostaglandin E1 infusion in a newborn with complex cyanotic heart disease: a case report. Orthopedic Research Online Journal, 4 (3). pp. 1-4. ISSN 2576-8875 https://crimsonpublishers.com/oproj/pdf/OPROJ.000588.pdf 10.31031/OPROJ.2018.04.000588
repository_type	Digital Repository
institution_category	Local University
institution	International Islamic University Malaysia
building	IIUM Repository
collection	Online Access
language	English
topic	RD701 Orthopedics
spellingShingle	RD701 Orthopedics Subramaniam, Kishan Rao Karupiah, Rajandra Kumar Mor Japar Khan, Ed Simor Khan Zakaria@Mohamad, Zamzuri Infantile cortical hyperostosis (ICH) secondary to prostaglandin E1 infusion in a newborn with complex cyanotic heart disease: a case report
description	Infantile cortical hyperostosis (ICH), also known as Caffey’s disease, is a benign self-limiting condition affecting young infants. The initial symptom of the classical ICH is usually irritability, fever and soft tissue swelling affecting one or more bones. Diagnosis is usually delayed because its clinical presentation mimics many other clinical conditions such as osteomyelitis, scurvy, hypervitaminosis A, child abuse, bone tumor or even fracture. There are no laboratory tests to confirm the diagnosis. There are two types of ICH been described; the classical mild infantile form and the more severe prenatal form. Most of the cases of ICH are sporadic, but autosomal dominant and recessive patterns have been reported as well. It is linked with missense mutation in COL1A1, the gene encoding the α1 chain of type I collagen and has raised some doubts whether it is a type of collagen disorder, like osteogenesis imperfect. In this case, the newborn developed ICH due to prolonged infusion with prostaglandin E1 which was given in view of congenital cyanotic heart disease. Prostaglandin E1 infusion is used for maintaining the patency of ductus arteriosus in ductus dependent congenital heart defects in neonates and is usually administered before corrective heart surgery. ICH is mostly self-limiting and resolves within 12-24 months and usually does not require any treatment. Anti-inflammatory drugs such as NSAIDS(indomethacin and naproxen) and steroids has been used in symptomatic cases with success. The outcome is generally good with symptoms resolution usually before the age of one year. Relapses are uncommon.
format	Article
author	Subramaniam, Kishan Rao Karupiah, Rajandra Kumar Mor Japar Khan, Ed Simor Khan Zakaria@Mohamad, Zamzuri
author_facet	Subramaniam, Kishan Rao Karupiah, Rajandra Kumar Mor Japar Khan, Ed Simor Khan Zakaria@Mohamad, Zamzuri
author_sort	Subramaniam, Kishan Rao
title	Infantile cortical hyperostosis (ICH) secondary to prostaglandin E1 infusion in a newborn with complex cyanotic heart disease: a case report
title_short	Infantile cortical hyperostosis (ICH) secondary to prostaglandin E1 infusion in a newborn with complex cyanotic heart disease: a case report
title_full	Infantile cortical hyperostosis (ICH) secondary to prostaglandin E1 infusion in a newborn with complex cyanotic heart disease: a case report
title_fullStr	Infantile cortical hyperostosis (ICH) secondary to prostaglandin E1 infusion in a newborn with complex cyanotic heart disease: a case report
title_full_unstemmed	Infantile cortical hyperostosis (ICH) secondary to prostaglandin E1 infusion in a newborn with complex cyanotic heart disease: a case report
title_sort	infantile cortical hyperostosis (ich) secondary to prostaglandin e1 infusion in a newborn with complex cyanotic heart disease: a case report
publisher	Crimson Publishers
publishDate	2018
url	http://irep.iium.edu.my/64154/ http://irep.iium.edu.my/64154/ http://irep.iium.edu.my/64154/ http://irep.iium.edu.my/64154/1/2018%20Infantile%20Cortical%20Hyperostosis%20Secondary%20to%20Prostaglandin%20E1%20Infusion%20in%20a%20Newborn%20with.pdf
first_indexed	2023-09-18T21:30:59Z
last_indexed	2023-09-18T21:30:59Z
_version_	1777412508957540352

Infantile cortical hyperostosis (ICH) secondary to prostaglandin E1 infusion in a newborn with complex cyanotic heart disease: a case report

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