| Hyperprostaglandin E syndrome: use of indomethacin and steroid, and death due to necrotizing enterocolitis and sepsis. | |
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MedLine Citation:
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PMID: 19014056 Owner: NLM Status: MEDLINE |
Abstract/OtherAbstract:
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Hyperprostaglandin E syndrome (HPS) is the antenatal variant of Bartter syndrome and characterized by polyhydramnios and preterm delivery in the antenatal period and salt-wasting, isosthenuric or hyposthenuric polyuria, hypercalciuria and nephrocalcinosis in the postnatal period. We report a one-month-old infant with HPS with a 15-year-old sister with Bartter syndrome. The infant's birth weight was 2750 g and she had severe dehydration on the 2nd day of life. She had hypercalcemia, hyponatremia, hypokalemia, metabolic alkalosis and elevated plasma renin and aldosterone levels. We instituted indomethacin therapy accompanied by steroid therapy for hypercalcemia. However, the patient developed abdominal distention on the 30th day, which was due to diffuse pneumatosis in sigmoid colon revealed by a subsequent surgical intervention. Following surgery, the patient developed fever, electrolyte abnormalities and subsequently sepsis. The patient died due to sepsis 10 days after surgery. We conclude that indomethacin and steroid therapy must be used cautiously in infants with HPS. |
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Authors:
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Merih Cetinkaya; Nilgün Köksal; Hilal Ozkan; Osman Dönmez; Halil Sağlam; Irfan Kiriştioğlu |
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Publication Detail:
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Type: Case Reports; Journal Article |
Journal Detail:
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Title: The Turkish journal of pediatrics Volume: 50 ISSN: 0041-4301 ISO Abbreviation: Turk. J. Pediatr. Publication Date: 2008 Jul-Aug |
Date Detail:
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Created Date: 2008-11-18 Completed Date: 2008-12-04 Revised Date: - |
Medline Journal Info:
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Nlm Unique ID: 0417505 Medline TA: Turk J Pediatr Country: Turkey |
Other Details:
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Languages: eng Pagination: 386-90 Citation Subset: IM |
Affiliation:
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Division of Neonatology, Department of Pediatrics, Uludağ University Faculty of Medicine, Bursa, Turkey. |
Export Citation:
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| MeSH Terms | |
Descriptor/Qualifier:
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Anti-Inflammatory Agents, Non-Steroidal
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therapeutic use Bartter Syndrome / drug therapy, etiology, physiopathology* Enterocolitis, Necrotizing / complications* Fatal Outcome Female Humans Indomethacin / therapeutic use Infant, Newborn Polyhydramnios Pregnancy Sepsis / complications* Steroids / therapeutic use |
| Chemical | |
Reg. No./Substance:
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0/Anti-Inflammatory Agents, Non-Steroidal; 0/Steroids; 53-86-1/Indomethacin |
From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine
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