A Rare Cause of Persistent Pulmonary Hypertension Resistant to Therapy in The Newborn: Short-Rib Polydactyly Syndrome

Short-rib polydactyly syndrome is an autosomal recessively inherited lethal skeletal dysplasia. The syndrome is characterized by marked narrow fetal thorax, short extremities, micromelia, cleft palate/lip, polydactyly, cardiac and renal abnormalities, and genital malformations. In cases with pulmona...

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Main Authors: Nihat Demir, Erdal Peker, İbrahim Ece, Sultan Kaba, Kemal Ağengin, Oğuz Tuncer
Format: Article
Language:English
Published: Wiley 2015-01-01
Series:Case Reports in Pulmonology
Online Access:http://dx.doi.org/10.1155/2015/274639
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author Nihat Demir
Erdal Peker
İbrahim Ece
Sultan Kaba
Kemal Ağengin
Oğuz Tuncer
author_facet Nihat Demir
Erdal Peker
İbrahim Ece
Sultan Kaba
Kemal Ağengin
Oğuz Tuncer
author_sort Nihat Demir
collection DOAJ
description Short-rib polydactyly syndrome is an autosomal recessively inherited lethal skeletal dysplasia. The syndrome is characterized by marked narrow fetal thorax, short extremities, micromelia, cleft palate/lip, polydactyly, cardiac and renal abnormalities, and genital malformations. In cases with pulmonary hypoplasia, persistent pulmonary hypertension of the newborn can develop. In this paper, we present a term newborn with persistent pulmonary hypertension of the newborn, which has developed secondary to short-rib polydactyly syndrome and was resistant to therapy with inhaled nitric oxide and oral sildenafil.
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institution Kabale University
issn 2090-6846
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language English
publishDate 2015-01-01
publisher Wiley
record_format Article
series Case Reports in Pulmonology
spelling doaj-art-13ffa4fc26b64a0eb2f208b018a91a6d2025-08-20T03:54:33ZengWileyCase Reports in Pulmonology2090-68462090-68542015-01-01201510.1155/2015/274639274639A Rare Cause of Persistent Pulmonary Hypertension Resistant to Therapy in The Newborn: Short-Rib Polydactyly SyndromeNihat Demir0Erdal Peker1İbrahim Ece2Sultan Kaba3Kemal Ağengin4Oğuz Tuncer5Department of Pediatrics, Division of Neonatology, Yuzuncu Yil University School of Medicine, 65080 Van, TurkeyDepartment of Pediatrics, Division of Neonatology, Yuzuncu Yil University School of Medicine, 65080 Van, TurkeyDepartment of Pediatrics, Division of Cardiology, Yuzuncu Yil University School of Medicine, 65080 Van, TurkeyDepartment of Pediatrics, Division of Endocrinology, Yuzuncu Yil University School of Medicine, 65080 Van, TurkeyDepartment of Pediatric Surgery, Yuzuncu Yil University School of Medicine, 65080 Van, TurkeyDepartment of Pediatrics, Division of Neonatology, Yuzuncu Yil University School of Medicine, 65080 Van, TurkeyShort-rib polydactyly syndrome is an autosomal recessively inherited lethal skeletal dysplasia. The syndrome is characterized by marked narrow fetal thorax, short extremities, micromelia, cleft palate/lip, polydactyly, cardiac and renal abnormalities, and genital malformations. In cases with pulmonary hypoplasia, persistent pulmonary hypertension of the newborn can develop. In this paper, we present a term newborn with persistent pulmonary hypertension of the newborn, which has developed secondary to short-rib polydactyly syndrome and was resistant to therapy with inhaled nitric oxide and oral sildenafil.http://dx.doi.org/10.1155/2015/274639
spellingShingle Nihat Demir
Erdal Peker
İbrahim Ece
Sultan Kaba
Kemal Ağengin
Oğuz Tuncer
A Rare Cause of Persistent Pulmonary Hypertension Resistant to Therapy in The Newborn: Short-Rib Polydactyly Syndrome
Case Reports in Pulmonology
title A Rare Cause of Persistent Pulmonary Hypertension Resistant to Therapy in The Newborn: Short-Rib Polydactyly Syndrome
title_full A Rare Cause of Persistent Pulmonary Hypertension Resistant to Therapy in The Newborn: Short-Rib Polydactyly Syndrome
title_fullStr A Rare Cause of Persistent Pulmonary Hypertension Resistant to Therapy in The Newborn: Short-Rib Polydactyly Syndrome
title_full_unstemmed A Rare Cause of Persistent Pulmonary Hypertension Resistant to Therapy in The Newborn: Short-Rib Polydactyly Syndrome
title_short A Rare Cause of Persistent Pulmonary Hypertension Resistant to Therapy in The Newborn: Short-Rib Polydactyly Syndrome
title_sort rare cause of persistent pulmonary hypertension resistant to therapy in the newborn short rib polydactyly syndrome
url http://dx.doi.org/10.1155/2015/274639
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