{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,11,2]],"date-time":"2025-11-02T10:33:35Z","timestamp":1762079615405},"reference-count":13,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2017,12,2]],"date-time":"2017-12-02T00:00:00Z","timestamp":1512172800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Int J Dermatology"],"published-print":{"date-parts":[[2018,1]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>Incontinentia pigmenti (<jats:styled-content style=\"fixed-case\">IP<\/jats:styled-content>) is a rare inherited genodermatosis that usually involves the skin, and also teeth, oral cavity, central nervous system, eyes, blood with eosinophilia, and rarely skeletal system, breast, heart, and lungs. Skin lesions usually appear early, at birth or within the first 2\u00a0weeks of life, with four different phases tending to follow Blaschko lines that may overlap.<\/jats:p><\/jats:sec><jats:sec><jats:title>Case report<\/jats:title><jats:p>We report a rare case of a neonate with transient reversible pulmonary hypertension that presented at day 9 of life. She manifested increasing dyspnea and deterioration of respiratory dynamics with a serious pulmonary hypertension without a primary pulmonary disease. Hence, oxygen therapy at high flows and nitric oxide have been administered with an initial response, but, subsequently, because of the worsening of the respiratory activity, she underwent sildenafil and bosentan treatment with respiratory dynamics improvement and progressive decrease of the pulmonary pressures.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>In literature only a few cases of cardiopulmonary anomalies in <jats:styled-content style=\"fixed-case\">IP<\/jats:styled-content> have been described with different outcomes, and these rare complications are probably underestimated by physicians. We could suppose that microangiopathic damages may have a critical role in endothelial alterations, and these processes are probably shared by multiple organs involved in <jats:styled-content style=\"fixed-case\">IP<\/jats:styled-content> and rarely by lungs and heart.<\/jats:p><\/jats:sec>","DOI":"10.1111\/ijd.13835","type":"journal-article","created":{"date-parts":[[2017,12,2]],"date-time":"2017-12-02T06:56:39Z","timestamp":1512197799000},"page":"40-45","update-policy":"http:\/\/dx.doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":10,"title":["Cardiopulmonary anomalies in incontinentia pigmenti patients"],"prefix":"10.1111","volume":"57","author":[{"given":"Giuliana","family":"Onnis","sequence":"first","affiliation":[{"name":"Dermatology Unit Department of Surgical, Microsurgical and Medical Sciences University of Sassari Sassari Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Andrea","family":"Diociaiuti","sequence":"additional","affiliation":[{"name":"Dermatology Unit Academic Department of Pediatrics Children's Hospital Bambino Ges\u00f9 \u2010IRCCS Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Paola","family":"Zangari","sequence":"additional","affiliation":[{"name":"Unit of Immune and Infectious Diseases Academic Department of Pediatrics Children's Hospital Bambino Ges\u00f9 Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Patrizia","family":"D'Argenio","sequence":"additional","affiliation":[{"name":"Unit of Immune and Infectious Diseases Academic Department of Pediatrics Children's Hospital Bambino Ges\u00f9 Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Caterina","family":"Cancrini","sequence":"additional","affiliation":[{"name":"Unit of Immune and Infectious Diseases Academic Department of Pediatrics Children's Hospital Bambino Ges\u00f9 Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lorenzo","family":"Iughetti","sequence":"additional","affiliation":[{"name":"Department of Pediatrics University of Modena and Reggio Emilia Modena Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"May","family":"El Hachem","sequence":"additional","affiliation":[{"name":"Dermatology Unit Academic Department of Pediatrics Children's Hospital Bambino Ges\u00f9 \u2010IRCCS Rome Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2017,12,2]]},"reference":[{"key":"e_1_2_4_2_1","first-page":"404","article-title":"Eigentumliche, bisher nicht and beschriebene pigmentaffection (incontinentia pigmenti)","volume":"7","author":"Bloch B","year":"1926","journal-title":"Pigmentaffection Schweiz Med Wschr"},{"key":"e_1_2_4_3_1","doi-asserted-by":"crossref","first-page":"19","DOI":"10.1007\/BF01828398","article-title":"Uber eine bisher nicht beschriebene congenitale pigmentanomalie (incontinentia pigmenti)","volume":"154","author":"Sulzeberger MB","year":"1928","journal-title":"Archiv f\u00fcr Dermatologie und Syphilis"},{"key":"e_1_2_4_4_1","doi-asserted-by":"publisher","DOI":"10.1590\/abd1806-4841.20142584"},{"key":"e_1_2_4_5_1","first-page":"496","article-title":"Incontinentia pigmenti (Bloch\u2010Sulzberger syndrome) in neonates","volume":"41","author":"Buinauskiene J","year":"2005","journal-title":"Medicina (Kaunas)"},{"key":"e_1_2_4_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0887-8994(03)00150-4"},{"key":"e_1_2_4_7_1","doi-asserted-by":"crossref","first-page":"289","DOI":"10.1038\/sj.jp.7211250","article-title":"Unusual neonatal presentation of incontinentia pigmenti with persistent pulmonary hypertension of the newborn: a case report","volume":"2","author":"Godambe S","year":"2005","journal-title":"J Perinatol"},{"key":"e_1_2_4_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.30698"},{"key":"e_1_2_4_9_1","first-page":"773","article-title":"Cerebral complications of incontinentia pigmenti. 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