{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,13]],"date-time":"2026-10-13T12:23:09Z","timestamp":1791894189057,"version":"4.3.4"},"reference-count":14,"publisher":"American Academy of Pediatrics (AAP)","issue":"1","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2016,1,1]]},"abstract":"<jats:p>Severe vitamin D deficiency and rickets are highly prevalent among children with congenital ichthyosis. We report an incidental observation of a dramatic and excellent clinical response with regard to skin scaling and stiffness in children with congenital ichthyosis after short-term high-dose vitamin D supplementation that has not been previously described. Seven children with congenital ichthyosis (5 with autosomal recessive congenital ichthyosis; 2 with epidermolytic ichthyosis) and severe vitamin D deficiency (and\/or rickets) were given 60\u2009000 IU of oral cholecalciferol daily for 10 days under supervision. All children were subsequently put on recommended daily allowance of 400 to 600 IU of cholecalciferol. The main outcome measures observed and studied were reduction in skin scaling and stiffness of the extremities. All cases had severe vitamin D deficiency (serum 25-hydroxyvitamin D &amp;lt; 4 ng\/mL) and secondary hyperparathyroidism. Six patients had clinical and radiologic evidence of rickets. Significant improvement in scaling was noticeable by day 5, showing further improvement by day 10, in 6 of the 7 cases. At 1 month, the skin had become near normal in all the cases of autosomal recessive congenital ichthyosis. Remarkable reduction in stiffness was also observed in all children. Supplementation with high-dose vitamin D followed by recommended daily allowance appears to be an effective form of therapy in the management of congenital ichthyosis with vitamin D deficiency.<\/jats:p>","DOI":"10.1542\/peds.2015-1313","type":"journal-article","created":{"date-parts":[[2016,1,1]],"date-time":"2016-01-01T02:58:26Z","timestamp":1451617106000},"source":"Crossref","is-referenced-by-count":33,"title":["Vitamin D: A New Promising Therapy for Congenital Ichthyosis"],"prefix":"10.1542","volume":"137","author":[{"given":"Gomathy","family":"Sethuraman","sequence":"first","affiliation":[{"name":"Departments of Dermatology"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Raman K","family":"Marwaha","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Institute of Nuclear Medicine and Allied Sciences, DRDO, New Delhi, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Apoorva","family":"Challa","sequence":"additional","affiliation":[{"name":"Departments of Dermatology"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vamsi K","family":"Yenamandra","sequence":"additional","affiliation":[{"name":"Departments of Dermatology"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lakshmy","family":"Ramakrishnan","sequence":"additional","affiliation":[{"name":"Cardiac Biochemistry, and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sanjay","family":"Thulkar","sequence":"additional","affiliation":[{"name":"Radiodiagnosis, All India Institute of Medical Sciences, New Delhi, India; and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vinod K","family":"Sharma","sequence":"additional","affiliation":[{"name":"Departments of Dermatology"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"417","published-online":{"date-parts":[[2016,1,1]]},"reference":[{"key":"2021120907191157400_B1","doi-asserted-by":"crossref","first-page":"603","DOI":"10.1111\/j.1365-2133.2007.08355.x","article-title":"Ichthyosiform erythroderma with rickets: report of five cases.","volume":"158","author":"Sethuraman","year":"2008","journal-title":"Br J Dermatol"},{"issue":"3","key":"2021120907191157400_B2","doi-asserted-by":"crossref","first-page":"608","DOI":"10.1111\/j.1365-2133.2011.10672.x","article-title":"Vitamin D deficiency and rickets in children and adolescents with ichthyosiform erythroderma in type IV and V skin.","volume":"166","author":"Chouhan","year":"2012","journal-title":"Br J Dermatol"},{"issue":"1","key":"2021120907191157400_B3","doi-asserted-by":"crossref","first-page":"208","DOI":"10.1111\/bjd.13131","article-title":"Threshold levels of 25-hydroxyvitamin D and parathyroid hormone for impaired bone health in children with congenital ichthyosis and type IV and V skin.","volume":"172","author":"Sethuraman","year":"2015","journal-title":"Br J Dermatol"},{"issue":"2","key":"2021120907191157400_B4","doi-asserted-by":"crossref","first-page":"398","DOI":"10.1542\/peds.2007-1894","article-title":"Vitamin D deficiency in children and its management: review of current knowledge and recommendations.","volume":"122","author":"Misra","year":"2008","journal-title":"Pediatrics"},{"issue":"1","key":"2021120907191157400_B5","doi-asserted-by":"crossref","first-page":"e119","DOI":"10.1542\/peds.114.1.e119","article-title":"Nutritional rickets in ichthyosis and response to calcipotriene.","volume":"114","author":"Thacher","year":"2004","journal-title":"Pediatrics"},{"issue":"1-2","key":"2021120907191157400_B6","doi-asserted-by":"crossref","first-page":"80","DOI":"10.1016\/j.mce.2011.05.017","article-title":"Vitamin D metabolism and function in the skin.","volume":"347","author":"Bikle","year":"2011","journal-title":"Mol Cell Endocrinol"},{"key":"2021120907191157400_B7","author":"Holick"},{"issue":"4","key":"2021120907191157400_B8","doi-asserted-by":"crossref","first-page":"778","DOI":"10.1111\/j.0022-202X.2005.23641.x","article-title":"Transcriptional profiling of keratinocytes reveals a vitamin D-regulated epidermal differentiation network.","volume":"124","author":"Lu","year":"2005","journal-title":"J Invest Dermatol"},{"issue":"12","key":"2021120907191157400_B9","doi-asserted-by":"crossref","first-page":"686","DOI":"10.5694\/j.1326-5377.2010.tb03702.x","article-title":"Efficacy and safety of oral continuous low-dose versus short-term high-dose vitamin D: a prospective randomised trial conducted in a clinical setting.","volume":"192","author":"Hackman","year":"2010","journal-title":"Med J Aust"},{"issue":"3","key":"2021120907191157400_B10","doi-asserted-by":"crossref","first-page":"203","DOI":"10.1093\/tropej\/fmt105","article-title":"A Randomized controlled trial on safety and efficacy of single intramuscular versus staggered oral dose of 600 000IU vitamin D in treatment of nutritional rickets.","volume":"60","author":"Mondal","year":"2014","journal-title":"J Trop Pediatr"},{"issue":"1","key":"2021120907191157400_B11","doi-asserted-by":"crossref","first-page":"23","DOI":"10.1159\/000248505","article-title":"A trial of oral 1 alpha-hydroxyvitamin D3 for ichthyosis.","volume":"177","author":"Okano","year":"1988","journal-title":"Dermatologica"},{"issue":"1","key":"2021120907191157400_B12","doi-asserted-by":"crossref","first-page":"52","DOI":"10.1080\/00015550310002738","article-title":"Bullous congenital ichthyosiform erythroderma: safe and effective topical treatment with calcipotriol ointment in a child.","volume":"83","author":"Bogenrieder","year":"2003","journal-title":"Acta Derm Venereol"},{"issue":"4","key":"2021120907191157400_B13","doi-asserted-by":"crossref","first-page":"546","DOI":"10.1111\/j.1365-2133.1994.tb08558.x","article-title":"Effect of topical calcipotriol on congenital ichthyoses.","volume":"131","author":"Lucker","year":"1994","journal-title":"Br J Dermatol"},{"issue":"1","key":"2021120907191157400_B14","doi-asserted-by":"crossref","first-page":"26","DOI":"10.1111\/j.1529-8019.2012.01527.x","article-title":"Systemic retinoids in the management of ichthyoses and related skin types.","volume":"26","author":"Digiovanna","year":"2013","journal-title":"Dermatol Ther (Heidelb)"}],"container-title":["Pediatrics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/publications.aap.org\/pediatrics\/article-pdf\/doi\/10.1542\/peds.2015-1313\/1061106\/peds_20151313.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/publications.aap.org\/pediatrics\/article-pdf\/doi\/10.1542\/peds.2015-1313\/1061106\/peds_20151313.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,12,10]],"date-time":"2021-12-10T01:56:12Z","timestamp":1639101372000},"score":1,"resource":{"primary":{"URL":"https:\/\/publications.aap.org\/pediatrics\/article\/137\/1\/e20151313\/52803\/Vitamin-D-A-New-Promising-Therapy-for-Congenital"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2016,1,1]]},"references-count":14,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2016,1,1]]},"published-print":{"date-parts":[[2016,1,1]]}},"URL":"https:\/\/doi.org\/10.1542\/peds.2015-1313","relation":{},"ISSN":["0031-4005","1098-4275"],"issn-type":[{"value":"0031-4005","type":"print"},{"value":"1098-4275","type":"electronic"}],"subject":[],"published":{"date-parts":[[2016,1,1]]},"article-number":"e20151313"}}