{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,12,22]],"date-time":"2025-12-22T10:55:44Z","timestamp":1766400944179},"reference-count":10,"publisher":"Walter de Gruyter GmbH","issue":"9","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2018,9,25]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec id=\"j_jpem-2018-0049_s_999_w2aab3b7c18b1b6b1aab1c16b1Aa\">\n                  <jats:title>Background<\/jats:title>\n                  <jats:p>Van Wyk-Grumbach syndrome (VWGS) is characterized by juvenile primary hypothyroidism, delayed bone age and isosexual incomplete precocious puberty with reversal to the prepubertal state following thyroid hormone replacement.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2018-0049_s_998_w2aab3b7c18b1b6b1aab1c16b2Aa\">\n                  <jats:title>Case presentation<\/jats:title>\n                  <jats:p>In this case, an 18-month-old girl presented with premature menarche since 9 months of age, delayed bone age and enlarged bilateral multicystic ovaries along with a superficial infantile hemangioma over the upper anterior chest. VWGS was diagnosed based on the clinical features. High serum thyroid stimulating hormone and low free thyroxine with the absence of any carpal bones in the wrist X-ray were suggestive of congenital hypothyroidism. Interestingly, the coexisting hemangioma could also play a role in the etiology of the hypothyroidism through \u201cconsumptive hypothyroidism\u201d. Thyroid hormone replacement resulted in the complete resolution of signs and symptoms.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2018-0049_s_997_w2aab3b7c18b1b6b1aab1c16b3Aa\">\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>Untreated congenital hypothyroidism of short duration, onset of symptoms in infancy and association of an infantile hemangioma in VWGS were the unique features in our case.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1515\/jpem-2018-0049","type":"journal-article","created":{"date-parts":[[2018,7,20]],"date-time":"2018-07-20T22:16:32Z","timestamp":1532124992000},"page":"1057-1060","source":"Crossref","is-referenced-by-count":5,"title":["Van Wyk-Grumbach syndrome with hemangioma in an infant"],"prefix":"10.1515","volume":"31","author":[{"given":"Moumita","family":"Biswas","sequence":"first","affiliation":[{"name":"Department of Pediatric Medicine , Institute of Medical Education and Research , Kolkata, West Bengal , India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Malay Kumar","family":"Sinha","sequence":"additional","affiliation":[{"name":"Department of Pediatric Medicine , Institute of Medical Education and Research , Kolkata, West Bengal , India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mrinal Kanti","family":"Das","sequence":"additional","affiliation":[{"name":"Department of Pediatric Medicine , Institute of Medical Education and Research , Kolkata, West Bengal , India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sumantra","family":"Sarkar","sequence":"additional","affiliation":[{"name":"Department of Pediatric Medicine , Institute of Medical Education and Research , Kolkata, West Bengal , India"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"374","published-online":{"date-parts":[[2018,7,20]]},"reference":[{"key":"2021021318143891261_j_jpem-2018-0049_ref_001_w2aab3b7c18b1b6b1ab2b1b1Aa","doi-asserted-by":"crossref","unstructured":"Kendle FW. Case of precocious puberty in a female cretin. Br Med J 1905;1:246.","DOI":"10.1136\/bmj.1.2301.246"},{"key":"2021021318143891261_j_jpem-2018-0049_ref_002_w2aab3b7c18b1b6b1ab2b1b2Aa","doi-asserted-by":"crossref","unstructured":"VanWyk JJ, Grumbach MM. Syndrome of precocious menstruation and galactorrhea in juvenile hypothyroidism. An example of hormonal overlap in pituitary feedback. J Pediatr 1960; 57:416\u201335.","DOI":"10.1016\/S0022-3476(60)80250-8"},{"key":"2021021318143891261_j_jpem-2018-0049_ref_003_w2aab3b7c18b1b6b1ab2b1b3Aa","doi-asserted-by":"crossref","unstructured":"Baranowsky E, Hogler W. An unusual presentation of acquired hypothyroidism: the Van Wyk-Grumbach syndrome. Eur J Endocrinol 2012;166:537\u201342.","DOI":"10.1530\/EJE-11-0494"},{"key":"2021021318143891261_j_jpem-2018-0049_ref_004_w2aab3b7c18b1b6b1ab2b1b4Aa","doi-asserted-by":"crossref","unstructured":"Dutta D, Kumar M, Jain R, Thukral A, Ghosh S, et al. Pituitary hyperplasia and Van Wyk Grumbach syndrome: a consequence of chronic untreated congenital hypothyroidism. J ASEAN Fed Endocr Soc 2013;28:88\u20139.","DOI":"10.15605\/jafes.028.01.16"},{"key":"2021021318143891261_j_jpem-2018-0049_ref_005_w2aab3b7c18b1b6b1ab2b1b5Aa","doi-asserted-by":"crossref","unstructured":"Kroeze WK, Sheffler DJ, Roth BL. G protein coupled receptors at a glance. J Cell Sci 2003;116:4867\u20139.","DOI":"10.1242\/jcs.00902"},{"key":"2021021318143891261_j_jpem-2018-0049_ref_006_w2aab3b7c18b1b6b1ab2b1b6Aa","doi-asserted-by":"crossref","unstructured":"Christens A, Sevenants L, Toelen J, Bullens D, Casteels K. Van Wyk and Grumbach syndrome: an unusual form of precocious puberty. Gynecol Endocrinol 2014;30:272\u20136.","DOI":"10.3109\/09513590.2013.871523"},{"key":"2021021318143891261_j_jpem-2018-0049_ref_007_w2aab3b7c18b1b6b1ab2b1b7Aa","unstructured":"Garibaldi LR, Chemaitilly W. Disorder of pubertal development. In: Behrman RE, Kliegman RM, Staton BF, St. Geme JW, Schor NF, editors. Nelson textbook of pediatrics. 20th ed. Philadelphia: Elsevier, 2016:2656."},{"key":"2021021318143891261_j_jpem-2018-0049_ref_008_w2aab3b7c18b1b6b1ab2b1b8Aa","doi-asserted-by":"crossref","unstructured":"Ayub SS, Ruzic A, Taylor JA. Ovarian cysts, vaginal bleeding and hypothyroidism in a 4-year-old female with down syndrome: a case of Van Wyk-Grumbach syndrome. J Pediatr Surg Case Rep 2017;25:5\u20139.","DOI":"10.1016\/j.epsc.2017.07.007"},{"key":"2021021318143891261_j_jpem-2018-0049_ref_009_w2aab3b7c18b1b6b1ab2b1b9Aa","doi-asserted-by":"crossref","unstructured":"Chakraborty PP, Bera M, Patra S, Roy S. BMJ Case Rep. Published Online First: 19th July 2017. doi:10.1136\/bcr-2017-221366.","DOI":"10.1136\/bcr-2017-221366"},{"key":"2021021318143891261_j_jpem-2018-0049_ref_010_w2aab3b7c18b1b6b1ab2b1c10Aa","doi-asserted-by":"crossref","unstructured":"Luongo C, Trivisano L, Alfano F, Salvatore D. 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