{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,1]],"date-time":"2026-10-01T07:25:33Z","timestamp":1790839533677,"version":"4.1.0"},"reference-count":43,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2010,12,14]],"date-time":"2010-12-14T00:00:00Z","timestamp":1292284800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Pediatric Blood &amp; Cancer"],"published-print":{"date-parts":[[2011,2]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>Patients with Langerhans cell histiocytosis (LCH) may develop neurodegeneration and other central nervous system (CNS) dysfunctions revealed by brain magnetic resonance imaging (MRI). We estimated the incidence and pattern of pathological brain MRI findings in a well\u2010defined, population\u2010based cohort of children with LCH.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Among children under 15 years of age diagnosed with LCH in the Stockholm County during 1992\u20132001, brain MRI was performed at a single center in children with clinical and\/or laboratory signs of CNS involvement, including endocrine dysfunction.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Out of the 29 children (16 males, 13 females) diagnosed with LCH, brain MRI was performed based on clinical indications in 16 children (55%) with either abnormal endocrine findings (n\u2009=\u20096), such as diabetes insipidus (n\u2009=\u20095), low IGF\u20101 (n\u2009=\u20091), or panhypopituitarism (n\u2009=\u20091), or clinical CNS symptoms (n\u2009=\u200910). CNS MRI abnormalities were demonstrated in eight children (28%), at a median time of 3.5 years after LCH diagnosis (range 1\u201311.4 years). Altogether 7 of the 29 children (24%) had MRI findings associated with neurodegeneration, corresponding to a minimal incidence of 2.1\/10<jats:sup>6<\/jats:sup> children per year. Neurodegenerative abnormalities tended to be more frequent in patients with craniofacial involvement (<jats:italic>P<\/jats:italic>\u2009=\u20090.12).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>The minimal annual incidence rate of neurodegenerative associated radiographic findings in LCH is estimated at 2.1\/10<jats:sup>6<\/jats:sup> children (24% of all children with LCH). An important question is whether all patients with LCH, or certain forms of LCH, should be recommended for a late follow\u2010up examination including MRI. In patients with CNS\u2010LCH, neurological, neuropsychological, neurophysiological, neurochemical and neuroradiological follow\u2010up assessment is suggested. Pediatr Blood Cancer 2011;56:250\u2013257. \u00a9 2010 Wiley\u2010Liss, Inc.<\/jats:p><\/jats:sec>","DOI":"10.1002\/pbc.22791","type":"journal-article","created":{"date-parts":[[2010,12,14]],"date-time":"2010-12-14T22:27:29Z","timestamp":1292365649000},"page":"250-257","source":"Crossref","is-referenced-by-count":69,"title":["Incidence and pattern of radiological central nervous system Langerhans cell histiocytosis in children: A population based study"],"prefix":"10.1002","volume":"56","author":[{"given":"Evaldas","family":"Laurencikas","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"D\u00e9sir\u00e9e","family":"Gavhed","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Helen","family":"St\u00e5lemark","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ingrid van't","family":"Hooft","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daniela","family":"Prayer","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicole","family":"Grois","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jan\u2010Inge","family":"Henter","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2010,12,14]]},"reference":[{"key":"e_1_2_6_2_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0959-8049(99)00039-8"},{"key":"e_1_2_6_3_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0889-8588(05)70508-6"},{"key":"e_1_2_6_4_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(95)70248-2"},{"key":"e_1_2_6_5_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.critrevonc.2004.01.002"},{"key":"e_1_2_6_6_2","doi-asserted-by":"publisher","DOI":"10.1038\/nm1694"},{"key":"e_1_2_6_7_2","doi-asserted-by":"publisher","DOI":"10.1097\/00043426-198100320-00009"},{"key":"e_1_2_6_8_2","doi-asserted-by":"publisher","DOI":"10.1002\/mpo.2950080106"},{"key":"e_1_2_6_9_2","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.20102"},{"key":"e_1_2_6_10_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1651-2227.2005.tb02048.x"},{"key":"e_1_2_6_11_2","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.20021"},{"key":"e_1_2_6_12_2","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.20760"},{"key":"e_1_2_6_13_2","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2265.2000.01125.x"},{"key":"e_1_2_6_14_2","doi-asserted-by":"publisher","DOI":"10.1001\/archpedi.155.7.778"},{"key":"e_1_2_6_15_2","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.1996.14.7.2073"},{"key":"e_1_2_6_16_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2003.12.030"},{"key":"e_1_2_6_17_2","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.21656"},{"key":"e_1_2_6_18_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0889-8588(05)70511-6"},{"key":"e_1_2_6_19_2","first-page":"880","article-title":"MR imaging presentation of intracranial disease associated with Langerhans cell histiocytosis","volume":"25","author":"Prayer D","year":"2004","journal-title":"Am J Neuroradiol"},{"key":"e_1_2_6_20_2","first-page":"S24","article-title":"Central nervous system disease in Langerhans cell histiocytosis","volume":"23","author":"Grois N","year":"1994","journal-title":"Br J Cancer Suppl"},{"key":"e_1_2_6_21_2","first-page":"1365","article-title":"MR of the hypothalamic\u2010pituitary axis in Langerhans cell histiocytosis","volume":"13","author":"Maghnie M","year":"1992","journal-title":"Am J Neuroradiol"},{"key":"e_1_2_6_22_2","doi-asserted-by":"publisher","DOI":"10.1007\/s00330-006-0200-y"},{"key":"e_1_2_6_23_2","doi-asserted-by":"publisher","DOI":"10.1007\/s002340050781"},{"key":"e_1_2_6_24_2","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.21504"},{"key":"e_1_2_6_25_2","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-911X(199709)29:3<157::AID-MPO1>3.0.CO;2-C"},{"key":"e_1_2_6_26_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2007.12.042"},{"key":"e_1_2_6_27_2","doi-asserted-by":"publisher","DOI":"10.1002\/mpo.2950240407"},{"key":"e_1_2_6_28_2","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.20425"},{"key":"e_1_2_6_29_2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM200010053431403"},{"key":"e_1_2_6_30_2","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.2003.05.048"},{"key":"e_1_2_6_31_2","doi-asserted-by":"publisher","DOI":"10.3174\/ajnr.A0509"},{"key":"e_1_2_6_32_2","doi-asserted-by":"publisher","DOI":"10.1210\/jcem.85.4.6501"},{"key":"e_1_2_6_33_2","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/awh403"},{"key":"e_1_2_6_34_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(00)90062-3"},{"key":"e_1_2_6_35_2","first-page":"529","article-title":"High\u2010intensity signals within the posterior pituitary fossa: A study with fat\u2010suppression MR techniques","volume":"12","author":"Mark LP","year":"1991","journal-title":"Am J Neuroradiol"},{"key":"e_1_2_6_36_2","first-page":"1749","article-title":"Persistent high MR signal of the posterior pituitary gland in central diabetes insipidus","volume":"18","author":"Maghnie M","year":"1997","journal-title":"Am J Neuroradiol"},{"key":"e_1_2_6_37_2","first-page":"1921","article-title":"Demyelinating and gliotic cerebellar lesions in Langerhans cell histiocytosis","volume":"15","author":"Poe LB","year":"1994","journal-title":"Am J Neuroradiol"},{"key":"e_1_2_6_38_2","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.20.4.305"},{"key":"e_1_2_6_39_2","doi-asserted-by":"publisher","DOI":"10.1038\/nm0509-483"},{"key":"e_1_2_6_40_2","doi-asserted-by":"publisher","DOI":"10.1038\/nm1651"},{"key":"e_1_2_6_41_2","doi-asserted-by":"publisher","DOI":"10.1046\/j.1440-1681.2000.03259.x"},{"key":"e_1_2_6_42_2","doi-asserted-by":"publisher","DOI":"10.1212\/01.wnl.0000327680.74910.93"},{"key":"e_1_2_6_43_2","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.22238"},{"key":"e_1_2_6_44_2","doi-asserted-by":"publisher","DOI":"10.2214\/ajr.174.1.1740217"}],"container-title":["Pediatric Blood &amp; Cancer"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fpbc.22791","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fpbc.22791","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/pbc.22791","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,10]],"date-time":"2023-10-10T01:28:10Z","timestamp":1696901290000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/pbc.22791"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2010,12,14]]},"references-count":43,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2011,2]]}},"alternative-id":["10.1002\/pbc.22791"],"URL":"https:\/\/doi.org\/10.1002\/pbc.22791","archive":["Portico"],"relation":{},"ISSN":["1545-5009","1545-5017"],"issn-type":[{"value":"1545-5009","type":"print"},{"value":"1545-5017","type":"electronic"}],"subject":[],"published":{"date-parts":[[2010,12,14]]}}}