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• W2058531100 abstract "To document the frequency and outcome of endocrine involvement in pediatric-onset Langerhans' cell histiocytosis (LCH), and the association with other types of organ involvement.This retrospective nationwide multicenter study involved 589 patients with pediatric-onset LCH, 148 of whom had endocrine dysfunction. Median follow-up was 11.6 years.Pituitary dysfunction was present in 145 patients, and 141 had diabetes insipidus (DI). The estimated 10-year risks of pituitary involvement were 24.2% +/- 1.8%. GH deficiency occurred in 61 patients. Median age at onset was 2.8 years for LCH, 3.9 years for DI, and 7.7 years for GH deficiency. The risk of cranial involvement; ear, nose, and throat involvement; pneumothorax; and cholangitis was significantly higher in patients with endocrinopathy. The chronology of episodes did not support a causal link between pituitary involvement and involvement of other organs. Systemic treatment of LCH did not prevent pituitary involvement. The most severe complication was a neurodegenerative syndrome, which affected 4.3% and 10.8% of patients, respectively, 5 and 15 years after initial diagnosis, and appeared to be linked to pituitary involvement.Patients who develop endocrine LCH disorders are at a high risk of neurodegenerative LCH and require long-term follow-up." @default.
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• W2058531100 date "2004-03-01" @default.
• W2058531100 modified "2023-10-16" @default.
• W2058531100 title "Endocrine involvement in pediatric-onset langerhans' cell histiocytosis: a population-based study" @default.
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• W2058531100 doi "https://doi.org/10.1016/j.jpeds.2003.12.030" @default.
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