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• W1969132177 abstract "From its very beginnings, the history of Langerhans cell histiocytosis (LCH) has been inseparable from its central nervous system (CNS) manifestations. Hand36 in 1893 reported on a patient with “polyuria and tuberculosis,” which is now recognized as a typical case of diabetes insipidus and multisystem disease with exophthalmos. Other early reports14, 28, 44, 71 described characteristic cases with skull lesions and diabetes insipidus (DI). A comprehensive treatise on LCH and the nervous system has been provided by Kepes,45 who illustrated the wide range of clinical and histopathologic presentations of the disease. Apart from the common manifestations in the hypothalamic-pituitary axis, virtually all other parts of the CNS can be affected by LCH. The plethora of symptoms depends on the type and site of involvement. Clinically, four groups of patients can be distinguished: (1) those with a disorder of the hypothalamic-pituitary system; (2) patients presenting with site-dependent symptoms from space-occupying lesions, such as headaches or seizures; (3) those who exhibit a neurologic dysfunction mostly following a cerebellar-pontine pathway, including reflex abnormalities, ataxia, intellectual impairment, tremor, or dysarthria with variable progression to severe CNS deterioration; and (4) patients who present with an overlap of the symptoms mentioned previously. During the last few years, a number of anecdotal case reports and small series of patients have appeared describing patients with variable CNS findings that were evaluated by modern imaging techniques.1, 27, 62, 65 In particular, MR imaging has provided an appropriate tool to facilitate the diagnosis and follow-up of CNS changes, even in patients with no or minimal neurologic symptoms.7, 26, 29, 35, 57, 64, 66, 75 There are still a number of unanswered questions regarding LCH-CNS disease. The actual incidence is unknown and the natural history still remains poorly understood. In this article, the wide spectrum of clinical manifestations of LCH-CNS are presented and correlated with the morphologic changes seen on imaging studies together with the histopathologic findings. Finally, the authors speculate on the pathogenesis, suggest guidelines for management of such patients, and discuss the therapeutic options." @default.
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• W1969132177 date "1998-04-01" @default.
• W1969132177 modified "2023-10-14" @default.
• W1969132177 title "CENTRAL NERVOUS SYSTEM DISEASE IN LANGERHANS CELL HISTIOCYTOSIS" @default.
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• W1969132177 doi "https://doi.org/10.1016/s0889-8588(05)70511-6" @default.
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