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- W2085406905 abstract "In this issue, Spagnolo et al. described a case of 58-yearold adult presenting Langerhans cell histiocytosis (LCH) pathology, in which psychic changes, dysarthria, dysphagia, opsoclonus, dysmetria and tremor were associated. MRI detected bilateral T2-hyperintense alterations within the dentate nuclei and adjacent white matter involving the middle cerebellar peduncles. In the past medical history, at the age of 38, diabetes insipidus was diagnosed and treated with desmopressin with a good response [1]. This report focuses the attention on the neurodegeneration in the course of LCH, mainly characterized by ataxia. In an interesting article, Wnorowski et al. collected 83 LCH cases, 25 % of them presenting with neurological disorders (tremor, cerebellar ataxia, dysarthria, dysdiadochokinesia, psychomotor retardation and psychosis), with onset 3–15 years after initial LCH diagnosis. The immunopathological pathogenesis of LCH suggests that this condition may be considered as a paraneoplastic disorder of the CNS, in which cerebellum is a special target [2]. Very often, contrary to this case in which neurodegeneration was detected after the LCH diagnosis, patients may have an infantile LCH onset with minimal signs followed by a slow progressive neurological disorder mimicking a neurodegenerative disease. A delayed diagnosis was reported by Jarquin-Valdivia and Buchhalter [3] in 2001 in a boy who presented with primarily neurological and endocrinological findings without pain, from a series of LCH paediatric patients (65) seen at Mayo Clinic. As an example of this, we here report a 29-year-old male recently referred to us for progressive gait disturbances and dysarthria." @default.
- W2085406905 created "2016-06-24" @default.
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- W2085406905 date "2012-05-09" @default.
- W2085406905 modified "2023-09-26" @default.
- W2085406905 title "Progressive neurodegenerative syndrome associated with Langerhans cell histiocytosis: a rare condition that we have to consider in patients with sporadic spastic ataxia and diabetes insipidus" @default.
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- W2085406905 doi "https://doi.org/10.1007/s10072-012-1107-9" @default.
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