Central Nervous System Langerhans Cell Histiocytosis

  • Milen Minkov
  • Jennifer Picarsic
  • Karoly Lakatos


Langerhans cell histiocytosis (LCH) can affect virtually any organ of the human body, including the central nervous system (CNS). Based on correlation of clinical, imaging, and pathology findings, two patterns of CNS-LCH have been identified: granulomatous (“tumorous”) and non-granulomatous “neurodegenerative” (ND)-CNS-LCH. Granulomatous CNS-LCH can manifest with polyuria and polydipsia, seizures, increased intracranial pressure, or other signs, depending on location. The typical location on MRI is extra-axial (hypothalamic-pituitary region, meninges, pineal gland, choroid plexus), and pathology in a proliferative phase reveals CD1a/langerin+ histiocytes. ND-CNS-LCH manifests insidiously with cerebellar and bulbar symptoms. MRI typically reveals parenchymal signal alterations in the cerebellum, pons, basal ganglia, and supratentorial white matter consistent with neuronal loss and demyelination. Biopsies of such lesions are rare. In the few published cases biopsied, variable pathologies are described with perivascular inflammatory changes, variable neuronal loss, demyelination, and gliosis, but are nondiagnostic for CD1a+/CD207+ LCH cells. Granulomatous LCH can be responsive to chemotherapy agents such as vinblastine, cytarabine, or cladribine, while the optimal treatment of ND-CNS-LCH is yet to be defined.


Langerhans cell histiocytosis Central nervous system Granulomatous Tumorous Non-granulomatous Neurodegeneration Sequelae 



Central nervous system


Cerebrospinal fluid


Diabetes insipidus


Erdheim-Chester disease


Expanded Disability Status Scale


Fluorodeoxyglucose positron emission tomography


Hypothalamic-pituitary region


International Cooperative Ataxia Rating Scale


Juvenile xanthogranuloma


Langerhans cell histiocytosis


Mitogen-activated protein kinase


Magnetic resonance imaging


Multisystem LCH




Permanent consequences


Rosai-Dorfman disease


Single system LCH


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Copyright information

© Springer International Publishing AG 2018

Authors and Affiliations

  • Milen Minkov
    • 1
    • 2
  • Jennifer Picarsic
    • 3
  • Karoly Lakatos
    • 2
  1. 1.International LCH Study Reference Center, CCRI, St. Anna Kinderkrebsforschung, Department of Pediatrics and Adolescent Medicine,Medical University of ViennaViennaAustria
  2. 2.St. Anna Children’s Hospital, Department of Pediatrics and Adolescent Medicine, Medical University of ViennaViennaAustria
  3. 3.Department of Pathology, University of Pittsburgh School of Medicine, Children’s Hospital of Pittsburgh of UPMCPittsburghUSA

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