Altered cerebral perfusion in children with Langerhans cell histiocytosis after chemotherapy

Pediatr Blood Cancer. 2020 Mar;67(3):e28104. doi: 10.1002/pbc.28104. Epub 2019 Dec 4.

Abstract

Background and purpose: Children with Langerhans cell histiocytosis (LCH) may develop a wide array of neurological symptoms, but associated cerebral physiologic changes are poorly understood. We examined cerebral hemodynamic properties of pediatric LCH using arterial spin-labeling (ASL) perfusion magnetic resonance imaging (MRI).

Materials and methods: A retrospective study was performed in 23 children with biopsy-proven LCH. Analysis was performed on routine brain MRI obtained before or after therapy. Region of interest (ROI) methodology was used to determine ASL cerebral blood flow (CBF) (mL/100 g/min) in the following bilateral regions: angular gyrus, anterior prefrontal cortex, orbitofrontal cortex, dorsal anterior cingulate cortex, and hippocampus. Quantile (median) regression was performed for each ROI location. CBF patterns were compared between pre- and posttreatment LCH patients as well as with age-matched healthy controls.

Results: Significantly reduced CBF was seen in posttreatment children with LCH compared to age-matched controls in angular gyrus (P = .046), anterior prefrontal cortex (P = .039), and dorsal anterior cingulate cortex (P = .023). Further analysis revealed dominant perfusion abnormalities in the right hemisphere. No significant perfusion differences were observed in the hippocampus or orbitofrontal cortex.

Conclusion: Perfusion in specific cerebral regions may be consistently reduced in children with LCH, and may represent effects of underlying disease physiology and/or sequelae of chemotherapy. Studies that combine a formal cognitive assessment and hemodynamic data may further provide insight into perfusion deficits associated with the disease and the potential neurotoxic effects in children treated by chemotherapy.

Keywords: Langerhans cell histiocytosis; chemotherapy; neurotoxity of therapy; pediatric oncology.

MeSH terms

  • Antineoplastic Combined Chemotherapy Protocols / adverse effects*
  • Blood Flow Velocity / drug effects*
  • Case-Control Studies
  • Cerebral Arteries / drug effects
  • Cerebral Arteries / pathology*
  • Cerebrovascular Circulation / drug effects*
  • Child
  • Child, Preschool
  • Female
  • Follow-Up Studies
  • Histiocytosis, Langerhans-Cell / drug therapy*
  • Histiocytosis, Langerhans-Cell / pathology
  • Humans
  • Infant
  • Magnetic Resonance Angiography
  • Male
  • Neuroimaging / methods*
  • Perfusion
  • Prognosis
  • Retrospective Studies