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September 3, 2020

Atypical Presentation of Langerhans Cell Histiocytosis of the Skull

Author Affiliations
  • 1Section of Neurosurgery, Carilion Clinic, Roanoke
  • 2Virginia Tech Carilion School of Medicine, Roanoke
  • 3Virginia Tech School of Neuroscience, Blacksburg
  • 4Department of Pathology, Carilion Clinic, Roanoke
JAMA Otolaryngol Head Neck Surg. Published online September 3, 2020. doi:10.1001/jamaoto.2020.2399

Langerhans cell histiocytosis is a rare myeloid neoplasm predominantly affecting the pediatric population with a broad clinical continuum, ranging from an isolated skin or bone lesion to multiorgan disease dissemination. Although the cranial vault is the most common site affected, it is exceedingly rare for this neoplasm to present as frontal sinusitis, with only 2 reports to our knowledge in the pediatric literature.1,2 We present, to our knowledge, the first case of Langerhans cell histiocytosis of the skull presenting as frontal sinusitis in an adult.