Background Neuroborreliosis is involvement of the nervous system in Lyme disease and can often be overlooked as a diagnosis in patients presenting with cranial neuropathies and meningitis in an endemic area. There can be variability in presentation with respect to the involved cranial nerves, making the diagnosis challenging. To the best of our knowledge, there have been no reported cases of Lyme disease cranial neuropathy occurring with simultaneous periorbital skin and soft tissue inflammation in the literature. Case We present the case of a healthy 25-year-old man who presented to the emergency department with headache, diplopia, and left eye pain after experiencing fever, malaise, and a recent history of unilateral facial palsy that improved a few weeks prior. He lived in a Lyme disease endemic area and often encountered ticks. He was found to have an abduction deficit in his left eye, preseptal and postseptal orbital inflammation with bilateral optic perineuritis, and a lymphocytic pleocytosis in the cerebrospinal fluid accompanied by an elevated antibody index, consistent with a diagnosis of Lyme neuroborreliosis. He was treated with doxycycline and empiric bacterial coverage, with symptom resolution at follow-up. Conclusion This case represents an unusual combination of cranial neuropathies and is the first report of orbital inflammation driven by Lyme neuroborreliosis, thought to be caused by contiguous spread of inflammation from affected portions of the nervous system. Although neuroborreliosis is less commonly encountered in the United States than in Europe, providers should maintain a high index of suspicion for this entity when patients present with multiple cranial neuropathies in an endemic area and not exclude Lyme disease on the basis of skin and soft tissue inflammation.
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