To report a case of Lyme disease presenting with subacute multiple cranial neuropathies and orbital myositis. 

A 69-year-old male with a history of hyperthyroidism status post thyroidectomy, presented with a new onset headache, double vision, numbness of the face, and dysphagia. On examination, he had left lower motor neuron facial weakness, left eye proptosis, and findings suggestive of III and VI nerve palsies.  Brain MRI with contrast revealed abnormal enhancement along the left V2 and V3 nerves and enhancement of the bilateral III cranial nerve. Abnormal enhancement was also seen at the left orbital apex in addition to enhancement along the left retro-orbital optic nerve. MRI of the orbit showed asymmetric enlargement and subtle enhancement of the left extra orbital muscles with mild orbital fat infiltration, compatible with orbital myositis. Lyme IgG/IgM antibody screen was positive which was confirmed by Lyme immunoblot assay which was also positive for Lyme IgG (5 bands) and IgM (3 bands) antibodies. B2 Glycoprotein IgM was elevated. Cerebrospinal fluid (CSF) analysis revealed lymphocytic pleocytosis and elevated protein. CSF cytology and flow cytometry were negative for malignant cells. TSH, thyroxin, and anti-thyroid peroxidase antibodies were unremarkable. Sarcoidosis, IgG4-related disease, autoimmune, and infectious workups were unrevealing. The patient received IV ceftriaxone with significant improvement and with near resolution of the symptoms and clinical findings after 1 month.