Ocular manifestations of Lyme disease (LD) remain a rare feature of the disease, but it may present a wide range of clinical presentations with different combinations. LD related optic neuritis or cranial nerve (CN) six palsy have been reported in the literature. However, this is the first case report of simultaneous involvement of CN 2 and CN 6 in a patient with LD. The diagnosis of LD can be challenging and initial laboratory tests can be a false negative. It is paramount important to repeat the diagnostic test if clinical suspicious is ongoing. With this case, we aim to increase awareness of clinicians for possible ocular manifestations of LD and its complex diagnostic process.

Lyme disease related central and peripheral nervous system manifestations can occur in isolation or together. Radiculitis or inflammation of the nerve root can be seen 3-5% of the time in acute neuroborreliosis affecting the PNS with a typical presentation and meningitis affecting the CNS is usually seen 1% of the time. The appropriate diagnosis and management of neuroborelliosis can be challenging and require meticulous medical approaches. Herein we present a unique case of Lyme disease with neurologic manifestations including both radiculitis and meningitis due to its atypical and challenging clinical presentation and management with updated literature review.

In the US, Lyme disease (LD) has become the most common vector-borne disease. Less than 10% of patients develop cranial nerve palsy or meningitis. There are few reports on cases of Lyme disease with more than one cranial neuropathy. Herein, we will discuss a case of persistent neurological deficits as a result of chronic Lyme disease resistant to standard therapy. Our case is unique due to involvements of cranial seven and eight nerves at the same time. Our case illustrates an extreme example of treatment resistance. However, early diagnosis and prompt establishment of adequate antibiotic treatment are still important to prevent progression to further stages of disease.