Adult-onset opsoclonus-myoclonus-ataxia syndrome as a manifestation of brazilian lyme disease-like syndrome: a case report and review of literature

Abstract

Described in 1962, the opsoclonus-myoclonus-ataxia syndrome (OMAS) is a rare, neurologically debilitating disorder with distinct characteristics that may begin in childhood or adult life. Although many cases remain without etiological diagnosis, others are related to neoplasms and infectious diseases. We report a 41-year-old previously healthy male with an 8-day history of headache, vertigo, nausea, vomiting, and nystagmus. After a normal brain computed tomography and lymphocytic pleocytosis in cerebral spinal fluid (CSF), intravenous acyclovir therapy was initiated in the emergency room. On the third day of hospitalization, the diagnosis of OMAS was made based on the presence of chaotic and irregular eye movements, dysarthric speech, gait instability, generalized tremor, and myoclonic jerks. In the face of his neurological worsening, ampicillin followed by nonspecific immunotherapy (methylprednisolone and intravenous immunoglobulin) was prescribed, with mild clinical improvement. After a thorough laboratory workup, the definite diagnosis of neuroborreliosis was established and ceftriaxone (4 g/daily/3 wks) and doxycycline (200 mg/day/2 mo) was administered. Toward the end of the ceftriaxone regimen, the neurologic signs substantially improved. We believe this to be the first case description of OMAS as clinical presentation of Brazilian Lyme disease-like syndrome (Baggio-Yoshinari syndrome).

Paraneoplastic opsoclonus-myoclonus syndrome as a presentation of high grade serous ovarian cancer

“…The pathophysiology of OMS is not definitively understood. It has been suggested that antibodies against cell surface or intracellular structures of the nervous system, such as anti-hu, anti-ri, and anti-yo may play a role in OMS (Lino et al, 2014). Specifically, a finding of anti-ri antibodies in a patient presenting with OMS is highly specific for an underlying neoplasm (Jongen et al, 1998, Luque et al, 1991).…”

Borreliae Part 1: Borrelia Lyme Group and Echidna-Reptile Group

“…The first manifestation is often EM as in classic Lyme disease; however, there are epidemiological, clinical and laboratory-related differences to define the Brazilian Lyme-like disease, which in 1993 was named Baggio-Yoshinari syndrome (BYS) [250]. In a study of 19 patients, Yoshinari reported that about 30% had skin lesions, 30% arthritis and 42% had neurological disorders; ocular symptoms were observed in 37.5% of patients, especially in the initial phase of the disease [251,252]. BYS is caused by a Borrelia which resembles Borrelia burgdorferi in clinical and laboratory characteristics.…”

Scrub typhus-associated opsoclonus: Clinical course and longitudinal outcomes in an Indian cohort

“…[ 8 ] Adult-onset opsomyoclonus occurs in 1–2 people/10 million/year, with a 2012 review spanning 65 years at Mayo Clinic, Minnesota, identifying 137 patients. [ 9 10 ]…”