BMJ Case Reports
14 februari 2013
Case study - 'Seronegative lyme neuroborreliosis in a patient using rituximab' by Willemijn Alexandra van Dop, Marie-José Kersten, Bob de Wever, and Joppe Willem Hovius; Bron https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3604215/
A 66-year-old woman presented with severe shooting pains throughout her back and legs, followed by progressive deafness, weight loss and headache. She had a history of marginal zone B-cell lymphoma stage IV-B, for which she was successfully treated with immunochemotherapy and rituximab maintenance therapy. A relapse was suspected, but chemotherapy was not administered, since, despite elaborate investigations, malignancy could not be proven. Because of a history of tick bites she was tested for antibodies against Borrelia burgdorferi in serum and cerebrospinal fluid (CSF), which were negative. However, a B burgdorferi PCR on CSF came back positive. The patient was treated for seronegative Lyme neuroborreliosis with ceftriaxone intravenously and dramatically improved. This case presentation demonstrates that, in immunocompromised patients, it is important not to solely rely on antibody testing and to use additional diagnostic tests to avoid missing or delaying the diagnosis.
Outcome and follow-up
During these 3 weeks she improved dramatically, which further strengthened the diagnosis. She gained weight, the complaints of headache and vertigo disappeared and her hearing loss slowly improved. Paired serology on blood and CSF 7 weeks later still revealed no antibodies against B burgdorferi.
One-and-a-half years later, a routine MRI of the myelum showed no evidence for relapse of the lymphoma and the hearing ability had completely returned. During neurological follow-up the patient reported a slight unsteadiness of gait, which was attributed to residual postinfectious complications. The rituximab maintenance therapy had been discontinued during her hospital admission, but despite the slow repopulation of B-lymphocyte subsets, 1.5 years after her initial presentation, there were still no antibodies against B burgdorferi (figure 3).
De Lyme (neuroborreliose) patiënte is succesvol behandeld met ceftriaxone.Discussion
In conclusion, this case report is an important reminder of the fact that in patients treated with drugs such as rituximab, which interfere with the immune response, serological diagnostics are not always reliable. In immunocompromised patients with a high suspicion of infectious diseases, such as infection with B burgdorferi, it is therefore important not to rely solely on antibody testing but to use several additional diagnostic tests (e.g, PCR and culture) to avoid missing or delaying the diagnosis. Lyme neuroborreliosis, although sometimes easy to diagnose, can be difficult to diagnose, and when the clinical suspicion is high, like in our case, additional diagnostic tests or sometimes even empiric treatment should be considered.