Acute febrile illness with choreoathetosis: a rare presentation of brucellosis

Abstract

© Author(s) (or their employer(s)) 2021. No commercial reuse. See rights and permissions. Published by BMJ. CASE DESCRIPTION A 15yearold Indian girl presented with fever and involuntary movements of the limbs and face of about a weeks’ duration. These stereotypic choreoathetosis movements involved both upper limbs, lower limbs and face (figure 1A,B; video 1). No other neurological deficits were noted and cardiorespiratory, abdominal examinations were normal. Evaluation revealed bicytopenia with neutrophilic leucocytosis. Blood cultures were sterile and cerebrospinal fluid analysis was unremarkable including herpes simplex virus—PCR, Venereal Disease Research Laboratory and NmethylDaspartate receptor antibodies. MRI of the brain was normal. Twodimensional echocardiography did not show vegetations or valvular abnormalities. Testing for antistreptolysin O titers, antinuclear antibodies and thyroid functions was within normal limits. ELISA for HIV antibodies was nonreactive. The absent Kayser–Fleischer ring, normal ceruloplasmin and 24hour urine copper ruled out a possibility of Wilson’s disease. The thoracoabdominal CT revealed mild hepatosplenomegaly. Workup for other infections including malaria, leptospirosis, dengue, scrub typhus and enteric fever was negative. The patient did not respond to empirical antimicrobials (ceftriaxone, doxycycline and acyclovir). Finally, as the patient belonged to a rural background and had a history of exposure to goats and cows, a possibility of Brucella infection was considered. The ELISA for Brucella IgM was strongly positive. The diagnosis was supported by the resolution of fever and choreoathetosis with the addition of rifampicin and streptomycin to doxycycline. Gradually, her cytopenias, leucocytosis, also settled. The patient received intramuscular streptomycin for the first 2 weeks and rifampicin and doxycycline for a total duration of 6 months. No residual neurologic defect was observed on a 3year followup. Acute febrile illness with choreoathetosis is rarely observed in clinical practice. The aetiologies include acute rheumatic fever, autoimmune encephalitis, viral infections (eg, herpes simplex virus and HIV), systemic lupus erythematosus and tropical infections (eg, malaria and leptospirosis). Brucellosis continues to cause disease with innumerable ways of presentation in the highrisk population; thus, a heightened awareness of this infection is essential. Neurological involvement can occur in up to 10% of cases. The usual presentation is meningoencephalitis, peripheral or cranial neuropathies, cerebellar dysfunction, intracerebral abscess, and ruptured mycotic aneurysms. 2 Involuntary body movements including chorea and athetosis are rarely reported.