Brucellosis Complicated by Kikuchi-Fujimoto Disease and Doxycycline-Induced Intracranial Hypertension

Excerpt

To the Editor: Brucellosis is a zoonosis, which rarely has autoimmune manifestations [1]. An adolescent boy presented with persistent fever despite oral antibiotics for 2 wk, cervical lymphadenopathy and hepato-splenomegaly. After 7 d of ceftriaxone and doxycycline, he progressed to develop axillary lymphadenopathy, epididymitis and scrotal ulcer. He had anemia (9.7 g%), lymphopenia (600/μL), monocytosis (15%), elevated ESR (87 mm), CRP (31 mg/L), transaminases (>5-times normal), globulin (3.9 g/dl), ferritin (1012 ng/ml), but normal procalcitonin, triglyceride and fibrinogen. Imaging revealed enlarged periportal and peri-pancreatic nodes. Bone marrow had increased reticulo-endothelial activity. Evaluation for malaria, dengue, typhoid, tuberculosis, leptospirosis, leishmania, HIV, hepatitis B & C, toxoplasmosis, cytomegalovirus and Epstein-Barr-virus were negative. All cultures, and 16 s-rDNA-PCR-sequencing, were negative. ELISA for Brucella-IgM was reactive (signal-strength: 1.87, normal: <0.8; IgG non-reactive). One day after switching to rifampicin and gentamicin, while continuing doxycycline, he developed increased temperature spikes (104 °F), arthralgia and maculopapular rash. Autoimmune markers (ANA, pANCA/cANCA, ASO, C3/C4, RF) were negative. Naproxen was started suspecting delayed Jarisch-Herxheimer (JH) reaction [2]. The next day he developed severe headache, right VI-nerve palsy, bilateral papilledema and enlarged blind spots. MRI (brain, orbit) and CSF analysis were normal. Doxycycline was stopped. Acetazolamide and prednisolone (1 mg/kg/d) were started. Lymph node biopsy suggested Kikuchi-Fujimoto disease (KFD). Symptoms resolved after starting steroids, allowing discharge on rifampicin and trimethoprim-sulfamethoxazole. Serology for Brucella, repeated after 7 and 14 d, demonstrated rising titers (IgM: 19, 41.58 U/ml; IgG: 5, 20.22 U/ml). All anomalies normalized, prednisolone was tapered after 2 wk, and the antibiotics were continued for 8 wk. …