Hepatic Brucelloma Diagnosis and Long-Term Treatment, France

We report a case of hepatic brucelloma in France. This diagnosis may be suspected in any patient who has a liver abscess after traveling to a brucellosis-endemic area. Brucella spp. may be detected by PCR in the liver tissue or suppuration. Abscess drainage and prolonged antimicrobial therapy help achieve healing.

We report a case of hepatic brucelloma in France.This diagnosis may be suspected in any patient who has a liver abscess after traveling to a brucellosis-endemic area.Brucella spp.may be detected by PCR in the liver tissue or suppuration.Abscess drainage and prolonged antimicrobial therapy help achieve healing.
In April 2015, a previously healthy 55-year-old woman was referred to Grenoble University Hospital after 7 days of fever, asthenia, and weight loss.She had lived in France for 20 years, but had been born in and had traveled every year to Algeria.Her clinical examination was unrevealing.Blood tests showed moderate inflammation and anicteric cholestasis (Table ).Hepatic ultrasound (HUS) and computed tomography (CT) confirmed a defect 60 mm in diameter in liver segments IV and VIII with several subcapsular liquid collections and central calcification (Appendix Figure, panel A, https://wwwnc.cdc.gov/EID/article/25/5/18-0613-App1.pdf).
Blood cultures remained sterile.Serologic test results were negative for HIV, amebiasis, and echinococcosis, but positive for Yersinia enterocolitica serotype O:9 and Brucella sp.(Table ).HUS-guided drainage of the abscess yielded thick purulent fluid.Fluid cultures were negative, but we detected Brucella melitensis DNA by PCR amplification and sequencing of the 16S rRNAencoding gene.Histological findings of liver tissue were compatible with a chronic abscess.We confirmed diagnosis on 2 occasions by PCR detection of Brucella DNA in the liver abscess, as previously reported (1,3,5-7).The serologic profile was suggestive of chronic brucellosis combining low IgM but strong IgG Brucella antibody titers (1,3,5,7).However, Brucella serologic diagnosis is poorly specific, due to antigenic cross-reactions (e.g.,

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Y. enterocolitica 0:9) and residual antibodies related to past exposure in patients from Brucella-endemic areas (3).Brucella cultures of blood or tissue samples and PCR on serum are usually not contributing factors in chronic focal brucellosis (1,3,(5)(6)(7).
Upon diagnosis of hepatic brucellosis, we administered doxycycline plus rifampin, replacing initial empirical treatment with ceftriaxone and metronidazole for suspected pyogenic abscess.After 2 months of treatment, clinical and biologic abnormalities had regressed.CT with contrast revealed substantial reduction of the abscess, but also thrombosis of the median hepatic vein, infiltration of the gallbladder, and new lesions in segment VIII (Appendix Figure , panel B).We continued the antimicrobial therapy and surgery was recused.After 6 months of treatment, antibody titers were decreasing (Table ).Three months later, positron emission tomography highlighted intense focal uptake in segments IV and VIII.Fluid from a second HUS-guided drainage was positive by Brucella-specific PCR test (Table ).We changed the treatment to trimethoprim/sulfamethoxazole plus doxycycline for another 6 months.At completion, magnetic resonance imaging showed abscess reduction with stable central calcification.
HB patients are difficult to manage because of lack of optimized treatment (1,8), and clinical course is the only reliable evidence in disease control (1,3,9).Our treatment was conservative, including 15 months of antimicrobial therapy and repeated HUS drainages.We continued treatment despite worsening radiological findings (including onset of cholecystitis and deep vein thrombosis) on the basis of early favorable clinical course and patient compliance (1-3,8,9).We monitored the improvement of radiological lesions using positron emission tomographycomputed tomography and magnetic resonance imaging, both of which are sensitive for diagnosis although not assessed for monitoring, for 15 months after treatment.Few studies have described the radiological evolution over time in complicated cases (1)(2)(3)5,8), but persistence of calcifications is usual in HB (2,3,9).
Administration of doxycycline plus rifampin, an aminoglycoside, or both is currently recommended for brucellosis (1-3); choice and duration of treatment must be individualized to the patient's symptomatology and treatment tolerability (1)(2)(3)8,9) and the involved tissues (2,8).Surgical resection and percutaneous drainage of the abscess are both reported as effective adjunctive therapy, with surgery indicated when evacuation of pus is ineffective or clinical symptoms persist despite drainage (1)(2)(3)5,6).A short course of an aminoglycoside could have been added at the time of radiological diagnosis of complications, as reported in other focal brucellosis with poor clinical evolution (2,8,9), in which a prolonged regimen (6-52 weeks) is also described (3,8,9).As of January 2019, after 2.5 years of treatment and follow-up care, our patient is considered cured.Relapses mainly occur within a few months of treatment completion but may occasionally occur later (2,5).
Any patient with characteristic liver abscess after traveling in brucellosis-endemic areas should undergo serologic testing for a presumptive diagnosis, and PCR testing of abscess fluid or hepatic tissue for confirmation of brucelloma.A conservative treatment combining long-term antimicrobial therapy and repeated HUS drainage may be effective.Long-term clinical comprehensive follow-up is required.

Table .
Diagnostic and blood tests on patient with hepatic brucellosis, Grenoble, France* *NA, not applicable.†Tissues were cultured for bacteria, fungi, and mycobacteria.