An unusual case of double jeopardy

Abstract

Brucellosis is an underrated and often neglected re-emerging zoonosis in India with palpably ill public health outcomes. In some cases, the diagnosis is not forthright especially when plagued by mixed infection. We bring to light a highly uncommon case of brucellosis coinfection with Mycobacterium tuberculosis (MTB) and review the literature.

Keywords: Brucella, mixed infection, mycobacterium, streptomycin

How to cite this article:
Gude D, Bansal DP, Kotari HR. An unusual case of double jeopardy. Ann Trop Med Public Health 2013;6:125-7

 

How to cite this URL:
Gude D, Bansal DP, Kotari HR. An unusual case of double jeopardy. Ann Trop Med Public Health [serial online] 2013 [cited 2017 Nov 14];6:125-7. Available from: https://www.atmph.org/text.asp?2013/6/1/125/115185

 

Introduction

 Brucellosis (undulant fever/ Malta fever / Gibraltar fever / Mediterranean fever) is a zoonotic infection transmitted to humans from infected animals (cattle, sheep, goats, pigs, etc.) or derived food products such as unpasteurized milk and cheese. It has a varied incidence of about 0.01-200 per 100,000 [1] and could be underreported for 25 times. Brucellosis may progress to grave severity involving various organ systems such as the nervous and/or cardiovascular systems. Coinfection with Mycobacterium tuberculosis (MTB) is very rare and may complicate the presentation.

Case Report

A 36-year-old female with a history of tubercular cervical lymphadenitis 2months ago [biopsy proven, [Figure 1] received antitubercular therapy (ATT) for the same, which is presented with recurring fever, neck swelling, nausea, vomiting, and decreased appetite from 1month. General examination revealed tender painful swelling in the neck, and cardiorespiratory and abdominal exam was unremarkable. Her laboratory reports showed Hb-11.1g/dl, TWBC-4200 (with 50% lymphocytes), platelets-2.1 L, TBilirubin-0.55mg/dl, DB-0.26 mg/dl; ALT-279U/l. Widal test was negative. In view of suspected hepatitis (?prior ATT induced), modified ATT was started with streptomycin, ethambutol, and levofloxacin. X-ray chest was unremarkable. Smear for malarial parasite was repeatedly negative and so were blood cultures. Ultrasonography of neck showed an ill-defined hypo-echoic lesion (cold abscess) and cervical lymphadenopathy. Piperacillin and tazobactamwere added.Excision biopsy of the cold abscess was done and a drain placed [Figure 2]. Two-dimensional echo did not reveal any vegetations.  Brucella More Details serology was sent as a part of work up for persistent fever which was highly positive at 1:10,240. The purulent fluid from the drain showed acid fast bacilli (AFB) bacilli. Doxycycline was added to the regimen, and over the period of next few weeks, fever responded along with the patient’s feeling of well-being. The titers of brucella serology fell to 1:320 over the week.

Figure 1: High power view showing caseating granuloma, Langerhans giant cell (arrow), and surrounding lymphocytic infiltrate

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Figure 2: Photograph showing swelling in the neck (post-cold asbcess drainage). Healed scar of the drain site is also seen

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Discussion

To our knowledge, ours is the second reported case of human brucellosis with coinfection of MTB. The first was by Karsen et al., [2] which was a case of meningitis with positive brucella-tube agglutination test in the cerebrospinal fluid (CSF) and serum, which was later found to have MTB grown in CSF culture. Settings like these could pose a diagnostic and therapeutic dilemma to clinicians demanding exhaustive resources and attention. We looked into the literature to rule out if our brucellaserology test could be a false positive and found that infections with  Escherichia More Details coli O157, Francisellatularensis,  Moraxella More Details phenylpyruvica,  Yersinia More Details enterocolitica, and certain  Salmonella More Details have a propensity to give false positive results. [3] But given the very high titers, the characteristic clinical presentation, and high unlikelihood of those infections in our scenario, we believe Brucella melitensis as the main culprit in our MTB-infected patient. Studies conducted in India reported a brucella sero-prevalence of 8.5% among dairy personnel in contact with infected animals and 4.2% among women with abortions and that B. melitensisis is the predominant strain. [4] The paucity of history pointing to risk from dairy animals or consumption of unpasteurized dairy products in our patient reckons clinicians to understand and be aware that the lack of such classical risk factors/presentations does not rule out brucella infection. Also in scenarios of coinfection, the clinical manifestations may vary greatly.

The pathophysiological mechanism involves the ingestion of brucella, followed by invasion of the enteral mucosa and its usual elimination by phagolysosome fusion. But up to 30% of the organisms may survive and replicate in the endoplasmic reticulum without lysis of the host cell (inhibits apoptosis) and eventually get released. [5] Interferon-gamma, TNF-alpha, and a variety of humoral and T cell-mediated mechanisms also play roles in its pathogenesis.

Brucella is an important and established cause of fever of unknown origin (FUO) and as was in our case, the diagnosis was clinched only after the work up for FUO began. A recent study showed fever (91%) and constitutive symptoms such as malaise, arthralgias (26%), hepatomegaly (17%), and splenomegaly (16%) to be the commonly observed signs and symptoms. Also complications such as peripheral arthritis (22%), spondylitis (19%), epididymoorchitis (5.7%), central nervous system disorders (3%), and respiratory disorders are reported. [6] Brucella typically causes relative lymphocytosis and transaminasemia although MTB for the former and ATT for the latter may cloud the specificity.

Cultures in brucella can pose a problem as the organism is slow growing, is not reliably recovered by routine blood culture techniques, and is positive in only 50-80% of the times. [7] Culturing it could be hazardous to laboratory workers, which further hinders the ubiquity of culture methods in developing countries. Serology is a better alternative with >1:160 (>1:10,240 in our case) or fourfold rise in titers strongly suggestive.

We have used the recommended doxycycline plus streptomycin (also as a part of ATT which would be continued) regimen with satisfactory results. Some have tried gentamycin or rifampin in place of streptomycin with doxycycline. Treatment in complicated states such as neurobrucellosis or endocarditis is prolonged (over a year). Health education, hygiene, and preventive measures (such as pasteurization), animal vaccination, and early recognition can help curb this zoonotic infection.

We reiterate the possibility of mixed infections when dealing with refractory clinical entities of convoluted presentations especially with the dearth of risk factors.

Acknowledgment

We thank our colleagues and staff of Internal Medicine and Critical Care, Princess Durru Shehvar and Medwin Hospitals.

References

 

1. Boschiroli ML, Foulongne V, O’Callaghan D. Brucellosis: A worldwide zoonosis. Curr Opin Microbiol 2001;4:58-64.
2. Karsen H, Karahocagil MK, Irmak H, Demiröz AP. A meningitis case of Brucella and tuberculosis co-infection. Mikrobiyol Bul 2008;42:689-94.
3. Corbel MJ. Brucellosis: An overview. Emerg Infect Dis 1997;3:213-21.
4. Mantur BG, Amarnath SK. Brucellosis in India -A review. J Biosci 2008;33:539-47.
5. Arenas GN, Staskevich AS, Aballay A, Mayorga LS. Intracellular trafficking of Brucellaabortus in J774 macrophages. Infect Immun 2000;68:4255-63.
6. Pappas G, Akritidis N, Bosilkovski M, Tsianos E. Brucellosis. N Engl J Med 2005;352:2325-36.
7. Manuselis G, MacGill T. Brucellosis (Brucellosis). In: Mahon CR, Lehman DC, Manuselis WB, editors. Textbook of Diagnostic Microbiology. vol. 3. USA: Saunders; 2007. p. 477-82.

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/1755-6783.115185

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[Figure 1], [Figure 2]

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