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Year : 2022  |  Volume : 24  |  Issue : 3  |  Page : 122-124

Demystifying pyrexia of unknown origin: A case of splenic brucellosis

Department of General Medicine, Army Hospital (Research and Referral) New Delhi, India

Date of Submission24-May-2021
Date of Decision06-Oct-2021
Date of Acceptance14-Dec-2021
Date of Web Publication21-Jan-2022

Correspondence Address:
Dr. Bhairavabhatla V S. P. Viswanadh
Army Hospital (Research and Referral) New Delhi
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jmms.jmms_78_21

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Brucellosis is a zoonotic infectious disease, which mainly involves lymphoreticular system. Our case report describes an isolated splenic lesion diagnosed in a 48-year-old individual who is a farmer by occupation, had a history of animal contact, and has the habit of consuming raw milk. He was admitted as a case of pyrexia of unknown origin (PUO) and detected to have splenic Space occupying lesion (SOL) on imaging. His serum Brucella IgM was positive and ultrasonography-guided fine-needle aspiration cytology of splenic SOL showed features of necrotizing granulomatosis inflammation or an organizing abscess. The patient was successfully treated for brucellosis with oral doxycycline and rifampin for 6 weeks. Hereby, we report a case of PUO with splenic SOLs, Diagnosed on the basis of clinical, radiological, histopathological, and serological basis as a case of spleenic brucellosis. This case required a very high index of clinical suspicion and further highlighted the fact that even in present times of advanced diagnostic modalities, clinical inputs cannot be overlooked. Thereby, we were able to demystify a case of PUO successfully.

Keywords: Brucellosis, doxycycline, necrotizing granulomas, pyrexia of unknown origin, splenic SOL, zoonosis

How to cite this article:
Kumar M, Singhal A, Singh V, Bushra A, Viswanadh BV, Kiran K K. Demystifying pyrexia of unknown origin: A case of splenic brucellosis. J Mar Med Soc 2022;24, Suppl S1:122-4

How to cite this URL:
Kumar M, Singhal A, Singh V, Bushra A, Viswanadh BV, Kiran K K. Demystifying pyrexia of unknown origin: A case of splenic brucellosis. J Mar Med Soc [serial online] 2022 [cited 2022 Nov 27];24, Suppl S1:122-4. Available from: https://www.marinemedicalsociety.in/text.asp?2022/24/3/122/336197

  Introduction Top

Brucellosis (also known as “undulant fever,” “Mediterranean fever,” or “Malta fever”) is a zoonotic infection caused by Gram-negative bacilli of the genus Brucella and transmitted to humans from infected animals (cattle, sheep, goats, camels, pigs, or other animals) by ingestion of food products (such as unpasteurized dairy products) or by contact with tissue or fluids. It is the most common zoonosis worldwide. Brucellosis presents with fever, malaise, night sweats, and arthralgias.[1] Brucella bacilli after inoculation is taken up by local tissue lymphocytes, enter the circulation through regional lymph nodes, and seed throughout the body, with tropism for the reticuloendothelial system. It can affect any organ in the body. Involvement of the liver and spleen may be attributed to intracellular survival and replication of bacteria in the mononuclear-phagocytic system.

  Case Report Top

In November 2020, a 48-year-old man was brought to the outpatient department with high-grade fever with chills, generalized malaise, and significant weight loss (>10 kg in 45 days). The patient is a farmer by occupation from Jammu and routinely consumes unpasteurized or raw milk. The patient's medical records and history from relatives revealed fever with night sweats and generalized malaise and significant weight loss from 18 months, for which he was evaluated as fever of unknown origin.

On clinical examination, at admission, he was afebrile, with normal vital signs. He had no pallor, icterus, cyanosis, clubbing, pedal edema, or lymphadenopathy. Systemic examination of the abdomen showed splenomegaly 10 cm below the costal margin. No abdominal lumps. Other systems examination was normal.

Investigations revealed hemoglobin of 11.4 g/dl with a total leukocyte count of 10,000/cumm and platelet count of 1.25 lakhs. His blood sugar, renal function tests, lipid profile, serum electrolytes, and liver function tests are within normal limits. Screening for viral markers was negative. Peripheral blood smears reveal normocytic normochromic red blood cells with mild poikilocytosis, no hairy cells, and no malaria parasites. Immunochromatographic test for malaria parasite, typhidot test for salmonella, dengue serology, and rapid test for Leishmania were negative. Sputum for acid-fast bacillus, staining, MTB GeneXpert, culture, and sensitivity were negative. Mantoux test was negative. Blood, urine, stool for culture, and sensitivity revealed no growth. Urine routine and microscopy were normal. Tumor markers were negative.

Bone marrow aspiration and biopsy suggestive of hypocellular marrow aspirate showed trilineage hematopoiesis and no significant hemophagocytosis. Bone marrow flow for chronic lymphoproliferative disorder and GeneXpert was negative. Ultrasonography (USG) was suggestive of massive splenomegaly 20 cm. Contrast-enhanced computed tomography abdomen revealed massive splenomegaly 20.1 cm in long span with multiple, well-defined round-to-oval variable-sized hypodense lesions (Average attenuation ~50–55 HU) scattered in the spleen, largest 12-mm largest diameter. Brucella serology was IgM positive. Positron emission tomography (PET) WB scan showed, few fluorodeoxyglucose avid and nonavid iso hypodense lesions noted in the spleen, largest ~11 mm SUV max 5.0 along superior border shown in [Figure 1]. USG-guided fine-needle aspiration cytology (FNAC) of splenic SOL showed features of necrotizing granulomatosis inflammation/organizing abscess. Diagnosis of systemic brucellosis with splenic SOLs was made.
Figure 1: Photomicrographs of Fine needle aspiration cytology findings, a –shows cellular smear comprised of mixed inflammatory infiltrate composed of neutrophils, lymphocytes, and histiocytes arranged in a necrotic background (black arrow) (Leishman Giemsa stain, 10x )b - Occasional ill-formed epithelioid cell granulomas are seen(black arrowhead) (Leishman Giemsa stain, 40x), c - Occasional multinucleated giant cells are also seen (Leishman Giemsa stain, 40x)

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To summarize, we had successfully diagnosed a case of systemic brucellosis who had presented with pyrexia of unknown origin (PUO) with splenic SOLs on the basis of clinical, radiological, histopathological, and serological tests.

The patient was successfully treated with oral doxycycline (200 mg/day) and rifampin (600 mg/day) for 6 weeks. The patient improved symptomatically with the absence of fever. The patient was discharged. The patient is in follow-up.

Take-home message, to manage this case, it not only required clinical skills but also pathological and radiological investigations. This case further enforces the importance of splenic FNAC practices at times to clinch the diagnosis.

  Discussion Top

Brucellosis is a multisystem disease with protean manifestations and myriad presentations. The spectrum can range from multisystem involvement to asymptomatic infection, hence diagnosis can be difficult. Commonly fever, night sweats, fatigue, myalgia, and arthralgias are encountered, and complications such as spondylitis, sacroiliitis, osteomyelitis, endocarditis, and epididymo-orchitis are known to occur. Bone marrow involvement can lead to pancytopenia. Microangiopathic hemolytic anemia, thrombotic thrombocytopenic purpura, and hemolytic–uremic syndrome are also known to occur in brucellosis.[1]

On physical examination, hepatosplenomegaly can be apparent; liver function tests may reveal elevated transaminases, nonspecific hepatic inflammation, and granulomatous hepatitis.[2],[3] Liver biopsy may reveal a granuloma formation in the liver parenchyma and in the periportal areas.[3] Visceral organ abscess formation in the spleen, liver, or other organs can be secondary to Brucella bacteremia, and sometimes, soft-tissue abscesses are known to occur.[4] Isolated splenic abscess is extremely rare[5] and in few cases only it presents as splenic subcapsular hematoma. The incidence of splenic abscess in patients with acute brucellosis is low and does not exceed 2%–3% of the cases. In a large study by Pourbagher et al. from Turkey (2006), 251 cases of brucellosis were reviewed and pleural effusion was found in 2.8%, splenic abscess in 1.6%, splenic cyst in 0.8%, and acute acalculous cholecystitis in 0.4% of the cases with an ultrasound.[6] In another large study by Colmenero et al. which had a total of 805 patients, only 7 hepatosplenic abscesses were detected (0.8%)[7] and the ratio of hepatic to splenic abscesses was identified as 57.1% and 42.9%, respectively. Our case had massive palpable splenomegaly and the CT and PET-CT imaging findings revealed multiple hypodense splenic lesions and FNAC of these lesions further revealed ill-formed granulomas with organizing abscess.

The blood culture did not grow Brucella, however, Brucella serology was positive. Workup for other pathogens such as tuberculosis, Leishmania, HIV was negative. Clinical history of consuming unpasteurized milk and family history of brucellosis a year back. Fine needle aspiration cytology (FNAC) of splenic SOL showed features of necrotizing granulomatosis inflammation/organizing abscess. Hence diagnosed as case of Splenic brucellosis. The patient was treated with 6 weeks of rifampin and doxycycline and he has clinically improved. This is a rare case report of brucellosis as the patient with a positive family history of brucellosis along with occupational history of being a farmer who consumed unpasteurized dairy products had PUO with massive splenomegaly and isolated splenic abscess with granulomas with Brucella IgM serology positive and responded to appropriate therapy.

  Conclusion Top

We hereby presented a rare case of brucellosis who had presented as splenic granulomas. To manage this case, it not only required sound clinical judgment but also required sophisticated imaging like PET-CT and risky procedures like splenic FNAC. This case further enforces the importance of splenic FNAC practices at times to clinch the diagnosis.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

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Conflicts of interest

There are no conflicts of interest.

  References Top

Söker M, Devecioglu C, Yaramis A, Ipek S, Özbek MN, Tüzün H. Microangiopathic hemolytic anemia, thrombocytopenia and acute renal failure associated with acute brucellosis. Int Pediatr 2001;16:105-8.  Back to cited text no. 1
Williams RK, Crossley K. Acute and chronic hepatic involvement of brucellosis. Gastroenterology 1982;83:455-8.  Back to cited text no. 2
Akritidis N, Tzivras M, Delladetsima I, Stefanaki S, Moutsopoulos HM, Pappas G. The liver in brucellosis. Clin Gastroenterol Hepatol 2007;5:1109-12.  Back to cited text no. 3
Calişkan AC, Barut S, Köseoğlu D, Aytan H, Demirtürk F. Obturatory abscess and pelvic pain caused by Brucella melitensis. Mikrobiyol Bul 2009;43:325-9.  Back to cited text no. 4
Naseem A. Splenic abscess secondary to Brucella melitensis. J Coll Physicians Surg Pak 2002;12:488-90.  Back to cited text no. 5
Pourbagher MA, Pourbagher A, Savas L, Turunc T, Demiroglu YZ, Erol I, et al. Clinical pattern and abdominal sonographic findings in 251 cases of brucellosis in southern Turkey. AJR Am J Roentgenol 2006;187:W191-4.  Back to cited text no. 6
Colmenero Jde D, Queipo-Ortuño MI, Maria Reguera J, Angel Suarez-Muñoz M, Martín-Carballino S, Morata P. Chronic hepatosplenic abscesses in Brucellosis. Clinico-therapeutic features and molecular diagnostic approach. Diagn Microbiol Infect Dis 2002;42:159-67.  Back to cited text no. 7


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