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CASE REPORT
Year : 2018  |  Volume : 21  |  Issue : 4  |  Page : 222-226

Multi-organ dysfunction syndrome as the first presentation of acute brucellosis, a case report and literature review


Department of Internal Medicine, College of Medicine, Taif University, Taif, KSA

Date of Web Publication31-Dec-2018

Correspondence Address:
Dr. Ibrahim Masoodi
Department of Internal Medicine, College of Medicine, Taif University, Taif
KSA
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DOI: 10.4103/smj.smj_11_18

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  Abstract 


Multiple organ dysfunction syndrome (MODS) is a continuous process with incremental degrees of physiologic derangements in individual organs. The alteration in organ function can vary widely from a mild degree of organ dysfunction to completely irreversible organ failure. MODS, as defined, is a clinical syndrome characterized by the development of progressive and potentially reversible physiologic dysfunction in two or more organs or organ systems that are induced by a variety of acute insults, including sepsis. The cornerstone in the management remains correction of the triggering agent. The clinical scenario of a young male who presented with progressive shortness of breath, renal failure, and epistaxis (MODS) at presentation is discussed in this report. The diagnosis of this patient was clinically quite challenging. On evaluation, he proved to have multi-organ failure due to Brucella melitensis. The patient improved with conventional B. melitensis treatment. The Brucella infection is a male predominant disease and frequently presents with typical symptoms of fever, fatigue, etc., and the diagnosis often is straightforward, but the reports of atypical presentation have been described in the literature. The heaviest disease burden of B. melitensis lies in countries of the Mediterranean basin and Arabian Peninsula. However, the disease is not uncommon in India, Mexico, and Central America. Hence, clinicians need to be aware of this clinical entity all over the globe. The report of the case and brief review of literature are presented in this report.

Keywords: Acute brucellosis, hepatitis, multi-organ dysfunction syndrome, renal failure, thrombocytopenia


How to cite this article:
Masoodi I. Multi-organ dysfunction syndrome as the first presentation of acute brucellosis, a case report and literature review. Sahel Med J 2018;21:222-6

How to cite this URL:
Masoodi I. Multi-organ dysfunction syndrome as the first presentation of acute brucellosis, a case report and literature review. Sahel Med J [serial online] 2018 [cited 2019 Jan 16];21:222-6. Available from: http://www.smjonline.org/text.asp?2018/21/4/222/249073




  Introduction Top


 Brucellosis More Details ( Mediterranean fever More Details, undulant fever) is the most common zoonotic infection caused by the bacterial genus  Brucella More Details. These small aerobic intracellular coccobacilli, localize in the reproductive organs of the host animals. The organisms are shed in large numbers in the animal's urine, milk, placental fluid, and other fluids. Even though humans are accidental hosts, brucellosis continues to be a major public health concern all over the world. Brucellosis is estimated to cause >500,000 infections/year worldwide. Its geographic distribution is limited by effective public and animal health programs and the prevalence of the disease varies widely from country to country.[1] Of the eight species discovered so far, Brucella melitensis (from sheep; highest pathogenicity), Brucella suis (from pigs; high pathogenicity) Brucella abortus (from cattle; moderate pathogenicity), and Brucella canis (from dogs; moderate pathogenicity) have moderate-to-significant human pathogenicity

Brucellosis in humans is a multisystem disease that may present with a broad spectrum of clinical manifestations. Overall, the frequency of brucellosis is higher in more agrarian societies and in places where handling of animal products and dairy products is less stringent. While hepatic involvement in brucellosis is not rare, it may rarely involve the kidney or display with cardiac manifestations. Central nervous system involvement in brucellosis sometimes can cause demyelinating syndromes. In areas endemic for brucellosis, this infection can be associated with hematuria, proteinuria, and renal failure. Brucellosis is endemic in Saudi Arabia and presents with classical symptoms of fever, fatigue, and malaise. In a study from Northern Saudi Arabia, the authors observed that for unknown reasons majority of brucellosis cases (60%) were in the age range of 13–40 years, whereas 21% occurred in those younger than 13 years, 16% in those aged 40–60 years, and 2.5% in those older than 60 years.[2] Report of multi-organ dysfunction syndrome due to acute brucellosis is presented in this report.


  Case Report Top


A 22 year old male student was brought by emergency medical services from his home to the Accident Emergency Department of King Abdulaziz Specialist Hospital in Taif, Saudi Arabia, with the history of acute onset of shortness of breath of 1-day duration. The patient was conscious but in distress and had oxygen saturation of 82% on room air. He was given high-flow oxygen by the face mask and admitted to the intensive care of the hospital. The history was taken from his father as the patient had shortness of breath. The father narrated that the patient had been unwell for almost 6 weeks with low-grade fever and malaise. He had been on a local doctor's treatment without any relief. He denied any high-risk behavior, trauma, or long travel. Fever had been low grade with no diurnal variation associated with occasional chills and the patient had lost 3 kgms of weight during this period. The examination of the patient revealed pallor, scleral icterus, mild pedal edema, and few petechial spots could be seen on his extremities. The pulse was 80 beats/minute regular, blood pressure was 110/70 mmHg, and JVP was normal. The temperature documented in the hospital was 38°C. The abdominal examination showed soft hepatomegaly 3 cm below costal margin. There was mild splenomegaly, but no free fluid could be appreciated. Chest examination showed few crackles and mild variable diffuse wheeze. The cardiovascular examination was normal. He was moving all four limbs and the detailed neurological examination next morning was normal. Septic screen was sent soon after the admission and the laboratory reports in the evening as shown in [Table 1] were suggestive of pancytopenia. There was high blood urea nitrogen and creatinine levels were 2.4 mg/dl. An impression of prerenal azotemia was made and the patient was given a fluid challenge and keeping in view fever pending culture reports, broad-spectrum antibiotics with renal modifications were started. The bedside X-ray chest showed prominent bronchovascular markings and no consolidation or pleural fluid was noted. Keeping in view shortness of breath, the echocardiography was done which was unremarkable. After fluid therapy and oxygen supplementation, the patient's breathlessness settled and he passed adequate urine. On day 2, the patient had a unprovoked epistaxis from the right nostril and an immediate ENT consultation was sought who ruled out any local pathology. After anterior nasal packing, the patient was transfused four units of platelet-rich plasma as the platelet count had fallen next morning to 18,000 × 103. The bleeding was stopped and nasal pack was removed; however, the patient continued to be febrile next day and bone marrow aspiration (BMA) was done. Keeping in view, high liver enzymes and predominantly direct hyperbilirubinemia viral markers (hepatitis A virus, hepatitis B virus surface antigen, anti-hepatitis C virus, and IgM hepatitis E virus) and HIV serology were sent which turned out be negative days later. An ultrasound of the abdomen showed hepatosplenomegaly and normal intrahepatic biliary radicles. The portal vein and hepatic veins were normal. Day 4th reports of Brucella serology was available which was strongly positive (titers were >1:620 [titers >l: 160 positive]). The blood culture grew B. melitensis and renal dose-modified standard anti-Brucella (injection streptomycin 0.75 g intramuscular once daily and tablet doxycycline 100 mg twice daily) regimen was started to begin with and later full dose once creatinine levels returned to normal for total of 6 weeks. BMA did not reveal any abnormal hematological lineage. On repeat questioning, the patient admitted raw camel milk ingestion few weeks before the current presentation. On day 7, the patient's fever settled and he was off oxygen and was shifted to the ward. The patient had progressive clinical improvement and his laboratory parameters normalized as shown in [Table 1], but the liver enzymes remained partially high. He was discharged in stable condition with an advice of liver biopsy on follow-up if liver enzymes continued to remain high. However, liver enzymes normalized on week 3rd of follow-up and the liver biopsy was averted.
Table 1: Investigations of the index case before and after the treatment

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  Discussion Top


This young male was brought to emergency in a very sick state and keeping in view his low oxygen saturation patient was immediately shifted to intensive care where he was treated with high-flow oxygen (8 l/min) and overnight his oxygen saturation improved. In the beginning of his hospital course brucellosis was difficult to predict as he had features of bone marrow suppression and hematological malignancy like presentation with multiple organ dysfunction syndrome. The bedside X-ray chest did not reveal any major lung lesion and his echocardiography was normal as well. It was only after the results of Brucella serology were available on day 4th of his admission, he was started on brucellosis treatment regimen. He showed progressive improvement with the treatment. Pulmonary involvement in acute brucellosis is one of the rare complications. In one of the largest retrospective series, the data on 133 patients with brucellosis and respiratory complication was analyzed by Erdem et al.[3] The authors observed that the most frequent radiological pattern was consolidation followed by pleural effusion. However, the index case had prominent bronchovascular markings and no evidence of consolidation or pleural effusion were observed. The sputum analysis in the aforementioned study had not revealed brucella while as pleural fluid analysis in 2 out of 19 samples were positive for brucellosis suggesting that pleural fluid may be screened for Brucella in a given case. The authors in this study concluded that brucellosis with pulmonary involvement is rare but has a good prognosis following treatment with appropriate antibiotics. Brucellosis may involve multiple organs and progress with complications. Brucella endocarditis, disseminated intravascular coagulopathy, and chronic renal insufficiency are rare complications of brucellosis. Capillary leak syndrome due to brucellosis has been reported.[4] The index case had features of hemopoietic involvement, renal involvement, liver involvement, and respiratory system involvement and he improved with treatment. In a study from Turkey, data on 52 patients with brucellosis showed the presence of pancytopenia in 11 patients. The authors observed that the most frequent admission symptoms and findings of the patients with pancytopenia were fever and fatigue. Splenomegaly followed by hepatomegaly had been constant clinical finding in their series as was observed in the index case.[5] In their study, pancytopenia improved with treatment of brucellosis as in the index case. The authors concluded that brucellosis can show great similarity with hematological disorder and Crimean-Cango hemorrhagic fever, another zoonotic disease endemic in Turkey. They further concluded that brucellosis should be considered in the differential diagnosis of pancytopenia, treatment-resistant immune thrombocytopenia, and viral hemorrhagic disease, especially in countries where brucellosis is endemic.[5] The patient had clinical jaundice and on evaluation high bilirubin levels and high liver enzymes. He had negative viral serologies and no biliary obstruction was noted on ultrasound examination. The patient's liver enzymes were last to return to normal which averted the need of liver biopsy as was planned during the hospital stay. The index case also had severe thrombocytopenia and epistaxis. In a retrospective data on 622 patients with brucellosis from Turkey, Karaman et al.[6] observed that 28.6% of patients had anemia, alone or in combination with leukopenia, or thrombocytopenia. In their study, 16% of patients had thrombocytopenia while 13.9% had leukopenia and 7.7% had pancytopenia. The authors in this study concluded that the hematologic complications such as anemia, thrombocytopenia, and leukopenia were more frequently seen in acute brucellosis cases. The authors were further of the opinion that acute brucellosis should also be considered in the differential diagnosis when hematologic abnormalities such as severe thrombocytopenia, bicytopenia, and pancytopenia are clinically encountered. The index case had epistaxis due to severe thrombocytopenia and initially was managed with platelet-rich plasma and later the platelet counts steadily improved with antibiotic treatment and he did not require further support with platelet-rich plasma. It is very important that brucellosis with hemorrhage should be differentiated from viral hemorrhagic fever, malignancy, and other blood disorders. In this context, BMA is mandatory to differentiate from other blood disorders. The BMA of the index case did not show any abnormality. Thrombocytopenia in brucellosis could be immune-mediated and steroids have been used in combination with antibiotics in brucellosis.[7] However, the index case had progressive increase in platelet counts after Brucella treatment and no steroids were used to improve the platelet count. In acute brucellosis, the platelet count may go drastically down and as low as 4000 responding to Brucella treatment has been reported.[8] While the index case had epistaxis as bleeding diatheses due to thrombocytopenia hematemesis and melena in a 59-year-old female patient has been reported responding to Brucella treatment.[9]

The index case had predominantly hepatitis and he had no clinical features of cholestasis. However, patients with brucellosis can present with cholestasis as well. The data on 325 brucellosis patients with significant hepatobiliary involvement from 30 centers between 2000 and 2013 were analyzed by Ozturk-Engin et al.[10] The authors in this study observed clinical hepatitis in 284 patients (87.3%), while cholestasis was detected in 215 (66.1%) patients suggesting high index of clinical suspicion of acute brucellosis in endemic areas. Antibiotic treatment remains the cornerstone modality to manage hepatitis in a case of brucella hepatitis. The authors in the same study while comparing various regimens observed that hepatobiliary involvement in brucellosis has a benign course with suitable antibiotics and the use of doxycycline and an aminoglycoside regimen seems a better strategy in patients. Brucella hepatitis in the index case responded very well to antibiotics.

Another Brucella-related complication was acute renal failure in the index case which improved with fluid replacement. A combination of clinical nephritis and acute renal failure due to brucellosis in a 42-year-old male has been reported with complete recovery following treatment.[11] In addition, many diverse etiologies can play a role in the renal involvement associated with Brucella infection. In a retrospective analysis data on 15 patients with acute renal failure due to Brucellosis, Ceylan et al.[12] observed that renal failure due to brucellosis could have multiple causes ranging from prerenal azotemia to brucellar tubulointerstitial nephritis. The causes even could be drug-related toxicities in a particular case. In their series, the authors had observed complete recovery of renal failure in all patients and only one had progressed to chronic renal disease. Five patients in their series required hemodialysis. While in the index case, renal failure was possibly due to prerenal factors which improved after fluid replacement and he did not need any artificial support. However, progression to end-stage renal disease due to B. melitensis has been reported despite antibiotic and steroid therapy in a 24-year-old female.[13]

In endemic areas, the clinical suspicion in a given case has to be very high. A 26-year-old shepherd presenting with fever, ataxia, and dysarthria and weight loss was proved to have brucellosis by blood culture. He had low standard tube agglutination test and 2-mercaptoethanol (2-ME) titers 1:80 and 1:40, respectively. The authors in this report have highlighted that low titers should be considered an indication of brucellosis in an endemic area.[14]

To conclude in endemic areas, clinicians should consider brucellosis in any unusual presentation involving multiple organ systems, even if serology is inconclusive.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Pappas G, Papadimitriou P, Akritidis N, Christou L, Tsianos EV. The new global map of human brucellosis. Lancet Infect Dis 2006;6:91-9.  Back to cited text no. 1
    
2.
Fallatah SM, Oduloju AJ, Al-Dusari SN, Fakunle YM. Human brucellosis in Northern Saudi Arabia. Saudi Med J 2005;26:1562-6.  Back to cited text no. 2
    
3.
Erdem H, Inan A, Elaldi N, Tekin R, Gulsun S, Ataman-Hatipoglu C, et al. Respiratory system involvement in brucellosis: The results of the Kardelen study. Chest 2014;145:87-94.  Back to cited text no. 3
    
4.
Karsen H, Koruk ST, Calisur C, Karagac L, Duygu F. A case of brucellosis complicated with fatal capillary leak syndrome. Bratisl Lek Listy 2012;113:511-3.  Back to cited text no. 4
    
5.
Karli A, Sensoy G, Albayrak C, Koken O, Cıraklı S, Belet N, et al. Pancytopenia as the initial manifestation of brucellosis in children. Vector Borne Zoonotic Dis 2015;15:545-9.  Back to cited text no. 5
    
6.
Karaman K, Akbayram S, Bayhan GI, Dogan M, Parlak M, Akbayram HT, et al. Hematologic findings in children with brucellosis: Experiences of 622 patients in Eastern Turkey. J Pediatr Hematol Oncol 2016;38:463-6.  Back to cited text no. 6
    
7.
Guzel Tunccan O, Dizbay M, Senol E, Aki Z, Ozdemir K. Isolated severe immune thrombocytopenia due to acute brucellosis. Indian J Hematol Blood Transfus 2014;30:27-9.  Back to cited text no. 7
    
8.
Elmas Dal S, Ersoy Y, Ali Erkurt M, Yetkin F, Kuzucu C, Akdogan O, et al. An uncommon case of acute brucellosis presenting with severe thrombocytopenia. Intern Med 2012;51:3291-3.  Back to cited text no. 8
    
9.
Baldane S, Sivgin S, Alkan TS, Kurnaz F, Pala C, Keklik M, et al. An atypical presentation of brucellosis in a patient with isolated thrombocytopenia complicated with upper gastrointestinal tract bleeding. Case Rep Med 2012;2012:473784.  Back to cited text no. 9
    
10.
Ozturk-Engin D, Erdem H, Gencer S, Kaya S, Baran AI, Batirel A, et al. Liver involvement in patients with brucellosis: Results of the Marmara study. Eur J Clin Microbiol Infect Dis 2014;33:1253-62.  Back to cited text no. 10
    
11.
Dagli O, Dokur M, Guzeldag G, Ozmen Y. Acute renal failure due to Brucella melitensis. J Infect Dev Ctries 2011;5:893-5.  Back to cited text no. 11
    
12.
Ceylan K, Karahocagil MK, Soyoral Y, Sayarlioğlu H, Karsen H, Dogan E, et al. Renal involvement in Brucella infection. Urology 2009;73:1179-83.  Back to cited text no. 12
    
13.
Bakri FG, Wahbeh A, Mahafzah A, Tarawneh M. Brucella glomerulonephritis resulting in end-stage renal disease: A case report and a brief review of the literature. Int Urol Nephrol 2008;40:529-33.  Back to cited text no. 13
    
14.
Khorvash F, Keshteli AH, Behjati M, Salehi M, Emami Naeini A. An unusual presentation of brucellosis, involving multiple organ systems, with low agglutinating titers: A case report. J Med Case Rep 2007;1:53.  Back to cited text no. 14
    



 
 
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