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   Table of Contents     
CASE REPORT  
Year : 2014  |  Volume : 6  |  Issue : 2  |  Page : 79-81
Acute arthritis in Crimean-Congo hemorrhagic fever


1 Infectious Disease Clinic, University Clinical Centre of Kosova, Faculty of Medicine, Prishtina University, Rrethi i spitalit, p.n., 10000 Pristina, Kosova
2 Main Family Health Center of Prizren, Xhemil Fluku, p.n., 20000 Prizren, Kosova

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Date of Web Publication7-May-2014
 

   Abstract 

Crimean-Congo hemorrhagic fever is a severe viral disease caused by a Nairovirus. An atypical manifestation in the form of acute arthritis was found in a confirmed Crimean-Congo hemorrhagic fever virus Kosova-Hoti strain positive patient. Acute arthritis in Crimean-Congo hemorrhagic fever (CCHF) may be as a result of immune mechanisms or the bleeding disorder underlying CCHF.

Keywords: Arthritis, Crimean-Congo hemorrhagic fever, Kosova-Hoti virus

How to cite this article:
Ahmeti S, Ajazaj-Berisha L, Halili B, Shala A. Acute arthritis in Crimean-Congo hemorrhagic fever. J Global Infect Dis 2014;6:79-81

How to cite this URL:
Ahmeti S, Ajazaj-Berisha L, Halili B, Shala A. Acute arthritis in Crimean-Congo hemorrhagic fever. J Global Infect Dis [serial online] 2014 [cited 2018 Nov 14];6:79-81. Available from: http://www.jgid.org/text.asp?2014/6/2/79/132052



   Introduction Top


Crimean-Congo hemorrhagic fever (CCHF) is an acute tick-borne viral disease caused by CCHF virus, a Nairovirus genus member of Bunyaviridae family. CCHF zoonosis produces asymptomatic infection in affected animals. Humans appear to be the only hosts where Crimean-Congo hemorrhagic fever virus (CCHFV) produces a severe hemorrhagic disease with a mortality rate up to 30%. [1] CCHF is mainly spread via Hyalomma genus ticks and direct contact with blood and tissues of infected persons and viremic animals. Kosova is a frequently affected CCHF country with the first reported case in 1954. Since 1989 epidemic outbreaks are recorded every 4-5 years. [2] The genome of Kosova's authentic CCHF virus strain Kosova-Hoti has been completely sequenced revealing characteristic nucleotide sequences especially in the L segment of the genome that may explain its high pathogenicity and mortality. [3],[4] Incubation, prehemorrhagic, hemorrhagic and convalescence phases characterize CCHF. The pre-hemorrhagic phase is characterized by fever, chills, headache, muscle pain, dizziness, nausea, vomiting, and diarrhea. The hemorrhagic phase follows with petechiae, echymosis, gingival bleeding, epistaxis, hematemesis, melena and visceral bleedings. Fatal outcomes of CCHF can occur in hemorrhagic phase because of cerebral hemorrhage, severe anemia, hemorrhagic shock, multiple organ failure and coma. [5] An atypical manifestation of CCHF was found in an adolescent patient in the form of acute arthritis. This finding according to current literature review has not been reported before.


   Case Report Top


Here we report a case of a 15-year-old boy who comes from a rural area known to be a natural CCHF focus and presents with fever (40°C), fatigue, anorexia, myalgia, polyarthralgia, backache, 2 days after his brother's illness. He denies any tick bite, but admits that he was taking care of his sick brother who first became ill. On day 1 of disease, he was hospitalized in a regional hospital with the diagnosis of acute febrile illness. On day 3, as his condition was not improving, he is transferred in University Clinical Center in Prishtina as his ill brother tested CCHFV positive. In admission he was very ill-appearing, febrile with general intoxication symptoms. The blood was drawn for laboratory investigations and the measured parameters were as following : e0 rythrocyte count of 4.0 × 10 6 cells/mm 3 ; hemoglobin level of 11.0 g/dL; leukocyte count of 2.0 × 10 3 /mm 3 ; thrombocyte count of 28 × 10 3 /mm 3 ; liver enzyme levels: Aspartate aminotransferase-377.5 U/L, alanine aminotransferase-154 U/L, lactate dehydrogenase-1558 U/L, creatine phosphokinase-2380 U/L; C-reactive protein-12 mg/L; fibrinogen-3.2 g/L; rheumatoid factor-negative; serum antistreptolysin O antibody titer- 132 units/ml; blood cultures-negative. Leucopenia, thrombocytopenia and liver dysfunction were noted. Blood real-time-polymerase chain reaction (RT-PCR) on day 3 resulted positive for CCHFV. On day 4, enathema, petechiae and gingival bleeding dominated. On day 6, his condition deteriorated further with massive bleedings-epistaxis, melena, spontaneous bleeding from injection sites; hypotension, bradycardia. On abdominal ultrasound hepatosplenomegaly, pleural effusion, acute cholecystitis and hemoperitoneum were found [Figure 1]. Treatment with ribavirin was initiated. On day 8 his temperature marked 39°C, his clinical condition was severely affected with intense chest pain and muscle aches. Chest X-ray showed right lower lobe infiltrations; chest computed tomography-scan revealed right lower lobe pleuropneumonia with abscess tendency formation [Figure 2]. On day 9, urine RT-PCR was positive for CCHFV. On day 12, patient complained of pain on his left knee, upon examination swelling and limited joint movements were noted. A left knee X-ray was ordered, orthopedic surgeon was consulted and in the meantime brucellosis, leptospirosis, hepatitis A, B, C and post-streptococcal arthritis were serologically ruled out. Synovial membrane hypertrophy with joint effusion in the level of suprapatellar recess was noted on left knee ultrasound [Figure 3]. Positive patella Ballottment test was assessed clinically. After day 16, knee pain and other signs of inflammation regressed gradually. From day 22 his condition gradually improved and a week later he was discharged from hospital.
Figure 1: Abdominal ultrasound: (a) Enlarged liver; disseminated anechogenic nodules implying punctiform bleeding are present; (b) slightly enlarged spleen; (c) gallbladder has edematous walls, an intraluminal anechogenic zone (liquid-blood) and pericholecystic fluid; (d) fluid in pleural cavity and abdominal cavity-pleural effusion and hemoperitoneum

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Figure 2: Diagnostic testing for chest pain: (a) chest-X-ray showing right lower lobe infiltration; (b) chest computed tomography (CT) scan shows right lower lobe pleuropneumonia with abscess tendency formation; (c) chest CT scan evidencing pleural effusion

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Figure 3: Left Knee Joint: (a) marked swelling is noted; (b) left knee X-ray: Anterior-posterior and lateral views, soft-tissue swelling above and below patella is noted, synovitis was suggested; (c) and (c') left knee ultrasound: synovial membrane hypertrophy and joint effusion are noted

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   Discussion Top


CCHF exact mechanism of disease is not well-understood. Nevertheless, endothelial cells, immune mechanisms, viral load and coagulation cascade are implicated in its pathogenesis. [6] Viral replication takes place in endothelial cells, followed by CD4+ T-helper cell activation that by secreting enormous amounts of tumor necrosis factor (TNF), IL-1, IL-6 causes macrophage co-activation. [7] Both viral replication and massive cytokines release damage the endothelium of blood vessels promoting vasodilatation, platelet aggregation and activation of coagulation cascade, hemorrhage, disseminated intravascular coagulation and multi-organ failure. [6] Activated macrophages can lead to hemophagocytosis syndrome, where macrophages phagocytize blood cells causing the characteristic cytopenia encountered in CCHF. A study by Ergonul et al. found increased levels of TNF, IL-1 and IL-6 in CCHF patients, supporting the crucial role of cytokines in CCHF pathogenesis. [7] Acute arthritis is an unusual CCHF disease manifestation. Viruses are a known cause of arthritis and arthralgias and how they elicit arthritis depends both on host and viral factors. Viruses mainly initiate rheumatic symptoms by direct invasion of synovium or by deposition of immune complexes into joint spaces. Patients present with symmetric polyarticular arthralgias or arthritis that can be associated by a rash, which generally occurs before or with the clinical onset of infection. [8] Considering CCHF mechanisms of disease development such as endothelial viral replication, intense activation of macrophages and massive cytokine release it can be thought that the same may apply to the arthritic symptoms encountered in CCHF associated arthritis, as activated synovial macrophages can make joint inflammation unavoidable. [6],[7],[8] Immune mediated mechanisms in the pathogenesis of CCHF associated acute arthritis cannot be excluded. However, the involvement of a single large joint and the development of acute arthritis later in disease course imply that arthritic symptoms may be a consequence of bleeding disorder underlying CCHF. Acute arthritis might be a part of hemorrhagic syndrome present in CCHF virus infection, even though its manifestation through monoarticular joint swelling and pain does not belong to the CCHF characteristic clinical picture. Affected joint with acute hemarthrosis is swollen, warm, painful with reduced range of motion and usually only one joint is affected at a time [9] which can explain our patient's knee symptoms. Synovial hypertrophy [Figure 3] is a known early consequence of toxic effects of blood products and if inflammation is ongoing fibrosis and impaired joint movement may result. Our patient recovered fully from acute arthritis regardless of possible mechanisms that led to its development.

 
   References Top

1.Appannanavar SB, Mishra B. An update on Crimean Congo hemorrhagic Fever. J Glob Infect Dis 2011;3:285-92.  Back to cited text no. 1
    
2.Humolli I. Epidemiological and Serological Features of Crimean-Congo Hemorrhagic Fever and Determination of Endemic Zones of Disease in Kosova 1995-2002. [dissertaton] University of Prishtina, Faculty of Medicine; 2003.  Back to cited text no. 2
    
3.Duh D, Nichol ST, Khristova ML, Saksida A, Hafner-Bratkovic I, Petrovec M, et al. The complete genome sequence of a Crimean-Congo hemorrhagic fever virus isolated from an endemic region in Kosovo. Virol J 2008;5:7.  Back to cited text no. 3
    
4.Frias-Staheli N, Giannakopoulos NV, Kikkert M, Taylor SL, Bridgen A, Paragas J, et al. Ovarian tumor domain-containing viral proteases evade ubiquitin- and ISG15-dependent innate immune responses. Cell Host Microbe 2007;2:404-16.  Back to cited text no. 4
    
5.Swanepoel R, Gill DE, Shepherd AJ, Leman PA, Mynhardt JH, Harvey S. The clinical pathology of Crimean-Congo hemorrhagic fever. Rev Infect Dis 1989;11 Suppl 4:S794-800.  Back to cited text no. 5
    
6.Duru F, Fisgin T. Hematological aspects of Crimean-Congo hemorrhagic fever. Turk J Hematol 2009;26:161-6.  Back to cited text no. 6
    
7.Ergonul O, Tuncbilek S, Baykam N, Celikbas A, Dokuzoguz B. Evaluation of serum levels of interleukin (IL)-6, IL-10, and tumor necrosis factor-alpha in patients with Crimean-Congo hemorrhagic fever. J Infect Dis 2006;193:941-4.  Back to cited text no. 7
    
8.Moore TL, Syed R. Specific viruses that cause arthritis. In: Basow DS, editor. UpToDate. Waltham, MA: UpToDate; 2013.  Back to cited text no. 8
    
9.Nigrovic PA. Hemarthrosis. In: Basow DS, editor. UpToDate. Waltham, MA: UpToDate; 2013.  Back to cited text no. 9
    

Top
Correspondence Address:
Dr. Salih Ahmeti
Infectious Disease Clinic, University Clinical Centre of Kosova, Faculty of Medicine, Prishtina University, Rrethi i spitalit, p.n., 10000 Pristina
Kosova
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0974-777X.132052

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2008 Journal of Global Infectious Diseases | Published by Wolters Kluwer - Medknow
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