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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 7  |  Issue : 3  |  Page : 285-287

The paradoxical evolution in an unusual case of disseminated tuberculosis: Spontaneous resolution with disease progression


Department of Infectious Diseases, Singapore General Hospital, Singapore, Singapore

Date of Web Publication6-Sep-2018

Correspondence Address:
Dr. Shuwei Zheng
Department of Infectious Diseases, Singapore General Hospital, Outram Road
Singapore
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijmy.ijmy_103_18

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  Abstract 


Mycobacterium tuberculosis can cause a myriad of clinical manifestations. We describe a case of a patient with end-stage renal failure, who presented with disseminated tuberculosis over the course of five months, manifesting with a self-resolving mediastinal mass, progressive lymphadenopathy, genitourinary, and musculoskeletal tuberculosis.

Keywords: Anterior mediastinal mass, disseminated tuberculosis, end-stage renal failure


How to cite this article:
Thien SY, Zheng S. The paradoxical evolution in an unusual case of disseminated tuberculosis: Spontaneous resolution with disease progression. Int J Mycobacteriol 2018;7:285-7

How to cite this URL:
Thien SY, Zheng S. The paradoxical evolution in an unusual case of disseminated tuberculosis: Spontaneous resolution with disease progression. Int J Mycobacteriol [serial online] 2018 [cited 2020 May 30];7:285-7. Available from: http://www.ijmyco.org/text.asp?2018/7/3/285/240686




  Introduction Top


Mycobacterium tuberculosis is an ancient bacterium known to cause a myriad of clinical manifestations. Herein, we highlight the clinical and radiological evolution of a patient with a delayed diagnosis of disseminated tuberculosis, manifesting with a self-resolving tuberculous mediastinal mass.


  Case Report Top


A 64-year-old female presented with an incidental anterior mediastinal mass measuring 5.9 cm × 2.9 cm × 5.8 cm on computed tomography (CT) scan of the thorax [Figure 1]a and [Figure 1]b as part of a precoronary artery bypass graft evaluation. There were no pulmonary lesions or lymphadenopathy seen. She had recently been diagnosed with triple-vessel coronary artery disease following an out-of-hospital collapse with ventricular fibrillation. She has a known history of end-stage renal failure (ESRF), secondary to diabetic nephropathy, and has been on hemodialysis for the past 2 months. Her last glycated hemoglobin then was 5.3%. In view of the concerns for malignancy, she underwent a CT imaging of her abdomen and pelvis, which only revealed nonspecific thickening of the rectum and thinning of the renal parenchyma. A CT-guided biopsy of the anterior mediastinal mass was performed; however, the results were nonyielding. Serological testing for human immunodeficiency virus was negative.
Figure 1: (a and b) Coronal view of lobulated anterior mediastinal mass (white arrowheads), indenting on to the pericardium. (c and d) Retrocaval lymphadenopathy and right external iliac lymphadenopathies (white arrows) on computed tomography scan 5 months after initial presentation

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A month later, she presented with hematochezia. She underwent a flexible sigmoidoscopy revealing evidence of proctitis in the lower rectum without any biopsy performed. A CT colonography showed mural thickening at the rectum and new multiple small retroperitoneal and pelvic lymphadenopathy. She underwent a repeat CT-guided biopsy of the mediastinal mass, noting an interval reduction in the size of this lesion. Histology returned, showing the evidence of necrotizing granulomatous inflammation. Unfortunately, she defaulted follow-up.

Five months later, she presented with recurrent right-sided knee pain and fever over the course of 2 weeks. On examination, she was febrile but hemodynamically stable. Cardiorespiratory and abdominal examinations were unremarkable. She had a limited range of motion over her knee with a tender, soft-tissue swelling medially. Her hemoglobin was 11.3 g/dL, white blood cell (WBC) count was 35.69 × 109/L, and platelet count was 538 × 109/L. Her C-reactive protein (CRP) was 183 mg/L, and the erythrocyte sedimentation rate was 84 mm/h. A bedside arthrocentesis yielded minimal fluid with negative bacterial gram stain and cultures. She was commenced on IV cefazolin. This improved but did not resolve her knee pain and swelling. Her WBC count and CRP reduced to 11.18 × 109/L and 44.5 mg/L, respectively, but fever persisted. A CT imaging of her chest, abdomen, and pelvis was repeated to reevaluate the abnormalities seen previously. There was a complete resolution of the mediastinal mass and rectal thickening, but an interval increase in the size of the previous intra-abdominal and pelvic lymphadenopathy [Figure 1]c and [Figure 1]d. A magnetic resonance imaging of her right knee revealed a 7.3 cm × 3.8 cm × 6.4 cm lobulated mass at the posteromedial aspect, likely representing a fluid-distended bursa [Figure 2]. There was also evidence of knee effusion, marrow edema in the patella and proximal tibia, and an irregularity of the subchondral bone plates suggestive of osteomyelitis. A CT-guided aspiration of the fluid collection at the right knee yielded 50 ml of frank pus. This tested negative for bacterial gram stain and culture, fungal microscopy, and acid-fast bacilli (AFB) smears. M. tuberculosis complex (MTC) deoxyribonucleic acid (DNA) was detected. Three samples of early morning urine were negative for MTC DNA amplification and AFB smears, pending cultures. She underwent a right knee arthroscopic washout. Intraoperative knee fluid and tissue returned positive for MTC DNA in two of three specimens sent, although the AFB smears were negative. She was started on MTC induction therapy with rifampicin, isoniazid, pyrazinamide, and ethambutol. At the outpatient review, her clinical symptoms resolved with normalization of her WBC count and CRP. Eventually, two of the three early morning urine specimens and all four knee fluid/tissue specimens grew pan-susceptible MTC. After 6 months of treatment, a repeat CT imaging of her abdomen and pelvis showed a resolution of the intra-abdominal lymphadenopathy. As such, she had disseminated tuberculosis involving the right knee joint with osteomyelitis, genitourinary tract, intra-abdominal lymph nodes, and a self-resolving mediastinal necrotizing granulomatous mass. She completed a total of 2 months of induction followed by 7 months of maintenance therapy with rifampicin and isoniazid.
Figure 2: (a) Magnetic resonance imaging of the right knee showing T1 coronal view of a collection at medial aspect of the joint. (b) T2 transverse view of the same collection and proximal tibial bone marrow edema (white arrow)

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  Discussion Top


ESRF is a risk factor for tuberculosis as a result of decreased cell-mediated immunity (CMI). Extrapulmonary tuberculosis is also known to be more common among this group of patients in comparison to the general population.[1] Disseminated tuberculosis, defined as isolation of Mycobacterium tuberculosis from the blood or bone marrow from a liver biopsy specimen, or from specimens from two or more contiguous organs in a single patient, has been rarely described in patients with ESRF.[2],[3],[4],[5],[6],[7],[8] Among 164 patients in a single center with disseminated tuberculosis, the most common comorbidity was the acquired immunodeficiency syndrome, followed by diabetes mellitus and malignancy. Only 8.5% had ESRF in this series.[8] We present a rare case of disseminated tuberculosis in a patient with ESRF who developed new symptoms over the course of 6 months before the diagnosis of tuberculosis was confirmed.

In the initial stage of infection, MTC disseminates before the onset of an acquired macrophage-activating CMI. With the development of CMI, granulomatous lesions form. A tissue-damaging response triggered by MTC antigens results in the destruction of inactivated macrophages that contain multiplying MTC and necrosis. Where these two host responses predominate, latency ensues; where host responses fail, expanding infection results in disease.[9] Our patient demonstrated an interesting ability to contain infectious tubercles within the anterior mediastinum but with uncontrolled dissemination and disease at other body sites (abdominal lymph nodes, genitourinary tract, and musculoskeletal system). While such a phenomenon of a self-limited end-organ involvement of tuberculosis is well described in tuberculous pleural effusions where spontaneous resolution within weeks is the norm,[10] this is not widely described in other disease sites. Studies about the natural history of tuberculosis are surprisingly few and mostly based on prechemotherapy data.[11] Our case demonstrated an interesting intricate balance between the tubercle bacilli and the innate host immune response pretuberculous chemotherapy.

Acknowledgment

The authors would like to thank the Medical Publication Support Unit, National University Health System, for their editorial services.

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.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Drain PK, Bajema KL, Dowdy D, Dheda K, Naidoo K, Schumacher SG, et al. Incipient and subclinical tuberculosis: A clinical review of early stages and progression of infection. Clin Microbiol Rev 2018;31. pii: e00021-18.  Back to cited text no. 1
    
2.
Hussein MM, Mooij JM, Roujouleh H. Tuberculosis and chronic renal disease. Semin Dial 2003;16:38-44.  Back to cited text no. 2
    
3.
Gavriilaki E, Sabanis N, Paschou E, Kalaitzoglou A, Michalaki K, Zarampoukas T, et al. Disseminated tuberculosis: A neglected entity in immunocompromised hemodialysis patients. Hemodial Int 2015;19:E8-11.  Back to cited text no. 3
    
4.
Hasegawa J, Wakai S. A case of disseminated tuberculosis after the initiation of hemodialysis. CEN Case Rep 2013;2:252-5.  Back to cited text no. 4
    
5.
Ko YC, Lee CT, Cheng YF, Hung KH, Kuo CY, Huang CC, et al. Hypercalcaemia and haemophagocytic syndrome: Rare concurrent presentations of disseminated tuberculosis in a dialysis patient. Int J Clin Pract 2004;58:723-5.  Back to cited text no. 5
    
6.
Ogutmen B, Tuglular S, Al Ahdab H, Akoglu E, Ozener Q. Tuberculosis peritonitis with clear fluid accompanying disseminated tuberculosis in a CAPD patient. Perit Dial Int 2003;23:95-6.  Back to cited text no. 6
    
7.
Peces R, Alvarez J. Hypercalcemia and elevated 1,25(OH) 2D3 levels in a dialysis patient with disseminated tuberculosis. Nephron 1987;46:377-9.  Back to cited text no. 7
    
8.
Wang JY, Hsueh PR, Wang SK, Jan IS, Lee LN, Liaw YS, et al. Disseminated tuberculosis: A 10-year experience in a medical center. Medicine (Baltimore) 2007;86:39-46.  Back to cited text no. 8
    
9.
Raviglione MC. Tuberculosis. In: Kasper D, Fauci A, Hauser S, Longo D, Jameson J, Loscalzo J, editors. Harrison's Principles of Internal Medicine. 19th ed. New York: McGraw-Hill; 2014. Available from: http://www.accessmedicine.mhmedical.com/content.aspx?bookid=1130 & sectionid= 79737003. [Last accessed on 2018 Jul 18].  Back to cited text no. 9
    
10.
Zhai K, Lu Y, Shi HZ. Tuberculous pleural effusion. J Thorac Dis 2016;8:E486-94.  Back to cited text no. 10
    
11.
Tiemersma EW, van der Werf MJ, Borgdorff MW, Williams BG, Nagelkerke NJ. Natural history of tuberculosis: Duration and fatality of untreated pulmonary tuberculosis in HIV negative patients: A systematic review. PLoS One 2011;6:e17601.  Back to cited text no. 11
    


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