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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 9  |  Issue : 4  |  Page : 442-444

Xanthogranulomatous pleuritis - An unusual presentation of tuberculosis


1 Department of Pulmonary Medicine, Rajagiri Hospital, Kochi, Kerala, India
2 Department of Pathology, Rajagiri Hospital, Kochi, Kerala, India

Date of Submission10-Aug-2020
Date of Decision11-Aug-2020
Date of Acceptance16-Aug-2020
Date of Web Publication15-Dec-2020

Correspondence Address:
Jolsana Augustine
Department of Pulmonary Medicine, Rajagiri Hospital, Aluva, Kochi, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijmy.ijmy_146_20

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  Abstract 


In pulmonary practice, pleural effusion is a commonly encountered entity and has various etiologies. Pleural effusions in postpartum women can be an incidental self-limiting finding. The presence of systemic or respiratory symptoms, however, calls for prompt etiological workup and targeted therapy. Tuberculous pleuritis and lupus-related pleural disease are well known to flare up in the postpartum period. We describe the case of a young healthcare worker with no previous comorbidities who presented with fever, breathlessness, and chest pain 2 weeks after an uneventful confinement. Chest radiograph revealed moderate left-sided pleural effusion. Pleural fluid analysis was biochemically consistent with tubercular effusion. Pleural biopsy histological examination showed features of xanthomatous pleuritis and Cartridge based nucleic acid amplification test (CB-NAAT) showed evidence of Mycobacterium tuberculosis(MTB). She was initiated on antitubercular medicines to which she responded well with the resolution of clinical symptoms and pleural collection. This is the first case report describing an association of xanthogranulomatous pleuritis with tuberculosis.

Keywords: Granulomatous pleuritis, xanthogranulomas, xanthomatous pleuritis


How to cite this article:
Augustine J, Venkitakrishnan R, Ramachandran D, Abraham L. Xanthogranulomatous pleuritis - An unusual presentation of tuberculosis. Int J Mycobacteriol 2020;9:442-4

How to cite this URL:
Augustine J, Venkitakrishnan R, Ramachandran D, Abraham L. Xanthogranulomatous pleuritis - An unusual presentation of tuberculosis. Int J Mycobacteriol [serial online] 2020 [cited 2021 Jan 24];9:442-4. Available from: https://www.ijmyco.org/text.asp?2020/9/4/442/303440




  Introduction Top


Xanthogranulomatous inflammation is an entity that, although uncommon, has been reported in several organs, most commonly the kidney, female genital tract, and gallbladder.

Xanthogranulomatous inflammation of the pleura is very rare. The etiology of xanthogranulomatous inflammation is unknown. We present the case of a young lady who presented with exudative pleural effusion and was detected to have xanthogranulomatous pleuritis and tubercular involvement of the pleura. This is the first case report linking xanthogranulomatous inflammation of the pleura to tuberculosis.


  Case Report Top


A 27-year-old woman, healthcare worker by profession, presented to the pulmonary medicine outpatient department in the postpartum period with left-sided chest pain, fever, and breathlessness for 2 weeks after an uneventful confinement. She denied any history of breathlessness or allergic background before. Initial hemogram and renal and liver functions were within normal limits. Her C-reactive protein was elevated (55 mg/l). Her chest radiograph showed left-sided moderate pleural effusion [Figure 1]a. Computed tomography of the chest was done which revealed moderate left-sided effusion along with the left upper lobe and lingular patchy consolidation [Figure 1]b. Thoracentesis was done from the left pleural cavity, and 1.2 l of straw-colored fluid was aspirated. The reports revealed an exudative lymphocytic effusion with high Adenosine Deaminase (ADA) (72 U/l). Mantoux test was nonreactive.
Figure 1: (a) Chest radiograph showing left moderate effusion. (b) Computed tomography chest revealing moderate left-sided effusion along with the left lower lobe passive collapse

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Echocardiogram was unremarkable. Since there was re-accumulation of the fluid leading to respiratory distress, she was admitted and stabilized. Connective tissue serology (RA factor and ANA) was sent to rule out any autoimmune-related effusion, all of which returned nonreactive. Ultrasound of the abdomen and pelvis showed no intra-abdominal pathology. Medical thoracoscopy was done under conscious sedation, pleural surfaces were found to be inflamed, and fibrinous adhesions between the layers of pleura were visualized [Figure 2]a. Multiple biopsy samples taken from suspicious areas were sent for mycobacterial and histopathological evaluations. Intrapleural fibrinolysis with streptokinase was performed after which her X-ray showed significant clearance and reasonable lung expansion. Bacterial culture of the pleural fluid was negative, and GeneXpert was positive for Mycobacterium tuberculosis (MTB). Bronchoscopy was done, and bronchoalveolar lavage was negative for bacterial culture and MTB. Pleural biopsy showed fibrocollagenous tissue with fibrinoid change and small foci of necrosis. Scattered lymphocytic infiltration was also noted along with a few macrophages, some showing foamy cytoplasm with occasional multinucleated giant cell rendering a pathological diagnosis of xanthogranulomatous inflammation [Figure 2]b. No definite granulomas were seen. Immunohistochemical analysis was not performed in our case as the patient opted out.
Figure 2: (a) Thoracoscopic image showing inflamed pleura with fibrinous adhesions. (b) Histopathological examination showing fibrocollagenous tissue with fibrinoid change and small foci of necrosis along with scattered lymphocytic infiltration and a few macrophages some showing foamy cytoplasm with occasional multinucleated giant cell

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She was started on antitubercular medications to which she responded promptly with defervescence of fever and clearance of pleural collection. She was kept on close follow-up during and after completion of antitubercular treatment. Her chest radiograph after treatment cessation and 1-year follow-up showed total clearance of effusion [Figure 3].
Figure 3: Chest radiograph after 1 year showing almost total clearance of effusion with some pleural thickening

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  Discussion Top


Xanthomatous inflammation is a rare form of chronic inflammation characterized by the presence of lipid-laden macrophages admixed with lymphocytes, plasma cells, neutrophils, and often multinucleated giant cells with or without cholesterol clefts.[1] The necrosis and hemorrhage are evidence of on-going inflammation. The entity was initially described in the kidney by Osterlind in 1944.[2] It has also been reported in other organs such as prostate, epididymis, ovary, urinary bladder, appendix, and breast,[3],[4] though most commonly described in gallbladder and kidneys.[5] It is more common in the Indian population compared to the western world.[5] In most of the cases, histopathological assessment is needed to establish correct diagnosis.

The exact etiology of xanthomatous inflammation is unclear. Proposed etiologies include defective lipid transport, immunologic disturbances, infection by low virulence organisms, and lymphatic obstruction.[6] It has been postulated that hemorrhage plays a major role in the development of foamy macrophages. The ingested erythrocytes and platelets at the bleeding site overwhelm the lysosomal system of the macrophages causing deposition of phospholipids, which results in a foamy appearance of the macrophages.[1]

Xanthomatous inflammation in the pleura is histomorphologically similar to xanthomatous inflammation in gallbladder, kidney, and appendix. There have been case reports of pleural involvement in xanthogranulomatous pyelonephritis.[7] Very rarely, it has been reported as isolated pleural effusion.[8]

Xanthomatous pleuritis mimicking mesothelioma has been reported in the literature necessitating extensive immunohistochemistry studies for conclusive diagnosis.[9] Etiology of xanthomatous pleuritis could be long-standing inflammation from infections such as tuberculosis or malignancy. Tuberculous involvement of the pleura is identified by biochemical and cytological analysis of fluid. Usually, exudative lymphocytic fluid with high adenosine deaminase level suggests the effusion to be tubercular.[10] If pleural fluid or biopsy CB-NAAT can detect MTB, we can confirm the diagnosis of tuberculosis. In our case, instead of classical tuberculosis granulomas, we got xanthogranulomatous involvement along with pleural biopsy material positivity for MTB by CB-NAAT. No evidence of any malignancy was noted in the cytological and histopathological analysis.

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.


  Conclusion Top


Xanthogranulomatous pleuritis is a rare entity with very few case reports in the literature till date. This is essentially a histopathological diagnosis. Its association with tuberculosis has not been reported before. More common organs affected include kidneys, gallbladder, and female genital tract. We report the first case of xanthogranulomatous pleuritis associated with tuberculosis, which responded successfully to antituberculosis chemotherapy.

Acknowledgment

Dr. Melcy for her efforts in taking part in care of the patient is acknowledged.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Cozzutto C, Carbone A. The xanthogranulomatous process. Xanthogranulomatous inflammation. Pathol Res Pract 1988;183:395-402.  Back to cited text no. 1
    
2.
Osterlind S. Uber pyelonephritis xanthomatosa. Acta Chir Scand. 1944;90:369–76.  Back to cited text no. 2
    
3.
Koo JS, Jung W. Xanthogranulomatous mastitis: Clinicopathology and pathological implications. Pathol Int 2009;59:234-40.  Back to cited text no. 3
    
4.
Al-Rawabdeh SM, Prasad V, King DR, Kahwash SB. Xanthogranulomatous appendicitis in a child: Report of a case and review of the literature. Case Rep Med 2013;2013:498191.  Back to cited text no. 4
    
5.
Hale MD, Roberts KJ, Hodson J, Scott N, Sheridan M, Toogood GJ. Xanthogranulomatous cholecystitis: A European and global perspective. HPB (Oxford) 2014;16:448-58.  Back to cited text no. 5
    
6.
Munichor M, Kerner H, Cohen H, Bickel A, Iancu TC. Xanthogranulomatous appendicitis--An incidental finding of localized pathology. Ultrastruct Pathol 2000;24:33-9.  Back to cited text no. 6
    
7.
Huang M, Sum R, Deshpande S, Joosten SA. Xanthogranulomatous pyelonephritis presenting as a left-sided pleural effusion. Respirol Case Rep 2018;6:e00377.  Back to cited text no. 7
    
8.
Singh RK, Pattari S. Xanthomatous pleuritis. Respir Med Case Rep 2018;23:143-4.  Back to cited text no. 8
    
9.
McGuire FR, Gourdin T, Finley JL, Downie G. Xanthomatous pleuritis mimicking mesothelioma. Respiration 2009;77:215-8.  Back to cited text no. 9
    
10.
Amer S, Hefnawy AE, Wahab NA, Okasha H, Baz A. Evaluation of different laboratory methods for rapid diagnosis of tuberculous pleurisy. Int J Mycobacteriol 2016;5:437-45.  Back to cited text no. 10
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    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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