AASM Society
Xanthogranulomatous pleuritis - An unusual presentation of tuberculosis
Jolsana Augustine1, Rajesh Venkitakrishnan1, Divya Ramachandran1, Latha Abraham2
1 Department of Pulmonary Medicine, Rajagiri Hospital, Kochi, Kerala, India
2 Department of Pathology, Rajagiri Hospital, Kochi, Kerala, India
Date of Decision 11-Aug-2020
Date of Acceptance 16-Aug-2020
Jolsana Augustine
Department of Pulmonary Medicine, Rajagiri Hospital, Aluva, Kochi, Kerala
DOI: 10.4103/ijmy.ijmy_146_20
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
Augustine J, Venkitakrishnan R, Ramachandran D, Abraham L. Xanthogranulomatous pleuritis - An unusual presentation of tuberculosis. Int J Mycobacteriol 2020;9:442-4
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
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.
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
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
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
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.
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.
Dr. Melcy for her efforts in taking part in care of the patient is acknowledged.
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Augustine J
Venkitakrishnan R
Ramachandran D
Abraham L
Granulomatous pleuritis, xanthogranulomas, xanthomatous pleuritis
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