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AN UNUSUAL CASE OF A PLEURAL EFFUSION WITH AN ABDOMINAL MASS

Preetam R Acharya1, Rameshchandra Sahoo2 and Poornima Baliga3

1. Assistant Professor 2. Professor & Head 3. ProfessorDepartment of Tuberculosis and Respiratory Diseases, Kasturba Medical College & Hospital, Attavara, Mangalore (Karnataka).Correspondence: Dr. Preetam R. Acharya, Department of Tuberculosis & Respiratory Diseases, Kasturba Medical College & Hospital, N. G. Road,Attavara, Mangalore – 575 001 (Karnataka); E-mail: dokpreet@yahoo.co.in; Telephone (land): (0824) 2445858; Telephone (mobile): 9448953430;Fax: (0824) 2428379

(Received on 10.12.2007. Accepted after revision on 22.4.2008)

Summary: A 38-year old man presented to us with a left sided pleural effusion. Pleural fluid was aspirated and analysisrevealed it to be an exudate with predominant lymphocytes and an elevated ADA level. He was discharged on anti-tuberculous treatment. Patient returned with re-accumulation of pleural fluid. Computed tomography done in ourinstitute picked up not only parenchymal disease in the lung which was not evident on chest radiographs but also pickedup an abdominal mass in the left renal fossa. Pathological examination of excised mass revealed its tuberculous nature.The repeated recollection of pleural fluid was attributed to a “paradoxical response”; the patient was reassured and hisanti-tuberculous treatment continued. Recognition of the fact that evidence of tuberculosis at distant sites may occasionallybe needed to substantiate the diagnosis of tuberculous pleural effusion in a difficult and bacteriologically “negative” caseprompted us to report this case.

Key words: Tuberculous Pleural Effusion, Paradoxical Response, Incidentaloma

[Indian J Tuberc 2008; 55: 153-156]

INTRODUCTION

Establishing Tuberculosis as the cause ofpleural effusion can be difficult at times. Thediagnosis of Tuberculous Pleuritis is commonlymade from observation of granulomas in pleuralbiopsy specimens or a culture positive forMycobacterium Tuberculosis from pleural tissue orpleural fluid. However, the diagnosis can beuncertain or missed in “bacteriologically negative”cases. Proof of co-existing pulmonary tuberculosisor getting evidence of the presence ofmycobacterium at other extra-pulmonary sites mayindicate dissemination and establish sufficientgrounds for institution of anti-tuberculous treatmentin an otherwise microbiologically ½ negative½ case.

CASE REPORT

A-38-year old man presented to us withleft sided pleuritic chest pain and exertionalbreathlessness (Medical Research Council GradeI) for last seven days. He also had cough withminimal expectoration for a month. He deniedhaving had haemoptysis, fever, chills or night

sweats. On review of systems, the patientreported that he had lost some weight since lastone month. He had no past history of respiratorydisease and was a non-smoker. Family history forpulmonary tuberculosis was positive in an eldersibling who had completed a course of anti-tuberculous treatment 15 years back.

Respiratory system examination wassuggestive of a left sided pleural effusion. Chestradiograph revealed a moderate left sided pleuraleffusion with contra lateral tracheal shift and noevidence of any parenchymal abnormalities (Fig.1).Routine blood investigations like haemogram, ESR,fasting blood glucose, total bilirubin, transaminasesand creatinine were all within normal limits. Humanimmuno-deficiency virus (HIV) ELISA was negative.Three samples of sputum examined for AFB by Ziehl-Neelsen method were negative. Thoracentesis wasdone and serosanguinous fluid aspirated. Pleural fluidwas an exudate (Protein: 7.1 gm/dl) with a leukocytecount of 4,800 cells/cumm and a lymphocytedifferential of 95 %. Pleural fluid examination byGram stain and ZN stain were negative. AFB cultureof the pleural fluid sample did not show any

Case Report

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mycobacterial colonies at the end of eight weeks ofincubation. Cytological examination of pleural fluidwas negative for malignant cells. Pleural fluidadenosine deaminase (ADA) levels were elevated(patient value: 122.61 iu/L, normal: up to 36.0iu/L). A provisional diagnosis of tuberculous pleuraleffusion was made and the patient discharged ona four drug antituberculous regimen consistingof Isoniazid, Rifampicin, Ethambutol andPyrazineamide (HERZ).

Over a period of next four weeks, ourpatient did not show any improvement. A chestradiograph taken at follow-up at this point showedre-accumulation of pleural fluid. A closed pleuralbiopsy was inconclusive. Pleural fluidcarcinoembryonic antigen (CEA) was normal (1.7).Pleural fluid was re-aspirated and a contrastenhanced computed tomography of chest wasadvised. This revealed calcified nodular opacities inapices of both the lungs. Additionally, a hypo dense

Fig. 1: Chest radiograph showing left sided effusionFig. 3: Chest radiograph at treatment end.

Fig. 2: Follow-up chest radiograph at 12 weeks

Fig. 4: Image showing granulomas consisting ofepitheloid cells and Langhan’s giant cells(H & E X10)

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enhancing mass lesion with a few areas of hyperintensity was seen to arise from the upper pole ofthe left kidney. CT guided biopsy and FNAC wasdone from the mass which showed only necroticmaterial.

Because of the rapid re-accumulation ofpleural fluid despite regular anti-tuberculoustreatment, a decision was taken to surgicallyexplore the abdomen and excise the mass to ruleout a malignant etiology. Surgical explorationrevealed a pale brown mass measuringapproximately 8×7×4 cms not involving thekidney. The mass was excised in its entirety.Histopathological examination revealed caseatinggranulomas consisting of epitheloid cells,Langhan’s giant cells and mature lymphocytessuggesting tuberculosis (Fig.4).

The presence of caseating granulomas inthe excised mass in addition to the pleural fluidcharacteristics helped us to substantiate our initialdiagnosis of tuberculous pleural effusion. Patientwas asked to continue his anti-tuberculous drugs.Follow-up at regular intervals and serial chestradiographs (Fig.2, Fig.3) have not revealed anyfurther re-accumulation of pleural fluid.

DISCUSSION

Pleural effusions can occur in any formof pulmonary tuberculosis. It is a well-known factthat neither the clinical features nor any of theimaging modalities are of much help in thediagnosis of a tuberculous pleural effusion. Co-existing parenchymal disease detectedradiographically in about one third of the patientswith an effusion serves as a marker of activepulmonary tuberculosis. Computed tomographyof chest may show lymphadenopathy, pulmonaryinfi l trates or cavitation not seen on chestradiographs; which although non-specific, mayhelp to distinguish tuberculous pleurisy from othercauses like malignancy1.

The onus of proving tuberculosis as a causeof pleural effusion rests on microbiological (smear/culture), and histological analysis of aspirated pleural

fluid and biopsied pleural tissue. Mycobacteria areseen in pleural fluid only in 10 - 20 % of caseswhereas a culture though positive in 25 - 50 % ofthe cases takes a minimum of 6-8 weeks byconventional methods to be of any clinical utility.Pleural biopsy will show granulomas in the parietalpleura in 50 – 80 % of patients and if cultured willgrow mycobacterium in 55 – 80% of the cases2.High levels of pleural fluid ADA (The reported cut-off value for PADA varies from 47 to 60 U/l) maybe helpful in distinguishing tuberculous effusionsfrom malignant effusions. Several reports havesuggested that an elevated pleural fluid ADA levelpredicts tuberculous pleuritis with a sensitivity of90 to 100% and a specificity of 89 to 100%3.However, controversy surrounds the use of elevatedpleural fluid ADA as a diagnostic aid for tuberculouseffusion4.

Treatment with anti-tuberculous drugs isthe mainstay of management of extra pulmonarytuberculosis. The term “paradoxical response” refersto enlargement of old lesions or unexpectedappearance of new lesions during anti-tuberculoustherapy. Re-crudescence of fever, enlargingadenopathies, worsening of pulmonary infiltrates,pleural effusion, ascites and appearance ofintracranial tuberculomas have all been described.An incidence of 16% of paradoxical worsening oftuberculous effusion following the start of anti-tuberculous treatment has been observed5,6. Such aparadoxical worsening can occur in both HIVuninfected and infected starting TB therapy. Thesyndrome poses a diagnostic challenge since theapparent clinical deterioration may raise the suspicionof drug resistant tuberculosis, non- compliance toprescribed regimen or presence of a concomitantdisorder unrelated to TB. These patients generallyneed no alteration in their drug regimen.

An incidentaloma is a tumor (-oma) foundby coincidence (- incidental). The widespread useof ultrasound, CT and MRI has resulted in theincidental discovery of asymptomatic adrenalmasses. Adrenal incidentaloma is not a singlepathological entity and the differential diagnosisincludes adenoma, adrenocortical carcinoma,phaeochromocytoma, metastases, Cushing’s

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syndrome, primary aldosteronism etc.The incidentaladrenal masses may also be infiltrative disease, fungaland tuberculous granulomas , hemorrhage and lesionsthat masquerade as adrenal but arise from adjacentorgans (e.g. kidneys, pancreas, gall bladder, spleen,lymph nodes). The adrenal gland is also a commonsite for metastases from the breast, lung, renal,melanoma, lymphoma7. Tuberculosis may affect manyof the endocrine glands the commonest being theadrenal gland; acute tuberculosis adrenalitis producingmass-like enlargement of the gland8. Ultrasonographyand computed tomography do not always differentiatebetween adrenal and extra-adrenal masses and betweenmalignancy and non-malignancy; surgical excisiontherefore seems to be desirable in such cases9.

In our patient, CT thorax was requestedfor purpose of evaluating lung parenchymalabnormalities. The upper abdominal cuts in the CThelped us to pick up the incidental adrenal mass.Unfortunately, transcutaneous needle biopsy ofadrenal mass proved to be inconclusive promptingus to subject the patient to exploratory laparatomywith excision of the mass in its entirety. Histologicalexamination of the excised mass showed featuressuggesting tuberculosis. Considering the pleuralfluid characteristics i.e. exudate withlymphocytic predominance and a high pleuralfluid ADA levels, the diagnosis of tuberculouspleural effusion was substantiated. Inretrospect, the increase in effusion size onanti-tuberculous treatment may be attributedto Immune reconstitution syndrome – asyndrome of paradoxical worsening known to

occur during treatment with ATT. Ourassumption is strengthened by the fact thatthe patient has completed treatment and hashad no recurrence of the effusion on follow-up.

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