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CASE REPORT
Year : 2016  |  Volume : 30  |  Issue : 2  |  Page : 102-104

A case of polyserositis in a 56-year-old female patient


Pulmonary Medicine, R N Tagore Hospital, Kolkata, West Bengal, India

Date of Web Publication5-Dec-2016

Correspondence Address:
Aratrika T Das
188/11, Prince Anwar Shah Road, Kolkata - 700 045, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-6691.195267

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  Abstract 

Polyserositis is defined as general inflammation of serous membranes associated with serous effusion due to many causes. In this case, we report polyserositis as a complication of connective tissue disorder. A 56-year-old female with a history of hypertension, hypothyroidism, and bipolar disorder having dyspnea along with pleural effusion which was repeatedly tapped and treated on suspicion of tuberculosis. She became better on steroid treatment and her respiratory distress resolved.

Keywords: Ascites, Concato′s disease, pericardial effusion, pleural effusion, polyserositis


How to cite this article:
Das AT. A case of polyserositis in a 56-year-old female patient. Indian J Allergy Asthma Immunol 2016;30:102-4

How to cite this URL:
Das AT. A case of polyserositis in a 56-year-old female patient. Indian J Allergy Asthma Immunol [serial online] 2016 [cited 2017 Mar 27];30:102-4. Available from: http://www.ijaai.in/text.asp?2016/30/2/102/195267


  Introduction Top


Polyserositis is defined as chronic inflammation of several serous membranes with effusions in serous cavities, [1] resulting in fibrous thickening of the serous membranes and sometimes constrictive pericarditis. This illness is also called Concato's disease. [2]

Polyserositis can be a syndrome within an underlying disease (tuberculosis [TB], systemic lupus erythematosus [SLE], asbestosis, silicosis) or can be a complication of chest surgery, radiation therapy, or drug usage, and very rarely, it may be idiopathic. Herein, we present a case of polyserositis probably secondary to mixed connective tissue disorder (MCTD).


  Case Report Top


Mrs. MM, 56-year-old female, postmenopausal, nonsmoker, nonalcoholic, homemaker presented to our outpatient department in November 2010 with a history of recurrent left-sided pleural effusion large enough to cause respiratory distress and require thoracocentesis (pleurocentesis).

Previously, the patient was investigated in a multispecialty hospital in July 2010 where she was found to have high triglyceride (233 mg/dl), urea (70.70 mg/dl), serum transaminases (serum glutamate-oxaloacetate transaminase -193, serum glutamate-pyruvate transaminase -146 U/L). Her echocardiography showed normal findings, except concentric left ventricular hypertrophy.

She was a known case of hypertension, hypothyroidism and was suffering from bipolar disorder, for which she was on regular medication.

She became symptomatic of dyspnea on exertion, panniculitis, and mild fever of 1-day duration in August 2010. However, she did not have a history of significant cough or loss of weight or anorexia. The patient underwent routine blood test on August 5, 2010, which revealed anemia (hemoglobin 10.1 g/dl) and raised erythrocyte sedimentation rate (55 mm) and her chest X-ray (CXR) on August 27, 2010, showed left-sided pleural effusion.

She was put on antithyroid drug (ATD) on September 22, 2010, by her family physician and was asked to consult a pulmonologist.

Subsequently, therapeutic aspiration followed by a pleural fluid examination revealed it to be having high protein level and very low lactate dehydrogenase with lymphocyte predominance and adenosine deaminase of <30. After 2 months of ATD therapy, her breathlessness worsened and a repeat CXR revealed a large effusion again on the left side [Figure 1]. She was then hospitalized for further investigations and treatment. She however did complain of pain in both knee joints on direct questioning. A contrast-enhanced computed tomography (CT) scan on September 6, 2010, of the chest revealed massive to moderate left-sided pleural effusion with mediastinal shift and enlarged anterior mediastinal space-occupying lesion (SOL) which was probably lymph node swelling [Figure 2] and [Figure 3]. Although repeat CT scans on November and December 2010 did not suggest the presence of any such SOL. Pleural effusion was transudative (serous) in nature and revealed mainly predominance of lymphocyte with a few neutrophils and many mesothelial cells on microscopic examination on several occasions. Pleural fluid was never positive for acid-fast Bacilli (AFB) and malignancy. During this period, only positive finding was positive rheumatoid arthritis factor repeated twice. A pleural biopsy with Cope's needle did not prove helpful. A bronchoalveolar lavage showed a normal cytology and no growth of AFB on culture. She was a known case of bipolar disorder on lithium and nexito. Her cardiac assessment was normal.
Figure 1: Chest X-ray of the patient

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Figure 2: Computed tomography scan of the patient

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Figure 3: Computed tomography scan of the patient

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At the time when this patient presented to us, she had bilateral pleural effusion, a mild pericardial effusion, edema, and ascites. Her cardiac assessment was normal. Positive laboratory findings at this stage were positive antinuclear antibodies and positive anti-Sm (U1) ribonucleoprotein. Hence, we could not conclude as to why the polyserositis occurred. A diagnosis of MCTD was high on the cards; hence, to confirm a diagnosis, we decided to go ahead with a visceral adipose tissue biopsy of visceral and parietal pleura. The biopsy revealed presence of fibrosed tissue amid pleural tissue. A provisional diagnosis of idiopathic fibrosing polyserositis was made as clinically no sign of MCTD was found. She was put on oral corticosteroids and colchicine along with methotrexate. Colchicine was given for 3 months and the steroid was tapered off over 6 months. The patient responded well, her pleural effusions resolved, and she remained healthy for 2 years of follow-up.


  Discussion Top


A large number of cases of polyserositis discussed by various authors in the literature describe sequel of constrictive pericarditis secondary to TB or a sequel of connective tissue disease, especially SLE. However, in our case, all investigations for TB or connective tissue disorder proved negative. However, the diametric response to steroids along with positive MCTD marker with disappearance of effusions and ascites could be a pointer toward MCTD or idiopathic fibrosing polyserositis.


  Conclusion Top


To conclude treatment of MCTD, 80% respond to steroids therapy. In refractory cases, other forms of immunosuppressant such as mycophenolic acid and tamoxifen have been found to be useful. Surgical intervention is considered in patients who have multifocal fibrosclerosis or extensive fibrosing mediastinitis or those who do not respond immunosuppressant therapy. Recurrences are unpredictable, so long-term follow-up is recommended. Sometimes, recurrences can occur up to 10 years after initial diagnosis.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Dorland WA. Dorland's Illustrated Medical Dictionary. 26 th ed. Philadelphia, London: W.B. Saunders and Co.; 1981. p. 1051.  Back to cited text no. 1
    
2.
Thomas EA, Charles CJ. Cecil Essential of Medicine. 5 th ed. Philadelphia: Saunders; 2001. p. 768.  Back to cited text no. 2
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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