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EXPERT VIEW
Year : 2021  |  Volume : 1  |  Issue : 1  |  Page : 28-30

Bleeding cysts in the lungs – A clinicians' dilemma!!


Department of Pulmonary Medicine, DM Wayanad Institute of Medical Sciences, Wayanad, Kerala, India

Date of Submission28-Nov-2020
Date of Decision02-Dec-2020
Date of Acceptance03-Dec-2020
Date of Web Publication28-Jan-2021

Correspondence Address:
Dr. Sanjeev Shivashankaran
Assistant Professor, Department of Pulmonary Medicine, DM Wayanad Institute of Medical Sciences, Wayanad, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jalh.jalh_10_20

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How to cite this article:
Shivashankaran S. Bleeding cysts in the lungs – A clinicians' dilemma!!. J Adv Lung Health 2021;1:28-30

How to cite this URL:
Shivashankaran S. Bleeding cysts in the lungs – A clinicians' dilemma!!. J Adv Lung Health [serial online] 2021 [cited 2021 Feb 24];1:28-30. Available from: http://www.jalh.com/text.asp?2021/1/1/28/308266



A 28-year-old unmarried female patient presented to the emergency department with a history of hemoptysis for the past 2 days. She was also having a fever. She had expectorated about 100–150 ml per episode which was 2–3 times a day. The patient had a history of recurrent episodes of hemoptysis over the past 8 years. She was treated for pulmonary tuberculosis (TB) 8 years back, but the sputum status was not known. There is no history of pneumonia in the past. There was no history of significant childhood illness. No other family member had a similar illness. The patient gave a history of massive hemoptysis 1 year back, for which she underwent bronchial artery embolization. On examination, she was a thin built, conscious patient with anemia. There is no clubbing or lymph node enlargement.

Her erythrocyte sedimentation rate was raised. Hemoglobin was 8.8 gm%. Sputum acid-fast staining was negative. Sputum culture reported growth of normal flora. Bronchoscopy did not reveal any significant abnormality. Bronchial washing was evaluated by acid-fast Bacilli smear, culture, and cytology. All were negative. X-ray of the chest [Figure 1] showed a large cystic lesion with air–fluid level occupying mid-lower zones on the left side and a small cavity with fibrosis left upper zone. The right side appeared hyperinflated and there was significant volume loss on the left side. In the hospital, the patient was managed with antibiotics, bed rest, antipyretics, and other supportive measures. Computed tomography (CT) of the abdomen was taken to rule out any cystic lesions in the liver or kidney. It was reported as normal.
Figure 1: X-ray of the chest posteroanterior showing volume loss in the left hemithorax, large cavity with fluid level occupying left mid-lower zones, and a small cavity with fibrosis left upper zone

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  Computed tomography Thorax findings – Dr. Ravindran C, Professor of Pulmonary Medicine Top


This patient was evaluated with CT of the thorax in 2015, 2019, and 2020. All the series showed almost similar findings. Mediastinal windows did not show any significant lesions including lymph node enlargement. The lesions detected in lung windows are multiple and complex. I will enumerate the predominant findings:

  1. Multiple thin walled cysts in the right upper lobes and lower lobes with normally looking surrounding lung [Figure 2] and [Figure 3]
  2. A bunch of three thick-walled cystic lesions seen in the right upper lobe toward the subpleural location
  3. Few bronchiectatic lesions are also seen in the right upper lobe [Figure 2]
  4. A small thin-walled cyst seen in the right lower lobe subpleurally [Figure 4]
  5. There is hyperinflation of the right lung [Figure 5]
  6. There is significant volume loss on the left side [Figure 2] and [Figure 3]
  7. A large thick-walled cyst or cavity is seen in the left upper lobe. The cyst extends to the pleura with pleural thickening [Figure 2] and [Figure 4]
  8. There are multiple small cysts surrounding the large cyst with fibrotic lesions [Figure 3]
  9. Few thick-walled cysts are seen subpleurally, with one of the cyst harboring a solid dense opacity with halo sign [Figure 4]
  10. Multiloculated cyst is seen in the left lower lobe with few small cysts, nodular lesions, and pleural thickening [Figure 5],[Figure 6],[Figure 7].
Figure 2: Computed tomography of the thorax - sections through the upper lobes showing a thin.walled cyst in the right upper lobe with bronchiectasis, multiloculated large cyst in the left upper lobe, subpleural cysts, and pleural thickening. Solid density with halo sign seen in one of the cysts

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Figure 3: Multiple thin-walled cysts with normal surrounding lung are seen on the right upper lobe with multiple thick-walled cysts (cavities) with fibrosis in the left upper lobe

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Figure 4: Multiple small cysts on the right side (cysts are seen in the subpleural location), large cyst with irregular wall on the left side, multiple subpleural nodules, and traction bronchiectasis

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Figure 5: High-resolution computed tomography cuts through lower lobes showing multiple thin-walled cysts right lower lobe surrounded by the normal lung and large thick-walled cyst extending to the pleura surrounded by multiple small cysts on the left side

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Figure 6: Hyperinflated right lung, large thick-walled cyst, and multiple small thick-walled cysts on the left side. There is significant volume loss on the left side

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Figure 7: Left lower lobe shows large multiloculated thick-walled cyst with surrounding small cysts and pleural thickening

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  Expert View – 1: Dr. Jayaprakash Balakrishnan, Additional Professor of Pulmonary Medicine Top


High-resolution computed tomography (HRCT), 2015 (March), shows multiple varying sized cysts predominantly in the right upper lobe with fibrotic bands. The left upper lobe shows multiple cavities, bronchiectasis, mycetoma, and volume loss. HRCT taken in 2019 shows multiple cysts in the right upper lobe. Cavities, bronchiectasis, and fibrotic bands in are noted in the left upper lobe. HRCT of April 2020 shows multiple cysts in the right upper lobe and cavities and bronchiectasis in the left upper lobe. There are few lenticular shaped cysts in the left upper lobe and a blood vessel coursing through one of the cyst.

The CT pictures suggest the possibility of a cystic? interstitial lung disease (ILD) with postinfective complications (TB). In view of the varying sized cyst with normal intervening lung, the possibility of? Lymphangioleomyomatosis (LAM) with postinfective complications (cavity, bronchiectasis, fibrosis, and mycetoma) should be the first possibility. The lenticular shape of the cyst and a blood vessel coursing sign indicate the rare possibility of Birt–Hogg–Dube (BHD) syndrome even though the cysts are usually seen in the lower lobes in BHD.


  Expert View – 2: Dr. Suraj KP, Professor of Pulmonary Medicine Top


Available sections of CT shows features suggestive of bronchiectasis in both upper lung fields, left more than right. There is evidence of fibrosis and volume reduction in the left upper part. There are cavities/thick-walled cysts on the left side. One cavity shows fluid level, may be due to infection, or retained blood. One cavity shows soft-tissue density lesion which can be due to fungal ball or blood clot. There is pleural thickening on the left upper part. There are multiple thin-walled cysts in upper and mid lung fields, more on the right side. The intervening lung appears normal. There are a few ground-glass opacities due to aspirated blood.

Possibilities

  1. Post TB sequelae – fibrocavitary lesion with a fungal ball
  2. Bronchiectasis with a fungal ball.


Congenital pulmonary airway malformation is a possibility for left lung lesion.

The presence of multiple thin-walled cysts with normal intervening lung in a young lady raises the possibility of coexisting LAM.

Selective resection of left lung segments with significant involvement may be beneficial as hemoptysis persists even after bronchial-artery embolisation. histo-pathological examination (HPE) of the resected specimen may help to confirm the rare possibility of congenital pulmonary airway malformation.

Vascular endothelial growth factor (VEGF)-D blood-based diagnostic test can distinguish LAM from other cystic lung diseases. In patients with HRCT chest suggestive of LAM, a high VEGF-D value (more than 800 pg/ml) is diagnostic for LAM. However, a normal VEGF-D level does not rule out the diagnosis of LAM.

As the CT picture is not typical of any particular cystic lung disease, she may be followed up with HRCT of the thorax and further evaluation based on imaging results. If the HRCT picture is not typical of LAM or if there is no other clinical or serological evidence, it is better to proceed with TBLB or preferably surgical lung biopsy.


  Expert View – 3: Dr. Rajendran VR, Professor of Radiodiagnosis Top


On evaluating the CT of the thorax of this young female patient, it is evident that the left upper lobe is almost destroyed with fibrocavitary changes and traction bronchiectasis. Cavity has a fungal ball on the posterior aspect. The mediastinum is shifted to left. The left pulmonary artery is small in size. There are thin-walled cysts in the right upper lobe and tiny thick-walled cysts in the middle lobe. Lung intervening the cyst appears normal. The most probable diagnosis is sequelae of recurrent childhood infections with fibrosis and bronchiectasis. However, thin-walled cysts on the right side surrounded with normally looking lung in a young female should raise the suspicion of lymphangileiomyomatosis (LAM) with secondary infection.

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.




    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]



 

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