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Congenital lobar emphysema presenting at late childhood: A rare case report.

Santra A, Dutta P, Manjhi R, Pothal S - Lung India (2014)

View Article: PubMed Central - PubMed

Affiliation: Critical Care Unit, Fortis Hospital, Anandapur, Kolkata, India.

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Sir, Congenital lobar emphysema (CLE) is a rare congenital abnormality that is characterized by overdistension and air trapping of one or more lobes of lung, leading to a compression atelectasis of ipsilateral lung and subsequently of contralateral lung... A 15-year old boy presented with progressive exertional breathlessness since 5 months... There was no other significant history... The left lung was normal except for compression atelectasis... No evidence of pneumothorax or intraluminal foreign body was found... Patients who present later in life usually have history of recurrent chest infections and frequent antibiotic use, which was not the scenario in this case... Exact aetiology is not known and no apparent cause is found in 50% cases... Lobectomy is the standard surgical procedure that allows expansion of compressed normal lung tissue... Long-term outcome was found to be excellent with complete cure in over 85% of cases... Recently endoscopic parenchymal sparing resection has been performed in a case of CLE with mild symptoms... Also right middle and lower lobectomy was unlikely to produce any improvement in our patient as he had hypoplastic right upper lobe bronchus and atelectasis of right upper lobe was not due to compression... In conclusion, CLE presenting in late childhood or adulthood for the first time may lead to a diagnostic challenge to the physician and it can be detected only if a high diagnostic suspicion is maintained.

No MeSH data available.


Chest X-ray shows hyperlucent and hyperexpanded right lung with mediastinal shifting to the left side along with attenuated but maintained vascularity of the right lung
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Figure 1: Chest X-ray shows hyperlucent and hyperexpanded right lung with mediastinal shifting to the left side along with attenuated but maintained vascularity of the right lung

Mentions: But chest X-ray in PA view [Figure 1] revealed hyperlucency of the entire right lung with leftward mediastinal shift without any visible visceral pleural lining. Attenuated but maintained vascularity was noted in the hyperlucent right lung. ABG analysis and other blood investigations were normal except Hb-10.4 gm/dl. Electrocardiography showed only left axis deviation without any evidence of left ventricular hypertrophy. Two-dimensional echocardiography did not reveal any cardiac abnormality. Spirometry showed a mixed ventilatory pattern with poor bronchodilator reversibility. Computed tomographic scan of thorax [Figure 2] showed hyperlucent and hyperexpanded right middle and lower lobe with collapsed right upper lobe along with mediastinal shift to left. Pulmonary vasculature on the right side appeared attenuated and displaced. Herniation of lung tissue was noted in retrocardiac space and in anterior mediastinum. The left lung was normal except for compression atelectasis. No evidence of pneumothorax or intraluminal foreign body was found. Subsequent fibreoptic bronchoscopy [Figure 3] showed a hypoplastic right upper lobe bronchus along with a normal trachea and left main bronchus.


Congenital lobar emphysema presenting at late childhood: A rare case report.

Santra A, Dutta P, Manjhi R, Pothal S - Lung India (2014)

Chest X-ray shows hyperlucent and hyperexpanded right lung with mediastinal shifting to the left side along with attenuated but maintained vascularity of the right lung
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4129613&req=5

Figure 1: Chest X-ray shows hyperlucent and hyperexpanded right lung with mediastinal shifting to the left side along with attenuated but maintained vascularity of the right lung
Mentions: But chest X-ray in PA view [Figure 1] revealed hyperlucency of the entire right lung with leftward mediastinal shift without any visible visceral pleural lining. Attenuated but maintained vascularity was noted in the hyperlucent right lung. ABG analysis and other blood investigations were normal except Hb-10.4 gm/dl. Electrocardiography showed only left axis deviation without any evidence of left ventricular hypertrophy. Two-dimensional echocardiography did not reveal any cardiac abnormality. Spirometry showed a mixed ventilatory pattern with poor bronchodilator reversibility. Computed tomographic scan of thorax [Figure 2] showed hyperlucent and hyperexpanded right middle and lower lobe with collapsed right upper lobe along with mediastinal shift to left. Pulmonary vasculature on the right side appeared attenuated and displaced. Herniation of lung tissue was noted in retrocardiac space and in anterior mediastinum. The left lung was normal except for compression atelectasis. No evidence of pneumothorax or intraluminal foreign body was found. Subsequent fibreoptic bronchoscopy [Figure 3] showed a hypoplastic right upper lobe bronchus along with a normal trachea and left main bronchus.

View Article: PubMed Central - PubMed

Affiliation: Critical Care Unit, Fortis Hospital, Anandapur, Kolkata, India.

AUTOMATICALLY GENERATED EXCERPT
Please rate it.

Sir, Congenital lobar emphysema (CLE) is a rare congenital abnormality that is characterized by overdistension and air trapping of one or more lobes of lung, leading to a compression atelectasis of ipsilateral lung and subsequently of contralateral lung... A 15-year old boy presented with progressive exertional breathlessness since 5 months... There was no other significant history... The left lung was normal except for compression atelectasis... No evidence of pneumothorax or intraluminal foreign body was found... Patients who present later in life usually have history of recurrent chest infections and frequent antibiotic use, which was not the scenario in this case... Exact aetiology is not known and no apparent cause is found in 50% cases... Lobectomy is the standard surgical procedure that allows expansion of compressed normal lung tissue... Long-term outcome was found to be excellent with complete cure in over 85% of cases... Recently endoscopic parenchymal sparing resection has been performed in a case of CLE with mild symptoms... Also right middle and lower lobectomy was unlikely to produce any improvement in our patient as he had hypoplastic right upper lobe bronchus and atelectasis of right upper lobe was not due to compression... In conclusion, CLE presenting in late childhood or adulthood for the first time may lead to a diagnostic challenge to the physician and it can be detected only if a high diagnostic suspicion is maintained.

No MeSH data available.