Right Lung Agenesis with Tracheal Stenosis due to Complete Tracheal Rings and Postpneumonectomy Like Syndrome Treated with Tissue Expander Placement

Agrawal, Yashwant; Patri, Sandeep; Kalavakunta, Jagadeesh K

doi:10.1155/2016/4397641

Cited by 9 publications

(14 citation statements)

References 14 publications

(13 reference statements)

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“…Two cases involved patients with malignancy, one with long-standing history of Hodgkin’s lymphoma involving the left lung and mediastinum [18] and one with lung cancer [6]. Another case involved a patient with destruction of the right lung due to multiple episodes of pneumonia [19] Several cases of postpneumonectomy-like syndrome due to congenital lung agenesis or hypoplasia have been described [5, 20, 21]. Among seventy-three cases of postpneumonectomy syndrome in adults reported in the literature, the median interval between pneumonectomy and onset of symptoms was 2 years (range 1 month—49 years) [14].…”

Section: Discussionmentioning

confidence: 99%

“…Radiologic findings are usually related to bronchial obstruction and include associated atelectasis, air trapping, obstructing pneumonia and mucoid impaction [4]. Postpneumonectomy-like syndrome is a rare condition caused by either destructive or neoplastic unilateral lung disease or by congenital lung agenesis or hypoplasia, resulting in herniation of the normal lung into the diseased hemithorax with compensatory hyperinflation and air trapping in that lung [5]. Its pathogenic mechanism and treatment are analogous to those of postpneumonectomy syndrome, but without previous lung resection [6].…”

Section: Introductionmentioning

confidence: 99%

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Postpneumonectomy-like syndrome due to bronchial carcinoid: a unique case report

et al. 2019

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Background Postpneumonectomy-like syndrome is a rare condition resulting from unilateral lung disease with severe lung volume loss leading to excessive mediastinal shift and herniation of the healthy lung into the contralateral hemithorax, mimicking the mediastinal shift observed in postpneumonectomy syndrome after pneumonectomy. We report a unique case of postpneumonectomy-like syndrome caused by an atypical bronchial carcinoid completely occluding the left main bronchus. Case presentation A 25-year-old woman presented with symptoms of chronic exertional dyspnea and productive cough. Imaging studies showed complete left lung atelectasis due to a mass occluding the left main bronchus, as well as extreme mediastinal deviation and substantial herniation of the right lung into the left hemithorax. Bronchoscopic biopsy of the tumor and subsequent left pneumonectomy with concurrent lymph node dissection revealed an atypical carcinoid. Sixteen months after surgery the patient has been asymptomatic with repeat imaging studies showing no change in mediastinal shifting. Conclusion Bronchial carcinoids are notorious for causing bronchial obstruction. The present case represents an extreme complication of centrally located bronchial carcinoid, resulting in postpneumonectomy-like syndrome with severe mediastinal shift and herniation of the healthy lung into the diseased hemithorax.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Postpneumonectomy-like syndrome due to bronchial carcinoid: a unique case report

et al. 2019

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“…Most patients with right lung agenesis suffer from airway problems as a result of tracheal or bronchial compression. 2 translocation or the application of tissue expander in the empty pleural cavity), we selected aortopexy and near total thymectomy to relieve tracheal compression, as suggested by Dohlemann et al [1][2][3] As the use of tissue expanders requires some time to fill the pleural cavity, we would not have achieved a rapid response for our patient. We did not consider the use of rapid expansion of a tissue expander for fast weaning from the ventilator to be appropriate because of the risk of haemodynamical instability.…”

Section: Discussionmentioning

confidence: 99%

Aortopexy and thymectomy through lower partial sternotomy for the treatment of airway compression in a patient with right lung agenesis

2018

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“…Congenital single lung (CSL) is a rare condition, affecting about 1–3.4 per 100,000 live births [ 1 ]. Although CSL can be unilateral or bilateral, right-sided CSL is more likely to have mediastinal shift causing compression of vascular structures or airway, causing recurrent respiratory infection or respiratory distress during childhood [ 2 ].…”

Section: Introductionmentioning

confidence: 99%

Case report: a step-wise management of concurrent presentation of congenital single lung and aberrant right subclavian artery in an infant girl

Park

Janek

Hermsen

et al. 2021

J Cardiothorac Surg

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Introduction Congenital single lung (CSL) is a rare condition, and symptomatic patients often present with respiratory distress or recurrent respiratory infection due to mediastinal shift causing vascular or airway compression. Aberrant right subclavian artery (ARSA) is another rare congenital anomality that can lead to tracheal or esophageal compressions. There is only one other case of concurrent presentation of CSL and ARSA reported, which presented unique challenge in surgical management of our patient. Here we present a step-wise, multidisciplinary approach to manage symptomatic CSL and ARSA. Case presentation An infant girl with a prenatal diagnosis of CSL developed worsening stridor and several episodes of respiratory illnesses at 11 months old. Cross-sectional imaging and bronchoscopic evaluation showed moderate to severe distal tracheomalacia with anterior and posterior tracheal compression resulting from severe mediastinal rotation secondary to right-sided CSL. It was determined that her tracheal compression was mainly caused by her aortic arch wrapping around the trachea, with possible additional posterior compression of the esophagus by the ARSA. She first underwent intrathoracic tissue expander placement, which resulted in immediate improvement of tracheal compression. Two days later, she developed symptoms of dysphagia lusoria due to increased posterior compression of her esophagus by the ARSA. She underwent transposition of ARSA to the right common carotid with immediate resolution of dysphagia lusoria. As the patient grew, additional saline was added to the tissue expander due to recurrence in compressive symptoms. Conclusions Concurrent presentation of CSL and ARSA is extremely rare. Asymptomatic CSL and ARSA do not require surgical interventions. However, if symptomatic, it is crucial to involve a multidisciplinary team for surgical planning and to take a step-wise approach as we were able to recognize and address both tracheomalacia and dysphagia lusoria in our patient promptly.

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Right Lung Agenesis with Tracheal Stenosis due to Complete Tracheal Rings and Postpneumonectomy Like Syndrome Treated with Tissue Expander Placement

Cited by 9 publications

References 14 publications

Postpneumonectomy-like syndrome due to bronchial carcinoid: a unique case report

Postpneumonectomy-like syndrome due to bronchial carcinoid: a unique case report

Aortopexy and thymectomy through lower partial sternotomy for the treatment of airway compression in a patient with right lung agenesis

Case report: a step-wise management of concurrent presentation of congenital single lung and aberrant right subclavian artery in an infant girl

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