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      Clinical and surgical aspects of congenital lobar over-inflation: a single center retrospective study

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          Abstract

          Background

          Congenital lobar overinflation (CLOI) is one of the most important causes of infantile respiratory distress (RD). We aim to evaluate our experience in CLOI management emphasizing on clinical features, diagnostic modalities, surgery and outcomes.

          Methods

          This is a retrospective study for all CLOI cases undergoing surgical management at Qena University Hospital. Demographic data, clinical data, radiographic findings, surgery and postoperative follow-up were reviewed.

          Results

          A total of 37 neonates and infants with CLOI were presented to our center between January 2015 and January 2019; their mean age was 111.43 ± 65.19 days and 22 were males. All cases presented with RD; and cyanosis in 19 cases. 15 cases presented with recurrent pneumonia and fever. Diminished breath sounds on the affected side and wheezes were the main clinical findings in 30 and 22 cases respectively. On CXR, emphysema was detected in all cases. A confirmatory CT chest was done for all cases. Left upper lobe was affected in 23 cases, right middle lobe in 7 and right upper lobe in 7 cases.

          Lobectomy was done in thirty-one cases; their mean age at surgery was 147.58 ± 81.49 days and 19 were males. Postoperative complications were noted in 5 cases and postoperative ventilation was required for 2 of them. No morbidity or mortality was reported. The follow-up duration ranged from 3 months to 1 year and all patients were doing well except one case that lost follow up after 3 months.

          Conclusion

          CLOI is a rare bronchopulmonary malformation that requires a high index of clinical suspicion, especially in persistent and recurrent infantile RD. CT chest is the most useful diagnostic modality. Early management of CLOI improves outcome and avoid life-threatening complications. Surgical management is the treatment of choice in our center without recorded mortality.

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          Most cited references41

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          Congenital anomalies of the tracheobronchial tree, lung, and mediastinum: embryology, radiology, and pathology.

          Congenital anomalies of the chest are an important cause of morbidity in infants, children, and even adults. The evaluation of affected patients frequently requires multiple imaging modalities to diagnose the anomaly and plan surgical correction. The authors analyze and illustrate practical aspects of certain common and uncommon congenital anomalies affecting the tracheobronchial tree, lung, and mediastinum, with emphasis on radiologic manifestations. Other thoracic anomalies such as rib anomalies and vascular rings are discussed when they are associated with anomalies of the tracheobronchial tree. The usefulness of the various imaging modalities in the diagnosis and treatment of these conditions is also evaluated. Specific topics addressed include tracheal conditions such as tracheal stenosis, tracheomalacia, tracheal bronchus, tracheal atresia, and bronchogenic cyst; anomalies of the lung such as lung underdevelopment (agenesis and hypoplasia), scimitar syndrome, congenital cystic adenomatoid malformation, congenital lobar emphysema, and pulmonary sequestration; esophageal anomalies such as esophageal atresia, tracheoesophageal fistula, and esophageal duplications; and vascular rings. The embryologic and pathologic basis of the radiologic findings are discussed in appropriate cases. Differential diagnoses, as well as pitfalls and diagnostic difficulties, are included. Copyright RSNA, 2003
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            Congenital lobar emphysema: diagnostic and therapeutic considerations.

            Congenital lobar emphysema (CLE) is characterized by overinflation of a pulmonary lobe and may present as a diagnostic and therapeutic dilemma. The authors have reviewed their experience to clarify the controversies about the diagnosis and management of CLE in children. Children who had CLE and were treated at our department between 1991 and 1998, inclusive, were reviewed. Fourteen children consisting of 8 boys and 6 girls (age range, 25 days to 2.5 years) had CLE. Eight children (57%) were symptomatic at birth. Major presenting symptoms were dyspnea (n = 8, 57%), cyanosis (n = 6, 43%) and recurrent respiratory tract infection (n = 4, 28.5%). Chest x-rays and computerized tomography scans showed hyperaeration of the affected lobes in 14 and 12 patients, respectively. Pulmonary perfusion scan was performed in 8 patients and showed loss of perfusion in the affected lobe in 7 cases. The affected sites were left upper lobe (n = 8, 57%), right middle lobe (n = 4, 28.5%) and right upper lobe (n = 2, 14.5%). Congenital cardiac anomalies were encountered in 2 patients (14.5%). Ten children underwent lobectomy, and postoperative course was uneventful. Nonoperative management was performed in 4 cases (28.5%) presenting at older age with milder symptoms. In the latter, although symptoms subsided, radiological abnormalities persisted during the follow-up period of 3 months to 4 years. No mortality was encountered in the series. The diagnosis of CLE is established by combined radiological and scintigraphic imaging methods. Surgical excision of the affected lobe is the appropriate treatment in all infants under 2 months of age and in infants older than 2 months presenting with severe respiratory symptoms. Infants older than 2 months presenting with mild to moderate respiratory symptoms associated with normal bronchoscopic findings can be treated conservatively. In conservative management, the children should be under close follow-up, and the family should be alarmed about the disease. Further studies are required to determine the long-term outcome of conservative management.
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              Congenital lung malformations.

              Congenital lung malformations are rare and vary widely in their clinical presentation and severity, depending mostly on the degree of lung involvement and their location in the thoracic cavity. They can manifest at any age and can be the source of significant morbidity and mortality in infants and children. Individuals with congenital lung malformations can present with respiratory symptoms at birth or can remain asymptomatic for long periods. Recently, there has been an increase in the early diagnosis of these malformations, a change that is attributable to the routine use of prenatal ultrasound. The clinical manifestation of these malformations varies from respiratory distress in the immediate postnatal period to an incidental finding on chest X-rays. Early diagnosis and prompt treatment offer the possibility of absolutely normal lung development. The treatment of asymptomatic patients with lung malformations is controversial, because the prognosis of these diseases is unpredictable. The management of these lesions depends on the type of malformation and symptoms. Because of the risk of complications, most authors recommend resection of the lesion at the time of diagnosis. Lobectomy is the procedure of choice and yields excellent long-term results. This article describes the principal congenital lung malformations, their diagnosis, and the controversies regarding treatment.
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                Author and article information

                Contributors
                Dr_abdelbary@med.svu.edu.eg
                monbasser@yahoo.com
                Ahmed.radiology@yahoo.com
                zakifm2000@yahoo.com
                khaledabdalla20@yahoo.com
                Journal
                J Cardiothorac Surg
                J Cardiothorac Surg
                Journal of Cardiothoracic Surgery
                BioMed Central (London )
                1749-8090
                19 May 2020
                19 May 2020
                2020
                : 15
                : 102
                Affiliations
                [1 ]GRID grid.412707.7, ISNI 0000 0004 0621 7833, Department of Cardiothoracic Surgery, , Qena Faculty of Medicine, South Valley University, ; Safaga Road, Qena, 83523 Egypt
                [2 ]GRID grid.252487.e, ISNI 0000 0000 8632 679X, Department of Anaesthesia and ICU, Assiut Faculty of Medicine, , Assiut University, ; Assiut, Egypt
                [3 ]GRID grid.412707.7, ISNI 0000 0004 0621 7833, Department of Radiology, Qena Faculty of Medicine, , South Valley University, ; Qena, Egypt
                [4 ]GRID grid.412659.d, ISNI 0000 0004 0621 726X, Department of Radiology, Sohag Faculty of Medicine, , Sohag University, ; Sohag, Egypt
                [5 ]GRID grid.412707.7, ISNI 0000 0004 0621 7833, Department of Pediatrics, Qena Faculty of Medicine, , South Valley University, ; Qena, Egypt
                Author information
                https://orcid.org/0000-0002-7734-9089
                https://orcid.org/0000-0002-3798-2322
                https://orcid.org/0000-0002-5761-0614
                Article
                1145
                10.1186/s13019-020-01145-8
                7236189
                32429981
                3b7dae79-f2a9-40f2-9287-d36787b8cb35
                © The Author(s) 2020

                Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.

                History
                : 27 February 2020
                : 4 May 2020
                Categories
                Research Article
                Custom metadata
                © The Author(s) 2020

                Surgery
                congenital lobar overinflation,lobectomy,neonatal respiratory distress
                Surgery
                congenital lobar overinflation, lobectomy, neonatal respiratory distress

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