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Yu H, Kim H, Cheong H. Extralobar Pulmonary Sequestration: A Case of Incidental Autopsy Diagnosis. Am J Forensic Med Pathol 2024; 45:e94-e96. [PMID: 38228310 DOI: 10.1097/paf.0000000000000894] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/18/2024]
Affiliation(s)
| | | | - Harin Cheong
- From the Department of Forensic Medicine, College of Medicine, The Catholic University of Korea, Seoul
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2
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Wang YJ, Chen YY, Lin GH. Relationship between intralobar pulmonary sequestration and type A aortic dissection: A case report. World J Clin Cases 2023; 11:3658-3663. [PMID: 37383900 PMCID: PMC10294189 DOI: 10.12998/wjcc.v11.i15.3658] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/06/2023] [Revised: 03/26/2023] [Accepted: 04/17/2023] [Indexed: 05/25/2023] Open
Abstract
BACKGROUND Pulmonary sequestrations often lead to serious complications such as infections, tuberculosis, fatal hemoptysis, cardiovascular problems, and even malignant degeneration, but it is rarely documented with medium and large vessel vasculitis, which is likely to result in acute aortic syndromes.
CASE SUMMARY A 44-year-old man with a history of acute Stanford type A aortic dissection status post-reconstructive surgery five years ago. The contrast-enhanced computed tomography of the chest at that time had also revealed an intralobar pulmonary sequestration in the left lower lung region, and the angiography also presented perivascular changes with mild mural thickening and wall enhancement, which indicated mild vasculitis. The intralobar pulmonary sequestration in the left lower lung region was long-term unprocessed, which was probably associated with his intermittent chest tightness since no specific medical findings were detected but only positive sputum culture with mycobacterium avium-intracellular complex and Aspergillus. We performed uniportal video-assisted thoracoscopic surgery with wedge resection of the left lower lung. Hypervascularity over the parietal pleura, engorgement of the bronchus due to a moderate amount of mucus, and firm adhesion of the lesion to the thoracic aorta were histopathologically noticed.
CONCLUSION We hypothesized that a long-term pulmonary sequestration-related bacterial or fungal infection can result in focal infectious aortitis gradually, which may threateningly aggravate the formation of aortic dissection.
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Affiliation(s)
- Yi-Jie Wang
- Department of Surgery, Tri Service General Hospital, National Defense Medical Center, Taipei 114, Taiwan
| | - Ying-Yi Chen
- Division of Thoracic Surgery, Department of Surgery, Tri-Service General Hospital, National Defense Medical Center, Taipei 114, Taiwan
| | - Gang-Hua Lin
- Division of General Surgery, Department of Surgery, Tri-Service General Hospital, National Defense Medical Center, Taipei 114, Taiwan
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3
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Affiliation(s)
- Z Zeng
- Department of Thoracic Surgery, West China Hospital, Sichuan University, No. 37, Guoxue Alley, Chengdu, Sichuan 610041, China and
- Western China Collaborative Innovation Center for Early Diagnosis and Multidisciplinary Therapy of Lung Cancer, Sichuan University, No. 37, Guoxue Alley, Chengdu 610041, China
| | - Y Zhu
- Department of Thoracic Surgery, West China Hospital, Sichuan University, No. 37, Guoxue Alley, Chengdu, Sichuan 610041, China and
- Western China Collaborative Innovation Center for Early Diagnosis and Multidisciplinary Therapy of Lung Cancer, Sichuan University, No. 37, Guoxue Alley, Chengdu 610041, China
| | - C Liu
- Department of Thoracic Surgery, West China Hospital, Sichuan University, No. 37, Guoxue Alley, Chengdu, Sichuan 610041, China and
- Western China Collaborative Innovation Center for Early Diagnosis and Multidisciplinary Therapy of Lung Cancer, Sichuan University, No. 37, Guoxue Alley, Chengdu 610041, China
| | - F Lin
- Department of Thoracic Surgery, West China Hospital, Sichuan University, No. 37, Guoxue Alley, Chengdu, Sichuan 610041, China and
- Western China Collaborative Innovation Center for Early Diagnosis and Multidisciplinary Therapy of Lung Cancer, Sichuan University, No. 37, Guoxue Alley, Chengdu 610041, China
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4
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Motohashi Y, Kato T, Kato N, Aragaki M, Fujiwara-Kuroda A, Tachibana T, Hida Y, Kaga K, Wakasa S. Intralobar pulmonary sequestration associated with left main coronary artery obstruction and mitral regurgitation. Gen Thorac Cardiovasc Surg 2021; 69:1575-1579. [PMID: 34546531 DOI: 10.1007/s11748-021-01708-z] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/31/2021] [Accepted: 09/04/2021] [Indexed: 10/20/2022]
Abstract
A 4-year-old boy with left intralobar pulmonary sequestration associated with left main coronary artery obstruction (LMCAO) and severe mitral regurgitation (MR) was admitted to our hospital. Since the patient presented with dyskinesia of the cardiac apex and increased left ventricular end-diastolic volume (LVEDV), left main coronary artery reconstruction and mitral annuloplasty were performed. The enlargement of the left ventricle was improved after sequential surgeries. There was a risk of deterioration of MR and regrowth of LVEDV due to shunt blood flow; therefore, left lower lobectomy and aberrant artery division were performed. This is a very rare case of a patient with pulmonary sequestration associated with LMCAO and severe MR.
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Affiliation(s)
- Yusuke Motohashi
- Department of Cardiovascular and Thoracic Surgery, Hokkaido University Graduate School of Medicine, N15W7, Kita-ku, Sapporo, Hokkaido, 060-8638, Japan
| | - Tatsuya Kato
- Department of Cardiovascular and Thoracic Surgery, Hokkaido University Graduate School of Medicine, N15W7, Kita-ku, Sapporo, Hokkaido, 060-8638, Japan.
| | - Nobuyasu Kato
- Department of Cardiovascular and Thoracic Surgery, Hokkaido University Graduate School of Medicine, N15W7, Kita-ku, Sapporo, Hokkaido, 060-8638, Japan
| | - Masato Aragaki
- Department of Cardiovascular and Thoracic Surgery, Hokkaido University Graduate School of Medicine, N15W7, Kita-ku, Sapporo, Hokkaido, 060-8638, Japan
| | - Aki Fujiwara-Kuroda
- Department of Cardiovascular and Thoracic Surgery, Hokkaido University Graduate School of Medicine, N15W7, Kita-ku, Sapporo, Hokkaido, 060-8638, Japan
| | - Tsuyoshi Tachibana
- Department of Cardiovascular and Thoracic Surgery, Hokkaido University Graduate School of Medicine, N15W7, Kita-ku, Sapporo, Hokkaido, 060-8638, Japan
| | - Yasuhiro Hida
- Department of Cardiovascular and Thoracic Surgery, Hokkaido University Graduate School of Medicine, N15W7, Kita-ku, Sapporo, Hokkaido, 060-8638, Japan
| | - Kichizo Kaga
- Department of Cardiovascular and Thoracic Surgery, Hokkaido University Graduate School of Medicine, N15W7, Kita-ku, Sapporo, Hokkaido, 060-8638, Japan
| | - Satoru Wakasa
- Department of Cardiovascular and Thoracic Surgery, Hokkaido University Graduate School of Medicine, N15W7, Kita-ku, Sapporo, Hokkaido, 060-8638, Japan
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5
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Sun Y, Shao F, Zhang Q, Wang Z. Feasibility investigation of uniportal video-assisted thoracoscopic anatomical lung resection for pulmonary sequestration. J Cardiothorac Surg 2020; 15:93. [PMID: 32404207 PMCID: PMC7222582 DOI: 10.1186/s13019-020-01126-x] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/10/2020] [Accepted: 04/28/2020] [Indexed: 11/23/2022] Open
Abstract
Background Uniportal video-assisted thoracic surgery (UVATS) technique has been increasingly used for many thoracic diseases. Whether UVATS has equivalent or better perioperative outcomes for pulmonary sequestration (PS) patients remains controversial. Our study aimed to evaluate the feasibility of UVATS in anatomical lung resection for pulmonary sequestration. Methods A total of 24 patients with PS including fifteen males and nine females with the mean age of 40 (range, 18–65) years old, who had received completely UVATS anatomical lung resection for PS in Nanjing Chest Hospital between January 2016 and December 2018 were retrospectively reviewed. Related clinical data were retrieved from hospital records and analyzed. Results All 24 patients had been treated with the UAVTS approach successfully without aberrant artery ruptured or massive hemorrhage, and no patients died during the perioperative period. Overall mean surgery time was 102 mins (range, 55–150 min), the mean blood loss was 94 ml (range, 10-300 ml), the mean days of chest tube maintained were 4 days (range,1-10 days), and the mean postoperative hospitalization days was 6 days (range,2-11 days). All patients were cured, without cough, fever, hemoptysis, and so on, associated with PS, occurring during the average follow-up of 17 months (range, 3-35 months). Conclusions Our preliminary results revealed that anatomical lung resection by UVATS is a safe and feasible mini-invasive technique for PS patients, which might be associated with less postoperative pain, reduced paresthesia, better cosmetic results, and faster recovery.
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Affiliation(s)
- Yungang Sun
- Department of Thoracic Surgery, Nanjing Chest Hospital, Nanjing Brain Hospital Affiliated to Nanjing Medical University, 264 Guangzhou Road, Nanjing, 210029, China.,Pulmonary Nodule Diagnosis and Treatment Research Center, Nanjing Medical University, Nanjing, China
| | - Feng Shao
- Department of Thoracic Surgery, Nanjing Chest Hospital, Nanjing Brain Hospital Affiliated to Nanjing Medical University, 264 Guangzhou Road, Nanjing, 210029, China. .,Pulmonary Nodule Diagnosis and Treatment Research Center, Nanjing Medical University, Nanjing, China.
| | - Qiang Zhang
- Department of Thoracic Surgery, Nanjing Chest Hospital, Nanjing Brain Hospital Affiliated to Nanjing Medical University, 264 Guangzhou Road, Nanjing, 210029, China.,Pulmonary Nodule Diagnosis and Treatment Research Center, Nanjing Medical University, Nanjing, China
| | - Zhao Wang
- Department of Thoracic Surgery, Nanjing Chest Hospital, Nanjing Brain Hospital Affiliated to Nanjing Medical University, 264 Guangzhou Road, Nanjing, 210029, China.,Pulmonary Nodule Diagnosis and Treatment Research Center, Nanjing Medical University, Nanjing, China
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6
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Sha JM, Zhao H, Lin ZB. Anomalous Systemic Arterial Supply to the Lung: To Which Category Should This Belong? Heart Lung Circ 2020; 29:1292-1300. [PMID: 32349946 DOI: 10.1016/j.hlc.2019.08.009] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/17/2019] [Revised: 06/23/2019] [Accepted: 08/04/2019] [Indexed: 10/26/2022]
Abstract
BACKGROUND The nomenclature of both intralobar pulmonary sequestration (ILS) and aortic origin of a pulmonary artery (AOPA) remains controversial. According to this review, both ILS and AOPA have an anomalous systemic arterial supply to all or part of the lung with venous drainage into the pulmonary veins, which leads to pulmonary hypertension, congestive heart failure, and fatal pulmonary haemorrhage. The purpose of this review was to consider whether these two rare congenital anomalies have similar anatomical, clinical and pathological characteristics. METHODS This review was conducted by researching relevant literature using PubMed and MEDLINE databases to January 2019. All researched literature was related to the anatomical, associated anomalies, pathophysiology and clinical features of the extralobar pulmonary sequestration (ELS), ILS, and AOPA, and the therapeutic method for ILS and AOPA. RESULTS Through research literature, it was found that ILS and AOPA may differ in terms of embryonic origin, but some of the anatomical, histopathological, physiological and clinical features of these two congenital malformations are similar. However, ELS and ILS have significant differences in their anatomical, histopathological, physiological, and clinical features. CONCLUSIONS This study proposes that ILS and AOPA could be classified as one single condition - systemic arterialisation of the lung - and further divided into three subtypes, namely: types I, II and III. This new classification nomenclature permits the appropriate change of novel surgical techniques, which obviate the need for lobectomy or segmentectomy in specific cases, thereby minimising fatal postoperative complications.
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Affiliation(s)
- Ji-Ming Sha
- Department of Cardiothoracic Surgery, The Second Affiliated Hospital of Anhui Medical University, Anhui, China.
| | - Hui Zhao
- Department of Respiratory Medicine, The Second Affiliated Hospital of Anhui Medical University, Anhui, China
| | - Ze-Bang Lin
- Department of Cardiothoracic Surgery, The Second Affiliated Hospital of Anhui Medical University, Anhui, China
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7
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Sergi CM. Lower Respiratory Tract. PATHOLOGY OF CHILDHOOD AND ADOLESCENCE 2020:139-253. [DOI: 10.1007/978-3-662-59169-7_2] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 08/30/2023]
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8
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Schierz IAM, Giuffrè M, Piro E, Leone MC, Pinello G, Corsello G. Clinical cardiac assessment in newborns with prenatally diagnosed intrathoracic masses. Ital J Pediatr 2018; 44:98. [PMID: 30134984 PMCID: PMC6106891 DOI: 10.1186/s13052-018-0543-4] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/06/2018] [Accepted: 08/13/2018] [Indexed: 11/17/2022] Open
Abstract
Background Congenital space-occupying thoracic malformations and diaphragmatic hernia have in common pulmonary hypoplasia. Our study aims to assess cardiac involvement during post-natal adaptation. Methods A retrospective study was carried out on newborns with prenatally diagnosed intrathoracic mass. Gathering for respiratory distress syndrome (RDS), 35 neonates were compared for clinical course, cardiovascular enzymes, ECG, and ultrasound. Results The analysis revealed a high left heart defect rate in patients with severe RDS, without being influenced by the laterality. Ultrasound or laboratory assessment did not detect altered cardiac dimension or cardiomyopathy. Solely ECG signs of right ventricular strain were found. Increased QT-dispersion, T-wave and cardiac variability alterations in the first hours were all expression of non-specific cardiac repolarization disorders but predict worse outcome. Conclusions Although RDS is the predominant symptom, slight cardiac dysfunctions should be recognized for prompt treatment. Conventional examinations for postnatal adaption should be integrated by complementary investigations.
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Li Q, Xie D, Sihoe A, Dai J, Wang H, Gonzalez-Rivas D, Zhu Y, Jiang G. Video-assisted thoracic surgery is associated with better short-term outcomes than open thoracotomy in adult patients with intralobar pulmonary sequestration. Interact Cardiovasc Thorac Surg 2017; 26:284-287. [DOI: 10.1093/icvts/ivx311] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/22/2017] [Accepted: 08/13/2017] [Indexed: 01/16/2023] Open
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10
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Das S, Ramamurthy A, Venkataramanan R, Madhan K, Balaji V. Inferior vena cava obstruction as a rare manifestation of bronchopulmonary sequestration. SURGICAL PRACTICE 2017. [DOI: 10.1111/1744-1633.12241] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/29/2022]
Affiliation(s)
- Somak Das
- Department of Surgical Gastroenterology, School of Digestive and Liver Diseases; Institute of Post Graduate Medical Education and Research; Kolkata India
| | - Anand Ramamurthy
- Department of Gastrointestinal Surgery and Liver Transplantation; Apollo Hospital; Chennai India
| | | | - Kumar Madhan
- Department of Cardiothoracic Surgery; Apollo Hospital; Chennai India
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11
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Genç O, Gürkök S, Dakak M, Gözübüyük A, Ozkan M, Caylak H. Pulmonary Sequestration and Surgical Treatment. Asian Cardiovasc Thorac Ann 2016; 14:3-6. [PMID: 16432109 DOI: 10.1177/021849230601400102] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/16/2022]
Abstract
Pulmonary sequestration is nonfunctioning, abnormal pulmonary tissue existing inside or outside the visceral pleura. It is not connected to the tracheobronchial tree. In this study, the results of 8 patients, who were diagnosed with pulmonary sequestration and treated during hospitalization in Gülhane Military Medical Academy between 1996 and 2003, were reported. Sex, age, symptoms, diagnostic approach, operative findings, procedures and postoperative outcomes were reviewed. No postoperative complications were seen. In the long-term follow-up all patients were seen to be asymptomatic. We believe surgical resection is mandatory in order to avoid infection and destruction of the normal pulmonary parenchyma even in asymptomatic cases. Furthermore, when infection occurs, major resection such as lobectomy may be necessary rather than segmentectomy or wedge resection, which involves removal of minimal pulmonary parenchyma.
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Affiliation(s)
- Onur Genç
- Department of Thoracic Surgery, Gülhane Military Medical Academy, Etlik, Ankara 06018, Turkey
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12
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Sihoe ADL, Luo Q, Shao G, Li Y, Li J, Pang D. Uniportal thoracoscopic lobectomy for intralobar pulmonary sequestration. J Cardiothorac Surg 2016; 11:27. [PMID: 26868145 PMCID: PMC4750188 DOI: 10.1186/s13019-016-0425-z] [Citation(s) in RCA: 10] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/07/2015] [Accepted: 02/05/2016] [Indexed: 04/01/2024] Open
Abstract
Background Pulmonary sequestration is an uncommon congenital condition for which surgical resection is usually indicated – either via open thoracotomy or conventional multi-port Video-Assisted Thoracoscopic Surgery (VATS). Of the two types of sequestration, intralobar sequestration is technically more challenging to resect. Case Presentation We report the management of a 34 year old male patient with a long history of respiratory symptoms, and an extensively diseased right lower lobe. A diagnosis of sequestration was confirmed by CT scanning, showing three separate anomalous feeding vessels arising from the abdominal aorta. A right lower lobectomy using a Uniportal VATS approach was performed, and the patient discharged home on the fourth post-operative day. Conclusion This is the first report to our knowledge demonstrating the safety and feasibility of the Uniportal approach for the resection of a relatively challenging intralobar sequestration.
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Affiliation(s)
- Alan D L Sihoe
- Division of Thoracic Surgery, Department of Surgery, The University of Hong Kong Shenzhen Hospital, Shenzhen, China.
| | - Qigang Luo
- Division of Thoracic Surgery, Department of Surgery, The University of Hong Kong Shenzhen Hospital, Shenzhen, China
| | - Guangqiang Shao
- Division of Thoracic Surgery, Department of Surgery, The University of Hong Kong Shenzhen Hospital, Shenzhen, China
| | - Yue Li
- Division of Thoracic Surgery, Department of Surgery, The University of Hong Kong Shenzhen Hospital, Shenzhen, China
| | - Jinglong Li
- Division of Thoracic Surgery, Department of Surgery, The University of Hong Kong Shenzhen Hospital, Shenzhen, China
| | - Dazhi Pang
- Division of Thoracic Surgery, Department of Surgery, The University of Hong Kong Shenzhen Hospital, Shenzhen, China
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Tsubouchi H, Matsumoto N, Yanagi S, Ashitani JI, Nakazato M. Successful treatment of chronic lower respiratory tract infection by macrolide administration in a patient with intralobar pulmonary sequestration and primary ciliary dyskinesia. Respir Med Case Rep 2015; 15:62-5. [PMID: 26236606 PMCID: PMC4501538 DOI: 10.1016/j.rmcr.2015.05.008] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/27/2015] [Revised: 04/19/2015] [Accepted: 05/12/2015] [Indexed: 12/21/2022] Open
Abstract
Primary ciliary dyskinesia (PCD) is a genetic disease associated with abnormalities in ciliary structure and function. Although recurrent respiratory infection associated with ciliary dysfunction is a common clinical feature, there is no standardized treatment or management of respiratory infection in PCD patients. Here, we report that respiratory infection with PCD and intralobar sequestration (ILS) were treated successfully with clarithromycin before the surgical resection of ILS. A 15-year-old non-smoking Japanese woman was admitted for productive cough and dyspnea on exertion. Chest CT scan on admission showed complex cystic LESIONS with air-fluid level in the right lower lobe, and diffuse nodular shadows in the whole lobe of the lung. On flexible bronchoscopy examination, sputum and bronchiolar fluid cultures revealed Staphylococcus aureus (S. aureus). An electron microscopic examination of the cilia showed inner dynein arm deficiency. Administration of clarithromycin improved the lower respiratory tract infection associated with S. aureus. CT angiography after clarithromycin treatment demonstrated an aberrant systemic artery arising from the celiac trunk and supplying the cystic mass lesions that were incorporated into the normal pulmonary parenchyma without their own pleural covering. Based on these results, the patient was diagnosed with PCD and ILS. Because of the clarithromycin treatment, resection of the ILS was performed safely without any complications. Although further observation of clarithromycin treatment is needed, we believe that clarithromycin may be considered one of the agents for treating PCD.
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Affiliation(s)
- Hironobu Tsubouchi
- Division of Neurology, Respirology, Endocrinology and Metabolism, Department of Internal Medicine, Faculty of Medicine, University of Miyazaki, Kiyotake, Miyazaki, 889-1692, Japan
| | - Nobuhiro Matsumoto
- Division of Neurology, Respirology, Endocrinology and Metabolism, Department of Internal Medicine, Faculty of Medicine, University of Miyazaki, Kiyotake, Miyazaki, 889-1692, Japan
| | - Shigehisa Yanagi
- Division of Neurology, Respirology, Endocrinology and Metabolism, Department of Internal Medicine, Faculty of Medicine, University of Miyazaki, Kiyotake, Miyazaki, 889-1692, Japan
| | - Jun-Ichi Ashitani
- Division of Neurology, Respirology, Endocrinology and Metabolism, Department of Internal Medicine, Faculty of Medicine, University of Miyazaki, Kiyotake, Miyazaki, 889-1692, Japan
| | - Masamitsu Nakazato
- Division of Neurology, Respirology, Endocrinology and Metabolism, Department of Internal Medicine, Faculty of Medicine, University of Miyazaki, Kiyotake, Miyazaki, 889-1692, Japan
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Chatelain S, Comp RA, Grace RR, Sabbath AM. Cardiomyopathy induced by pulmonary sequestration in a 50-year-old man. Tex Heart Inst J 2015; 42:63-5. [PMID: 25873803 DOI: 10.14503/thij-13-3728] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/23/2022]
Abstract
A 50-year-old black man presented at the emergency department with midsternal, nonradiating chest pressure and chronic dyspnea on exertion. Four years before the current admission, he had been diagnosed with nonischemic cardiomyopathy at another facility. After our complete evaluation, we suspected that his symptoms arose from left-to-left shunting in association with pulmonary sequestration, a congenital malformation. Our preliminary diagnosis of secondary dilated cardiomyopathy was confirmed by normalization of the patient's ventricular size and function after lobectomy. To our knowledge, this patient is the oldest on record to present with cardiomyopathy consequent to pulmonary sequestration. His case is highly unusual because of his age and the rapid resolution of his symptoms after lobectomy. We believe that pulmonary sequestration should be included in the differential diagnosis of dilated cardiomyopathy.
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15
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Hui DS, Shavelle DM, Starnes VA, McFadden PM. Concurrent aortic valvular disease and pulmonary sequestration: clinical implications. Tex Heart Inst J 2015; 41:649-52. [PMID: 25593534 DOI: 10.14503/thij-13-3907] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/23/2022]
Abstract
Pulmonary sequestration refers to segmental lung tissue that has no connection with the bronchial tree or pulmonary arteries. In adults, the clinical sequelae are usually related to infection. Patients are typically referred for sequestrectomy even when they are asymptomatic. There are no guidelines for treating patients who have pulmonary sequestration and coexisting cardiac valvular disease, in which case the venous drainage patterns of sequestra pose the additional risks of infective endocarditis and volume overload. We present the cases of 2 adult patients--one symptomatic and one asymptomatic--who had concurrent aortic valvular disease and pulmonary sequestration, and we discuss the factors involved in our evaluation of their cardiac risk and our treatment decisions. In view of the sparse data to predict cardiac risks, we think that pulmonary sequestrectomy in adult patients with concurrent valvular conditions should be considered on a case-by-case basis.
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16
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Malik S, Khurana S, Vasudevan V, Gupta N. A rare case of underlying pulmonary sequestration in a patient with recently diagnosed medium and large vessel vasculitis. Lung India 2014; 31:176-8. [PMID: 24778487 PMCID: PMC3999684 DOI: 10.4103/0970-2113.129871] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022] Open
Abstract
Vasculitis of medium- and large-sized arteries is an inflammatory and stenotic disease characterized by a strong predilection for the aortic arch and its branches. It presents with symptoms and signs as per the vessels and organs involved. Pulmonary sequestration is a rare abnormality characterized by a mass of nonfunctioning lung tissue that receives its vascular supply from a systemic artery and is separated from the normal tracheobronchial tree. The following is a rare case report showing the presence of pulmonary sequestration in a patient with recently diagnosed hypertension and intestinal angina due to medium and large vessel vasculitis.
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Affiliation(s)
- Sarthak Malik
- Department of Medicine, University College of Medical Sciences, Delhi University, Dilshad Garden, Delhi, India
| | - Sakshi Khurana
- Department of Radio-Diagnosis, University College of Medical Sciences, Delhi University, Dilshad Garden, Delhi, India
| | - Vishnu Vasudevan
- Department of Medicine, University College of Medical Sciences, Delhi University, Dilshad Garden, Delhi, India
| | - Nikhil Gupta
- Department of Medicine, University College of Medical Sciences, Delhi University, Dilshad Garden, Delhi, India
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Liu C, Pu Q, Ma L, Mei J, Xiao Z, Liao H, Liu L. Video-assisted thoracic surgery for pulmonary sequestration compared with posterolateral thoracotomy. J Thorac Cardiovasc Surg 2013; 146:557-61. [DOI: 10.1016/j.jtcvs.2013.04.027] [Citation(s) in RCA: 35] [Impact Index Per Article: 2.9] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/04/2013] [Revised: 03/30/2013] [Accepted: 04/19/2013] [Indexed: 10/26/2022]
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18
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He W, Luo SJ, Chu L. Extralobar pulmonary sequestration in the abdominal cavity: an ultrasound case report. Quant Imaging Med Surg 2013; 3:59-60. [PMID: 23476899 DOI: 10.3978/j.issn.2223-4292.2013.02.02] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/24/2013] [Accepted: 02/20/2013] [Indexed: 11/14/2022]
Affiliation(s)
- Wei He
- Department of Ultrasonography, Third Xiangya Hospital of Central South University, Changsha, Hunan 410013, China
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19
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Orsini B, Doddoli C, Brioude G, D'Journo XB, Trousse D, Gaubert JY, Thomas PA. [Non-tumoral vascular disorders of the lung in the adulthood]. REVUE DE PNEUMOLOGIE CLINIQUE 2012; 68:146-151. [PMID: 22361065 DOI: 10.1016/j.pneumo.2012.01.012] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Accepted: 12/19/2011] [Indexed: 05/31/2023]
Abstract
Non-tumoral vascular disorders of the lung are multiple, even if cases diagnosed in the adulthood are rare. They include congenital or acquired conditions, which related symptoms, if present, are non specific. This explains why their diagnosis is challenging and usually delayed. Surgery is the cornerstone of their treatment, although interventional radiology represents currently a less invasive alternative option for some of them.
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Affiliation(s)
- B Orsini
- Service de chirurgie thoracique et des maladies de l'œsophage, université de la Méditerranée, hôpital Nord, AP-HM, chemin des Bourrely, Marseille cedex 20, France
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Medford ARL, Chandratreya L. Intralobar lung sequestration as a cause of unexplained recurrent chest infections. Br J Hosp Med (Lond) 2011; 72:230-231. [PMID: 21537242 DOI: 10.12968/hmed.2011.72.4.230] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/05/2023]
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Gompelmann D, Eberhardt R, Heussel CP, Hoffmann H, Dienemann H, Schuhmann M, Böckler D, Schnabel PA, Warth A, Lopez-Benitez R, Herth FJF. Lung sequestration: a rare cause for pulmonary symptoms in adulthood. ACTA ACUST UNITED AC 2011; 82:445-50. [PMID: 21311173 DOI: 10.1159/000323562] [Citation(s) in RCA: 13] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/13/2010] [Accepted: 12/02/2010] [Indexed: 12/14/2022]
Abstract
BACKGROUND Lung sequestration is a rare congenital pulmonary disorder and is usually diagnosed in children with recurrent pulmonary infections. Lung sequestrations are not commonly found to be a cause of respiratory symptoms in adults. OBJECTIVES It was the aim of this study to show that pulmonary sequestration is rare in advanced age and can be accompanied by severe pulmonary symptoms. METHODS We conducted a case series analysis of patient characteristics, symptoms, diagnosis and treatment of 11 adults with a lung sequestration at the Thoraxklinik Heidelberg between 2001 and 2009. RESULTS From 2001 to 2009, intralobar lung sequestration was diagnosed and treated in 11 adults aged 19 to 58 years with an average age of 39.9 ± 11.3 years and a male:female distribution of 5:6. In 3 patients (27.3%), the predominant symptom was hemoptysis. Recurrent pulmonary infections occurred in 1 patient (9.1%); pneumonia and lung abscess were detected in 2 patients (18.2%). In 3 cases (27.3%), dry cough was the predominant symptom, and in only 2 cases (18.2%), lung sequestration was asymptomatic. Eight patients (72.7%) were diagnosed by imaging techniques prior to surgery. In 3 cases (27.3%), diagnosis was made intraoperatively and by pathological examination. Surgical intervention included 7 lobectomies (63.6%), 3 wedge resections (27.3%) and 1 (9.1%) segmentectomy. CONCLUSION Lung sequestration in adults is rare, but it can cause severe pulmonary symptoms. In cases of recurrent pulmonary infections of identical localization or recurrent hemoptysis, lung sequestration should be considered in order for the diagnosis to be made rapidly. Surgical resection is the treatment of choice.
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Affiliation(s)
- Daniela Gompelmann
- Department of Pneumology and Respiratory Care Medicine, Thoraxklinik, University of Heidelberg, Heidelberg, Germany.
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Laje P, Liechty KW. Postnatal management and outcome of prenatally diagnosed lung lesions. Prenat Diagn 2008; 28:612-8. [PMID: 18330859 DOI: 10.1002/pd.1966] [Citation(s) in RCA: 56] [Impact Index Per Article: 3.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/20/2022]
Abstract
Advancements in fetal diagnostic imaging have increased prenatal diagnosis of many fetal anomalies. The purpose of this chapter is to review the etiology and natural history of prenatally diagnosed cystic lung lesions, including congenital cystic adenomatoid malformations (CCAM), pulmonary sequestrations (PSs), hybrid lesions, and bronchogenic cysts, and then discuss current concepts in the management and outcome of these lesions.
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Affiliation(s)
- Pablo Laje
- Children's Hospital of Philadelphia, PA 19104, USA
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23
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[Surgical treatment of lung sequestration]. VOJNOSANIT PREGL 2008; 65:33-9. [PMID: 18368936 DOI: 10.2298/vsp0801033c] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/27/2022] Open
Abstract
BACKGROUND/AIM Pulmonary sequestration is a congenital malformation which consists of afunctional part of the lung, separated of the normal airway, and vascularisated with anomal systemic artery instead of pulmonary artery. There are two kinds of sequestration. Intralobar is surrounded with normal lung and its pleura, and extralobar which has extrapulmonary position and pleura of its own. This anomaly is very rare and appears in 1.1-1.8% of all congenital lung malformations. The illness is revealed either in early childhood whith other life-threatening anomalies or in adulthood and middle age when secondary infection arises. The aim of this paper was to show our own experience in surgical treatment of pulmonary sequestration and to emphasize sequestration as a real differential-diagnostic possibility with patients with recidive bronchopneumonias. METHODS We retrospectively analyzed medical records for the period from 1967-2007 and found 15 patients with pulmonary sequestration at the average age of 30 years. We pointed out the well known problems with identification of this anomaly, preoperative diagnostic procedures and surgical possibilities of treatment. RESULTS There were 13 patients with intralobar and two patients with extralobar sequestration. By the use of preoperative angiography, seven patients were found to have intralobar pulmonary sequestration. All intralobar sequestrations were clinically manifested, the most often with recidive bronchopneumonia. Six patients had no preoperative diagnosis of lung sequestration. The most common locality of intralobar sequestration was the left lower lobe (eight patients). We performed nine lobectomies, three sequestrectomies, two segmentectomies and one pneumonectomy. Both extralobar sequestrations were diagnosed intraoperatively. CONCLUSION Pulmonary sequestration is a rare malformation. Diagnosis is established by angiography. Treatment is exclusively surgical. In the last three years we have had one patient per year. This experience obliges to consider pulmonary sequestration as a real differn tial diagnostic possibility in patients with localised repeated bronchopneumonias.
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Abstract
Pulmonary vascular malformations have historically been diagnosed in a wide range of age groups, but the extensive use of prenatal imaging studies has resulted in the majority of lesions being diagnosed in utero. Among this group of lesions, bronchopulmonary sequestrations (BPS), hybrid lesions with both congenital cystic adenomatoid malformation (CCAM) and BPS, aberrant systemic vascular anastomoses, and pulmonary arteriovenous malformations (PAVM), are the most common. The biologic behavior of these lesions and the subsequent therapy is, in large part, determined by the age of the patient at diagnosis. In the fetus, large BPS or hybrid lesions can result in fetal hydrops and in utero fetal demise. In the perinatal period, pulmonary hypoplasia from the mass effect or air trapping within the cystic component of hybrid lesions can result in life-threatening respiratory distress. In the postnatal period, communication of the lesion with the aero-digestive system can result in recurrent pneumonia. Alternatively, increased pulmonary blood flow from the systemic arterial supply can result in hemorrhage, hemoptysis, or high output cardiac failure. In addition, there have been several reports of malignant degeneration. Finally, the broad spectrum encompassed by these lesions makes classification and subsequent communication of the lesions confusing and difficult. This paper will review the components of these lesions, their associated anomalies, the diagnosis and natural history, and finally, current concepts in the management of pulmonary vascular malformations.
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Affiliation(s)
- Kenneth W Liechty
- Center for Fetal Research, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania 19104, USA.
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25
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Gezer S, Taştepe I, Sirmali M, Findik G, Türüt H, Kaya S, Karaoğlanoğlu N, Cetin G. Pulmonary sequestration: a single-institutional series composed of 27 cases. J Thorac Cardiovasc Surg 2007; 133:955-9. [PMID: 17382633 DOI: 10.1016/j.jtcvs.2006.11.003] [Citation(s) in RCA: 68] [Impact Index Per Article: 3.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/25/2006] [Revised: 10/02/2006] [Accepted: 11/02/2006] [Indexed: 12/12/2022]
Abstract
OBJECTIVE Large series about pulmonary sequestration from a single institute are rare in the literature. In this study, we aimed to evaluate diagnosis, treatment, and outcomes of pulmonary sequestration in a single institute. METHODS Records of patients with pulmonary sequestration between January 1982 and January 2006 were reviewed retrospectively. Age, sex, symptoms, diagnostic procedures, operative findings, operative techniques, postoperative complications, and follow-up results were evaluated. RESULTS Twenty-seven patients, 17 male and 10 female, with an average age of 23.3 were operated on for pulmonary sequestration. Twenty patients had preoperative symptoms including recurrent pneumonia attacks, chest pain, hemoptysis, and shortness of breath. Chest radiography, thoracic computed tomography, aortography, magnetic resonance imaging, and bronchoscopy were used as diagnostic methods. Of the cases, 19 (70%) were intralobar pulmonary sequestration and 8 (30%) were extralobar pulmonary sequestration. Surgical procedures were lower lobectomy in 18 and segmentectomy in 1 of the patients with intralobar pulmonary sequestration and simple mass excision in all of those with extralobar pulmonary sequestration. Postoperative histopathologic examinations excluded any other alternative diagnosis. Furthermore, it detected an aspergilloma ball in 1 of the intralobar pulmonary sequestration specimens. Two patients had a postoperative complication (prolonged air leak in 1 patient and empyema in the other). During the follow-up period (mean 2.3 years), none of the patients presented a problem. No mortality was encountered. CONCLUSION Owing to the potentially severe complications they can cause, pulmonary sequestrations should be removed whenever they are diagnosed. Since careful dissection provides sufficient surgical comfort, preoperative identification of the aberrant vessels is not a rule for the success of the operation.
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Affiliation(s)
- Suat Gezer
- Atatürk Chest Diseases and Thoracic Surgery Training and Research Hospital, Clinic of Thoracic Surgery, Ankara, Turkey.
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Abstract
Anesthesia for adults with congenital heart disease has many challenging physiologic considerations. Collaborative relationships of a multidisciplinary team including cardiology, cardiac surgery, anesthesiology, and intensive care are essential to ensure positive outcomes in this population for noncardiac and cardiac surgery.
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Affiliation(s)
- Jane Heggie
- Department of Anesthesia, Toronto General Hospital, 3 Eaton North, 200 Elizabeth Street, Toronto, Ontario M5G 2C4, Canada
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27
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Freedom RM, Yoo SJ, Goo HW, Mikailian H, Anderson RH. The bronchopulmonary foregut malformation complex. Cardiol Young 2006; 16:229-51. [PMID: 16725062 DOI: 10.1017/s104795110600031x] [Citation(s) in RCA: 33] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 07/05/2005] [Indexed: 12/24/2022]
Affiliation(s)
- Robert M Freedom
- Department of Paediatrics, Division of Cardiology, The University of Toronto, Toronto, Canada
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Behnia MM, Catalano PW, Brooks WS. Hemoptysis in a 38-Year-Old Woman Receiving an Oral Contraceptive. Chest 2004; 125:1944-7. [PMID: 15136411 DOI: 10.1378/chest.125.5.1944] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/10/2023] Open
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Affiliation(s)
- Claire Langston
- Department of Pathology, Texas Children's Hospital, Houston, TX 77030, USA
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Shope AL, Islam S, Zaher A. Pathologic quiz case: a 30-year-old man with recurrent pneumonia. Intralobar bronchopulmonary sequestration. Arch Pathol Lab Med 2002; 126:623-4. [PMID: 12013015 DOI: 10.5858/2002-126-0623-pqcayo] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/06/2022]
Affiliation(s)
- Adam L Shope
- Department of Pathology, Medical College of Ohio, Toledo, Ohio, USA
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Martí V, Pujadas S, Casan P, García J, Guiteras P, Augé JM. Reversible dilated cardiomyopathy after lobectomy for pulmonary sequestration. J Thorac Cardiovasc Surg 2001; 121:1001-2. [PMID: 11326252 DOI: 10.1067/mtc.2001.112320] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/22/2022]
Affiliation(s)
- V Martí
- Department of Cardiology and Cardiac Surgery, Hospital de la Santa Creu i Sant Pau, Av Sant Antoni M. Claret 167, 08025-Barcelona, Spain.
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Van Raemdonck D, De Boeck K, Devlieger H, Demedts M, Moerman P, Coosemans W, Deneffe G, Lerut T. Pulmonary sequestration: a comparison between pediatric and adult patients. Eur J Cardiothorac Surg 2001; 19:388-95. [PMID: 11306301 DOI: 10.1016/s1010-7940(01)00603-0] [Citation(s) in RCA: 97] [Impact Index Per Article: 4.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/23/2023] Open
Abstract
OBJECTIVE Modern large single institutional reports on pulmonary sequestration (PS) are extremely rare. We were interested in comparing patients with PS referred by our pediatric versus adult pulmonologists. METHODS Hospital notes of all patients operated on between 1978 and 1997 for a congenital broncho-pulmonary malformation were reviewed. In 28 patients, the parenchymal lesion was vascularized by a systemic artery and was separated from the bronchial tree, thus matching the strict definition of PS. Patient characteristics and outcome were analyzed comparing the pediatric group (< or =16 years: n=13; mean age, 3+/-5 years) versus the adult group (>16 years: n=15; mean age, 33+/-13 years). RESULTS No significant differences between both groups were observed in sex, side, type of sequestration, pulmonary venous drainage, associated anomalies, hospital and late outcome, and patient's overall score. Patients (n=21) with the intralobar type of sequestration presented significantly more often with an infection when compared with patients (n=7) with the extralobar type (91 versus 14%; P=0.0033). When compared with the pediatric group, patients in the adult group had significantly more respiratory infections (87 versus 38%; P=0.016), and also required a lobectomy more often (67 versus 31%; P=0.056). CONCLUSIONS The extralobar type of sequestration often remains asymptomatic, and is usually an incidental finding during infancy. The intralobar type mostly presents with recurrent infections in adulthood resulting in more lobectomies. We believe these findings support our current policy to remove any pulmonary malformation whenever diagnosed in order to: (1), prevent infection and other potentially serious late complications which may compromise the surgical outcome; and (2), enhance the chance of a parenchymal-sparing resection.
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Affiliation(s)
- D Van Raemdonck
- Department of Thoracic Surgery, University Hospital Gasthuisberg, Herestraat 49, B-3000 Leuven, Belgium.
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