肺隔离症4例临床分析并文献复习
摘要
目的
探讨肺隔离症的临床表现及影像学特点,提高对肺隔离症的诊疗水平,减少误诊误治。
方法
收集2008年1月至2010年6月期间就诊于浙江大学附属邵逸夫医院呼吸内科,并最终诊断为肺隔离症的病例共4例,对4例患者的临床特点、影像学表现、纤维支气管镜检查及治疗转归进行讨论和分析,并结合相关文献进行复习。
结果
叶内型隔离肺3例,为男性1例,女性2例,年龄24～31岁,叶外型隔离肺1例,女性,60岁。1例叶内型表现为反复咯血,病史长达18年,2例表现为咳嗽咳痰伴发热,其中1例女性患者同时患有肺结核；1例叶外型为常规体检发现,无不适主诉。3例叶内型患者首次就诊均被误诊为支气管扩张、肺脓肿等,1例叶外型初诊时考虑肺占位性病变。叶内型隔离肺好发于左下肺,胸部cT可表现为极淡薄阴影,也可表现为大片团块状密度增高影,伴或不伴囊肿样改变及充气支气管征,并可见来自体循环的异常供血动脉；叶外型隔离肺常表现为与周围肺组织分界清晰的囊性包块,也可见异常供血动脉。无论叶内型还是叶外型,纤维支气管镜表现均无特异性,可无异常改变或仅表现为支气管黏膜轻度充血。2例叶内型予病变肺叶切除,随访4个月至1年半,疗效确切；1例同时合并肺结核的叶内型先予抗结核治疗,现随访中；1例叶外型未予治疗。
结论
肺隔离症以叶内型多见,常见症状为反复呼吸道感染及咯血,叶外型肺隔离症一般无临床症状,胸部多层螺旋CT及血管三维重建一般可确诊。一旦确诊为肺隔离症均建议积极外科治疗,病变肺叶切除预后良好。
Objectives
To investigate the clinical presentations and features of pulmonary sequestration, and therefore to improve the diagnostic accuracy.
Methods
4 patients diagnosed as pulmonary sequestration in Sir Run Run Shaw Hospital from Jan,2008 to Jun,2010 were reviewed and analyzed. After preparation of clinical information materials of these patients, we analyzed the clinical characteristics, imaging, fibrobronchoscopy, treatment and prognosis retrospectively, and the literature were reviewed as well.
Results
3 cases,1 male and 2 females, aged from 24 to 31, were diagnosed with intralobar sequestration(ILS); 1 case, female, aged 60, with extralobar sequestration(ELS). Intermittent hemoptysis was presented in 1 ILS patient with the history of 18 years and other 2 had cough, expectoration and fever, including 1 female case with a concurrence of tuberculosis; The ELS patient was detected in a routine chest radiography, without any recorded complaints. The three ILS cases were initially mistaken for bronchiectasis or lung abscess. The ELS case was firstly misdiagnosed as a lung mass. The spiral CT of the ILS patients demonstrated thin shadows or massive consolidation with or without cystiform alterations and air bronchogram, mostly located at the left lower lung, and often with finding of aberrant feeding artery from systemic artery. With the ELS case, a well-defined cystic mass and aberrant feeding artery can be found in CT scan. In fibrobronchoscopy, no other abnormality can be found both in ILS and ELS cases except light congestion of bronchial mucosa.2 cases of ILS underwent lobectomy with excellent result and long-term favorable outcome,1 case concomitant with TB treated with antituberculosis, and 1 case of ELS was not treated.
Conclusions
In pulmonary sequestration, ILS occurs more frequently than ELS, the common symptoms are recurrent respiratory infection and hemoptysis. In ELS patients, usually there is no complaint. Chest spiral CT scan and three-dimensional reconstruction of blood vessels are usually helpful for the diagnosis. Once PS is diagnosed, surgery is suggested, and the common treatment is resection of the sequestered lobe and the clinical outcome is favorable.
探讨肺隔离症的临床表现及影像学特点,提高对肺隔离症的诊疗水平,减少误诊误治。
方法
收集2008年1月至2010年6月期间就诊于浙江大学附属邵逸夫医院呼吸内科,并最终诊断为肺隔离症的病例共4例,对4例患者的临床特点、影像学表现、纤维支气管镜检查及治疗转归进行讨论和分析,并结合相关文献进行复习。
结果
叶内型隔离肺3例,为男性1例,女性2例,年龄24～31岁,叶外型隔离肺1例,女性,60岁。1例叶内型表现为反复咯血,病史长达18年,2例表现为咳嗽咳痰伴发热,其中1例女性患者同时患有肺结核；1例叶外型为常规体检发现,无不适主诉。3例叶内型患者首次就诊均被误诊为支气管扩张、肺脓肿等,1例叶外型初诊时考虑肺占位性病变。叶内型隔离肺好发于左下肺,胸部cT可表现为极淡薄阴影,也可表现为大片团块状密度增高影,伴或不伴囊肿样改变及充气支气管征,并可见来自体循环的异常供血动脉；叶外型隔离肺常表现为与周围肺组织分界清晰的囊性包块,也可见异常供血动脉。无论叶内型还是叶外型,纤维支气管镜表现均无特异性,可无异常改变或仅表现为支气管黏膜轻度充血。2例叶内型予病变肺叶切除,随访4个月至1年半,疗效确切；1例同时合并肺结核的叶内型先予抗结核治疗,现随访中；1例叶外型未予治疗。
结论
肺隔离症以叶内型多见,常见症状为反复呼吸道感染及咯血,叶外型肺隔离症一般无临床症状,胸部多层螺旋CT及血管三维重建一般可确诊。一旦确诊为肺隔离症均建议积极外科治疗,病变肺叶切除预后良好。
Objectives
To investigate the clinical presentations and features of pulmonary sequestration, and therefore to improve the diagnostic accuracy.
Methods
4 patients diagnosed as pulmonary sequestration in Sir Run Run Shaw Hospital from Jan,2008 to Jun,2010 were reviewed and analyzed. After preparation of clinical information materials of these patients, we analyzed the clinical characteristics, imaging, fibrobronchoscopy, treatment and prognosis retrospectively, and the literature were reviewed as well.
Results
3 cases,1 male and 2 females, aged from 24 to 31, were diagnosed with intralobar sequestration(ILS); 1 case, female, aged 60, with extralobar sequestration(ELS). Intermittent hemoptysis was presented in 1 ILS patient with the history of 18 years and other 2 had cough, expectoration and fever, including 1 female case with a concurrence of tuberculosis; The ELS patient was detected in a routine chest radiography, without any recorded complaints. The three ILS cases were initially mistaken for bronchiectasis or lung abscess. The ELS case was firstly misdiagnosed as a lung mass. The spiral CT of the ILS patients demonstrated thin shadows or massive consolidation with or without cystiform alterations and air bronchogram, mostly located at the left lower lung, and often with finding of aberrant feeding artery from systemic artery. With the ELS case, a well-defined cystic mass and aberrant feeding artery can be found in CT scan. In fibrobronchoscopy, no other abnormality can be found both in ILS and ELS cases except light congestion of bronchial mucosa.2 cases of ILS underwent lobectomy with excellent result and long-term favorable outcome,1 case concomitant with TB treated with antituberculosis, and 1 case of ELS was not treated.
Conclusions
In pulmonary sequestration, ILS occurs more frequently than ELS, the common symptoms are recurrent respiratory infection and hemoptysis. In ELS patients, usually there is no complaint. Chest spiral CT scan and three-dimensional reconstruction of blood vessels are usually helpful for the diagnosis. Once PS is diagnosed, surgery is suggested, and the common treatment is resection of the sequestered lobe and the clinical outcome is favorable.
引文
1、Pryce DM. Lower accessory pulmonary with intralobar sequestration of lung: report of seven cases [J]. J Pathol,1946,58(4):457-467
2、Shibli M, Connery C, Shapiro JM. Intralobar and extralobar bronchopulmonary sequestration complicated by nocardia asteroides infection [J]. Southern Med J, 2003,96(1):78-80.
3、Savic B, Britel FJ, Tholen W, et al. Lung sequestration:report of seven cases and review of 540 published cases [J]. Thorax,1979,34:96-101.
4、Mehrdad MB, Phillip WC, William SB. Hemoptysis in a 38-Year-Old Woman Receiving an Oral Contraceptive [J]. Chest,2004,125:1944-1947.
5、Ammar J, Ghrairi H, Ismail, et al. Pulmonary sequestrations:from radiologic to preoperative diagnosis:2 cases with review of the literature[J]. Tunis Med, 2006,84(2):118-121.
6、王新卫,刘新年,张维.以大咯血为突出表现的肺隔离症1例[J].医学新知杂志,2007,2：124.
7、纪建松,章士正,王丽华,等.螺旋CT及重建技术对肺隔离症的诊断价值[J].中华结核和呼吸杂志,2007,8：620-621.
8、Kang M, Khandelwal N, Oj ili V, et al. Multidetector CT angiography in pulmonary sequestration[J]. J Comput Assist Tomogr,2006,30(6):926-932.
9、 李义,田建明,曾宪强,等.肺隔离症的影像诊断和介入治疗[J].临床放射学杂志2004,2：122-124.
10、Stern R, Berger S, Casaulta C, et al. Bilateral intralobar pulmonary sequestration in a newborn. cases report and review of the literature on bilateral pulmonary sequestrations[J]. Journal of Pediatric Surgery,2007,42(4):19-23.
11、李畅,马海涛,赵军,等.肺隔离症的流行病学调查及临床诊断治疗分析[J]. 实用预防医学2007,12：1856-1857.
12、Bell-Thomson J, Missier P, Sommers S C. lung carcinoma arising in bronchopulmonary sequestration[J]. Cancer,1979,44(1):334-339.
13、Gustafson RA, Murray GF, Warden HE, et al. Intralobar sequestration:a missed diagnosis[J]. Ann Thorac Surg,1989,47(6):841-847.
14、杨异,陈文虎,王勇文,等.27例肺隔离症的临床分析[J].心肺血管病杂志,2003,1：11-13.
15、王秋梅,谢兰品,李素娟,等.肺隔离症并结核感染1例[J].临床荟萃,2007,2：138.
16、Okamoto T, Masuya D, Nakashima T, et al. Successful treatment for lung caucer associated with pulmonary sequestration[J]. Ann Thorac Surg,2005,80(6): 2344-2346.
17、张卫国,周清华.叶内型肺隔离症的诊断和治疗[J].医学研究杂志.2009,3：33-35.
18、Loscertales J, CongregadoM, ArroyoA, et al. Treatment of pulmonary sequestration by video-assisted thoracic surgery (VATS). Surg Endosc,2003,8:1323.
19、唐晓军,谭仲俊,蔡义兵,等.肺隔离症引发大咯血的介入治疗[J].介入放射学杂志,2007,11：734-736.
1、Pryce DM. Lower accessory pulmonary with intralobar sequestration of lung: report of seven cases [J]. J Pathol,1946,58(4):457-467
2、Shibli M, Connery C, Shapiro JM. Intralobar and extralobar bronchopulmonary sequestration complicated by nocardia asteroides infection [J]. Southern Med J, 2003,96(1):78-80.
3、Savic B, Britel FJ, Tholen W, et al. Lung sequestration:report of seven cases and review of 540 published cases [J]. Thorax,1979,34:96-101.
4、Mehrdad MB, Phillip WC, William SB. Hemoptysis in a 38-Year-Old Woman Receiving an Oral Contraceptive [J]. Chest,2004,125:1944-1947.
5、Hashemzadeh S, Aslanabadi S, Jafari Rouhi AH, et al. Congenital malformations of the lung [J]. Indian J Pediatr,2007; 74(2):192-4.
6、Ammar J, Ghrairi H, Ismail, et al. Pulmonary sequestrations:from radiologic to preoperative diagnosis:2 cases with review of the literature[J]. Tunis Med, 2006,84(2):118-121.
7、 韩志青,罗艳蓉,徐晓雯.肺隔离症误诊为支气管哮喘1例[J].实用医学杂志,2009,4：662.
8、王新卫,刘新年,张维.以大咯血为突出表现的肺隔离症1例[J].医学新知杂志,2007,2：124.
9、纪建松,章士正,王丽华,等.螺旋CT及重建技术对肺隔离症的诊断价值[J].中华结核和呼吸杂志,2007,8：620-621.
10、Stern R, Berger S, Casaulta C, et al. Bilateral intralobar pulmonary sequestration in a newborn, cases report and review of the literature on bilateral pulmonary sequestrations[J]. Journal of Pediatric Surgery,2007,42(4):19-23.
11, Gustafson RA, Murray GF, Warden HE, et al. Intralobar sequestration:a missed diagnosis[J]. AnnThorac Surg,1989,47(6):841-847.
12、杨异,陈文虎,王勇文,等.27例肺隔离症的临床分析[J].心肺血管病杂志,2003,1：11-13.
13. Berna P, Lebied el D, Assouad J, et al. Pulmonary sequestration and aspergillosis [J].Eur J Cardiothorac Surg2005; 27:28-31.
14、李畅,马海涛,赵军,等.肺隔离症的流行病学调查及临床诊断治疗分析[J].实用预防医学2007,12：1856-18577.
15、Bell-Thomson J, Missier P, Sommers S C. lung carcinoma arising in bronchopulmonary sequestration[J]. Cancer,1979,44(1):334-339.
16、Sakuma T, Sugita M, Sagawa M, et al. Video-assisted thoracoscopic wedge resection for pulmonary sequestration[J]. Ann Thorac Surg 2004,78:1844-5.
17、Tanaka T, Ueda K, Sakano H, et al. Videoassisted thoracoscopic surgery for intralobar pulmonary sequestration[J], Surgery 2003,133:216-8.
18、张卫国,周清华.叶内型肺隔离症的诊断和治疗[J].医学研究杂志.2009,3：33-35.
19、Loscertales J, CongregadoM, Arroyo A, et al. Treatment of pulmonary sequestration by video-assisted thoracic surgery (VATS). Surg Endosc,2003,8: 1323.
20、Diego Gonzalez, Jose Garcia, Eva Fieira, et al. Video-assisted thoracoscopic lobectomy in the treatment of intralobar pulmonary sequestration[J]. Interactive Cardio Vascular and Thoracic Surgery 2011; 12:77-79.
21、Dimitrios Avgerinos, Angelo Reyes, Eduardo Plantilla, et al. Video-assisted thoracoscopic surgery for intralobar pulmonary sequestration[J]. Cases Journal 2008,1:269.
2、Shibli M, Connery C, Shapiro JM. Intralobar and extralobar bronchopulmonary sequestration complicated by nocardia asteroides infection [J]. Southern Med J, 2003,96(1):78-80.
3、Savic B, Britel FJ, Tholen W, et al. Lung sequestration:report of seven cases and review of 540 published cases [J]. Thorax,1979,34:96-101.
4、Mehrdad MB, Phillip WC, William SB. Hemoptysis in a 38-Year-Old Woman Receiving an Oral Contraceptive [J]. Chest,2004,125:1944-1947.
5、Ammar J, Ghrairi H, Ismail, et al. Pulmonary sequestrations:from radiologic to preoperative diagnosis:2 cases with review of the literature[J]. Tunis Med, 2006,84(2):118-121.
6、王新卫,刘新年,张维.以大咯血为突出表现的肺隔离症1例[J].医学新知杂志,2007,2：124.
7、纪建松,章士正,王丽华,等.螺旋CT及重建技术对肺隔离症的诊断价值[J].中华结核和呼吸杂志,2007,8：620-621.
8、Kang M, Khandelwal N, Oj ili V, et al. Multidetector CT angiography in pulmonary sequestration[J]. J Comput Assist Tomogr,2006,30(6):926-932.
9、 李义,田建明,曾宪强,等.肺隔离症的影像诊断和介入治疗[J].临床放射学杂志2004,2：122-124.
10、Stern R, Berger S, Casaulta C, et al. Bilateral intralobar pulmonary sequestration in a newborn. cases report and review of the literature on bilateral pulmonary sequestrations[J]. Journal of Pediatric Surgery,2007,42(4):19-23.
11、李畅,马海涛,赵军,等.肺隔离症的流行病学调查及临床诊断治疗分析[J]. 实用预防医学2007,12：1856-1857.
12、Bell-Thomson J, Missier P, Sommers S C. lung carcinoma arising in bronchopulmonary sequestration[J]. Cancer,1979,44(1):334-339.
13、Gustafson RA, Murray GF, Warden HE, et al. Intralobar sequestration:a missed diagnosis[J]. Ann Thorac Surg,1989,47(6):841-847.
14、杨异,陈文虎,王勇文,等.27例肺隔离症的临床分析[J].心肺血管病杂志,2003,1：11-13.
15、王秋梅,谢兰品,李素娟,等.肺隔离症并结核感染1例[J].临床荟萃,2007,2：138.
16、Okamoto T, Masuya D, Nakashima T, et al. Successful treatment for lung caucer associated with pulmonary sequestration[J]. Ann Thorac Surg,2005,80(6): 2344-2346.
17、张卫国,周清华.叶内型肺隔离症的诊断和治疗[J].医学研究杂志.2009,3：33-35.
18、Loscertales J, CongregadoM, ArroyoA, et al. Treatment of pulmonary sequestration by video-assisted thoracic surgery (VATS). Surg Endosc,2003,8:1323.
19、唐晓军,谭仲俊,蔡义兵,等.肺隔离症引发大咯血的介入治疗[J].介入放射学杂志,2007,11：734-736.
1、Pryce DM. Lower accessory pulmonary with intralobar sequestration of lung: report of seven cases [J]. J Pathol,1946,58(4):457-467
2、Shibli M, Connery C, Shapiro JM. Intralobar and extralobar bronchopulmonary sequestration complicated by nocardia asteroides infection [J]. Southern Med J, 2003,96(1):78-80.
3、Savic B, Britel FJ, Tholen W, et al. Lung sequestration:report of seven cases and review of 540 published cases [J]. Thorax,1979,34:96-101.
4、Mehrdad MB, Phillip WC, William SB. Hemoptysis in a 38-Year-Old Woman Receiving an Oral Contraceptive [J]. Chest,2004,125:1944-1947.
5、Hashemzadeh S, Aslanabadi S, Jafari Rouhi AH, et al. Congenital malformations of the lung [J]. Indian J Pediatr,2007; 74(2):192-4.
6、Ammar J, Ghrairi H, Ismail, et al. Pulmonary sequestrations:from radiologic to preoperative diagnosis:2 cases with review of the literature[J]. Tunis Med, 2006,84(2):118-121.
7、 韩志青,罗艳蓉,徐晓雯.肺隔离症误诊为支气管哮喘1例[J].实用医学杂志,2009,4：662.
8、王新卫,刘新年,张维.以大咯血为突出表现的肺隔离症1例[J].医学新知杂志,2007,2：124.
9、纪建松,章士正,王丽华,等.螺旋CT及重建技术对肺隔离症的诊断价值[J].中华结核和呼吸杂志,2007,8：620-621.
10、Stern R, Berger S, Casaulta C, et al. Bilateral intralobar pulmonary sequestration in a newborn, cases report and review of the literature on bilateral pulmonary sequestrations[J]. Journal of Pediatric Surgery,2007,42(4):19-23.
11, Gustafson RA, Murray GF, Warden HE, et al. Intralobar sequestration:a missed diagnosis[J]. AnnThorac Surg,1989,47(6):841-847.
12、杨异,陈文虎,王勇文,等.27例肺隔离症的临床分析[J].心肺血管病杂志,2003,1：11-13.
13. Berna P, Lebied el D, Assouad J, et al. Pulmonary sequestration and aspergillosis [J].Eur J Cardiothorac Surg2005; 27:28-31.
14、李畅,马海涛,赵军,等.肺隔离症的流行病学调查及临床诊断治疗分析[J].实用预防医学2007,12：1856-18577.
15、Bell-Thomson J, Missier P, Sommers S C. lung carcinoma arising in bronchopulmonary sequestration[J]. Cancer,1979,44(1):334-339.
16、Sakuma T, Sugita M, Sagawa M, et al. Video-assisted thoracoscopic wedge resection for pulmonary sequestration[J]. Ann Thorac Surg 2004,78:1844-5.
17、Tanaka T, Ueda K, Sakano H, et al. Videoassisted thoracoscopic surgery for intralobar pulmonary sequestration[J], Surgery 2003,133:216-8.
18、张卫国,周清华.叶内型肺隔离症的诊断和治疗[J].医学研究杂志.2009,3：33-35.
19、Loscertales J, CongregadoM, Arroyo A, et al. Treatment of pulmonary sequestration by video-assisted thoracic surgery (VATS). Surg Endosc,2003,8: 1323.
20、Diego Gonzalez, Jose Garcia, Eva Fieira, et al. Video-assisted thoracoscopic lobectomy in the treatment of intralobar pulmonary sequestration[J]. Interactive Cardio Vascular and Thoracic Surgery 2011; 12:77-79.
21、Dimitrios Avgerinos, Angelo Reyes, Eduardo Plantilla, et al. Video-assisted thoracoscopic surgery for intralobar pulmonary sequestration[J]. Cases Journal 2008,1:269.