Extralobar pulmonary sequestration in children with abdominal pain:Four case reports

BACKGROUND Extralobar pulmonary sequestration(ELS)with torsion is extremely rare,consequently,the diagnosis of ELS with torsion in children presents a challenge for clinicians.Herein,we report four cases of ELS with torsion that presented with abdominal pain,and further review the relevant literature to summarize the clinical features.CASE SUMMARY Four children presented to our department with abdominal pain.All underwent chest computed tomography,which revealed an intrathoracic soft tissue mass with pleural effusion.All four children underwent thoracoscopic resection of the identified pulmonary sequestration,and the vascular pedicle was clipped and excised.None of the patients experienced any postoperative complications.CONCLUSION Clinicians should consider the possibility of ELS with torsion in children presenting with abdominal pain as the chief complaint.

Surgical Treatment of Intralobar Pulmonary Sequestration

Objective To evaluate the clinical features,diagnosis,treatment,and outcome of intralobar pulmonary sequestration (ILS). Methods Patients who were diagnosed with ILS in our hospital between January 1988 and January 2009 were retrospectively reviewed. We recorded the clinical symptoms,imaging findings,operative technique,complications,and outcome of these patients. Results Forty-seven patients (25 men and 22 women) with an average age of 32.3 years were enrolled. Forty-two patients had symptoms including cough and hemoptysis. Chest X-ray,computed tomography (CT),magnetic resonance imaging (MRI),and angiography were performed. Thoracotomy was performed in 45 patients,while thoracoscopy was performed in 2 patients. Lobectomy was the most common treatment procedure. Massive bleeding developed in 2 patients due to injury of aberrant supplying artery intraoperatively,1 patient had atrial fibrillation,1 patient had thrombosis of upper extremity postoperatively. All patients were confirmed the diagnosis pathologically,4 accompanied with bronchogenic cyst,15 with bronchiectasis,8 with infection,2 with aspergilloma,and 1 with carcinoid. No late complications occurred. Conclusions ILS is rare,surgery is recommended because some patients may have potential severe complications. Contrast enhanced CT and three-dimensional reconstruction is the best diagnostic method. Both thoracotomy and thoracoscopy are appropriate for the selected candidates.

Rare presentation of intralobar pulmonary sequestration associated with repeated episodes of ventricular tachycardia

Arterial supply of an intralobar pulmonary sequestration(IPS) from the coronary circulation is extremely rare. A significant coronary steal does not occur because of dual or triple sources of blood supply to sequestrated lung tissue. We present a 60-year-old woman who presented to us with repeated episodes of monomorphic ventricular tachycardia(VT) in last 3 mo. Radio frequency ablation was ineffective. On evaluation, she had right lower lobe IPS with dual arterial blood supply, i.e., right pulmonary artery and the systemic arterial supply from the right coronary artery(RCA). Stress myocardial perfusion scan revealed significant inducible ischemia in the RCA territory. Coronary angiogram revealed critical stenosis of proximal RCA just after the origin of the systemic artery supplying IPS. The critical stenosis in the RCA was stented. At 12 mo follow-up, she had no further episodes of VT or angina.

Pulmonary sequestration infected with nontuberculous mycobacteria:a report of two cases and literature review

We report two cases of pulmonary sequestration infected with nontuberculous mycobacteria(NTM): Mycobacterium avium and Mycobacterium abscessus.Chest computed tomography showed pneumonic consolidation in the right lower lobe,which received a systemic blood supply from the descending aorta in both patients.Video-assisted thoracoscopic surgeries were successfully performed and pathologica]examinations revealed multiple caseating granulomas.A review of the literature revealed only seven previous case reports of pulmonary sequestration infected with NTM,and no case with Mycobacterium abscessus has been reported.

Pulmonary Sequestration: Three Dimensional Dynamic Contrast-enhanced MR Angiography and MRI

In order to evaluate the diagnostic value of three dimentional contrast enhanced MR angiography and MRI for pulmonary sequestration, 5 patients with pulmonary sequestration underwent 3D fast imaging by steady state precession (FISP) with a contrast medium and breath holding following chest radiography, CT and MR scans. The reconstructed MR angiography was performed using maximum intensity projection (MIP) and multiplanar reconstruction (MPR) techniques. It was found that the chest radiography showed pulmonary sequestration as a persistent area of opacity in the posterior basal segment of the left lower lobe, which was close to mediastinum in 2 cases and close to diaphragma in 3 cases. CT revealed a soft issue mass beyond descending aorta and lobar emphysema around the pulmonary sequestration. And the supplying vessel was documented in 2 cases on enhanced CT. MRI demonstrated a hyperintensity mass with respect to normal lung parenchyma on T1WI and T2WI, and the origin of the supplying vessel in 3 cases. The reconstructed CE MRA using MIP or MRP techniques clearly showed the supplying vessel and its course, branches as well as draining vessels. It was concluded that 3D CE MRA of demonstrating the supplying and draining vessels to pulmonary sequestration, together with plain MRI, can provide a diagnosis and aid in surgical planning without the need for DSA.

A case report of extralobar pulmonary sequestration in a dog

Pulmonary sequestration is a rare congenital anomaly in the veterinary literature.This malformation is characterized by a cystic mass of non-functioning primitive lung tissue that does not communicate with the tracheobronchial tree or with the pulmonary arteries.This article describes gross and histopathological characteristics of extralobar pulmonary sequestration in a dog.Crossly,a mass was observed in the left side of the thoracic cavity,closed to the caudal lobes of the lung,without communication with the tracheobronchial tree and the pulmonary arteries that was separated by pleural covering.Histopathologic examination showed emphysematous alveoli and bronchi,hypertrophy of smooth muscles and presence of the undifferentiated mesenchymal tissue.Therefore,based on microscopic findings,extralobar pulmonary sequestration was diagnosed.To the best of our knowledge,this is the first report of extralobar pulmonary sequestration in dog.

Nocardia cyriacigeorgica infection in a patient with pulmonary sequestration: A case report

BACKGROUND Nocardia cyriacigeorgica (N. cyriacigeorgica) infection is uncommon in clinicalpractice. Pulmonary sequestration complicated with N. cyriacigeorgica has notbeen reported in the literature. Here, we present a case of pulmonary sequestrationcomplicated with N. cyriacigeorgica infection in an immunocom-petentwoman.CASE SUMMARYA 37-year-old woman complaining of a recurrent cough was admitted to ourhospital. Pulmonary sequestration in the lower lobe of the left lung wasdiagnosed by enhanced computed tomography. Bronchoalveolar lavage fluid wasthen collected, which showed gram positive bacilli with weakly positive modifiedacid-fast staining. The pathogen was identified as N. cyriacigeorgica after bacterialculture and mass spectrometry analysis. The patient was diagnosed withpulmonary sequestration complicated with N. cyriacigeorgica infection, and hersymptoms quickly improved following anti-infective therapy.CONCLUSION Nocardiosis is considered to be an opportunistic infection. This is the first reportof pulmonary sequestration complicated with N. cyriacigeorgica infection in apatient with normal immunity.

Treatment for hemoptysis after percutaneous closure of a large intralobar pulmonary sequestration in elderly patient

Pulmonary sequestration is a rare congenital cardiovascular malformation.[1,2]Open surgical excision is the usual treatment,although thorascopic intervention and transcatheter embolization have been reported.However,only a limited number of articles involving percutaneous closure the rare pulmonary malformations have been published in the literature.[3]What’s more,previous studies involving percutaneous pulmonary sequestration closure with repeated respiratory symptoms at the very young age suggested that the optimal therapeutic strategy has yet to be determined.[4]We hereby describe a particular case of percutaneous closure of a large intralobar pulmonary sequestration at elderly age.

Posterior mediastinal extralobar pulmonary sequestration misdiagnosed as a neurogenic tumor:A case report

BACKGROUND Pulmonary sequestration-both intralobar and extralobar-is a rare congenital developmental malformation.Extralobar pulmonary sequestrations(EPS)have their own pleura but are separated from the bronchus and usually occur in the left lung.They are mainly found mainly between the lower lobe and the mediastinum.EPS is rarely found within the mediastinum itself,even rarer so in the posterior mediastinum.CASE SUMMARY We report the case of a 27-year-old man who was misdiagnosed with a neurogenic tumor based on preoperative contrast-enhanced computed tomography(CT)and magnetic resonance imaging findings.Contrast-enhanced chest CT revealed a posterior mediastinal mass measuring 1.2 cm×1.4 cm×3.3 cm,which consisted of some cystic areas and showed slight enhancement.The mass was in the 11th paravertebral region and attached to the 11th thoracic vertebra behind the descending aorta in the posterior mediastinum.An arteriole originating from the intercostal artery and a vein originating directly from the hemiazygos vein were found in the pedicle of the mass.The mass was resected in a uniport videoassisted thoracoscopic surgery.During the operation,the pyramid-shaped mass appeared well-encapsulated.Postoperative histopathology established a diagnosis of EPS.One month later,a follow-up CT of the thorax showed good recovery.CONCLUSION Although EPS rarely occurs in the posterior mediastinum,its diagnosis should be considered when posterior mediastinal tumors are suspected.

Relationship between intralobar pulmonary sequestration and type A aortic dissection:A case report

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.

Intralobar Pulmonary Sequestration with Rete Mirabile as Part of Its Systemic Artery

Intralobar sequestration (ILS) is a complex lung lesion where part of a lower lobe lacks communication with the tracheobronchial tree and receives an aberrant arterial blood supply from the systemic circulation. That systemic artery is usually large, originating from the thoracic aorta or the abdominal aorta and its upper branches. We describe a case of ILS where a large systemic artery coming up from below the diaphragm is formed by the convergence of many very small serpiginous arteries coming up from the area of the celiac trunk and common hepatic artery: a kind of “rete mirabile”. This peculiar morphology was not described before.

Patterns of Blood Supply and Venous Drainage in Pediatric Intralobar Pulmonary Sequestration: A Retrospective Analysis of 30 Pediatric Cases from a Single Center

Purposes: Intralobar pulmonary sequestration is a rare congenital anomaly that causes nonspecific respiratory symptoms. This report presented our surgical experience for pediatric cases of intralobar pulmonary sequestration in our institute. Methods: Between 2002 and 2014, 30 pediatric patients with intralobar pulmonary sequestration underwent lobectomy. The clinical characteristics of patients were retrospectively analyzed using medical records and imaging data. Results: The median age at operation was 23 months. The sequestered lung was situated in the right lower lung in 15 cases and in the left lower lung in 15 cases. Eight cases were prenatally diagnosed by fetal ultrasound and magnetic resonance imaging. One aberrant artery was seen in 26 cases and two aberrant arteries in 4 cases. The drainage vein from the sequestered lung was: 1) the pulmonary vein in 27 cases;2) the inferior vena cava in 1 case;and 3) the azygos vein and the pulmonary vein in 2 cases. All patients underwent lower lobectomy of the affected side without postoperative early and long-term complications. Conclusion: Due to the increased rate of prenatal diagnoses, the age at operation has decreased. With respect to safety, it is vital to ascertain the number of aberrant arteries and the venous drainage route before lobectomy.

A Rare Case of Diaphragmatic Extralobar Pulmonary Sequestration

Context: Bronchopulmonary sequestration is defined as a congenital malformation in which a portion of lung tissue does not communicate with the normal bronchial tree and the pulmonary arteries. Case Report: We present a case of a 35-year-old male complaining of left lower back pain and with a mass in the left thoracoabdominal transition. Imaging studies have suggested a pulmonary sequestration and its diagnosis was confirmed by histopathological study. Conclusion: Diaphragmatic extralobar pulmonary sequestration is a rare anomaly whose radiological diagnosis is difficult. This case is illustrative and demonstrates the importance of imaging methods to the correct diagnosis and effective treatment.

联合应用PS、HFOV和iNO治疗新生儿重度呼吸衰竭的疗效观察

目的探讨联合应用肺表面活性物质(PS)、高频振荡通气(HFOV)、吸入一氧化氮(iNO)治疗新生儿重度呼吸衰竭的疗效。方法选取2013年2月至2016年2月间武汉市儿童医院新生儿重症监护室收治的86例重度呼吸衰竭新生儿,均采用PS、HFOV、iNO联合治疗。观察比较患儿治疗前后酸碱度(pH)、二氧化碳分压(PaCO_2)、氧分压(PaO_2)动脉血气分析指标,吸入氧浓度(FiO_2)、平均气道压(MAP)、氧合指数(OI)呼吸功能指标,并观察临床疗效及并发症发生情况。结果在86例患儿中,总有效率为76.7%(66/86),并发症的发生率为4.7%(4/86)。治疗前与治疗后1、6、24h患儿的pH、PaCO_2、PaO_2、FiO_2、MAP、OI比较差异均具有统计学意义(F值分别为19.064、63.158、42.790、62.163、17.737,38.944,均P<0.05);且与治疗前相比,治疗后1、6、24h患儿的pH、PaO_2均升高,PaCO_2、FiO_2、MAP、OI均下降,差异均具有统计学意义(t=-18.354~32.403,均P<0.05)。结论联合应用PS、HFOV和iNO治疗新生儿重度呼吸衰竭的疗效显著,可有效改善患儿的动脉血气分析指标和呼吸功能指标,值得应用推广于临床。

闭环通气模式适应性支持通气与智能化脱机Smart Care/PS对COPD患者机械通气脱机的疗效对比研究

目的比较闭环通气模式适应性支持通气与智能化脱机Smart Care/PS对慢性阻塞性肺疾病(COPD)患者机械通气脱机的疗效。方法选取接受有创机械通气的70例COPD患者随机分为A组和B组各35例,A组适应性支持通气脱机法,B组智能化脱机Smart Care/PS脱机,比较疗效。结果 A组脱机时间、总机械通气时间明显短于B组(P<0.01),1次脱机成功率高于B组(P<0.05),脱机后无创通气率、住院期间患者病死率两组比较差异无统计学意义(P>0.05)。结论闭环通气模式下对机械通气的COPD患者脱机,适应性支持通气脱机较智能化脱机Smart Care/PS脱机时间更短、脱机成功率更高。

PS联合BiPAP治疗NRDS临床疗效及对新生儿肺氧合功能 肺泡灌洗液Bcl-2 Caspase-3和TGF-β1水平的影响

目的 探讨肺表面活性物质联合双水平持续气道正压通气治疗新生儿呼吸窘迫综合征的临床效果及对新生儿肺氧合功能、肺泡灌洗液Bcl-2、半胱氨酸蛋白酶-3和转化生长因子-β1水平的影响。方法 将142例新生儿呼吸窘迫综合征患儿按治疗方法不同分为两组,观察组(66例)采用肺表面活性物质联合双水平持续气道正压通气治疗,对照组(76例)采用肺表面活性物质联合持续气道正压通气治疗。比较两组一般治疗指标、肺氧合功能及肺泡灌洗液Bcl-2、半胱氨酸蛋白酶-3、转化生长因子-β1水平,并统计两组并发症及预后转归状况。结果 观察组上机时间、氧疗时间、总住院时间均显著短于对照组(P<0.05或0.01),总住院费用显著少于对照组(P<0.01),治疗成功率显著高于对照组(P<0.05),两组有创通气、外源性肺表面活性物质时龄、二次应用外源性肺表面活性物质占比比较差异无统计学意义(P>0.05)。治疗后各时点两组动脉血氧分压/吸入氧浓度均较治疗前显著升高(P<0.05),氧合指数、呼吸指数均较治疗前显著下降(P<0.05),观察组治疗后各时点动脉血氧分压/吸入氧浓度显著高于对照组(P<0.05),氧合指数、呼吸指数显著低于对照组(P<0.05)。治疗后观察组肺泡灌洗液Bcl-2水平显著高于对照组(P<0.01),半胱氨酸蛋白酶3和转化生长因子-β1水平显著低于对照组(P<0.01)。治疗后观察组鼻损失、腹胀发生率显著低于对照组(P<0.05),两组气漏、支气管肺发育不良、颅内出血、肺出血、通气失败及死亡发生率比较差异均无统计学意义(P>0.05)。结论 在应用肺表面活性物质治疗基础上无论是应用持续气道正压或双水平持续正压通气治疗新生儿呼吸窘迫综合征均可取得满意效果,但双水平持续正压通气对患儿肺氧合功能的改善作用、肺泡细胞凋亡的抑制作用、机体炎症反应的缓解作用均更为显著,并能在一定程度上缩短上机时间、氧疗时间。

不同剂量PS治疗新生儿呼吸窘迫综合征的临床研究

目的比较不同剂量牛肺表面活性物质(PS)治疗新生儿呼吸窘迫综合征(NRDS)的疗效。方法选择2009年1月至2014年9月西电集团医院新生儿科收治的44例NRDS患儿,随机分为3组,均予国产牛肺表面活性物质治疗,首剂大剂量组100mg/kg为15例,中剂量组70mg/kg为14例,小剂量组40mg/kg为15例。比较3组的动脉血氧分压(PaO_2)、肺泡动脉氧分压差[P(A-a)O_2]、动脉血二氧化碳分压(PaCO_2)、氧合指数(OI)、用药后通气时间、总用氧时间、肺部X线改变及常见并发症的发生率等。结果 3组患儿治疗前一般情况无差异,应用PS治疗1h后,PaO_2、P(A-a)O_2较使用前明显好转,且组间比较差异有统计学意义(F值分别为5.752、7.184,均P<0.05);用药6h后,各组血气分析指标均出现显著改变,大剂量组PaCO_2和OI均低于中、小剂量组(F值分别为6.215、8.341,均P<0.05);用药12h及24h后X线评分,大剂量组亦明显优于中、小剂量组(F值分别为8.233、6.865,均P<0.05)。应用PS次数、氧疗时间、通气时间、住院时间、治愈率3组比较差异均有统计学意义(x^2=7.325;F值分别为8.295、4.598、7.155;x^2=5.126,均P<0.05)。各组均无严重不良反应发生。结论用PS治疗NRDS时,尽可能满足首剂≥70mg/kg是安全有效的。

机械通气联合PS对早产儿RDS的治疗效果

目的:探讨机械通气下肺表面活性物质(PS)开始使用时间对早产儿呼吸窘迫综合征(RDS)的治疗效果。方法:100例机械通气联合PS治疗的RDS的早产儿分为A组(PS开始治疗时间为新生儿出生后小时龄<6 h)、B组(PS开始治疗时间为新生儿出生后小时龄≥6 h~<12 h)和C组(PS开始治疗时间为新生儿出生后小时龄≥12 h),收集并比较3组早产儿的临床资料及转归情况;于PS治疗前、治疗后3~6 h、12 h及24 h时取各组患儿左手桡动脉血,检测血气指标pH值、动脉氧分压(PaO2)及动脉二氧化碳分压(PaCO2),并计算氧合指数(PaO2/FiO2)。结果:PS治疗后,3组患儿的pH值、PaO2及PaO2/FiO2较用药前上升,PaCO2较用药前下降,且以A组改变最明显(P<0.05);A组早产儿的插管呼吸机使用率、肺部感染率、BPD发生率及病死率均低于C组(P<0.05)。结论:呼吸机辅助通气联合PS治疗对RDS患儿有效,尽早使用PS效果更好。

PS应用于新生儿奶汁吸入性肺炎治疗的临床疗效观察

目的探讨PS在新生儿奶汁吸入性肺炎中的应用。方法以使用PS气管内注入治疗的10例新生儿奶汁吸入性肺炎为研究组,以同期拒绝使用PS治疗的8例新生儿奶汁吸入性肺炎病例为对照组,对照组仅给予预防感染、营养支持等常规治疗,研究组在常规治疗的基础上于入院2 h内给予PS气管内注入治疗,定时监测两组Pa O2、Pa CO2和p H值变化,统计并比较两组氧疗时间、无创呼吸支持例数和住院天数。结果研究组应用PS后6 h、24 h监测Pa O2、Pa CO2和p H值较用药前显著改善,两组对应时间点差异有统计学意义(P<0.05)。研究组氧疗时间、NCPAP治疗例数及住院天数均优于对照组(P<0.05)。两组患儿合并肺部感染、气漏、支气管肺发育不良及死亡情况比较差异无统计学意义(P>0.05)。结论 PS应用于新生儿奶汁吸入性肺炎治疗可取得显著疗效。

Solitary AAH Arising from Extralobar Sequestration in A Less Than 3-year-old Boy: A Case Report

We present a case of two-year old boy with solitary atypical adenomatous hyperplasia(AAH)in extralobar sequestration(ELS),which was misdiagnosed as diaphragmatic hernia before surgery.Review of AAH and pulmonary sequestration(PS)revealed that the present case is the youngest of solitary AAH and also the first report of solitary AAH arising in ELS without a primary lung cancer.In a sense,the present case firstly supports the hypothesis that ELS may be an underlying cancer predisposition syndrome,so aggressive surgical therapy should be recommended for ELS.