Case Of Pulmonary Sequestration Research Articles

An Unusual Presentation of Pulmonary Sequestration - A Case Report

Pulmonary sequestration is an uncommon congenital anomaly. It is usually diagnosed at a young age but can present in adults with recurrent episodes of pneumonia and cough. Isolated shortness of breath is an uncommon presentation of pulmonary sequestration and is rarely reported. We describe an adult male who presented with exertional dyspnea at a high altitude and was eventually diagnosed as a case of pulmonary sequestration. It is an uncommon developmental anomaly that can go undetected at a young age. In adults, it can present as isolated dyspnea, and a thorough work-up can lead to an accurate diagnosis.

"A Rare Case of Pulmonary Sequestration in a Young Male Patient"

"A Rare Case of Pulmonary Sequestration in a Young Male Patient"

Bronchopulmonary sequestration A rare congenital anomaly presented in an adult female patient

Pulmonary sequestration is a congenital anomaly of lung parenchyma without a normal connection to the tracheobronchial tree and an anomalous systemic arterial supply. The presentation of this condition is variable ranging from no symptoms to hemoptysis. We report a case of pulmonary sequestration in an adult female who presented with recurrent chest infections. Her chest x-ray showed non-homogenous opacity in left lower lobe of lung and CT scan findings were suggestive of intralobar pulmonary sequestration in left lower lobe having arterial blood supply from the descending aorta and venous drainage in left inferior pulmonary vein. She subsequently underwent resection of the left, lower lobe of the lung.

Rare Case of Pulmonary Sequestration

Pulmonary sequestration is a rare congenital lung malformation resulting in a mass of dysplastic lung tissue supplied by aberrant systemic vessel and separated from the normal bronchopulmonary tree, commonly misdiagnosed, and not treated in a timely manner, patient often exposed to unnecessary investigations and invasive procedures before diagnosis been made. We present one of our patients who was misdiagnosed and had two surgeries before diagnosis was made and successfully treated.

Perinatal Safe Management by Repeated Fetal Therapies for a Case of Pulmonary Sequestration With Fetal Hydrops

Perinatal Safe Management by Repeated Fetal Therapies for a Case of Pulmonary Sequestration With Fetal Hydrops

A LUNG WITHIN A LUNG? A CASE REPORT OF MASSIVE HEMOPTYSIS SECONDARY TO PULMONARY SEQUESTRATION

TOPIC: Lung Pathology TYPE: Medical Student/Resident Case Reports INTRODUCTION: Bronchopulmonary sequestration (BPS), is defined as a non-functional mass of lung tissue with systemic arterial blood supply, but without normal tracheobronchial communication. BPS often presents in adulthood with recurrent pulmonary infections and even fatal hemoptysis. We present a case of a 53-year-old African American male with large volume hemoptysis while hiking in upstate New York, who was found to have right lower lobe intralobar pulmonary sequestration. CASE PRESENTATION: A 53-year-old African American male with a past medical history of hypertension, hyperlipidemia, and diabetes mellitus presented with a subacute cough for one month and one episode of large volume hemoptysis while hiking. Review of systems was negative for fever, chills, chest pain, pleurisy, history of tuberculosis, weight loss, and anticoagulation use. He admitted to recreational marajuana use, but denied smoking cigarettes. Initially he was tachypneic, saturating 91% on room air, but hemodynamically stable. Computed tomography angiography (CTA) revealed right lower lobe intralobar pulmonary sequestration with internal locules of air suggesting superimposed infection. Flexible bronchoscopy revealed fresh blood in the right mainstem and lower lobe. Thoracotomy was followed by a lower lobectomy, which confirmed the diagnosis as it demonstrated an aberrant arterial supply from the descending thoracic aorta. There was no envelopment of pleura around the lung fed by the large pulsatile artery. DISCUSSION: Pulmonary airway malformations are the most common type of congenital abnormalities of the lower respiratory tract, diagnosed 1 in 10,000 to 35,000 live births [1]. An intralobar sequestration (IS) is located in a normal lobe and an extralobar sequestration (ES) is outside, with both having their own visceral pleura. Hybrid lesions are most common and have features of both. ES presents early with associated congenital malformations, whereas IS presents later with hemoptysis and recurrent pulmonary infections [2]. BPS can appear as other pathologies on CT such as a mass lesion, cyst, cavity lesion and localized emphysema [3,4]. Lobectomy should be prompt to prevent complications, such as fatal hemoptysis [5]. Shorter recovery times have been noted with video-assisted thoracoscopic surgery (VATS) [6,7]. Postoperative complications include empyema, hemoptysis, prolonged air leak, and fistula formation. Berna et al described their surgical approach in 25 patients in 2011, and all patients were doing well at long-term follow-up [7]. CONCLUSIONS: BPS can be fatal despite its rarity and innocent presentation. Radiographic ambiguity and nonspecific symptoms may delay appropriate management. It is imperative to include BPS on the initial query when managing hemoptysis. In the era of COVID-19, where hemoptysis may herald a deadly disease, understanding the broad differential of hemoptysis expedites appropriate management. REFERENCE #1: [1] Durell J, Thakkar H, Gould S, Fowler D, Lakhoo K. Pathology of asymptomatic, prenatally diagnosed cystic lung malformations. J Pediatr Surg. 2016;51(2):231-235. doi:10.1016/j.jpedsurg.2015.10.061 REFERENCE #2: [2] Van Raemdonck D, De Boeck K, Devlieger H, et al. Pulmonary sequestration: a comparison between pediatric and adult patients. Eur J Cardiothorac Surg. 2001;19(4):388-395. doi:10.1016/s1010-7940(01)00603-0 REFERENCE #3: [3] Wei Y, Li F. Pulmonary sequestration: a retrospective analysis of 2625 cases in China. Eur J Cardiothorac Surg. 2011;40(1):e39-e42. doi:10.1016/j.ejcts.2011.01.080[4] Qi W, Zhao J, Shi G, Yang F. Intralobar pulmonary sequestration displayed as localized emphysema on computed tomography image. J Cardiothorac Surg. 2017;12(1):83. Published 2017 Sep 8. doi:10.1186/s13019-017-0646-9[5] Rubin EM, Garcia H, Horowitz MD, Guerra JJ Jr. Fatal massive hemoptysis secondary to intralobar sequestration. Chest. 1994 Sep;106(3):954-5. doi: 10.1378/chest.106.3.954. PMID: 8082388.[6] Sun X, Xiao Y. Pulmonary sequestration in adult patients: a retrospective study. Eur J Cardiothorac Surg. 2015;48(2):279-282. doi:10.1093/ejcts/ezu397[7] Polaczek, M., Baranska, I., Szolkowska, M., Zych, J., Rudzinski, P., Szopinski, J., Orlowski, T., & Roszkowski-Sliz, K. (2017). Clinical presentation and characteristics of 25 adult cases of pulmonary sequestration. Journal of thoracic disease, 9(3), 762–767. https://doi.org/10.21037/jtd.2017.03.107 DISCLOSURES: No relevant relationships by Christian Castaneda, source=Web Response no disclosure on file for Rammohan Gumpeni; No relevant relationships by Sophia Ji, source=Web Response No relevant relationships by Parmjyot Singh, source=Web Response No relevant relationships by Anthony Smith, source=Web Response

Debating the embolization of a large aberrant systemic artery for pulmonary sequestration using an Amplatzer duct occluder: a case report and literature review

Here, we report two rare cases of pulmonary sequestration that were fed by large systemic arteries and embolized with a large Amplatzer duct occluder and their 3-year follow-up, and we discuss the efficacy and safety of the embolization of a large aberrant systemic artery to pulmonary sequestration using an Amplatzer duct occluder. A 9-year-old boy complained of chest pain for 1 month, and a 6-year-old boy initially complained of recurrent cough for 3 months. A series of examinations was launched to evaluate any possible malformation or abnormalities in the patients. Chest CT and CTA identified a right lower pulmonary sequestration with infection. After admission, transcatheter device occlusion was planned after essential antibiotic treatment, and postoperative infection prevention and anti-inflammatory treatment were given. In the following 2 years of follow-up, neither of the children had recurrent chest pain, cough or other related symptoms. However, the CT follow-up demonstrated that a residual mass was visible in both patients. The same chest scan section revealed slight reductions in lung lesions from 38.344 cm2 to 37.119 cm2 (3% reduction) and 14.243 cm2 to 13.178 cm2 (7.5% reduction) for each patient. No follow-up data demonstrated the long-term clinical outcomes of the residual lesion. We do not recommend that embolization be performed for large pulmonary sequestration lesions with an aberrant artery larger than 6 mm that is planned to receive a device larger than 10 mm, as their outcomes showed a higher possibility of rebuilding the vascularization network feeding the pulmonary sequestration, indicating a higher risk for long-term complications.

STARD-compliant article: Comparison of pulmonary sequestrations with thoracic and abdominal aortic arterial supply.

Pulmonary sequestrations (PS) are typically supplied by a vessel originating from thoracic aorta, or abdominal aorta. Differences in imaging features between these PS subtypes have not been described.To analyze the imaging features of PS with arterial supply from the thoracic and abdominal aorta.Retrospectively, 23 pathologically proven cases of pulmonary sequestration were analyzed and compared based on the site of feeding artery origin.In 21 cases (21/23), the PS was soft tissue density. 1 (1/23) PS was purely cystic and another heterogeneous with both cystic and solid components (1/23). In 16/23 cases, the feeding vessel(s) arose from the thoracic aorta (male:female ratio 1:7) and in 7/23 cases from the abdominal aorta (male:female ratio 4:3). Feeding vessels from the thoracic aorta were duplicated in 7/16 cases. PS location (P <.05) and size (P <.001) differed based on the origin of the feeding vessel (thoracic aorta: 14/16 left lower lobe, mean volume 962.97 mL; abdominal aorta: 3/7 left lower lobe, mean volume 1120.89 mL). The feeding arteries themselves differed in size depending on their site of origin (thoracic aorta: mean diameter 7.0mm ± 2.7 mm, mean length 44.6mm ± 10.9 mm; abdominal aorta: mean diameter 3.3mm ± 0.6 mm, mean length 103.6mm ± 34.5 mm).PS size and distribution differ depending upon the site of feeding vessel origin as does the size of the feeding vessel itself.

Pulmonary Sequestration as an Incidental Finding of Pediatric Abdominal Ultrasound - Two Cases

Abdominal ultrasound (US) is a commonly indicated examination in pediatrics. However, lung bases are often notthoroughly examined. Pulmonary sequestration is mostly detected prenatally, but also postnatally – incidentally or withthe appearance of recurrent pulmonary infections. It can be detected by US, yet our research has not found a singlereported case where sequestration was incidentally detected by US, without any previous knowledge of its existence.Here, we report on 2 such cases of pulmonary sequestrations detected during routine pediatric abdominal US. Tubularanechogenic structure above the left hemidiaphragm should bring attention to the potential diagnosis of pulmonarysequestration.

A Rare Case of Pulmonary Sequestration Masquerading as Lung Abscess

A Rare Case of Pulmonary Sequestration Masquerading as Lung Abscess

Clinical presentation and characteristics of 25 adult cases of pulmonary sequestration.

Pulmonary sequestration (PS) is a rare congenital abnormality of lung tissue. Only few series of adult cases are reported. The aim was to describe clinical characteristics in adult cases of PS and to compare outcomes in different clinical situations. Using MSD engine we searched for cases of PS that have been diagnosed between Jan 1st, 2005 and Dec 31st, 2015. Clinical data was retrospectively gathered. Statistica v.12 (StatSoft, Inc.) was used for statistical analyses. We found 25 cases (18 females, 7 males), which underwent surgery and were histologically proven. There were 22 cases of intralobar PS. 7 cases were asymptomatic, 12 had infectious history (including 3 cases of lung abscess and pleural empyema), 4 presented with hemoptysis, 2 with chest pain. The average age to undergo surgery was 38.24, in the asymptomatic group 34, in symptomatic 39.89. In the latter the symptoms preceded the surgery for 2.45-year. Great majority of sequestrations was located in lower lobes (96%), 52% on the left. Symptomatic cases were at higher than expected risk of surgical complications, comparing to asymptomatic (chi2, P=0.04). In most cases there were surgical and histological signs of infection, only in 9 cases etiological factor was determined: in 5 cases it was A. fumigatus. A 0.53-day longer post-surgical hospital stay was observed in the symptomatic group, no statistical significance was found (U-test, P=0.45). Surgical treatment of symptomatic cases of PS is characterized by slightly longer post-surgical hospital stay and higher risk of surgical complications. Fungal infections are the most likely to occur in PS.

小児肺分画症の 1 例 : ならびに本邦小児報告例 86 例の統計的観察

小児肺分画症の 1 例 : ならびに本邦小児報告例 86 例の統計的観察

乳児期に心雑音で発見された肺底区動脈大動脈起始症(いわゆるPryce I型肺葉内肺分画症)の1例

乳児期に心雑音で発見された肺底区動脈大動脈起始症(いわゆるPryce I型肺葉内肺分画症)の1例

EP09.08: Prenatal diagnosis and outcome of fetuses with echogenic lung masses

To describe the antenatal ultrasound findings and outcome of fetuses with echogenic lung masses. Ten fetuses with echogenic lung masses suspected prenatally by ultrasound were observed in a total of 2916 examinations performed in our unit from 2011 up to 2014. Eleven cases of congenital diaphragmatic hernia were excluded. There were three cases of congenital pulmonary airway malformation (CPAM). Five cases of pulmonary sequestration (PS), three of them were considered to have also CPAM. One case of congenital high airway obstruction syndrome (CHAOS) and one case of unilateral bronchial obstruction (UBO). In all cases, a displacement of the heart and abnormal lung was observed and presence of an aberrant vessel rising from the descending aorta was seen in all PS cases. In 8 cases, magnetic resonance imaging (MRI) was performed, in 7/8 helped to clarify the diagnosis. The prenatal management in all cases was expectant and in cases of CPAM and PS + CPAM, a course of corticosteroids was administered. We observed prenatal remission of the lung mass in 6 cases (complete: 4 and partial: 2). One case of PS had hydrothorax, which decreased completely along gestation. The fetus with CHAOS develop hydrops and died in utero. The fetus with UBO had a right aortic arch and during pregnancy the obstruction decreases and the lung recovered to its normal size. The fetuses with echogenic lung masses is a rare congenital abnormality (1/1,000 birth). Clinical diagnosis can be suspected by US, but the final classification may be difficult and others diagnostic tools (MRI and histology) should be employed. In most cases prognosis is good (even in a case with PS and hydrothorax), except in CHAOS. Corticosteroid therapy should be offered in cases of CPAM and PS (mixed).

Intralobar pulmonary sequestration complicating with cryptococcal infection

Pulmonary sequestration is a rare congenital malformation characterized by accessory lung tissue supplied by an aberrant systemic artery. It often presents with recurrent infections. But fungus infection is exceptional. To help other clinicians deal with patients with pulmonary sequestration complicating with cryptococcal infection. We reported a case of pulmonary sequestration complicating with pulmonary and eye cryptococcosis and did literature review. A 20-year-old immunocompetent male patient suffered from eye pain and blurred vision, and received antibiotics and dexamethasone therapy with no improvement. Bacteriology inspection and computerized tomography scan indicated a diagnosis of cryptococcal infection in both eye and lung. Vitrectomy and antifungal agents were administered. Since symptoms fluctuations, lobectomy was operated. The surgery revealed the presence of pulmonary sequestration. Literature review showed that there is no report about pulmonary sequestration complicating with cryptococcal infection so far. Pulmonary sequestration is a very important but extremely negligent cause for pulmonary cryptococcal infection.

Pryce I型肺分画症を合併した肺癌の1切除例

症例は77歳男性．慢性咳嗽精査の胸部CTで左肺S10に51 mmの腫瘤影を認めた．経気管支肺生検にて肺扁平上皮癌（cT2bN0M0-IIA）と診断され，左下葉切除術ND2a-1を施行した．手術時，下行大動脈より下葉へ流入する異常血管を認め，Pryce I型肺分画症と診断した．最終診断は肺分画症合併肺扁平上皮癌（pT2aN0M0-IB）であった．肺分画症合併肺癌は稀であり，文献的考察を含め報告する．

Role and importance of Doppler ultrasound in the diagnosis of pulmonary sequestration: Report of two adolescent cases.

Pulmonary sequestration is a rare pulmonary parenchymal anomaly. The localization of the aberrant artery and its relation with sequestration are of importance in the process of diagnosis in the preoperative period. The feeding artery can be identified by color Doppler ultrasonography. We report two cases of pulmonary sequestration in two adolescents, aged 11 and 13 years old, and this report documents the use of in which color Doppler ultrasound demonstrated the abnormal feeding artery. Color Doppler ultrasonography, a noninvasive and radiation-free imaging modality should be considered in the diagnostic algorithm of pulmonary sequestration.

術前化学療法を行った非結核性抗酸菌感染を伴った肺分画症の1例

術前化学療法を行った非結核性抗酸菌感染を伴った肺分画症の1例

Pulmonary sequestration presented as massive left hemothorax and associated with primary lung sarcoma

Pulmonary sequestration is an uncommon disease, accounting for only approximately 1.5% of all congenital pulmonary malformations. In most cases, the diagnosis is a result of accidental radiological findings; it is rarely accompanied by clinical symptoms, and is more commonly associated with other congenital malformations. Herein, we reported a case of pulmonary sequestration presented as massive left hemothorax and associated with primary lung sarcoma. A pneumonectomy via thoracotomy was attended with complete resection of sequestration and of sarcoma. The postoperative course was unremarkable, and the patient was discharged on postoperative day 11.

分画肺内に非結核性抗酸菌症を合併した肺葉内肺分画症の1例

症例は37歳女性．35歳時に乾性咳嗽が出現し，近医受診．胸部レントゲンで異常影を認め，喀痰検査でGaffky 2号，Mycobacterium avium（以下M. avium）が検出された．胸部CTでは右下葉に浸潤影と石灰化，さらに大動脈から分岐する異常血管を認め，肺葉内肺分画症（Pryce III型）に合併した非結核性抗酸菌症と診断された．まずはCAM＋RFP＋EBの3剤併用で治療し，1年8ヵ月後には排菌は消失，陰影は改善を認め，肺分画症に対する手術目的に当科紹介となった．手術は胸腔鏡下右下葉切除術を施行した．切除肺の病理所見では右肺底区近傍の分画肺に流入する異常血管を認め，分画肺内には乾酪壊死と類上皮細胞性肉芽腫の形成を認める部位があり，同部はZiehl-Neelsen染色で抗酸菌が多数同定された．肺分画症に非結核性抗酸菌症を合併した症例は稀であり，文献的考察を加え，報告する．