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Department of Radiology
Seth G.S. Medical College and K.E.M. Hospital, Mumbai , India
Introduction:
Introduction:
Pulmonary sequestration is characterized by an abnormal mass of dysplastic lung tissue supplied by an anomalous systemic artery and does not have a connection with the tracheobronchial tree [1]. The sequestrated tissue itself is often non-functional. Pulmonary sequestration can present as a major respiratory issue, often diagnosed in infancy or childhood. It can lead to respiratory distress and recurrent infections and hemoptysis especially if left untreated. Early diagnosis and surgical intervention are critical for preventing long-term complications and improving patient outcomes.
Pulmonary sequestration is characterized by an abnormal mass of dysplastic lung tissue supplied by an anomalous systemic artery and does not have a connection with the tracheobronchial tree [1]. The sequestrated tissue itself is often non-functional. Pulmonary sequestration can present as a major respiratory issue, often diagnosed in infancy or childhood. It can lead to respiratory distress and recurrent infections and hemoptysis especially if left untreated. Early diagnosis and surgical intervention are critical for preventing long-term complications and improving patient outcomes.
This is a patient with intralobar pulmonary sequestration who presented during adulthood. We have described its endovascular management.
This is a patient with intralobar pulmonary sequestration who presented during adulthood. We have described its endovascular management.
Case presentation:
Case presentation:
A 26 year old man was referred to our hospital with recurrent episodes of pulmonary infections and fever. The patient has been having persistent cough with expectoration since four years, The cough was associated with episodes of fever every 15 days. The patient did not mention seasonal or diurnal variation of these symptoms. He had been clinically diagnosed as a case of pulmonary tuberculosis and started on Anti Koch's treatment (AKT) which he received for six months at the local hospital. As symptoms persisted the AKT regimen was extended for another six months. The patient underwent bronchoscopy in February 2024 and bronchoalveolar lavage was performed. This showed gram positive cocci but not acid fast bacilli. In the following few months, the HRCT of the chest (Fig 1 and 2), that was reported as as Congenital Pulmonary Airway Malformation.
A 26 year old man was referred to our hospital with recurrent episodes of pulmonary infections and fever. The patient has been having persistent cough with expectoration since four years, The cough was associated with episodes of fever every 15 days. The patient did not mention seasonal or diurnal variation of these symptoms. He had been clinically diagnosed as a case of pulmonary tuberculosis and started on Anti Koch's treatment (AKT) which he received for six months at the local hospital. As symptoms persisted the AKT regimen was extended for another six months. The patient underwent bronchoscopy in February 2024 and bronchoalveolar lavage was performed. This showed gram positive cocci but not acid fast bacilli. In the following few months, the HRCT of the chest (Fig 1 and 2), that was reported as as Congenital Pulmonary Airway Malformation.
Figure 1, 2
Fig 1, 2: Axial section of thorax with lung window showing areas of air space opacification with cystic changes involving left lower lobe, misdiagnosing the condition as Congenital Pulmonary Airway Malformation.
Later, the patient underwent contrast enhanced CT of the chest (Fig 3 and 4, Video 1 and 2), which diagnosed as Pulmonary sequestration.
Later, the patient underwent contrast enhanced CT of the chest (Fig 3 and 4, Video 1 and 2), which diagnosed as Pulmonary sequestration.
Fig 3: Axial section of thorax showing the air space opacification with areas of cystic changes in the left lower lobe. The solid cystic lesion is supplied by the artery arising from descending thoracic aorta.
Fig 4: Coronal section of thorax showing the air space opacification with areas of cystic changes in the left lower lobe. The solid cystic lesion is supplied by the artery arising from descending thoracic aorta.
Video 1.mp4Video 2.mp4Video 1,2 : Axial section of thorax showing the air space opacification with areas of cystic changes in the left lower lobe. The feeder artery arising from descending thoracic aorta.
The patient was referred to our hospital for further management.
The patient was referred to our hospital for further management.
Interventional technique:
Interventional technique:
After assessment of all the relevant clinical history and examination with review of radiological findings, it was decided to embolise the feeder artery to cut off vascular supply to the sequestrated segment.
After assessment of all the relevant clinical history and examination with review of radiological findings, it was decided to embolise the feeder artery to cut off vascular supply to the sequestrated segment.
DSA of the aorta was performed for evaluation of the feeder arteries to sequestration and to plan embolization procedure.
DSA of the aorta was performed for evaluation of the feeder arteries to sequestration and to plan embolization procedure.
After adequate pre-procedural patient and relative counselling, obtaining of an informed consent, and pre-operative preparation-medical, surgical and anaesthesia fitness was completed.
After adequate pre-procedural patient and relative counselling, obtaining of an informed consent, and pre-operative preparation-medical, surgical and anaesthesia fitness was completed.
Right femoral arterial access was established using a 6F sheath. The aortogram showed feeder artery supplying the sequestration arising from the descending thoracic aorta at D11 vertebral level (Fig 5). The feeder artery was catheterised using a 5F Cobra catheter. A selective angiogram was performed to delineate the blood supply (Fig 6 , Video 3,4,5)
Right femoral arterial access was established using a 6F sheath. The aortogram showed feeder artery supplying the sequestration arising from the descending thoracic aorta at D11 vertebral level (Fig 5). The feeder artery was catheterised using a 5F Cobra catheter. A selective angiogram was performed to delineate the blood supply (Fig 6 , Video 3,4,5)
Fig 5: Aortogram showing an aberrant artery arising from descending thoracic aorta, supplying the sequestrated left lower lobe.
Fig 6: Selective angiogram of the aberrant artery with a 5 F C1 catheter to delineates multiple branches.
Video 3.mp4Video 4.mp4Video 5.mp4Video 3 : DSA showing aberrant artery arising from descending thoracic aorta, supplying PS in left lower lobe.
Video 4: Selective angiogram of the aberrant artery following cannulation with 5 F C1 catheter to delineate multiple branches.
Video 5 : Pulmonary angiogram to check feeders from pulmonary artery. In this case no arterial feeder arising from pulmonary artery.
After reviewing the angiographic images embolization of the feeder artery was planned to interrupt the blood supply to sequestered tissue. Through a right common femoral arterial access. with a 6F sheath, the feeder artery was cannulated using 5 F cobra catheter. A 2.7 F microcatheter was advanced through Cobra catheter and multiple branches of feeder artery were embolized using multiple steel coils (Video 6).
After reviewing the angiographic images embolization of the feeder artery was planned to interrupt the blood supply to sequestered tissue. Through a right common femoral arterial access. with a 6F sheath, the feeder artery was cannulated using 5 F cobra catheter. A 2.7 F microcatheter was advanced through Cobra catheter and multiple branches of feeder artery were embolized using multiple steel coils (Video 6).
(VID 6).mp4Video 6 : Endovascular coiling of the multiple branches of feeder artery using microcatheter.
Post embolization angiogram revealed significant reduction in vascularity and no antegrade flow (Fig 8 and 9, Video 7).
Post embolization angiogram revealed significant reduction in vascularity and no antegrade flow (Fig 8 and 9, Video 7).
Video 7.mp4Fig 8: Post embolization DSA showing no forward flow.
Fig 9: Post embolization DSA showing near complete obliteration of the feeder artery with no forward flow.
Video 7: Post embolization angiography showing near complete obliteration with no forward flow.
Discussion:
Discussion:
Pulmonary sequestration (PS) is a rare congenital lung malformation representing 0.15%- 6.4% of all congenital pulmonary malformations [1,2]. Intralobar sequestration (ILS) is more common (75% of cases) than the extralobar sequestration (ELS), roughly in a 3:1 ratio [2]
Pulmonary sequestration (PS) is a rare congenital lung malformation representing 0.15%- 6.4% of all congenital pulmonary malformations [1,2]. Intralobar sequestration (ILS) is more common (75% of cases) than the extralobar sequestration (ELS), roughly in a 3:1 ratio [2]
Almost 97% of PS cases were located in the lower lobe, and PS in the left lower lobe is two to three times more common than that in the right lower lobe [2-4]. As in our patient, the sequestration was located in the left lower lobe.
Almost 97% of PS cases were located in the lower lobe, and PS in the left lower lobe is two to three times more common than that in the right lower lobe [2-4]. As in our patient, the sequestration was located in the left lower lobe.
Multiple theories of the pathogenesis of pulmonary sequestration have been proposed, but all remain controversial. The most widely accepted hypothesis is that it results from formation of an accessory lung bud inferior to the normal lung buds during development and gets its blood supply from foregut vascular [5]. If the accessory lung bud develops before formation of the pleura, both the normal and sequestered lung tissue are covered by the same pleura, resulting in ILS; If the accessory lung bud develops after formation of the pleura, the sequestered lung tissue forms its own pleural covering, resulting in ELS [6].
Multiple theories of the pathogenesis of pulmonary sequestration have been proposed, but all remain controversial. The most widely accepted hypothesis is that it results from formation of an accessory lung bud inferior to the normal lung buds during development and gets its blood supply from foregut vascular [5]. If the accessory lung bud develops before formation of the pleura, both the normal and sequestered lung tissue are covered by the same pleura, resulting in ILS; If the accessory lung bud develops after formation of the pleura, the sequestered lung tissue forms its own pleural covering, resulting in ELS [6].
Pryce originally divided pulmonary sequestration into Intralobar and extralobar based on its pleural covering.
Pryce originally divided pulmonary sequestration into Intralobar and extralobar based on its pleural covering.
1. Intralobar Sequestration (ILS): In this form, the sequestrated tissue is contained within the normal visceral pleura of functioning lung and venous drainage is via the pulmonary veins. It is typically located in the lower lobes of the lung. Approximately two-thirds of intralobar sequestrations are found in the posterior basal segment of the left lower lobe. Intralobar sequestration is more common in older children and adults and has a reported incidence of 75% to 86% among all cases of sequestration.
1. Intralobar Sequestration (ILS): In this form, the sequestrated tissue is contained within the normal visceral pleura of functioning lung and venous drainage is via the pulmonary veins. It is typically located in the lower lobes of the lung. Approximately two-thirds of intralobar sequestrations are found in the posterior basal segment of the left lower lobe. Intralobar sequestration is more common in older children and adults and has a reported incidence of 75% to 86% among all cases of sequestration.
An exception to the left-sided preponderance is pulmonary sequestrations associated with Scimitar syndrome (partial anomalous pulmonary venous connection), where right-sided lesions are more common.[7]
An exception to the left-sided preponderance is pulmonary sequestrations associated with Scimitar syndrome (partial anomalous pulmonary venous connection), where right-sided lesions are more common.[7]
2. Extralobar Sequestration (ELS):
2. Extralobar Sequestration (ELS):
The sequestrated tissue is located outside the normal pleural space. It has its separate pleura and venous drainage. Arterial supply is from systemic vessels–most commonly descending thoracic aorta (73%) as reported by Savic et al.,[8] the cranial portion of the abdominal aorta, celiac trunk, splenic artery (21%), as well as intercostal arteries. Rare cases of arterial supply from pericardiophrenic, right coronary artery, subclavian, and internal thoracic arteries have been reported.[9]
The sequestrated tissue is located outside the normal pleural space. It has its separate pleura and venous drainage. Arterial supply is from systemic vessels–most commonly descending thoracic aorta (73%) as reported by Savic et al.,[8] the cranial portion of the abdominal aorta, celiac trunk, splenic artery (21%), as well as intercostal arteries. Rare cases of arterial supply from pericardiophrenic, right coronary artery, subclavian, and internal thoracic arteries have been reported.[9]
Venous drainage is into the systemic veins, frequently the lower lobe vein, azygos vein, or hemiazygos vein. There have been reported cases of drainage into the subclavian or portal vein as well.
Venous drainage is into the systemic veins, frequently the lower lobe vein, azygos vein, or hemiazygos vein. There have been reported cases of drainage into the subclavian or portal vein as well.
They are often found between the diaphragm and the lower lobes and rarely infra-diaphragmatically. In spite of the overall male prevalence of pulmonary sequestration, intraabdominal sequestration is most often found in women (75%), usually as a left retroperitoneal mass.
They are often found between the diaphragm and the lower lobes and rarely infra-diaphragmatically. In spite of the overall male prevalence of pulmonary sequestration, intraabdominal sequestration is most often found in women (75%), usually as a left retroperitoneal mass.
Extralobar sequestration is more commonly diagnosed in neonates and infants and may be associated with other congenital abnormalities, such as diaphragmatic hernias, congenital heart disease, or scoliosis.
Extralobar sequestration is more commonly diagnosed in neonates and infants and may be associated with other congenital abnormalities, such as diaphragmatic hernias, congenital heart disease, or scoliosis.
The diagnosis may be easily missed in adults, as many of the symptoms and the computed tomogram (CT) manifestations overlap with other pulmonary processes. So many tend to be misdiagnosed as hydatid cyst, lung abscess of lung mass.
The diagnosis may be easily missed in adults, as many of the symptoms and the computed tomogram (CT) manifestations overlap with other pulmonary processes. So many tend to be misdiagnosed as hydatid cyst, lung abscess of lung mass.
In most cases, PS has a single feeding artery, occasionally there are multiple systemic arteries supplying the PS. Arterial supply of PS mainly originated from thoracic aorta (46.1-86.1%) and abdominal aorta (6.9%-31.6%), the other feeding arteries include intercostal artery, diaphragmatic artery, aortic arch, subclavian artery, pulmonary artery, left gastric artery, coronary artery, celiac trunk and renal artery . The key to establishing the diagnosis of the PS lies in identifying the aberrant arterial supply. In our patient, the enhanced CT scan demonstrated that the single feeding vessel coursing from the thoracic aorta directly into the cystic solid mass.
In most cases, PS has a single feeding artery, occasionally there are multiple systemic arteries supplying the PS. Arterial supply of PS mainly originated from thoracic aorta (46.1-86.1%) and abdominal aorta (6.9%-31.6%), the other feeding arteries include intercostal artery, diaphragmatic artery, aortic arch, subclavian artery, pulmonary artery, left gastric artery, coronary artery, celiac trunk and renal artery . The key to establishing the diagnosis of the PS lies in identifying the aberrant arterial supply. In our patient, the enhanced CT scan demonstrated that the single feeding vessel coursing from the thoracic aorta directly into the cystic solid mass.
PS usually presents during childhood, but half of patients present after the age of 20. Usually patients present with recurrent pulmonary infection as in our case. Most patients are asymptomatic and carry the abnormality for years, only to be diagnosed during a routine physical examination or with recurrent bacterial pneumonia in the affected lower lobe. Symptoms are often nonspecific, the most common symptom of PS was cough or expectoration, fever, hemoptysis and chest pain.
PS usually presents during childhood, but half of patients present after the age of 20. Usually patients present with recurrent pulmonary infection as in our case. Most patients are asymptomatic and carry the abnormality for years, only to be diagnosed during a routine physical examination or with recurrent bacterial pneumonia in the affected lower lobe. Symptoms are often nonspecific, the most common symptom of PS was cough or expectoration, fever, hemoptysis and chest pain.
Imaging:
Imaging:
PS has diverse imaging features. Plain chest radiograph is usually nonspecific, showing an ill- defined consolidation that mimics pneumonia, or shows a solitary soft tissue mass or nodule, or a cystic or multicystic lesion [9]. On CT, the most common manifests of ILS as a mass lesion, cystic lesion that may be filled with fluid, air, or both and cavitary lesion [8,9].
PS has diverse imaging features. Plain chest radiograph is usually nonspecific, showing an ill- defined consolidation that mimics pneumonia, or shows a solitary soft tissue mass or nodule, or a cystic or multicystic lesion [9]. On CT, the most common manifests of ILS as a mass lesion, cystic lesion that may be filled with fluid, air, or both and cavitary lesion [8,9].
CT scan with intravenous contrast and preferably CT angiography (CTA) is the method of choice for identifying the arterial supply.
CT scan with intravenous contrast and preferably CT angiography (CTA) is the method of choice for identifying the arterial supply.
Treatment ;
Treatment ;
Pulmonary sequestration needs to be addressed in the event of recurrent pulmonary infection as in our case, hemoptysis, chest pain, failure to thrive, respiratory distress or shortness of breath.
Pulmonary sequestration needs to be addressed in the event of recurrent pulmonary infection as in our case, hemoptysis, chest pain, failure to thrive, respiratory distress or shortness of breath.
Pulmonary lobectomy is the treatment of choice for established pulmonary sequestration. It is recommended even in asymptomatic patients to avoid infection), and progressive inflammation of the lung parenchyma. However, in the largest retrospective case series in adults by Xiao et al., there was a lack of clear evidence of the benefit between preemptive surgery and a conservative approach in asymptomatic patients.[10]. Resection can be achieved by open thoracotomy or video-assisted thoracoscopic procedures (VATS). In recent years uniportal VATS guided lobectomy has also been successfully performed [11,12].
Pulmonary lobectomy is the treatment of choice for established pulmonary sequestration. It is recommended even in asymptomatic patients to avoid infection), and progressive inflammation of the lung parenchyma. However, in the largest retrospective case series in adults by Xiao et al., there was a lack of clear evidence of the benefit between preemptive surgery and a conservative approach in asymptomatic patients.[10]. Resection can be achieved by open thoracotomy or video-assisted thoracoscopic procedures (VATS). In recent years uniportal VATS guided lobectomy has also been successfully performed [11,12].
Surgical intervention is associated with the complications such as prolonged hospital stay, prolonged air leak, bronchopleural fistula, hemoptysis, erythema, arrhythmia.
Surgical intervention is associated with the complications such as prolonged hospital stay, prolonged air leak, bronchopleural fistula, hemoptysis, erythema, arrhythmia.
In the past few years, endovascular embolization and coiling have emerged as a therapeutic alternative.[13] Endovascular embolization reduces blood flow to the sequestered tissue leading to necrosis, fibrosis and progressive involution. Based on operators preference detachable interlock coils and pushable coils are used to pack the entire feeder vessel. Apart from micro coils, polyvinyl alcohol particles, n-butyl cyanoacrylate glue, Amplatzer plugs, gelatin, and alcohol has been used as embolic agents. A combination of agents has also been reported for use.[13][14][15] Micro coils are preferred in infants as an embolization agent because they can be delivered through smaller diameter catheters.[16] In any setting, more than one vessel needs to be embolized, and more than one coil is necessary to embolize each vessel.[17]
In the past few years, endovascular embolization and coiling have emerged as a therapeutic alternative.[13] Endovascular embolization reduces blood flow to the sequestered tissue leading to necrosis, fibrosis and progressive involution. Based on operators preference detachable interlock coils and pushable coils are used to pack the entire feeder vessel. Apart from micro coils, polyvinyl alcohol particles, n-butyl cyanoacrylate glue, Amplatzer plugs, gelatin, and alcohol has been used as embolic agents. A combination of agents has also been reported for use.[13][14][15] Micro coils are preferred in infants as an embolization agent because they can be delivered through smaller diameter catheters.[16] In any setting, more than one vessel needs to be embolized, and more than one coil is necessary to embolize each vessel.[17]
Embolization is associated with a recurrence rate of 25% to 47%. Etiology of recurrence could be incomplete closure, displacement of the embolic agent, opening of collaterals, formation of shunts. Some authors have explored the role of preoperative embolization of sequestration, to minimize bleeding risk, as the area of sequestration arteries with anomalous blood vessels with fragile wall integrity, thus prone to hemorrhagic complications during surgery.[18]
Embolization is associated with a recurrence rate of 25% to 47%. Etiology of recurrence could be incomplete closure, displacement of the embolic agent, opening of collaterals, formation of shunts. Some authors have explored the role of preoperative embolization of sequestration, to minimize bleeding risk, as the area of sequestration arteries with anomalous blood vessels with fragile wall integrity, thus prone to hemorrhagic complications during surgery.[18]
References:
References:
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