[Right upper lobe pulmonary sequestration as a rare cause of recurrent spontaneous pneumothorax]. / Sekvestratsiya verkhnei doli pravogo legkogo ­ redkaya prichina pravostoronnego retsidiviruyushchego spontannogo pnevmotoraksa.

A 19-year-old patient after previous wedge resection of the right upper pulmonary lobe a year ago urgently admitted with recurrent right-sided spontaneous pneumothorax. According to standard management of spontaneous pneumothorax, we performed diagnostic thoracoscopy and drainage of the right pleural cavity with regular X-ray examinations. However, these measures were ineffective. The patient was scheduled for surgery, and we intraoperatively observed an unusual cause of pneumothorax. Thus, we present spontaneous pneumothorax following right upper lobe pulmonary sequestration. The uniqueness of this case is associated with unusual manifestation and non-standard localization of rare lesion. A few cases of pneumothorax in similar patients are described in the world literature. The key limiting factor in diagnosis of such defects (identification of aberrant vessel supplying abnormal lung parenchyma) is the lack of routine CT angiography in patients diagnosed with pneumothorax. That is why CT changes were interpreted as postoperative ones, and the true cause was established only during redo surgery. A thorough inspection of the pleural cavity and alertness regarding unusual appearance of the right upper pulmonary lobe made it possible to suggest a non-standard diagnosis, avoid complications (bleeding from afferent vessel) and perform adequate lung resection.

Congenital diaphragmatic hernia with associated broncho-pulmonary sequestration: A report of two cases and a literature review.

BACKGROUND: Congenital Diaphragmatic Hernia (CDH) is a severe congenital anomaly with significant morbidity and mortality. It can be isolated or can be associated with other congenital anomalies, including broncho-pulmonary sequestration (BPS). The association of CDH with BPS (CDH+BPS) is uncommon but has been previously reported, and it can complicate the course of the disease in patients with CDH. We report two cases of CDH+BPS that were recently treated at our CDH-Qatar (CDH-Q) program. METHODS: We reviewed CDH-Q program registry to search for CDH+BPS and extracted the data for the identified cases. We also reviewed the previously published literature available on PubMed for similar cases. RESULTS: Out of 53 cases of CDH referred to CDH-Q from January 2018 to December 2022, two cases of CDH+BPS were identified, with an estimated prevalence of 3.8% of this association in our CDH population. Both cases were born at term. Case 1 was diagnosed with CDH+BPS postnatally, while case 2 was diagnosed with CDH antenatally but BPS was diagnosed after birth. Both cases underwent a surgical repair of the CDH with resection of the associated BPS, and the histopathology of the resected lung tissue confirmed the presence of BPS in both. Both cases survived to discharge. CONCLUSION: The association of CDH+BPS is uncommon; however, it can have significant consequences on the management and the prognosis of patients with CDH. Reporting these cases is important to provide a better understanding of this association and its impact on CDH patients.

Bilateral pulmonary sequestrations: Distinct types sharing a common celiac artery origin, presented with pneumonia and associated findings in a pediatric case.

A 14-year-old boy presented with shortness of breath, cough, and mild chest pain, with a history of intermittent milder symptoms. Physical examination and initial tests showed mild iron deficiency anemia, elevated C-reactive protein, and normal vital signs. Chest radiograph revealed abnormalities in the left lower zone, leading to contrast-enhanced chest CT. The CT scan revealed bilateral intrapulmonary sequestrations, both deriving blood supply from a common trunk originating from the celiac artery. The patient's symptoms initially attributed to a pulmonary infection improved with antibiotic therapy. Pulmonary sequestration is a congenital anomaly characterized by aberrant lung tissue lacking connections to bronchial tree or pulmonary arteries. It can lead to recurrent pulmonary infections and postinfectious sequelae. This case presented a unique bilateral sequestration, both originating from the celiac artery. Radiologists should be aware of sequestration types and associated anomalies, even in atypical locations. Blood supply can originate from various arteries, not just the aorta.

Unexpected pulmonary sequestration in a pregnant patient.

A pregnant woman in her early 30s, at 20 weeks of gestational age, presented with recurrent haemoptysis, pleuritic chest pain and a productive cough of 6 months duration. She underwent CT pulmonary angiogram which demonstrated right pulmonary sequestration and right-sided consolidation. Pre-existing pulmonary comorbidities such as chronic inflammation, structural abnormalities or weakened blood vessels within the lungs can encourage the growth of abnormal blood vessels. During pregnancy, these dynamics can be further aggravated by increasing cardiac output to promote blood flow to the placenta and increasing oxygen delivery to the developing foetus. These changes likely cause increased blood flow to the pulmonary sequestration, resulting in haemoptysis. The patient was treated conservatively for community-acquired pneumonia with a course of oral amoxicillin 500 mg three times a day for 5 days, and she is doing well on follow-up.

Manejo quirúrgico del secuestro pulmonar complicado. Reporte de caso / Surgical management of the complicated pulmonary sequestration. A case report

Introducción: el secuestro pulmonar es una patología poco frecuente en la edad adulta, su mayor incidencia es en etapas tempranas de la vida. Se acompaña de infecciones pulmonares a repetición o crónicas severas, y puede asociarse a malformaciones congénitas. El diagnóstico se fundamenta en la detección de la irrigación anómala a través de un vaso de la circulación sistémica. Este caso nos permite plantear un diagnóstico diferencial en pacientes de edad adulta, aún cuando se trate de patología con mayor incidencia en niños y jóvenes, y poder orientar el manejo de acuerdo a esta posibilidad diagnóstica. Caso clínico: paciente femenino, de 19 años, con antecedente de asma e infecciones respiratorias a repetición de un mes de evolución. Ingresa con dificultad respiratoria, fiebre y tos productiva. Se realiza tomografía y Radiografía de tórax, en la que se evidencia imágenes compatibles con colección en campo pulmonar izquierdo y derrame pleural. Se realiza BAAR en esputo con resultado negativo para tuberculosis. Se realiza toracotomía posterolateral izquierda, resección de lóbulo inferior izquierdo, y se coloca dren pleural. Se identifica absceso pulmonar en lóbulo inferior izquierdo, y hallazgos anatómicos de secuestro pulmonar. Paciente presenta evolución favorable; el control clínico y radiográfico en el postoperatorio inmediato y mediato fue satisfactorio. Conclusión: presentamos el caso de edad adulta, donde un diagnóstico adecuado y un manejo multidisciplinario permiten una evolución satisfactoria de los pacientes.(AU)

Introduction: pulmonary sequestration is a rare disease in adulthood; its highest incidence is in early stages of life. It is accompanied by repeated or severe chronic lung infections, and may be associated with congenital malformations. The diagnosis is based on the detection of anomalous irrigation through a vessel of the systemic circulation.This case allows us to propose a differential diagnosis in patients of adulthood, even when it is pathology with higher incidence in children and young people, and guide the management according to this diagnostic possibility.Clinical case: female patient, 19 years old, with a history of asthma and respiratory infections after a month of evolution. The patient was admitted with respiratory distress, fever and productive cough.A tomography and chest X-ray are performed; that show images compatible with collection in the left pulmonary field and pleural effusion. BAAR is performed in sputum with a negative result for tuberculosis.Posterolateral left thoracotomy is performed, left lower lobe resection, and pleural drain is placed. Pulmonary abscess is identified in the left lower lobe, and findings of anatomical pulmonary sequestration. Patient presents positive evolution; The clinical and radiographic control in the immediate and intermediate postoperative period was satisfactory.Conclusion: we present the case of adulthood, where an adequate diagnosis and multidisciplinary management allows a satisfactory evolution of the patient.(AU)

Dolor torácico y hemotórax como presentación atípica de secuestro pulmonar en adulto / Chest pain and hemothorax: an atypical presentation of a pulmonary sequestration

Resumen El secuestro pulmonar es una malformación pulmonar rara, presentándose generalmente en edades tempranas. Se presenta mayoritariamente con neumonías e infecciones repetidas, distrés respiratorio y falla cardíaca; raramente en pacientes de mayor edad se presenta con hemoptisis y dolor torácico. En este artículo se describe el caso clínico de un paciente de 60 años de edad que se presenta con un infarto de un secuestro pulmonar y hemotórax.

Bronchopulmonary sequestration is a rare pulmonary malformation, usually occurring at an early age. It presents mainly with pneumonia and repetitive infections, respiratory distress and heart failure; rarely in aged patients presents with hemoptysis and chest pain. This article describes the clinical case of a 60-year-old male patient who presented an ischemic pulmonary sequestration and hemothorax.

Secuestro pulmonar extralobular como causa de hemorragia pulmonar recurrente en un recién nacido / Extralobar pulmonary sequestration as a cause of recurrent pulmonary hemorrhage in a neonate

El secuestro pulmonar es una malformación pulmonar congenita, quística o sólida, compuesta de tejido pulmonar no funcional sin conexión con el árbol traqueobronquial y que recibe sangre arterial de la circulación sistemica. Existen dos formas de secuestro: intralobular y extralobular. El tratamiento se realiza mediante resección quirúrgica. Se describe el caso de un recién nacido prematuro con secuestro pulmonar extralobular que presentó insuficiencia respiratoria y hemorragia pulmonar recurrente. Luego de la cirugía, el paciente mejoró en forma clínicamente significativa.

Pulmonary sequestration is acystic or solid congenital lung malformation comprised of non functional lung tissue that does not communicate with the normal tracheobronchial tree and has a systemic arterial blood supply. There aretwo forms of sequestration: intralobar and extralobar. Its treatment is surgical resection. Here we presented a case of premature neonate with extralobar pulmonary sequestration who had respiratory failure and recurrent pulmonary hemorrhage. Following surgery, the patient showed significant clinical improvement.

Paciente com sequestro pulmonar intralobar: uma rara anomalia congênita / Patient with intralobar pulmonary sequestration: a rare congenital anomaly

Paciente do sexo masculino, com 51 anos de idade, com história de pneumonias de repetição desde a segunda década de vida. As radiografias de tórax curiosamente demonstravam omesmo padrão radiológico com envolvimento do lobo inferioresquerdo. Foi realizada tomografia helicoidal de tórax, que revelouvaso arterial anômalo emergindo da aorta torácica descendente em direção ao lobo pulmonar inferior esquerdo,caracterizando sequestro pulmonar. Os achados foram confirmados por aortografia torácica, sendo o paciente encaminhado para tratamento cirúrgico.

A 51-year-old male patient had a history of recurrent pneumonia since the second decade of life. Interestingly enough, chest X-rays showed the same radiological pattern with involvement of the left lower lobe. A multislice spiral CT scan of the chest was performed and depicted an anomalous arterial vessel emerging from the descending thoracic aorta towards the left lower pulmonary lobe, characterizing pulmonary sequestration. The findings were confirmed by thoracic aortography, and the patient was referred for surgical treatment.

A utilidade da TC de tórax no diagnóstico do sequestro pulmonar / Usefulness of chest CT in the diagnosis of pulmonary sequestration

O sequestro pulmonar é uma rara anomalia congênita, caracterizada por tecido pulmonar embrionário não funcionante, perfazendo 0,15-6,40 por cento de todas as malformações pulmonares congênitas. Essa anomalia envolve o parênquima e a vascularização pulmonar, sendo classificado como intralobar ou extralobar. Neste relato, descrevemos o caso de um paciente de 56 anos com hemoptise e imagem hipotransparente retrocardíaca em base de hemitórax esquerdo na radiografia de tórax. Após a realização de TC com contraste endovenoso, foi evidenciada a presença de sequestro pulmonar. O paciente foi submetido à cirurgia para a retirada do tecido anômalo, que foi realizada com sucesso. Apresentou boa evolução pós-operatória e recebeu alta com acompanhamento ambulatorial.

Pulmonary sequestration is a rare congenital anomaly, characterized by nonfunctional embryonic pulmonary tissue. Pulmonary sequestration accounts for 0.15-6.40 percent of all congenital pulmonary malformations. This anomaly, which is classified as intralobar or extralobar, involves the lung parenchyma and its vascularization. We report the case of a 56-year-old male presenting with hemoptysis. A chest X-ray showed an area of opacity behind the cardiac silhouette in the base of the left hemithorax. Chest CT scans with intravenous contrast revealed pulmonary sequestration. The patient underwent surgery, in which the anomalous tissue was successful resected. The postoperative evolution was favorable, and the patient was discharged to outpatient treatment.

Secuestro pulmonar asociado a hidrotórax, un hallazgo infrecuente: caso clínico / Sequestration pulmonary associated to hydrotorax, an infrequent find

Pulmonary sequestration is a rare lung congenital malformation that can be diagnosed in the rutine prenatal ultrasound scan. It has a wide spectrum of clinical manifestations, that range from asymptomatic fetuses to hydrops fetalis. Although prenatal Ultrasound (US) has been the traditional diagnostic tool in these cases, fetal magnetic resonance imaging (MRI) may prove useful in differential diagnosis with cistic adenomatous malformation (CCAM) and also in the evaluation of the mass efect exerted by the malformation. We present a case of a 38 year old pregnant woman with a diagnosis of fetal hydrothorax in the 29th week of gestation. Ultrasound and MRI evaluation revealed pulmonary sequestration. Management involved prenatal thoracocentesis, planned delivery and neonatal support. This case highlights the importance of a multidisciplinary approach when encountering fetal lung anomalies including radiologists and pediatricians, a thorough assessment of the lesion using US, MRI and planned delivery in a tertiary center with adecuate neonatal support.

El secuestro pulmonar es una malformación congénita pulmonar infrecuente que puede ser diagnosticada mediante el ultrasonido durante una evaluación prenatal de rutina. Sus manifestaciones clínicas van desde fetos asintomáticos hasta el hidrops fetal. Si bien la ultrasonografía ha sido el método tradicional de evaluación de estas malformaciones, la utilización de resonancia puede demostrar utilidad, en especial en el diagnóstico diferencial con malformación adenomatosa quística, como también en evaluar el efecto de compresión ejercido por la lesión, y estimar el volumen pulmonar residual. Se presenta un caso de una paciente embarazada de 38 años con diagnóstico de secuestro pulmonar a las 29 semanas y que a las 32semanas evolucionó con hidrotórax que requirió toracocentesis, interrupción programada y soporte neonatal. Este caso resalta la importancia de usar un enfoque multidisciplinario al enfrentarse con lesiones pulmonares fetales que incluya neonatólogo, radiólogo, obstetra, y una evaluación detallada de la lesión utilizando ultrasonografía y resonancia magnética como también interrupción programada en un centro terciario con adecuado soporte neonatal.

Pulmón esofágico: caso clínico / Esophageal lung: a case-report

Introducción: El pulmón esofágico es una malformación congénita muy infrecuente que se produce por una alteración en el desarrollo normal del intestino anterior. Consiste en la presencia de tejido pulmonar conectado con el esófago. Clínicamente se puede manifestar como neumonías recurrentes, dificultad respiratoria o disnea que aparece junto con la alimentación. Objetivo: Reportar el primer paciente portador de esta malformación tratado en nuestra institución. Caso Clínico: Lactante masculino, 4 meses de edad, con neumonías recurrentes del lóbulo superior derecho. El estudio con imágenes y endoscopía, confirmó la presencia de un pulmón supernumerario que estaba comunicado con el tercio medio del esófago. El paciente fue sometido a una resección quirúrgica de la malformación. Evoluciona favorablemente con mejoría de sintomatología. Conclusión: El pulmón esofágico es una malformación poco frecuente que debe ser sospechada en pacientes con sintomatología respiratoria recurrente. El estudio con imágenes y endoscopía permite precisar el diagnóstico.

Seqüestro broncopulmonar intralobar: relato de caso / Intralobar pulmonary sequestration: a case report

Neste trabalho é descrito um caso de uma paciente de 49 anos de idade que apresentava infecções pulmonares de repetição. Na telerradiografia de tórax observou-se massa no segmento basal posterior do pulmão direito, que na angiotomografia e nas reconstruções em 3D notou-se a presença de um suprimento sanguíneo proveniente da artéria aorta descendente. No exame da peça cirúrgica ratificou-se a presença de seqüestro pulmonar intralobar com cavidade preenchida por muco.

We report the case of a 49-year-old patient with repeated lung infections. Chest x-rays showed a mass in the posterior basal segment of the right lung. Angiotomography and 3D reconstructions showed a blood supply coming from the descending aorta. The analysis of the surgical specimen confirmed the occurrence of intralobar pulmonary sequestration with a cavitation filled with mucus.

Sequestro pulmonar: apresentação clínica / Pulmonary sequestration: clinical presentation

Os autores relatam o caso de um paciente do sexo masculino de 60 anos de idade, assintomático respiratório, não tabagista, cuja radiografia de tórax evidenciava a presença de um nódulo pulmonar localizado no segmento 10. Após complementação do estudo radiológico com tomografia convencional e arteriografia foi confirmado o diagnóstico de sequestro pulmonar. A seguir tecem comentários acerca do quadro clínico e radiológico desta afecção além de considerações sobre a patogenia e, finalmente, o tratamento proposto no caso

Sequestro pulmonar intra-abdominal em adulto: relato de um caso operado e revisao da literatura / Intra-adbominal bronchopulmonary sequestration in adults: a report of a surgical case and review of literature

E relatado caso incomum de sequestro pulmonar extralobar de localizacao extratoracica em paciente adulto operado com quadro de abdome agudo inflamatorio, sendo o diagnostico estabelecido pelo exame anatomo-patologico da peca cirurgica. Os AA discutem os aspectos clinicos e diagnosticos desta malformacao com enfase em sua localizacao atipica. E feita revisao de literatura.

Cancer injertado en una secuestración pulmonar: caso clínico / Ingrafted cancer in pulmonary sequestration: a clinical case

Se hace una breve descripción de lo que es la "secuestración pulmonar". Se presenta el caso clínico de un portador de secuestración pulmonar comprobada por aortografía, en el que se injerta un cáncer pulmonar comprobado histológicamente por biopsia quirúrgica. Se revisa la literatura, no encontrándose ningún caso de asociación descrito en Chile y no más de tres en la literatura mundial

Secuestro pulmonar: comentario sobre 14 casos operados / Pulmonary sequestration: experience with 14 cases operated

Se presenta una serie de 14 enfermos operados por secuestro pulmonar. En uno de ellos hay que señalar la coexistencia con secuestro extralobar. Todos ingresaron con síntomas. Se resalta la importancia de la aortografía en el diagnóstico y en la resección del secuestro

Secuestro pulmonar

En un período de 20 años, se operaron 15 casos de secuestro pulmonar (SP); 11 fueron hombres, y la edad media fue de 34 años (rango 10-56 años). Doce pacientes fueron sintomáticos. La angiografía preoperatoria practicada a trece pacientes, fue diagnóstica en todos los casos. En 86 por ciento de los pacientes el SP se localizó en el segmento basal posterior. La cirugía fue practicada en todos los casos sin mortalidad; catorce fueron intralobares (SIL) y no fue estralobar (SEL). En un paciente se encontró incidentalmente un carcinoma bronquioloalveolar. El SP aunque infrecuente es importante de tener en mente ya que puede asociarse a infecciones pulmonares crónicas severas y otras enfermedades muy incapacitantes o potencialmente letales, tales como el cáncer de pulmón. La cirugía es el tratamiento de elección, e idealmente requiere que el diagnóstico se haga preoperatoriamente, para detectar el vaso anómalo y evitar accidentes quirúrgicos que puede ser fatales; esto es ahora más fácil con la nueva tecnología no invasiva en imágenes, sin embargo un alto índice de sospecha es necesario.