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1.
Medicine (Baltimore) ; 99(20): e20052, 2020 May.
Artigo em Inglês | MEDLINE | ID: mdl-32443310

RESUMO

INTRODUCTION: Pulmonary sequestration (PS) is a rare pulmonary congenital malformation characterized by disconnection with the tracheobronchial tree or the pulmonary arterial blood supply thus impeding the connection to the arterial blood supply from systemic circulation, ultimately resulting in a non-functional lung. PATIENT CONCERNS: A 73-day-old boy with rhabdomyomatoid hyperplasia was hospitalized for cough and fever 2 months after birth. DIAGNOSES: Routine B-ultrasound revealed a cystic malformation in the right lung. CT showed increased volume of the right lung accompanied with cystic low-density shadows of different sizes and a blood vessel leading from the abdominal aorta into the lesion lung. Thus, he was diagnosed with PS. INTERVENTIONS: The boy underwent an entire lobectomy of the lower lobe of the right lung. The operation field revealed several malformed blood vessels from the apposition to the right lower lobe. Most of the lung had cystic adenomatoid malformations. Other parts consisted of well-differentiated cystic dilated bronchus and striated muscles. Histopathological examination revealed rhabdomyus fibroblasts distributed throughout the bronchi. Nuclei were located at the margins of the cells. Striated muscle fibers observed as immunological markers (myogenin and desmin) of striated muscles were positively detected. OUTCOMES: The boy recovered uneventfully after his operation and no abnormal clinical findings were found at the 10-month follow-up. CONCLUSION: PS can not only accompany other developmental abnormalities, but can also associate with rhabdomyomatoid hyperplasia. This lesion is not a fatal congenital malformation and instead is a benign lesion affecting only a single lobe. It has a relatively good prognosis.


Assuntos
Sequestro Broncopulmonar/diagnóstico por imagem , Sequestro Broncopulmonar/cirurgia , Sequestro Broncopulmonar/complicações , Malformação Adenomatoide Cística Congênita do Pulmão/complicações , Malformação Adenomatoide Cística Congênita do Pulmão/diagnóstico por imagem , Malformação Adenomatoide Cística Congênita do Pulmão/cirurgia , Humanos , Lactente , Pulmão/diagnóstico por imagem , Pulmão/cirurgia , Masculino , Tomografia Computadorizada por Raios X , Ultrassonografia
2.
Rev. Fac. Cienc. Méd. Univ. Cuenca ; 37(3): [63-68], Dic.2019. ilus; tab.
Artigo em Espanhol | LILACS | ID: biblio-1100438

RESUMO

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)


Assuntos
Humanos , Feminino , Adulto , Sequestro Broncopulmonar/complicações , Sequestro Broncopulmonar/diagnóstico por imagem , Pneumonectomia/efeitos adversos
10.
Semin Pediatr Surg ; 28(4): 150821, 2019 Aug.
Artigo em Inglês | MEDLINE | ID: mdl-31451177

RESUMO

Diagnosis and management of congenital lung malformations has evolved dramatically over the past several decades. Advancement in imaging technology has enabled earlier, more definitive diagnoses and, consequently, more timely intervention in utero or after birth, when indicated. These advancements have increased overall survival rates to around 95% from historical rates of 60%. However, further refinement of diagnostic technique and standardization of treatment is needed, particularly as the increased sensitivity of diagnostic imaging results in more frequent diagnoses. In this article, we provide an updated review of the diagnostic strategies, management, and prognosis of congenital lung malformations.


Assuntos
Cisto Broncogênico/diagnóstico por imagem , Sequestro Broncopulmonar/diagnóstico por imagem , Malformação Adenomatoide Cística Congênita do Pulmão/diagnóstico por imagem , Enfisema Pulmonar/diagnóstico por imagem , Cisto Broncogênico/congênito , Cisto Broncogênico/terapia , Sequestro Broncopulmonar/terapia , Malformação Adenomatoide Cística Congênita do Pulmão/terapia , Humanos , Imagem por Ressonância Magnética , Cuidado Pós-Natal , Diagnóstico Pré-Natal , Enfisema Pulmonar/congênito , Enfisema Pulmonar/terapia , Ultrassonografia Pré-Natal
11.
Curr Med Sci ; 39(4): 658-662, 2019 Aug.
Artigo em Inglês | MEDLINE | ID: mdl-31347005

RESUMO

This study aimed to evaluate the prognosis of pulmonary sequestration (PS) by measuring congenital cystic adenomatoid malformation volume ratio (CVR) value in fetal congenital PS. The fetal CVR in 49 cases of fetal PS diagnosed by prenatal ultrasound in Xiangyang No. 1 People's Hospital from March 2010 to June 2017 were measured, and the clinical outcomes were observed. According to the prenatal ultrasound CVR value, 49 fetuses diagnosed with PS were divided into 2 groups: group 1 with CVR≥1.26, and group 2 with CVR<1.26. The incidence rate of fetal edema, respiratory distress symptoms and survival rate were compared between the two groups. The risk factors of the fetal PS were evaluated by single and multiple Logistic regression analysis. The correlation between CVR and fetal prognosis was analyzed. Of the 49 fetuses, there were 34 cases of PS (ILS) type (69.39%, 34/49), 10 cases of PS (ELS) type I (20.41%, 10/49) and 5 cases of PS (ELS) type II (10.20%, 5/49). Forty-six cases (93.88%, 46/49) were born alive, there was 1 case (CVR ≥1.26) (2.04%, 1/49) of induced abortion, and 2 cases (CVR ≥1.26) (4.08%, 2/49) of stillbirths. In group 1 (n=24), 21 cases were born alive, and the incidence rate of newborn respiratory distress and fetal edema was 100% (21/21) and 79.17% (19/24) respectively. In group 2 (n=25), there were 3 cases (12%,3/25) of newborn respiratory distress, 3 cases (12%, 3/25) of fetal edema, and the rate of live birth was 100%. There were statistically significant differences between the two groups in the incidence of fetal edema, postpartum respiratory symptoms and survival rate. CVR was a risk factor for PS and was associated with fetal prognosis. CVR in the midtrimester of pregnancy is an effective index to evaluate the prognosis of fetal PS. CVR ≥1.26 is associated with an increased risk of fetal edema, infant respiratory distress and intrauterine or postnatal death.


Assuntos
Sequestro Broncopulmonar/diagnóstico , Malformação Adenomatoide Cística Congênita do Pulmão/diagnóstico por imagem , Feto/diagnóstico por imagem , Pulmão/diagnóstico por imagem , Sequestro Broncopulmonar/diagnóstico por imagem , Sequestro Broncopulmonar/patologia , Malformação Adenomatoide Cística Congênita do Pulmão/fisiopatologia , Feminino , Feto/fisiopatologia , Humanos , Recém-Nascido , Nascimento Vivo , Pulmão/fisiopatologia , Gravidez , Prognóstico , Resultado do Tratamento
12.
J Med Case Rep ; 13(1): 228, 2019 Jul 27.
Artigo em Inglês | MEDLINE | ID: mdl-31349794

RESUMO

BACKGROUND: The potential complications of pulmonary sequestration are serious and may include recurrent pulmonary infection, hemoptysis, and tumorigenesis. Therefore, the gold standard of treatment has been surgery. Although an adequate boundary between the nonfunctional lung and normal lung is required for the resection of pulmonary sequestration, the boundaries have been conventionally identified intraoperatively with inflation/deflation of the target segment by clamping and unclamping the relevant bronchus. The technique of visualizing the demarcation line based on near-infrared fluorescence imaging with indocyanine green was recently developed. CASE PRESENTATION: A 42-year-old Japanese woman with right Pryce III intralobar sequestration was admitted to our hospital. We planned video-assisted thoracoscopic wedge resection of the right sequestration using near-infrared fluorescence imaging with indocyanine green because of the small volume of the nonfunctional region. The aberrant artery was recognized in the pulmonary ligament; the artery was cut off after ligation. Indocyanine green at 5 mg/body was rapidly injected into the peripheral vein, and the boundary of the sequestration was clearly identified under near-infrared fluorescence imaging. CONCLUSION: Near-infrared fluorescence imaging with indocyanine green is safe and useful for the identification of the boundary of a pulmonary sequestration.


Assuntos
Sequestro Broncopulmonar/cirurgia , Pneumonectomia/métodos , Cirurgia Torácica Vídeoassistida/métodos , Sequestro Broncopulmonar/diagnóstico por imagem , Sequestro Broncopulmonar/patologia , Corantes/administração & dosagem , Feminino , Humanos , Verde de Indocianina/administração & dosagem , Margens de Excisão , Pessoa de Meia-Idade , Tomografia Computadorizada por Raios X
14.
Am J Perinatol ; 36(S 02): S99-S105, 2019 07.
Artigo em Inglês | MEDLINE | ID: mdl-31238368

RESUMO

OBJECTIVE: This study was aimed to evaluate effectiveness of lung ultrasound (LUS) in the management of congenital pulmonary airway malformation and pulmonary sequestration in NICUs. STUDY DESIGN: This is a nonconsecutive case series of neonates admitted to the academic NICU of Policlinico of Bari, Italy, from 2010 to 2018, for suspected lung malformations and examined by LUS. RESULTS: Seven neonates were admitted for suspected pulmonary malformations, four neonates were diagnosed with pulmonary sequestration and three with congenital pulmonary airway malformation either type I (two cases) or type II (one case) according to Adzick classification. Prenatal scans had described lung malformations in six patients. Two underwent surgical intervention during the 1st month of life. All were successfully discharged home and their follow-up has been uneventful thereafter. In all the seven neonates, LUS easily detected the lesion showing a significantly high correspondence with computed tomography (CT) scan findings. CONCLUSION: We described the first case series of neonates affected by complex pulmonary malformations, assessed by LUS. In our experience, LUS was safe and effective for the diagnosis with high degree of consistency with CT scan findings. We suggest that LUS might be an important diagnostic method for lung malformations in newborns and a useful technique for their follow-up and late management, avoiding multiple exposures to radiations.


Assuntos
Sequestro Broncopulmonar/diagnóstico por imagem , Hérnias Diafragmáticas Congênitas/diagnóstico por imagem , Pulmão/anormalidades , Pulmão/diagnóstico por imagem , Ultrassonografia , Humanos , Recém-Nascido , Masculino , Ultrassonografia/métodos , Ultrassonografia Pré-Natal
15.
J Cardiothorac Surg ; 14(1): 113, 2019 Jun 20.
Artigo em Inglês | MEDLINE | ID: mdl-31221172

RESUMO

BACKGROUND: Extralobar sequestration is a rare congenital malformation of lung tissue, which can be combined with other foregut and cardiac abnormalities. Our case is the first to report extralobar sequestration, absence of pericardium and atrial septal defect in the same patient. CASE PRESENTATION: A 22-year-old female with atrial septal defect came for her recent atypical symptom of intermittent palpitation and shortness of breath. Her computed tomography showed a cystic mass located in left superior anterior mediastinum near the pulmonary trunk. With specious of cystic teratoma prior to video-assisted thoracoscopic surgery, she finally was diagnosed as extralobar sequestration, while incidentally found with congenital absence of pericardium during surgery. CONCLUSIONS: Extralobar sequestration, absence of pericardium and atrial septal defect can occur in the same patient. The preoperative diagnostic rate of extralobar sequestration and asymptomatic absence of pericardium remains low attributed to atypical imaging features. A cardiac magnetic resonance imaging is highly recommended if necessary. Regular follow-up is essential to asymptomatic absence of pericardium and atrial septal defect patients. To patients with extralobar sequestration, an operation could be performed.


Assuntos
Sequestro Broncopulmonar/diagnóstico , Comunicação Interatrial/diagnóstico , Neoplasias do Mediastino/diagnóstico , Pericárdio/anormalidades , Teratoma/diagnóstico , Anormalidades Múltiplas/diagnóstico , Anormalidades Múltiplas/diagnóstico por imagem , Anormalidades Múltiplas/cirurgia , Sequestro Broncopulmonar/complicações , Sequestro Broncopulmonar/diagnóstico por imagem , Sequestro Broncopulmonar/cirurgia , Diagnóstico Diferencial , Feminino , Comunicação Interatrial/complicações , Comunicação Interatrial/diagnóstico por imagem , Comunicação Interatrial/cirurgia , Humanos , Imagem por Ressonância Magnética , Neoplasias do Mediastino/complicações , Neoplasias do Mediastino/diagnóstico por imagem , Neoplasias do Mediastino/cirurgia , Teratoma/complicações , Teratoma/diagnóstico por imagem , Teratoma/cirurgia , Cirurgia Torácica Vídeoassistida , Tomografia Computadorizada por Raios X , Adulto Jovem
17.
Eur Radiol ; 29(9): 4544-4554, 2019 Sep.
Artigo em Inglês | MEDLINE | ID: mdl-30796572

RESUMO

OBJECTIVES: To compare postnatal magnetic resonance imaging (MRI) with the reference standard computed tomography (CT) in the identification of the key features for diagnosing different types of congenital lung malformation (CLM). METHODS: Respiratory-triggered T2-weighted single-shot turbo spin echo (ss-TSE), respiratory-triggered T1-weighted turbo field echo (TFE), balanced fast field echo (BFFE), and T2-weighted MultiVane sequences were performed at 1.5 T on 20 patients prospectively enrolled. Two independent radiologists examined the postnatal CT and MRI evaluating the presence of cysts, hyperinflation, solid component, abnormal arteries and/or venous drainage, and bronchocele. Diagnostic performance of MRI was calculated and the agreement between the findings was assessed using the McNemar-Bowker test. Interobserver agreement was measured with the kappa coefficient. RESULTS: CT reported five congenital pulmonary airway malformations (CPAMs), eight segmental bronchial atresias, five bronchopulmonary sequestrations (BPS), one congenital lobar overinflation, one bronchogenic cyst, and three hybrid lesions. MRI reported the correct diagnosis in 19/20 (95%) patients and the malformation was correctly classified in 22/23 cases (96%). MRI correctly identified all the key findings described on the CT except for the abnormal vascularization (85.7% sensitivity, 100% specificity, 100% PPV, 94.1% NPV, 95% accuracy for arterial vessels; 57.1% sensitivity, 100% specificity, 100% PPV, 84.2% NPV, 87% accuracy for venous drainage). CONCLUSIONS: MRI can represent an effective alternative to CT in the postnatal assessment of CLM. In order to further narrow the gap with CT, the use of contrast material and improvements in sequence design are needed to obtain detailed information on vascularization, which is essential for surgical planning. KEY POINTS: • Congenital lung malformations (CLMs) can be effectively studied by MRI avoiding radiation exposure. • Crucial features of CLM have similar appearance when comparing CT with MRI. • MRI performs very well in CLM except for aberrant vessel detection and characterization.


Assuntos
Pulmão/anormalidades , Pulmão/diagnóstico por imagem , Cisto Broncogênico/diagnóstico por imagem , Sequestro Broncopulmonar/diagnóstico por imagem , Meios de Contraste , Feminino , Humanos , Lactente , Recém-Nascido , Pneumopatias/diagnóstico por imagem , Imagem por Ressonância Magnética/métodos , Masculino , Projetos Piloto , Cuidado Pós-Natal/métodos , Reprodutibilidade dos Testes , Sensibilidade e Especificidade , Tomografia Computadorizada por Raios X/métodos
18.
Eur J Pediatr Surg ; 29(1): 49-52, 2019 Feb.
Artigo em Inglês | MEDLINE | ID: mdl-30103238

RESUMO

INTRODUCTION: Rhabdomyomatous dysplasia (RD) is a pathologic finding in CPAMs that was incorrectly attributed to their malignant potential. The increasing recognition of extrathoracic (intradiaphragmatic and intraabdominal) congenital pulmonary airway malformations (CPAMs) offers a clue to the origin of RD. We hypothesize that the presence of RD is related to the CPAM's anatomic location. MATERIALS AND METHODS: Retrospective review was performed of all children who underwent resection of a CPAM during a 10-year period. The age at the time of operation, location of the CPAM, and pathologic findings were collected. Peridiaphragmatic location was defined as within the inferior pulmonary ligament, deep to the diaphragmatic portion of the parietal pleura ("intradiaphragmatic") or adjacent to the abdominal side of the diaphragm. Statistical analysis was performed using Fisher's exact test for 2 × 2 tables. RESULTS: Twenty-six patients with CPAM were identified. Preoperative imaging was performed by computed tomography (CT) scan (16/26), ultrasound (5/26), magnetic resonance imaging (MRI) (1/26), and chest radiograph (4/26). The median age at resection was 15 months. Of these, 16 were pure cystic adenomatoid malformations, 4 were extralobar sequestrations, 4 were intralobar sequestrations, and 2 were bronchogenic cysts. Nine lesions were peridiaphragmatic with four being intradiaphragmatic (44%). Eight of the nine resected peridiaphragmatic lesions contained histologic evidence of rhabdomyomatous changes (89%, confidence interval [CI] 52-99%). None of the other lesions contained RD (CI 0-19%, p < 0.001). CONCLUSION: RD was seen exclusively, and in virtually all peridiaphragmatic CPAMs. While the exact significance of RD remains unclear, it may represent incorporation of striated muscle tissue associated with the developing diaphragm.


Assuntos
Diafragma/patologia , Pulmão/anormalidades , Pulmão/patologia , Cisto Broncogênico/diagnóstico por imagem , Cisto Broncogênico/patologia , Cisto Broncogênico/cirurgia , Sequestro Broncopulmonar/diagnóstico por imagem , Sequestro Broncopulmonar/patologia , Sequestro Broncopulmonar/cirurgia , Malformação Adenomatoide Cística Congênita do Pulmão/diagnóstico por imagem , Malformação Adenomatoide Cística Congênita do Pulmão/patologia , Malformação Adenomatoide Cística Congênita do Pulmão/cirurgia , Diafragma/diagnóstico por imagem , Diafragma/cirurgia , Humanos , Lactente , Pulmão/diagnóstico por imagem , Pulmão/cirurgia , Estudos Retrospectivos
19.
J Pediatr Surg ; 54(7): 1286-1292, 2019 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-30291025

RESUMO

OBJECTIVE: This study was performed to explore the clinical features, typing, distribution, and treatment of pulmonary sequestration (PS), with the aim of improving the awareness and treatment of this condition. METHODS: Clinical data regarding surgical procedures, outcomes, and prognosis of 208 pediatric patients with PS who were treated in our center from January 2005 to October 2017 were retrospectively analyzed. RESULTS: PS was confirmed by ultrasonography, enhanced computed tomography (CT), and/or magnetic resonance imaging (MRI) before surgery, and the surgeries were smoothly performed in all 208 patients (138 males, 70 females; age, 1 month to 14 years; mean age, 19.70 ±â€¯48.82 months). The operative time ranged from 10 to 230 min (mean, 70 ±â€¯48.75 min), and the intraoperative blood loss volume ranged from 1 to 200 ml (mean, 5 ±â€¯18 ml). PS was located in multiple sites of the thoracic cavity and was also found in some rare locations such as the neck and abdomen. The feeding arteries of the PS mainly arose from the thoracic aorta or abdominal aorta, and a few of them originated from other vessels in the systemic circulation. The venous drainage differed between intralobar and extralobar PS: in patients with intralobar PS, the venous drainage was mainly via the pulmonary veins, especially the lower pulmonary veins; in patients with extralobar PS, the venous drainage was via the azygos vein and hemiazygos vein or reached the right atrium via the vena cava. The infection rate in children with intralobar sequestration was 71.17% (79/111), and that in children with extralobar sequestration was 31.37% (16/51). CONCLUSION: PS has increasingly been detected by prenatal ultrasonography, and enhanced CT and MRI are the main techniques for diagnosing PS. Once confirmed, PS should be surgically resected. We choose an age of 6 to 12 months for surgical resection. Minimally invasive video-assisted thoracic surgery has many advantages in the treatment of PS and can be the treatment of choice for this condition. TYPE OF STUDY: Treatment Study. LEVEL OF EVIDENCE: Level III.


Assuntos
Sequestro Broncopulmonar/diagnóstico , Sequestro Broncopulmonar/cirurgia , Adolescente , Sequestro Broncopulmonar/diagnóstico por imagem , Sequestro Broncopulmonar/fisiopatologia , Criança , Pré-Escolar , Diagnóstico por Imagem , Feminino , Humanos , Lactente , Masculino , Prognóstico , Estudos Retrospectivos , Cirurgia Torácica Vídeoassistida/métodos , Toracotomia , Resultado do Tratamento
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