Tratamiento conservador del quilotórax posquirúrgico en paciente pediátrico / Conservative treatment of post-surgical chylothorax in pediatric patient

INTRODUCCIÓN. El quilotórax resulta de un daño al conducto torácico por ruptura, laceración, desgarro o compresión. Es una patología rara de derrame pleural en la edad pediátrica, pero frecuente como complicación posterior a cirugía cardiotorácica. La base del tratamiento conservador se ha fundamentado en: drenaje inicial, modificación de la dieta, uso de somatostatina o análogos sintéticos como octreotide, cirugía, prevención y manejo de complicaciones. Fue preciso describir la experiencia institucional clínica así como su abordaje. CASO CLÍNICO. Paciente masculino de 4 meses de edad, que ingresó a la Unidad Pediátrica Área de Emergencias del Hospital de Especialidades Carlos Andrade Marín, el 13 de septiembre de 2019 con antece-dente quirúrgico de atresia de esófago corregida en etapa neonatal. Acudió con dificultad respiratoria, radiografía de tórax que evidenció derrame pleural derecho, toracentesis diagnóstica con salida de líquido de aspecto turbio y lechoso; se colocó tubo de tórax derecho. Se prescribió ayuno inicial, nutrición parenteral durante 4 semanas hasta comprobar resolución del quilotórax. Fue dado de alta en condición estable tras 43 días de hospitalización. DISCUSIÓN. La evidencia científica registró que el tratamiento conservador del quilotórax se basó en: drenaje, reposo digestivo inicial, nutrición parenteral, modificación cualitativa de la dieta enteral y uso de octreotide; el mismo que fue aplicado al paciente de este caso clínico con evolución favorable. CONCLUSIÓN. El tratamiento conservador y multidisciplinario en el abordaje del qui-lotórax fue exitoso y no necesitó manejo quirúrgico.

INTRODUCTION. Chylothorax results from damage to the thoracic duct by rupture, la-ceration, tear or compression. It is a rare pathology of pleural effusion in pediatric age, but frequent as a complication after cardiothoracic surgery. The basis of conservative treatment has been based on: initial drainage, diet modification, use of somatostatin or synthetic analogues such as octreotide, surgery, prevention and management of complications. It was necessary to describe the clinical institutional experience as well as its approach. CLINICAL CASE. A 4-month-old male patient was admitted to the Emergency Area Pediatric Unit of the Carlos Andrade Marín Specialties Hospital on september 13, 2019 with a surgical history of esophageal atresia corrected in the neonatal stage. He went with respiratory distress, chest X-ray that showed right pleural effusion, diagnostic thoracentesis with outflow of cloudy and milky fluid; a right chest tube was placed. Initial fasting was prescribed, parenteral nutrition for 4 weeks until resolution of the chylothorax was verified. He was discharged in stable condition after 43 days of hospitalization. DISCUSSION. The scientific evidence recorded that the conservative treatment of chylothorax was based on: drainage, initial digestive rest, parenteral nutrition, qualitative modification of enteral diet and use of octreotide; the same that was applied to the patient of this clinical case with favorable evolu-tion. CONCLUSION. Conservative and multidisciplinary treatment in the approach to chylothorax was successful.

Tratamiento de quilotórax y quiloascitis refractaria en paciente cirrótico. Caso clínico

Chylous Ascites (CA) and chylothorax (CTx) are associated with obstruction, disruption or insufficiency of the lymphatic system. We report a 68-year-old male, with a history of alcoholic cirrhosis, who had recurrent events of CTx and CA. After a complete study, no other etiologies other than portal hypertension were found. Therapy with diuretics, nothing per mouth, parenteral feeding plus octreotide did not relieve symptoms. A transjugular intrahepatic portosystemic shunt (TIPS) was successfully placed and pleural effusion subsided. This case shows that CA and CTx can be caused by portal hypertension and they may subside employing a multimodal management strategy.

Fetal pleural effusion in the uterus and dyspnea after birth / 中国当代儿科杂志

Neonatal chylothorax is a common cause of neonatal congenital pleural effusion and is often caused by the accumulation of chylous fluid in the thoracic cavity due to the rupture of the thoracic duct and its branched lymphatic vessels for a variety of reasons. Neonatal chylothorax caused by malignant tumors is extremely rare, and this is the first case of neonatal mediastinal neuroblastoma with chylothorax in China. The boy was found to have pleural effusion in the left thoracic cavity in the uterus, and experienced apnea at birth, as well as dyspnea and cyanosis as the main manifestations after birth. He was diagnosed with left chylothorax based on conventional biochemical analysis of pleural effusion. After the treatment including persistent chest drainage and symptomatic and supportive treatment, the drainage of the left thoracic cavity reached a volume of 90-180 mL per day. Neonatal refractory chylothorax was considered. Chest radiograph on day 13 after birth showed lesions in the upper left lung field, and contrast-enhanced plain CT scan of the chest suggested the possibility of posterior mediastinal neuroblastoma. The autopsy confirmed giant posterior mediastinal neuroblastoma (poorly differentiated), which involved the C7-T6 spinal canal and the nearby erector spinae, with a small amount of tumor tissue in the liver and both adrenal glands. Mediastinal tumor is considered the underlying cause of chylothorax in this case.

Generalidades del quilotórax en el paciente adulto / Generalities of the chylothorax in the adult patient

Resumen El quilotórax se produce ante la ruptura, desgarro u obstrucción del conducto torácico o sus afluentes principales, lo que resulta en la liberación de quilo al espacio pleural. Ocurre más frecuentemente asociado a trauma o a lesiones malignas; pero han sido descritas otras causas. El diagnóstico se obtiene mediante toracocentesis y la determinación de las concentraciones de triglicéridos y colesterol en el líquido pleural. Las complicaciones incluyen la desnutrición, inmunosupresión y compromiso respiratorio. El tratamiento puede ser conservador o agresivo en función de la situación clínica.

Abstract Chylothorax occurs when there is rupture, laceration or obstruction of the thoracic duct or its main tributaries, resulting in the release of chyle into the pleural space. It most commonly occurs from trauma or malignancy, but other causes have been described. Diagnosis involves thoracocentesis and cholesterol and triglyceride measurement in the pleural fluid. Complications include malnutrition, immunosuppression and respiratory distress. Treatment may be either conservative or aggressive depending on the clinical scenario.

Reporte de un caso de quilotórax tuberculoso / A case report of tuberculous chylothorax

El quilotórax tuberculoso es una patología infecciosa infrecuente, que se produce como consecuencia del bloqueo del conducto torácico. Su tratamiento está dirigido a combatir la infección tuberculosa. Se presenta el caso de un varón de 55 años de edad, chofer, natural de Trujillo-Perú, que acudió a emergencia por disnea progresiva y tos seca de cinco días de evolución. El examen físico reveló frémito vocal, matidez y murmullo vesicular disminuido en 2/3 inferiores del hemitórax izquierdo. La radiografía y ecografía torácica evidenciaron derrame pleural significativo, y la toracocentesis reveló quilotórax. Posteriormente, se colocó un tubo de drenaje torácico, con disminución progresiva del volumen del líquido pleural y cambios citoquímicos. Se realizó videobroncoscopía diagnóstica con aspirado broncoalveolar, revelando bacilos ácido-alcohol resistentes. El paciente recibió tratamiento antituberculoso, con evolución favorable. El quilotórax tuberculoso constituye una causa importante de quilotórax a considerar en zonas endémicas de tuberculosis. El tratamiento adecuado de la infección, conlleva a resolución de la enfermedad.

Tuberculous chylothorax is a rare infectious disease that occurs when the thoracic duct is obstructed. Treatment is directed to the tuberculosis infection. A 55-year-old male, driver, born in Trujillo (Peru) is admitted to the emergency department with increasing dyspnea and a 5-day dry cough. The physical examination revealed vocal fremitus, dullness to percussion, and a vesicular murmur that was decreased on the lower 2/3 of the left hemithorax. The X-ray and the thoracic ultrasound revealed significant left pleural effusion. The thoracocentesis drained fluid identified as chylothorax. Subsequently, a thoracic tube was placed, with a decrease in pleural fluid volume and later normalization of the cytochemical changes. Diagnostic video bronchoscopy was performed with a bronchoalveolar aspirate, revealing acid-fast bacilli. The patient received antituberculosis treatment with a favorable outcome. Tuberculous chylothorax is an important cause of chylothorax to be considered in endemic areas of tuberculosis. Proper treatment of the infection leads to resolution of the disease.

Lymphangiographic Interventions to Manage Postoperative Chylothorax / 대한흉부외과학회지

BACKGROUND: Postoperative chylothorax may be caused by iatrogenic injury of the collateral lymphatic ducts after thoracic surgery. Although traditional treatment could be considered in most cases, resolution may be slow. Radiological interventions have recently been developed to manage postoperative chylothorax. This study aimed to compare radiological interventions and conservative management in patients with postoperative chylothorax.METHODS: We retrospectively reviewed periprocedural drainage time, length of hospital stay, and nil per os (NPO) duration in 7 patients who received radiological interventions (intervention group [IG]) and in 9 patients who received conservative management (non-intervention group [NG]).RESULTS: The baseline characteristics of the patients in the IG and NG were comparable; however, the median drainage time and median length of hospital stay after detection of chylothorax were significantly shorter in the IG than in the NG (6 vs. 10 days, p=0.036 and 10 vs. 20 days, p=0.025, respectively). NPO duration after chylothorax detection and total drainage duration were somewhat shorter in the IG than in the NG (5 vs. 7 days and 8 vs. 14 days, respectively).CONCLUSION: This study showed that radiological interventions reduced the duration of drainage and the length of hospital stay, allowing an earlier return to normal life. To overcome several limitations of this study, a prospective, randomized controlled trial with a larger number of patients is recommended.

Povidone-Iodine Pleurodesis for Chylothorax in an Extremely Low Birth Weight Infant

Chylothorax, the accumulation of chyle in the pleural space, is a rare condition, but can lead to serious complications in neonates. Conservative therapy for chylothorax includes enteral feeding with medium-chain triglyceride-enriched diet or parenteral nutrition and administration of octreotide. Surgical management is considered in cases where there is no response to conservative therapy; however, the standardized approach to refractory neonatal chylothorax is still controversial. Chemical pleurodesis can be used when medical therapies for chylothorax fail, to avoid more invasive surgical procedures. We report an extremely preterm infant born at 26 weeks of gestation with refractory chylothorax after patent ductus arteriosus ligation. The infant was successfully treated with pleurodesis using 4% povidone-iodine, without long-term side effects.

Chylous Manifestations and Management of Gorham-Stout Syndrome / 대한흉부외과학회지

Gorham-Stout disease (GSD) was first described by Gorham and colleagues in 1954, but its precise mechanism and cause remain to be elucidated. In this condition, voluminous and potentially fatal chylous effusions into the thorax can occur. Herein, we describe a case of GSD in which the patient presented with massive pleural effusions and mottled osteolytic bone lesions. We performed multiple operations, including thoracic duct ligation using video-assisted thoracoscopic surgery and thoracotomic decortication, but these procedures did not succeed in preventing recurrent pleural effusion and chest wall lymphedema. After administering sirolimus (0.8 mg/m2, twice a day) and propranolol (40 mg, twice a day), the process of GSD in this patient has been controlled for more than 2 years.

Clinical characteristics of nontraumatic chylothorax in pediatric patients

PURPOSE: To evaluate clinical characteristics of pediatric nontraumaitc chylothorax and to suggest appropriate therapeutic managements. METHODS: We retrospectively reviewed medical records of 22 patients with nontraumatic chylothorax from January 2005 to December 2018 in the Children's Hospital of Seoul National University. We analyze their etiology, treatment, complications and outcome. RESULTS: Of the 22 patients, 16 were diagnosed before 1 year old and 6 after 1 year old. The causes of chylothorax under 1-year-old children were related to congenital factors (n=9), unknown causes (n=5), and high central venous pressure (n=2). The causes of chylothorax over 1-year-old children were related to congenital factors (n=3), unknown causes (n=1), high venous pressure (n=1), and lymphoma (n=1). All patients had dietary modification. Eight of them were cured by dietary modification, but there was no improvement in over 1-year-old children. Medication was added to patients refractory to dietary modification. Four patients with medication were improved and 5 were improved by surgical management. Nutritional, immunological and other complications occurred in many patients. Five death cases were reported. Four patients were under 1 year old and 1 was over 1 year old. The causes of nontraumatic chylothorax in dead patients were high central venous pressure (n=3), congenital (n=1), and unknown (n=1). CONCLUSION: Nontraumatic chylothorax more frequently occurs in under 1-year-old children. The most common cause is congenital factors. Stepwise management is effective in many patients, but specific treatment is needed in some cases. The prognosis is related to the onset of age and underlying diseases.

Nonintubated Uniportal Video-Assisted Thoracoscopic Surgery: A Single-Center Experience

BACKGROUND: We report our surgical technique for nonintubated uniportal video-assisted thoracoscopic surgery (VATS) pulmonary resection and early postoperative outcomes at a single center. METHODS: Between January and July 2017, 40 consecutive patients underwent nonintubated uniportal VATS pulmonary resection. Multilevel intercostal nerve block was performed using local anesthesia in all patients, and an intrathoracic vagal blockade was performed in 35 patients (87.5%). RESULTS: Twenty-nine procedures (72.5%) were performed in patients with lung cancer (21 lobectomies, 6 segmentectomies, and 2 wedge resections), and 11 (27.5%) in patients with pulmonary metastases, benign lung disease, or pleural disease. The mean anesthesia time was 166.8 minutes, and the mean operative duration was 125.9 minutes. The mean postoperative chest tube duration was 3.2 days, and the mean hospital stay was 5.8 days. There were 3 conversions (7.5%) to intubation due to intraoperative hypoxemia and 1 conversion (2.5%) to multiportal VATS due to injury of the segmental artery. There were 7 complications (17.5%), including 3 cases of prolonged air leak, 2 cases of chylothorax, 1 case of pleural effusion, and 1 case of pneumonia. There was no in-hospital mortality. CONCLUSION: Nonintubated uniportal VATS appears to be a feasible and valid surgical option, depending on the surgeon’s experience, for appropriately selected patients.

Surgical Reconstruction for High-Output Chylothorax Associated with Thrombo-Occlusion of Superior Vena Cava and Left Innominate vein in a Neonate

We report a case of high-output chylothorax associated with thrombo-occlusion of the superior vena cava (SVC) and left innominate vein (LIV) following an arterial switch operation in a neonate. The chylothorax was resolved by 3 weeks after surgical reconstruction of the SVC and LIV using fresh autologous pericardium. We confirmed the patency of the SVC and LIV with a 1-year follow-up computed tomographic scan at our outpatient clinic.

Outcomes of the Multimodal Treatment of Malignant Pleural Mesiothelioma: The Role of Surgery / 대한흉부외과학회지

BACKGROUND: The treatment of malignant pleural mesothelioma (MPM) is challenging, and multimodal treatment including surgery is recommended; however, the role of surgery is debated. The treatment outcomes of MPM in Korea have not been reported. We analyzed the outcomes of MPM in the context of multimodal treatment, including surgery. METHODS: The records of 29 patients with pathologically proven MPM from April 1998 to July 2015 were retrospectively reviewed. The treatment outcomes of the surgery and non-surgery groups were compared. RESULTS: The overall median survival time was 10.6 months, and the overall 3-year survival rate was 25%. No postoperative 30-day or in-hospital mortality occurred in the surgery group. Postoperative complications included tachyarrhythmia (n=4), pulmonary thromboembolism (n=1), pneumonia (n=1), chylothorax (n=1), and wound complications (n=3). The treatment outcomes between the surgery and non-surgery groups were not significantly different (3-year survival rate: 31.3% vs. 16.7%, respectively; p=0.47). In a subgroup analysis, there was no significant difference in the treatment outcomes between the extrapleural pneumonectomy group and the non-surgery group (3-year survival rate: 45.5% vs. 16.7%, respectively; p=0.23). CONCLUSION: Multimodal treatment incorporating surgery did not show better outcomes than non-surgical treatment. A nationwide multicenter data registry and prospective randomized controlled studies are necessary to optimize the treatment of MPM.

Postoperative Chylothorax: the Use of Dynamic Magnetic Resonance Lymphangiography and Thoracic Duct Embolization

Dynamic enhanced magnetic resonance lymphangiography can be used to provide anatomic and dynamic information for various lymphatic diseases, including thoracic duct injury, and can also help to guide the thoracic duct embolization procedure. We present a case of postoperative chylothorax demonstrated by dynamic enhanced MR lymphangiography. In this case, the chyle leakage site and location of cisterna chyli were clearly visualized by dynamic enhanced MR lymphangiography, thus allowing for management with thoracic duct embolization.

Incidence and treatment of chylothorax in children undergoing corrective surgery for congenital heart diseases

Abstract Introduction: Chylothorax is a lymphatic extravasation into pleural cavity and its incidence is 0.25%-5.3% in children undergoing cardiac surgery. Objective: To evaluate the incidence of chylothorax in pediatrics patients operated, linking it in each surgical intervention. Evaluate treatment types and efficiency. Methods: Retrospective study using medical records of children undergoing cardiac surgery in the Hospital do Coração between 2004 and 2014. For statistical analysis, qualitative variables by absolute frequency and relative frequency; quantitative variables, by median of 25 and 75 percentiles, as they did not present normal distribution (Shapiro-Wilk, P<0.05). The Chi-square test was used for the association between type of treatment and result. The adopted confidence level was 95%. Results: Incidence of chylothorax was 2.1% (0.9% in intracardiac surgery, 1.7% correction of patent ductus arteriosus and aortic coarctation, 8.3% Glenn's surgery, 11.8% total cavopulmonary surgery and 3% in others). Among treatments, fasting associated with total parenteral nutrition (TPN) resolved 51% of the cases. Hypoglossal diet had failed treatment and surgical referral in 22% of the cases. Fasting with TPN associated with octreotide had success in the treatment of chylothorax in a period exceeding 15 days in 78% of cases, and 3.7% were referred for surgery. Conclusion: According to the results, incidence of chylothorax was 2.18%. Treatment with fasting and TPN leads to resolutions in 86.5%, and the association with octreotide was successful in 85.1% of cases, showing an efficient option, while the treatment with hypoglossal diet had therapeutic failure in 22% of the cases in which it was used.

Recién nacido pretérmino con quilotórax congénito bilateral, una entidad infrecuente que amenaza la vida / Preterm newborn with bilateral congenital chylothorax, a rare life-threatening entity

RESUMEN El quilotórax corresponde a la acumulación de linfa en el espacio pleural. El diagnóstico se confirma por la presencia de quilomicrones en el líquido pleural, sin embargo en la práctica clínica se utilizan los criterios de Büttiker, para establecer el diagnóstico. El objetivo de esta publicación es presentar el caso de un recién nacido con quilotórax congénito bilateral, realizando una revisión actualizada del tema, que incentive la recolección de datos y la generación de guías nacionales para su abordaje estandarizado. Se trató de un recién nacido pretérmino, quien tras parto por cesárea presentó síndrome de distrés respiratorio, ameritó terapia temprana de surfactante logrando retiro de ventilación mecánica al día de vida, con posterior presentación de signos de dificultad respiratoria y hallazgo radiológico y ecográfico de derrame pleural bilateral. Se realizó toracentesis con citoquímico compatible con quilotórax. Nuevamente ameritó soporte ventilatorio invasivo, toracostomía cerrada bilateral y nutrición parenteral, con posterior evolución clínica satisfactoria. MÉD.UIS. 2017;30(1):87-92.

ABSTRACT Chylothorax refers to the accumulation of lymphatic fluid in the pleural space. Diagnosis is confirmed by the presence of chylomicrons in the pleural fluid, however in clinical practice Büttiker criteria are widely used. The aim of this publication is to present the case of a newborn with bilateral congenital chylothorax with an updated review of the literature that encourages data collection and generation of national guidelines for standardized approach. This was a newborn preterm, who after a cesarean delivery presented respiratory distress syndrome who required early surfactant therapy achieving withdrawal of mechanical ventilation on his first day, with subsequent presentation of signs of respiratory distress, and a radiological and ultrasound finding of bilateral pleural effusion. Thoracentesis was performed obtaining cytochemical compatible with chylothorax. Again required invasive ventilatory support, closed bilateral thoracostomy and parenteral nutrition, with a satisfactory clinical course. MÉD.UIS. 2017;30(1):87-92.

A Case of Congenital Lymphatic Dysplasia Complicated by Hydrops Fetalis

Congenital lymphatic dysplasia is a rare congenital maldevelopment of the lymphatic system, in which dysfunction of the lymphatic system may cause leakage of lymph fluid into the limbs and the pleural, pericardial, or peritoneal cavity. We experienced a case of hydrops fetalis with subcutaneous lymphedema, chylothorax, chylous ascites and pericardial effusion. Lymphangiography revealed a critical defect of lymphatic system. Here, we report the first case of premature infant with congenital lymphatic dysplasia confirmed by lymphangiography, which is the first reported in Korea.

Simultaneous chylothorax and chylous ascites / 영남의대학술지

Chylothorax or chylous ascites are rare manifestations of liver cirrhosis. We report a rare case of simultaneous chylothorax and chylous ascites in a patient with hepatitis B virus-related liver cirrhosis. A 76-year-old woman was referred to our hospital with a pleural effusion on her right side. She had no history of recent medical procedures, trauma or tumor. There was no evidence of mass or thoracic duct obstruction in a computed tomography scan. Pleural fluid and ascites were confirmed as chylothorax and chylous ascites by chemistry analysis. Despite thorough conservative care, there was no improvement. Pleurodesis was planned, but hepatic encephalopathy developed suddenly and she did not recover.

Simultaneous chylothorax and chylous ascites / 영남의대학술지

Chylothorax or chylous ascites are rare manifestations of liver cirrhosis. We report a rare case of simultaneous chylothorax and chylous ascites in a patient with hepatitis B virus-related liver cirrhosis. A 76-year-old woman was referred to our hospital with a pleural effusion on her right side. She had no history of recent medical procedures, trauma or tumor. There was no evidence of mass or thoracic duct obstruction in a computed tomography scan. Pleural fluid and ascites were confirmed as chylothorax and chylous ascites by chemistry analysis. Despite thorough conservative care, there was no improvement. Pleurodesis was planned, but hepatic encephalopathy developed suddenly and she did not recover.

Peripheral T-cell Lymphoma Presenting with Chylothorax / 대한내과학회지

A 72-year-old male presented with respiratory discomfort. A simple chest X-ray and abdominal computed tomography showed pleural effusion and multiple lymph node enlargement. The pleural effusion was determined by thoracentesis to be chylothorax. An inguinal lymph node biopsy showed peripheral T-cell lymphoma. Following three cycles of cyclophospamide, hydroxyl doxorubicin, vincristine, prednisolone (CHOP) chemotherapy, a partial response was observed. Chylothorax is an extremely rare complication of T-cell lymphoma. We present a case of peripheral T-cell lymphoma presenting with chylothorax. We suggest that clinicians should consider chylothorax when examining patients with lymphoma who present with atypical pleural effusion.

Chylothorax after Blunt Chest Trauma: A Case Report

Traumatic chylothorax after blunt chest trauma alone is considered rare. Our patient was a 27-year-old female who was in a motorcycle accident and sustained blunt thoracic and traumatic thoracic aortic injuries with T1–T2 vertebral subluxation. She underwent thoracic endovascular aortic repair from T4 to T9 without any thoracic or spinal surgery. On postoperative day 7, the drainage from her left chest turned into a milky-white fluid indicative of chyle leakage. The patient was treated conservatively for 2 weeks and then the chest drain was safely removed. The results show that traumatic chylothorax can be successfully managed with conservative treatment.