Intranodal lipiodol injection for the treatment of chyle leak in children - a preliminary experience.

OBJECTIVE: To assess the effectiveness of intranodal lymphangiography using ethiodised oil (Lipiodol; Guerbet Japan, Tokyo, Japan) for the treatment of refractory cases of chylothorax and chylous ascites in the paediatric population. METHODS: Between 2016 and 2020, eight children having chyle leak resistant to conservative management underwent intranodal lymphangiography using lipiodol injection. After ethical approval by the Institutional Review Board, these patients' data were retrospectively analysed. Technical success was defined by opacification of inguinal and retroperitoneal lymphatics while injection on fluoroscopy. Clinical success was defined as progressively decreasing drain output and eventual cessation of output within a week after the procedure. Long-term follow up was done as feasible. RESULTS: Technical success was achieved in all the patients. Complete cessation of drain output was noted within 1 week of procedure in all patients indicating clinical success. One patient had recurrence of chylous leakage after an interval of 1 month and intranodal lymphangiography was repeated for that patient. The child had technical as well as clinical success after the repeat procedure. Hence a total of 9 procedures were performed in 8 patients. CONCLUSION: Intranodal lymphangiography may prove to be a valuable minimally invasive therapeutic tool in cases of refractory chylous leakage in paediatric patients with minimal risk of complications. ADVANCES IN KNOWLEDGE: Intranodal lymphangiography using lipiodol may prove to be a minimally invasive alternative in paediatric patients with refractory lymphatic leaks.

Conservative management of postoperative chylous fistula with octreotide and peripheral total parenteral nutrition.

Postoperative chylous fistula after neck dissection is an uncommon complication associated with significant patient morbidity. Octreotide acetate is a somatostatin analogue established in the treatment of chylothorax; however, its utility in the management of cervical chylous fistulae has not been fully evaluated. The investigators hypothesized that chylous fistula can be managed by a combination of octreotide and peripheral total parenteral nutrition (TPN). A retrospective review of cases compiled at our institution from 2009 to 2015 was conducted. Ten patients, all men, were identified as having a postoperative chylous fistula after a neck dissection. All patients were treated with peripheral TPN and intravenous octreotide. Mean age of the patients was 63.0 years (range 49 to 82). Five (50.0%) had a neck dissection for the management of metastatic nasopharyngeal carcinoma and had previous neck irradiation. In 8 (80%) patients, chylous fistula occurred in the left neck. Seven (70.0%) of the leaks occurred within the first 2 postoperative days. Eight (80%) leaks were controlled using TPN and octreotide, with 2 (20%) patients requiring surgical intervention. No factors were significant in the successful conservative management of chylous fistulae. One patient with a chylous fistula of 1,800 ml/day was managed successfully without surgical intervention. The results of this case series suggest that chylous fistulae may be managed conservatively with octreotide and TPN. However, long-term evaluation is needed to define if and when surgical intervention is required for control.

Cervical chylous leakage following esophagectomy that was successfully treated by intranodal lipiodol lymphangiography: a case report.

BACKGROUND: Chylous leakage is a well-known complication after esophagectomy, but cervical chylous leakage is relatively rare, and considerable controversy remains regarding the appropriate management strategies. We herein report a case of cervical chylous leakage treated successfully by lipiodol lymphangiography. CASE PRESENTATION: The patient, a 70-year-old man with middle thoracic esophageal cancer, underwent radical esophagectomy with 3-field lymph node dissection and subsequently developed cervical chylous leakage. From the second postoperative day (POD2), the amount of fluid in the cervical drainage tube increased by 200-300 ml/day. We started octreotide (300 µg/day) on POD5 and etilefrine (120 mg/day) on the POD6. However, the amount of cervical discharge did not decrease. We performed lipiodol lymphangiography on POD8. Thereafter, the amount of cervical discharge finally began to decrease. We removed the drainage tube on POD13, and the patient was discharged from the hospital on POD23. CONCLUSIONS: Our case suggests the clinical efficacy of lipiodol lymphangiography for cervical chylous leakage after esophagectomy.

Nontraumatic Chylothorax: Diagnostic Algorithm and Treatment Options.

Nontraumatic chylothorax is a relatively rare condition in which the intestinal lymph (chyle) leaks into the pleural cavity. Nontraumatic chylothorax is more difficult to treat than the more common traumatic chylothorax because the site of chylous leak may occur in less predictable locations. In the past, patients with nontraumatic chylothoraces were offered traditional fluoroscopically guided lymphangiography and thoracic duct embolization similar to traumatic chylothorax. However, the observation that thoracic duct embolization outcomes for nontraumatic chylothorax differed based on the imaging findings during lymphangiography has led to the development of a treatment algorithm, which incorporates noninvasive diagnostic studies, such as magnetic resonance lymphangiography. The development of this systematic approach allows better delineation of the source of the chylous leak and selection of the appropriate method of embolization. In this article, we will review the etiologies of nontraumatic chylothorax, the diagnostic work-up for managing this condition, and the treatment algorithm to care for these patients.

Therapeutic role of ultrasound-guided intranodal lymphangiography in refractory cervical chylous leakage after neck dissection: Report of a case and review of the literature.

BACKGROUND: Chylous leakage is a well-recognized but rare complication of head and neck surgery, affecting approximately 1% to 2.5% of head and neck dissections. It is a potentially life-threatening condition characterized by electrolyte imbalance, immunosuppression, delayed wound healing, risk of infection, and generalized sepsis. Management can be problematic and prolonged. METHODS: We present a case of refractory cervical chylous leakage after neck dissection treated with ultrasound-guided intranodal lymphangiography. RESULTS: Ultrasound-guided intranodal lymphangiography alone resulted in rapid and complete resolution of chylous leakage with minimal morbidity. CONCLUSION: Based on our clinical experience and after a thorough literature review, we propose that ultrasound-guided intranodal lymphangiography with contrast agent could be considered a viable therapeutic option for persistent chylous leakages in selected patients.

REPEATED INTRANODAL LYMPHANGIOGRAPHY FOR THE TREATMENT OF LYMPHATIC LEAKAGE.

Treatment of patients with chylous or non-chylous lymphatic leakage can be difficult. An approach using therapeutic lymphangiography can reduce the lymphatic leakage, but it seldom stops the leakage immediately and subsequent conservative treatment is necessary. We report three cases in which intranodal lymphangiography was performed multiple times to inhibit lymphatic leakage. In each case, the lymph node was punctured under ultrasound guidance using a 23-gauge needle and lipiodol was injected manually at a rate of 1 ml/3 min. The procedure was repeated twice in two cases of gastrointestinal carcinoma and four times in one case of lymphoma. In all three cases, the postoperative lymphatic leakage stopped after the repeated intranodal lymphangiography.

Good response of low-fat/high-protein diet in a patient with chyluria.

UNLABELLED: Chyluria is an inappropriate urinary excretion of chyle that turns the urine milky. A nutritional approach based on low-fat/high-protein content diet associated or not with medium-chain triglyceride (MCT) showed to be an efficient conservative treatment to improve the milky urine appearance in a patient with chyluria. CASE REPORT: A 30-year-old female patient was admitted with chyluria of unknown etiology. An ureteropyeloscopy revealed a single lesion in each kidney, both with linear aspect and measuring 5 mm in extension. These lesions were located close to the renal papillae and were leaking a cloudy and milky fluid. Both lesions were laser cauterized followed by improvement of the milky urine. However, the chyluria relapsed after few months and a low-fat/high-protein content diet with 10 g of soybean oil to meet the requirements essential fatty acids (EFA) and with MCT from coconut oil as alternative to prepare foods was started. Few weeks later the patient returned reporting consistent improvement of the milky urine appearance related with the use of the diet. However since the diet was tasteless and time consuming to prepare, she reported low compliance to diet with MCT and the milky urine relapsed. The MCT was discontinued and the diet with EFA source was maintained with better compliance. Since then the chyluria remains in remission. In conclusion, the dramatic improvement of the milky urine with low-fat/high-protein diet with EFA source observed in our patient demonstrates that this nutritional approach is efficient with fast results to treat chyluria during long term.

Lymphangiography in the diagnosis and localization of various chyle leaks.

PURPOSE: Chyle leaks are rare entities infrequently encountered by most physicians. However, large centers providing advanced surgical care are inevitably confronted with chyle leaks as a complication of surgery, an extension of disease, or as a primary disorder. Regardless of the etiology, proper diagnosis and localization are paramount in the management of any chyle leak. MATERIALS AND METHODS: Here we present 16 patients with 17 chyle leaks (5 chyluria, 8 chylothorax, and 4 chylous ascites) who underwent bipedal lymphangiography (LAG) and postprocedure computed tomography (CT) imaging. RESULTS: In each case, the source of the chyle leak was identified and properly localized to guide further treatment. Of the 16 patients who underwent LAG and postprocedure CT imaging, the initial LAG alone provided the diagnosis and localized the chyle leak in 4 patients (25%); the postprocedure CT imaging provided the diagnosis and localized the chyle leak in 6 patients (37.5%); and the two modalities were equal in the diagnosing and localizing the chyle leak in the remaining 6 patients (37.5%) CONCLUSION: These cases highlight the unparalleled abilities of LAG and the added benefit of post-LAG CT imaging in the diagnosis and fine anatomic localization of chyle leaks. In addition, these cases demonstrate the retained utility of LAG in these investigations despite the development of alternative tests involving CT, magnetic resonance imaging, and nuclear medicine imaging.

Chyluria associated with nephrotic-range proteinuria: pathophysiology, clinical picture and therapeutic options.

Chyluria denotes the urinary excretion of chyle, which is a lymphatic fluid rich in chylomicrons. Chyle flows from the intestinal lacteals to the left subclavian vein through the thoracic duct. When an abnormal connection between these structures and the urinary tract develops, chyluria appears. The syndrome is often associated with a nephrotic-range proteinuria, and this could be a wrong indication to perform renal biopsy. Chyluria is classified as parasitic or nonparasitic, the former being induced by lymphatic filariasis, whereas the latter is caused by medical, traumatic or inherited diseases. The patient usually reports excretion of milky urines, monolateral flank pain, malnutrition, weight loss and weakness. Urinalysis demonstrates lymphocyturia associated with chylomicrons and triglycerides in the supernatant. The diagnostic approach is aimed to define the site of lymphourinary fistula. A selective ureteral catheterization allows to collect urine samples from each kidney, demonstrating a monolateral source of proteins and lipids and making renal biopsy superfluous. Other diagnostic tools include nuclear magnetic resonance urography and lymphoangiography. Many therapeutic options have been proposed. Sclerosing solution instillation into the renal pelvis and laparoscopic renal pedicle disconnection are the invasive procedures most commonly employed. Among the medical alternatives, a low-fat diet supplemented with medium-chain triglycerides is often followed by complete clinical and biochemical remission.

Looking at appearance of urine before performing a renal biopsy in nephrotic syndrome.

Chyluria results from an abnormal connection between lymphatic bed and urinary tract, causing lymph leakage into the urine. The clinical picture often begins with the appearance of cloudy, milky urines accompanied by monolateral flank pain, malnutrition, weight loss and weakness. We report a case of chyluria that occurred in a young woman who was referred to our unit for nephrotic-range proteinuria. Before performing a renal biopsy, we found that urine analysis demonstrated a massive lipiduria. Therefore, we collected urine samples from each kidney with a selective ureteral catheterization, demonstrating a monolateral source of lipids and proteins. We suspended the renal biopsy and performed a lymphography that showed an inherited lymphangioma on the left lumbar lymphatic bed. Sclerosing solution instillation, renal pedicle lymphatic disconnection or laser therapy are invasive therapeutical options that may cause severe adverse effects. Instead of these procedures, a conservative therapy based on a low-fat diet supplemented with medium-chain triglycerides was chosen. This dietetic schedule was followed by complete resolution of proteinuria and lipiduria. The patient progressively gained body weight and improved quality of life. No relapses were observed after 3 years of follow-up. This case emphasizes the possible role of a noninvasive therapeutical option for patients with chyluria.

Fístula linfática cervical: manejo conservador / Neck chylous fistula: conservative treatment

La fístula quilosa, por daño del conducto torácico, aparece en el 1-2,5% de los pacientes sometidos a disección cervical. Las complicaciones asociadas incluyen desnutrición, compromiso inmune, formación de fístulas y necrosis cutánea con exposición carotídea, e incluso ruptura. De la literatura actual no se puede deducir ningún algoritmo de tratamiento definitivo, pero parece existir un consenso en realizar un manejo conservador de la mayoría de los casos. El manejo médico se basa en la teoría de que si se disminuye el flujo de quilo se permitirá el cierre espontáneo de la fístula, e incluye: drenaje conectado a vacío, reposo, vendaje compresivo (en discusión),intervención nutricional y uso de análogos de somatostatina. El manejo nutricional implica el uso de dietas bajas en grasa suplementadas con Triglicéridos de Cadena Media (MCT), Nutrición Enteral con TCM o Nutrición Parenteral total (NPT) (AU)

Injury to the thoracic duct, leading to chyle leak, occursin 1-2,5% of patients who undergo neck dissection. Associated complications include malnutrition, immunecompromise, fistula formation and carotid blowout. No definitive treatment algorithm can be deduced from the current literature, but on last reviews, there is an agreementon the conservative management. Medical managementis based on that decreasing chyle flow will allow for spontaneous closure of the chyle leak. Conservative treatment includes: closed vacuum drainage, bed-rest,nutrition modification and synthetic somatostatin analog. Nutrition modification involves a low-fat diet supplemented with medium-chain triglycerides (MCT),enteral nutrition with high percentage of MCT or parenteral nutrition (AU)

[Neck chylous fistula: conservative treatment]. / Fístula linfática cervical: manejo conservador.

Injury to the thoracic duct, leading to chyle leak, occurs in 1-2,5% of patients who undergo neck dissection. Associated complications include malnutrition, immune compromise, fistula formation and carotid blowout. No definitive treatment algorithm can be deduced from the current literature, but on last reviews, there is an agreement on the conservative management. Medical management is based on that decreasing chyle flow will allow for spontaneous closure of the chyle leak. Conservative treatment includes: closed vacuum drainage, bed-rest, nutrition modification and synthetic somatostatin analog. Nutrition modification involves a low-fat diet supplemented with medium-chain triglycerides (MCT), enteral nutrition with high percentage of MCT or parenteral nutrition.

Octreotide in the treatment of thoracic duct injuries.

Anecdotal reports support the use of octreotide in the treatment of traumatic thoracic duct injuries and chylothorax, but no prospective studies have proved its efficacy. We evaluated the effects of octreotide in treating thoracic duct transection in a canine model. Eight mongrel dogs (27.8+/-5.1 kg) were fed one pint of 10.5 per cent milkfat 2 hours before operation. Through a left supraclavicular neck incision, the thoracic duct was identified and transected, producing free flow of chyle. A quarter-inch drain was tunneled subcutaneously from the wound and attached to closed suction. After wound closure dogs were randomized to a control group (n = 4) receiving sham injections of saline subcutaneously three times per day, or a treatment group (n = 4) given 3 microg/kg octreotide three times per day. Postoperatively all dogs were fed a standard low-fat (5-7%) crude fat diet. Drain output was measured each day, and on odd-numbered postoperative days the drainage was analyzed for cholesterol, triglycerides, albumin, and total protein. Fistula closure was defined as drainage <10 ml/24-hour period. Treated dogs achieved fistula closure significantly faster than controls: 3.5+/-1.3 days versus 7.8+/-1.0 days (P = 0.0037). Whereas equivalent amounts of drainage occurred on the day of surgery and on postoperative day one in both groups, by postoperative day 2 the treatment group had significantly less drainage over 24 hours: 63+/-69 ml versus 195+/-79 ml (P = 0.046); this significant difference persisted through postoperative day 5 when drainage began to decrease in the control group. No significant differences between groups were seen in levels of cholesterol, triglycerides, albumin, or protein in the drainage at any time point. We conclude that octreotide is effective in treating thoracic duct injury, leading to an early decrease in drainage and early fistula closure. The mechanism for this effect remains to be clarified.

[Immunological observation on chyluric patients with heat-clearing and hemostatic drugs].

In order to investigate the pathogenesis and therapeutic mechanism of chyluria, an experiment with a basic Heat-clearing and hemostatic prescription was conducted in treating 30 patients of chyluria. The result, 26 cases were cured completely. The cell-mediated and humoral immunity observation showed that OKT3 and OKT4 levels were commonly low in chyluric cases, and OKT8 as higher than normal value, the OKT4/OKT8 ratio was inverted before treatment. While OKT3 and OKT4 increased commonly, and OKT8 decreased with the OKT4/OKT8 ratio adjusted after treatment. Meanwhile, humoral immunity level was also commonly low before treatment, it increased after treatment.

[Electron microscopy observation on pre- and post-treatment skin of dorsum pedis of chyluric patient treated with heat-clearing and hemostatic drugs--the lymphangial wall damage being the pathogenesis of chyluria].

Heat-clearing and hemostatic drugs were effective in treating chyluria. In order to explore the curative mechanism, a group of 10 in-patients with chyluria were chosen and treated with heat-clearing and hemostatic drugs for 45 days. Some pre- and post-treatment skin from dorsum pedis of the patients were taken off and observed under electron microscope. It showed that the walls of lymphatic capillaries were seriously damaged before treatment and rehabilitated wall after treatment. The pinocytotic vesicles were packed tightly in the endothelial cells of capillaries before treatment and their number was decreased remarkably after treatment; the mast cells were rare in number and abnormal as spindle shape before treatment and they increased in number and became round or oval in shape, the nucleus became large, the granules they contained became more in number and larger in size after treatment, lymph edema was noticed before treatment and it disappeared after treatment. The result showed that the extensive lesion of lymphangial walls is the pathogenesis of chyluria, while the rehabilitation of them is the curative mechanism. It seems that in the process of rehabilitation the mast cells act took the role of "soldier-engineer".

[The diagnosis of lymphatic vascular dysplasia and lymphatic fistula formation]. / Zur Diagnostik von Lymphgefässdysplasie und Lymphfistelbildung.

The importance of lymphangiography for diagnosing the formation of lymphatic fistulas and vascular dysplasias of the lymphatic vessels is on the increase within the scope of therapeutic invasiveness. Its importance and ranking in diagnosing lymphatic circular disorders is demonstrated and exemplified by case reports on three patients suffering from chylothorax of various origin, on one patient with chyluria, and one patient with lymphoedema in the soft parts of the neck. In two chylothorax patients, lymphangiography resulted in successful surgery. It is the decisive method in diagnosis and therapy planning in fistulas and dysplasias of the lymphatic vessel system.

[Preliminary study of therapeutical mechanism on chyluria by heat-cleaning and hemostasis drugs].

Ten cases of filarial chyluria were treated with Chinese medical herbs. The results showed that the average concentration of serum histamine of 142 ng/ml before treatment dropped to 93 ng/ml after treatment. The average lymph-vessel pressure of 9.93 cmH2O in dorsalis pedis dropped to 4.74 cmH2O after cure. The prevalent microcirculation obstacle of medium to serious grade also was improved or recovered. The average serum viscosity of 1,884 before treatment dropped to 1,725. These results indicated that the chronic lymphatic inflammation occurring in pelvis or behind peritoneum due to perennial stimulation of adult filariae, caused the high concentration of inflammatory medium tissues, the expansion of micro-vessels and the increase of lymph-vessel pressure. Therefore, the microcirculation obstacle was the principle pathogeny of filarial chyluria.

Dietary modification of chyle composition in chylothorax.

Nutrition support has played a major role in the treatment of chylothorax, both to prevent malnutrition and to minimize chyle production and flow. This report evaluates chyle composition in a patient with chylothorax who was placed on a low-fat diet, medium-chain triglyceride diet, and total parenteral nutrition in sequence. Both triglyceride content and volume of chyle declined, but drainage persisted, ultimately requiring thoracic duct ligation. The chyle triglyceride while on total parenteral nutrition, which presumably originates from both the intestine and plasma, contained more long-chain unsaturated fatty acids than the circulating serum triglyceride. Of particular interest was the detection of an appreciable amount of medium-chain fatty acids in the chyle triglyceride, constituting 20% of the triglyceride fatty acids when an enteral formulation with medium-chain triglyceride as a sole fat source was administered. The finding of almost threefold more decanoic acid (C10:0) than octanoic acid (C8:0), despite the presence of considerably more octanoic acid in the original diet, suggests that trioctanoin may be a preferable medium-chain triglyceride substrate for the nonsurgical treatment of chylothorax.