Indocyanine Green Fluorescent Lymphography During Open Inguinal Hernia Repair: Relationship Between Lymphatic Vessel Injury and Postoperative Hydrocele.

BACKGROUND: This prospective case series analyzed patients who underwent indocyanine green (ICG) fluorescent lymphography during open inguinal hernia repair. The aim of this study was to investigate the association between ICG leakage and postoperative hydroceles in patients who underwent inguinal hernia repair. MATERIALS AND METHODS: Data were analyzed from 40 patients who underwent primary open hernia repair between October 2020 and June 2021 (44 cases in total). Hydroceles were categorized into two types: symptomatic and "ultrasonic" (detected only by ultrasound imaging). RESULTS: In the univariate analysis, hernia type (p = 0.044) and ICG leakage (p = 0.007) were independent risk factors for postoperative ultrasonic hydroceles. Additionally, mesh type (p = 0.043) and ICG leakage (p = 0.025) were independent risk factors for postoperative symptomatic hydroceles. In the multivariate analysis, ICG leakage (p = 0.034) was an independent risk factor for postoperative ultrasonic hydroceles. CONCLUSIONS: ICG leakage after inguinal hernia repair was independently associated with postoperative ultrasonic and symptomatic hydroceles. These findings suggest a relationship between lymphatic vessel injury and the incidence of postoperative hydroceles.

Chylothorax associated with primary membranous nephropathy: a case report.

BACKGROUND: Chylothorax is a state in which pleurisy is induced by chylomicron leakage due to lymphatic injury. Membranous nephropathy (MN) is one of the relatively common glomerular diseases that cause nephrotic syndrome in adults. Chylothorax at the onset of nephrotic syndrome is very rare in adult patients. CASE DESCRIPTION: We report a case of chylothorax associated with primary MN. A 64-year-old man visited the hospital complaining of lower extremity edema and dyspnea for 4 weeks. Laboratory findings showed no azotemia but hypercholesterolemia, hypoalbuminemia, nephrotic-range proteinuria, and microscopic hematuria. Chest and abdominal computed tomography (CT) revealed no ascites, venous thrombosis, or malignancy with the presence of right-side pleurisy. Biochemical analysis of the pleural fluid was consistent with chylothorax. The patient was confirmed to have MN by percutaneous kidney biopsy. An angiotensin receptor blocker, diuretics, and a hypolipidemic agent were prescribed; non-per os, total parenteral nutrition (TPN), and subcutaneous injection of octreotide were added for management of chylothorax. As serum anti-phospholipase receptor 2 antibody (Ab) concentration increased again, immunosuppressive therapy (IST) consisting of alternating monthly cycles of glucocorticoids and oral cyclophosphamide was instituted. With no improvement in chylothorax and deteriorating nutritional status despite 3 weeks of medical therapy, lymphangiography was performed, followed by thoracic duct embolization (TDE). The patient was discharged from the hospital on day 53 with clinical improvement. At 9 months after discharge, clinical remission of primary MN was achieved without recurrence of chylothorax. CONCLUSIONS: Patients with nephrotic syndrome may rarely exhibit refractory chylothorax without chylous ascites, increasing the risk of serious metabolic complications such as severe malnutrition. Therefore, upon confirming chylothorax associated with primary nephrotic syndrome, prompt radiologic intervention for lymphatic leakage must be considered in addition to specific IST.

Therapeutic effect of lymphography in the postoperative chylothorax - case reports and literature review.

Postoperative chylothorax is a well-known rare complication of thoracic surgery. It is a serious complication that is fatal in cases of inadequate treatment. The authors present 2 cases of postoperative chylothorax that were successfully treated by performing pedal and/or intranodal lymphography. In one case, the patient underwent lymphography after previous unsuccessful surgical ligation of the thoracic duct. The presented case reports describe therapeutic importance of conventional lymphography as a minimally invasive treatment of the postoperative chylothorax.

Therapeutic lymphangiography with ethiodized oil for the management of lymphoceles and chylous ascites.

PURPOSE: The purpose of this study was to analyze the safety, technical success and clinical outcome of percutaneous intranodal ethiodized oil (Lipiodol®) based lymphangiography (L-LAG) for the management of refractory pelvic lymphoceles or chylous ascites using high doses of ethiodized oil. MATERIALS AND METHODS: Thirty-four patients presenting with symptomatic, refractory postoperative pelvic lymphocele or chylous ascites referred for theranostic, inguinal, intranodal L-LAG treatment between May 2018 and November 2021 were retrospectively included. There were 21 men and 13 women, with a mean age of 62.7 ± 16.2 (standard deviation) years (age range: 9-86 years), who underwent a total of 49 L-LAG for the management of lymphoceles (n = 14), chylous ascites (n = 18) or a combination of lymphocele and chylous ascites (n = 2). Clinical and radiological pre-interventional, procedural and follow-up data up to January 2022 were collected from patients' electronic medical records and imaging files. RESULTS: Technical success was obtained in 48 out of 49 L-LAG (98%). No complications related to L-LAG were noted. After one or more L-LAG, clinical success was obtained in 30 patients (88%) with a mean of 1.4 interventions per patient and mean intranodal injected volume of 29 mL of ethiodized oil per session. The remaining four patients (12%), with one or more failed L-LAG, underwent additional surgical intervention to definitively treat the postoperative lymphatic leakage. CONCLUSION: L-LAG using high doses of ethiodized oil is a minimally invasive, safe and effective treatment of postoperative pelvic lymphocele or chylous ascites. Multiple sessions may be needed to obtain a meaningful clinical result.

Thoracic duct leakage in a patient with Type B-Non-Hodgkin lymphoma treated with transvenous retrograde access embolization: a case report.

Thoracic duct (TD) is the largest lymphatic vessel in the body and drains the lymph at the junction between the left subclavian and jugular veins. Chylothorax (CTX) represents an accumulation of lymphatic fluid in the pleural space. We present a case of a 65 years-old man with an histologically diagnosed mediastinal type B non-Hodgkin Lymphoma, treated with chemo-immunotherapy. CT scan during follow up showed significant left side pleural effusion, amounting to 2.8 litres after drainage. Conservative treatment with low fat parenteral nutrition was started without reduction of drainage output, then lymphangiography (LP) with Lipiodol was performed demonstrating a leak in the distal TD. CTX increased in the following days, and a further LP was performed. Using transvenous retrograde access we catheterized TD at the left subclavian jugular veins using a microcatheter. The leak was treated with multiple conventional and controlled delivery microcroils and cyanoacrylate, obtaining complete embolization without residual leak.

Popliteal Vascular Lymph Node Resection in the Rabbit Hindlimb for Secondary Lymphedema Induction.

Lymphedema is a common condition often associated with cancer and its treatment, which leads to damage to the lymphatic system, and current treatments are mostly palliative rather than curative. Its high incidence among oncologic patients indicates the need to study both normal lymphatic function and pathologic dysfunction. To reproduce chronic lymphedema, it is necessary to choose a suitable experimental animal. Attempts to establish animal models are limited by the regenerative capacity of the lymphatic system. Among the potential candidates, the rabbit hindlimb is easy to handle and extrapolate to the human clinical scenario, making it advantageous. In addition, the size of this species allows for better selection of lymphatic vessels for vascularized lymph node resection. In this study, we present a procedure of vascular lymph node resection in the rabbit hindlimb for inducing secondary lymphedema. Anesthetized animals were subjected to circumferential measurement, patent blue V infiltration, and indocyanine green lymphography (ICG-L) using real-time near-infrared fluorescence, a technique that allows the identification of single popliteal nodes and lymphatic channels. Access to the identified structures is achieved by excising the popliteal node and ligating the medial and lateral afferent lymphatics. Special care must be taken to ensure that any lymphatic vessel that joins the femoral lymphatic system within the thigh without entering the popliteal node can be identified and ligated. Postoperative evaluation was performed at 3, 6, and 12 months after induction using circumferential measurements of the hindlimb and ICG-L. As demonstrated during follow-up, the animals developed dermal backflow that was maintained until the 12th month, making this experimental animal useful for novel long-term evaluations in the management of lymphedema. In conclusion, the approach described here is feasible and reproducible. Additionally, during the time window presented, it can be representative of human lymphedema, thus providing a useful research tool.

[Plastic bronchitis].

Plastic bronchitis (PB) is a rare disease caused by abnormal lymphatic vessels in the thorax. These vessels drain into the bronchi creating solid casts and potential life-threatening airway obstruction. This is a case report of a 30-year-old man diagnosed with PB after several years of extensive examinations due to symptoms misconceived as non-allergic asthma. We describe the first interventional treatment in Denmark using special T2 weighed MR imaging and dynamic contrast MR lymphangiography with subsequent embolisation of abnormal lymphatic vessels in the thorax.

Real-time image-sharing to educate a patient with lymphoedema on self-care: a case study.

INTRODUCTION: Compression therapy is important in oedema control in lymphoedema. However, some patients have difficulties starting compression therapy because standard self-care education does not enable them to fully understand lymphoedema and recognise it as their problem. To overcome this, real-time image-sharing education, using a combination of indocyanine green lymphography (ICG) and ultrasonography, may be used to educate patients. In this case study, real-time image-sharing education promoted decision-making and behaviour change in a patient with lower extremity lymphoedema so she would wear elastic stockings. CASE: A 51-year-old woman with a BMI of 31.7 kg/m2 and secondary lower extremity lymphoedema following cervical cancer surgery did not adhere to self-care instructions regarding wearing elastic stockings for 5 years. The oedema worsened, her limb circumference increased and she had two episodes of cellulitis within a year. Because the patient had a negative attitude towards elastic stockings, real-time image-sharing education was used to promote recognition of illness using ICG and an understanding of the condition of lymphoedema using ultrasonography. After the images were shared and explained, the patient discussed her recognition and understanding of lymphoedema, then decided to use compression stockings. She continued to wear them for 4 months, and her limb circumference decreased. CONCLUSION: Real-time image-sharing education using ICG and ultrasonography as self-care support for a lymphoedema patient who would not start compression therapy could result in behavioural changes and the patient starting and continuing to wear elastic stockings.

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.

Neonatal lymphatic flow disorders: central lymphatic flow disorder and isolated chylothorax, diagnosis and treatment using novel lymphatic imaging and interventions technique.

PURPOSE OF REVIEW: Neonatal lymphatic disorders (NLDs) are conditions that are relatively rare and difficult to treat. The recent development of lymphatic imaging, such as Dynamic Contrast-Enhanced MR Lymphangiography and Intranodal Lymphangiography has led to a new, better understanding of the anatomical substrate and pathophysiological mechanisms of the diseases. Consequently, this has allowed the development of new targeted therapeutic interventions as well as prognostication for this population with lymphatic flow disorders. RECENT FINDINGS: The underlying causes of all NLD is an obstruction or altered flow of the central lymphatic flow. Two types of NLD have been described: isolated neonatal chylothorax and central lymphatic flow disorder (CLFD). Isolated neonatal chylothorax can be treated successfully with oil-based contrast (lipiodol) embolization. CLFD secondary to obstruction of the thoraco-venous junction can be successfully treated with surgical thoracic duct-venous anastomosis. CLFD caused by elevated central pressure and/or thoracic duct dysplasia can be treated medically, including with new systemic therapies such as mammalian target of rapamycin inhibitors. SUMMARY: New diagnostic and interventional tools have recently allowed for classification, prognostication, and targeted interventions for neonatal patients with lymphatic flow disorders. Further research will build on these discoveries.

Contemporary Management of Chylous Ascites after Retroperitoneal Surgery: Development of an Evidence-Based Treatment Algorithm.

PURPOSE: Chylous ascites (CA) is an uncommon complication that occurs from traumatic disruption of lymphatic channels after retroperitoneal surgery. The purpose of this study was to generate an evidence-based management strategy for CA by reviewing the current literature and available treatment modalities. MATERIALS AND METHODS: A MEDLINE® literature review was performed for "chylous ascites." Individual patient data were extracted from case series and reports to create an efficacy analysis. Treatment modality, drain output, time to escalation of care and time to resolution were recorded. The efficacy analysis was utilized to generate a data-driven treatment algorithm. RESULTS: The literature review yielded 1,953 articles, from which 146 studies contributed data for 523 patients. The efficacy analysis included 245 patients, 168 (69%) of whom were managed successfully with conservative management (CM), at a median time to resolution of 11 days. Forty-eight patients underwent lymphangiography±embolization after CM, with a success rate of 85%. Thirty-one (12%) patients underwent surgical exploration. When treating CA, the patients who underwent stepwise management with CM followed by lymphangiography if CM failed experienced a resolution rate of 96.7%. An evidence-based treatment algorithm was created to guide treatment selection and duration of therapy before escalating to additional forms of therapy. CONCLUSIONS: In this report, we describe the largest conglomeration of iatrogenic CA cases from a literature review (523 cases) and efficacy analysis (245 cases), and created the first evidence-based treatment algorithm for this condition. Treatment success is substantial when using a stepwise combination of CM followed by lymphangiography±embolization.

PIEZO1-gene gain-of-function mutations with lower limb lymphedema onset in an adult: Clinical, scintigraphic, and noncontrast magnetic resonance lymphography findings.

Primary lymphedema, a rare disease, has a genetic cause in ~40% of patients. Recently, loss-of-function mutations in PIEZO1, which encodes the mechanotransducer protein PIEZO1, were described as causing primary lymphedema, when gain-of-function PIEZO1 mutations were attributed to dehydrated hereditary stomatocytosis type-1 (DHS), a dominant red cell hemolytic disorder, with ~20% of patients having perinatal edema. Lymphedema was diagnosed in a 36-year-old man from a three-generation DHS family, with a PIEZO1-allele harboring 3 missense mutations in cis. Four affected family members had severe fetal and neonatal edema, most severe in the proband, whose generalized edema with prevailing ascites resolved after 8 months. Our patient's intermittent lower limb-lymphedema episodes during hot periods appeared at puberty; they became persistent and bilateral at age 32. Clinical Stemmer's sign confirmed lymphedema. Lower leg lymphoscintigraphy showed substantial dermal backflow in both calves, predominantly on the right. Noncontrast magnetic resonance lymphography showed bilateral lower limb lymphedema, dilated dysplastic lymphatic iliac, and inguinal trunks. Exome-sequencing analysis identified no additional pathogenic variation in primary lymphedema-associated genes. This is the first description of well-documented lymphedema in an adult with PIEZO1-DHS. The pathophysiology of PIEZO1-associated primary lymphedema is poorly understood. Whether it infers overlapping phenotypes or different mechanisms of gain- and loss-of-function PIEZO1 mutations deserves further investigation.

Retrograde transvenous selective lymphatic duct embolization in post donor nephrectomy chylous ascites.

Chylous ascites is a rare, potentially sinister complication in post donor nephrectomy patients which may cause significant morbidity in form of severe malnutrition and an immuno-compromised state. We present two patients with post donor nephrectomy-related chylous leaks who failed conservative treatment. In both cases, lymphangiography was done first to detect the chylous leak site in the left renal fossa, and thereafter transvenous retrograde approach via left subclavian vein with selective lymphatic duct embolization of chylous leak was done with coils and glue successfully. Chylous ascites resolved in both patients after the embolization. Hence retrograde transvenous embolization technique appears to be an effective management option for refractory chylous leaks.

Computed tomography-guided lymphangiography from the para-aortic lymph node: a useful approach for chylothorax after esophagectomy.

BACKGROUND: Chylothorax is a relatively rare complication of esophagectomy but can lead to fatal conditions. The aim of this report was to present a case of lymphangiography from the para-aortic lymph node (PALN) under computed tomography (CT) guidance, which could be an alternative modality to ultrasound sonography from the groin. CASE PRESENTATION: A 58-year-old man was diagnosed as having postoperative chylothorax after esophagectomy following neoadjuvant chemotherapy. As the pleural effusion did not improve, intranodal lipiodol lymphangiography from the inguinal lymph node was performed but was unsuccessful. Therefore, CT-guided lymphangiography from the left PALN was performed, and the pleural effusion drainage significantly disappeared. CONCLUSIONS: CT-guided lipiodol lymphangiography from the PALN for chylothorax after esophagectomy was safe and thus can be considered an alternative treatment option to ultrasonography-guided intranodal lymphangiography. The flexible use of CT and ultrasonography for lymphangiography may improve the treatment outcomes of chylothorax after esophagectomy.

Reverse Lymph Node Mapping Using Indocyanine Green Lymphography: A Step Forward in Minimizing Donor-Site Morbidity in Vascularized Lymph Node Transfer.

BACKGROUND: Reverse lymphatic mapping before harvesting a lymph node flap is crucial to avoid donor-site lymphedema; however, the technique is complex and unavailable in many centers. The authors introduce radioisotope-free reverse lymphatic mapping using indocyanine green and Patent Blue dye. METHODS: The authors conducted a prospective study in patients undergoing free vascularized groin lymph node transfer for postmastectomy upper extremity lymphedema. The day before surgery, 0.2 ml of technetium-99 was injected into the first and second web spaces of the ipsilateral foot. The following day, once the patient was anesthetized, indocyanine green was injected into the same web spaces of the same foot and Patent Blue dye was injected just proximal to the upper margin of the skin paddle of the lymph node flap. The main lymph nodes draining the limb were localized using indocyanine green lymphography and gamma probe. RESULTS: Thirty-nine patients underwent vascularized groin lymph node transfer with or without deep inferior epigastric artery perforator flap breast reconstruction. Navigation of the main lower extremity draining inguinal lymph nodes using the gamma probe and indocyanine green lymphography was identical in all patients. The blue-stained lymphatics in the skin paddle drained to the superficial proximal inguinal lymph node and were targeted for transfer. No donor-site lymphedema was reported, and lymphatic drainage of the lower extremity was preserved in all cases. CONCLUSIONS: Reverse lymphatic mapping using indocyanine green lymphography provides identical results to those using technetium-99 isotope scanning. However, indocyanine green is preferable in terms of safety and reproducibility and also avoids the complexity and hazards of radioisotope mapping. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, IV.

[Postoperative Chylothorax Likely to be Successfully Treated by Lymphangiography;Report of a Case].

A 74-year-old man underwent right upper lobectomy and systemic lymph node dissection with video-assisted thoracoscopic surgery. Chylothorax occurred on postoperative day (POD) 1st. Under fasting management, the patient underwent pleurodesis on POD 5th and 7th, subcutaneous octreotide acetate injection on POD 6th, and lymphangiography on POD 9th. The amount of drainage decreased on POD 10th, and the drainage tube was removed on POD 12nd. Lymphangiography has been reported as one of effective procedures to treat postoperative chylothorax. The present case is also considered to be successfully treated by lymphangiography.

Devastating cerebral Lipiodol® embolization related to therapeutic lymphangiography for refractory chylothorax in a patient with Behçet's disease.

Onset of neurological symptoms early after intranodal lymphangiography can occur due to Lipiodol droplet migration through intrapulmonary lymphovenous communication. Patients with Behçet's disease may be at higher risk of developing this devastating complication.

A Novel Technique in the Treatment of Lymphoceles After Renal Transplantation: C-Arm Cone Beam CT-Guided Percutaneous Embolization of Lymphatic Leakage After Lymphangiography.

BACKGROUND: We aimed to evaluate the efficacy of percutaneous embolization after lymphangiography using C-arm cone-beam computed tomography (CBCT) performed at the site of lymphatic leakage in patients with postrenal transplant lymphocele. METHODS: Between July 2014 and August 2017, 13 patients not responding to percutaneous ethanol sclerotherapy and conservative treatment for recurrent lymphocele after renal transplant were included. The mean age of the patients was 56.38 ± 9.91 (range, 36-70) years, and it comprised 9 men and 4 women. All patients underwent intranodal lymphangiography. C-arm CBCT-guided percutaneous embolization was performed in patients with confirmed lymphatic leakage. Patients who had no lymphatic leakage underwent drainage with fibrin glue injection. RESULTS: Lymphatic leakage was observed in 9 patients after lymphangiography, and they underwent CBCT-guided percutaneous N-butyl-2-cyanoacrylate embolization. The volume of lymphatic drainage reduced to less than 10 mL in 8 patients. One patient who was not responding to embolization was treated surgically, after percutaneous drainage and fibrin glue injection. Lymphatic leakage was not observed in 4 patients after lymphangiography. Of these, 3 patients showed a reduction in the amount of lymphatic drainage after lymphangiography. All 4 patients underwent percutaneous drainage and fibrin glue injection. One patient did not respond to the treatment and was treated surgically. Prelymphangiography and postlymphangiography and embolization, the volume of lymphatic drainage was 113.07 ± 21.75 mL, and 53.84 ± 30.96 mL, respectively, and statistically significant decrease was detected (P < 0.005). CONCLUSIONS: Lymphangiography and CBCT-guided percutaneous embolization procedures might be an effective treatment method for patients with lymphocele refractory to treatment.

Feasibility of fluorescence lymph node imaging in colon cancer: FLICC.

BACKGROUND: In colon cancer, appropriate tumour excision and associated lymphadenectomy directly impact recurrence and survival outcomes. Currently, there is no standard for mesenteric lymphadenectomy, with a lymph node yield of 12 acting as a surrogate quality marker. Our goal was to determine the safety and feasibility of indocyanine green (ICG) fluorescence imaging to demonstrate lymphatic drainage in colon cancer in a dose-escalation study. METHODS: A prospective pilot study of colon cancer patients undergoing curative laparoscopic resection was performed. At surgery, peritumoural subserosal ICG injection was done to demonstrate lymphatic drainage of the tumour. A specialized fluorescence system excited the ICG and assessed lymphatics in real time. The primary outcome was the feasibility of ICG fluorescent lymphangiography for lymphatic drainage in colon cancer. Secondary outcomes were the optimal protocol for dose, injection site, and ICG lymphatic mapping timing. RESULTS: Ten consecutive patients were evaluated (six males, mean age 69.5 years). In all, lymphatic channels were seen around the tumour to a varying extent. Eight (80%) had drainage to the sentinel node. In all cases where the lymphatic map was seen, there was no further spread 10 min after injection. In 2 patients (20%), additional lymph nodes located outside of the proposed resection margins were demonstrated. In both cases the resection was extended to include the nodes and in both patients these nodes were positive on histopathology. Factors contributing to reduced lymphatic visualization were inadequate ICG concentrations, excess India ink blocking drainage, and inflammation from tattoo placement. CONCLUSIONS: ICG can be safely injected into the peritumoural subserosal and demonstrate lymphatic drainage in colon cancer. This proof of concept and proposed standards for the procedure can lead to future studies to optimize the application of image-guided precision surgery in colon cancer. Furthermore, this technique may be of value in indicating the need for more extended lymphadenectomy.