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Management of chyluria using percutaneous thoracic duct stenting

Management of chyluria using percutaneous thoracic duct stenting Background: Thoracic duct stenosis or obstruction is one of the causes of chyluria. Although the diagnosis of chy- luria is not difficult, treatment is still challenging. Although there have been no standard guidelines for the treatment of chyluria, interventional techniques now offer minimally invasive treatment options for chyluria such as interstitial lymphatic embolization, ductoplasty with balloon, or thoracic duct stenting. Case presentation : Here, we report a case of chyluria due to obstruction of the junction between the thoracic duct and subclavian vein in a 64 -year- old female patient. The patient was treated with balloon plasty for lymphovenous junction obstruction and interstitial lymphatic embolization for chyluria. However, chyluria was recurrent after 6 months so intranodal lymphangiography was performed. Anterograde thoracic duct was accessed through a transab- dominal to the cisterna chyli which showed that the thoracic venous junction was re-obstruction. The patient was successfully treated by placing a uncovered drug-eluting stent with the size of 2.5 mm x 15 mm in length for resolv- ing the thoracic occlusion. Conclusion: This report demonstrates the feasibility of using thoracic duct stenting in the treatment chyluria due to lymphovenous junction obstruction. Keywords: Chyluria, Lymphatic, Thoracic duct, Stenosis, Obstruction, Balloon, Stent Introduction diet is the first step. Sclerotherapy using a ureteroscope Chyluria is divided into parasitic and non-parasitic cat- has also been reported, but it is not a well-established egories (Stainer et  al. 2020). Non-parasitic chyluria is a treatment and its effectiveness is limited. As a result, not rare condition and caused by: trauma, surgery, infections, all patients respond well to these therapies (Lovrec Krstić malignancy, lymphatic malformation, radiation, urinary et al. 2021). In the literature, some new alternative treat- retention, congenital fistula between lymphatics and the ments were applied for the management of chyluria such urinary tract, pregnancy and stenosis of the thoracic duct as interstitial lymphatic embolization through percuta- (TD) (Stainer et al. 2020; Shah et al. 2020). There are no neous or retrograde thoracic duct access, and interstitial guidelines for the management of chyluria. The treatment lymphatic embolization with balloon plasty (Gurevich approach depends on the etiology and the site of the lym- et  al. 2018; Hur et  al. 2021; Nguyen et  al. 2020). In this phatic system damage; and mostly should be tailored on a article, we would like to present a new interventional case-by-case basis. Conservative treatment with a low-fat technique for a patient with chyluria associated with the lymphovenous junction (LVJ) obstruction. Case report *Correspondence: cuongcdha@gmail.com A 64 -year- old female patient has suffered from chylu - Diagnostic Imaging and Interventional Center, Hanoi Medical University ria for 6 months. She has no history of parasite infection, Hospital, No1, Tonthattung, Dongda, Hanoi, Vietnam Full list of author information is available at the end of the article no abdominal trauma, and no history of renal operation. © The Author(s) 2022. Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http:// creat iveco mmons. org/ licen ses/ by/4. 0/. Cuong et al. CVIR Endovascular (2022) 5:54 Page 2 of 5 Cystoscopy showed the milky chyle efflux from the right was advanced into the superior vena cava. From the right ureteric orifice suggesting that the origin of chyluria was femoral vein, we advanced a snare into the superior vena from the right kidney. The patient underwent dynamic cave. The snare caught the guide wire and then pulled the contrast-enhanced magnetic resonance lymphangiog- guide wire out of the sheath at the right femoral vein. TD raphy (DCMRL). DCMRL visualized the thoracic duct venous junction was dilated at a pressure of 8 atm using and showed the presence of dilated lymphatic vessels in a balloon 2.5  mm x 20  mm (Pantera LEO, Biotronik, the right renal pelvis confirming the chylo calyceal fistula Bulach, Switzerland) (Fig. 2 C). The interstitial lymphatic (Fig. 1). embolization was then performed with total of 2 ml mix- Bilateral inguinal intranodal lymphangiography was ture of N-butyl cyanoacrylate (NBCA) and lipiodol with then performed. There was a stagnation of Lipiodol the ratio of 1:5 (Fig. 2D) as describe in the literature dur- (Guerbet, France) in the lymphatic system and the cis- ing the inflation of the balloon at the junction TD vein terna chyli appeared after 50 minutes since the injection (Gurevich et al. 2018). of contrast started. In addition, reflux of oil contrast into The patient recovered well with no longer chyluria; the right renal pelvis was also observed. On lymphogra- no complications were noticed. But 6 months after, the phy, the thoracic duct was also dilated and the contrast patient come back to our institution because of recur- was ended at the TD venous junction without draining rent symptoms of chyluria. Cystoscopy showed the efflux in the subclavian vein. Neck ultrasound and echocar- of chyle on the right side, and DCMRL found again the diography revealed there was no neck tumor or central afferent lymphatic vessels in the right kidney. We per - vein thrombosis. There were no anatomical abnormali - formed intranodal lymphangiography and found that ties of the lymphatic system, LVJ as well as the left sub- there was re-obstruction of the TD venous junction. clavian vein. The laboratory tests gave negative results Dilated and tortuous right retroperitoneal lymphatics, with Wuchereria bancrofti, Toxocariasis, Taenia echi- right kidney lymphatics, and filling of kidney calyces nococcus. The diagnosis was made as chyluria from the like the previous lymphangiography was also revealed. right kidney due to an obstruction at the junction TD- Because re-obstruction occurred after balloon plasty subclavian vein. Because the guide wire (0.018” Terumo) 6 months, we planned to re-delate the junction by bal- could pass through the occlusion, the LVJ was dilated by loon and place a metallic stent. The stent we used was balloon and then interstitial lymphatic embolization was a coronary stent with the size of 2.5  mm x 15  mm in done to occlude the lymphatic communication between length (drug-eluting stent) because of the compatible size retroperitoneal lymphatic vessels and the right renal pel- (Fig. 3). After the stent deployment, direct lymphangiog- vis as described in the literature (Gurevich et  al. 2018; raphy demonstrated contrast medium from the thoracic Kariya et al. 2019). We punctured the cisterna chyli by a duct to the left subclavian vein without disruption or 21-gauge-needle (chiba, Cook, USA) and then inserted leakage. Venography of the left subclavian vein showed a 0.018” guide wire into the thoracic duct. A 2.7-french- no reflux flow from the vein into the thoracic duct. Then, microcatheter (progreat, terumo, Japan) was advanced we intended to puncture the interstitial lymphatic ves- into the thoracic duct over the gude wire. By injecting sels at the lumbar region, but all attempts failed. There - the contrast into microcatheter in order to opacify the fore, no embolization of interstitial lymphatic vessels was thoracic duct, we found that there was an occlusion at performed. After procedure, the patient had transient the TD venous junction (Fig.  2 A and B). The long guide hematuria but no chyluria. The patient had remained wire (0.014”, 300 cm, transend soft tip, Boston Scientific, asymptomatic for 1 year later and still under follow- USA) was easily passed through the occlusion and then ing up. The computed tomography (CT) scan at the 6th Fig. 1 DCMRL showed dilated lymphatic vessels in the right renal pelvis (A) and the chylo calyceal fistula (B) C uong et al. CVIR Endovascular (2022) 5:54 Page 3 of 5 Fig. 3 Second intervention. A TD lymphangiography showed re-occlusion of the junction TD vein and collateral circulation of lymphatic vessels in the left neck (arrows). B The stent was inflated in the LVJ by the balloon (arrow). C Injection of contrast after deploying the stent showed the flow into subclavian vein. D superior venogram showed that the contrast material in the left subclavian vein did not reflux to the TD Fig. 2 First intervention. A Lymphangiography revealed dilated the upper part of TD and remained the contrast in the TD (arrow). In terms of management of the chyluria symptoms, con- B injection of contrast into TD showed occlusion of the TD and dilatation at the junction (arrow). C The 0.014”-guidewire passed servative treatment such as a high-protein and low-fat through the occlusion into superior vena cava then the femoral vein diet has a possibility to reduce chyluria. Other treatment by a snare. A 6 French-guiding catheter was placed at the junction options are sclerotherapy and embolization. Sclerosants TD subclavian vein (arrow). D Right retroperitoneal lymphatic system induce an inflammatory reaction in the lymphatic ves - communicates with renal collecting system and the contrast material sels and blockade the communicating channels by fibrosis presented in the kidney calyces (arrow), the interstitial lymphatic embolization was then performed (Lovrec Krstić et  al. 2021; Gurevich et  al. 2018) presented three cases of chyluria and two of them underwent inter- stitial lymphatic embolization by percutaneously accessing month showed the stent fully opened and was in the cor- the retroperitoneum lymphatic channels or retroperito- rect position (Fig.  4  A) and DCMRL lymphangiography neal lymph node under fluoroscopic guidance. These two at the 12th month showed the patency of TD (Fig. 4B). patients had chyluria due to increased thoracic duct pres- sure and they were completely resolved within 1 year and 7 months after the second embolization up to 1 year. Discussion One patient had improved symptoms with a low-fat diet. The TD can be injured via trauma, surgery, or congested Nguyen et al. (2020) also used interstitial lymphatic embo- by central venous occlusion (An et  al. 2021; Chick et  al. lization and accessed the lymphatic channel by puncturing 2018). It may be caused by compression from outside. the retroperitoneal lymph node through the abdominal wall Other causes include lymphatic malformation, radiation, and they put a balloon catheter in the TD in order to pre- congenital abnormalities, and stenosis of the TD (Lovrec vent reflux of embolic agent into the TD. A recent article Krstić et  al. 2021). Valvular insufficiency in the thoracic by Hur et al. (2021) presented another approach into TD by duct results in elevated pressure in the lymphatic circu- retrograde through the left brachial vein or directly punc- lation and backflow of lymphatic fluid into other major turing the TD followed by lymphatic embolization through lymphatic branches. When the thoracic duct pressure is a micro catheter. These treatments method for chyluria increased, it may lead to rupture or reflux of lymphatic were based on disruption of the connection between renal channels and may cause chylous ascites or chylothorax calyces and lumbar interstitial lymphatic vessels. (Kariya et al. 2019). Cuong et al. CVIR Endovascular (2022) 5:54 Page 4 of 5 Fig. 4 Follow up images. A CT scans 6 months post-stenting revealed the stent was in the right position. B DCMRL after one year showed the patency of thoracic duct Regarding to management of the causes, especially TD ducts. There is no blood reflux into the TD, possibly obstruction, there are only a few articles about the treat- because the pressure in the subclavian vein is low and the ment methods. Thoracic ductoplasty with a balloon is same flow direction of the stent and the subclavian vein reported to reduce lymphatic circulation pressure and blood. Moreover, because we used drug-eluting stents, restore the patency of the TD to treat chylothorax and and there was no coagulation factor in the TD so antico- chylous ascites (Kariya et  al. 2019). The results showed agulation was not used in this patient. that after 6 months, the symptoms did not recurrent. The Thoracic duct stenting seems a feasible and practical use of stent in the management of TD obstruction has approach in the treatment of LVJ obstruction. Follow-up not been reported. is necessary to access the patency, location of the stent. Therefore, stenting in management LVJ obstruction of Further studies are needed to confirm these results. the thoracic duct has not been reported and this article may be the first case reported in the literature regard - Conclusion ing to treat TD occlusion. Until now, there has been one Thoracic duct stent is a new procedure with success - article describing TD stenting treatment for TD hyper- ful approach in chyluria with thoracic duct stenosis or tension by Ghelfi et al. (2022). In this article, Ghelfi et al. obstruction. reported two cases with cirrhosis and refractory chylous ascites for which transjugular intrahepatic portosystemic shunt. In these cases, portal hypertension increases lym- Abbreviations TD: Thoracic duct; LVJ: Lymphovenous junction; DCMRL: Dynamic con- phatic flow and may cause the pressure gradient without trast-enhanced magnetic resonancelymphangiography; CT: Computed TD obstruction or stenosis. TD stenting may resolve the tomography. lymph-venous pressure gradient and chylous ascites. The Acknowledgements stent used in our patient was a drug-eluting stent. A part None. of the stent was located in the subclavian vein aimed to keep the stent in the right location. The drug-eluting may Informed consent Informed consent was obtained from patient in the study. prevent thrombosis. Our patient had short-term success with balloon dila- Authors’ contributions tation; however, re-obstruction appeared later. The cause NNC actually engaged in treatment, created the initial draft and contributed to editing and submitting the revised manuscript. LTL performed the proce- of this condition may be fibrosis, intimal hyperplasia, or dure and edited the revised manuscript. TTTM, TQH obtained the images and inflammation. In this case, the internal pressure of the edited the revised manuscript; contributed to the content of the discussion. thoracic duct was not measured before and after the pro- HL, LH and MI provided edits and improvements to the revised manuscript. All authors read and approved the final manuscript. cedure because of a lack of measuring tools. The cause of chyluria in this patient may be increased intratho- Funding racic pressure leading to rupture or reflux of lymphatic The authors have no relevant financial or non-financial interests to disclose. This study was not supported by any funding. channels into the urinary tract. The objective treatment in the present case was to release a blockage in the tho- Availability of data and materials racic duct to reduce pressure in the peripheral lymphatic All data are referenced from the medical records and stored in the hospital. C uong et al. CVIR Endovascular (2022) 5:54 Page 5 of 5 Declarations Ethics approval and consent to participate All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional ethics committee and with the 1964 Helsinki declaration and its later amendments or compara- ble ethical standards. Consent for publication Written informed consent was obtained from the patient for publication of this case report and any accompanying images. Competing interests All authors declare that they have no conflict of interest. Author details Diagnostic Imaging and Interventional Center, Hanoi Medical University Hospital, No1, Tonthattung, Dongda, Hanoi, Vietnam. Surgery of Urology Department, Hanoi Medical University Hospital, Hanoi Medical University, Hanoi, Vietnam. Respiratory Department, Hanoi Medical University Hospital, Hanoi, Vietnam. Keio University, Tokyo, Japan. Received: 13 September 2022 Accepted: 14 October 2022 References An R, Xia S, Sun Y, Chang K, Li Y, Shen W (2021) New application of direct lym- phangiography in the diagnosis and treatment of chylothorax after lung cancer surgery: a case series. Ann Palliat Med 10(4):4768–4776 Chick JFB, Hage AN, Patel N, Gemmete JJ, Meadows JM, Srinivasa RN (2018) Chylothorax secondary to venous outflow obstruction treated with transcervical retrograde thoracic duct cannulation with embolization and venous reconstruction. J Vascular Surg Cases Innovations Techniques 4(3):193–196 Ghelfi J, Brusset B, Thony F, Decaens T (2022) Successful management of refractory ascites in non-TIPSable patients using percutaneous thoracic duct stenting. J Hepatol 76(1):216–218 Gurevich A, Nadolski GJ, Itkin M (2018) Novel Lymphatic Imaging and Percutaneous Treatment of Chyluria. Cardiovasc Intervent Radiol 41(12):1968–1971 Hur S, Gurevich A, Nadolski G, Itkin M (2021) Lymphatic Interventional Treat- ment for Chyluria via Retrograde Thoracic Duct Access. J Vasc Interv Radiol 32(6):896–900 Kariya S, Nakatani M, Ono Y et al (2019) Percutaneous Balloon Plasty for Tho- racic Duct Occlusion in a Patient with Chylothorax and Chylous Ascites. Cardiovasc Intervent Radiol 42(5):779–783 Lovrec Krstić T, Šoštarič K, Caf P, Žerdin M (2021) The Case of a 15-Year-Old With Non-Parasitic Chyluria. Cureus 13(8):e17388 Nguyen CN, Le LT, Inoue M et al (2020) Interstitial Lymphatic Embolization with Balloon Assistance for Treatment of Chyluria. J Vasc Interv Radiol 31(3):523–526 Shah UH, Ngoc H, Gupta S (2020) Milky White Urine After Relief of Urinary Retention. J Emerg Med 58(3):e149–e152 Stainer V, Jones P, Juliebø S, Beck R, Hawary A (2020) Chyluria: what does the clinician need to know? Ther Adv Urol 12:175628722094089 Publisher’s Note Springer Nature remains neutral with regard to jurisdictional claims in pub- lished maps and institutional affiliations. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png CVIR Endovascular Springer Journals

Management of chyluria using percutaneous thoracic duct stenting

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Abstract

Background: Thoracic duct stenosis or obstruction is one of the causes of chyluria. Although the diagnosis of chy- luria is not difficult, treatment is still challenging. Although there have been no standard guidelines for the treatment of chyluria, interventional techniques now offer minimally invasive treatment options for chyluria such as interstitial lymphatic embolization, ductoplasty with balloon, or thoracic duct stenting. Case presentation : Here, we report a case of chyluria due to obstruction of the junction between the thoracic duct and subclavian vein in a 64 -year- old female patient. The patient was treated with balloon plasty for lymphovenous junction obstruction and interstitial lymphatic embolization for chyluria. However, chyluria was recurrent after 6 months so intranodal lymphangiography was performed. Anterograde thoracic duct was accessed through a transab- dominal to the cisterna chyli which showed that the thoracic venous junction was re-obstruction. The patient was successfully treated by placing a uncovered drug-eluting stent with the size of 2.5 mm x 15 mm in length for resolv- ing the thoracic occlusion. Conclusion: This report demonstrates the feasibility of using thoracic duct stenting in the treatment chyluria due to lymphovenous junction obstruction. Keywords: Chyluria, Lymphatic, Thoracic duct, Stenosis, Obstruction, Balloon, Stent Introduction diet is the first step. Sclerotherapy using a ureteroscope Chyluria is divided into parasitic and non-parasitic cat- has also been reported, but it is not a well-established egories (Stainer et  al. 2020). Non-parasitic chyluria is a treatment and its effectiveness is limited. As a result, not rare condition and caused by: trauma, surgery, infections, all patients respond well to these therapies (Lovrec Krstić malignancy, lymphatic malformation, radiation, urinary et al. 2021). In the literature, some new alternative treat- retention, congenital fistula between lymphatics and the ments were applied for the management of chyluria such urinary tract, pregnancy and stenosis of the thoracic duct as interstitial lymphatic embolization through percuta- (TD) (Stainer et al. 2020; Shah et al. 2020). There are no neous or retrograde thoracic duct access, and interstitial guidelines for the management of chyluria. The treatment lymphatic embolization with balloon plasty (Gurevich approach depends on the etiology and the site of the lym- et  al. 2018; Hur et  al. 2021; Nguyen et  al. 2020). In this phatic system damage; and mostly should be tailored on a article, we would like to present a new interventional case-by-case basis. Conservative treatment with a low-fat technique for a patient with chyluria associated with the lymphovenous junction (LVJ) obstruction. Case report *Correspondence: cuongcdha@gmail.com A 64 -year- old female patient has suffered from chylu - Diagnostic Imaging and Interventional Center, Hanoi Medical University ria for 6 months. She has no history of parasite infection, Hospital, No1, Tonthattung, Dongda, Hanoi, Vietnam Full list of author information is available at the end of the article no abdominal trauma, and no history of renal operation. © The Author(s) 2022. Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http:// creat iveco mmons. org/ licen ses/ by/4. 0/. Cuong et al. CVIR Endovascular (2022) 5:54 Page 2 of 5 Cystoscopy showed the milky chyle efflux from the right was advanced into the superior vena cava. From the right ureteric orifice suggesting that the origin of chyluria was femoral vein, we advanced a snare into the superior vena from the right kidney. The patient underwent dynamic cave. The snare caught the guide wire and then pulled the contrast-enhanced magnetic resonance lymphangiog- guide wire out of the sheath at the right femoral vein. TD raphy (DCMRL). DCMRL visualized the thoracic duct venous junction was dilated at a pressure of 8 atm using and showed the presence of dilated lymphatic vessels in a balloon 2.5  mm x 20  mm (Pantera LEO, Biotronik, the right renal pelvis confirming the chylo calyceal fistula Bulach, Switzerland) (Fig. 2 C). The interstitial lymphatic (Fig. 1). embolization was then performed with total of 2 ml mix- Bilateral inguinal intranodal lymphangiography was ture of N-butyl cyanoacrylate (NBCA) and lipiodol with then performed. There was a stagnation of Lipiodol the ratio of 1:5 (Fig. 2D) as describe in the literature dur- (Guerbet, France) in the lymphatic system and the cis- ing the inflation of the balloon at the junction TD vein terna chyli appeared after 50 minutes since the injection (Gurevich et al. 2018). of contrast started. In addition, reflux of oil contrast into The patient recovered well with no longer chyluria; the right renal pelvis was also observed. On lymphogra- no complications were noticed. But 6 months after, the phy, the thoracic duct was also dilated and the contrast patient come back to our institution because of recur- was ended at the TD venous junction without draining rent symptoms of chyluria. Cystoscopy showed the efflux in the subclavian vein. Neck ultrasound and echocar- of chyle on the right side, and DCMRL found again the diography revealed there was no neck tumor or central afferent lymphatic vessels in the right kidney. We per - vein thrombosis. There were no anatomical abnormali - formed intranodal lymphangiography and found that ties of the lymphatic system, LVJ as well as the left sub- there was re-obstruction of the TD venous junction. clavian vein. The laboratory tests gave negative results Dilated and tortuous right retroperitoneal lymphatics, with Wuchereria bancrofti, Toxocariasis, Taenia echi- right kidney lymphatics, and filling of kidney calyces nococcus. The diagnosis was made as chyluria from the like the previous lymphangiography was also revealed. right kidney due to an obstruction at the junction TD- Because re-obstruction occurred after balloon plasty subclavian vein. Because the guide wire (0.018” Terumo) 6 months, we planned to re-delate the junction by bal- could pass through the occlusion, the LVJ was dilated by loon and place a metallic stent. The stent we used was balloon and then interstitial lymphatic embolization was a coronary stent with the size of 2.5  mm x 15  mm in done to occlude the lymphatic communication between length (drug-eluting stent) because of the compatible size retroperitoneal lymphatic vessels and the right renal pel- (Fig. 3). After the stent deployment, direct lymphangiog- vis as described in the literature (Gurevich et  al. 2018; raphy demonstrated contrast medium from the thoracic Kariya et al. 2019). We punctured the cisterna chyli by a duct to the left subclavian vein without disruption or 21-gauge-needle (chiba, Cook, USA) and then inserted leakage. Venography of the left subclavian vein showed a 0.018” guide wire into the thoracic duct. A 2.7-french- no reflux flow from the vein into the thoracic duct. Then, microcatheter (progreat, terumo, Japan) was advanced we intended to puncture the interstitial lymphatic ves- into the thoracic duct over the gude wire. By injecting sels at the lumbar region, but all attempts failed. There - the contrast into microcatheter in order to opacify the fore, no embolization of interstitial lymphatic vessels was thoracic duct, we found that there was an occlusion at performed. After procedure, the patient had transient the TD venous junction (Fig.  2 A and B). The long guide hematuria but no chyluria. The patient had remained wire (0.014”, 300 cm, transend soft tip, Boston Scientific, asymptomatic for 1 year later and still under follow- USA) was easily passed through the occlusion and then ing up. The computed tomography (CT) scan at the 6th Fig. 1 DCMRL showed dilated lymphatic vessels in the right renal pelvis (A) and the chylo calyceal fistula (B) C uong et al. CVIR Endovascular (2022) 5:54 Page 3 of 5 Fig. 3 Second intervention. A TD lymphangiography showed re-occlusion of the junction TD vein and collateral circulation of lymphatic vessels in the left neck (arrows). B The stent was inflated in the LVJ by the balloon (arrow). C Injection of contrast after deploying the stent showed the flow into subclavian vein. D superior venogram showed that the contrast material in the left subclavian vein did not reflux to the TD Fig. 2 First intervention. A Lymphangiography revealed dilated the upper part of TD and remained the contrast in the TD (arrow). In terms of management of the chyluria symptoms, con- B injection of contrast into TD showed occlusion of the TD and dilatation at the junction (arrow). C The 0.014”-guidewire passed servative treatment such as a high-protein and low-fat through the occlusion into superior vena cava then the femoral vein diet has a possibility to reduce chyluria. Other treatment by a snare. A 6 French-guiding catheter was placed at the junction options are sclerotherapy and embolization. Sclerosants TD subclavian vein (arrow). D Right retroperitoneal lymphatic system induce an inflammatory reaction in the lymphatic ves - communicates with renal collecting system and the contrast material sels and blockade the communicating channels by fibrosis presented in the kidney calyces (arrow), the interstitial lymphatic embolization was then performed (Lovrec Krstić et  al. 2021; Gurevich et  al. 2018) presented three cases of chyluria and two of them underwent inter- stitial lymphatic embolization by percutaneously accessing month showed the stent fully opened and was in the cor- the retroperitoneum lymphatic channels or retroperito- rect position (Fig.  4  A) and DCMRL lymphangiography neal lymph node under fluoroscopic guidance. These two at the 12th month showed the patency of TD (Fig. 4B). patients had chyluria due to increased thoracic duct pres- sure and they were completely resolved within 1 year and 7 months after the second embolization up to 1 year. Discussion One patient had improved symptoms with a low-fat diet. The TD can be injured via trauma, surgery, or congested Nguyen et al. (2020) also used interstitial lymphatic embo- by central venous occlusion (An et  al. 2021; Chick et  al. lization and accessed the lymphatic channel by puncturing 2018). It may be caused by compression from outside. the retroperitoneal lymph node through the abdominal wall Other causes include lymphatic malformation, radiation, and they put a balloon catheter in the TD in order to pre- congenital abnormalities, and stenosis of the TD (Lovrec vent reflux of embolic agent into the TD. A recent article Krstić et  al. 2021). Valvular insufficiency in the thoracic by Hur et al. (2021) presented another approach into TD by duct results in elevated pressure in the lymphatic circu- retrograde through the left brachial vein or directly punc- lation and backflow of lymphatic fluid into other major turing the TD followed by lymphatic embolization through lymphatic branches. When the thoracic duct pressure is a micro catheter. These treatments method for chyluria increased, it may lead to rupture or reflux of lymphatic were based on disruption of the connection between renal channels and may cause chylous ascites or chylothorax calyces and lumbar interstitial lymphatic vessels. (Kariya et al. 2019). Cuong et al. CVIR Endovascular (2022) 5:54 Page 4 of 5 Fig. 4 Follow up images. A CT scans 6 months post-stenting revealed the stent was in the right position. B DCMRL after one year showed the patency of thoracic duct Regarding to management of the causes, especially TD ducts. There is no blood reflux into the TD, possibly obstruction, there are only a few articles about the treat- because the pressure in the subclavian vein is low and the ment methods. Thoracic ductoplasty with a balloon is same flow direction of the stent and the subclavian vein reported to reduce lymphatic circulation pressure and blood. Moreover, because we used drug-eluting stents, restore the patency of the TD to treat chylothorax and and there was no coagulation factor in the TD so antico- chylous ascites (Kariya et  al. 2019). The results showed agulation was not used in this patient. that after 6 months, the symptoms did not recurrent. The Thoracic duct stenting seems a feasible and practical use of stent in the management of TD obstruction has approach in the treatment of LVJ obstruction. Follow-up not been reported. is necessary to access the patency, location of the stent. Therefore, stenting in management LVJ obstruction of Further studies are needed to confirm these results. the thoracic duct has not been reported and this article may be the first case reported in the literature regard - Conclusion ing to treat TD occlusion. Until now, there has been one Thoracic duct stent is a new procedure with success - article describing TD stenting treatment for TD hyper- ful approach in chyluria with thoracic duct stenosis or tension by Ghelfi et al. (2022). In this article, Ghelfi et al. obstruction. reported two cases with cirrhosis and refractory chylous ascites for which transjugular intrahepatic portosystemic shunt. In these cases, portal hypertension increases lym- Abbreviations TD: Thoracic duct; LVJ: Lymphovenous junction; DCMRL: Dynamic con- phatic flow and may cause the pressure gradient without trast-enhanced magnetic resonancelymphangiography; CT: Computed TD obstruction or stenosis. TD stenting may resolve the tomography. lymph-venous pressure gradient and chylous ascites. The Acknowledgements stent used in our patient was a drug-eluting stent. A part None. of the stent was located in the subclavian vein aimed to keep the stent in the right location. The drug-eluting may Informed consent Informed consent was obtained from patient in the study. prevent thrombosis. Our patient had short-term success with balloon dila- Authors’ contributions tation; however, re-obstruction appeared later. The cause NNC actually engaged in treatment, created the initial draft and contributed to editing and submitting the revised manuscript. LTL performed the proce- of this condition may be fibrosis, intimal hyperplasia, or dure and edited the revised manuscript. TTTM, TQH obtained the images and inflammation. In this case, the internal pressure of the edited the revised manuscript; contributed to the content of the discussion. thoracic duct was not measured before and after the pro- HL, LH and MI provided edits and improvements to the revised manuscript. All authors read and approved the final manuscript. cedure because of a lack of measuring tools. The cause of chyluria in this patient may be increased intratho- Funding racic pressure leading to rupture or reflux of lymphatic The authors have no relevant financial or non-financial interests to disclose. This study was not supported by any funding. channels into the urinary tract. The objective treatment in the present case was to release a blockage in the tho- Availability of data and materials racic duct to reduce pressure in the peripheral lymphatic All data are referenced from the medical records and stored in the hospital. C uong et al. CVIR Endovascular (2022) 5:54 Page 5 of 5 Declarations Ethics approval and consent to participate All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional ethics committee and with the 1964 Helsinki declaration and its later amendments or compara- ble ethical standards. Consent for publication Written informed consent was obtained from the patient for publication of this case report and any accompanying images. Competing interests All authors declare that they have no conflict of interest. Author details Diagnostic Imaging and Interventional Center, Hanoi Medical University Hospital, No1, Tonthattung, Dongda, Hanoi, Vietnam. Surgery of Urology Department, Hanoi Medical University Hospital, Hanoi Medical University, Hanoi, Vietnam. Respiratory Department, Hanoi Medical University Hospital, Hanoi, Vietnam. Keio University, Tokyo, Japan. 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Ther Adv Urol 12:175628722094089 Publisher’s Note Springer Nature remains neutral with regard to jurisdictional claims in pub- lished maps and institutional affiliations.

Journal

CVIR Endovascular – Springer Journals

Published: Oct 19, 2022

Keywords: Chyluria; Lymphatic; Thoracic duct; Stenosis; Obstruction; Balloon; Stent

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