Advertisement

Supermicrosurgical lymphatic venous anastomosis for intractable lymphocele after great saphenous vein harvesting graft

Open AccessPublished:November 22, 2021DOI:https://doi.org/10.1016/j.jvscit.2021.11.003

      Abstract

      Lymphocele results either due to trauma to lymphatic vessels or following a vein graft harvest (as observed in 10–16% of patients). When a lymphocele persists despite conservative treatment, patients may be subjected to undue distress. We report a case of successful treatment of an intractable lymphocele––which had been refractory to conservative treatment including stretch bandage, drainage and local injection for two years––after a great saphenous vein harvest, through lymphatic venous anastomosis (LVA). The lymphocele resolved shortly after LVA without any adverse effect. LVA can be a useful and minimally invasive alternative treatment for lymphocele after harvesting the great saphenous vein.

      Key words

      Introduction

      A lymphocele may occur after trauma to the lymphatic vessels, and 10–16% of patients develop lymphoceles or lymphorrhea after a vein graft harvest [
      • Reifsnyder T.
      • Bandyk D.
      • Seabrook G.
      • Kinney E.
      • Towne J.B.
      Wound complications of the in situ saphenous vein bypass technique.
      ]. Large lymphatic vessels lie adjacent to the great saphenous vein and are likely to be injured during vein grafting [
      • Heitink M.V.
      • Schurink G.W.
      • de Pont C.D.
      • van Kroonenburgh M.J.
      • Veraart J.C.
      Lymphedema after Greater Saphenous Vein Surgery.
      ]. When a lymphocele persists despite conservative treatment including stretch bandage, drainage, and sclerotherapy, patients might suffer due to uncontrollable lymphocele.
      Lymphatic venous anastomosis (LVA) is a surgical treatment that improves lymphatic drainage by anastomosing the lymphatic vessels to a cutaneous vein under surgical microscopy. LVA is one of the alternative methods for the treatment of lymphocele and lymphorrhea [
      • Kobayashi H.
      • Iida T.
      • Yamamoto T.
      • Ikegami M.
      • Shinoda Y.
      • Tanaka S.
      • et al.
      Following resection of soft-tissue sarcomas of the adductor compartment: A report of two cases.
      ,
      • Yamamoto T.
      • Koshima I.
      • Yoshimatsu H.
      • Narushima M.
      • Miahara M.
      • Iida T.
      Simultaneous multi-site lymphaticovenular anastomoses for primary lower extremity and genital lymphoedema complicated with severe lymphorrhea.
      ].
      Herein, we report a rare case of lymphocele that occurred as a postoperative complication of great saphenous vein graft harvest; treatment was successful using LVA. This study was approved by the Institutional Review Board of Hiroshima University Hospital. The patient provided informed consent for the publication of this report.
      Case
      A 70-year-old male patient had renal transplantation following renal failure and his great saphenous vein was harvested for the transplant. He had a history of glomerulonephritis since childhood and underwent renal transplantation at the age of 68 years. During his renal transplantation, the great saphenous vein was harvested from the right thigh to the groin. The patient has been receiving immunosuppressive drugs since then. After the great saphenous vein was harvested, the patient developed a hydrocele under the dermis of the medial side of the right thigh. Approximately 30 ml of fluid was aspirated from the hydrocele once every 2–3 days for two years. After failed treatment using local injection including glucose and minocycline at the site of the hydrocele, the patient visited our hospital for the treatment of an intractable hydrocele.
      The patient had a protuberance over the medial side of the thigh along the scar at the site of where the great saphenous vein was harvested (Fig 1A). Ultrasound study revealed a 3.0 × 3.0 cm-sized hydrocele containing 35 ml clear yellow serum confirmed by fine needle aspiration. Indocyanine green lymphography (ICG) and lymphoscintigraphy findings demonstrated pooling on the medial side of the right thigh (Fig 1B, C); therefore, the patient was diagnosed with a lymphocele. After puncture of the lymphocele, we tried the high pressure of the bandage; however, the lymphocele recurred after 3 days. To treat the intractable lymphocele, we performed LVA to drain the lymph into the vein.
      Figure thumbnail gr1
      Fig 1The patient’s lower extremities. (A) Lymphocele (arrow) in the medial side of the thigh along the scar at the site where the great saphenous vein was harvested. Indocyanine green lymphography (B) and lymphoscintigraphy (C) findings revealed pooling (arrow) at the site of lymphocele.
      One LVA was performed on the medial side of the right thigh and four LVA were performed in the right lower leg (Fig 2). Using 12-0 50-μ needles, four end-to-end and one end-to-side anastomoses were performed. The lymphocele gradually became smaller and disappeared four weeks postoperatively without any puncture. The patient did not wear the bandage after the operation. Eight months after surgery, lymphoscintigraphy demonstrated lymph fluid upstream over the site of the lymphocele and several pathways were found on the medial side of the right thigh, which were more than those observed preoperatively (Fig 3). No recurrence of lymphocele was reported 15 months postoperatively.
      Figure thumbnail gr2
      Fig 2Operative findings in lymphatic vein anastomosis (LVA). One LVA was performed on the medial side of the right thigh, and four LVAs were performed in the right lower leg.
      Figure thumbnail gr3
      Fig 3Comparison of lymphoscintigraphy before and after the surgery. (A) Lymphoscintigraphy before surgery. (B) After surgery, lymphoscintigraphy demonstrated upstream lymph flow over the site of the lymphocele and several pathways were found over the proximal side of right medial thigh (arrow).

      Discussion

      We have described a single case that demonstrated the effectiveness of LVA in treating intractable lymphocele which had been refractory to conservative treatment including stretch bandage, drainage, and local for two years after harvesting the great saphenous vein. Lymphocele and lymphorrhea occur in 10–16% of patients after a great saphenous vein graft is harvested [
      • Reifsnyder T.
      • Bandyk D.
      • Seabrook G.
      • Kinney E.
      • Towne J.B.
      Wound complications of the in situ saphenous vein bypass technique.
      ]. In this case, the patient had been receiving immunosuppressive drugs that correlated to the occurrence of peripheral lymphedema [
      • Ersoy A.
      • Koca N.
      Everolimus-induced lymphedema in a renal transplant recipient: a case report.
      ,
      • Crespo H.S.
      • Roach E.
      • Sakpal S.V.
      • Auvenshine C.
      • Steers J.
      Spontaneous chylous ascites after liver transplantation secondary to everolimus: a case report.
      ]. Intractable lymphocele at the site where a vein is harvested is relatively uncommon and generally underrated by physicians, thereby increasing the risk of morbidity in patients. However, when it occurs, it may be difficult to manage. Furthermore, disruption of the lymphatic vessels during vein harvesting is associated with postoperative lymphedema after harvesting of the great saphenous vein [
      • Unno N.
      • Yamamoto N.
      • Suzuki M.
      • Tanaka H.
      • Mano Y.
      • Sano M.
      • et al.
      Intraoperative lymph mapping with preoperative vein mapping to prevent postoperative lymphorrhea following paramalleolar bypass surgery in patients with critical limb ischemia.
      ].
      Radical treatment for intractable lymphocele is not established. A variety of conservative treatments including stretch bandage, drainage, and local injection have been performed [
      • Karcaaltincaba M.
      • Akhan O.
      Radiologic imaging and percutaneous treatment of pelvic lymphocele.
      ]; however, in some cases failed to conservative treatment. Lymphatic embolization and macroscopic ligation of ruptured lymphatic vessels in the subcutaneous fat were performed; however, disruption of lymph drainage may result in lymphedema [
      • Cha J.G.
      • Lee S.Y.
      • Hong J.
      • Ryeom H.K.
      • Kim G.C.
      • Do Y.W.
      Transpedal lymphatic embolization for lymphorrhea at the graft harvest site after coronary artery bypass grafting.
      ,
      • Sommer C.M.
      • Pieper C.C.
      • Offensperger F.
      • Pan F.
      • Killguss H.J.
      • Köninger J.
      • et al.
      Radiological management of postoperative lymphorrhea.
      ]. LVA, which allows the flow of lymph into the venous circulation, is considered to be the most appropriate therapeutic approach for lymphoedema for it addresses the pathophysiology of the condition. Yamamoto et al. [
      • Yamamoto T.
      • Koshima I.
      • Yoshimatsu H.
      • Narushima M.
      • Miahara M.
      • Iida T.
      Simultaneous multi-site lymphaticovenular anastomoses for primary lower extremity and genital lymphoedema complicated with severe lymphorrhea.
      ] and Yoshida et al. [
      • Yoshida S.
      • Hamada Y.
      • Koshima I.
      • Imai H.
      • Uchiki T.
      • Sasaki A.
      • et al.
      Role of lymphatico venular anastomosis for treatment of lymphorrhea in lower limbs.
      ] performed LVA to treat lymphorrhea accompanied with genital lymphedema and resection of soft tissue sarcomas, respectively, and reported that lymphorrhea resolved soon after LVA. LVA can be an alternative treatment for lymphocele and lymphorrhea; however, no study has reported using LVA for treating lymphocele at the graft harvest site. In this case, multi-site LVA was performed but microsurgical ligation of disrupted lymphatics was considered because of the ligation-induced lymphedema, which was possibly due to the immunosuppressive drugs [
      • Ersoy A.
      • Koca N.
      Everolimus-induced lymphedema in a renal transplant recipient: a case report.
      ,
      • Crespo H.S.
      • Roach E.
      • Sakpal S.V.
      • Auvenshine C.
      • Steers J.
      Spontaneous chylous ascites after liver transplantation secondary to everolimus: a case report.
      ,
      • Karcaaltincaba M.
      • Akhan O.
      Radiologic imaging and percutaneous treatment of pelvic lymphocele.
      ,
      • Cha J.G.
      • Lee S.Y.
      • Hong J.
      • Ryeom H.K.
      • Kim G.C.
      • Do Y.W.
      Transpedal lymphatic embolization for lymphorrhea at the graft harvest site after coronary artery bypass grafting.
      ]. It could also have been completed in a single LVA procedure presenting good results for lymphocele; however, multi-site LVA can be more effective and therapeutic for lymphedema [
      • Mihara M.
      • Hara H.
      • Tange S.
      • Zhou H.P.
      • Kawahara M.
      • Shimizu Y.
      • et al.
      Multisite lymphaticovenular bypass using supermicrosurgery technique for lymphedema management in lower lymphedema cases.
      ]. To divert the flow of the lymphocele-causing lymph to the venous circulation, we performed multi-site LVA by targetting the lymph channel linked to the lymphocele as described by ICG findings.
      Lymphoscintigraphy may help to diagnose as well as treat lymphocele. In case of a lymphocele, lymphoscintigraphy enables successful visualization of the leakage [
      • Lee E.W.
      • Shin J.H.
      • Ko H.K.
      • Park J.
      • Kim S.H.
      • Sung K.B.
      Lymphangiography to treat postoperative lymphatic leakage: A technical review.
      ]. Lymphoscintigraphy demonstrated the pooling on the medial side of the right thigh along the harvesting site of the great saphenous vein; however, upstream lymph flow toward the site of the lymphocele and several pathways were found over the medial side of the right thigh, which were more than those observed preoperatively. These findings suggest that the flow of lymph into the venous circulation by LVA decreased the subcutaneous lymphocele, and the lymph flow reactivation at the proximal medial side of the thigh occurred by decreased lymphocele.
      In conclusion, we performed LVA after failure of conservative treatment of a lymphocele. The surgery was successful with no adverse effects within the patient’s postoperative course. LVA has the potential to be the first line treatment for intractable lymphocele resistant to conservative treatment due to its minimally invasive approach. However, the present report is limited to a single case. Additional studies with a large number of patients are required to prove the efficacy of LVA for the treatment of intractable lymphocele after harvesting of the great saphenous vein.

      Declaration of conflicting interests:

      The authors of this manuscript received JSPS KAKENHI Grant Number JP21K09768.

      Acknowledgements

      None

      References

        • Reifsnyder T.
        • Bandyk D.
        • Seabrook G.
        • Kinney E.
        • Towne J.B.
        Wound complications of the in situ saphenous vein bypass technique.
        J Vasc Surg. 1992; 15: 843-848
        • Heitink M.V.
        • Schurink G.W.
        • de Pont C.D.
        • van Kroonenburgh M.J.
        • Veraart J.C.
        Lymphedema after Greater Saphenous Vein Surgery.
        EJVES Extra. 2009; 18: 41-43
        • Kobayashi H.
        • Iida T.
        • Yamamoto T.
        • Ikegami M.
        • Shinoda Y.
        • Tanaka S.
        • et al.
        Following resection of soft-tissue sarcomas of the adductor compartment: A report of two cases.
        JBJS Case Connect. 2017; 7: e80
        • Yamamoto T.
        • Koshima I.
        • Yoshimatsu H.
        • Narushima M.
        • Miahara M.
        • Iida T.
        Simultaneous multi-site lymphaticovenular anastomoses for primary lower extremity and genital lymphoedema complicated with severe lymphorrhea.
        J Plast Reconstr Aesthet Surg. 2011; 64: 812-815
        • Ersoy A.
        • Koca N.
        Everolimus-induced lymphedema in a renal transplant recipient: a case report.
        Exp Clin Transplant. 2012; 10: 296-298
        • Crespo H.S.
        • Roach E.
        • Sakpal S.V.
        • Auvenshine C.
        • Steers J.
        Spontaneous chylous ascites after liver transplantation secondary to everolimus: a case report.
        Transplant Proc. 2020; 52: 638-640
        • Unno N.
        • Yamamoto N.
        • Suzuki M.
        • Tanaka H.
        • Mano Y.
        • Sano M.
        • et al.
        Intraoperative lymph mapping with preoperative vein mapping to prevent postoperative lymphorrhea following paramalleolar bypass surgery in patients with critical limb ischemia.
        Surg Today. 2014; 44: 436-442
        • Karcaaltincaba M.
        • Akhan O.
        Radiologic imaging and percutaneous treatment of pelvic lymphocele.
        Eur J Radiol. 2005; 55: 340-354
        • Cha J.G.
        • Lee S.Y.
        • Hong J.
        • Ryeom H.K.
        • Kim G.C.
        • Do Y.W.
        Transpedal lymphatic embolization for lymphorrhea at the graft harvest site after coronary artery bypass grafting.
        Yeungnam Univ J Med. 2021; 38: 74-77
        • Sommer C.M.
        • Pieper C.C.
        • Offensperger F.
        • Pan F.
        • Killguss H.J.
        • Köninger J.
        • et al.
        Radiological management of postoperative lymphorrhea.
        Langenbecks Arch Surg. 2021; 406: 945-969
        • Yoshida S.
        • Hamada Y.
        • Koshima I.
        • Imai H.
        • Uchiki T.
        • Sasaki A.
        • et al.
        Role of lymphatico venular anastomosis for treatment of lymphorrhea in lower limbs.
        J Plast Reconstr Aesthet Surg. 2020; 73: 1357-1404
        • Mihara M.
        • Hara H.
        • Tange S.
        • Zhou H.P.
        • Kawahara M.
        • Shimizu Y.
        • et al.
        Multisite lymphaticovenular bypass using supermicrosurgery technique for lymphedema management in lower lymphedema cases.
        Plast Reconstr Surg. 2016; 138: 262-272
        • Lee E.W.
        • Shin J.H.
        • Ko H.K.
        • Park J.
        • Kim S.H.
        • Sung K.B.
        Lymphangiography to treat postoperative lymphatic leakage: A technical review.
        Korean J Radiol. 2014; 15: 724