British Journal of Radiology (2005) 78, 854-857
© 2005 British Institute of Radiology
doi: 10.1259/bjr/61177542
Spontaneous healing of chyle leakage after lymphangiography
T Yamagami, MD, PhD1,
T Masunami, MD1,
T Kato, MD1,
O Tanaka, MD1,
T Hirota, MD1,
T Nomoto, MD, PhD2,
K Mikami, MD, PhD2,
T Miki, MD, PhD2 and
T Nishimura, MD, PhD1
Departments of 1 Radiology and 2 Urology, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, 465 Kajii-chyo, Kawaramachi-Hirokoji, Kamigyo, Kyoto, 602-8566, Japan
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Abstract
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We report a 34-year-old man with the complication of chylous ascites after retroperitoneal lymphadenectomy that was refractory to various conservative therapies. Because surgical treatment for chylous ascites was considered, lymphangiography was performed to identify the area of leakage of chyle, after which the chylous ascites spontaneously healed.
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Introduction
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Chylous ascites is a rare complication after retroperitoneal lymph node resection, reported to occur in 2% of such cases according to Baniel et al [1]. Most cases of post-operative chylous ascites heal with conservative therapy such as therapeutic paracentesis, a high protein, low fat, medium-chain triglyceride diet and total parenteral nutrition [2, 3]. If uncontrolled, chylous ascites can be life-threatening because of significant loss of fluid, plasma protein, fats and immunoregulatory lymphocytes, which increases the susceptibility to infection [4]. Thus, surgical treatment such as surgical closure of the lympho-peritoneal fistula and creation of a peritoneovenous fistula has been performed [2]. Recently, a treatment less invasive than surgery, that is, embolisation via percutaneous transabdominal catheterization of the cisterna chyli or lymphatic ducts, has been reported [5]. However, this [5] may be the only report of such a procedure and further evaluation is necessary before the procedure can be recommended for routine management of chylous ascites.
Recently, we experienced a case of chylous ascites occurring after retroperitoneal lymphadenectomy that was refractory to conservative therapies, but healed spontaneously after lymphangiography, with the result that invasive surgical treatment could be avoided.
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Case report
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A 34-year-old man with retroperitoneal lymph node metastasis from cancer of the right testis underwent retroperitoneal lymphadenectomy by surgical laparotomy. The principal site of lymph node dissection was the juxta-aortic, bilateral common and external iliac area. 5 months previously, this patient had undergone surgical resection of the right testis and subsequently systemic intravenous chemotherapy was performed. The removed testicular tumour had been pathologically diagnosed as a mixture of seminoma, immature teratoma and embryonal carcinoma. Before surgical removal of the right testis, retroperitoneal lymph node and multiple lung metastasis had been revealed on CT images. After systemic chemotherapy, retroperitoneal lymph node metastasis had decreased while the lung metastasis disappeared completely, as shown on CT images.
After surgical lymphadenectomy, abdominal distention increased. The drainage tube inserted percutaneously 19 days after the lymphadenectomy revealed milk-like fluid, which led us to strongly suspect post-operative chyle leakage. Volume drained was more than 1000 ml day–1. Chylous ascites did not decrease in spite of conservative therapy such as diet therapy, which included a low fat, medium-chain triglyceride diet and total parenteral nutrition.
In considering surgical intervention to diagnose and localize the chyle leakage, bipedal ascending lymphangiography was performed 55 days after surgical retroperitoneal lymphadenectomy, as follows. First, 2.5 ml of indigocarmine (Daiichi Pharmaceutical, Tokyo, Japan), a dye that may stain the lymphatics, was injected into the web space between the first and second toes on each foot. 30 min later, linear cut-down was performed on the dorsum of the foot below the ankle and the lymphatic vessel was isolated. After cannulation of a lymphatic vessel on the dorsum of each foot using a 30 gauge needle, 6 ml of iodized oil (Lipiodol; Laboratoire Guerbet, Roissy, France), which is a contrast agent for lymphangiography, was injected at a rate of 0.1 ml min–1.
Roentgenogram obtained 1 h after initiating insertion of the contrast agent showed contrast pooling in the retroperitoneal space close to the left common iliac lymph vessels (Figure 1a
). Transverse CT images obtained 3 h after this roentgenography revealed contrast agent distributed mainly in the retroperitoneal space at the ventral site of the fifth lumbar vertebrae (Figure 1b). Pooling of the contrast agent was also seen at the right iliac fossa in the abdominal space. These CT images also showed a large amount of fluid collected in the entire abdominal cavity. Roentgenogram obtained the next day revealed that the contrast agent in the lymphatic vessel on the left side did not ascend beyond the level of the fifth lumbar vertebrae and that the area of leaked contrast agent outside the lymphatic vessel was greater compared with that obtained the day before (Figure 1c, d). From these images, we confirmed that chyle leakage originated from the lymph vessel running in the left common iliac lymph vessel and that leaked fluid expanded into the left retroperitoneal space and flowed into the abdominal cavity. Because the area of surgical lymphadenectomy corresponded to the area of leakage, this chyle leakage was strongly suspected to result from surgical disruption of the retroperitoneal lymph vessel.
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Figure 1. A 34-year-old man with chylous ascites as a complication of retroperitoneal lymphoidectomy. (a) Roentgenogram obtained 1 h after initiating administration of contrast agent for lymphangiography shows contrast pooling at the level of the fifth lumbar vertebrae close to the left common iliac lymph vessels, which might show leakage of contrast agent (arrowhead) from the left common iliac lymph vessels (arrows). (b) Unenhanced CT image obtained 3 h after completion of injection of contrast agent for lymphangiography revealed contrast pooling in the retroperitoneum (arrowhead) and ascitic fluid in the right iliac fossa (thick arrow). Contrast distributed anterior to the fifth lumbar vertebrae (arrowhead) might be due to leakage from the left common iliac lymph vessels. Note that a large amount of collected fluid is evident in the abdominal cavity (thin arrow). (c, d) Roentgenogram obtained the day following lymphangiography revealed that the contrast agent in the lymphatic vessel of the left side did not ascend beyond the level of the fifth lumbar vertebrae and that the area of the leaked contrast agent had increased (arrowhead). Note that leaked contrast agent distributed in the retroperitoneal space at the left side from the level of the fifth lumbar vertebrae to the level of the second lumbar vertebrae (arrows). (e) Enhanced CT image obtained 19 days after lymphangiography shows no fluid collected in the abdominal space and an encapsulated cystic mass that contains some contrast agent peripherally in the retroperitoneal space at the ventral site of the lumbar vertebrae (arrowhead). The area of encapsulated cystic mass is consistent with the area of leakage from the left common iliac lymph vessel as shown on previous lymphangiography. Note that contrast agent injected at the time of lymphangiography remained in the bilateral common and external iliac lymph vessels with the exception of the left common iliac area above the level of the fifth lumbar vertebrae.
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From the day following the lymphangiography, the volume of drained fluid gradually decreased and finally stopped. The drainage tube was withdrawn 14 days after lymphangiography. On CT images obtained 19 days after lymphangiography, an encapsulated cyst that contained some contrast agent peripherally was apparent in the retroperitoneal space at the ventral site of the lumbar vertebrae. This was consistent with the area of leakage from lymph vessels as revealed on lymphangiography performed 19 days before (Figure 1e
). No other fluid was collected in the abdominal cavity. Contrast agent used at the time of lymphangiography remained in the bilateral common and external iliac lymph vessels with the exception of the left common iliac area above the level of the fifth lumbar vertebrae, as would be expected. This patient was completely relieved from abdominal distension and was not restricted in activities of daily life. Hence, he was discharged 34 days after lymphangiography. Currently, 9 months after lymphangiography, chylous ascites has not been observed in this patient.
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Discussion
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It is commonly considered that lymphangiography is useful to diagnose and identify chyle leakage, after which surgical repair can be performed [6]. Among complications of lymphangiography, pulmonary oil embolisation is recognized [7, 8]. This is known to be caused by Lipiodol, which is a contrast agent used in lymphangiography, acting as an embolus after passing the lymphatic-venous anastomoses [7, 8]. This theory can be applied to elucidate the mechanism of oil embolisation after lymphangiography occurring in other organs such as liver [9] and cerebrum [10]. Because of Lipiodol's ability to act as an embolus, this contrast agent is used in some situations in therapeutic embolisation. In a majority of procedures, Lipiodol is added to other embolic agents to enhance their therapeutic ability [11, 12]. Also, when mixed with anticancer drugs in addition to other embolic agents such as gelatin sponge in transcatheter arterial chemoembolisation for hepatic cancer [13], it is useful to prevent early release of the chemotherapeutic agent from Lipiodol that was lodged in the arterial branches that supply blood to the cancer lesion.
To our knowledge, there has been no report of the use of Lipiodol as a therapeutic embolic agent for lymphatic vessels. In the present case, chyle leakage as a complication of surgical lymphadenectomy that was refractory to conservative therapies disappeared spontaneously after lymphangiography. Of course, there remains a possibility that chylous ascites can just heal with repeated drainage. However, considering the fact that the chyle leakage was refractory to any therapies including repeated drainage with a volume of more than 1000 ml day–1 and that it began to decrease promptly after lymphangiography, there is no doubt that lymphangiography played an important role in the disappearance of chyle leakage in this patient. This can be explained by the effect of Lipiodol as an embolic agent through the following sequence of events. First, Lipiodol accumulated at the point of leakage outside the lymphatic vessel. Next, a regional inflammatory reaction occurred in the soft tissue adjacent to the area of Lipiodol retention. Third, the point of leakage of the lymphatic vessel was obstructed. Finally, Lipiodol retention inside the lymphatic vessel just at the distal side of the point of leakage played a role as a therapeutic embolic agent, as was demonstrated by the fact that Lipiodol had not advanced beyond that point on roentgenogram obtained 1 day after lymphangiography. In addition, compression by the cystic mass (Figure 1e) formed from the leaked chyle may have influenced the obstruction of the point of leakage of the lymphatic vessel.
With the recent rapid development of CT and MR imaging, including modalities to evaluate the lymphatic systems, the role of lymphangiography has diminished. However, this traditional imaging method is still important to diagnose and identify chylous leakage and should be performed in cases refractory to conservative therapies and when surgery is considered, because it is possible that, as in the present case, chyle leakage will disappear spontaneously after lymphangiography, resulting in avoidance of surgery, which is, of course, invasive. It goes without saying that in cases in which chyle leakage does not stop even after lymphangiography, other treatments such as embolisation via percutaneous transabdominal catheterization of the cisterna chyli or lymphatic ducts [5] or traditional surgical treatment should be administered.
Received for publication January 14, 2005.
Revision received March 7, 2005.
Accepted for publication April 12, 2005.
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Abstract
Introduction
