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Lymphography in a Patient with Unilateral Chyluria
THE JOURNAL OF UROLOGY
Vol. 92, No. 5 November 1964 Copyright © 1964 by The Williams & Wilkins Co. Printed in U.S.A.
LYMPHOGRAPHY IN A PATIENT WITH UNILATERAL CHYLURIA TAKASHI KISHIMOTO, TERUO HIGUCHI, MINOR! ENDO
AND
YOSHIO KAI
From the Department of Urology, Self-Defense Forces Central Hospital, Tokyo, Japan
Chyluria of filarial origin is not a rarity in Japan. This paper presents a case of unilateral hematochyluria in which lymphography clearly demonstrates abnormal pelvic and para-aortic lymphatics along with the retrograde flow of contrast material into the renal lymphatics and calyces. Reference is made to the surgical interception of perirenal lymph vessels by stripping of renal pedicle which successfully cured hematochyluria. CASE REPORTS
JVI. A., a 61-year-old male Japanese, was admitted to the Self-Defense Forces Central Hospital on June 6, 1963 with persistent chyluria 2 years in duration. The patient was born and has always lived in Kagoshima Prefecture, the southern extremity of Japan, where filariasis is endemic. In August 1960 and August 1961 the patient had temperatures up to 104F of unknown etiology, each lasting about 2 weeks on palliative treatment. In September 1962, the patient began complaining of an occasional dull ache in the right flank radiating toward the lower abdomen. At another hospital in March 1963, a diagnosis of right renal chyluria due to filariasis was made on the basis of laboratory data including discovery of microfilaria in the peripheral blood. The patient was then given a piperazine derivative 0.3 gm. a day orally. After approximately 3 months of therapy, microfilaria disappeared from the blood although chyluria persisted. During the 1month period prior to hospitalization, the milky urine became bloody and the patient began having dysuria. Physical examination revealed no gross abnormalities. There was no edema. The urine was sanguineous and chylous (hematochyluria and fibrinuria). No microfilaria were recovered from the blood even following provocative procedures. Cystoscopy revealed normal mucosa and efflux of bloody chylous urine on the right side. Excretory urography demonstrated a normal collecting system and prompt dye excretion of either kidney, but it seemed to be somewhat unusual that the right kidney wa8 located slightly above the level Accepted for publication April 8, 1964.
of the left kidney. On retrograde pyelography considerable pyelolymphatic reflux from each calyx toward the hilum was observed on the right side (fig. 1, A). Seven days following retrograde pyelography, lymphography (lymphangioadenography) was performed. The technique used was the one described by Kinmonth and modified by Wallace and associates. Ten milliliters of domestic oil contrast material (almost identical with ethiodol) were slowly injected into a lymphatic vessel in the right foot. No injection was made on the left side. A lymphangiogram taken immediately after the injection is shown in figure 1, B. A ureteral catheter had been inserted to the right renal pelvis in order to investigate the relation between the lymphatic channels and the upper urinary tract. A massively dilated vermiform plexus of lymphatic channels was visualized in the right pelvic region. Contrast material injected only into the right foot entered the para-aortic lymphatic channels of the opposite side abundantly above the sacral region. This finding is totally abnormal. Bilateral para-aortic lymphatics were also markedly dilated and tortuous though the left side was more extensively affected. The lymphatics of the right kidney were well visualized, as shown by the retrogTade pyelogram. A portion of contrast material was present in the right renal calyces. Such filling of the renal lymphatics on a lymphangiogram is definitely abnormal and indicates backflow of lymph to the kidney. Despite seemingly more advanced change of the left abdominal lymphatic channels, no obvious retrograde flow of contrast material into the left renal lymphatics was demonstrable. The thoracic duct was not visualized. Like the immediate lymphang-iogram, a 24-hour-film showed many right pelvic and left para-aortic lymph nodes (fig. 2, A). Nevertheless, no right abdominal lymph nodes were visualized except for a few present on the right side of the first and second lumbar vertebrae. Some of the lymph nodes visualized were irregular in contour and density just like "motheaten" nodes observed in metastatic canrer.
574
LYMPHOGRAPHY IN PATIENT WITH UNILATERAL CHYLURIA
575
Fm. 1. A, retrograde pyelogram demonstrates definite pyelolymphatic reflux on right side. B, immediate lymphangiogram following lymphatic vessel injection in right foot.
These are pathological findings and their significance will be discussed later. Two days following lymphography, the patient experienced severe right flank pain with acute urinary retention. The bladder was filled with bloody, chylous clots which had to be removd with an evacuator. It is not known whether this aggravation of chyluria was a coincidental remission of the disease or a result of promoted communication already existing between lymphatics and the urinary tract. The patient was kept on bed rest and given a low fat diet without remarkable change in chyluria. Instillation of 1 per cent silver nitrate solution into the right renal pelvis ,vas ineffectual. Inasmuch as the hematochyluria was refractory to traditional measures, surgical treatment was decided upon. The operation, performed on June 24, consisted of removing all lymphatic and connective tissue around the renal vascular pedicle, thereby skeletonizing the blood vessels. Numerous dilated lymphatic vessels, the thickest reaching 3 mm. in outside diameter, could be seen
entangled with the renal vascular peclicle. This finding is compatible with that of lymphangiography. Histological study of the resected lymphatics revealed dilatation of the lumen and swelling of the wall without inflammatory change. The result was dramatic. Immediately after operation the hematochyluria disappeared. Retrograde pyelography performed on the ninth postoperative day revealed no pyelolymphatic reflux (fig. 2, B). The film is also a 15-day lymphadenogram, showing almost the same nodes as seen on a 24-hour film. Six months after operation the chyluria had not recurred and the patient was leading a healthy, normal life. DISCUSSION
In 1945 Yamauchi published an excellent reveiw of 45 cases of chyluria observed in Hawaii. 1 In general, however, chyluria being a relatively infrequent condition, most of the previous reports were presentation of individual cases with a re1 Yamauchi, S.: Chyluria: Clinical laboratory and statistical study of 45 personal cases observed in Hawaii. J. Urol., 54: 318-347, 1945.
576
KISHIMOTO, HIGUCHI, ENDO AND KAI
FIG. 2. A, 24-hour film of lymphography. B, retrograde pyelogram taken 9 days postoperatively. Film is also 15-day lymphadenogram. view of the literature.2-1 Chyluria is usually classified as parasitic (tropical) or non-parasitic (nontropical), though the latter is quite uncommon. All the cases recently reported are in Puerto Ricans, Filipinos or Chinese and are presumably due to filariasis. Japan, the southern part in particular, is where filariasis is endemic with the incidence of 10 to 20 per cent of the local population. Chyluria is not a rare complication of the disease. Almost all cases of chyluria observed in Japan are due to filariasis caused by Wuchereria 2 Torres, L. F. and Estrada, J.: Experiences in the treatment of chyluria. J. Urol., 87: 73-76, 1962. 3 Morgan, R.R. and Larotunda-Formato, M. L.: Chyluria: Report of a case and review of the literature. J. Urol., 87: 200-202, 1962. 4 Cockett, A. T. K. and Goodwin, W. E.: Chyluria: Attempted surgical treatment by lymphaticvenous anastomosis. J. Urol., 88: 566-568, 1962. 5 Tuller, M.A., Feuer, M. M., Schapira, H. E. and Ho, P. P.: Recumbent chyluria: Demonstration of unilateral renal-lymphatic communication. Amer. J. Med., 33: 951-956, 1962. 6 Kittredge, R. D., Hashim, S., Roholt, H. B., Van Itallie, T. B. and Finby, N.: Demonstration of lymphatic abnormalities in a patient with chyluria. Amer. J. Roentgenol., 90: 159-165, 1963. 7 Swanson, G. E.: Lymphangiography in chyluria. Radiology, 81: 473-478, 1963.
bancrofti. A statistical analysis of 2,222 cases of chyluria, discovered throughout Japan during 5 years from 1957 to 1961 showed that 77 per cent of the cases were encountered in southern Japan. 8 The mechanism of retrograde flow of lymph has been much debated upon, but it is generally agreed that lymph appears in the urine through the ruptured renal calyces or pelvis by virtue of abnormal communication between retroperitoneal lymphatics and the renal pelvis around the renal vascular pedicle. Support for this viewpoint has come from such sources as 1) pyelolymphatic reflux on retrograde pyelogram, 2) results of surgical removal of all the lymphatics around the renal vascular pedicle (perirenal lymph shunt), and 3) histological findings of extirpated kidneys. Since Kinmonth 9 first described a practical method of lymphography (lymphangioadenography), numerous excellent reports have attested to the value of this procedure. 10 - 19 Naturally, an 8 Okamoto, K.: Chyluria. Read before Symposium on Filariasis at 16th General Assembly, The Japan Medical Congress, Osaka, 1963. 9 Kinmonth, J. B.: Lymphangiography in man. Method of outlining lymphatic trunks at operation. Olin. Sc., 11: 13-20, 1952. 10 Sheehan, R., Hershchyshyn, M., Lin, R. K.
LYMPHOGRAPHY IN PATIENT WITH UNILATERAL CHYLURIA
attempt to visualize abnormal lymphatics in chyluria was considered as reasonable, and there was an opportunity for us to study the case reported here. Several similar accounts on lymphography in chyluric patients have been reported. 6· 7 • 20 The lymphograms illustrated m these articles showed abnormal lymphatics m the pelvic and para-aortic regions as well as retrograde flow of contrast material into the renal lymphatics and calyces as seen in our case, although there was some difference in the degree of visualization. In the second case reported the Swanson 7 there was so much contrast material traveling to the kidney that the calyces were completely outlined. In chyluria due to filariasis principal lesions are found in the lymphatics of the pelvic, lumbar and retroperitoneal regions. In some instances, however, as shown in several autopsy cases, the whole lymphatic system including the thoracic duct is extensively involved. Histologically, lymphatic and Lessmann, F. P.: The use of lymphography as a diagnostic method. Radiology, 76: 47-53, 1961. 11 Wallace, S., Jackson, L., Schaffer, B., Gould, J., Greening, R.R., Weiss, A. and Kramer, S.: Lymphangiograms: Their diagnostic and therapeutic potential. Radiology, 76: 179-199, 1961. 12 Fuchs, W. A. and Bo6k-Hederstr6m, G.: Inguinal and pelvic lymphography. A preliminary report. Acta radiol., 56: 340-354, 1961. 13 Ruttimann, V. A., Del Buono, JVI. S. and Cocchi, U.: Neue Fortschritte in der Lymphographie. Schweiz.Med. Wschr., 91: 1460-1466, 1961. 14 Viamonte, M., Jr., 11:yers, M. B., Soto, 11:., Kenyon, N. JVI. and Parks, R. E.: Lymphography: Its role in detection and therapeutic evaluation of carcinoma and neoplastic conditions of the genitourinary tract. J. Urol., 87: 85-90, 1962. 15 Jacobsson, S. and Johansson, S.: Lymphangiography in lymphedema. Acta radiol., 57: 81-89,
577
vessels lose elastic tissue with dilated lumen, thickened wall and incompetent valves, and reactive as well as cicatrical changes take place in lymph nodes, leading to obstruction of the afferent lymphatics. Lymphatic varicosities and poor or loss of visualization of lymph nodes on lymphogram correspond to those histological alterations. The degree of visualization may be quite variable, presumably reflecting the extent of an underlying pathological process. Behaving like varicose veins, lymphatic varicosities probably rupture into the urinary tract owing to the in~ creased hydrostatic pressure caused by lymph stasis because of incompetent valves, and become demonstrable on lymphography. In our case, the contrast material entered freely the contralateral abdominal lymphatics with cross-over on injection exclusively in the right foot. This was interpreted mainly as indicating abnormal dilation of the lymphatics in the sacral region communicating both sides. On the right side, where retrograde flow of contrast material into the renal lymphatics was \Nell visualized, few lymph nodes were demonstrable. On the contrary, there were numerous abnormal abdominal lymph nodes on the left side. These findings suggest more marked involvement
I
1962. 16 Fischer, H. W., Lawrence, M. S. and Thornbury, J. R.: Lymphography of the normal adult male: Observations and their relation to the diagnosis of metastatic neoplasm. Radiology, 78: 399-
406, 1962. 17 Wallace, S., Jackson, L. and Greening, R.R.: Clinical applications of lymphangiography. Amer. J. Roentgenol., 88: 97-109, 1962. 18 Viamonte, JVI., Jr., Altman, D., Parks, R., Blum, E., Bevilacqua, :i\I. and Recher, L.: Radiographic-pathologic correlation in the interpretation of lymphangioadenograms. Radiology, 80:
903-916, 1963.
19 Schaffer, B., Koehler, P. R., Daniel, C. R., Wohl, G. T., Rivera, E., ]\!foyers, W. A. and Skelley, J. F.: A critical evaluation of lymphangiography. Radiology, 80: 917-930, 1963. 20 Turiaf, J., Arvay, N., Picard, J. D., Servelle, M. and Gentilini, M.: Donnees de la lymphographie dans deux cas de chylurie filarienne. Bull. Soc. Med. d. hop. de Paris, 113: 753-766, 1962. Cited by Swanson, G. E. 7
Frn. 3. Immediate lymphangiogram of additional case.
I
578
KISHIMOTO, HIGUCHI, ENDO AND KAI
in the right abdominal lymphatics than in the left, although possibly chyluria might recur from the left side since considerable change is seen there. The measures to control chyluria include a low fat diet, bed rest, medication, instillation or irrigation of the renal pelvis with a sclerosing agent, and surgery. One must r(:1sort to surgery only after failure of other therapeutic means. As an operative procedure nephrectomy should be avoided unless inevitable. Conventional capsulectomy alone usually proves to be unsatisfactory as stated in previous reports. The method of choice appears to be removal of all lymphatics around the renal vascular pedicle. If this is done completely, concomitant capsulectomy is not necessary. Lately Torres and Estrada reported on this surgical approach along with two personal cases. 2 Another procedure, lymphaticovenous anastomosis, has been attempted. 4 Besides the case just reported as being cured of obstinate chyluria after removal of lymphatics around the renal vascular pedicle, the authors treated one more patient in the same manner without recurrence in 2 years postoperatively. In view of those roentgenograms, findings at operation and favorable outcome, this procedure is worthy to be recommended for cases in which other therapeutic measures are ineffective. According to the Japanese statistics cited earlier, 8 there were 70 patients who underwent surgery during the 5 years (1957-1961). Chyluria disappeared in 53 (75.7 per cent), improved in 14 (20.0 per cent), a11diwas unchanged in 3 (4.3 per
cent). The long-term recovery rate was 71.9 per cent, while recurrence on the operated side developed in 28.1 per cent. Better results may be obtained by more meticulous removal of the lymphatics around the renal vascular pedicle. SUMMARY
The case of a patient with unilateral hematochyluria has been described. Abnormal communication between the para-aortic and the renal lymphatics had been demonstrated on lymphography (lymphangioadenography). Retrograde fl.ow of some contrast material from the renallymphatics into the renal calyces had also been noted. Following an operation that completely removed numerous dilated lymphatics around the renal vascular pedicle shown on lymphography, chyluria disappeared immediately and has not recurred for the past 6 months. ADDENDUM
After this paper was completed, the authors had an opportunity to perform lymphography on one more patient with chyluria. The case was in a 60-year-old male Japanese who had left renal chyluria due to filariasis. The i=ediate lymphogram following lymphatic vessel injection of this patient is shown. Ten milliliters of co.ntrast material were injected in each foot, demonstrating. a considerable degree of retrograde fl.ow of the contrast material into the left renal lymphatics.
Vol. 92, No. 5 November 1964 Copyright © 1964 by The Williams & Wilkins Co. Printed in U.S.A.
LYMPHOGRAPHY IN A PATIENT WITH UNILATERAL CHYLURIA TAKASHI KISHIMOTO, TERUO HIGUCHI, MINOR! ENDO
AND
YOSHIO KAI
From the Department of Urology, Self-Defense Forces Central Hospital, Tokyo, Japan
Chyluria of filarial origin is not a rarity in Japan. This paper presents a case of unilateral hematochyluria in which lymphography clearly demonstrates abnormal pelvic and para-aortic lymphatics along with the retrograde flow of contrast material into the renal lymphatics and calyces. Reference is made to the surgical interception of perirenal lymph vessels by stripping of renal pedicle which successfully cured hematochyluria. CASE REPORTS
JVI. A., a 61-year-old male Japanese, was admitted to the Self-Defense Forces Central Hospital on June 6, 1963 with persistent chyluria 2 years in duration. The patient was born and has always lived in Kagoshima Prefecture, the southern extremity of Japan, where filariasis is endemic. In August 1960 and August 1961 the patient had temperatures up to 104F of unknown etiology, each lasting about 2 weeks on palliative treatment. In September 1962, the patient began complaining of an occasional dull ache in the right flank radiating toward the lower abdomen. At another hospital in March 1963, a diagnosis of right renal chyluria due to filariasis was made on the basis of laboratory data including discovery of microfilaria in the peripheral blood. The patient was then given a piperazine derivative 0.3 gm. a day orally. After approximately 3 months of therapy, microfilaria disappeared from the blood although chyluria persisted. During the 1month period prior to hospitalization, the milky urine became bloody and the patient began having dysuria. Physical examination revealed no gross abnormalities. There was no edema. The urine was sanguineous and chylous (hematochyluria and fibrinuria). No microfilaria were recovered from the blood even following provocative procedures. Cystoscopy revealed normal mucosa and efflux of bloody chylous urine on the right side. Excretory urography demonstrated a normal collecting system and prompt dye excretion of either kidney, but it seemed to be somewhat unusual that the right kidney wa8 located slightly above the level Accepted for publication April 8, 1964.
of the left kidney. On retrograde pyelography considerable pyelolymphatic reflux from each calyx toward the hilum was observed on the right side (fig. 1, A). Seven days following retrograde pyelography, lymphography (lymphangioadenography) was performed. The technique used was the one described by Kinmonth and modified by Wallace and associates. Ten milliliters of domestic oil contrast material (almost identical with ethiodol) were slowly injected into a lymphatic vessel in the right foot. No injection was made on the left side. A lymphangiogram taken immediately after the injection is shown in figure 1, B. A ureteral catheter had been inserted to the right renal pelvis in order to investigate the relation between the lymphatic channels and the upper urinary tract. A massively dilated vermiform plexus of lymphatic channels was visualized in the right pelvic region. Contrast material injected only into the right foot entered the para-aortic lymphatic channels of the opposite side abundantly above the sacral region. This finding is totally abnormal. Bilateral para-aortic lymphatics were also markedly dilated and tortuous though the left side was more extensively affected. The lymphatics of the right kidney were well visualized, as shown by the retrogTade pyelogram. A portion of contrast material was present in the right renal calyces. Such filling of the renal lymphatics on a lymphangiogram is definitely abnormal and indicates backflow of lymph to the kidney. Despite seemingly more advanced change of the left abdominal lymphatic channels, no obvious retrograde flow of contrast material into the left renal lymphatics was demonstrable. The thoracic duct was not visualized. Like the immediate lymphang-iogram, a 24-hour-film showed many right pelvic and left para-aortic lymph nodes (fig. 2, A). Nevertheless, no right abdominal lymph nodes were visualized except for a few present on the right side of the first and second lumbar vertebrae. Some of the lymph nodes visualized were irregular in contour and density just like "motheaten" nodes observed in metastatic canrer.
574
LYMPHOGRAPHY IN PATIENT WITH UNILATERAL CHYLURIA
575
Fm. 1. A, retrograde pyelogram demonstrates definite pyelolymphatic reflux on right side. B, immediate lymphangiogram following lymphatic vessel injection in right foot.
These are pathological findings and their significance will be discussed later. Two days following lymphography, the patient experienced severe right flank pain with acute urinary retention. The bladder was filled with bloody, chylous clots which had to be removd with an evacuator. It is not known whether this aggravation of chyluria was a coincidental remission of the disease or a result of promoted communication already existing between lymphatics and the urinary tract. The patient was kept on bed rest and given a low fat diet without remarkable change in chyluria. Instillation of 1 per cent silver nitrate solution into the right renal pelvis ,vas ineffectual. Inasmuch as the hematochyluria was refractory to traditional measures, surgical treatment was decided upon. The operation, performed on June 24, consisted of removing all lymphatic and connective tissue around the renal vascular pedicle, thereby skeletonizing the blood vessels. Numerous dilated lymphatic vessels, the thickest reaching 3 mm. in outside diameter, could be seen
entangled with the renal vascular peclicle. This finding is compatible with that of lymphangiography. Histological study of the resected lymphatics revealed dilatation of the lumen and swelling of the wall without inflammatory change. The result was dramatic. Immediately after operation the hematochyluria disappeared. Retrograde pyelography performed on the ninth postoperative day revealed no pyelolymphatic reflux (fig. 2, B). The film is also a 15-day lymphadenogram, showing almost the same nodes as seen on a 24-hour film. Six months after operation the chyluria had not recurred and the patient was leading a healthy, normal life. DISCUSSION
In 1945 Yamauchi published an excellent reveiw of 45 cases of chyluria observed in Hawaii. 1 In general, however, chyluria being a relatively infrequent condition, most of the previous reports were presentation of individual cases with a re1 Yamauchi, S.: Chyluria: Clinical laboratory and statistical study of 45 personal cases observed in Hawaii. J. Urol., 54: 318-347, 1945.
576
KISHIMOTO, HIGUCHI, ENDO AND KAI
FIG. 2. A, 24-hour film of lymphography. B, retrograde pyelogram taken 9 days postoperatively. Film is also 15-day lymphadenogram. view of the literature.2-1 Chyluria is usually classified as parasitic (tropical) or non-parasitic (nontropical), though the latter is quite uncommon. All the cases recently reported are in Puerto Ricans, Filipinos or Chinese and are presumably due to filariasis. Japan, the southern part in particular, is where filariasis is endemic with the incidence of 10 to 20 per cent of the local population. Chyluria is not a rare complication of the disease. Almost all cases of chyluria observed in Japan are due to filariasis caused by Wuchereria 2 Torres, L. F. and Estrada, J.: Experiences in the treatment of chyluria. J. Urol., 87: 73-76, 1962. 3 Morgan, R.R. and Larotunda-Formato, M. L.: Chyluria: Report of a case and review of the literature. J. Urol., 87: 200-202, 1962. 4 Cockett, A. T. K. and Goodwin, W. E.: Chyluria: Attempted surgical treatment by lymphaticvenous anastomosis. J. Urol., 88: 566-568, 1962. 5 Tuller, M.A., Feuer, M. M., Schapira, H. E. and Ho, P. P.: Recumbent chyluria: Demonstration of unilateral renal-lymphatic communication. Amer. J. Med., 33: 951-956, 1962. 6 Kittredge, R. D., Hashim, S., Roholt, H. B., Van Itallie, T. B. and Finby, N.: Demonstration of lymphatic abnormalities in a patient with chyluria. Amer. J. Roentgenol., 90: 159-165, 1963. 7 Swanson, G. E.: Lymphangiography in chyluria. Radiology, 81: 473-478, 1963.
bancrofti. A statistical analysis of 2,222 cases of chyluria, discovered throughout Japan during 5 years from 1957 to 1961 showed that 77 per cent of the cases were encountered in southern Japan. 8 The mechanism of retrograde flow of lymph has been much debated upon, but it is generally agreed that lymph appears in the urine through the ruptured renal calyces or pelvis by virtue of abnormal communication between retroperitoneal lymphatics and the renal pelvis around the renal vascular pedicle. Support for this viewpoint has come from such sources as 1) pyelolymphatic reflux on retrograde pyelogram, 2) results of surgical removal of all the lymphatics around the renal vascular pedicle (perirenal lymph shunt), and 3) histological findings of extirpated kidneys. Since Kinmonth 9 first described a practical method of lymphography (lymphangioadenography), numerous excellent reports have attested to the value of this procedure. 10 - 19 Naturally, an 8 Okamoto, K.: Chyluria. Read before Symposium on Filariasis at 16th General Assembly, The Japan Medical Congress, Osaka, 1963. 9 Kinmonth, J. B.: Lymphangiography in man. Method of outlining lymphatic trunks at operation. Olin. Sc., 11: 13-20, 1952. 10 Sheehan, R., Hershchyshyn, M., Lin, R. K.
LYMPHOGRAPHY IN PATIENT WITH UNILATERAL CHYLURIA
attempt to visualize abnormal lymphatics in chyluria was considered as reasonable, and there was an opportunity for us to study the case reported here. Several similar accounts on lymphography in chyluric patients have been reported. 6· 7 • 20 The lymphograms illustrated m these articles showed abnormal lymphatics m the pelvic and para-aortic regions as well as retrograde flow of contrast material into the renal lymphatics and calyces as seen in our case, although there was some difference in the degree of visualization. In the second case reported the Swanson 7 there was so much contrast material traveling to the kidney that the calyces were completely outlined. In chyluria due to filariasis principal lesions are found in the lymphatics of the pelvic, lumbar and retroperitoneal regions. In some instances, however, as shown in several autopsy cases, the whole lymphatic system including the thoracic duct is extensively involved. Histologically, lymphatic and Lessmann, F. P.: The use of lymphography as a diagnostic method. Radiology, 76: 47-53, 1961. 11 Wallace, S., Jackson, L., Schaffer, B., Gould, J., Greening, R.R., Weiss, A. and Kramer, S.: Lymphangiograms: Their diagnostic and therapeutic potential. Radiology, 76: 179-199, 1961. 12 Fuchs, W. A. and Bo6k-Hederstr6m, G.: Inguinal and pelvic lymphography. A preliminary report. Acta radiol., 56: 340-354, 1961. 13 Ruttimann, V. A., Del Buono, JVI. S. and Cocchi, U.: Neue Fortschritte in der Lymphographie. Schweiz.Med. Wschr., 91: 1460-1466, 1961. 14 Viamonte, M., Jr., 11:yers, M. B., Soto, 11:., Kenyon, N. JVI. and Parks, R. E.: Lymphography: Its role in detection and therapeutic evaluation of carcinoma and neoplastic conditions of the genitourinary tract. J. Urol., 87: 85-90, 1962. 15 Jacobsson, S. and Johansson, S.: Lymphangiography in lymphedema. Acta radiol., 57: 81-89,
577
vessels lose elastic tissue with dilated lumen, thickened wall and incompetent valves, and reactive as well as cicatrical changes take place in lymph nodes, leading to obstruction of the afferent lymphatics. Lymphatic varicosities and poor or loss of visualization of lymph nodes on lymphogram correspond to those histological alterations. The degree of visualization may be quite variable, presumably reflecting the extent of an underlying pathological process. Behaving like varicose veins, lymphatic varicosities probably rupture into the urinary tract owing to the in~ creased hydrostatic pressure caused by lymph stasis because of incompetent valves, and become demonstrable on lymphography. In our case, the contrast material entered freely the contralateral abdominal lymphatics with cross-over on injection exclusively in the right foot. This was interpreted mainly as indicating abnormal dilation of the lymphatics in the sacral region communicating both sides. On the right side, where retrograde flow of contrast material into the renal lymphatics was \Nell visualized, few lymph nodes were demonstrable. On the contrary, there were numerous abnormal abdominal lymph nodes on the left side. These findings suggest more marked involvement
I
1962. 16 Fischer, H. W., Lawrence, M. S. and Thornbury, J. R.: Lymphography of the normal adult male: Observations and their relation to the diagnosis of metastatic neoplasm. Radiology, 78: 399-
406, 1962. 17 Wallace, S., Jackson, L. and Greening, R.R.: Clinical applications of lymphangiography. Amer. J. Roentgenol., 88: 97-109, 1962. 18 Viamonte, JVI., Jr., Altman, D., Parks, R., Blum, E., Bevilacqua, :i\I. and Recher, L.: Radiographic-pathologic correlation in the interpretation of lymphangioadenograms. Radiology, 80:
903-916, 1963.
19 Schaffer, B., Koehler, P. R., Daniel, C. R., Wohl, G. T., Rivera, E., ]\!foyers, W. A. and Skelley, J. F.: A critical evaluation of lymphangiography. Radiology, 80: 917-930, 1963. 20 Turiaf, J., Arvay, N., Picard, J. D., Servelle, M. and Gentilini, M.: Donnees de la lymphographie dans deux cas de chylurie filarienne. Bull. Soc. Med. d. hop. de Paris, 113: 753-766, 1962. Cited by Swanson, G. E. 7
Frn. 3. Immediate lymphangiogram of additional case.
I
578
KISHIMOTO, HIGUCHI, ENDO AND KAI
in the right abdominal lymphatics than in the left, although possibly chyluria might recur from the left side since considerable change is seen there. The measures to control chyluria include a low fat diet, bed rest, medication, instillation or irrigation of the renal pelvis with a sclerosing agent, and surgery. One must r(:1sort to surgery only after failure of other therapeutic means. As an operative procedure nephrectomy should be avoided unless inevitable. Conventional capsulectomy alone usually proves to be unsatisfactory as stated in previous reports. The method of choice appears to be removal of all lymphatics around the renal vascular pedicle. If this is done completely, concomitant capsulectomy is not necessary. Lately Torres and Estrada reported on this surgical approach along with two personal cases. 2 Another procedure, lymphaticovenous anastomosis, has been attempted. 4 Besides the case just reported as being cured of obstinate chyluria after removal of lymphatics around the renal vascular pedicle, the authors treated one more patient in the same manner without recurrence in 2 years postoperatively. In view of those roentgenograms, findings at operation and favorable outcome, this procedure is worthy to be recommended for cases in which other therapeutic measures are ineffective. According to the Japanese statistics cited earlier, 8 there were 70 patients who underwent surgery during the 5 years (1957-1961). Chyluria disappeared in 53 (75.7 per cent), improved in 14 (20.0 per cent), a11diwas unchanged in 3 (4.3 per
cent). The long-term recovery rate was 71.9 per cent, while recurrence on the operated side developed in 28.1 per cent. Better results may be obtained by more meticulous removal of the lymphatics around the renal vascular pedicle. SUMMARY
The case of a patient with unilateral hematochyluria has been described. Abnormal communication between the para-aortic and the renal lymphatics had been demonstrated on lymphography (lymphangioadenography). Retrograde fl.ow of some contrast material from the renallymphatics into the renal calyces had also been noted. Following an operation that completely removed numerous dilated lymphatics around the renal vascular pedicle shown on lymphography, chyluria disappeared immediately and has not recurred for the past 6 months. ADDENDUM
After this paper was completed, the authors had an opportunity to perform lymphography on one more patient with chyluria. The case was in a 60-year-old male Japanese who had left renal chyluria due to filariasis. The i=ediate lymphogram following lymphatic vessel injection of this patient is shown. Ten milliliters of co.ntrast material were injected in each foot, demonstrating. a considerable degree of retrograde fl.ow of the contrast material into the left renal lymphatics.