| Abstract|| |
A 21- year-old lady presented with chyluria, severe malnutrition, secondary amenorrhea, profound hypoalbuminemia, heavy proteinuria and renal tubular abnormalities suggestive of Type IV renal tubular acidosis. No particular cause for chyluria could be ascertained. She was successfully treated with an injection of 2% silver nitrate into the left ureter and urinary bladder. She continues to be well after 15 years of follow-up.
Keywords: Chyluria, IgA deposition, Type IV RTA
|How to cite this article:|
Abraham G, Pratap B, Govindan P, Matthew M. Successful Treatment of Chyluria, Glomerular and Tubular Abnormalities in a Young Lady with Silver Nitrate Instillation. Saudi J Kidney Dis Transpl 2007;18:231-4
|How to cite this URL:|
Abraham G, Pratap B, Govindan P, Matthew M. Successful Treatment of Chyluria, Glomerular and Tubular Abnormalities in a Young Lady with Silver Nitrate Instillation. Saudi J Kidney Dis Transpl [serial online] 2007 [cited 2016 May 3];18:231-4. Available from: http://www.sjkdt.org/text.asp?2007/18/2/231/32315
| Introduction|| |
Chyluria is characterized by lymphuria and is commonly caused by Bancroftian filariasis in South India. It is usually caused by lymph drainage obstruction into the thoracic duct resulting in dilatation and rupture of the lymph varix into the urinary collecting system with retrograde lymph flow. This acute nephritic illness, as well as the more common nephritic syndrome, has been described with filarial nematodes, which on light microscopy show membranous, mesangial or diffuse proliferative glomerulonephritis.
Here, we describe a case of chyluria likely due to filariasis with IgA deposition in the glomerulus, hyperkalemia, Type IV renal tubular acidosis (RTA) and secondary amenorrhea, which reversed after successful treatment of chyluria with a silver nitrate injection.
| Case Report|| |
A 21-year-old female presented in July 1991 with complaints of passing milky urine for 11 months, pedal edema and 11 kg weight loss over a six month period, secondary amenorrhea for three months, exertional dyspnea for two weeks, fatigue, nausea and loss of appetite. Because she was from a Bancroftian filariasis endemic area, she was empirically treated with diethyl carbamazine citrate, 100 mg twice daily for three months elsewhere. On admission, she weighed 43 kg and her blood pressure (BP) was 80/50 mm Hg supine, pulse rate was 134/min and regular and she had anasarca.
Relevant laboratory investigations after admission showed a total serum protein of 3.47 gm/dl, albumin 1.9 gm/dl, blood urea nitrogen (BUN) 14 mg/dl, serum creatinine (Se Cr) 1.6 mg/dl, sodium (Na + ) 118 mmol/L, potassium (K + ) 4.75 mmol/L, chloride (Cl - ) 101 mmol/L, bicarbonate (HCO3 - ) 16.1 mmol/L, calcium (Ca ++ ) 9.6 mg/dl, inorganic phosphorous 5.8 mg/dl and alkaline phosphatase of 144 IU/L. Serum electrophoresis showed elevation of α-2 and β globulin with decrements in total protein, albumin and gamma globulin. Urinalysis showed pH of 5.0, specific gravity 1.015, color-milky white, albumin ++++, occult blood ++, red blood cells (RBC) 5-6/hpf, white blood cells (WBC) 3-4/hpf, and no casts or crystals.
The 24-hour urine protein excretion was 20 gm in a total volume of 2570 ml. Ultrasound showed right kidney measuring 8.5 x 3 cm, left kidney 8.3 x 3 cm, good cortico-medullary differentiation and no obstruction. Addison's disease was ruled out with appropriate investigations. As her electrolyte abnormalities were compatible with Type IV RTA, the patient was initiated on oral fludrocortisone 200 mcg/day. An ultrasound guided renal biopsy was performed two weeks later which contained 13 glomeruli. On light microscopy, the glomeruli showed focal and segmental increase in mesangial cellularity [Figure - 1]. The interstitium, tubules and blood vessels were normal. Immunofluorescence revealed mesangial IgA deposits ++++ and IgM + [Figure - 2].
As the milky urine persisted, a lymphadenogram was done through the right superficial inguinal node approach. It showed a lymphatic leak into the bladder and the collecting system of the right kidney [Figure - 3]. Subsequent pictures showed a communication with the renal collecting system on the left side. Imaging studies for extraneous obstruction at the level of the cisterna chyli and thoracic duct were negative. As the patient experienced lymphatic leak and persistent heavy chyluria, sclerosing therapy was performed using cystoscopy and bladder wash with three ml of 2% silver nitrate mixed in 100 ml of distilled water. A retrograde catheter was passed up to 15 cm in the left ureter and two ml of silver nitrate solution was injected into the renal pelvis. The remaining silver nitrate was left in the bladder for four hours before the patient voided again. Two days later, the milky urine disappeared.
She developed mild renal failure as shown by a BUN of 31 mg/dl, Se Cr 3.3mg/dl, total protein 5.1 gm/dl, albumin 3 g/dl, Na + 132 mmol/L, K + 3.9 mmol/L, Cl - 103 mmol/L, and HCO3 - 22.6 mmol/L. Her urine output was 1 ½ liters/day and clear. Ultrasound examination of the kidneys and the collecting systems and bladder was unremarkable. Four weeks later, laboratory investigations showed BUN 9 mg/dl, Se Cr 1.5 mg/dl, with normal electrolyte levels. Six weeks later, 24 hours urine protein was 158 mg in 1.6 liters. The patient's edema disappeared and she started to menstruate. In February 1993, urine examination showed no proteinuria and there was no abnormal sediment. During her first pregnancy in 1993, she had mild hypertension in the first trimester, which was treated with alpha-methyl dopa and was delivered by cesarean section full-term. She conceived again in 1995 and had a normal delivery. Current investigations showed BUN 9 mg/dl, Se Cr 1.1 mg/dl, uric acid 6.7 mg/dl, total protein 6.2 gm/dl, albumin 3.8 gm/dl and urine analysis did not show abnormal sediment.
| Discussion|| |
Chyluria can be persistent or intermittent and can present with turbid, milky or creamy urine due to the presence of thoracic or intestinal lymph in the urine. As the thoracic duct contains a higher concentration of IgA than other immunoglobulin fractions, we postulate that the IgA deposits in the mesangium were a result of the delivery of this immunoglobulin to the glomeruli along the lymphatics as a result of lymphatic obstruction. A turbid fluid due to leukocytes or tumor cells may be confused with chyluria. Alkalinization and ether extraction will lead to clearing of the urine in chyluria. However, in our patient, the cloudiness of the urine persisted even after alkalinization and mixing with ether. The observed hypogammaglobulinemia and hypoalbuminemia is due to the massive loss of chyle in the urine. Chyle contains 4-6 gms of protein/dl and a total of 100 ml of chyle is formed in the thoracic duct per hour.
Glomerular mesangial IgA deposits have been described in association with a number of systemic illnesses and in otherwise healthy normal subjects. Previously, in Bancroftian filariasis, mesangial or diffuse proliferative glomerulonephritis have been described. Immunofluorescence in these disorders showed deposits of IgM, IgG and C3 in the mesangium and along the capillary wall., To our knowledge, this is the first time the association of IgA deposition in the mesangium with chyluria has been reported. However, a biopsy was not repeated after the patient recovered.
Successful therapy of chyluria has produced long-term benefit; correction of metabolic acidosis, secondary amenorrhea, disappearrance of proteinuria and improvement in the globulin fraction of the plasma protein. Silver nitrate injection is a successful treatment modality executed by urologists in India for recurrent and refractory chyluria. One study found that the three day eight-hour instillation was a more economical and less cumbersome regimen as compared to the weekly instillation for six weeks. We must mention that silver nitrate injection, which is a heavy metal, can produce reversible renal failure, as published in a previous case report. Silver has been shown in experimental models to produce interstitial edema and tubular degeneration, leading to acute tubular necrosis and acute renal failure in film developers. A previous study showed anuria and pelvi-calyceal casts following silver nitrate injection, which was observed in imaging studies. Povidone iodine 0.2% is also considered to be as effective as 1% silver nitrate therapy. The cumulative success rate after two courses of therapy was 82% (silver nitrate) and 83% (povidone).
As our patient's serum potassium was consistently high, a diagnosis of Type IV RTA was considered however, she was resistant to mineralocorticoid therapy. We believe that the Type IV RTA was due to the direct toxic effect of chyle on the distal tubular cells and the hyponatremia could be due to the nonosmotic ADH secretion as a result of hypovolemia as well as the urinary loss of sodium ions. Chronic hypoalbuminemia, malnutrition and fat loss could have been the cause of her amenorrhea, which also spontaneously corrected after her nutritional status improved following recovery from chyluria.
In conclusion, we present a patient with chyluria, glomerular IgA deposition, tubular abnormalities suggestive of Type IV RTA, malnutrition and secondary amenorrhea, who was successfully treated with silver nitrate injection. She has been followed for over 15 years with no evidence of chronic kidney disease
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Department of Medicine, Sri Ramachandra Medical College and Research Institute, Chennai - 600 116,Tamil Nadu
[Figure - 1], [Figure - 2], [Figure - 3]