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Saudi Journal of Kidney Diseases and Transplantation
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CASE REPORT  
Year : 2020  |  Volume : 31  |  Issue : 6  |  Page : 1407-1410
Chyluria with massive proteinuria: Do not reach for the biopsy gun!


1 Department of Urology, Christian Medical College, Vellore, Tamil Nadu, India
2 Department of Nephrology, Christian Medical College, Vellore, Tamil Nadu, India

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Date of Web Publication29-Jan-2021
 

   Abstract 


We report on a patient presenting with persistent chyluria due to filariasis, whose clinical course was complicated by massive proteinuria and severe hypoalbuminemia. Treatment with dietary manipulation, antifilarials, and sclerotherapy resulted in successful reversal of the above abnormalities. It has been reported that chyluria is not associated with massive proteinuria, or that even in cases of massive proteinuria, hypoalbuminemia is not seen and implies a glomerular pathology. We argue that chyluria is always associated with proteinuria, which may be massive, and does not warrant a kidney biopsy unless proteinuria persists despite resolution of chyluria.

How to cite this article:
Sudrania MK, Valson AT, Dangi AD, Kekre NS. Chyluria with massive proteinuria: Do not reach for the biopsy gun!. Saudi J Kidney Dis Transpl 2020;31:1407-10

How to cite this URL:
Sudrania MK, Valson AT, Dangi AD, Kekre NS. Chyluria with massive proteinuria: Do not reach for the biopsy gun!. Saudi J Kidney Dis Transpl [serial online] 2020 [cited 2021 May 15];31:1407-10. Available from: https://www.sjkdt.org/text.asp?2020/31/6/1407/308357



   Introduction Top


Based on findings in patients with non-parasitic (congenital) chyluria, it has been previously reported that proteinuria in chyluria is not massive, does not result in hypo-albuminemia, and if present, suggests a coexistent glomerular pathology such as mesangioproliferative glomerulonephritis, membranous nephropathy, or membranoprolife-rative glomerulonephritis.[1],[2],[3],[4] However, as this case illustrates, the presence of large amounts of protein in the urine in a patient with chyluria need not imply glomerular pathology; treatment of chyluria may itself result in “remission”of the proteinuria, thus obviating the need for a kidney biopsy.


   Case Report Top


Informed consent was obtained from the patient for the publication of this case report.

A 63-year-old man from West Bengal with no previous comorbidities presented to his local physician with a six-month history of anorexia, weight loss, and passage of white-colored urine along with gross painless hema-turia with clots. He failed to respond to dietary manipulation and was prescribed diethylcarba-mazine (DEC) for presumed filarial infection, which he discontinued due to severe nausea and vomiting. He presented to our center with persistent passage of white-colored urine and gross hematuria and, on examination, was found to be malnourished [body mass index (BMI): 16.86 kg/m2] and had bilateral pitting pedal edema. Blood pressure was 100/60 mm Hg with a pulse rate of 96/min. Systemic examination was unremarkable.

Laboratory investigations are shown in [Table 1]. Noncontrast computed tomography showed retroperitoneal lymphangiectasia without retroperitoneal lymphadenopathy [Figure 1]. Cystoscopy following a challenge with fatty meal localized hematochyluria to the right side.
Figure 1: Multiple tiny serpiginous lymphatic channels in the retroperitoneum on computed tomography.

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Table 1: Laboratory investigations at presentation.

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Due to salt-wasting state, he was given 3% hypertonic saline. With improvement in volume status, serum creatinine declined to 1.2 mg/dL, and after correction of coagulopathy with parenteral Vitamin K, he underwent right retrograde pyelography and sclerotherapy with 0.5% w/w (in distilled water) Betadine. Cystourethroscopy revealed two ureteric orifices on the right side. Pyelolymphatic channels could be demonstrated on injecting radiocontrast into both renal moieties [Figure 2]. Both ureteric orifices on the right side were cannulated with 4 Fr ureteric catheters, and Betadine instillation was carried out nine times in all over three days. His urine became grossly clear. Due to documented intolerance to DEC, he was given a single dose of oral ivermectin 400 μg/kg followed by doxycycline 100 mg twice daily for 6 weeks and was discharged on restricted fat diet with medium-chain triglycerides.
Figure 2: Pyelolymphatic channels demonstrated in both right renal moieties on retrograde pyelography.

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At follow-up six months later, urine protein creatinine ratio was 0.09, serum albumin was 4.4 g/dL, no chylomicrons were detected in urine, and he had gained 6 kg weight (BMI 20.6).


   Discussion Top


Chyle has an alkaline pH with electrolyte composition similar to plasma. It has 5–30 g/L of fat, 20–30 g/L of protein, caloric composition of 200 kcal/L, and fat-soluble vitamins.[5] Persistent chyluria results in prolonged loss of immunoglobulins, lymphocytes, and complement fractions, which results in lymphopenia and impaired cell-mediated immunity,[6],[7] while the loss of large amounts of protein, fat, calories, electrolytes,and vitamins in urine results in severe nutritional and electrolyte disturbances.[6]

Chyluria is, therefore, always associated with proteinuria. Because filariasis is associated with various glomerular pathologies,[1],[2],[3] and various authors have also described the presence of glomerular lesions coexisting with filarial chyluria,[4] it is important to rule out concomitant glomerular involvement in patients who present with chyluria and significant proteinuria. Features that distinguish the proteinuria in chyluria from that due to a glomerular disease are the presence of leukocyturia (lympho-cyturia) and isomorphic hematuria and the absence of dysmorphic red blood cells (RBCs), RBC casts, fatty droplets, or fatty casts on urinalysis.[8] In a patient who presents with chyluria and proteinuria, these urinary findings, in combination with the absence of hypercholesterolemia and hypertriglyceridemia, suggest that proteinuria can be entirely attributed to chyluria and a renal biopsy need not be performed.[8],[9] In fact, in patients with these findings, renal biopsy often demonstrates minimal change disease, and patients show no response to immunosuppressive therapy, pro-teinuria only disappearing with antifilarial therapy, sclerotherapy, or open surgical interv-ention.[10]

Our patient had chylomicrons in urine, retroperitoneal lymphangiectasia on cross-sectional imaging, and milky urine efflux was demonstrated through the right ureteric orifices, thus confirming the diagnosis of chyluria. He belonged to an area endemic for filariasis, and responded to second-line antifilarials, thus suggesting that the primary etiology was filariasis.The unique feature, in this case, was the degree of proteinuria seen and the coexistent hypoalbuminemia. Massive proteinuria >10 g/day has been described in chyluria even without glomerular involvement, however, the authors reported that even this degree of proteinuria did not result in hypoalbuminemia.[10] It is likely that because only 30% of patients in the series had continuous chyluria (the duration of chyluria was not reported), hypoalbuminemia did not occur. Our patient had persistent chyluria for over six months and manifested various nutritional deficiencies apart from hypoalbuminemia, namely low BMI, coagulopathy (due to loss of fat-soluble vitamins), lymphopenia, and hypo-natremia. He had isomorphic hematuria and leukocyturia, no dysmorphic RBCs or fatty casts, normal serum cholesterol, and trigly-cerides, all of which suggested that proteinuria was due to chyluria. Surgical intervention, combined with antifilarial and nutritional therapy, led to significant improvement in all metabolic parameters at follow-up.

To conclude, chyluria is always associated with proteinuria, and this proteinuria may be massive. A renal biopsy is not indicated unless proteinuria persists despite resolution of chyluria.

Conflict of interest: None declared.



 
   References Top

1.
Chugh KS, Singhal PC, Tewari SC, Nath IV, Datta BN. Acute glomerulonephritis associated with Filariasis. Am J Trop Med Hyg 1978;27: 630-1.  Back to cited text no. 1
    
2.
Pillay VK, Kirch E, Kurtzman NA. Glomerulopathy associated with filarial loiasis. JAMA 1973;225:179.  Back to cited text no. 2
    
3.
Yap HK, Woo KT, Yeo PP, et al. The nephrotic syndrome associated with filariasis. Ann Acad Med Singap 1982;11:60-3.  Back to cited text no. 3
    
4.
Lai KN, Siu D, Chan KW, Lai FM, Yan KW. The clinical significance of proteinuria in patients with nonparasitic chyluria. Am J Kidney Dis 1986;7:381-5.  Back to cited text no. 4
    
5.
Merrigan BA, Winter DC, O’Sullivan GC. Chylothorax. Br J Surg 1997;84:15-20.  Back to cited text no. 5
    
6.
Date A, John TJ, Chandy KG, et al. Abnormalities of the immune system in patients with chyluria. Br J Urol 1981;53:384-6.  Back to cited text no. 6
    
7.
Date A, Shastry JC, Vaska PH, et al. Complement in chyluria. Trans R Soc Trop Med Hyg 1982;76:238-9.  Back to cited text no. 7
    
8.
Graziani G, Cucchiari D, Verdesca S, et al. Chyluria associated with nephrotic-range proteinuria: Pathophysiology, clinical picture and therapeutic options. Nephron Clin Pract 2011;119:c248-53.  Back to cited text no. 8
    
9.
Cheng JT, Mohan S, Nasr SH, D’Agati VD. Chyluria presenting as milky urine and nephrotic-range proteinuria. Kidney Int 2006;70:1518-22.  Back to cited text no. 9
    
10.
Kaul A, Bhadhuria D, Bhat S, et al. Chyluria: A mimicker of nephrotic syndrome. Ann Saudi Med 2012;32:593-5.  Back to cited text no. 10
    

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Correspondence Address:
Anna T Valson
Department of Nephrology, Christian Medical College, Vellore, Tamil Nadu - 632004
India
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DOI: 10.4103/1319-2442.308357

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