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CASE REPORT
Year : 2020  |  Volume : 13  |  Issue : 1  |  Page : 86-88  

Imaging in a case of chyluria presenting as hematuria


1 Department of Radiology, Command Hospital, Kolkata, West Bengal, India
2 Department of Urology, Command Hospital, Kolkata, West Bengal, India

Date of Submission10-Mar-2019
Date of Decision29-May-2019
Date of Acceptance03-Sep-2019
Date of Web Publication16-Dec-2019

Correspondence Address:
Vinay Maurya
Department of Radiology, Command Hospital, Kolkata, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/mjdrdypu.mjdrdypu_78_19

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  Abstract 


Chyluria is a debilitating disease characterized by passage of milky white urine. The cases of chyluria usually pose no problem in diagnosis as the characteristic milky white urine on naked examination is virtually diagnostic. However, if hematuria is a predominant finding it may mask the chyluria and lead to delayed diagnosis. In such cases imaging may play a vital role in diagnosing these cases. MRI findings of dilated retroperitoneal lymphatics are characteristic and help in early diagnosis and management of these cases.

Keywords: Chyluria, hematochyluria, hematuria, magnetic resonance imaging


How to cite this article:
Maurya V, Gopinath M, Sarkar K, Aggarwal P. Imaging in a case of chyluria presenting as hematuria. Med J DY Patil Vidyapeeth 2020;13:86-8

How to cite this URL:
Maurya V, Gopinath M, Sarkar K, Aggarwal P. Imaging in a case of chyluria presenting as hematuria. Med J DY Patil Vidyapeeth [serial online] 2020 [cited 2020 Jan 28];13:86-8. Available from: http://www.mjdrdypv.org/text.asp?2020/13/1/86/272888




  Introduction Top


Chyluria is the passage of milky white urine due to abnormal communication developing between the retroperitoneal lymphatics and the urinary collecting system.[1] It more commonly occurs in filarial endemic areas such as Southeast Asia, India, and Northern Africa and is infrequent in the Western world.[2] The cases of chyluria usually pose no problem in diagnosis as the characteristic milky white urine on naked examination is virtually pathognomonic. However, if hematuria is a predominant finding, it may mask the chyluria and lead to delayed diagnosis. In such cases, imaging may play a vital role in diagnosing these cases. Here, we report a case of hematochyluria in a young female.


  Case Report Top


A 22-year-old female patient with no significant previous history of relevant illness presented with painless, gross, and total hematuria for 2 days with intermittent passage of blood clots in the urine. There was no history of ureteric colic, fever, urgency, or frequency of micturition. On clinical examination, pallor was present. On palpation, the abdomen was soft with no palpable lump. Routine hematological investigations revealed Hb, 10.2 g/dL; total leukocyte count, 10,800/mm 3; and erythrocyte sedimentation rate, 30 mm/h. Urine was red on naked eye examination and showed numerous red blood cells on microscopy and urine culture was sterile. Radiograph kidney, ureter, and bladder was normal. On ultrasound, the kidney was normal and there was no ureteric dilatation. The bladder showed clots with no intravesical growth. On cystoscopy, there was a large bladder clot which was removed; after clot evacuation, hemorrhagic urine was seen coming from the left ureteric orifice, suggesting left upper urinary tract bleed necessitating further investigations [Figure 1]. Contrast-enhanced computed tomography (CECT) abdomen showed normal appearance of both kidneys and ureter. However, subtle sleeve of hypodensity was noted along the left para-aortic region and the left renal pelvis and along the iliac vessels and ureter on the left side [Figure 2]a-d]. Based on the above findings, the possibility of lymphatic obstruction with dilated retroperitoneal lymphatics was raised. Repeat urine examination for chylomicrons was done in light of CT findings, which showed urine triglyceride level of 7.8 μg/dL. Magnetic resonance imaging (MRI) was done, which showed T2 and short inversion time inversion recovery (STIR) hyperintensities in the retroperitoneum encasing and insinuating between the great vessels, along the renal pelvis and ureters and extending into the pelvic cavity bilaterally with changes being more marked on the left side [Figure 3]a, [Figure 3]b, [Figure 3]c, [Figure 3]d. MRI confirmed the diagnosis of obstructed and dilated retroperitoneal lymphatics with possible fistulous communication with urinary collecting system. The patient was diagnosed as a case of hematochyluria due to dilated retroperitoneal lymphatics with fistulous communication and managed initially with intravenous (IV) antibiotics and IV fluids. In the cystoscopic suite, a ureteric catheter was placed and renal pelvic instillation sclerotherapy was carried out by instilling 0.2% povidone iodine into the left ureter and pelvicalyceal system every 8 hourly for the next 3 days to induce sclerosis in the fistulous communication. Her hematuria stopped after 3 days and she became asymptomatic. She was worked up for filariasis which came out to be negative. However, the patient was given antifilarial treatment with diethylcarbamazine for 21 days empirically and advised low-fat, high-protein diet and advised regular follow-up. She has been asymptomatic for the last 4 months without any fresh episode of hematochyluria.
Figure 1: Cystoscopy showing hemorrhagic urine coming from the left ureteric orifice

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Figure 2: (a-d) Axial contrast-enhanced computed tomography – sleeve of hypodensity (arrow) is seen surrounding the left renal hilum (a). Hypodensity (arrow) in the left para-aortic region (b). Hypodense sleeve insinuating between the great vessels and encasing the inferior vena cava and common iliac artery bifurcation (c) (arrows). Delayed computed tomography image showing hypodensity (arrow) on medial aspect of contrast-filled ureter (d)

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Figure 3: (a-d) Axial magnetic resonance imaging showing short inversion time inversion recovery hyperintensities (arrow) encasing the left renal hilum (a). Fluid intensity insinuating and encasing retroperitoneal great vessels (arrows) (b). Fluid intensity along the bilateral internal iliac vessels (arrows) more marked on the left side (c). Coronal magnetic resonance image showing short inversion time inversion recovery hyperintensities in the retroperitoneum (arrow), note the blood clot in the catheterized urinary bladder (d)

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  Discussion Top


Chyluria occurs when dilated retroperitoneal lymphatics rupture into the renal tubules or collecting system. It has been graded into three grades: in Grade I, the urine is milky white; in Grade II, whitish clots or clot retention occurs; and when associated with hematuria, it is labeled as hematochyluria and termed as Grade III as was seen in our case.

The etiopathogenesis of chyluria is spontaneous rupture of dilated retroperitoneal lymphatics into pyelocalyceal system or ureter.[1]

Causes of chyluria can be parasitic or nonparasitic in nature. In India, various parasitic agents such as Wuchereria bancrofti, Echinococcus granulosus, and ascariasis are mainly responsible for this, out of which filarial worm is the leading cause and chyluria should be considered filarial unless proven otherwise particularly in the filarial belt.[2] The nonparasitic causes can be retroperitoneal lymphatic malformation, obstruction to lymphatics due to malignancy, trauma, tuberculosis, and pregnancy.[3],[4]

The clinical presentation is typically milky white urine, but sometimes when associated with hematuria, it can present as hematochyluria. If the hemorrhage element is more dominant and the patient passes clots, it may be mistakenly labeled as a case of hematuria as happened in this case. In our case, the patient's predominant complaint was hematuria, and hematochyluria was only suspected after CECT abdomen.

Diagnosis of chyluria is clinical, and typical milky white appearance of urine is virtually diagnostic. Urine examination of triglycerides has 100% specificity in diagnosing chyluria. The role of imaging is in identifying cases where the clinical picture is not clear and hematuria is the predominant symptom as happened in our case. The ultrasound has limited role in identifying the dilated lymphatics. CECT abdomen can demonstrate fluid density in the retroperitoneum, which encases and insinuates between the great vessels and renal hilum and can extend along the ureters. These findings were elicited in our case, which lead to suspicion of hematochyluria [Figure 2]d. Another finding which has been described in literature but was not seen in our case is demonstration of fat–fluid level in the urinary bladder, which is typically identified as fat density (−20 to −30 HU) floating in the urine in nondependent part in axial sections.[5] MRI is the modality of choice due to its ability to demonstrate the fluid intensity and the true extent better than CT. MRI demonstrates the dilated tubular fluid-filled lymphatic channels in the retroperitoneum as T2/STIR hyperintensities which encase the great vessels and renal hilum or ureter as was seen in this case [Figure 3]a, [Figure 3]b, [Figure 3]c, [Figure 3]d. MRI can also help in differentiating the primary tropical cause from retroperitoneal lymphatic malformation which can also present as chyluria.[6] Therefore, MRI should be done in cases of chyluria or hematochyluria to map out the dilated retroperitoneal lymphatics and to rule out lymphatic malformation as cause.

The management of chyluria can be nonsurgical or surgical. Nonsurgical management includes dietary modifications and treatment with diethylcarbamazine for 3 weeks. Surgical management has two components: first to provide immediate relief and second to provide definitive cure to prevent recurrence. Instillation of sclerosant into the urinary tract induces chemical lymphangitis and edema, which blocks the communication and provides immediate relief. Once it heals, it produces fibrosis which provides lasting relief, but chance of recurrence remains. The sclerosants used are 1% AgNO3 and 0.2% povidone iodine.[7],[8] Suri and Kumar in their large series of 600 cases showed that 0.2% povidone iodine was better than 1% AgNO3 as it had equal efficacy, less complications, and was easy to prepare.[2] The permanent cure involves surgical lymphatics disconnection. The older techniques such as Katamine procedure, Patna operation, and Cockett and Goodwin procedure have now been replaced by laparoscopic and retroperitoneoscopy in management of these cases.


  Conclusion Top


Chyluria is easily diagnosed by typical milky appearance of urine, but hematochyluria may be misinterpreted as hematuria and the diagnosis may be delayed. It is in these cases the imaging can play a role in early diagnosis. MRI is the diagnostic modality of choice as it demonstrates the dilated and tortuous lymphatics in the retroperitoneum. Therefore, it is recommended that these patients undergo MRI for the evaluation of extent of disease and to rule out other causes such as lymphatic malformation which can also present as hematochyluria.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Singh I, Dargan P, Sharma N. Chyluria – A clinical and diagnostic stepladder algorithm with review of literature. Indian J Urol 2004;20:79-85.  Back to cited text no. 1
  [Full text]  
2.
Suri A, Kumar A. Chyluria – SGPGI experience. Indian J Urol 2005;21:59-62.  Back to cited text no. 2
  [Full text]  
3.
Esquirol Mallol R, Servelle M, Turpyn L, Verminck JP, Esquirol Caussa JR. Lymphatic malformation and chyluria: Presentation of a treated case without relapse. Med Clin (Barc) 1991;97:576-8.  Back to cited text no. 3
    
4.
Núñez Mora C, Cárcamo Valor P, de Cabo Ripoll M, Kabani MH, Martínez-Piñeiro Caramés JA. Recurrent nonparasitic chyluria. Arch Esp Urol 1998;51:932-4.  Back to cited text no. 4
    
5.
Miller FH, Keppke AL, Yaghmai V, Gabriel H, Hoff F, Chowdhry A, et al. CT diagnosis of chyluria after partial nephrectomy. AJR Am J Roentgenol 2007;188:W25-8.  Back to cited text no. 5
    
6.
Govil S, Justus A, Lakshminarayanan R, Nayak S, Devasia A, Gopalakrishnan G. Retroperitoneal lymphatics on CT and MR. Abdom Imaging 2007;32:53-5.  Back to cited text no. 6
    
7.
Shanmugam TV, Prakash JV, Sivashankar G. Povidone iodine used as a sclerosing agent in the treatment of chyluria. Br J Urol 1998;82:587.  Back to cited text no. 7
    
8.
Seleem MM, Eliwa AM, Elsayed ER, Desouky H, El Galaly H, Abdelwahab K, et al. Single versus multiple instillation of povidone iodine and urographin in the treatment of chyluria: A prospective randomised study. Arab J Urol 2016;14:131-5.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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