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Repeated Sclerotherapy Successfully Treats a Child with Idiopathic Chyluria: a Case Report

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Repeated Sclerotherapy Successfully Treats a Child with Idiopathic Chyluria: a Case Report SOUTHEAST ASIAN J TROP MED PUBLIC HEALTH REPEATED SCLEROTHERAPY SUCCESSFULLY TREATS A CHILD WITH IDIOPATHIC CHYLURIA: A CASE REPORT Narumon Densupsoontorn1, Anirut Pattaragarn1 and Phichaya Sujijantararat2 1Department of Pediatrics, 2Department of Surgery, Faculty of Medicine Siriraj Hospital, Mahidol University, Bangkok, Thailand Abstract. We describe a clinical course of idiopathic chyluria in a previously healthy 8-year-old Thai boy, with a 1-year history of cloudy white urine under- going antibiotic treatment. The patient was investigated for the causes but they all proved negative. Cystoscopy and retrograde pyelography demonstrated a renolymphatic fistula of the right kidney. The patient was refractory to a medium- chain triglyceride-rich diet. Ultimately, sclerotherapy with 1% povidone iodine was employed twice, which resulted in a resolution of the disorder. We emphasize that chyluria, even though a very rare condition of chylous disorders, but ultimately can be initially managed by providing a medium-chain triglyceride-rich diet and is curable by sclerotharapy. Importantly, medium-chain triglyceride-rich diet can be applied to treat other types of chylous disorders. Keywords: child, chyluria, medium-chain triglyceride-rich diet, sclerotherapy INTRODUCTION reported cases (Stalens et al, 1992; Ganesh et al, 2007; McNeil et al, 2008). Chyluria is a rare condition of chy- lous leakage from lymphatic vessels The etiology of chyluria is categorized into urinary system through abnormal into two groups, which are (i) parasitic communicating tracts. Chyuria is found cause, viz, lymphatic filariasis, and (ii) non- in 10% and 34% of patients suffering parasitic cause, viz, congenital anomalies from filariasis in India and China, re- of lymphatic system, trauma, lymphatic spectively (Cui et al, 1990; Tandon et al, obstruction after surgery, tumor, tuberculo- 2004). Its incidence from radiofrequency sis, thoracic aneurysm, mesenteric adenitis, ablation for treatment of renal tumors and granulomatous disease (McNeil et al, is 41% (Kaur et al, 2011). To the best of 2008). Patients with chyluria might present our knowledge, no incidence of chyluria with milky urine, chylous clot, hematochy- in the general population and pediatric luria, dysuria, frequent urination, weight group has been published except for three loss, urinary retention, low back pain, renal colic, edema, or protein-energy malnutri- Correspondence: Narumon Densupsoontorn, tion (Nandy et al, 2004). Department of Pediatrics, Faculty of Medicine Siriraj Hospital, Mahidol University, 2 Wang To date, in the Thai population only an Lang Road, Bangkok Noi, Bangkok 10700, elderly female with nephrotic syndrome Thailand. was reported to have chylous ascites and Tel: +66 (0) 2419 7000 ext 95947; Fax: +66 (0) chyluria, but evidences of lymphatic ob- 2411 3010 struction and lympho-renal fistula could E-mail: [email protected] not be substantiated (Lewsuwan et al, 360 Vol 48 No. 2 March 2017 SCLEROTHERAPY IN IDIOPATHIC CHYLURIA 2006). Herein, clinical mani- festation, investigation to demonstrate lympho-renal connection, management protocol and finally cure of a previously healthy Thai boy with idiopathic chyluria are described. CASE REPORT An 8-year-old boy pre- sented to a pediatric ne- phrologist for evaluation of passing cloudy white urine for 1 year. The patient had Fig 1–Gross appearance of cloudy white urine. difficulty in voiding urine particularly in the morning due to white ultra-heat temperature cow’s milk, fruits clots obstructing the urethra. Although and snacks. the patient had been treated for urinary Physical examination revealed afe- tract infection with oral antibiotics three brile, a blood pressure of 112/72 mm Hg, times during one year but with negative weight of 25 kg (at the 50th percentile), urine cultures, the symptoms still per- height of 124 cm (between the 25th and sisted. The results of urinalysis showed 50th percentiles), a visible BCG scar, no positive proteinuria off and on and the hardening and thickening of the skin, patient still grew well with the normal no pitting edema or swelling of legs, no growth percentiles during the previous abdominal mass, normal urethral opening year. The physician was unaware of other and scrotum, no phimosis, and no super- uncommon conditions. ficial lymphadenopathy. The consulting pediatric nephrologist A complete blood count showed noticed that by drinking large amounts of hemoglobin level of 12.5 g/dl, a white water, the patient’s urine became clearer blood cell count of 7,300/mm3 with 49.2% and the patient could void urine without neutrophils, 38.9% lymphocytes, 8.3% difficulty. The patient had no history of monocytes, 3.3% eosinophils (absolute prolonged fever, burning sensation dur- eosinophils 240 cell/mm3), and a normal ing urination, brownish/reddish urine or platelet count. Blood chemistry revealed unexplained edema of extremities. No blood urea nitrogen level of 8 mg/dl, cre- history of blunt trauma at abdomen or atinine 0.4 mg/dl, cholesterol 158 mg/dl, back, elephantiasis, chronic cough, weight and albumin 3.5 g/dl. loss, joint pain, hair loss or skin lesions Urinalysis showed cloudy white color occurred. The patient had traveled to (Fig 1), specific gravity of 1.025, pH 6.0, Myanmar four years ago. There was no 4+ albumin level, no presence of sugar, family history of tuberculosis, chronic positive occult blood, no nitrite present, cough or kidney diseases. Dietary his- white blood cells of 3-5/high power field tory revealed regular intake of solid food, (HPF), red blood cells of 10-20/HPF with- Vol 48 No. 2 March 2017 361 SOUTHEAST ASIAN J TROP MED PUBLIC HEALTH A A B B C Fig 2–Cystoscopy with retrograde pyelography. A) right side showing a lymphatic fistula tract at the right lower calyx (arrows); B) left side showing normal finding. out dysmorphic shape, and no presence of crystals. Urine triglyceride was 29 mg/dl whereas serum triglyceride was 137 mg/dl. A urine culture was negative. Fig 3–Examples of an MCT-rich diet menu. Based on the patient’s remarkable his- A) steamed rice, fried boiled egg white with tamarind sauce, and vegetable soup tory and urine triglyceride level, chyluria with chicken breast; B) fried rice with was diagnosed. The possible causes of egg white and chicken breast, vegetable chyluria were filariasis, abdominal tumor, soup with chicken breast, and papaya; C) or congenital anomaly. steamed rice, fried chicken breast, stirred Microfilaria in the blood was not fried mixed vegetables, and rose apple. 362 Vol 48 No. 2 March 2017 SCLEROTHERAPY IN IDIOPATHIC CHYLURIA found and serologic test for antifilarial quently, urine triglyceride became measur- IgG was negative. Stool examination for able (4 mg/dl) 6 days later. Although the parasites and parasite eggs was negative. MCT-rich diet was re-introduced again, Ultrasonography of the urinary the patient’s urine kept being cloudy and system, intravenous pyelography and urine analysis showed protein at 4+ level computer tomography of abdomen and and triglyceride of 1,356 mg/dl. These re- pelvis disclosed no detectable abnormal sulted from the patient’s poor compliance findings. Radionuclide lymphoscintigra- with the MCT-rich diet. phy was conducted to determine site of With parents’ consent, sclerotherapy chylous leakage by intradermal injection under retrograde pyelography using 10 of Tc-99m dextran in the webs between the ml of 1% povidone iodine solution in- first and second toes of both feet, 1 mCi jected through a ureteric catheter into the per point. Immediate imaging for whole right renal pelvis under fluoroscopy guide body and dynamic imaging for abdomen was performed. The patient continued to were performed. There appeared faint consume the MCT-rich diet for 1 month af- accumulation of radioactivity in an area ter sclerotherapy and then was placed on located at the left lateral and superior to a normal diet. Chyluria was not resolved the urinary bladder, which might indicate completely based on the presence of urine leakage of lymph at the distal part of the protein at 4+ level and urine triglyceride left ureter. of 43 mg/dl at day 51 after sclerotherapy. Cystoscopy demonstrated an efflux of On day 84 a second sclerotherapy with milky urine from the right but clear urine 20 ml of 1% povidone iodine solution from the left ureteric orifice. Retrograde was initiated. No adverse effects were pyelography demonstrated a lymphatic observed and the MCT-rich diet was dis- fistula tract at the right lower calyx (Fig continued a day after sclerotherapy. 2A) but normal left pyelography (Fig 2B). The patient’s progress was followed- Subsequent to a conservative treat- up several times post-sclerotherapy. The ment with a medium-chain triglyceride patient was asymptomatic and urine tests (MCT)-rich diet, urine became clearer showed the presence of neither protein and urine triglyceride and protein were nor triglyceride. Chyluria did not reap- undetectable. Neither clinical symptoms pear at a 4-year follow-up visit. nor signs of essential fatty acid deficiency and fat-soluble vitamin deficiencies were DISCUSSION observed. At 80 days of conservative MCT- rich diet treatment, the patient’s body We report a clinical progress of idio- weight and serum albumin level increased pathic chyluria in a previously healthy to 26.3 kg and 4.1 g/dl, respectively. At Thai boy. The patient was investigated this time the physician team thought that for possible causes but they all came up normal diet should be restored because all negative. The patient was refractory to an clinical symptoms and laboratory results MCT-rich diet. Finally, sclerotherapy with were normal without signs of essential 1% povidone iodine solution was em- fatty acid and fat-soluble vitamin defi- ployed twice and resolved the symptom. ciencies from the avoidance of long-chain Normally, urine does not contain tri- triglyceride in the treatment diet. Conse- glyceride and chyle is characterized by a Vol 48 No. 2 March 2017 363 SOUTHEAST ASIAN J TROP MED PUBLIC HEALTH high content of triglyceride (in a form of diotracers used in lymphoscintigraphy chylomicrons), albumin, globulin, lym- are colloidal (eg, Tc-99m sulfur, or Tc-99m phocytes and certain electrolytes.
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