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Spinal Cord (2004) 42, 7–13 & 2004 International Spinal Cord Society All rights reserved 1362-4393/04 $25.00 www.nature.com/sc Clinical Case Discussion Calculus anuria in a spina bifida patient, who had solitary functioning kidney and recurrent renal calculi S Vaidyanathan*,1, BM Soni1, J-J Wyndaele2, AZ Buczynski3, E Iwatsubo4, M Stoehrer5, H Madersbacher6, R Peschel6, G Singh1, JWH Watt1, PL Hughes7 and P Sett1 1Regional Spinal Injuries Centre, District General Hospital, Southport, UK; 2Centrum Urologische Revalidatie, Universitair Ziekenhuis Antwerpen, Edegem, Antwerpen, Belgium; 3Metropolitan Rehabilitation Centre, Department of Neurourology, Konstancin, Poland; 4Kyushu University School of Medicine, LWC Spinal Injuries Centre, Igisu, Iizuka, 820-8508, Japan; 5Urological Department, Berufsgenossenschaftliches, Murmau, Germany; 6University Klinik Innsbruck, Anichstrae 35, A-6020 Innsbruck, Austria; 7Department of Radiology, District General Hospital, Southport, UK Study design: Clinical case report with comments by colleagues from Austria, Belgium, Germany, Japan, and Poland. Objectives: To discuss challenges in the management of spinal bifida patients, who have marked kyphoscoliosis and no vascular access. Setting: Regional Spinal Injuries Centre, Southport, UK. Methods: A female patient, who was born with spina bifida, paraplegia and solitary right kidney, had undergone ileal loop urinary diversion. Renal calculi were noted in 1986. Percutaneous nephrostolithotomy was performed in 1989 and there was no residual stone fragment. However, she developed recurrence of calculi in the lower pole of the right kidney in 1991. Intravenous urography, performed in 1995, revealed right staghorn calculus and hydronephrosis. Chest X-ray showed markedly restricted lung volume due to severe kyphoscoliosis. In 2000, she was declared unsuitable for anaesthesia due to a lack of venous access and a high likelihood of difficulty in weaning off the ventilator in the postoperative period. In June 2002, she developed anuria (urine output ¼ 18 ml/24 h) due to ball-valve-type obstruction by a renal stone at the ureteropelvic junction. Urea: 14.4 mmol/l; creatinine: 236 ml/l. Ultrasound showed right hydronephrosis. Percutaneous nephrostomy was performed. Results: Following relief of urinary tract obstruction, there was postobstructive diuresis (3765 ml/24 h). However, the patient expired 19 days later due to progressive respiratory failure. Conclusion: In this spina bifida patient, who had reached the age of 35 years, severe kyphoscoliosis and lack of vascular access presented insurmountable challenges to implement the desired surgical procedure for removal of stones from a solitary kidney. Spinal Cord (2004) 42, 7–13. doi:10.1038/sj.sc.3101545 Keywords: spina bifida; anuria; kidney calculi Introduction A retrospective reviewof 327 patients withneural tube increased risk of renal calculus formation. Apart from a defects showed that there is a higher incidence of high incidence of nephrolithiasis, the prevalence of renal nephrolithiasis in patients with neural tube defects than parenchymal damage is also high in patients with spinal in the general population, and the risk of stone bifida. In all, 19.4% of the patients attending a recurrence is also elevated.1 Renal stones were noted multidisciplinary spina bifida clinic had renal parench- in 10.7% of those 12 years old or older. Interventional ymal damage.3 We present a case of calculus anuria in a management of the stones in these patients was 35-year-old female patient with spina bifida, who had associated with 87% recurrence rates after intervention. severe kyphoscoliosis and almost no vascular access. Rege et al2 observed that children with myelomeningo- coele, who had ileal conduit urinary diversion, had an Case history *Correspondence: S Vaidyanathan, Regional Spinal Injuries Centre, A 35-year-old female patient presented in 2002 with a District General Hospital, Town Lane, Southport PR8 6PN, UK history of not passing urine for approximately 24 h. The Calculus anuria in a spina bifida patient S Vaidyanathan et al 8 day before presentation, she had developed pain in the stomach and back. She had not passed urine from the urostomy. On the day of attending the hospital, she was noticed to be shivering and breathless. Past history included operations for hydrocephalus, spinal fixation, and renal stone. She had a solitary right kidney draining into an ileal loop urostomy. Percutaneous nephrostolithotomy had previously been performed in 1989, when she was 22 years old. The initial puncture was unsuccessful, but a second puncture was made further laterally and the mid-pole calyx was accessed. The track was dilated and lithotripsy of intrapelvic stone was performed. Rescreening showed solitary lower pole calculus remaining, which was inaccessible from within. Therefore, another track was established to reach the lower pole calyx. Electrohy- draulic lithotripsy was performed. The patient was absolutely free of renal stones at the end of the Figure 2 IVU of 18 May 1999 shows the right calyceal system procedure. dilated. There is marked cortical thinning In 1991, a routine intravenous urography (IVU) showed opaque calculi in the lower pole of right kidney, which was functioning. In 1995, a routine IVU showed The patient was assessed in 2000 for pyelolithotomy. right staghorn calculus (Figure 1) and hydronephrosis. Her weight was 53.7 kg. Blood pressure was 145/95 Marked thoraco-lumbar scoliosis was noted. In 1997, a mmHg. The respiratory rate was 30 per minute at rest. routine IVU showed excretion of contrast by the right The patient became dyspnoeic on mild exertion. She had kidney. There was loss of cortical thickness with no daytime somnolence. She would sleep on an air dilatation of the pelvicalyceal system consistent with mattress with two pillows in a propped-up position. She chronic pyelonephritis. In May 1999, the IVU revealed a was very short with a protuberant abdomen. She had a dilated right calyceal system with marked cortical scar on the right side of the neck from a ventriculo-atrial thinning (Figure 2). In August 1999, MAG 3 renogram shunt. She had a more extensive scar on the left side of showed a single functioning right kidney demonstrating the neck. Creatinine clearance was 18 ml/min. Tidal poor uptake of 2.9% at 2 min. The excretion was slow. volume: 150 ml. Vital capacity: 470 ml. The results of An ultrasound scan of the right kidney was performed in arterial blood gas analysis, which were performed on 20 December 1999; the scan showed dilatation of the upper March 2000, were as follows. pole calyces with rather good overlying cortical thick- ness. However, there was loss of cortical thickness over the mid- and lower poles of the right kidney. A staghorn calculus was noted. pH 7.29 (7.32–7.45) pCO2 4.4 kPa (4.6–6.0) pO2 11.8 kPa (12.0–13.3) Actual bicarbonate: 16.2 mmol/l (20.0–32.0) Base excess: À8.6 mmol/l (À2.0–2.0) Standard bicarbonate: 18.1 mmol/l (22.0–26.0) Comment: acute metabolic acidosis The patient was considered unsuitable for anaesthesia for pyelolithotomy on account of the very high like- lihood of difficulty in weaning off the ventilator after a surgical procedure, which is to be performed under general anaesthesia. There was almost no venous access; the usual subclavian and jugular veins were not accessible because of previous surgical procedures for the ventriculo-atrial shunt. In June 2002, the patient’s mother brought her to the spinal unit with a history of not passing urine for about 24 h. An electrocardiogram showed sinus tachycardia, but no features attributable to hyperkalaemia. Urea: Figure 1 X-ray KUB of 28 March 1995 shows staghorn 14.7 mmol/l; creatinine: 224 m/l; bicarbonate: 13 mmol/l; calculus in right kidney sodium: 130 mmol/l; potassium: 4.0 mmol/l. The patient Spinal Cord Calculus anuria in a spina bifida patient S Vaidyanathan et al 9 was given meropenem 500 mg intravenously. Intrave- nous infusion of sodium bicarbonate 1.26% was started. Ultrasound revealed moderate hydronephrosis with calculi in the right kidney. Right nephrostomy was planned. During the radiological procedure, the patient was turned prone from the supine position. The heart rate decreased to 44 and oxygen saturation dropped to 67% on air. The patient was given atropine sulphate 0.6 mg intravenously. The heart rate increased to 110 and oxygen saturation rose to 99% while breathing air with 4 l of added oxygen. A single puncture of the right kidney was performed and an 8-Fr, locking pigtail catheter was inserted. The radiological procedure took less than 10 min and was accomplished without anaes- thesia or sedation. Following relief of urinary tract obstruction, there was postobstructive diuresis (3765 ml/ 24 h). At 2 weeks after nephrostomy, plasma creatinine was 125 m/l; urea: 5.2 mmol/l. Figure 4 CT of abdomen, performed on 25 June 2002 (5 days Chest X-rays taken at 4, 6 and 11 days after nephrostomy), shows that there is still moderate right after nephrostomy showed clear lungs; but the lung hydronephrosis. A gallstone is seen in the gall bladder. The left volume was restricted due to kyphoscoliotic deformity kidney is not identified of the vertebral column (Figure 3). A CT scan of the abdomen, which was performed 5 days after nephrost- omy, showed the nephrostomy tube located in the proper position within the right kidney. There was still moderate right hydronephrosis (Figure 4). There was no evidence of any perinephric or other abdominal collec- tion. The left kidney was not identified. A nephrosto- gram was performed 8 days after percutaneous nephrostomy. Calculi were seen in the pelvicalyceal system. There was some narrowing at the level of the pelvi-ureteric junction. However, contrast flowed freely into the right ureter and then into the ileal loop (Figure 5). This led us to conclude that anuria was the result from a ball-valve-type obstruction due to one of the calculi nowlying freely in the renal pelvis.

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