泄殖腔異常患者之泌尿道結石：罕見病例報告

李岳霖¹、林嘉緯²、高偉棠²

行政院衛生福利部雙和醫院一般醫學科¹，泌尿科²

Left ureteral stone and left renal stone in patient with cloacal malformation:
 a rare case report

Lee Yueh-Lin¹, Lin Jia-Wei², Kao Wei-Tang²

¹Department of General Medicine, Taipei Medical University-Shuang Ho Hospital, New Taipei city, Taiwan; ²Department of Urology, Taipei Medical University-Shuang Ho Hospital, New Taipei city, Taiwan

Introduction:

Cloacal malformation is a complex malformation in which the rectum, vagina and urologic structures join into a single common channel. There are few studies about this anomaly and most of them are limited to surgical intervention and functional outcomes including urinary continence. In this report, we present a challenging case of ureterorenoscopic lithotripsy of a left ureteral and renal stone, complicated by sepsis and hydronephrosis, in a 40-year-old female with a past history of cloacal malformation status post anal reconstruction.

Case presentation:

This 40-year-old female has a past history of cloacal malformation status post two-step surgery of colostomy and anal reconstruction in infancy. She presented to our emergency department with three days of left lower abdominal pain. She also complained of on and off fever for three days, dysuria, and mild nausea. Upon arrival, she was febrile (38.8°C) with tachycardia and relative hypotension. Physical examination showed mild left lower quadrant tenderness with left costovertebral angle knocking pain. Laboratory data revealed leukocytosis, elevated CRP (31.4 mg/dL) with urinalysis indicating a urinary tract infection. Contrast-enhanced computed tomography (CT) of her abdomen and pelvis showed left upper ureteral stones with the SFU grade 3 hydronephrosis, acute pyelonephritis, and a focal renal abscess measuring 2.5 cm at middle pole region (Figure 1 and 2). We then performed percutaneous nephrostomy drainage of the renal abscess followed by treatment of sepsis including adequate fluid hydration and empirical administration of Flomoxef. Her left flank pain improved. Blood culture grew Klebsiella pneumoniae, and the antibiotics were changed to Cefmetazole once sensitivities were known. Once stabilized, a retrograde flexible ureteroscopic lithotripsy (URSL) was performed for removal of left lower ureteral stone. However, during the first half of the procedure, it took multiple attempts to access the left ureter. Furthermore, as we accessed the orifice of ureter via vagina-urethra-bladder, the upper ureter was not accessible due to middle ureteral stricture. Therefore, we changed our approach to a left mini-percutaneous nephrolithotomy and antegrade double-J stent plus retrograde flexible-URSL. The stent was then removed the following day and she was discharged. We discuss the presentation and literature review of cloacal malformations.

Conclusion: