骨盆腔放線菌症引發之輸尿管纖維化相關腎臟水腫之病例報告

江長和¹、范玉華^1,2、黃志賢^1,2 

¹臺北榮民總醫院泌尿部

²國立陽明交通大學醫學院泌尿學科 書田泌尿科學研究中心

A case report of hydronephrosis associated with

pelvic actinomycosis-related ureteral fibrosis

Chang-Ho Chiang¹, Yu-Hua Fan^1,2,William J. Huang^1,2

¹Department of Urology, Taipei Veterans General Hospital,

²Department of Urology, College of Medicine and Shu-Tien Urological Research Center, National Yang Ming Chiao Tung University, Taipei, Taiwan

Introduction:

Actinomycosis is a rare bacterial infection, and the estimated annual incidence of the disease is approximately 1 case per 300,000. Pelvic actinomycosis accounts for only 3% of all human actinomycosis cases.

Pelvic actinomycosis can cause ureteric stricture, which is a common complication of the disease. It can also lead to fibrosis and abscess formation in other pelvic organs, including the bladder, rectum and sigmoid colon. The disease can be misdiagnosed as ovarian malignant cancer due to similar imaging findings until pathology is reported. Herein, we present a case of hydronephrosis associated with pelvic actinomycosis-related ureteral fibrosis.

Case report:

A 38-year-old female had a medical history of pons infarction resulting in total paralysis and dysphagia, Type II diabetes mellitus and chronic hepatitis B virus infection. The patient initially presented with constipation and abdominal pain, which was not relieved by laxative agents. Then she had been experiencing persistent right flank pain for several months. She went to a local hospital where severe right CV angle pain was accidentally noticed, and right hydronephrosis due to external compression of a right ovarian cyst was diagnosed after an image survey. Therefore, she underwent a right ovariectomy first and then ureteral balloon dilatation 2 months after the ovariectomy. However right hydronephrosis persisted, and she still suffered from right flank pain.

She visited our hospital for a second opinion. A diagnostic ureteroscopy was arranged for to survey the hydronephrosis, which revealed a severe ureteral stricture in the lower third of the ureter, through which only a ureteral guidewire could pass. Right retrograde pyelography (RP) was also performed, showing that the right lower third ureteral stricture was too narrow to allow the passage of contrast medium. After shared decision-making at the outpatient clinic, she opted to undergo right RP and open ureterolysis. A long segmental stricture of the right lower third ureter, approximately 5cm proximal to the ureteral orifice, and about 4cm in length, was identified during the RP procedure. Severe adhesion of the right ureter with surrounding fibrotic tissue, which was also attached to the cecum and sigmoid colon, were observed in the surgical field. A colorectal surgeon was consulted for a colonoscopy, which showed no intraluminal tumor during the operation. A frozen section of the suspicious pelvic tumor revealed abscess and fibrosis during the operation. Antibiotics with Cefmetazole were administered after the operation, as Proteus mirabilis was identified in the urine culture. However, both the pathology of the pelvic soft tissue and the permanent sections of the frozen section reported abscess formation and fibrosis, with Actinomyces species detected. Tracing back her medical history, she had an intra-uterine device (IUD) implanted, which was removed about 1 month after the right ovariectomy at a local hospital. The right double-J stent was removed 1 month after the ureterolysis at our hospital.

The patient then underwent antibiotic treatment with Amoxicillin, 2000mg twice a day, at the infection outpatient department for 6 months. A sonography was arranged after the treatment had finished, revealing complete regression of right hydronephrosis.

However, two months later, bilateral flank pain was noted. A computed tomography scan showed the recurrence of right hydronephrosis and a 3.2 x 4.5 cm soft tissue mass in the right adnexal region with a stranding appearance. The mass was attached to the uterine body and rectum, consistent with actinomycosis. Subsequently, a right diagnostic ureteroscopy and RP were performed, which revealed a segmental stricture in the right lower third ureter, measuring approximately 3cm in length. A double-J stent was smoothly inserted.

The patient was admitted to the Infectious Disease wards due to a urinary tract infection three months later. Lab data showed an elevated C-reactive protein level and leukocytosis. Magnetic resonance imaging revealed an ill-defined soft tissue lesion with intermedial high signal on T2WI, regional stranding, and tethering. It measured approximately 3.7x4.6x5.0cm and was located on the right pelvic side wall, resulting in right hydronephrosis and hydroureter. After a two-week treatment with Ceftriaxone, 2000mg once a day, the patient was discharged with oral Amoxicillin, 2000mg twice a day, to be taken at home. The patient had been undergoing antibiotic treatment with Amoxicillin, 2000mg twice a day, for a total of 8 months so far.

Conclusion:

Pelvic actinomycosis is predominantly associated with the long-term use of intrauterine devices. Recently, some case reports of pelvic actinomycosis have claimed that many patients are cured with short-term (<6 months) of antibiotic therapy. However, the traditional treatment for pelvic actinomycosis involves high-dose penicillin for an extended period (6 months to 1 year). Radiological response should be closely monitored after treatment is finished. If a recurrence of pelvic actinomycosis occurs, intravenous penicillin may be considered, requiring hospitalization for at least one month.