Case Report

0 downloads 0 Views 138KB Size Report
Extragenitourinary Retroperitoneal Primary Hydatid Cyst: A Rare. Cause of ... vagina was pushed posteriorly. ... graphy showed multiple smooth-walled cystic.

[Downloaded free from on Wednesday, September 28, 2016, IP:]

Saudi J Kidney Dis Transpl 2013;24(5):991-994 © 2013 Saudi Center for Organ Transplantation

Saudi Journal of Kidney Diseases and Transplantation

Case Report Extragenitourinary Retroperitoneal Primary Hydatid Cyst: A Rare Cause of Bilateral Lower Ureteric Obstruction and Unilateral Limb Edema Amit Goel, Punit Tiwari, Pramod Kumar Sharma, Suresh Kumar, Anup Kumar Kundu Department of Urology, IPGME&R, S.S.K.M. Hospital, Kolkata, India ABSTRACT. Hydatid cyst is an endemic disease in our country. Most commonly, it occurs in the liver and lungs. Bilateral hydroureteronephrosis is one of the rare presentations of hydatid disease. Herein, we are reporting an unusual case of hydatid disease where the primary mode of presentation was external iliac vein compression with chronic renal failure because of bilateral ureteric involvement. The patient was treated with bilateral double-J stenting to improve the renal function and operated later for removal of hydatid cyst under albendazole drug treatment. Introduction Primary hydatid disease of the pelvis (0.752.25%) is a very rare disease. Most commonly, it occurs in the liver (80%) and lungs (15%). It is caused by the parasite Echinococcus granulosus. The dog is the definitive host and sheep is the intermediate host. Man is an accidental intermediate host.1 The hydatid cyst grows slowly over years and causes symptoms usually due to compression of adjacent structures. Case Report A 38-year-old female of rural habitat had a lump in her lower abdomen since 2005, which Correspondence to: Dr. Amit Goel, Department of Urology, IPGME&R, S.S.K.M. Hospital, Kolkata, 700020, India E-mail: [email protected]

was gradually increasing in size. The patient had dull pain in both loins for two months and later started having nausea, vomiting and anorexia with puffiness of the face, right limb pitting edema and decreased urine output and constipation. On examination, the patient looked sick and was dehydrated. A large irregular lump was visible in the lower right side of the abdomen. The lump had an irregular but smooth surface and a cystic consistency. On per vaginal examination, a mass was palpated anteriorly and the vagina was pushed posteriorly. Digital rectal examination revealed rectal compression. On investigation, the patient had Hb 6.2 gm/ dL, TLC 13,400, differential count revealed eosinophilia 8%, ESR 42 mm in the first hour, serum creatinine 4.6 mg/dL, blood urea 86 mg/ dL and liver function tests revealed hypoalbuminemia. The indirect hemagglutination test was positive at 1/2148 dilution. Ultrasonography showed multiple smooth-walled cystic

[Downloaded free from on Wednesday, September 28, 2016, IP:]


lesions with septations in the abdomen and pelvis, displacing the bladder, with moderate hydroureter and hydronephrosis (Figure 1). Noncontrast computed tomography (CT) showed multiple large smooth-walled cystic lesions with septa in the pelvis and abdomen displacing the bowel, compressing the rectum posteriorly and displacing the bladder anteriorly, with moderate dilatation of both ureters and pelvicalyceal system (Figures 2 and 3). The chest X-ray was normal. Lower limb venous Doppler revealed subcutaneous edema of the right lower limb but no evidence of deep vein thrombosis. The patient was hydrated and under cover of broad spectrum antibiotics and 1.2 g/day of albendazole, was taken for cystoscopy and bilateral double-J stenting. Cystoscopy revealed normal bladder neck with normal capacity bladder, but with raised and stretched trigone and increased distance between the ureteric orifices. Double-J stenting was performed with great difficulty using an angled Terumo guide wire. After two weeks, the serum creatinine level came down to 1.4 mg/dL and the blood urea level came down to 26 mg/dL, and patient was taken up for cyst excision. Surgical exploration via midline incision revealed multiple large cystic masses predominantly in the pelvis and the right side of the abdomen. The mass was displacing the bladder anteriorly and the bowel to the left side. The rectum and sigmoid colon were also compressed. The cyst was

Goel A, Tiwari P, Sharma PK, et al

dissected from the bladder, major vessels, ureter, rectum and bowel, but the entire cyst could not be excised intact, since it was adherent to the adjacent organs. The fluid of the cyst was drained and the cavity was irrigated with 30% saline and povidone iodine solution. The cyst wall was excised as much as possible. A drain was kept and closure was performed. Histology confirmed hydatid disease. The patient recovered well and was discharged with six months of albendazole treatment. There is no recurrence of disease noted till the 8th month of follow-up. Discussion Hydatid disease is an endemic parasitic disease with world wide distribution. Man is an accidental intermediate host due to ingestion of fertilized eggs. The larvae penetrate the intestine to enter the blood stream and lymphatics and travel to the liver, lungs and other organs.1 Juxtavesical and retrovesical location of the cyst are unusual, but location in the retroperitoneum is especially rare. The vast majority of abdominal and pelvic cysts are considered to be secondary to prior hepatic localization followed by spontaneous fissuring or rupture.2 The primary cyst might then heal and even disappear, leaving behind a scar that could be overlooked by any diagnostic modality. Retrovesical and retroperitoneal hydatid cyst

Figure 1. Abdominal ultrasound showing hydatid cyst in the peritoneal cavity.

[Downloaded free from on Wednesday, September 28, 2016, IP:]

Extragenitourinary retroperitoneal primary hydatid cyst

Figure 2. CT scan showing multiple hydatid cyst in the peritoneal cavity compressing the bilateral ureter with bilateral hydroureteronephrosis.

Figure 3. CT scan showing hydatid cysts extending up to the pelvic cavity, pushing the rectum posteriorly.

disease grows slowly over years and the patient presents with lump in the abdomen and symptoms due to compression of the adjacent structures, usually with chronic renal failure (CRF) due to bladder outlet obstruction3,4 and constipation due to rectal compression.5 Our case presented with lump in the abdomen with vomiting, decreased urine output and facial puffiness due to CRF, right lower limb pitting edema and constipation. There was no cyst in


the liver or lungs. CRF in our case was due to primary huge retrovesical and retroperitoneal hydatid cyst causing extrinsic compression over both the lower ureters, causing hydroureter and hydronephrosis, and not due to bladder outlet obstruction. No symptoms of difficulty passing urine and training were noted. Also, no signs of bladder outlet obstruction were found on cystoscopy. The scope could be easily introduced through the bladder neck. Posterior compression over the bladder wall with increased inter-ureteric distance and both ureteric orifices were seen high up. This was unlike the earlier reported cases of CRF.3,4 To the best of our knowledge, this is the first case reported in the English literature presenting with right lower limb pitting edema that was due to unilateral compression of the right external iliac vein by a hydatid cyst. Diagnosis of hydatid cyst disease is made by imaging modalities and serological tests. In our case, ultrasonography and non-contrast CT scan showed multiple large cysts in the abdomen and pelvis with septations. An indirect hemagglutination test performed also suggested hydatid disease. Management of hydatid cyst disease is by exploration and excision of as much cyst as feasible. In our case, because the patient presented with symptoms of CRF and was dehydrated, we managed her with hydration and antibiotics and albendazole followed by emergency bilateral double-J stenting thus saving nephrostomy tube placement and extra morbidity. Abdomen exploration and cyst excision was performed at a later date. After eight months of follow-up, the patient was doing well with no evidence of cyst recurrence. In conclusion, hydatid cyst should be considered in the differential diagnosis of bilateral ureteric obstruction, especially in endemic areas. The combination of history, imaging and serological tests usually help in diagnosing the condition. Primary retrovesical and retroperitoneal hydatid cyst disease can obstruct both ureters and cause CRF without causing bladder outlet obstruction. It can also cause, although very rarely, external iliac vein compression and unilateral lower limb pitting edema, as

[Downloaded free from on Wednesday, September 28, 2016, IP:]


Goel A, Tiwari P, Sharma PK, et al

seen in our case. Management of ureteric obstruction by bilateral ureteric stenting should be tried in difficult cases with angled terumo guide wire rather than directly resorting to nephrostomy, which carries extra morbidity. References 1.


Angulo JC, Sanchez-Chapado M, Diego A, Escribano J, Tamayo JC, Martin L. Renal echinococcosis: Clinical study of 34 cases. J Urol 1997;157:787-94. Angulo JC, Escribano J, Diego A, SanchezChapado M. Isolated retrovesical and extra-




renal retroperitoneal hydatidosis: Clinical study of 10 cases and literature review. J Urol 1998;159:76-82. Seenu V, Misra MC, Tiwari SC, Jab R, Chandrashekhar C. Primary pelvic hydatid cyst presenting with obstructive uropathy and renal failure. Postgrad Med J 1994;70:930-2. Porcaro AB, Novella G, Beltrami P, Ficarra V, Antoniolli SZ, Malossini G. Case report: Retroperitoneal pelvic hydatid disease. Int Urol Nephrol 2002;33:23-5. Yilmaz M, Ugraş M, Işik B, Yilmaz S, Kirimlioğlu V. Acute uremia and intestinal obstruction due to retroperitoneal hydatid cyst. Turk J Gastroenterol 2007;18:258-60.