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Saudi Journal of Kidney Diseases and Transplantation
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CASE REPORT Table of Contents   
Year : 2009  |  Volume : 20  |  Issue : 6  |  Page : 1057-1060
Hydatid cyst disease in a renal allograft recipient

1 Department of Nephrology, Transplantation and Clinical Immunology, University Hospital of Tours, Morocco
2 Department of Cytology, University Hospital of Tours, Morocco
3 Department of Digestive and Thoracic Surgery, University Hospital of Tours, Morocco
4 Department of Parasitology, University Hospital of Tours, Morocco

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Date of Web Publication27-Oct-2009

How to cite this article:
Sqalli TH, Buchler M, Al Najjar A, Paris A, Halimi JM, Nivet H, Anthonioz P, Bourlier P, Duong TH, Lebranchu Y. Hydatid cyst disease in a renal allograft recipient. Saudi J Kidney Dis Transpl 2009;20:1057-60

How to cite this URL:
Sqalli TH, Buchler M, Al Najjar A, Paris A, Halimi JM, Nivet H, Anthonioz P, Bourlier P, Duong TH, Lebranchu Y. Hydatid cyst disease in a renal allograft recipient. Saudi J Kidney Dis Transpl [serial online] 2009 [cited 2022 Sep 30];20:1057-60. Available from: https://www.sjkdt.org/text.asp?2009/20/6/1057/57263

   Introduction Top

Hydatid disease is a rare parasitic disease mainly involving the liver and lungs. It is caused by the parasitic tapeworm Echinococcus.

We report the case of a renal allograft reci­pient who was diagnosed with hepatic cystic echinococcosis five months after kidney trans­plantation. The development of tapeworm lar­vae in humans is rare, and in this case it could have been encouraged by immunosuppressive therapy. The patient was successfully treated by surgical removal of the cyst and periope­rative treatment with albendazole.

   Case Report Top

A 44-year-old Turkish man, resident of France since 1987, underwent cadaveric renal trans­plantation on 13 December 2000. The patient originally had end-stage renal disease secon­dary to nephrolithiasis and hemodialysis was initiated in February 1999. He underwent right nephrectomy in 1995 and left nephrectomy in 1999 for nephrolithisis before undergoing hemo­dialysis.

Quadruple sequential therapy, including anti­thymocyte globulin (ATG), Cyclosporin A (CsA), mycophenolate mofetil (MMF) and predniso­lone, was given as induction immunosuppre­ssive protocol. Delayed graft function required four dialysis sessions followed by a fall in serum creatinine to 103 μmol/L on day 40.

Two months later an episode of Banff 1b grade cellular graft rejection was treated with five pulses of methylprednisolone. The patient experienced concomitant cytomegalovirus in­fection treated with intravenous ganciclovir.

After five months post-transplantation, the pa­tient reported a 3-day history of fever of 39­40°C with shivering and dysuria. Physical exa­mination was essentially normal, but the serum creatinine level increased to 243 μmol/L and. a high C reactive protein level of 286 mg/L. Blood cultures and midstream urine specimen were positive for Enterococcus fecalis.

72 hours later on intravenous ampicillin and aminoglycoside, the fever remained elevated. Renal transplant and abdominal ultrasound so­nography showed a non-echogenic perihepatic cystic mass. A computed tomography (CT) scan of the abdomen revealed a 5 cm fluid contai­ning mass in the left hepatic lobe thought to be a hepatic abscess. A needle biopsy revealed free scolices and loose hooks suggesting hydatid cysts [Figure 1]. Neither the transplanted kid­ney nor the lung revealed an echinococcus focus and the echinococcal larvae had developed protoscolices. Intravenous albendazole therapy was started at 400 mg twice daily and was continued for 4 weeks.

During albendazole therapy, despite a 30% reduction in CsA from 350 mg daily to 250 mg daily, residual cyclosporine trough concentra­tions remained stable. The immunosuppressive therapy at the time also included MMF (500 mg twice daily) and prednisolone (10 mg daily).

The patient was afebrile throughout the pe­riod of treatment and his renal function gra­dually normalized, with latest serum creatinine of 116 μmol/L.

The hydatid cyst was removed surgically, and a communication between the cyst and left he­patic duct was noted during surgery. Pericys­tectomy, choledochotomy, and removal of re­maining daughter cysts from the common bile duct were performed, with sutures of the left hepatic duct. The patient recovered fully after the surgery.

Hydatid serology was negative. There was no hypereosinophilia. Liver function tests were always within the normal range. A detailed CT scan to assess extension of hydatid disease did not reveal any pulmonary, cerebral or other intra­abdominal localisations. Diagnosis of hydatid disease was confirmed by histopathology exa­mination.

Postoperative therapy (400 mg albendazole twice daily for six weeks) was initiated 7 days after surgery. A second course of albendazole therapy was administered two weeks later.

CT and ultrasound scans were repeated after completion of therapy. After 48 months of follow-up, the patient was in a good clinical condition, symptom-free and no sign of relapse.

   Discussion Top

This is the first case, to our knowledge, of hydatid cyst disease in a renal allograft reci­pient. Two forms of echinococcosis exist in Turkey x, i.e. E. granulosus and E. multilocu­laris, and they cause cystic echinococcosis (CE) and alveolar echinococcosis (AE), respectively.

In a patient with known liver hydatidosis who underwent heart transplantation the immuno­suppressive treatment seemed to have no effect on the growth of the cysts, which were re­moved surgically 14 months later. [1] In this pa­tient, the diagnosis of liver hydatidosis was based on an indirect hemagglutination test which was positive before transplantation. In our patient, no intra-hepatic cyst was observed on abdominal scan prior to transplantation. We can therefore only speculate the occurrence of cystic hydatidosis being enhanced by the immu­nunosuppressive treatment.

The diagnosis of hydatidosis in our patient was made by direct parasitology examination of the supposed hepatic abscess. We did not perform the antibody test; nevertheless, a dec­rease in antibody production has been reported with MMF. [2]

Surgery remains the first choice for treat­ment, with the potential to remove the parasite totally. Liver transplantation, in unresectable cases are encouraging, with a 5 year survival rate of 76.2% and lack of recurrence of disease despite the immunosuppressive regimen. [3]

Pre- and post-surgical treatment with alben­dazole or mebendazole has the advantage of reducing the risk of disease recurrence and intraperitoneal seeding of infection with spon­taneous cyst rupture and spillage or during sur­gery or needle drainage [4] .

Chemotherapy with benzimidazole compounds (albendazole or mebendazole) and the puncture­aspiration-injection-re-aspiration (PAIR) pro­cedure with concomitant chemotherapy offer options for treatment of hydatid disease. Alben­dazole is a clinically important antihelminthic agent with low oral bioavailability. Intestinal and hepatic albendazole metabolism leads to albendazole sulfoxide, the active metabolite, which is a CYP3A4 substrate. The concomitant use of benzimidazoles and immunosuppressive drugs, in particular calcineurin inhibitors, in transplanted patients has not been reported. Albendazole and cyclosporine A may interfere at CYP3A4 and P-glycoprotein levels and close pharmacokinetic monitoring may be needed to maintain both drugs at the therapeutic level. In our patient, the CsA dose was reduced by 30% during albendazole therapy. On the other hand, over exposure to albendazole may be attribu­ted to CsA as cyclosporin significantly reduces the intestinal elimination of albendazole sulfo­xide in rats and sheep. [5]

Antihelminthic properties of CsA, perhaps via cyclophilin, have been reported in vitro by Colebrook et al [6] CsA. Administration two days prior to infestation, resulted in significant re­duction in cyst establishment in mice, but the drug had no effect when administered 18 weeks after Echinococcus infestation. [7]

We do not know if the immunosuppressive treatment encouraged the development of hy­datid cysts, as a significant role of cellular im­munity in controlling the first stages of larva development has been shown. [8] Hildreth et al showed that cortisone treatment drastically increased both the number of Echinococcus cysts in mice and the average size of each cyst when treatment was administered at an early stage. [9]

In conclusion,we report the first case of hy­datid cyst disease in a kidney-transplanted pa­tient, treated by surgical removal of the cyst and perioperative albendazole. The interaction between albendazole and immunosuppressive drugs needs close therapeutic monitoring. No recurrence of hydatidosis occurred during 4 years' follow-up.

   References Top

1.Sobrino JM, Pulpon LA, Crespo MG, et al. Heart transplantation in a patient with liver hydat­idosis. J Heart Lung Transplant 1993;12(3): 531-3.  Back to cited text no. 1      
2.Lederer SR, Friedrich N, Banas B, Welser G, Albert ED, Sitter T. Effects of mycophenolate mofetil on donor-specific antibody formation in renal transplantation. Clin Transplan 2005; 19(2):168-74.  Back to cited text no. 2      
3.Loinaz C, Moreno-Gonzalez E, Gomez R, et al. Liver transplantation in liver disease: echi­nococcus granulosus. Transplant Proc 1998; 30(7):3268-9.  Back to cited text no. 3      
4.Smego RA Jr, Sebanego P. Treatment options for hepatic cystic echinococcosis. Int J Infect Dis 2005;9(2):69-76.  Back to cited text no. 4      
5.Merino G, Molina AJ, Garcia JL, Pulido MM, Prieto JG, Alvarez AI. Intestinal elimination of albendazole sulfoxide: pharmacokinetic effects of inhibitors. Int J Pharm 2003:263(1-2):123­-32.  Back to cited text no. 5      
6.Colebrook AL, Jenkins DD, Lightowlers MW. Anti-parasitic effect of cyclosporin A on Echinococcus granulosus and characterization of the associated cyclophilin protein. Parasi­tology 2002;125(Pt 5):485-93.  Back to cited text no. 6      
7.Hurd H, Mackenzie KS, Chappell LH. Anti­helmintic effects of cyclosporin A on proto­scoleces and secondary hydatid cysts of Echi­nococcus granulosus in the mouse. Int J Parasitol 1993;23(3):315-20.  Back to cited text no. 7      
8.Liance M, Bresson-Hadni S, Vuitton DA, Lenys D, Carbillet JP, Houin R. Effects of cyclos­porin A on the course of murine alveolar echi­nococcosis and on specific cellular and humoral immune responses against Echinococcus multi­locularis. Int J Parasitol 1992;22(1):23-8.  Back to cited text no. 8      
9.Hildreth MB, Granholm NH. Effect of mouse strain variations and cortisone treatment on the establishment and growth of primary Echino­coccus multilocularis hydatid cysts. J Parasitol 2003;89(3):493-5.  Back to cited text no. 9      

Correspondence Address:
Tarik Houssaini Sqalli
Lotissement Oulladia II, No B13, Km 8, Souissi, Rabat
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Source of Support: None, Conflict of Interest: None

PMID: 19861870

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