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  IN THIS Article
 ::  Introduction
 ::  Case report
 ::  Discussion
 ::  Acknowledgements
 ::  References

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Year : 1982  |  Volume : 28  |  Issue : 2  |  Page : 115-7

Splenic and orbital hydatid cysts and treatment with mebendazole (a case report).







How to cite this article:
Agarwal M B, Kamdar M S, Tilve G H, Bapat R D, Vakil N B, Desai M B. Splenic and orbital hydatid cysts and treatment with mebendazole (a case report). J Postgrad Med 1982;28:115


How to cite this URL:
Agarwal M B, Kamdar M S, Tilve G H, Bapat R D, Vakil N B, Desai M B. Splenic and orbital hydatid cysts and treatment with mebendazole (a case report). J Postgrad Med [serial online] 1982 [cited 2019 Nov 18];28:115. Available from: http://www.jpgmonline.com/text.asp?1982/28/2/115/5581




  ::   Introduction Top

Hydatid cyst is not an uncommon disorder in India. Liver is the most common site of this disorder since the route of spread is from the duodenum with the entry of the ova into the portal system. The right lobe is involved more often. Sometimes the ova that escape the liver produce pulmonary disease. Overall, about 60% of the cases have hepatic echinococcosis while about 25 have a pulmonary affection.[6], [7] The next two commonest sites which are affected are brain and bones.[6], [7] Affection of any other tissue is rare and only occasionally reported.[7]
Although surgery was considered as the only treatment for this disorder, recently medical management with mebendazole both in humans[1], [2] and animals[3], [4] has been described and by now, several optimistic reports have become available.[5], [9]
We are reporting a case who had hydatid cyst of spleen and an intra-occular cystic lesion which subsided with mebendazole.

  ::   Case report Top

M.P., a 38 year old male patient was admitted with history of painless proptosis of the left eye which was gradually progressive for 2 years. He had also noticed a lump in the left hypochondrium which had also painlessly increased over 2-3 years. He had no other complaints. There was no history of contact with dogs. His examination revealed a moderate proptosis of the left eye with intact external occular movements and vision. Perabdomen examination revealed a spleen of 18 cm below the costal margin; however, no notch was felt on the anterior border and this border was rounded and hence a suspicion was raised regarding the nature of swelling. Liver was not palpable and he had no other significant findings. Investigations revealed normal hemogram and normal serum biochemistry. There was no evidence of eosinophilia or hypersplenism. ESR was normal. X-ray chest was normal while plain X-ray of the abdomen showed no calcifications. The hepatosplenic scan using 99mTc showed a normal liver while splenic tissue was reduced to a narrow strip in the left subdiaphragmatic region with a rounded filling defect. The brain and bone scans were normal. Ophthalmic venography revealed an avascular space occupying lesion in the left orbit [Fig. 1]. Casoni's test was negative. Patient was subjected to splenectomy and the splenic cyst was proved to be a hydatid cyst [Fig. 2]. As he refused for any surgery on the eye, he was put on oral mebendazole in doses of 30 mg; kg day (1.2gd) in four divided doses. The drug was continued for 6 months; however, only after one month, the proptosis had totally disappeared.

  ::   Discussion Top

This case had two unusual features: (1) the unusual sites for occurrence of hydatid cyst, and (2) the response to mebendazole.
Splenomegaly is an important but common medical problem. Causes of splenomegaly form an exhaustive list for a student of medicine. Cysts, tumours and abscesses find a last place in such lists. However, a good clinical examination is often helpful as in this case. The abdominal lump in this case had all the characteristic features of an enlarged spleen but for two or three odd ones e.g. rounded borders, absence of a notch and absence of drop in Hb, (T) WBC, neutrophils and/or platelets (i.e. hypersplenism which is expected in a splenomegaly o: this size). It was these suspicions which led to the isotope scan and ultrasonic studies which turned out to be so informative.
Although hydatid cyst can occur in any organ or tissue of the body, its occurrence in the spleen and orbit without affecting the liver, lung, brain and bones is uncommon.[2] The Casoni's test has gone into a .total disrepute because of very high incidence of both false positive and negative results.[8] In the absence of calcification, the diagnosis at odd sites remains unproven except by surgery and histopathology.
In the present case, it can be assumed with confidence that the orbital mass was a hydatid cyst as it was gradually increasing for over two years; it was avascular and finally, the patient had hydatid cyst in the spleen.
Till now, the only treatment for hydatid cyst was considered to be surgical. Surgery, however, may not always be possible when the cysts are multiple or inaccessible. Several optimistic reports as mentioned before are now available regarding medical management of this entity by using mebendazole. This drug has been used both for Echinococcus granulosus[1] and Echinococus multilocularis[9] with interesting results. This drug kills the larvae stage of Echinococcus by limiting its glucose uptake.[5] It is clear that a high dose protocol is required and the treatment must be prolonged. The recommended doses at the moment appear to be about 30 mg/kg a day for four weeks.[5] However, the data on exact dosage and duration of therapy differ widely and the optimum treatment programme remains to be detarmined.[2], [9] We need to assess the value of serology for monitoring the effectiveness of this drug. Proof that the parasites have lost their viability from direct histological examination of the cyst's contents is at present the only criterion we have against which new tests can be assessed. The reports of its failure or relapses appear to be mainly due to short term therapy or inadequate dosages. The only risk of such therapy is said to be anaphylaxis[4] which, in some series, appear to be very common.[5] Occasional reports of gastritis, pruritus and certain biochemical abnormalities of liver function are available.[5] Overall, it can be said that mebendazole therapy has opened a new era in the management of hydatid disease.

  ::   Acknowledgements Top

We are thankful to Dr. C. K. Deshpande, Dean, Seth G.S. Medical College and K.E.M. Hospital for permission to publish the case report. We are also thankful to our Ophthalmology and Radiology Departments' colleagues for the help in investigating this case.

  ::   References Top

1.Beard, T. C., Rickard, M. D. and Goodman, H. T.: Editorial: Medical treatment for hydatids. Med. J. Aust., 1: 633-635, 1978.  Back to cited text no. 1    
2.Bekhti, A., Schapps, J. P., Capron, M., Dessaint, J. P., Santoro, F. and Capron, A.: Treatment of hepatic hydatid disease with mebendazole. Preliminary results in four cases. Brit. Med. J., 2: 1047-1051, 1977.  Back to cited text no. 2    
3.Heath, D. D., Christie, M. J. and Chevis, R. A. F.: The lethal effect of mebendazole on secondary Echinococcus granulosus, cysticerci of Taenia pisiformis and tetrathyridia of Mesocestoides cacti. Parasitology, 70: 273-285, 1975.  Back to cited text no. 3    
4.Kammerer, W. S. and Judge, D. M.: Chemotherapy of hydatid disease (Echinococcus granulosus) in mice with mebendazole and bithionol. Amer. J. Trop. Med. & Hyg., 25: 714-717, 1976.   Back to cited text no. 4    
5.Kern, P., Dietrich, M. and Volkmer; K. J.: Chemotherapy of echinococcus with mebendazole-clinical observations of seven patients. Tropenmed. Parasitol., 30: 65-72, 1979.  Back to cited text no. 5    
6.Lewis, J. W. Jr., Koss, N. and Kerstein, M. D.: A review of ecchinococcal disease. Ann. Surg., 181: 390-396, 1975.   Back to cited text no. 6    
7.Saidi, F.: "Surgery of Hydatid Disease". W. B. Saunders, Philadelphia 1976.  Back to cited text no. 7    
8.Scully, R. E., Galdabini, J. J. and McNeely, B. U.: Case reports of the Massachusetts General Hospital. Case 251979. New. Engl. J. Med., 300: 1429-1434, 1979.  Back to cited text no. 8    
9.Wilson, J. F., Davidson, M. and Rausch, R. L.: A clinical trial of mebendazole in the treatment of alveolar hydatid disease. Amer. Rev. Respir. Dis., 118: 747-757, 1978.  Back to cited text no. 9    

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Online since 12th February '04
2004 - Journal of Postgraduate Medicine
Official Publication of the Staff Society of the Seth GS Medical College and KEM Hospital, Mumbai, India
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