Abstract Hydatidosis or echinococcosis is a parasitic disease caused by Echinococcus granulosus or E. multilocularis, which forms cysts in the liver and lung.
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Musculoskeletal and Adipose Tissue Hydatidosis Based on the Iatrogenic Spreading of Cystic Fluid During Surgery: Report of a Case Luigi Iuliano, Angela Gurgo, Elisabetta Polettini, Gianfranco Gualdi, and Paolo De Marzio Institute of Clinical Medicine I, University “La Sapienza,” 00185 Rome, Italy
Abstract Hydatidosis or echinococcosis is a parasitic disease caused by Echinococcus granulosus or E. multilocularis, which forms cysts in the liver and lung after penetrating the duodenal mucosa and entering the portal circulation. The liver and lung act as a filter but some embryos enter the general circulation and disseminate throughout the body. Muscoloskeletal involvement is a rare manifestation of hydatidosis, which is usually reported to affect a single muscle. We report here a rare case of a 68-year-old man with widespread hydatidosis of the retroperitoneum and the subcutaneous adipose tissue, and with multiple muscle involvement in the absence of liver, lung, and spleen involvement. The patient underwent surgical excision of a subcutaneous hydatid cyst 7 years earlier. It is likely that the large dissemination of parasites resulted from accidental rupture of the primary focus during surgery with consequent release and spreading of scolices via lymphatics. Key words Hydatidosis · Parasitosis
Introduction Hydatidosis or echinococcosis is a parasitic disease caused by either Echinococcus granulosus or E. multilocularis, which forms cysts in the liver and lung.1 The pathology associated with hydatid disease results from pressure effects, obstruction, or allergic responses to cystic fluid. However, hydatidosis frequently remains asymptomatic, and hydatid cysts are often found by chance on physical examination or by imaging studies done for other reasons.
Reprint requests to: L. Iuliano Received: October 25, 1999 / Accepted: March 24, 2000
The therapeutic options for hydatid disease are relatively limited. Only symptomatic cysts require surgical removal.2 The effectiveness of surgery should be improved by perioperative chemotherapy with albendazole.3 For asymptomatic cysts albendazole is the treatment of choice.4 In this paper, we describe the rare case of a 68-year-old man with widespread hydatidosis of the retroperitoneum, involving the muscles ileopsoas, lumbar, adductor, and pectineus, and the subcutaneous adipose tissue of the right inguinal region and the adjacent part of the upper thigh, without any involvement of the liver, lung, or spleen.
Case Report A 68-year-old man was admitted to our hospital with a 2-month history of gradually increasing weakness, exertional dyspnea, and dizziness. Seven years earlier the patient had developed a subcutaneous hydatid cyst in the right lateral abdominal region which was removed by surgery. At the operation the cyst appeared to be a gelatinous mass, measuring about 6 cm in diameter, covered by a translucent membrane. A histological examination showed hydatid endocysts and exocysts with a foreign-body reaction. After surgical removal, no adjuvant treatment by either albendazole or any other types of anthelminthic chemotherapy was performed. On admission, laboratory analyses revealed only severe sideropenic anemia, and the patient was treated with a transfusion of packed red cells and iron therapy. Physical examination revealed a round swelling, measuring about 8 cm in diameter, palpable in the right inguinal region. The lump was painless and appeared to be fixed to the underlying tissue. A presumptive diagnosis of cyst or neoplasm was made and a computed tomography (CT) study was performed. CT scans revealed a voluminous multilocular cystic mass (10 3
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ment and left the hospital in disagreement with the physicians.
Discussion
Fig. 1. Computed tomography (CT) scan showing the hydatid cyst in the context of the ileopsoas muscle
Fig. 2. CT scan showing a hydatid cyst in the subcutaneous adipose tissue of the right inguinal region and the adjacent part of the upper thigh
8 cm) with calcified walls in the right iliac fossa, consistent with a hydatid cyst. The cyst was localized in the ileopsoas muscle, with compression and displacement of iliac vessels and small intestine (Fig. 1). Another cyst, measuring about 4 cm in diameter, was also found in the retroperitoneum, contiguous to the lumbar muscle. In addition, several smaller cysts were found in the subcutaneous adipose tissue of right inguinal region and the adjacent part of the upper thigh (Fig. 2), while other cysts infiltrated pectineus and adductor muscles. The liver, lung, spleen, and kidneys were all normal. An indirect hemoagglutination test for hydatidosis was positive (titer 1 : 12 800). As a result, a diagnosis of widespread musculoskeletal and subcutaneous hydatidosis was made, and a 6-week therapy regimen with albendazole before surgery was advised;4 however, the patient refused both the surgical and medical treat-
Humans acquire hydatidosis by ingesting viable parasite eggs excreted by a definitive host. Once in the intestinal tract, the embryos escape from the eggs, penetrate the duodenal mucosa, and enter the portal circulation. The liver and lung act as a filter but some embryos may nevertheless enter the general circulation and disseminate throughout the body. The most common sites of echinococcosis are the liver (50%–70%), lung (20%), and spleen (5%–8%), while the kidneys, bone, brain, spinal cord, and heart are rarely involved.1 Symptoms are often absent and, in many cases, an infection is incidentally detected by imaging studies. In our case, as frequently occurs, the diagnosis of hydatidosis was made by chance. In fact, the patient entered the hospital because of a severe anemia in the absence of any symptoms attributable to musculoskeletal hydatidosis. Musculoskeletal involvement is a rare manifestation of hydatidosis, since it is usually reported to affect only a single muscle.5 The interesting aspects of the case reported herein are the unusual and dramatic spread of hydatidosis to both different muscles and to adipose tissue, while the liver, lungs, and spleen were not involved. Our patient underwent a surgical excision of a subcutaneous hydatid cyst 7 years earlier. It is likely that the large dissemination of parasites resulted from an accidental rupture of the primary focus during surgery with the consequent release and spread of scolices. The absence of liver, lung, and spleen involvement suggests that spreading did not occur via the blood vessels. According to Cant,6 parasites could possibly have disseminated via the lymphatics. In conclusion, we have presented a patient with multifocal widespread hydatidosis who had a previous history of a surgically treated hydatid cyst in the lateral abdominal region 7 years earlier. The accidental release of cystic fluid during the previous surgery was likely responsible for parasitic dissemination through the lymphatics to the retroperitoneum, muscles, and adipose tissue, therefore bypassing the usual sites of the liver, lungs, and spleen.
References 1. Mandell G, Douglas R, Bennet J (1985) Principles and practice of infectious diseases. Churchill Livingstone, New York 2. Bengisun U, Tunc G, Kesenci M, Kuzu I, Demirci S, Alic B (1997) Surgical treatment of advanced alveolar hydatid disease of the liver: a report of five cases. Surg Today 27:261–265
L. Iuliano et al.: Musculoskeletal and Adipose Tissue Hydatidosis 3. Uhl W, Loffler H, Zimmerman A, Tcholakov O, Gloor B, Buchler MW (1999) Surgical treatment of the echinococcosis of the liver. Swiss Surg 5:126–132 4. Horton RJ (1989) Chemotherapy of Echinococcus infection in man with albendazole. Trans R Soc Trop Med Hyg 83:97
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5. Zambelli S, Carnevale G, Prata M, Barbero E, Mastinu A, Vergara E (1991) Hydatidosis of the psoas muscle: description of a case and review of the literature. Arch Esp Urol 44:864–866 6. Cant PJ (1995) Migration of Echinococcus granulosus via lymphatics. Lancet 345:393–394
