Infected primary intramuscular echinococcosis of thigh
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《美国医学杂志》
1 Department of Surgery, Pt. B.D. Sharma PGIMS, Rohtak, India
2 Department of Pathology, Pt. B.D. Sharma PGIMS, Rohtak, India
3 Department of Pediatric Surgery, Pt. B.D. Sharma PGIMS, Rohtak, India
Abstract
An unusual case of infected primary echinococcosis involving quadriceps muscles of the thigh and presenting as a cystic mass in a child is being reported. The diagnosis was made preoperatively with ultrasound examination and serology. After a cover of medical treatment the cyst was excised. Various clinical possibilities and management strategies are discussed.
Keywords: Echinococcosis; Primary intramuscular; Infected
Echinococcosis or hydatid disease may affect several organs in the body and thus represents a major challenge for the surgeons. It is rare to see echinococcosis of thigh muscles in children, like in adults cysts are seen in the liver and lungs. The causes for primary muscular localization of the disease are unknown. Preoperative diagnosis is important for proper management and to avoid the long term complication of recurrence.
Case Report
An eleven-year-old girl presented with a progressively increasing one-year-old swelling in the right thigh, following a doubtful history of trauma. Ten months prior, there was history of needle aspiration of thin fluid from the swelling and it subsided thereafter. The swelling recurred after a few months and became painful. On examination, there was a diffuse, intramuscular swelling occupying the anterior region of right thigh. It measured 11x7 cms, cystic in consistency and with signs of local inflammation. The clinical possibilities of soft tissue sarcoma and infected hematoma of muscle were kept. Ultrasound examination revealed multiple well-defined rounded cystic structures with honeycomb appearance in muscle planes of right thigh, suggestive of hydatid cyst. Serology for hydatid cyst was positive, confirming the diagnosis of echinococcosis. Chest X-ray and abdominal ultrasound ruled out the presence of hydatid cyst of lungs and abdominal viscera respectively. Thus, the diagnosis of infected primary echinococcosis of right thigh was made.
The child was given a course of antibiotics and antihelminthic treatment (Albendazole). In three weeks time, the signs of inflammation resolved and the swelling became half the initial size. Thereafter, the cyst was widely excised along with pericyst from the right quadriceps femoris. It was densely adherent to the surrounding muscle. The wound was irrigated with povidone iodine solution and closed over a negative suction drain. On opening the cyst, it contained multiple small fluid filled daughter cysts along with pus flakes Figure1. The pus was sterile on culture and sensitivity probably due to prior course of antibiotics. Postoperative period was uneventful. The diagnosis of hydatid cyst was confirmed on histopathology. The child was given antihelminthic treatment for another six weeks and she is disease free during one and half year of follow-up.
Discussion
Echinococcosis is an endemic surgical problem in the mediterranean countries. Hydatid cyst is the larval form of Echinococcus granulosus . Although it can involve any part of the body, the most commonly affected organs are liver (75 %), lungs (15.4 %) and spleen (5.1%).[1] The other rare sites reported to be involved by hydatid cyst are peritoneal cavity, pancreas, thyroid glands, breast, gall-bladder, thigh, kidney, brain, supraclavicular region, pericardium, diaphragm and pleural cavity.[1],[2] In a series of 272 cases of hydatid cyst, thigh is reported to be involved in only 0.37 % cases.[1] Although hydatid disease is not uncommon in India, there are very few reports of thigh involvement.[3],[4],[5] The occurrence of hydatid cyst thigh is still rare in children for unknown reasons.[6],[7] Infection in the hydatid cyst of thigh has not been reported in the available literature. In present case, needle aspiration of the cyst was probably responsible for infection in the cyst.
Primary hydatidosis of thigh muscles being very rare, can cause variety of diagnostic problems. The important differential diagnoses are soft tissue tumor, traumatic and developmental lesions.[8],[9]
Preoperative diagnosis is mandatory in order to prevent any rupture of the cyst during surgery so as to avoid anaphylactic shock and local recurrence.[10] Ultrasound, CT scan and MRI are very helpful in making preoperative diagnosis. "Water lily sign" has been described recently on MR imaging of intramuscular hydatid cyst of thigh, which is almost confirmatory of the diagnosis.[11] Although fine needle aspiration cytology (FNAC) should be discouraged for the diagnosis of hydatid cyst, it has been reported to be useful sometimes when serology is negative.[5],[9]
Since there is no effective medical treatment, surgery is the treatment of choice. Surgical treatment follows the principles of malignant tumor namely wide surgical excision in form of total pericystectomy so as to avoid local spillage and recurrence.[7] Combination adjunctive chemotherapy with antihelminthics is recommended to cover the risk of dissemination during initial exploration. Medical treatment should precede and follow the surgical treatment[2]. Hydatid cyst of thigh has been reported to be treated with hypertonic saline solution and reaspiration.[12] Since relapse is a possibility, a long-term follow up of the patient including clinical examination and serological investigation is advisable.
In conclusion, although rarely recorded in India, cystic hydatid disease should be kept in the list of differential diagnosis of any slow growing cystic mass of thigh so as to avoid the complications of infection, anaphylactic reaction and recurrence due to improper intervention.
References
1. Safioleas M, Misiakos EP, Kakisis J, Manti C, Papachristodoulou A, Lambrou P, Tsinari KK, Skalkeas G. Surgical treatment of human echinococcosis. Int Surg 2000; 85(4): 358-365.
2. Abu-Eshy SA. Some rare presentations of hydatid cyst (Echinococcus granulosus). J R Coll Surg Edinb 1998; 43(5): 347-352.
3. Singh CM. Hydatid cyst of adductor longus. J Indian Med Assoc 1990; 88(10): 288.
4. Chatterjee H, Colaco RJ, Shanmugam M, Subramaniam S, Sen SB. Hydatid cyst in rare sites: kidney, retroperitoneum and thigh. Indian J Med Sci 1973; 27(1): 20-33.
5. Kapila K, Verma K. Aspiration cytology diagnosis of echinococcosis. Diagn Cytopathol 1990; 6(5): 301-303.
6. Keskin D, Ezirmik N, Karsan O, Gursan N. Primary hydatidosis of the gracilis muscle in a girl. J Int Med Res 2002; 30(4): 449-451.
7. Dudkiewicz I, Salai M, Apter S. Hydatid cyst presenting as a soft-tissue thigh mass in a child. Arch Orthop Trauma Surg 1999; 119(7-8): 474-475.
8. Luhr T, Junginger T. Muscular echinococcal cysts. A rare differential diagnosis of a soft tissue swelling. Chirurg 1995; 66(12): 1275-1276.
9. Giuffre G, Mondello P, Inferrera A, Furchi A, Gentile HM, Speciale G. Unexpected cytological diagnosis of two cases of echinococcosis. Pathologica 1993; 85(1100): 747-753.
10. Fikry T, Harfaoui A, Sibai H, Zryouil B. Primary muscular echinococcosis. Apropos of 2 cases. J Chir Paris 1997; 134(7-8): 325-328.
11. Comert RB, Aydingoz U, Ucaner A, Arikan M. Water-lily sign on MR. Imaging of primary intramuscular hydatidosis of sartorius muscle. Skeletal Radiol 2003; 32(7): 420-423.
12. Belfiore G, Cioffi A, Marano I, Della Noce M. Report of a case of hydatid cyst of the thigh treated with hypertonic saline solution and reaspiration. Radiol Med (Torino) 1996; 92(6): 792-793.(Marwah Sanjay, Subramania)
2 Department of Pathology, Pt. B.D. Sharma PGIMS, Rohtak, India
3 Department of Pediatric Surgery, Pt. B.D. Sharma PGIMS, Rohtak, India
Abstract
An unusual case of infected primary echinococcosis involving quadriceps muscles of the thigh and presenting as a cystic mass in a child is being reported. The diagnosis was made preoperatively with ultrasound examination and serology. After a cover of medical treatment the cyst was excised. Various clinical possibilities and management strategies are discussed.
Keywords: Echinococcosis; Primary intramuscular; Infected
Echinococcosis or hydatid disease may affect several organs in the body and thus represents a major challenge for the surgeons. It is rare to see echinococcosis of thigh muscles in children, like in adults cysts are seen in the liver and lungs. The causes for primary muscular localization of the disease are unknown. Preoperative diagnosis is important for proper management and to avoid the long term complication of recurrence.
Case Report
An eleven-year-old girl presented with a progressively increasing one-year-old swelling in the right thigh, following a doubtful history of trauma. Ten months prior, there was history of needle aspiration of thin fluid from the swelling and it subsided thereafter. The swelling recurred after a few months and became painful. On examination, there was a diffuse, intramuscular swelling occupying the anterior region of right thigh. It measured 11x7 cms, cystic in consistency and with signs of local inflammation. The clinical possibilities of soft tissue sarcoma and infected hematoma of muscle were kept. Ultrasound examination revealed multiple well-defined rounded cystic structures with honeycomb appearance in muscle planes of right thigh, suggestive of hydatid cyst. Serology for hydatid cyst was positive, confirming the diagnosis of echinococcosis. Chest X-ray and abdominal ultrasound ruled out the presence of hydatid cyst of lungs and abdominal viscera respectively. Thus, the diagnosis of infected primary echinococcosis of right thigh was made.
The child was given a course of antibiotics and antihelminthic treatment (Albendazole). In three weeks time, the signs of inflammation resolved and the swelling became half the initial size. Thereafter, the cyst was widely excised along with pericyst from the right quadriceps femoris. It was densely adherent to the surrounding muscle. The wound was irrigated with povidone iodine solution and closed over a negative suction drain. On opening the cyst, it contained multiple small fluid filled daughter cysts along with pus flakes Figure1. The pus was sterile on culture and sensitivity probably due to prior course of antibiotics. Postoperative period was uneventful. The diagnosis of hydatid cyst was confirmed on histopathology. The child was given antihelminthic treatment for another six weeks and she is disease free during one and half year of follow-up.
Discussion
Echinococcosis is an endemic surgical problem in the mediterranean countries. Hydatid cyst is the larval form of Echinococcus granulosus . Although it can involve any part of the body, the most commonly affected organs are liver (75 %), lungs (15.4 %) and spleen (5.1%).[1] The other rare sites reported to be involved by hydatid cyst are peritoneal cavity, pancreas, thyroid glands, breast, gall-bladder, thigh, kidney, brain, supraclavicular region, pericardium, diaphragm and pleural cavity.[1],[2] In a series of 272 cases of hydatid cyst, thigh is reported to be involved in only 0.37 % cases.[1] Although hydatid disease is not uncommon in India, there are very few reports of thigh involvement.[3],[4],[5] The occurrence of hydatid cyst thigh is still rare in children for unknown reasons.[6],[7] Infection in the hydatid cyst of thigh has not been reported in the available literature. In present case, needle aspiration of the cyst was probably responsible for infection in the cyst.
Primary hydatidosis of thigh muscles being very rare, can cause variety of diagnostic problems. The important differential diagnoses are soft tissue tumor, traumatic and developmental lesions.[8],[9]
Preoperative diagnosis is mandatory in order to prevent any rupture of the cyst during surgery so as to avoid anaphylactic shock and local recurrence.[10] Ultrasound, CT scan and MRI are very helpful in making preoperative diagnosis. "Water lily sign" has been described recently on MR imaging of intramuscular hydatid cyst of thigh, which is almost confirmatory of the diagnosis.[11] Although fine needle aspiration cytology (FNAC) should be discouraged for the diagnosis of hydatid cyst, it has been reported to be useful sometimes when serology is negative.[5],[9]
Since there is no effective medical treatment, surgery is the treatment of choice. Surgical treatment follows the principles of malignant tumor namely wide surgical excision in form of total pericystectomy so as to avoid local spillage and recurrence.[7] Combination adjunctive chemotherapy with antihelminthics is recommended to cover the risk of dissemination during initial exploration. Medical treatment should precede and follow the surgical treatment[2]. Hydatid cyst of thigh has been reported to be treated with hypertonic saline solution and reaspiration.[12] Since relapse is a possibility, a long-term follow up of the patient including clinical examination and serological investigation is advisable.
In conclusion, although rarely recorded in India, cystic hydatid disease should be kept in the list of differential diagnosis of any slow growing cystic mass of thigh so as to avoid the complications of infection, anaphylactic reaction and recurrence due to improper intervention.
References
1. Safioleas M, Misiakos EP, Kakisis J, Manti C, Papachristodoulou A, Lambrou P, Tsinari KK, Skalkeas G. Surgical treatment of human echinococcosis. Int Surg 2000; 85(4): 358-365.
2. Abu-Eshy SA. Some rare presentations of hydatid cyst (Echinococcus granulosus). J R Coll Surg Edinb 1998; 43(5): 347-352.
3. Singh CM. Hydatid cyst of adductor longus. J Indian Med Assoc 1990; 88(10): 288.
4. Chatterjee H, Colaco RJ, Shanmugam M, Subramaniam S, Sen SB. Hydatid cyst in rare sites: kidney, retroperitoneum and thigh. Indian J Med Sci 1973; 27(1): 20-33.
5. Kapila K, Verma K. Aspiration cytology diagnosis of echinococcosis. Diagn Cytopathol 1990; 6(5): 301-303.
6. Keskin D, Ezirmik N, Karsan O, Gursan N. Primary hydatidosis of the gracilis muscle in a girl. J Int Med Res 2002; 30(4): 449-451.
7. Dudkiewicz I, Salai M, Apter S. Hydatid cyst presenting as a soft-tissue thigh mass in a child. Arch Orthop Trauma Surg 1999; 119(7-8): 474-475.
8. Luhr T, Junginger T. Muscular echinococcal cysts. A rare differential diagnosis of a soft tissue swelling. Chirurg 1995; 66(12): 1275-1276.
9. Giuffre G, Mondello P, Inferrera A, Furchi A, Gentile HM, Speciale G. Unexpected cytological diagnosis of two cases of echinococcosis. Pathologica 1993; 85(1100): 747-753.
10. Fikry T, Harfaoui A, Sibai H, Zryouil B. Primary muscular echinococcosis. Apropos of 2 cases. J Chir Paris 1997; 134(7-8): 325-328.
11. Comert RB, Aydingoz U, Ucaner A, Arikan M. Water-lily sign on MR. Imaging of primary intramuscular hydatidosis of sartorius muscle. Skeletal Radiol 2003; 32(7): 420-423.
12. Belfiore G, Cioffi A, Marano I, Della Noce M. Report of a case of hydatid cyst of the thigh treated with hypertonic saline solution and reaspiration. Radiol Med (Torino) 1996; 92(6): 792-793.(Marwah Sanjay, Subramania)