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Muscular cystic hydatidosis: case report.

Vicidomini S, Cancrini G, Gabrielli S, Naspetti R, Bartoloni A - BMC Infect. Dis. (2007)

Bottom Line: The patient, 34 years old, was admitted to the Department of Infectious and Tropical Diseases for ultrasonographic detection, with successive confirmation by magnetic resonance imaging, of an ovular mass (13 x 8 cm) in the big adductor of the left thigh, cyst-like, and containing several small cystic formations.Any post-surgery complications was observed during 6 following months.The diagnosis should be achieved by taking into consideration the clinical aspects, the epidemiology of the disease, the imaging and immunological tests but, as demonstrated in this case, without neglecting the numerous possibilities offered by new serological devices and modern day molecular biology techniques.

View Article: PubMed Central - HTML - PubMed

Affiliation: Malattie Infettive e Tropicali, Azienda Ospedaliero-Universitaria Careggi, Florence, Italy.svicidomini@hotmail.com <svicidomini@hotmail.com>

ABSTRACT

Background: Hydatidosis is a zoonosis caused by Echinococcus granulosus, and ingesting eggs released through the faeces from infected dogs infects humans. The location of the hydatid cysts is mostly hepatic and/or pulmonary, whereas musculoskeletal hydatidosis is very rare.

Case presentation: We report an unusual case of primary muscular hydatidosis in proximity of the big adductor in a young Sicilian man. The patient, 34 years old, was admitted to the Department of Infectious and Tropical Diseases for ultrasonographic detection, with successive confirmation by magnetic resonance imaging, of an ovular mass (13 x 8 cm) in the big adductor of the left thigh, cyst-like, and containing several small cystic formations. Serological tests for hydatidosis gave negative results. A second drawing of blood was done 10 days after the first one and showed an increase in the antibody titer for hydatidosis. The patient was submitted to surgical excision of the lesion with perioperatory prophylaxis with albendazole. The histopathological examination of the bioptic material was not diriment in the diagnosis, therefore further tests were performed: additional serological tests for hydatidosis for the evaluation of IgE and IgG serotype (Western Blot and REAST), and molecular analysis of the excised material. These more specific serological tests gave positive results for hydatidosis, and the sequencing of the polymerase chain reaction products from the cyst evidenced E. granulosus DNA, genotype G1. Any post-surgery complications was observed during 6 following months.

Conclusion: Cystic hydatidosis should always be considered in the differential diagnosis of any cystic mass, regardless of its location, also in epidemiological contests less suggestive of the disease. The diagnosis should be achieved by taking into consideration the clinical aspects, the epidemiology of the disease, the imaging and immunological tests but, as demonstrated in this case, without neglecting the numerous possibilities offered by new serological devices and modern day molecular biology techniques.

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MRI of the left thigh, sequence T1-weighted: a) precontrast image: "ovular mass in the adductor magnus muscle, with a cystic aspect, dimensions 13 × 8 cm containing numerous cystic formations with regular outlines; b) after contrast image: "the lesion appears surrounded by a thin wall homogeneously impregnated after the injection of paramagnetic contrast medium while there is no contrastographic enhancement of the content of the lesion".
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Figure 1: MRI of the left thigh, sequence T1-weighted: a) precontrast image: "ovular mass in the adductor magnus muscle, with a cystic aspect, dimensions 13 × 8 cm containing numerous cystic formations with regular outlines; b) after contrast image: "the lesion appears surrounded by a thin wall homogeneously impregnated after the injection of paramagnetic contrast medium while there is no contrastographic enhancement of the content of the lesion".

Mentions: The patient, C. L., a 34 year old male, was a bricklayer born in Sicily (Southern Italy), but had lived in the province of Florence (Tuscany region, Central Italy) for the past 10 years. He was admitted in our ward following the ultrasonographic detection of a cyst-like ovular mass (13 × 8 cm) which contained several small cystic formations. The cyst was in the adductor magnus muscle of the left thigh, and it was later confirmed by magnetic resonance imaging (MRI) (Figure 1). The patient had been complaining for approximately one month about a painful mass growing on his left thigh. Therefore, his physician prescribed imaging examinations, and eventually referred him to us. Our physical exam revealed a palpable mass with hard consistency, and no signs of erythema. The patient reported neither fever nor systemic symptomatology. The blood cell count was normal except for a relatively slight hypereosinophilia (white blood cells 6700/mmc, eosinophils 8.9%). Abdominal ultrasonography and chest X-ray were normal, and the serological tests for hydatidosis were negative (Echinococcosis IHA, Fumouze Diagnostics, Levallois Perret, France: 1:80, n.v. <1:160; Echinococcus granulosus IFA, Bios GmbH Munchen, Germany: negative). The same tests were repeated 10 days later and the results showed a weak increase in the antibody titre (IHA 1:320; IFA 1:40, n.v. <40). The patient was evaluated for, and then submitted to the surgical ablation of the lesion, while receiving chemoprophylaxis with albendazole 400 mg bid. Ten days after the surgical excision the patient stopped the perioperative prophylaxis. At a first glance, the excised material resembled a big cyst containing more than 60 translucent daughter cysts immersed in a clear liquid (Figure 2). The subsequent histopathological examination showed "an inflammatory tissue reaction surrounding a parasite-like cyst". Additional serological tests and bio-molecular diagnostics were then used to further examine the two available serum samples and the excised material respectively, with the aim to define the nature of the lesion. Enzyme immunoassay for specific IgG (EIA Echinococcus Ab, Cypress Diagnostics, Langdorp, Belgium) and reversed-enzyme-allergo-sorbent-test for specific IgE (REAST Allergyzen IgE, ZenTech, Angleur, Belgium) resulted positive only on the second blood sample. Whereas the Western Blot assay (WB), for specific E. granulosus proteins, corresponding to the band of 7 and 26–28 Kda (WB Echinococcus IgG, LDBIO Diagnostics, Lyon, France), resulted positive on both serum samples. As for the bio-molecular analysis, genomic DNA was extracted (Wizard SV Genomic DNA Purification kit, Promega, USA) from the cyst wall and from its liquid. A polymerase chain reaction (PCR) protocol was applied which amplified a 373 bp fragment of the mitochondrial 12S rRNA gene common to almost 12 Cestoda species, E. granulosus included [16]. Amplification products were gel-purified (Immolase DNA Purification Kit, Bioline, UK), and sequenced (MWG-Biotech AG, Germany). The sequences obtained were assembled using the program MEGA 3.1 [17]. A comparison with sequences available in the GenBank was made to identify genetic similarities with already known cestode sequences. Molecular diagnostics identified E. granulosus DNA, genotype G1 (98% homology). All five serological tests of the 8-week-after-surgery follow-up exam were negative. The patient underwent follow-up visits within the next 12 months without any occurrence of post-surgery complications.


Muscular cystic hydatidosis: case report.

Vicidomini S, Cancrini G, Gabrielli S, Naspetti R, Bartoloni A - BMC Infect. Dis. (2007)

MRI of the left thigh, sequence T1-weighted: a) precontrast image: "ovular mass in the adductor magnus muscle, with a cystic aspect, dimensions 13 × 8 cm containing numerous cystic formations with regular outlines; b) after contrast image: "the lesion appears surrounded by a thin wall homogeneously impregnated after the injection of paramagnetic contrast medium while there is no contrastographic enhancement of the content of the lesion".
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC1851964&req=5

Figure 1: MRI of the left thigh, sequence T1-weighted: a) precontrast image: "ovular mass in the adductor magnus muscle, with a cystic aspect, dimensions 13 × 8 cm containing numerous cystic formations with regular outlines; b) after contrast image: "the lesion appears surrounded by a thin wall homogeneously impregnated after the injection of paramagnetic contrast medium while there is no contrastographic enhancement of the content of the lesion".
Mentions: The patient, C. L., a 34 year old male, was a bricklayer born in Sicily (Southern Italy), but had lived in the province of Florence (Tuscany region, Central Italy) for the past 10 years. He was admitted in our ward following the ultrasonographic detection of a cyst-like ovular mass (13 × 8 cm) which contained several small cystic formations. The cyst was in the adductor magnus muscle of the left thigh, and it was later confirmed by magnetic resonance imaging (MRI) (Figure 1). The patient had been complaining for approximately one month about a painful mass growing on his left thigh. Therefore, his physician prescribed imaging examinations, and eventually referred him to us. Our physical exam revealed a palpable mass with hard consistency, and no signs of erythema. The patient reported neither fever nor systemic symptomatology. The blood cell count was normal except for a relatively slight hypereosinophilia (white blood cells 6700/mmc, eosinophils 8.9%). Abdominal ultrasonography and chest X-ray were normal, and the serological tests for hydatidosis were negative (Echinococcosis IHA, Fumouze Diagnostics, Levallois Perret, France: 1:80, n.v. <1:160; Echinococcus granulosus IFA, Bios GmbH Munchen, Germany: negative). The same tests were repeated 10 days later and the results showed a weak increase in the antibody titre (IHA 1:320; IFA 1:40, n.v. <40). The patient was evaluated for, and then submitted to the surgical ablation of the lesion, while receiving chemoprophylaxis with albendazole 400 mg bid. Ten days after the surgical excision the patient stopped the perioperative prophylaxis. At a first glance, the excised material resembled a big cyst containing more than 60 translucent daughter cysts immersed in a clear liquid (Figure 2). The subsequent histopathological examination showed "an inflammatory tissue reaction surrounding a parasite-like cyst". Additional serological tests and bio-molecular diagnostics were then used to further examine the two available serum samples and the excised material respectively, with the aim to define the nature of the lesion. Enzyme immunoassay for specific IgG (EIA Echinococcus Ab, Cypress Diagnostics, Langdorp, Belgium) and reversed-enzyme-allergo-sorbent-test for specific IgE (REAST Allergyzen IgE, ZenTech, Angleur, Belgium) resulted positive only on the second blood sample. Whereas the Western Blot assay (WB), for specific E. granulosus proteins, corresponding to the band of 7 and 26–28 Kda (WB Echinococcus IgG, LDBIO Diagnostics, Lyon, France), resulted positive on both serum samples. As for the bio-molecular analysis, genomic DNA was extracted (Wizard SV Genomic DNA Purification kit, Promega, USA) from the cyst wall and from its liquid. A polymerase chain reaction (PCR) protocol was applied which amplified a 373 bp fragment of the mitochondrial 12S rRNA gene common to almost 12 Cestoda species, E. granulosus included [16]. Amplification products were gel-purified (Immolase DNA Purification Kit, Bioline, UK), and sequenced (MWG-Biotech AG, Germany). The sequences obtained were assembled using the program MEGA 3.1 [17]. A comparison with sequences available in the GenBank was made to identify genetic similarities with already known cestode sequences. Molecular diagnostics identified E. granulosus DNA, genotype G1 (98% homology). All five serological tests of the 8-week-after-surgery follow-up exam were negative. The patient underwent follow-up visits within the next 12 months without any occurrence of post-surgery complications.

Bottom Line: The patient, 34 years old, was admitted to the Department of Infectious and Tropical Diseases for ultrasonographic detection, with successive confirmation by magnetic resonance imaging, of an ovular mass (13 x 8 cm) in the big adductor of the left thigh, cyst-like, and containing several small cystic formations.Any post-surgery complications was observed during 6 following months.The diagnosis should be achieved by taking into consideration the clinical aspects, the epidemiology of the disease, the imaging and immunological tests but, as demonstrated in this case, without neglecting the numerous possibilities offered by new serological devices and modern day molecular biology techniques.

View Article: PubMed Central - HTML - PubMed

Affiliation: Malattie Infettive e Tropicali, Azienda Ospedaliero-Universitaria Careggi, Florence, Italy.svicidomini@hotmail.com <svicidomini@hotmail.com>

ABSTRACT

Background: Hydatidosis is a zoonosis caused by Echinococcus granulosus, and ingesting eggs released through the faeces from infected dogs infects humans. The location of the hydatid cysts is mostly hepatic and/or pulmonary, whereas musculoskeletal hydatidosis is very rare.

Case presentation: We report an unusual case of primary muscular hydatidosis in proximity of the big adductor in a young Sicilian man. The patient, 34 years old, was admitted to the Department of Infectious and Tropical Diseases for ultrasonographic detection, with successive confirmation by magnetic resonance imaging, of an ovular mass (13 x 8 cm) in the big adductor of the left thigh, cyst-like, and containing several small cystic formations. Serological tests for hydatidosis gave negative results. A second drawing of blood was done 10 days after the first one and showed an increase in the antibody titer for hydatidosis. The patient was submitted to surgical excision of the lesion with perioperatory prophylaxis with albendazole. The histopathological examination of the bioptic material was not diriment in the diagnosis, therefore further tests were performed: additional serological tests for hydatidosis for the evaluation of IgE and IgG serotype (Western Blot and REAST), and molecular analysis of the excised material. These more specific serological tests gave positive results for hydatidosis, and the sequencing of the polymerase chain reaction products from the cyst evidenced E. granulosus DNA, genotype G1. Any post-surgery complications was observed during 6 following months.

Conclusion: Cystic hydatidosis should always be considered in the differential diagnosis of any cystic mass, regardless of its location, also in epidemiological contests less suggestive of the disease. The diagnosis should be achieved by taking into consideration the clinical aspects, the epidemiology of the disease, the imaging and immunological tests but, as demonstrated in this case, without neglecting the numerous possibilities offered by new serological devices and modern day molecular biology techniques.

Show MeSH
Related in: MedlinePlus