Hydatid disease of bone with adjacent joint involvement. A radiological follow-up of 12 years

Osseous lesions have been reported in only 1-2% of patients with hydatid disease. Joint involvement is usually due to secondary extension from the adjacent bone, although primary hydatid synovitis after haematogenous spread of the infection can be seen. We present a long-term radiological follow-up (12 yr) in a patient who developed hydatid disease of the left pelvic and femoral bones with cartilage destruction of the ipsilateral hip joint. After a Girdlestone arthroplasty, she received mebendazole (3 g/day) for 10 yr and albendazole (400 mg/day) for 2 yr with radiological impairment of the lesions. Complete surgical excision is the treatment of choice for osseous hydatid disease. Isolated medical therapy with mebendazole or albendazole is not adequate for controlling the process, but it can be added to surgery or, as in our case, used like isolated therapy when complete excision is not possible.      

Colohepatic fistula due to hydatid disease

A patient with colohepatic fistula due to hydatid disease is reported. Only two similar cases have been described in the medical literature. This case illustrates that medical treatment with mebendazole is ineffective.     

Treatment of Liver Hydatid Disease with Mebendazole: A Prospective Study of Thirteen Cases

We have prospectively studied the evolution of 13 patients with liver hydatid cysts treated with mebendazole. Two patients also had peritoneal hydatid disease in addition to liver cysts. One of the two also had a retrovesical cyst. Liver cysts disappeared in three patients. In one, the liver cyst reappeared and in another a new cyst was observed after treatment was stopped. Therapy was most effective in young patients, in small cysts, in one case of peritoneal hydatid disease and in one case of a retrovesical cyst. Side effects warranted interruption of therapy in three cases, although we cannot be sure that they were due to mebendazole treatment. We recommend long-term patient follow-up once the cyst can no longer be identified by ultrasound.     

甲苯咪唑新剂型（微丸）治疗小鼠细粒棘球蚴的效果观察

通过体外和动物实验证实甲苯咪唑（Ｍｅｂｅｎｄａｚｏｌｅ ＭＢＺ）微丸的抗细粒棘球蚴的作用。在试管内观测不同浓度甲苯咪唑微丸对原头节的致死作用,将甲苯咪唑微丸制成１００ｍｇ／ｋｇ．５００ｍｇ／ｋｇ和２５ｍｇ／ｋｇ三个不同剂量悬液对感染细粒棘球蚴６个月的小鼠经口灌胃,每日一次,连续给药２０ｄ,停药７ｄ后剖检,以棘球蚴囊重抑制率、塌囊率和组织病理改变为指标,同法将甲苯唑片剂和原粉制成悬液按相同剂量给药,     

A clinical-radiological evaluation of benzimidazoles in the management of Echinococcus granulosus cysts.

Nine patients with complicated hydatid disease managed with surgery and mebendazole/albendazole are presented. Five patients received albendazole (1 treatment course) and 5 patients received mebendazole (3 had 2 treatment courses, 1 had a switch-over from mebendazole to albendazole). The mean durations of treatment and follow-up were respectively 7 +/- 2.5 months and 7 +/- 2.5 months (albendazole); 13 +/- 10 months and 29 +/- 31 months (mebendazole). A superior clinical and radiological response was seen in 1 patient with disseminated intra-abdominal disease on switching therapy from mebendazole to albendazole. Radiological improvement occurred in 3/5 courses of albendazole and in 2/8 courses of mebendazole. Clinical improvement occurred in 3/5 courses of albendazole and 0/8 courses of mebendazole. Radiological deterioration was demonstrated in 0/5 courses of albendazole and 2/8 courses of mebendazole. Although the impression was that albendazole was superior, good responses were also seen with mebendazole. The heterogeneity of the patients, their disease, short follow-up time, lack of more sensitive noninvasive assay techniques urges caution before firm conclusions can be drawn.     

Hepatotoxicity of mebendazole. Relationship with serum concentrations of the drug

We report the case of a 47-year-old female patient, suffering from multiple hydatid cysts of the liver, in whom hepatitis developed after mebendazole treatment. Clinical manifestations of hypersensitivity were absent. A correlation was found between serum mebendazole concentrations and the degree of cytolysis; this is compatible with a direct hepatotoxic effect of mebendazole.     

Consequences of continuous mebendazole therapy in alveolar hydatid disease--with a summary of a ten-year clinical trial

The assessment of a ten-year clinical trial of continuous therapy in eight patients revealed further evidence of a significant therapeutic effect of mebendazole on alveolar hydatid disease. Life-expectancy was increased when compared to untreated historical controls, especially in the patients over 55 years of age. All symptomatic patients showed subjective improvement. In four patients, three had a 50% or greater reduction in the diameter of massive hepatic lesions, and in the fourth, progressively enlarging metastases were arrested. Fall in the IHA titre suggested that the causative organism had been destroyed in two additional patients. Of greater significance was the absence of progression of the disease process as measured by changes in the size of the hepatic lesion or lack of development of distant metastases in patients under therapy. In contrast, progressive enlargement of hepatic lesions or the appearance of distant metastases were cardinal features of untreated cases (15 of the 16 cases followed). In vivo determination of viability of tissues of the larval Echinococcus multilocularis from patients receiving long-term therapy was considered important in evaluating efficacy of the drug. Such tissues, obtained by autopsy from two patients under continuous therapy for four and ten years, failed to proliferate when inoculated into rodents (red-backed voles), whereas similar inoculations from untreated patients or those receiving 15 months' or less of therapy brought about production of vesicles in rodents in eight of 11 tests (73%). These two deaths, unrelated to therapy, resulted from late fibrotic constriction of end-stage parasitic lesions about the portal vein and major bile ducts. The clinical findings in combination with negative in vivo tests and other data indicate that the mebendazole therapy significantly alters the clinical course of alveolar hydatid disease. The evidence strongly indicates that long-term therapy may eventually have a lethal effect on the larval cestode in advanced disease.     

Resolution of hydatid liver cyst by spontaneous rupture into the biliary tract

Among the complications of hydatid liver disease, spontaneous cyst rupture into the biliary tract is unusual, occurring in 3.2–17% of cases. Its endoscopic management has been reported rarely, and corresponding complete photodocumentation is unique. Such a case is described and comprehensively illustrated in a 48-year-old immunocompromised man, presenting with supper abdominal pain, obstructive jaundice, and fever. Impaction of hydated material into the common bile duct and the papilla of Vater was relieved endoscopically, and the patient was consecutively treated with two courses of mebendazole. This management resulted incomplete clinical resolution of hepatic hydatosis after 8 months of follow-up, Complications of overt cyst perforation may be allergic, obstructive, secondary infectious, or metastatic. Ultrasound and computed tomography are completely tools for diagnosis of hepatic echninococcosis, with endoscopic retrograde cholagiography being the “gold standard” in confirming rupture into the biliary system. Laboratory results are usually non-specific. While surgical excision is the treatment of choice, selected patients may primarily be managed endoscopically, followed by anthelminthic theraphy.     

Mebendazole.

The broad-spectrum of activity and safety of mebendazole remain, after 5 years of clinical experience, unique features of this anthelmintic. Through microtubular destruction, mebendazole kills helminths by inhibiting glucose uptake into susceptible parasites. The drug's poor absorption does not appear to affect clinical efficacy except, perhaps, in the treatment of systemic helminth infections. Mebendazole is generally considered the drug of choice for trichuriasis and has therapeutic advantages over other anthelmintics in the treatment of enterobiasis and hookworm infections. Although mebendazole is an effective agent against ascariasis, there are preferable alternatives. Among its nonapproved uses, mebendazole shows great promise in the treatment of capillariasis and hydatid disease. Further investigation is needed to establish its role in the treatment of taeniasis, Hymenolepsis nana, strongyloidiasis, trichinosis, and Dipetalonema perstans. Undoubtedly, mebendazole will find its greatest value in the treatment of patients with multiple helminth infections.     

Experience in the chemotherapy of severe,inoperable echinococcosis in man

Summary The effect of albendazole and mebendazole was studied in 25 patients with severe, multiple, inoperable echinococcosis. Eleven patients were treated with albendazole 10 mg/kg/day for four courses of 30 days with 15-day drug-free intervals and 14 with mebendazole 50–70 mg/kg/day continuously for between 6 and 24 months. The determination of cyst response was based on the changes in cyst appearance as seen by radiography, ultrasound and computed tomography. Albendazole and mebendazole were found to have favourable effects in patients with severe, inoperable hydatid disease, although the degree of response varied. Treatment with albendazole was successful in seven cases, partially successful in two, and failure was noted in the other two patients. In patients treated with mebendazole, success was established in three, partial success in five and failure in six cases. The differences in response were mainly associated with cyst condition and the drug used. In some cases long-term therapy and retreatment should be recommended.

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Erfahrung mit der Chemotherapie der schweren, inoperablen Echinokokkose des Menschen

Zusammenfassung Die Wirkung von Albendazol und Mebendazol wurde bei 25 Patienten mit schwerer, multipler und inoperabler Echinokokkose untersucht. Elf Patienten wurden mit Albendazol in der Dosierung von 10 mg/kg Körpergewicht/die über 4 Kurse von 30 Tagen mit 15 Tagen zwischen Kursen und 14 Patienten mit Mebendazol 50–70 mg/kg/Körpergewicht/die über 6–24 Monate behandelt. Die Beurteilung der Therapiewirkung wurde aufgrund der Änderungen der röntgenologischen, sonographischen und computertomographischen Befunde durchgeführt. Es wurde festgestellt, daß Albendazol und Mebendazol eine günstige therapeutische Wirkung bei Patienten mit schwerer, inoperabler Echinokokkose haben. Die Therapie mit Albendazol war bei sieben Patienten erfolgreich, erzielte Teilerfolge bei zwei Patienten und war erfolglos bei zwei weiteren Patienten. Mit Mebendazol wurde bei drei Patienten ein Therapieerfolg, bei fünf ein Teilerfolg und in sieben Fällen ein Mißerfolg beobachtet. Die Unterschiede in den therapeutischen Ergebnissen wurden hauptsächlich mit dem Zustand der Zysten, aber auch mit dem benutzten Medikament in Verbindung gebracht. Bei manchen Patienten war eine längere und wiederholte Therapie nötig.

     

Effect of mebendazole on human cystic echinococcosis: the role of dosage and treatment duration

Fifty-three patients with single, multiple and/or multi-organ hydatid cysts were treated with mebendazole, in varying dosages (30-70 mg/kg.day) and over varying periods (6-24 months). Treatment failure was recorded if the mebendazole had no apparent effect on cyst morphology (monitored by radiology, ultrasonography and computed tomography) or only a parasitostatic effect (characterized by mixed and transitory, hypo- and hyper-echoic changes in the lesions). Cure was indicated by a parasitocidal effect (characterized by a totally echogenic picture, increased density of the cyst contents, reduction in the size or complete disappearance of the cysts, complete detachment of the endocysts and/or calcification of the cyst wall). Treatment failure was seen in about 40% of the patients and cure in about 38%; the other 23% had an intermediate result considered as an improvement. Cure rates increased both with dosage and duration, daily dosages of 60-70, 50 and 30-40 mg/kg curing 48%, 33% and 25%, respectively. The pre-treatment condition of any cysts should be taken into consideration when determining dosage and treatment duration.     

Changing echocardiographic features of a hydatid cyst of the heart.

A 24 year old woman with pulmonary embolism and a past history of echinococcal disease underwent echocardiography which detected two cysts in the right ventricle which became more solid after treatment with mebendazole. The surgical and pathological findings confirmed the presence of the two cysts and the hydatid nature of the lesion.     

Changing echocardiographic features of a hydatid cyst of the heart

A 24 year old woman with pulmonary embolism and a past history of echinococcal disease underwent echocardiography which detected two cysts in the right ventricle which became more solid after treatment with mebendazole. The surgical and pathological findings confirmed the presence of the two cysts and the hydatid nature of the lesion.     

Marrow aplasia during high dose mebendazole treatment

A patient with chronic liver disease was treated with large doses of mebendazole for a hepatic hydatid cyst. Eighteen days after beginning treatment he developed marrow aplasia which reverted to normal after the drug was stopped. This is the marrow aplasia which reverted to normal after the drug was stopped. This is the sixth patient described as developing marrow aplasia when treated with large doses of mebendazole. We suggest that the aplasia is related to the dose of the drug, and that the patient's chronic liver disease was an important factor in its genesis. Patients treated with large doses of mebendazole should have their blood counts monitored during treatment.     

The types and timing of the degenerative changes seen in the cysts during and after benzimidazole treatment of cystic echinococcosis

In a large-scale Bulgarian study, 122 patients with abdominal and/or lung echinococcosis were randomly selected for treatment with albendazole or mebendazole. The main aims were to evaluate the effect of each drug on the hydatid cysts, to follow the changes in cyst morphology during and after treatment, and to determine how quickly each drug produced the first, detectable, degenerative changes in the cysts. Follow-up was based on periodic ultrasonography, chest radiography and computed tomography. The abdominal cysts were categorised as small (<5 cm in diameter) or large. As albendazole treatment had effects that were almost identical to those of mebendazole treatment, the results for the two drugs were combined. In the lungs and, particularly, in the abdomen, the size of the cysts being treated influenced the character and timing of the degenerative changes seen in them.The initial change seen in each abdominal cyst was detachment of the endocyst, which occurred 1-3 months (small cysts) or 2-5 months (large cysts) after the initiation of treatment (P<0.05). The abdominal cysts then developed a hyper-echoic/hyper-dense appearance, became smaller, and finally disappeared 3.3-9.3 months (small cysts) or 5.6-13.9 months (large cysts) after treatment began (P<0.05).The first degenerative change noted in the lung cysts was cyst rupture, which occurred as early as day 10 of therapy but was generally observed 1 or 2 months after treatment began. After their complete evacuation, the ruptured lung cysts shrank and became deformed, some disappearing within 5-9 months of the initiation of treatment. The degenerative changes recorded, which began significantly earlier in the lung cysts than in abdominal cysts, indicate serious damage to the cysts and the parasiticidal, curative effect of each of the two benzimidazoles employed.     

Serologic tests for diagnosis and post-treatment evaluation of patients with alveolar hydatid disease (Echinococcus multilocularis)

The indirect hemagglutination (IHA) and immunoelectrophoresis (IEP) tests were used for diagnosis and follow-up evaluation of 17 patients with alveolar hydatid disease caused by Echinococcus multilocularis. Follow-up periods ranged from 2 to 22 years. At the time of diagnosis 16 (94%) patients' sera gave IHA titers greater than or equal to 1:128. Serum specimens from 13 patients were examined by IEP; nine (69%) revealed the arc 5, and three of the four arc 5-negative sera revealed one or more non-characterized bands. Titers declined markedly during the first year following radical surgical resection of the larval lesions; in three cases clinical evidence of recurrence was preceded by rising serologic titers. Antibody has persisted at high levels in non-resected patients treated continuously with high doses of mebendazole.     

Videolaparoscopic treatment of hepatic hydatid cyst

BACKGROUND/AIMS: Unroofing, cystopericystectomy, or cystic evaluation and omentoplasty have been used in videolaparoscopic treatment in hepatic hydatidosis since 1992. Currently it is shown that videolaparoscopic treatment has been carried out successfully in selected cases. METHODOLOGY: Fifteen hepatic hydatid cysts in 12 cases were treated by videolaparoscopic methods. Formerly in the 5 cases, the cysts were aspirated with a needle designed for a 5-mm trocar, leaving a cystic cavity that was tension-free, then scolicidal solution was injected and aspirated. In the last 7 patients an aspirator-grinder apparatus was used. Intraoperative ultrasonography was applied in all patients. RESULTS: All the cysts were treated by drainage and omentoplasty. In one case cystic cavity infection was diagnosed in the 2nd postoperative month (morbidity rate 8.33%). Another patient died due to cerebral hydatid cyst and multiple organ failure after the postoperative first month (mortality rate 8.33%). Operative mortality was not seen. CONCLUSIONS: Videolaparoscopic treatments of hepatic hydatid cysts may be carried out successfully in selected cases.     

Surgical treatment of liver hydatid cyst before autologous stem cell transplantation; presentation of two cases with literature review

Hydatid cyst, caused by Echinococcus granulosus, is a parasitic disease which is frequently seen in endemic areas. The disease can be seen as an isolated entity or simultaneously with other diseases. Treatment of hydatid cyst in patients with malignancy as well as in patients in need of transplantation is still unclear. Although there is information in the form of case reports, a large number of patient data are needed to create a consensus on the management of these patients. We reviewed two cases with the need of autologuous stem cell transplantation which underwent liver hydatic cystectomy before transplantation.     

Hydatid cyst of the liver: a case report and literature review

Taiwan is nearly free from hydatid disease. We report a case of hydatid cyst of the liver in a 37-year-old man who originally lived in India and had migrated to Taiwan 2 years earlier. He presented with right upper quadrant pain and intermittent low-grade fever. Both sonography and computed tomography (CT) demonstrated a cystic lesion with vesicles at its periphery in segments 6 and 7 of the liver. A hydatid cyst was diagnosed. The patient underwent radical excision of the cyst with total removal without opening the wall. He also received pre- and postoperative oral mebendazole. Pathology showed a hydatid cyst consisting of three layers: the inner single nucleated geminal layer, the middle acellular laminated layer, and the outer pericyst originating from inflammatory and hepatic cells. This case highlights that accurate preoperative diagnosis of hydatid disease can be made from personal history, typical sonography and CT study in non-endemic areas.