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Table 1 Main characteristics of non-randomized controlled trials, prospective or retrospective case series included in the qualitative analysis

From: Medical treatment of cystic echinococcosis: systematic review and meta-analysis

Author/s Year (Ref.no.)

Location, Country

Study design (follow-up period)

Participants

Sample size(N)

No. cysts

Cyst location

Objective

Anthelminthic drugs

Results/Conclusions

Aktan AO, et al. 1996 [13]

Istanbul, Turkey

A non-randomized controlled trial

Adult patients

70

89

Liver

To evaluate the effect of preoperative ABZ* treatment (3-weeks) in two groups: 1st group (experimental group) ABZ 3 weeks before surgery, 2nd group (control group) surgery (no preoperative treatment).

ABZ

The ICP values of viable cysts in the 1st group were significantly lower (p < 0.05). The number of non-viable cysts was also significantly higher in the 1st group (p < 0.05). ABZ has proved to be effective in decreasing the viability of liver hydatid cysts when given for 3 weeks preoperatively.

Di Matteo G, et al. 1996 [15]

Rome, Italy

A prospective, descriptive, non-comparative study (1985–1992)

Adult patients (mean age, 42)

95

No data

Liver

To show that radical surgery is most effective when it is associated with medical therapy of benzoimidazole drugs (MBZ) pre- and post-operatively.

MBZ*

The most effective treatment for echinococcus cystic disease of the liver is radical surgery. Results are best when surgery is combined with medical therapy of benzoimidazole drugs (MBZ) given pre- and post-operatively.

Doğru D, et al. 2005 [16]

Ankara, Turkey

A retrospective study

Pediatric patients

82

102

Lung

To demonstrate the safety and efficacy of medical treatment.

MBZ vs ABZ

The cure and the failure rates were statistically insignificant in cysts treated with MBZ and ABZ; however statistically significantly more cysts were improved with ABZ. The results were statistically insignificant between continuous and cyclic ABZ treatment. There was a positive, weak and statistically significant correlation between the cyst size and treatment results. These results cannot recommend a standard treatment regimen as the duration of treatment should be individualized for each patient.

el-Mufti M, et al. 1993 [17]

Benghazi, Libya

A prospective, descriptive, non-comparative study

Adult patients

40

63

Multi-organ

To assess the effectiveness of ABZ before surgery.

ABZ

It is suggested that patients suffering from uncomplicated hydatid disease should be given the benefit of a trial course of ABZ therapy before surgery.

Ghoshal AG, et al. 2012 [18]

Kolkata, India

A retrospective study (5 years)

Adult patients

106

No data

Lung

To determine the presentation, treatment (ABZ and surgery) and outcome of hydatid disease of lung.

ABZ

Surgery is a safe and effective way of treatment for thoracic hydatid cyst along with perioperative ABZ therapy. There is a scope for chemotherapy with ABZ in inoperable cases.

Larrieu E, et al. 2004 [21]

Rio Negro, Argentina

A prospective cohort study (5–6 years)

Pediatric patients

5745

Exposed cohort = 4644

Unexposed cohort = 1101

No data

Abdominal

To evaluate the results of a program carried out in endemic areas of the Province of Río Negro, Argentina, during the years 1997–2002 in asymptomatic children, screnning.

ABZ

Treatment with ABZ confirmed its action in modifying the prognosis of CE, presenting positive effects in 76% of patients receiving the drug. None of the treated cases required surgery. The combination of ultrasonographic screening and ABZ treatment showed promising results.

Li T, et al. 2011 [22]

Sichuan, China

A prospective, descriptive, non-comparative study

Adult patients

49

No data

Abdominal

A post-treatment follow-up study was carried out to assess the effectiveness of community based use of cyclic ABZ treatment in Tibetan CE cases.

ABZ

Cyclic ABZ treatment proved to be effective in the great majority of CE, but periodic abdominal ultrasound examination was necessary to guide appropriate treatment. Serology with recombinant antigen B could provide additional limited information about the effectiveness of ABZ in CE cases. Oral ABZ for over 18 months was more likely to result in CE cure.

Mikić D, et al. 1998 [23]

Republic of Serbia

A retrospective study

Adult and pediatric patients (female age range, 9–83; males age range, 6–72)

119

No data

Liver

To value the efficacy of ABZ and surgery.

ABZ

Surgical removal of the cyst takes a leading place in the treatment of hepatic echinococcosis. However, in well-selected cases and in the patients with high surgical risk, anthelminthic therapy and PD of echinococcus cyst are of more significance.

Nahmias J, et al. 1994 [24]

Moztkin, Israel

A prospective, descriptive, non-comparative study (3–7 years)

Adult patients

68

No data

Multi-organ

To assess long-term efficacy of ABZ.

ABZ

Follow-up for 3–7 years showed that this treatment alone eradicated the cysts in many patients; in most of the remainder, disease progression stopped. No patient worsened but a recurrence occurred in two patients at about 56 months.

Perez Molina JA, et al. 2011 [10]

Madrid, Spain

A case series

Adult patients (age range, 27–68)

7

No data

Multi-organ

To describe the clinical effectiveness and tolerability of nitazoxanide, combined with ABZ, with or without PZQ*, in patients affected by disseminated chronic CE.

ABZ vs ABZ + PZQ

Nitazoxanide combination therapy seems to be active for disseminated CE affecting soft tissues, muscles, or viscera, and apparently it has no role in chronic and extensive bony lesions.

Redzić B, et al. 1995 [24]

Republic of Serbia

A prospective, descriptive non-comparative study (from 1989 to 1993)

Adult patients

73

No data

Liver

To value the efficacy of PZQ.

PZQ

The drug treatment was the therapy of choice in patients with Echinococcus granulosus. It should be given prophylactically, preoperatively, to sterilize the cyst and also as a curative treatment.

Salinas JL, et al. 2011 [26]

Lima, Perú

A retrospective study (from January 1997 to December 2007)

Adult patients (mean age at diagnosis, 51 ± 14)

27

No data

Liver

To ascertain factors associated with the success of ABZ in the treatment of non-complicated hepatic CE, and to establish the frequency of long-term worsening and recurrence of disease after treatment completion in Peru.

ABZ

Long-term hepatic CE treatment outcomes and the success rate of ABZ were modest (3 cycles are few and needed treatment 6–12 months). It’s necessary to investigate into alternate therapeutic strategies for this neglected disease.

Tarnovetchi C, et al. 2010 [28]

Romania

A retrospective study (2004–2009 and 2000–2009)

Pediatric patients (age range, 2–17)

111

No data

Abdominal

To value the efficacy of ABZ and surgery (Lagrot partial pericystectomy).

ABZ

The treatment includes both surgical and medical means. There is a relatively high rate of postoperative complications (although some of them being minor) in 31 patients.

Todorov T, et al. 1992 [29]

Sofia, Bulgaria

A prospective descriptive study

Adult and pediatric patients (age range, 6–70)

51 (28 MBZ, 23 ABZ)

No data

Multi-organ

To test the efficacy of MBZ and ABZ.

MBZ or ABZ

Treatment with MBZ was successful in 8 (28.6%), partially successful in 8 (28.6%) and unsuccessful in 12 (42.8%). Treatment with ABZ was successful in 10 (43.5%), partially successful in 10 (43.5%) and unsuccessful in 3 (13.0%).

Yasawy MI, et al. 1993 [30]

Riyah, Saudi Arabia

A case series

Adult patients

4

No data

Pelvic, abdominal and thoracic

To value the response to combined medical treatment (ABZ and PZQ).

ABZ plus PZQ vs ABZ

This preliminary report shows that the response to combined treatment is better and much quicker compared to ABZ alone.

Yilmaz Y, et al. 2006 [32]

Van, Turkey

A retrospective study (10 years)

Adult and pediatric patients

372 (of them, 8 urinary hydatid disease)

No data

Liver, spleen, brain and kidneys(7)-retrovesical area(1)

To discuss therapeutic options and treatment results according to current literature.

ABZ

Treated surgically (271 cases) and drained percutaneously (99 cases). Kidneys were removed totally (4 cases), cystectomy and omentoplasty was performed in one case. ABZ was administered to 192 patients.

  1. *ABZ Albendazole, PZQ Praziquantel, MBZ Mebendazol