Introduction. Cystic echinococcosis (CE) is a neglected zoonotic disease caused by Echinococcus granulosus with major health and economic burden. The information on how the community members perceive the disease is crucial in order to recommend an effective preventive and control plan. The current study is aimed at finding out knowledge, attitude, and practices (KAP) of surgically operated cases of hydatid cyst in educational hospitals of Shiraz in Fars Province, southern Iran, toward the CE. Methods. A cross-sectional survey was conducted among 180 CE patients who underwent surgery due to CE. Using a well-designed questionnaire, a telephone-based survey was carried out to collect the data. The contents of the questionnaire included basic personal information and questions related to the participants’ knowledge, attitude, and behavioral patterns toward CE. Univariate and then multivariate linear regression analyses were used to identify factors associated with the KAP. Unstandardized regression coefficients (β) and odds ratios (ORs) and their 95% confidence intervals (CIs) were used to quantify the associations between variables and KAP. Results. A total of 180 CE patients with a mean age of 35.64 (±17.59) years were recruited. The mean score of participant’s knowledge was 8.7 (
SD
=
2.8
, range: 0-17), whereas these scores were 1.3 (
SD
=
0.7
, range 0-2) for attitude and 1.2 (
SD
=
1.0
, range 0-4) for practice. Findings of the study demonstrated that 20 of the participants (11.1%) had good knowledge towards CE, 82 (45.6%) demonstrated a positive attitude, and 57 (47.5%) without having dogs demonstrated a good practice towards CE (
score
=
2
/
2
), while from 60 dog owners, only 7 (11.6%) participants demonstrated good practice (score 3 and 4/4). Factors that were associated with knowledge were age (
OR
=
−
0.49
,
P
value = 0.001) and educational level (
OR
=
0.668
,
P
value = 0.001), where higher age was associated with lower knowledge and also higher educational levels were associated with higher knowledge regarding hydatid cyst. Regarding attitude, only living location had a significant association with participants’ attitude where those who were living in urban areas demonstrating a more positive attitude towards CE (
OR
=
0.261
,
P
value = 0.022). The practice of the participants was grouped into dog owners and participants with no dogs, in which among participants who did not own a dog, those living in urban areas demonstrating weaker practice towards CE (
OR
=
−
0.491
,
P
value>0.001). Moreover, a lack of counseling of patients after the surgery on how to prevent reinfection was noticed. Conclusion. Findings of the study revealed that the CE patients in southern Iran had poor knowledge and attitude toward the disease, and their practice may help in maintaining the disease in the community. Health education is highly needed to increase community awareness and to prevent and control this neglected parasitic infection in the area.