Hydatid Cyst of The Orbit: A Systematic Review with Meta-Data

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Abstarct Introduction Orbital hydatid cysts (HCs) constitute less than 1% of all cases of hydatidosis, yet their occurrence is often linked to severe visual complications. This study presents a systematic review of reported cases of orbital HCs. Methods A systematic review of the published studies of orbital HCs was conducted, the studies that met the following criteria were included: 1) The presence of the infection was confirmed through diagnostic methods, surgical findings, or histopathology. 2) The study provided a detailed case presentation. Results Thirty-two studies (56 cases) met the inclusion criteria. Ten patients were from Afghanistan (17.9%). There was no gender predilection, the distribution was almost equal. The ages ranged from three to 80 years old. The most common symptoms that the patients presented with were proptosis of the affected eye (98.2%) and visual impairment (64.3%). The therapeutic approach of orbital HC was primarily surgical removal of the cyst accompanied by anthelmintic drugs in 41 (73.2%) cases. Concurrent HC was reported in two cases (3.6%), and recurrence with subsequent recovery was reported in four (7.1%) cases. Conclusion Orbital HC is a rare condition, primarily diagnosed using MRI, with surgery as the definitive treatment. Concurrent hydatidosis increases the risk of recurrence, requiring thorough and ongoing follow-up. Introduction Hydatidosis or hydatid cyst (HC) is a commonly recognized zoonotic disease caused by the larval form of the tapeworm Echinococcus granulosus. Humans act as intermediate hosts for this parasite, acquiring infection through direct contact with definitive hosts (e.g., sheep, goats, cattle, dogs) or consuming contaminated food or water. [1]. The global incidence of hydatidosis varies, with higher rates observed in regions where livestock farming is widespread. Key risk factors for contracting hydatidosis include close contact with dogs, livestock-related activities, and residence in areas where the disease is endemic. These cysts typically occur in the liver (50-70%) and lungs (20-30%). The global burden of HC is significant, with an estimated 2 to 3 million cases reported worldwide [2]. However, orbital HC is uncommon, representing less than 1% of all cases, accounting for 19.8% in endemic countries [3]. The World Health Organization (WHO) has classified Echinococcosis as one of the 20 neglected tropical diseases that pose significant public health concerns. To ensure consistent global monitoring, the WHO Informal Working Group on echinococcosis has categorized cysts of echinococcosis into five distinct types, grouped into three main categories. Specifically, CE1 and CE2 are indicative of active infection, CE3 represents an intermediate stage, while CE4 and CE5 are associated with inactive cysts [4]. In endemic regions, environmental and climatic conditions play a crucial role in the survival of parasite eggs and the living conditions of livestock and stray dogs. For example, Echinococcus granulosus eggs remain viable in water and damp sand for up to three weeks at 30°C, 4.5 weeks at 10–21°C, and 32 weeks at 6°C. They can also survive for several months in green pastures and gardens [4]. Although the WHO classifies hydatidosis as a neglected disease, it continues to be a significant public health concern due to its status as the second most impactful foodborne parasitic disease, its endemic presence in certain regions, and its potential to cause substantial morbidity. The WHO prioritizes the control and prevention of hydatidosis, particularly given its impact on human health, animals, and the food supply chain. Orbital HC, although rare, is often linked to severe visual complications. As of the date of the current review, the available literature on orbital HC primarily consists of case reports and case series, with no reviews currently available. This study aims to provide and analyze a collection of data through a systematic review and a meta-data presentation. Methods Study design This systematic review was conducted following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines [5]. Data sources and search strategy A systematic review of the published studies of orbital HCs was conducted using Google Scholar and PubMed. Boolean operators (OR/AND) were used to refine the results. The keywords that were used in the search included: (eye OR orbital OR intraorbital OR ocular) AND (hydatid OR echinococcosis OR hydatidosis). Eligibility criteria Studies in languages other than English, as well as those not related to humans, were excluded either before or during the initial screening process. All studies on orbital HCs that met the following criteria were included: 1) The presence of the infection was confirmed through diagnostic methods, surgical findings, or histopathology. 2) The case presentation was detailed in the study. Studies published in non-peer-reviewed journals [6] or those failing to meet inclusion criteria were excluded. Selection and extraction of data The titles and abstracts of identified studies were first screened, followed by a thorough full-text review to assess eligibility. Key data were extracted from the included studies, including study design, country of origin, patient demographics (age, gender, residence), symptoms, history of HC, serological tests, diagnosis, management strategies, follow-up details, and recurrence rates. Data analysis Microsoft Excel (2019) was used to gather and organize the extracted data, while the Statistical Package for Social Sciences (SPSS) version 27.0 was utilized for data analysis (descriptive statistics). The findings were displayed as frequencies, percentages, ranges, and means with standard deviations. Results A total of 146 studies were retrieved. One was excluded as a duplicate, 14 were non-English, and 62 were unretrievable. After title and abstract screening, 21 studies did not meet the inclusion criteria. The remaining 48 underwent full-text review, with seven more excluded. Of the 41 studies assessed for eligibility, nine were excluded for being from non-peer-reviewed journals or preprints. Ultimately, 32 studies [3,7-37] (56 cases) met the inclusion criteria (Figure 1). Of the included studies [3,7-37], 28 (87.5%) were case reports, while the remaining 4 (12.5%) were case series (Table 1). The highest number of patients were from Afghanistan (10, 17.9%), followed by India (8, 14.3%), Azerbaijan (8, 14.3%), and Morocco and Turkey (6 each, 10.7%). Patient ages spanned from 3 to 80 years, with a mean age of 27.45 ± 19.57 years. The majority of the cases occurred between the first and fifth decades of life (47, 83.9%). The right side was affected in 33 (58.9%) cases and there were no cases with bilateral HC. Sixteen patients (28.6%) were from rural areas, and 13 (23.2%) reported contact with dogs, sheep, or other cattle (Table 2). Table 1. Baseline characteristics of the included studies. Author Type of study Country of the patients N. of Patients Age Sex Symptoms Affected side Cyst Size (Cm) Surgical approach Cyst removal approach Adjuvant therapy Outcome Follow up (months) Abouassi et al. [3] Case Report Syria 1 21 F Proptosis & visual impairment Right 4.2 Fronto-orbitozy-gomatic orbitotomy Cystectomy Albendazole Recovered 3 Ilhami et al. [7] Case series Morocco 3 13 F Proptosis & decreased visual acuity Right 4.2 Internal paracanthal orbitotomy Cystectomy Albendazole Recovered N/A 67 F Proptosis, pain, headache & chemosis Left 3.5 Superolateral orbitotomy Enucleation cystectomy Albendazole Recovered N/A 43 F Proptosis Left 2.9 Internal paracanthal orbitotomy Enucleation cystectomy Albendazole Recovered N/A Alabdullah et al. [8] Case Report Syria 1 10 M Proptosis, diplopia & decreased vision Left 2.7 Subperiosteal orbitotomy Lynch method Albendazole Recovered N/A Khan et al. [9] Case Series Pakistan 11 15 F Proptosis & visual impairment Left N/A Orbitotomy Unspecified Albendazole Re

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