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Chest Wall Hydatid Cysts: A Systematic Review
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Abstract
Introduction
Given the rarity of chest wall hydatid disease, information on this condition is primarily drawn from case reports. Hence, this study systematically reviews the disease's manifestation and management.
Methods
Google Scholar was searched with the following keywords: (hydatid OR hydatidosis OR tapeworms OR echinococcosis OR echinococcus OR granulosus AND chest OR wall OR thoracic OR thorax OR rib OR sternum OR sternal OR cartilage OR intercostal OR extra-pulmonary). Inclusion criteria involved a confirmed diagnosis of chest wall hydatid cyst. Only English-language studies published in legitimate journals were included.
Results
The reported cases were primarily from Turkey (41.5%). The mean age of the patients was 39.7 ± 17.1 years, with a male predominance (56.9%). The most common clinical presentations were swelling (47.1%) and chest or abdominal pain (45.1%). Only 10 cases (19.6%) had reported a history of animal contact. Among those with documented residency (35.3%), 16 (31.4%) resided in rural areas. The average mass size on the CT scan was 7.5 ± 2.4 cm. Surgery was the treatment of choice, with thoracotomy performed in 37.3% of cases, video-assisted thoracoscopy in 1.9%, and the surgical technique not specified in 60.8% of cases. The mean hospital stay was 8.6 ± 4.4 days, and no recurrences were reported.
Conclusion
Despite its rarity, chest wall hydatid cyst may have a good prognosis with few complications. Given its often-nonspecific presentation, reviewing the patient's medical history may help establish an accurate provisional diagnosis.
Introduction
Hydatid disease, caused by tapeworm parasites, is prevalent in sheep-rearing regions, including the Middle East, Mediterranean areas, Africa, South America, and Australia [1,2]. In humans, three types of echinococcosis are known to occur: cystic echinococcosis caused by Echinococcus granulosus, alveolar echinococcosis caused by Echinococcus multilocularis, and polycystic echinococcosis due to Echinococcus Vogeli or Echinococcus oligarthrus [3]. The most common causative organism of human hydatid disease is Echinococcus granulosus [1]. The parasite is a tapeworm with an approximate length of 2 to 7 mm [4]. Hydatid disease or echinococcosis is an old and well-known helminthic disease known since Hippocrates [5]. Rudolphi, in 1808, first used the term hydatid cyst [HC] to describe human echinococcosis [2]. Dogs are the definitive hosts, while farm animals are intermediate hosts. Although humans are not involved in the parasite's life cycle, they may be affected accidentally, either by direct contact with a dog or by ingesting contaminated food and fluid from parasite eggs [6]. After oral ingestion, the cyst hatches in the duodenum and initially spreads to the liver through the portal vein via hematogenous or occasionally lymphogenous routes. It then reaches the lungs through the venous system, and from the lungs, it can disseminate to other organs via the arterial system [7]. The disease can be seen in different body parts, like the thyroid, bladder, heart, and pulmonary artery [7-10]. The chest wall is a very uncommon localization for this disease [6,11]. Therefore, the literature has limited information regarding chest wall HCs. This study aims to systematically review the manifestation and management of the disease.
Methods
Study design
This study was conducted according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines.
Data sources and search strategy
Google Scholar was searched with the following keywords: [hydatid OR hydatidosis OR tapeworms OR echinococcosis OR echinococcus OR granulosus AND chest OR wall OR thoracic OR thorax OR rib OR sternum OR sternal OR cartilage OR intercostal OR extra-pulmonary].
Eligibility Criteria
Inclusion criteria involved a confirmed diagnosis of chest wall HC. Only English-language studies published in legitimate journals were included [12].
Data items
One author screened the studies to select those that met the eligibility criteria, and another rechecked his work. Key data, including the first author's name, study design, country and year of publication, sample size, patient demography, clinical presentation, management strategies, and outcomes, were extracted from the included studies.
Data analysis and synthesis
The data were collected in a Microsoft Excel sheet (2021), and descriptive statistics were performed using the Statistical Package for the Social Sciences (SPSS) version 27. The data were presented as frequencies, percentages, means, and standard deviations.
Results
Study Selection
A systematic search identified a total of 432 articles. After the initial screening, 29 studies presenting only abstracts, two duplicates, and three non-English publications were excluded, resulting in 398 articles for further evaluation. Title and abstract screening excluded 329 studies due to irrelevance, leaving 69 articles for full-text review. Of these, seven were excluded due to irrelevancy, eight due to unretrieved data, and two for being letters to the editor. During the final screening, 11 studies were removed due to publishing in predatory journals. Finally, 41 studies [1–3,5,6,11,13–26,28-48] met the inclusion criteria and were included in the review (Figure 1). The raw data for each included study are detailed in Tables 1, 2, and 3.
Table 1. Characteristics of the studies and patients’ demography with past medical history.
Author/reference
Year
Type of Study
Country
No. of Case
Age
Gender
Residency
Contact with Animal
PMH
PSH
Origin
Althobaity et al. [1]
2023
Case report
Saudi Arabia
1
22
Male
N\A
Yes
Insignificant
N\A
Primary
Goyal et al. [2]
2010
Case report
India
1
30
Female
N\A
N\A
N\A
N\A
N\A
Godazandeh et al. [3]
2020
Case report
Iran
1
40
Male
Urban
No
N\A
No
Primary
Basit et al. [5]
2021
Case report
Afghanistan
1
65
Male
Rural
N\A
N\A
No
Primary
Salih et al. [6]
2017
Case report
Iraq
1
20
Female
Rural
Yes
N\A
N\A
N\A
Döner et al. [11]
2019
Case report
Turkey
1
31
Female
N\A
N\A
N\A
N\A
Primary
MohIeldeen et al. [13]
2013
Case report
Iraq
1
42
Male
Rural
N\A
Hydatid cyst
Pulmonary hydatid cyst surgery
Secondary
Akkas et al. [14]
2016
Case report
Turkey
2
32
Male
N\A
N\A
Hepatic hydatid cyst 2 years ago
N\A
Secondary
24
Male
N\A
N\A
N\A
N\A
N\A
Alloubi et al. [15]
2012
Case report
Morocco
1
57
Male
Rural
N\A
N\A
N\A
N\A
Al-Qudah et al. [16]
2000
Case report
Jordan
1
24
Male
N\A
N\A
N\A
N\A
Primary
Afghani et al. [17]
2017
Case report
Iran
1
35
Female
N\A
N\A
N\A
N\A
Primary
Yekeler et al. [18]
2010
Case report
Turkey
1
57
Male
Rural
Yes
Insignificant
N\A
N\A
Ulger et al. [19]
2013
Case report
Turkey
1
62
Male
N\A
N\A
Hydatid cyst
Hydatid cyst surgery
N\A
Tulay et al. [20]
2015
Case report
Turkey
1
48
Male
N\A
N\A
Insignificant
No
Primary
Tomos et al. [21]
2005
Case report
Greece
1
26
Female
N\A
N\A
Hydatid cyst
Pulmonary and hepatic hydatid cyst surgery
Secondary
Tezcan et al. [22]
2014
Case report
Turkey
1
55
Male
N\A
N\A
Hydatid cyst
Pulmonary hydatid cyst surgery
Secondary
Tadasa et al. [23]
2023
Case report
Ethiopia
1
65
Male
N\A
N\A
N\A
N\A
N\A
Sevinc et al. [24]
2014
Case report
Turkey
1
31
Male
N\A
N\A
N\A
N\A
N\A
Sarkar et al. [25]
2015
Case report
India
1
58
Female
Rural
Yes
N\A
N\A
Primary
Sabzi et al. [26]
2023
Case report
Iran
1
53
Male
Rural
Yes
N\A
N\A
Secondary
Roman et al. [28]
2015
Case report
Romania
1
25
Male
Rural
Yes
History of trauma
N\A
N\A
Redington et al. [29]
2001
Case report
United Kingdom
1
72
Male
N\A
N\A
History of trauma
N\A
N\A
Raut et al. [30]
2004
Case report
India
1
28
Male
Rural
Yes
N\A
N\A
N\A
Ninos et al. [31]
2010
Case report
Greece
1
50
Male
N\A
N\A
Hydatid cyst
Pulmonary hydatid cyst surgery
Primary
Kiliç et al. [32]
2003
Case report
Turkey
1
54
Male
Rural
N\A
Insignificant
N\A
Primary
Karapolat et al. [33]
2012
Case report
Turkey
1
69
Female
N\A
N\A
N\A
N\A
Primary
Karaoğlanoğlu et al. [34]
2001
Case report
Turkey
1
63
Male
Rural
Yes
N\A
N\A
N\A
Kaplanoğlu et al. [35]
2017
Case report
Turkey
1
27
Female
N\A
N\A
N\A
No
N\A
Honda et al. [36]
2010
Case report
Japan
1
9
Female
Rural
N\A
Insignificant
N\A
Secondary
Han et al. [37]
2004
Case report
Turkey
1
N\A
Female
N\A
N\A
Insignificant
N\A
N\A
Gezer et al. [38]
2006
Case report
Turkey
1
57
Female
N\A
N\A
N\A
N\A
N\A
Foroulis et al. [39]
2003
Case report
Greece
1
28
Female
Urban
N\A
N\A
N\A
Primary
Findikcioglu et al. [40]
2010
Case report
Turkey
1
48
Female
N\A
N\A
N\A
N\A
N\A
Faber et al. [41]
2010
Case report
Israel
1
18
Female
Rural
N\A
Thoracic outlet syndrome
N\A
N\A
Demir et al. [42]
2010
Case report
Turkey
1
9
Male
Rural
Yes
N\A
N\A
Primary
Chafik et al. [43]
2009
Case report
Morocco
1
35
Male
N\A
N\A
History of trauma
Undiagnosed swelling drainage
N\A
Sinberg et al. [44]
1936
Case report
United States
1
15
Male
Rural
Yes
Insignificant
N/A
Primary
Rose et al. [45]
1893
Case report
United Kingdom
1
25
Female
N\A
N/A
Bronchitis
N/A
Primary
Machboua et al. [46]
2023
Case series
Morocco
5
26
Male
Rural
N\A
N\A
N\A
N\A
32
Female
N\A
N\A
N\A
N\A
N\A
28
Male
N\A
N\A
N\A
N\A
N\A
57
Male
N\A
N\A
N\A
N\A
N\A
63
Female
N\A
N\A
Hydatid cyst
Pulmonary and hepatic hydatid cyst surgery
Secondary
Özdemir et al. [47]
1994
Case series
Turkey
6
40
Female
N\A
N\A
Hydatid cyst
Pulmonary hydatid cyst surgery
N\A
38
Male
N\A
N\A
N\A
N\A
N\A
26
Female
N\A
N\A
N\A
N\A
N\A
35
Male
N\A
N\A
Hydatid cyst
Hepatic hydatid cyst surgery
Secondary
35
Female
N\A
N\A
Hydatid cyst
Pulmonary hydatid cyst surgery
Secondary
Avci et al. [48]
2005
Case report
Turkey
1
72
Female
N\A
N\A
History of trauma
No
Primary
N/A: non-available, PMH: past medical history, PSH: past surgical history
Table 2. Presentation and diagnosis of the cases.
Author/reference
Year
No. of Case
Presentation
Duration (months)
Size of the mass on CT scan (cm)
Hydatid serology
Provisional diagnosis
Pre-operative medication
Althobaity et al. [1]
2023
1
Chest pain
36
N\A
N\A
Hydatid disease
Albendazole
Goyal et al. [2]
2010
1
Swelling, Chest pain
N\A
N\A
N\A
N\A
No
Godazandeh et al. [3]
2020
1
Swelling
5
N\A
Negative
N\A
No
Basit et al. [5]
2021
1
Chest pain
0.66*
N\A
N\A
Chest wall tumor
No
Salih et al. [6]
2017
1
Swelling, Chest pain
60
N\A
N\A
N\A
No
Döner et al. [11]
2019
1
Swelling, Chest pain
N\A
4
N\A
Hydatid disease
No
MohIeldeen et al. [13]
2013
1
Chest pain, Numbness
4
N\A
N\A
Hydatid disease
No
Akkas et al. [14]
2016
2
Chest pain
N\A
10
N\A
N\A
No
N\A
N\A
10
N\A
N\A
No
Alloubi et al. [15]
2012
1
Chest pain
3
9
N\A
Chest wall tumor
No
Al-Qudah et al. [16]
2000
1
Dyspnea, Cough, Fever, Shoulder pain
12
N\A
Negative
N\A
No
Afghani et al. [17]
2017
1
Swelling, Chest pain
N\A
N\A
Negative
Hydatid disease
Albendazole
Yekeler et al. [18]
2010
1
N\A
N\A
N\A
Positive
Hydatid disease
No
Ulger et al. [19]
2013
1
Chest pain
70
N\A
N\A
Hydatid disease
Albendazole
Tulay et al. [20]
2015
1
Swelling
5
10
N/A
N\A
No
Tomos et al. [21]
2005
1
Chest pain
N\A
N\A
Negative
N\A
No
Tezcan et al. [22]
2014
1
Cough, Dyspnea, Shoulder pain
N\A
8
N\A
N\A
No
Tadasa et al. [23]
2023
1
Cough, Back pain, Heaviness, Paresthesia
12
N\A
N\A
Hydatid disease
Albendazole
Sevinc et al. [24]
2014
1
Swelling
N\A
5
N\A
Hydatid disease
No
Sarkar et al. [25]
2015
1
Swelling
36
N\A
Positive
Hydatid disease
Albendazole
Sabzi et al. [26]
2023
1
Swelling, Chest pain
24
N\A
Negative
Hydatid disease
No
Roman et al. [28]
2015
1
Chest pain
N\A
3
N\A
Hydatid disease
No
Redington et al. [29]
2001
1
Dizziness, Sweating, Horsness of voice, Swelling, Chest pain
24
7
Negative
Hydatid disease
Albendazole
Raut et al. [30]
2004
1
Back pain, Paraparesis
6
N\A
N\A
Hydatid disease
No
Ninos et al. [31]
2010
1
Swelling
N\A
N\A
Negative
Hydatid disease
Albendazole
Kiliç et al. [32]
2003
1
Swelling, Chest pain
3
N\A
N\A
N\A
No
Karapolat et al. [33]
2012
1
Abdominal pain
48
9
N\A
Chest wall tumor
No
Karaoğlanoğlu et al. [34]
2001
1
Cough
144
N\A
Negative
N\A
No
Kaplanoğlu et al. [35]
2017
1
Swelling
6
N\A
N\A
N\A
No
Honda et al. [36]
2010
1
Fever, General fatigue, Nausea
N\A
6
Positive
N\A
No
Han et al. [37]
2004
1
Swelling
8
N\A
Positive
N\A
No
Gezer et al. [38]
2006
1
Shoulder pain, Numbness, Arm weakness
3
N\A
N\A
N\A
No
Foroulis et al. [39]
2003
1
Swelling
N\A
N\A
Positive
N\A
Albendazole
Findikcioglu et al. [40]
2010
1
Swelling
N\A
N\A
N\A
N\A
No
Faber et al. [41]
2010
1
Shoulder pain, Swelling
12
N\A
N\A
Aneurysmal bone cyst
No
Demir et al. [42]
2010
1
Chest pain
1
N\A
Positive
Ewing sarcoma
No
Chafik et al. [43]
2009
1
Swelling
N\A
N\A
Negative
N\A
No
Sinberg et al. [44]
1936
1
Swelling, Chest pain
18
N\A
N/A
N\A
No
Rose et al. [45]
1893
1
Swelling
N\A
N\A
N/A
N\A
No
Machboua et al. [46]
2023
5
Chest pain, Back pain, Lower limb paresthesia
N\A
N\A
Positive
N\A
No
Chest pain, Back pain, Dyspnea
N\A
N\A
Positive
N\A
No
Chest pain, Dyspnea
N\A
N\A
N\A
N\A
No
Swelling, Chest pain
N\A
N\A
Positive
N\A
No
Chest pain
N\A
N\A
N\A
N\A
No
Özdemir et al. [47]
1994
6
Swelling
N\A
N\A
N\A
N\A
No
N\A
N\A
N\A
N\A
N\A
No
N\A
N\A
N\A
N\A
N\A
No
Empyema
N\A
N\A
N\A
N\A
No
Swelling
N\A
N\A
N\A
N\A
No
Swelling, Shoulder pain
N\A
N\A
N\A
N\A
No
Avci et al. [48]
2005
1
Chest pain
N\A
9
N\A
Hematoma
No
* Less than a month
Table 3. Management and outcome.
Author/reference
Year
No. of Case
Surgical approach
Scolicidal agent
Site of involvement
Post-operative medication
Complication
Recurrence
Follow-up (month)
Althobaity et al. [1]
2023
1
Video-assisted thoracoscopy [VATS]
Hypertonic saline
N\A
Albendazole
Pneumothorax
No
5
Goyal et al. [2]
2010
1
Thoracotomy
Hypertonic saline
Rib and intercostal muscle
No
No
N\A
N\A
Godazandeh et al. [3]
2020
1
N\A
N\A
N\A
Albendazole
No
No
24
Basit et al. [5]
2021
1
N\A
Hypertonic saline
N\A
Albendazole
No
N\A
N\A
Salih et al. [6]
2017
1
N\A
N\A
N\A
N\A
No
No
3
Döner et al. [11]
2019
1
N\A
N\A
Intercostal muscle
Albendazole
No
N\A
N\A
MohIeldeen et al. [13]
2013
1
N\A
N\A
8th rib
Albendazole
No
No
24
Akkas et al. [14]
2016
2
Thoracotomy
N\A
Intercostal muscle
Albendazole
No
No
36
Thoracotomy
N\A
N\A
Albendazole
No
No
18
Alloubi et al. [15]
2012
1
Thoracotomy
N\A
6th and 7th ribs
Albendazole
No
No
6
Al-Qudah et al. [16]
2000
1
Thoracotomy
N\A
1st rib
No
No
N\A
N\A
Afghani et al. [17]
2017
1
N\A
N\A
N\A
Albendazole
No
No
24
Yekeler et al. [18]
2010
1
Thoracotomy
N\A
6th rib
Albendazole
No
No
10
Ulger et al. [19]
2013
1
N\A
N\A
Sternum
No
N\A
No
12
Tulay et al. [20]
2015
1
N\A
Hypertonic saline
N\A
Albendazole
N\A
No
6
Tomos et al. [21]
2005
1
Thoracotomy
N\A
6th rib
No
N\A
N\A
N\A
Tezcan et al. [22]
2014
1
N\A
N\A
Sternum
No
No
N\A
N\A
Tadasa et al. [23]
2023
1
No
N\A
N\A
No
N\A
N\A
N\A
Sevinc et al. [24]
2014
1
N\A
Hypertonic saline
N\A
Albendazole
No
N\A
N\A
Sarkar et al. [25]
2015
1
N\A
N\A
N\A
Albendazole
No
No
12
Sabzi et al. [26]
2023
1
N\A
N\A
N\A
Albendazole
No
N\A
N\A
Roman et al. [28]
2015
1
Thoracotomy
Formalin
8th rib
Albendazole, Chemotherapy
No
No
1
Redington et al. [29]
2001
1
No
N\A
N\A
No
N\A
N\A
N\A
Raut et al. [30]
2004
1
N\A
N\A
9th rib
Albendazole
No
N\A
N\A
Ninos et al. [31]
2010
1
N\A
N\A
Intercostal muscle
Albendazole
N\A
No
12
Kiliç et al. [32]
2003
1
Thoracotomy
N\A
5th, 6th and 7th ribs
Albendazole
No
N\A
N\A
Karapolat et al. [33]
2012
1
N\A
N\A
N\A
Albendazole
No
No
12
Karaoğlanoğlu et al. [34]
2001
1
N\A
N\A
5th and 6th ribs, Serratus anterior muscle, Latissimus dorsi muscle
Albendazole
No
No
6
Kaplanoğlu et al. [35]
2017
1
N\A
N\A
Intercostal muscle
No
N\A
No
12
Honda et al. [36]
2010
1
N\A
Hypertonic saline
N\A
Albendazole
N\A
No
16
Han et al. [37]
2004
1
N\A
N\A
7th rib
Albendazole
N\A
N\A
N\A
Gezer et al. [38]
2006
1
Thoracotomy
N\A
1st rib
Albendazole
No
No
15
Foroulis et al. [39]
2003
1
Thoracotomy
Hypertonic saline
10th vertebra, 9th and 10th ribs
Albendazole
No
No
48
Findikcioglu et al. [40]
2010
1
Thoracotomy
N\A
Riband vertebra
No
N\A
N\A
N\A
Faber et al. [41]
2010
1
N\A
N\A
1st rib
No
No
N\A
N\A
Demir et al. [42]
2010
1
N\A
N\A
N\A
Albendazole
N\A
N\A
N\A
Chafik et al. [43]
2009
1
Thoracotomy
N\A
N\A
Albendazole
No
N\A
N\A
Sinberg et al. [44]
1936
1
N\A
Hypertonic saline
Sternum
No
Slight superficial infection
N\A
N\A
Rose et al. [45]
1893
1
N\A
N\A
N\A
No
Hemorrhage
N\A
N\A
Machboua et al. [46]
2023
5
Thoracotomy
N\A
N\A
No
Pleurocutaneous fistula, Disabling parietal pain and neurological pain, Disorder of the Shoulder Girdle.
No
24
Thoracotomy
N\A
Rib and vertebra
No
No
N\A
N\A
N\A
N\A
N\A
No
No
N\A
N\A
Thoracotomy
N\A
N\A
No
No
N\A
N\A
Thoracotomy
N\A
N\A
No
N\A
N\A
N\A
Özdemir et al. [47]
1994
6
N\A
N\A
N\A
No
N\A
N\A
N\A
N\A
N\A
N\A
No
N\A
N\A
N\A
Thoracotomy
N\A
N\A
No
N\A
N\A
N\A
Thoracotomy
N\A
N\A
No
N\A
N\A
N\A
N\A
N\A
N\A
No
N\A
N\A
N\A
N\A
N\A
N\A
No
N\A
N\A
N\A
Avci et al. [48]
2005
1
N\A
N\A
N\A
No
No
N\A
N\A
N/A:non-available
Main findings
The reported cases were primarily from Turkey (41.5%). The mean age of the patients was 39.7 ± 17.1 years, with a male predominance (56.9%). The past medical history for HC was positive in 10 cases (19.6%). The most common clinical presentations were swelling in 24 cases (47.1%) and chest or abdominal pain in 23 cases (45.1%). Only 10 cases (19.6%) had reported a history of animal contact, while one patient (2%) reported no such history. Data on the history of animal contact was unavailable in 78.4% of cases. Among those with documented residency (35.3%), 16 (31.4%) resided in rural areas, while two (3.9%) were from urban settings. The disease was primary in sixteen cases (31.4%), and in nine (17.6%), it was secondary. The disease origin was unknown in 26 cases (51%). The average mass size on the CT scan was 7.5 ± 2.4 cm. Hydatid serology was performed in 18 patients, yielding positive results in nine (50%). A provisional diagnosis of hydatid disease was considered in 14 cases (27.5%). Pre-operative treatment with albendazole was administered to eight patients (15.7%). Surgery was the treatment of choice, with thoracotomy performed in 37.3% of cases, video-assisted thoracoscopy (VATS) in 1.9%, and the surgical technique not specified in 60.8% of cases. The average hospital stay was 8.6 ± 4.4 days. The ribs (31.4%) were the primary involved sites, followed by the muscles (11.8%). Postoperative medication included albendazole in 25 cases (49%) and albendazole with chemotherapy in one case (1.9%). Twenty-four cases (47.1%) had received no postoperative treatment. Only four cases (7.8%) faced complications. No recurrence was reported after a mean follow-up period of 14.1± 10.3 months (Table 4).
Table 4. Summary and baseline characteristics of the included studies.
Variables
Frequency/percentage
Mean Age (years)
39.7 ± 17.1
Sex
Male
Female
29 (56.9%)
22 (43.1%)
Country of study
Turkey
Morocco
Greece
India
Iran
Iraq
United Kingdom
Afghanistan
Ethiopia
Israel
Japan
Jordan
Romania
Saudi Arabia
United States
17 (41.5%)
3 (7.3%)
3 (7.3%)
3 (7.3%)
3 (7.3%)
2 (5.0%)
2 (5.0%)
1 (2.4%)
1 (2.4%)
1 (2.4%)
1 (2.4%)
1 (2.4%)
1 (2.4%)
1 (2.4%)
1 (2.4%)
Past medical history of hydatid cyst
Positive
Negative
10 (19.6%)
41 (80.4%)
Contact with an animal
Yes
No
N\A
10 (19.6%)
1 (2.0%)
40 (78.4%)
Residency
Rural
Urban
N\A
16 (31.4%)
2 (3.9%)
33 (64.7%)
Origin
Primary
Secondary
N\A
16 (31.4%)
9 (17.6%)
26 (51.0%)
Size of the mass on CT scan
7.5 ± 2.4 cm
Presentation
Swelling
Chest/Abdominal pain
Dyspnea
Cough
Shoulder/back pain
Numbness
Others
N\A
24 (47.1%)
23 (45.1%)
4 (7.8%)
4 (7.8%)
9 (17.6%)
5 (9.8%)
6 (11.8%)
4 (7.8%)
Hydatid serology test
Positive
Negative
N\A
9 (17.6%)
9 (17.6%)
33 (64.7%)
Provisional diagnosis
Hydatid disease
Chest wall tumor
Aneurysmal bone cyst
Ewing sarcoma
Hematoma
N\A
14 (27.5%)
3 (5.9%)
1 (1.9%)
1 (1.9%)
1 (1.9%)
31 (60.8%)
Pre-operative medication
Albendazole
No
8 (15.7%)
43 (84.3%)
Surgical approach
Thoracotomy
Video-assisted thoracoscopy
N\A
19 (37.3%)
1 (1.9%)
31 (60.8%)
Injection of scolicidal agent with surgery
Hypertonic saline
Formalin
No
8 (15.7%)
1 (1.9%)
42 (82.4%)
Site of involvement
Rib
Muscle
Sternum
Vertebra
N\A
16 (31.4%)
6 (11.8%)
3 (5.9%)
3 (5.9%)
28 (54.9%)
Post-operative medication
Albendazole
Albendazole and chemotherapy
No
N/A
25 (49.0%)
1 (1.9%)
24 (47.1%)
1 (1.9%)
Mean postoperative hospital stay (days)*
8.6 ± 4.4
Outcome of surgery
Recovered with no complication
Recovered with complication
N\A
29 (56.9%)
4 (7.8%)
18 (35.3%)
Average duration of follow-up (months)
14.1± 10.3
Recurrence
Yes
No
N\A
0 (0.0%)
21 (41.0%)
30 (59.0%)
N/A: non-available, CT: computed tomography.
*For 12 patients out of 51 patients who underwent surgery.
#For 23 patients out of 51 patients who underwent surgery.
Discussion
Human echinococcosis, a prevalent parasitic infection, presents a considerable health and economic burden to society, yet it remains largely neglected as a disease [49]. The findings of this study provide a comprehensive review of chest wall HC, with the majority of the studies conducted in Turkey (41.5%), followed by Morocco (7.3%), Greece (7.3%), India (7.3%), and Iran (7.3%). The higher incidence in these countries may reflect the endemic nature of hydatid disease, likely due to closer contact with livestock and domestic animals [50]. However, it has become a pressing global health issue, primarily attributed to increasing immigration rates and travel activities [7].
The mean age of 39.7 years, with a slight male predominance (56.9%), suggests that both demographic groups are at risk. However, occupational or environmental exposure factors could contribute to the observed gender disparity.
Clinically, the most common presentation was swelling (47.1%), followed by chest or abdominal pain (45.1%). This indicates that chest wall HCs may present as localized symptoms rather than systemic manifestations [51]. The relatively low percentage (19.6%) of patients with a history of animal contact suggests that direct exposure may not always be reported or remembered, or that indirect exposure through contaminated water or food could also be significant transmission routes [26,27]. In addition, a review by Possenti and colleagues suggested that the primary route of human cystic echinococcosis transmission may be the direct or indirect contamination of hands with Echinococcus granulosus eggs excreted by dogs [52].
Schantz et al. indicated that individuals who own livestock are three times more likely to be diagnosed with this disease compared to those who do not own livestock [53]. In accordance with this finding, 16 (88.9%) of the 18 cases with known residency were from rural areas.
Ten cases (19.6%) had a positive history for HC at other sites, and the primary origin of cysts in 31.4% of patients and secondary origin in 17.6% highlights the importance of considering both primary infection or possible spread from other sites, such as the liver or lungs, as the diagnosis and treatment strategies may be different [1].
In the present study, the average cyst size on the CT scan was 7.5 cm, reflecting potentially large cysts that could cause significant local pressure effects or complications; this might be because the course of infection is slow, and most infected individuals remain either asymptomatic for years or exhibit non-specific symptoms leading to accidental diagnosis [54]. Parasite eggs can remain viable from several months up to a few years in the environment, and diverse conditions [55].
Only nine (50%) out of 18 patients with available data in this systematic review had positive hydatid serology, suggesting that serological tests alone may not be sufficient for diagnosis and imaging, particularly CT scans, plays a crucial role in the diagnostic process [56]. The provisional diagnosis of hydatid disease in only 27.5% of the cases underscores the diagnostic challenges faced by clinicians, especially when the clinical presentation is atypical or when imaging findings are inconclusive [57].
The importance and priority of infection prevention by practicing several preventive actions, especially in developing countries, has been proposed. The most significant of them included self-hygiene practices, proper washing and cooking of foods, and avoiding eating raw foods or groceries [27]. Regarding management, surgical intervention was the primary treatment modality [1,2,7,58]. A study indicated that while VATS has been successfully applied in managing pulmonary HCs, its adoption remains limited. It has been pointed out that many surgeons are still hesitant to use VATS due to a lack of familiarity with the technique and concerns about potential complications, such as cyst rupture or spillage of infectious material [59]. In this review, authors came across similar findings, such as thoracotomy being performed in 37.3% of cases and VATS being performed in 1.9%. This may depend on cyst size and location. Furthermore, the low utilization of minimally invasive approaches like thoracoscopy may suggest either a preference for traditional open techniques or a lack of resources or expertise in minimally invasive surgery in some settings. The postoperative outcomes were generally favorable, with only 7.8% of patients facing complications, and no recurrence was reported, emphasizing the effectiveness of surgical management.
Machboua et al. on intra-thoracic extra-pulmonary hydatidosis noted that the average duration of hospitalization was only seven days, which is similar to what was found in this study (8.58 ± 4.44 days) [46].
The administration of post-operative albendazole alone (49%) or in combination with other medications (1.9%) may appear to be a standard practice to prevent recurrence, even though no recurrences were reported in this study. This could reflect a preventive strategy against the possible dissemination of HCs during surgery [1,14,15,17,18]. Overall, the study may provide valuable insights into the epidemiology, clinical presentation, diagnostic strategies, and management outcomes of chest wall HCs; however, the lack of essential data in a number of reviewed studies might generate biased or non-conclusive findings.
Conclusion
Despite its rarity, chest wall HC may have a good prognosis with few complications. Given its often-nonspecific presentation, reviewing the patient's medical history may help establish an accurate provisional diagnosis.
Declarations
Conflicts of interest: The authors have no conflicts of interest to disclose.
Ethical approval: Not applicable, as systematic reviews do not require ethical approval.
Patient consent (participation and publication): Not applicable.
Funding: The present study received no financial support.
Acknowledgements: None to be declared.
Authors' contributions: FHK and BAA were significant contributors to the conception of the study and the literature search for related studies. HOA and DHH involved in the literature review, study design, and manuscript writing. HKA, AHH, AHA, AAM, SMA, SOK, FA, SSA, and SHM were involved in the literature review, the study's design, the critical revision of the manuscript, and data collection. FHK and HOA confirm the authenticity of all the raw data. All authors approved the final version of the manuscript.
Use of AI: ChatGPT-3.5 was used to assist in language editing and improving the clarity of the manuscript. All content was reviewed and verified by the authors. Authors are fully responsible for the entire content of their manuscript.
Data availability statement: Not applicable.
Title: Chest Wall Hydatid Cysts: A Systematic Review
Description:
Abstract
Introduction
Given the rarity of chest wall hydatid disease, information on this condition is primarily drawn from case reports.
Hence, this study systematically reviews the disease's manifestation and management.
Methods
Google Scholar was searched with the following keywords: (hydatid OR hydatidosis OR tapeworms OR echinococcosis OR echinococcus OR granulosus AND chest OR wall OR thoracic OR thorax OR rib OR sternum OR sternal OR cartilage OR intercostal OR extra-pulmonary).
Inclusion criteria involved a confirmed diagnosis of chest wall hydatid cyst.
Only English-language studies published in legitimate journals were included.
Results
The reported cases were primarily from Turkey (41.
5%).
The mean age of the patients was 39.
7 ± 17.
1 years, with a male predominance (56.
9%).
The most common clinical presentations were swelling (47.
1%) and chest or abdominal pain (45.
1%).
Only 10 cases (19.
6%) had reported a history of animal contact.
Among those with documented residency (35.
3%), 16 (31.
4%) resided in rural areas.
The average mass size on the CT scan was 7.
5 ± 2.
4 cm.
Surgery was the treatment of choice, with thoracotomy performed in 37.
3% of cases, video-assisted thoracoscopy in 1.
9%, and the surgical technique not specified in 60.
8% of cases.
The mean hospital stay was 8.
6 ± 4.
4 days, and no recurrences were reported.
Conclusion
Despite its rarity, chest wall hydatid cyst may have a good prognosis with few complications.
Given its often-nonspecific presentation, reviewing the patient's medical history may help establish an accurate provisional diagnosis.
Introduction
Hydatid disease, caused by tapeworm parasites, is prevalent in sheep-rearing regions, including the Middle East, Mediterranean areas, Africa, South America, and Australia [1,2].
In humans, three types of echinococcosis are known to occur: cystic echinococcosis caused by Echinococcus granulosus, alveolar echinococcosis caused by Echinococcus multilocularis, and polycystic echinococcosis due to Echinococcus Vogeli or Echinococcus oligarthrus [3].
The most common causative organism of human hydatid disease is Echinococcus granulosus [1].
The parasite is a tapeworm with an approximate length of 2 to 7 mm [4].
Hydatid disease or echinococcosis is an old and well-known helminthic disease known since Hippocrates [5].
Rudolphi, in 1808, first used the term hydatid cyst [HC] to describe human echinococcosis [2].
Dogs are the definitive hosts, while farm animals are intermediate hosts.
Although humans are not involved in the parasite's life cycle, they may be affected accidentally, either by direct contact with a dog or by ingesting contaminated food and fluid from parasite eggs [6].
After oral ingestion, the cyst hatches in the duodenum and initially spreads to the liver through the portal vein via hematogenous or occasionally lymphogenous routes.
It then reaches the lungs through the venous system, and from the lungs, it can disseminate to other organs via the arterial system [7].
The disease can be seen in different body parts, like the thyroid, bladder, heart, and pulmonary artery [7-10].
The chest wall is a very uncommon localization for this disease [6,11].
Therefore, the literature has limited information regarding chest wall HCs.
This study aims to systematically review the manifestation and management of the disease.
Methods
Study design
This study was conducted according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines.
Data sources and search strategy
Google Scholar was searched with the following keywords: [hydatid OR hydatidosis OR tapeworms OR echinococcosis OR echinococcus OR granulosus AND chest OR wall OR thoracic OR thorax OR rib OR sternum OR sternal OR cartilage OR intercostal OR extra-pulmonary].
Eligibility Criteria
Inclusion criteria involved a confirmed diagnosis of chest wall HC.
Only English-language studies published in legitimate journals were included [12].
Data items
One author screened the studies to select those that met the eligibility criteria, and another rechecked his work.
Key data, including the first author's name, study design, country and year of publication, sample size, patient demography, clinical presentation, management strategies, and outcomes, were extracted from the included studies.
Data analysis and synthesis
The data were collected in a Microsoft Excel sheet (2021), and descriptive statistics were performed using the Statistical Package for the Social Sciences (SPSS) version 27.
The data were presented as frequencies, percentages, means, and standard deviations.
Results
Study Selection
A systematic search identified a total of 432 articles.
After the initial screening, 29 studies presenting only abstracts, two duplicates, and three non-English publications were excluded, resulting in 398 articles for further evaluation.
Title and abstract screening excluded 329 studies due to irrelevance, leaving 69 articles for full-text review.
Of these, seven were excluded due to irrelevancy, eight due to unretrieved data, and two for being letters to the editor.
During the final screening, 11 studies were removed due to publishing in predatory journals.
Finally, 41 studies [1–3,5,6,11,13–26,28-48] met the inclusion criteria and were included in the review (Figure 1).
The raw data for each included study are detailed in Tables 1, 2, and 3.
Table 1.
Characteristics of the studies and patients’ demography with past medical history.
Author/reference
Year
Type of Study
Country
No.
of Case
Age
Gender
Residency
Contact with Animal
PMH
PSH
Origin
Althobaity et al.
[1]
2023
Case report
Saudi Arabia
1
22
Male
N\A
Yes
Insignificant
N\A
Primary
Goyal et al.
[2]
2010
Case report
India
1
30
Female
N\A
N\A
N\A
N\A
N\A
Godazandeh et al.
[3]
2020
Case report
Iran
1
40
Male
Urban
No
N\A
No
Primary
Basit et al.
[5]
2021
Case report
Afghanistan
1
65
Male
Rural
N\A
N\A
No
Primary
Salih et al.
[6]
2017
Case report
Iraq
1
20
Female
Rural
Yes
N\A
N\A
N\A
Döner et al.
[11]
2019
Case report
Turkey
1
31
Female
N\A
N\A
N\A
N\A
Primary
MohIeldeen et al.
[13]
2013
Case report
Iraq
1
42
Male
Rural
N\A
Hydatid cyst
Pulmonary hydatid cyst surgery
Secondary
Akkas et al.
[14]
2016
Case report
Turkey
2
32
Male
N\A
N\A
Hepatic hydatid cyst 2 years ago
N\A
Secondary
24
Male
N\A
N\A
N\A
N\A
N\A
Alloubi et al.
[15]
2012
Case report
Morocco
1
57
Male
Rural
N\A
N\A
N\A
N\A
Al-Qudah et al.
[16]
2000
Case report
Jordan
1
24
Male
N\A
N\A
N\A
N\A
Primary
Afghani et al.
[17]
2017
Case report
Iran
1
35
Female
N\A
N\A
N\A
N\A
Primary
Yekeler et al.
[18]
2010
Case report
Turkey
1
57
Male
Rural
Yes
Insignificant
N\A
N\A
Ulger et al.
[19]
2013
Case report
Turkey
1
62
Male
N\A
N\A
Hydatid cyst
Hydatid cyst surgery
N\A
Tulay et al.
[20]
2015
Case report
Turkey
1
48
Male
N\A
N\A
Insignificant
No
Primary
Tomos et al.
[21]
2005
Case report
Greece
1
26
Female
N\A
N\A
Hydatid cyst
Pulmonary and hepatic hydatid cyst surgery
Secondary
Tezcan et al.
[22]
2014
Case report
Turkey
1
55
Male
N\A
N\A
Hydatid cyst
Pulmonary hydatid cyst surgery
Secondary
Tadasa et al.
[23]
2023
Case report
Ethiopia
1
65
Male
N\A
N\A
N\A
N\A
N\A
Sevinc et al.
[24]
2014
Case report
Turkey
1
31
Male
N\A
N\A
N\A
N\A
N\A
Sarkar et al.
[25]
2015
Case report
India
1
58
Female
Rural
Yes
N\A
N\A
Primary
Sabzi et al.
[26]
2023
Case report
Iran
1
53
Male
Rural
Yes
N\A
N\A
Secondary
Roman et al.
[28]
2015
Case report
Romania
1
25
Male
Rural
Yes
History of trauma
N\A
N\A
Redington et al.
[29]
2001
Case report
United Kingdom
1
72
Male
N\A
N\A
History of trauma
N\A
N\A
Raut et al.
[30]
2004
Case report
India
1
28
Male
Rural
Yes
N\A
N\A
N\A
Ninos et al.
[31]
2010
Case report
Greece
1
50
Male
N\A
N\A
Hydatid cyst
Pulmonary hydatid cyst surgery
Primary
Kiliç et al.
[32]
2003
Case report
Turkey
1
54
Male
Rural
N\A
Insignificant
N\A
Primary
Karapolat et al.
[33]
2012
Case report
Turkey
1
69
Female
N\A
N\A
N\A
N\A
Primary
Karaoğlanoğlu et al.
[34]
2001
Case report
Turkey
1
63
Male
Rural
Yes
N\A
N\A
N\A
Kaplanoğlu et al.
[35]
2017
Case report
Turkey
1
27
Female
N\A
N\A
N\A
No
N\A
Honda et al.
[36]
2010
Case report
Japan
1
9
Female
Rural
N\A
Insignificant
N\A
Secondary
Han et al.
[37]
2004
Case report
Turkey
1
N\A
Female
N\A
N\A
Insignificant
N\A
N\A
Gezer et al.
[38]
2006
Case report
Turkey
1
57
Female
N\A
N\A
N\A
N\A
N\A
Foroulis et al.
[39]
2003
Case report
Greece
1
28
Female
Urban
N\A
N\A
N\A
Primary
Findikcioglu et al.
[40]
2010
Case report
Turkey
1
48
Female
N\A
N\A
N\A
N\A
N\A
Faber et al.
[41]
2010
Case report
Israel
1
18
Female
Rural
N\A
Thoracic outlet syndrome
N\A
N\A
Demir et al.
[42]
2010
Case report
Turkey
1
9
Male
Rural
Yes
N\A
N\A
Primary
Chafik et al.
[43]
2009
Case report
Morocco
1
35
Male
N\A
N\A
History of trauma
Undiagnosed swelling drainage
N\A
Sinberg et al.
[44]
1936
Case report
United States
1
15
Male
Rural
Yes
Insignificant
N/A
Primary
Rose et al.
[45]
1893
Case report
United Kingdom
1
25
Female
N\A
N/A
Bronchitis
N/A
Primary
Machboua et al.
[46]
2023
Case series
Morocco
5
26
Male
Rural
N\A
N\A
N\A
N\A
32
Female
N\A
N\A
N\A
N\A
N\A
28
Male
N\A
N\A
N\A
N\A
N\A
57
Male
N\A
N\A
N\A
N\A
N\A
63
Female
N\A
N\A
Hydatid cyst
Pulmonary and hepatic hydatid cyst surgery
Secondary
Özdemir et al.
[47]
1994
Case series
Turkey
6
40
Female
N\A
N\A
Hydatid cyst
Pulmonary hydatid cyst surgery
N\A
38
Male
N\A
N\A
N\A
N\A
N\A
26
Female
N\A
N\A
N\A
N\A
N\A
35
Male
N\A
N\A
Hydatid cyst
Hepatic hydatid cyst surgery
Secondary
35
Female
N\A
N\A
Hydatid cyst
Pulmonary hydatid cyst surgery
Secondary
Avci et al.
[48]
2005
Case report
Turkey
1
72
Female
N\A
N\A
History of trauma
No
Primary
N/A: non-available, PMH: past medical history, PSH: past surgical history
Table 2.
Presentation and diagnosis of the cases.
Author/reference
Year
No.
of Case
Presentation
Duration (months)
Size of the mass on CT scan (cm)
Hydatid serology
Provisional diagnosis
Pre-operative medication
Althobaity et al.
[1]
2023
1
Chest pain
36
N\A
N\A
Hydatid disease
Albendazole
Goyal et al.
[2]
2010
1
Swelling, Chest pain
N\A
N\A
N\A
N\A
No
Godazandeh et al.
[3]
2020
1
Swelling
5
N\A
Negative
N\A
No
Basit et al.
[5]
2021
1
Chest pain
0.
66*
N\A
N\A
Chest wall tumor
No
Salih et al.
[6]
2017
1
Swelling, Chest pain
60
N\A
N\A
N\A
No
Döner et al.
[11]
2019
1
Swelling, Chest pain
N\A
4
N\A
Hydatid disease
No
MohIeldeen et al.
[13]
2013
1
Chest pain, Numbness
4
N\A
N\A
Hydatid disease
No
Akkas et al.
[14]
2016
2
Chest pain
N\A
10
N\A
N\A
No
N\A
N\A
10
N\A
N\A
No
Alloubi et al.
[15]
2012
1
Chest pain
3
9
N\A
Chest wall tumor
No
Al-Qudah et al.
[16]
2000
1
Dyspnea, Cough, Fever, Shoulder pain
12
N\A
Negative
N\A
No
Afghani et al.
[17]
2017
1
Swelling, Chest pain
N\A
N\A
Negative
Hydatid disease
Albendazole
Yekeler et al.
[18]
2010
1
N\A
N\A
N\A
Positive
Hydatid disease
No
Ulger et al.
[19]
2013
1
Chest pain
70
N\A
N\A
Hydatid disease
Albendazole
Tulay et al.
[20]
2015
1
Swelling
5
10
N/A
N\A
No
Tomos et al.
[21]
2005
1
Chest pain
N\A
N\A
Negative
N\A
No
Tezcan et al.
[22]
2014
1
Cough, Dyspnea, Shoulder pain
N\A
8
N\A
N\A
No
Tadasa et al.
[23]
2023
1
Cough, Back pain, Heaviness, Paresthesia
12
N\A
N\A
Hydatid disease
Albendazole
Sevinc et al.
[24]
2014
1
Swelling
N\A
5
N\A
Hydatid disease
No
Sarkar et al.
[25]
2015
1
Swelling
36
N\A
Positive
Hydatid disease
Albendazole
Sabzi et al.
[26]
2023
1
Swelling, Chest pain
24
N\A
Negative
Hydatid disease
No
Roman et al.
[28]
2015
1
Chest pain
N\A
3
N\A
Hydatid disease
No
Redington et al.
[29]
2001
1
Dizziness, Sweating, Horsness of voice, Swelling, Chest pain
24
7
Negative
Hydatid disease
Albendazole
Raut et al.
[30]
2004
1
Back pain, Paraparesis
6
N\A
N\A
Hydatid disease
No
Ninos et al.
[31]
2010
1
Swelling
N\A
N\A
Negative
Hydatid disease
Albendazole
Kiliç et al.
[32]
2003
1
Swelling, Chest pain
3
N\A
N\A
N\A
No
Karapolat et al.
[33]
2012
1
Abdominal pain
48
9
N\A
Chest wall tumor
No
Karaoğlanoğlu et al.
[34]
2001
1
Cough
144
N\A
Negative
N\A
No
Kaplanoğlu et al.
[35]
2017
1
Swelling
6
N\A
N\A
N\A
No
Honda et al.
[36]
2010
1
Fever, General fatigue, Nausea
N\A
6
Positive
N\A
No
Han et al.
[37]
2004
1
Swelling
8
N\A
Positive
N\A
No
Gezer et al.
[38]
2006
1
Shoulder pain, Numbness, Arm weakness
3
N\A
N\A
N\A
No
Foroulis et al.
[39]
2003
1
Swelling
N\A
N\A
Positive
N\A
Albendazole
Findikcioglu et al.
[40]
2010
1
Swelling
N\A
N\A
N\A
N\A
No
Faber et al.
[41]
2010
1
Shoulder pain, Swelling
12
N\A
N\A
Aneurysmal bone cyst
No
Demir et al.
[42]
2010
1
Chest pain
1
N\A
Positive
Ewing sarcoma
No
Chafik et al.
[43]
2009
1
Swelling
N\A
N\A
Negative
N\A
No
Sinberg et al.
[44]
1936
1
Swelling, Chest pain
18
N\A
N/A
N\A
No
Rose et al.
[45]
1893
1
Swelling
N\A
N\A
N/A
N\A
No
Machboua et al.
[46]
2023
5
Chest pain, Back pain, Lower limb paresthesia
N\A
N\A
Positive
N\A
No
Chest pain, Back pain, Dyspnea
N\A
N\A
Positive
N\A
No
Chest pain, Dyspnea
N\A
N\A
N\A
N\A
No
Swelling, Chest pain
N\A
N\A
Positive
N\A
No
Chest pain
N\A
N\A
N\A
N\A
No
Özdemir et al.
[47]
1994
6
Swelling
N\A
N\A
N\A
N\A
No
N\A
N\A
N\A
N\A
N\A
No
N\A
N\A
N\A
N\A
N\A
No
Empyema
N\A
N\A
N\A
N\A
No
Swelling
N\A
N\A
N\A
N\A
No
Swelling, Shoulder pain
N\A
N\A
N\A
N\A
No
Avci et al.
[48]
2005
1
Chest pain
N\A
9
N\A
Hematoma
No
* Less than a month
Table 3.
Management and outcome.
Author/reference
Year
No.
of Case
Surgical approach
Scolicidal agent
Site of involvement
Post-operative medication
Complication
Recurrence
Follow-up (month)
Althobaity et al.
[1]
2023
1
Video-assisted thoracoscopy [VATS]
Hypertonic saline
N\A
Albendazole
Pneumothorax
No
5
Goyal et al.
[2]
2010
1
Thoracotomy
Hypertonic saline
Rib and intercostal muscle
No
No
N\A
N\A
Godazandeh et al.
[3]
2020
1
N\A
N\A
N\A
Albendazole
No
No
24
Basit et al.
[5]
2021
1
N\A
Hypertonic saline
N\A
Albendazole
No
N\A
N\A
Salih et al.
[6]
2017
1
N\A
N\A
N\A
N\A
No
No
3
Döner et al.
[11]
2019
1
N\A
N\A
Intercostal muscle
Albendazole
No
N\A
N\A
MohIeldeen et al.
[13]
2013
1
N\A
N\A
8th rib
Albendazole
No
No
24
Akkas et al.
[14]
2016
2
Thoracotomy
N\A
Intercostal muscle
Albendazole
No
No
36
Thoracotomy
N\A
N\A
Albendazole
No
No
18
Alloubi et al.
[15]
2012
1
Thoracotomy
N\A
6th and 7th ribs
Albendazole
No
No
6
Al-Qudah et al.
[16]
2000
1
Thoracotomy
N\A
1st rib
No
No
N\A
N\A
Afghani et al.
[17]
2017
1
N\A
N\A
N\A
Albendazole
No
No
24
Yekeler et al.
[18]
2010
1
Thoracotomy
N\A
6th rib
Albendazole
No
No
10
Ulger et al.
[19]
2013
1
N\A
N\A
Sternum
No
N\A
No
12
Tulay et al.
[20]
2015
1
N\A
Hypertonic saline
N\A
Albendazole
N\A
No
6
Tomos et al.
[21]
2005
1
Thoracotomy
N\A
6th rib
No
N\A
N\A
N\A
Tezcan et al.
[22]
2014
1
N\A
N\A
Sternum
No
No
N\A
N\A
Tadasa et al.
[23]
2023
1
No
N\A
N\A
No
N\A
N\A
N\A
Sevinc et al.
[24]
2014
1
N\A
Hypertonic saline
N\A
Albendazole
No
N\A
N\A
Sarkar et al.
[25]
2015
1
N\A
N\A
N\A
Albendazole
No
No
12
Sabzi et al.
[26]
2023
1
N\A
N\A
N\A
Albendazole
No
N\A
N\A
Roman et al.
[28]
2015
1
Thoracotomy
Formalin
8th rib
Albendazole, Chemotherapy
No
No
1
Redington et al.
[29]
2001
1
No
N\A
N\A
No
N\A
N\A
N\A
Raut et al.
[30]
2004
1
N\A
N\A
9th rib
Albendazole
No
N\A
N\A
Ninos et al.
[31]
2010
1
N\A
N\A
Intercostal muscle
Albendazole
N\A
No
12
Kiliç et al.
[32]
2003
1
Thoracotomy
N\A
5th, 6th and 7th ribs
Albendazole
No
N\A
N\A
Karapolat et al.
[33]
2012
1
N\A
N\A
N\A
Albendazole
No
No
12
Karaoğlanoğlu et al.
[34]
2001
1
N\A
N\A
5th and 6th ribs, Serratus anterior muscle, Latissimus dorsi muscle
Albendazole
No
No
6
Kaplanoğlu et al.
[35]
2017
1
N\A
N\A
Intercostal muscle
No
N\A
No
12
Honda et al.
[36]
2010
1
N\A
Hypertonic saline
N\A
Albendazole
N\A
No
16
Han et al.
[37]
2004
1
N\A
N\A
7th rib
Albendazole
N\A
N\A
N\A
Gezer et al.
[38]
2006
1
Thoracotomy
N\A
1st rib
Albendazole
No
No
15
Foroulis et al.
[39]
2003
1
Thoracotomy
Hypertonic saline
10th vertebra, 9th and 10th ribs
Albendazole
No
No
48
Findikcioglu et al.
[40]
2010
1
Thoracotomy
N\A
Riband vertebra
No
N\A
N\A
N\A
Faber et al.
[41]
2010
1
N\A
N\A
1st rib
No
No
N\A
N\A
Demir et al.
[42]
2010
1
N\A
N\A
N\A
Albendazole
N\A
N\A
N\A
Chafik et al.
[43]
2009
1
Thoracotomy
N\A
N\A
Albendazole
No
N\A
N\A
Sinberg et al.
[44]
1936
1
N\A
Hypertonic saline
Sternum
No
Slight superficial infection
N\A
N\A
Rose et al.
[45]
1893
1
N\A
N\A
N\A
No
Hemorrhage
N\A
N\A
Machboua et al.
[46]
2023
5
Thoracotomy
N\A
N\A
No
Pleurocutaneous fistula, Disabling parietal pain and neurological pain, Disorder of the Shoulder Girdle.
No
24
Thoracotomy
N\A
Rib and vertebra
No
No
N\A
N\A
N\A
N\A
N\A
No
No
N\A
N\A
Thoracotomy
N\A
N\A
No
No
N\A
N\A
Thoracotomy
N\A
N\A
No
N\A
N\A
N\A
Özdemir et al.
[47]
1994
6
N\A
N\A
N\A
No
N\A
N\A
N\A
N\A
N\A
N\A
No
N\A
N\A
N\A
Thoracotomy
N\A
N\A
No
N\A
N\A
N\A
Thoracotomy
N\A
N\A
No
N\A
N\A
N\A
N\A
N\A
N\A
No
N\A
N\A
N\A
N\A
N\A
N\A
No
N\A
N\A
N\A
Avci et al.
[48]
2005
1
N\A
N\A
N\A
No
No
N\A
N\A
N/A:non-available
Main findings
The reported cases were primarily from Turkey (41.
5%).
The mean age of the patients was 39.
7 ± 17.
1 years, with a male predominance (56.
9%).
The past medical history for HC was positive in 10 cases (19.
6%).
The most common clinical presentations were swelling in 24 cases (47.
1%) and chest or abdominal pain in 23 cases (45.
1%).
Only 10 cases (19.
6%) had reported a history of animal contact, while one patient (2%) reported no such history.
Data on the history of animal contact was unavailable in 78.
4% of cases.
Among those with documented residency (35.
3%), 16 (31.
4%) resided in rural areas, while two (3.
9%) were from urban settings.
The disease was primary in sixteen cases (31.
4%), and in nine (17.
6%), it was secondary.
The disease origin was unknown in 26 cases (51%).
The average mass size on the CT scan was 7.
5 ± 2.
4 cm.
Hydatid serology was performed in 18 patients, yielding positive results in nine (50%).
A provisional diagnosis of hydatid disease was considered in 14 cases (27.
5%).
Pre-operative treatment with albendazole was administered to eight patients (15.
7%).
Surgery was the treatment of choice, with thoracotomy performed in 37.
3% of cases, video-assisted thoracoscopy (VATS) in 1.
9%, and the surgical technique not specified in 60.
8% of cases.
The average hospital stay was 8.
6 ± 4.
4 days.
The ribs (31.
4%) were the primary involved sites, followed by the muscles (11.
8%).
Postoperative medication included albendazole in 25 cases (49%) and albendazole with chemotherapy in one case (1.
9%).
Twenty-four cases (47.
1%) had received no postoperative treatment.
Only four cases (7.
8%) faced complications.
No recurrence was reported after a mean follow-up period of 14.
1± 10.
3 months (Table 4).
Table 4.
Summary and baseline characteristics of the included studies.
Variables
Frequency/percentage
Mean Age (years)
39.
7 ± 17.
1
Sex
Male
Female
29 (56.
9%)
22 (43.
1%)
Country of study
Turkey
Morocco
Greece
India
Iran
Iraq
United Kingdom
Afghanistan
Ethiopia
Israel
Japan
Jordan
Romania
Saudi Arabia
United States
17 (41.
5%)
3 (7.
3%)
3 (7.
3%)
3 (7.
3%)
3 (7.
3%)
2 (5.
0%)
2 (5.
0%)
1 (2.
4%)
1 (2.
4%)
1 (2.
4%)
1 (2.
4%)
1 (2.
4%)
1 (2.
4%)
1 (2.
4%)
1 (2.
4%)
Past medical history of hydatid cyst
Positive
Negative
10 (19.
6%)
41 (80.
4%)
Contact with an animal
Yes
No
N\A
10 (19.
6%)
1 (2.
0%)
40 (78.
4%)
Residency
Rural
Urban
N\A
16 (31.
4%)
2 (3.
9%)
33 (64.
7%)
Origin
Primary
Secondary
N\A
16 (31.
4%)
9 (17.
6%)
26 (51.
0%)
Size of the mass on CT scan
7.
5 ± 2.
4 cm
Presentation
Swelling
Chest/Abdominal pain
Dyspnea
Cough
Shoulder/back pain
Numbness
Others
N\A
24 (47.
1%)
23 (45.
1%)
4 (7.
8%)
4 (7.
8%)
9 (17.
6%)
5 (9.
8%)
6 (11.
8%)
4 (7.
8%)
Hydatid serology test
Positive
Negative
N\A
9 (17.
6%)
9 (17.
6%)
33 (64.
7%)
Provisional diagnosis
Hydatid disease
Chest wall tumor
Aneurysmal bone cyst
Ewing sarcoma
Hematoma
N\A
14 (27.
5%)
3 (5.
9%)
1 (1.
9%)
1 (1.
9%)
1 (1.
9%)
31 (60.
8%)
Pre-operative medication
Albendazole
No
8 (15.
7%)
43 (84.
3%)
Surgical approach
Thoracotomy
Video-assisted thoracoscopy
N\A
19 (37.
3%)
1 (1.
9%)
31 (60.
8%)
Injection of scolicidal agent with surgery
Hypertonic saline
Formalin
No
8 (15.
7%)
1 (1.
9%)
42 (82.
4%)
Site of involvement
Rib
Muscle
Sternum
Vertebra
N\A
16 (31.
4%)
6 (11.
8%)
3 (5.
9%)
3 (5.
9%)
28 (54.
9%)
Post-operative medication
Albendazole
Albendazole and chemotherapy
No
N/A
25 (49.
0%)
1 (1.
9%)
24 (47.
1%)
1 (1.
9%)
Mean postoperative hospital stay (days)*
8.
6 ± 4.
4
Outcome of surgery
Recovered with no complication
Recovered with complication
N\A
29 (56.
9%)
4 (7.
8%)
18 (35.
3%)
Average duration of follow-up (months)
14.
1± 10.
3
Recurrence
Yes
No
N\A
0 (0.
0%)
21 (41.
0%)
30 (59.
0%)
N/A: non-available, CT: computed tomography.
*For 12 patients out of 51 patients who underwent surgery.
#For 23 patients out of 51 patients who underwent surgery.
Discussion
Human echinococcosis, a prevalent parasitic infection, presents a considerable health and economic burden to society, yet it remains largely neglected as a disease [49].
The findings of this study provide a comprehensive review of chest wall HC, with the majority of the studies conducted in Turkey (41.
5%), followed by Morocco (7.
3%), Greece (7.
3%), India (7.
3%), and Iran (7.
3%).
The higher incidence in these countries may reflect the endemic nature of hydatid disease, likely due to closer contact with livestock and domestic animals [50].
However, it has become a pressing global health issue, primarily attributed to increasing immigration rates and travel activities [7].
The mean age of 39.
7 years, with a slight male predominance (56.
9%), suggests that both demographic groups are at risk.
However, occupational or environmental exposure factors could contribute to the observed gender disparity.
Clinically, the most common presentation was swelling (47.
1%), followed by chest or abdominal pain (45.
1%).
This indicates that chest wall HCs may present as localized symptoms rather than systemic manifestations [51].
The relatively low percentage (19.
6%) of patients with a history of animal contact suggests that direct exposure may not always be reported or remembered, or that indirect exposure through contaminated water or food could also be significant transmission routes [26,27].
In addition, a review by Possenti and colleagues suggested that the primary route of human cystic echinococcosis transmission may be the direct or indirect contamination of hands with Echinococcus granulosus eggs excreted by dogs [52].
Schantz et al.
indicated that individuals who own livestock are three times more likely to be diagnosed with this disease compared to those who do not own livestock [53].
In accordance with this finding, 16 (88.
9%) of the 18 cases with known residency were from rural areas.
Ten cases (19.
6%) had a positive history for HC at other sites, and the primary origin of cysts in 31.
4% of patients and secondary origin in 17.
6% highlights the importance of considering both primary infection or possible spread from other sites, such as the liver or lungs, as the diagnosis and treatment strategies may be different [1].
In the present study, the average cyst size on the CT scan was 7.
5 cm, reflecting potentially large cysts that could cause significant local pressure effects or complications; this might be because the course of infection is slow, and most infected individuals remain either asymptomatic for years or exhibit non-specific symptoms leading to accidental diagnosis [54].
Parasite eggs can remain viable from several months up to a few years in the environment, and diverse conditions [55].
Only nine (50%) out of 18 patients with available data in this systematic review had positive hydatid serology, suggesting that serological tests alone may not be sufficient for diagnosis and imaging, particularly CT scans, plays a crucial role in the diagnostic process [56].
The provisional diagnosis of hydatid disease in only 27.
5% of the cases underscores the diagnostic challenges faced by clinicians, especially when the clinical presentation is atypical or when imaging findings are inconclusive [57].
The importance and priority of infection prevention by practicing several preventive actions, especially in developing countries, has been proposed.
The most significant of them included self-hygiene practices, proper washing and cooking of foods, and avoiding eating raw foods or groceries [27].
Regarding management, surgical intervention was the primary treatment modality [1,2,7,58].
A study indicated that while VATS has been successfully applied in managing pulmonary HCs, its adoption remains limited.
It has been pointed out that many surgeons are still hesitant to use VATS due to a lack of familiarity with the technique and concerns about potential complications, such as cyst rupture or spillage of infectious material [59].
In this review, authors came across similar findings, such as thoracotomy being performed in 37.
3% of cases and VATS being performed in 1.
9%.
This may depend on cyst size and location.
Furthermore, the low utilization of minimally invasive approaches like thoracoscopy may suggest either a preference for traditional open techniques or a lack of resources or expertise in minimally invasive surgery in some settings.
The postoperative outcomes were generally favorable, with only 7.
8% of patients facing complications, and no recurrence was reported, emphasizing the effectiveness of surgical management.
Machboua et al.
on intra-thoracic extra-pulmonary hydatidosis noted that the average duration of hospitalization was only seven days, which is similar to what was found in this study (8.
58 ± 4.
44 days) [46].
The administration of post-operative albendazole alone (49%) or in combination with other medications (1.
9%) may appear to be a standard practice to prevent recurrence, even though no recurrences were reported in this study.
This could reflect a preventive strategy against the possible dissemination of HCs during surgery [1,14,15,17,18].
Overall, the study may provide valuable insights into the epidemiology, clinical presentation, diagnostic strategies, and management outcomes of chest wall HCs; however, the lack of essential data in a number of reviewed studies might generate biased or non-conclusive findings.
Conclusion
Despite its rarity, chest wall HC may have a good prognosis with few complications.
Given its often-nonspecific presentation, reviewing the patient's medical history may help establish an accurate provisional diagnosis.
Declarations
Conflicts of interest: The authors have no conflicts of interest to disclose.
Ethical approval: Not applicable, as systematic reviews do not require ethical approval.
Patient consent (participation and publication): Not applicable.
Funding: The present study received no financial support.
Acknowledgements: None to be declared.
Authors' contributions: FHK and BAA were significant contributors to the conception of the study and the literature search for related studies.
HOA and DHH involved in the literature review, study design, and manuscript writing.
HKA, AHH, AHA, AAM, SMA, SOK, FA, SSA, and SHM were involved in the literature review, the study's design, the critical revision of the manuscript, and data collection.
FHK and HOA confirm the authenticity of all the raw data.
All authors approved the final version of the manuscript.
Use of AI: ChatGPT-3.
5 was used to assist in language editing and improving the clarity of the manuscript.
All content was reviewed and verified by the authors.
Authors are fully responsible for the entire content of their manuscript.
Data availability statement: Not applicable.
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