Review Article


Presentation and management of hydatid cyst of the thigh: A systematic review of literature

,  ,  ,  ,  ,  ,  

1 Faculty of Medical Sciences, School of Medicine, Department Surgery, University of Sulaimani, François Mitterrand Street, Sulaimani,, Kurdistan Region, Iraq

2 Faculty of Medical Sciences, School of Medicine, Department Cardiothoracic and Vascular Surgery, University of Sulaimani, Sulaimani, Kurdistan, Iraq

3 Kscien Organization for Scientific Research, Sulaimani, Kurdistan, Iraq

Address correspondence to:

Fahmi H. Kakamad

Faculty of Medical Sciences, School of Medicine, Department Cardiothoracic and Vascular Surgery, University of Sulaimani, Sulaimani, Kurdistan,

Iraq

Message to Corresponding Author


Article ID: 100957Z01AS2018

doi: 10.5348/100957Z01AS2018RA

Access full text article on other devices

Access PDF of article on other devices

How to cite this article

Salih AM, Kakamad FH, Mohammed SH, Baba HO, Mohammed KK, Ahmmad DR, Kakamad SH. Presentation and management of hydatid cyst of the thigh: A systematic review of literature. Int J Case Rep Images 2018;9:100957Z01AS2018.

ABSTRACT


Hydatid cyst of the thigh is an extremely rare variant of the condition. The aim of this study is to systematically review the reported cases of hydatid cyst of the thigh. Science Direct, Web of Science, Medline on OVID, PubMed and Google scholar were scrutinized for articles which at least contain one new case of hydatid cyst of the thigh. Sixty-four patients were collected and discussed. In conclusion, hydatid cyst of the thigh is a very rare parasitic manifestation presenting as a painless swelling, diagnosed typically by magnetic resonance imaging and managed with en bloc resection.

Keywords: Hydatid cyst, Parasite, Thigh

INTRODUCTION


Echinococcus, a cestode of the Taeniidae family although lives outside the human body, causes a cystic parasitic infestation in mankind [1],[2]. The organism possesses both definitive and intermediate hosts. Wolves, dogs, and foxes (the definitive hosts) pass the worm’s eggs in their excretes into the surroundings, subsequently, the intermediate hosts (sheep, cattle, humans, horses, goats, and camel) develop hydatidosis [3]. The organs which are most incessantly affected by the disease are liver and lung, the latter is more common in the pediatric age group [4]. Other seldom affected organs are skeletal and smooth muscles, bone, viscera, and mediastinum [5]. Hydatid cysts (HCs) of the thigh is a very rare variant of the parasite manifestation, still, it occurs in endemic areas of the Middle East [6]. Presentation of the thigh HC, epidemiology, its clinical courses, diagnosis, and management are not well described. As there is no summary paper about thigh HC, this study was attempted to systematically review the reported cases of HC of the thigh worldwide.

METHODS


Search and information sources

Science direct, Web of Science, Medline on OVID, PubMed, Elsevier, Scopus, Wiley online and Google scholar were scrutinized for English-language articles distributed before 2018. The keywords were thigh hydatid cyst, thigh hydatidosis, hydatid cyst of the thigh, Echinococcosis of thigh. The data assemblage was enhanced by references from the included articles (Figure 1).

Eligibility criteria

For an article to be considered for this review, it had to include at least one new case of HC of the thigh. Exclusion criteria was articles without atleast one new case of HC of the thigh.

Data assemblage and review process

Data were excerpted from the included studies by two authors independently (second and fourth authors). Authors of the included articles have not been contacted to get and confirm the data. Several data were quoted and some of them were pooled including socio-demographic characteristics of the patients, sample size, presentations, duration of presentation, the method of diagnosis, options for management, recurrence rate and complications.

Summary measures and synthesis of results

Some data were calculated and re-analyzed. According to the variables, they were demonstrated as the percentage, mean values, and ranges of variation and percentages. Some other papers were illustrated and summarized in brief.

Figure 1: Flow chart of the included articles.

Share Image:

RESULTS


The search through literature found 70 papers. From which 20 papers were excluded by titles. Other three papers were ostracized either because of scanty information or incomplete report. Two papers were omitted as a result of failure to be retrieved. The remaining 45 papers were analyzed and 64 patients were collected (Table 1). Thirty-seven of them (57.8%) were females, 27 (43.2%) were males. The age of the patients ranged from eight to 83 years with a mean age of 32.6 years. Almost all the patient presented with slow-growing mass. One of the patients presented with signs and symptoms of abscess (erythema, tender and swelling) [7]. The duration of the presentation was variable ranging from three days to 30 years with a mean period of the presentation was two years. The left side was affected more commonly than the right side (41/58, 70.6%). All patients received antihelminthics from the time of diagnosis (pre or postoperatively) until a variable length of time. All of the cases were managed by total resection. Three of the patients (4%) had HC other than thigh HC [8],[9],[10]. Two of them had retroperitoneal HCs another case had liver HC.

Table 1: Sociodemographic and clinical summary of the included articles

Share Image:

DISCUSSION


The prevalence of thigh HCs is not notorious. Apart from two case series studies, all cases of thigh HCs accessible in the literature are case reports [11],[12]. Ammari and his colleagues published the registry of 13 years with nine cases of thigh HCs, seven patients were female, their mean age was 37 years [12]. Madhar et al presented seven cases of thigh HCs in a solitary paper from which six patients were female and their ages ranged from 19 to 56 years with a mean age of 30 years [11]. Thigh HCis either primary or secondary. In the latter, HCs should disturb concomitantly at least one of the common sites like liver, lung or spleen. Acu and his associates disclosed a case of thigh HC with concomitant HC of the left lobe of the liver occurring in a 20-year-female patient [8]. Sarda and his companions reported a 60-year-female with secondary HC of the thigh, the patient presented with swelling of the thigh with a palpable mass in the suprapubic region, during operation, they realized that there were two HCs. One of the thigh and other in the retroperitoneum communicating with each other via sub-facial plane [9]. In endemic countries, the disease might be prevented by several mechanisms including regular supplementation of praziquantel to the hosts, preventing dog’s access to intermediate hosts, vaccinating sheep, boiling or safe disposal of offal [13]. Up to date, 63 patients of thigh HCs have been reported in the medical literature [6],[7],[8], [9], [10], [11], [12], [13], [14], [15], [16], [17], [18], [19], [20], [21], [22], [23], [24], [25], [26], [27], [28], [29], [30], [31], [32], [33], [34], [35], [36], [37], [38], [39], [40], [41], [42], [43], [44], [45], [46], [47], [48], [49], [50],[51],[52],[53] The age of the cases ranged from 8 to 83 years [4],[23].

The prevalence of thigh HCs is not notorious. Apart from two case series studies, all cases of thigh HCs accessible in the literature are case reports [11],[12]. Ammari and his colleagues published the registry of 13 years with nine cases of thigh HCs, seven patients were female, their mean age was 37 years [12]. Madhar et al presented seven cases of thigh HCs in a solitary paper from which six patients were female and their ages ranged from 19 to 56 years with a mean age of 30 years [11]. Thigh HCis either primary or secondary. In the latter, HCs should disturb concomitantly at least one of the common sites like liver, lung or spleen. Acu and his associates disclosed a case of thigh HC with concomitant HC of the left lobe of the liver occurring in a 20-year-female patient [8]. Sarda and his companions reported a 60-year-female with secondary HC of the thigh, the patient presented with swelling of the thigh with a palpable mass in the suprapubic region, during operation, they realized that there were two HCs. One of the thigh and other in the retroperitoneum communicating with each other via sub-facial plane [9]. In endemic countries, the disease might be prevented by several mechanisms including regular supplementation of praziquantel to the hosts, preventing dog’s access to intermediate hosts, vaccinating sheep, boiling or safe disposal of offal [13]. Up to date, 63 patients of thigh HCs have been reported in the medical literature [6],[7],[8], [9], [10], [11], [12], [13], [14], [15], [16], [17], [18], [19], [20], [21], [22], [23], [24], [25], [26], [27], [28], [29], [30], [31], [32], [33], [34], [35], [36], [37], [38], [39], [40], [41], [42], [43], [44], [45], [46], [47], [48], [49], [50],[51],[52],[53] The age of the cases ranged from 8 to 83 years [4],[23].

CONCLUSION


Hydatid cyst of the thigh is a very rare parasitic manifestation presenting as a painless swelling, diagnosed typically by MRI and managed with en bloc resection.

REFERENCE


1.

Salih AM, Kakamad FH, Hammood ZD, Yasin B, Ahmed DM. Abdominal wall hydatid cyst: A review a literature with a case report. Int J Surg Case Rep 2017;37:154–6. [CrossRef] [Pubmed]   Back to citation no. 1  

2.

Mustafa HM, Muhammad NH, Kakamad FH, Essa RA, Rahem HM. Hydatid disease causing paraplegia: A case report with literature review. Edorium J Infect Dis 2017;3:17–20. [CrossRef]   Back to citation no. 1  

3.

Salih AM, Ahmed DM, Kakamad FH, Essa RA, Hunar AH, Ali HM. Primary chest wall hydatid cyst: Review of literature with report of a new case. Int J Surg Case Rep 2017 Nov 8;41:404–6.   Back to citation no. 1  

4.

Salih AM, Kakamad FH, Rauf GM. Isolated hydatid cyst of the diaphragm, a case report. Int J Surg Case Rep 2016;29:130–2. [CrossRef] [Pubmed]   Back to citation no. 1  

5.

Baram A, Kakamad FH, Alwan AA. Primary posterior mediastinal hydatid cyst mimicking malignant mediastinal neurogenic tumor. International Journal of Case Reports and Images 2014;5(1):54–7.   Back to citation no. 1  

6.

Argy N, Abou Bacar A, Boeri C, et al. Primary musculoskeletal hydatid cyst of the thigh: Diagnostic and curative challenge for an unusual localization. Can J Infect Dis Med Microbiol 2013 Fall;24(3):e99–e101. [Pubmed]   Back to citation no. 1  

7.

Madhar M, Aitsoultana A, Chafik R, Elhaoury H, Saidi H, Fikry T. Primary hydatid cyst of the thigh: on seven cases. Musculoskelet Surg 2013 Apr;97(1):77–9. [CrossRef] [Pubmed]   Back to citation no. 1  

8.

Ammari T, Zrig M, Annabi MR, et al. Hydatid Cyst of The Thigh: 8 Cases. The Journal of Bone and Joint Surgery British volume 2005;87-B.   Back to citation no. 1  

9.

Acu L, Acu B, Acu R, Sahin SA. Hydatid cyst presented as a subcutaneous thigh mass. J Ultrasound 2016 Apr 8;20(4):321–4. [CrossRef] [Pubmed]   Back to citation no. 1  

10.

Sarda DK, Lohiya SJ, Jawandiya VG, Kamble AT. Migration of retroperitoneal hydatid cyst into the thigh: A rare presentation. Indian Journal of Surgery 2006;68:5.   Back to citation no. 1  

11.

Abebe AA. Hydatid Cyst of the Left Thigh: A case report. East and Central African Journal of Surgery 2010;15(1):139–40.   Back to citation no. 1  

12.

Garagnani L, Sudanese A, Rimondi E, Bacchini P. Primary hydatid cyst of the root of the thigh: A case report and review of the literature. Chir Organi Mov 2008 Sep;92(2):113–8. [CrossRef] [Pubmed]   Back to citation no. 1  

13.

Bansiwal RK, Sharma R, Attri AK. A large primary hydatid cyst of thigh: A case report. Indian J Surg 2011 Apr;73(2):158–60. [CrossRef] [Pubmed]   Back to citation no. 1  

14.

Kayikçioglu A, Karamürsel S. Primary hydatidosis of the thigh. Plast Reconstr Surg 1999 Dec;104(7):2341–2. [Pubmed]   Back to citation no. 1  

15.

Salamone G, Licari L, Randisi B, et al. A primary subcutaneous hydatid cyst in the thigh. A case report. Ann Ital Chir 2014;85(ePub):pii/S2239253X14022208. [Pubmed]   Back to citation no. 1  

16.

Arslan M, Gulek B, Ogur HU, Adamhasan F. Primary hydatid cyst in the posterior thigh, and its percutaneous treatment. Skeletal Radiol 2018 Oct;47(10):1437– 42. [CrossRef] [Pubmed]   Back to citation no. 1  

17.

Barkati S, Butler-Laporte G, Ndao M, et al. A recurrent hydatid cyst of the thigh diagnosed 13 years after initial presentation. IDCases 2017 Nov 23;11:12– 15. [CrossRef] [Pubmed]   Back to citation no. 1  

18.

Bothale KA, Kolhe H, Mahore SD, Wilkinson AR. Diagnosis of primary hydatid cyst of thigh by fine needle aspiration cytology. Indian J Med Microbiol 2015 Jan–Mar;33(1):151–3. [CrossRef] [Pubmed]   Back to citation no. 1  

19.

Bouomrani S, Nouma H, Slama A, Béji M. Primary hydatid cyst of the thigh: An unusual location. European Journal of Internal Medicine 2013;24:201.   Back to citation no. 1  

20.

Chevalier X, Rhamouni A, Bretagne S, Martigny J, Larget-Piet B. Hydatid cyst of the subcutaneous tissue without other involvement: MR imaging features. AJR Am J Roentgenol 1994 Sep;163(3):645–6. [CrossRef] [Pubmed]   Back to citation no. 1  

21.

Dirican A, Unal B, Kayaalp C, Kirimlioglu V. Subcutaneous hydatid cysts occurring in the palm and the thigh: Two case reports. J Med Case Rep 2008 Aug 13;2:273. [CrossRef] [Pubmed]   Back to citation no. 1  

22.

Duygulu F, Karaoglu S, Erdogan N, Yildiz O. Primary hydatid cyst of the thigh: A case report of an unusual localization. Turk J Pediatr 2006 Jul-Sep;48 (3):256–9. [Pubmed]   Back to citation no. 1  

23.

Goel A, Dewanda NK. Hydatid cyst of thigh: A rare presentation. Journal of Evolution of Medical and Dental Sciences 2013;2(40):7761–4.   Back to citation no. 1  

24.

Gupta A, Singal RP, Gupta S, Singal R. Hydatid cyst of thigh diagnosed on ultrasonography-a rare case report. J Med Life 2012 Jun 12;5(2):196–7. [Pubmed]   Back to citation no. 1  

25.

Ibrahem SY. Rare presentation of thigh hydatid cyst (Case report). Diyala Journal of Medicine 2013;4(1):125–7.   Back to citation no. 1  

26.

Kazmi Z, Qureishi S, Quraishy MS, Mallick FA, Rizvi S. Atypical presentation of hydatid cyst in the thigh. J Coll Physicians Surg Pak 2017 Jan;27(1):51– 52. [Pubmed]   Back to citation no. 1  

27.

Koc Z, Agildere AM, Yalcin O, Pourbagher A, Pourbagher M. Primary hydatid cyst in the anterior thigh: Sonographic findings. Clin Ultrasound 2004 Sep;32 (7):358–60. [CrossRef] [Pubmed]   Back to citation no. 1  

28.

Kocakusak A, Koyuncu A, Arikan S, Senturk O. Primary hydatid cyst of vastus lateralis muscle. Acta Chir Belg 2004 Aug;104(4):471–2. [Pubmed]   Back to citation no. 1  

29.

Landolsi M, Kouki S, Abdennadher A. Hydatid cyst of the thigh: A challenging diagnosis. BMJ Case Rep 2017 Oct 11;2017. [CrossRef] [Pubmed]   Back to citation no. 1  

30.

Mahmoudi A, Rachidi CA, Khattala K, Chater L, Bouabdallah Y, Afifi MA. Primary subcutaneous hydatid cyst in the left distal thigh. Eur J Orthop Surg Traumatol 2012 Nov;22 Suppl 1:177–80. [CrossRef] [Pubmed]   Back to citation no. 1  

31.

Marwah S, Subramanian P, Marwah N, Rattan KN, Karwasra RK. Infected primary intramuscular echinococcosis of thigh. Indian J Pediatr 2005 Sep;72(9):799–800. [Pubmed]   Back to citation no. 1  

32.

Mourafiq O, Echchaoui A, Chafri B, Bouabid AS, Benchebba D, Boussouga M. Primary hydatid cyst: An unusual cause of a mass in the thigh. BMH Medical Journal 2017;4(4):139–41.   Back to citation no. 1  

33.

Pathak TK, Roy S, Das S, Achar A, Biswas AK. Solitary hydatid cyst in thigh without any detectable primary site. J Pak Med Assoc 2011 Dec;61(12):1244–5. [Pubmed]   Back to citation no. 1  

34.

Pawar I, Mittal A, Bugga P, Aggarwal A. Large hydatid cyst in thigh: A rare case with clinico-radio-pathological profile. Journal of musculoskeletal research 2010;13(03):153–7.   Back to citation no. 1  

35.

Perelman H, Gottlieb L. Hydatid cyst in muscle of the thigh. Calif Med 1961 Feb;94:101–3. [Pubmed]   Back to citation no. 1  

36.

Rodrigues G, Prabhu R. Primary subcutaneous hydatid cyst of the thigh: An unusual site with a diagnostic dilemma. J Cutan Med Surg 2016 May;20(3):266–8. [CrossRef] [Pubmed]   Back to citation no. 1  

37.

Abi Saad GS, Musallam KM, Korban ZR, Reslan OM, Mneimne M. Solitary hydatid cyst of the thigh: A challenging diagnosis. Vector Borne Zoonotic Dis 2009 Dec;9 (6):743–5. [CrossRef] [Pubmed]   Back to citation no. 1  

38.

Shaikh R. Primary musculoskeletal hydatid cyst of thigh. Journal of Islamabad Medical & Dental College 2017;6(2):113–5.   Back to citation no. 1  

39.

Thursky K, Torresi J. Primary muscle hydatidosis of the thigh: Management of a complicated case with combination adjunctive albendazole and praziquantel chemotherapy. Clin Infect Dis 2001 Feb 1;32(3):E65–8. [CrossRef] [Pubmed]   Back to citation no. 1  

40.

La Greca G, Pulvirenti E, Gagliardo S, Sofia M, Russello D. Muscle-sparing approach for recurrent hydatidosis of the thigh and psoas: Report of a rare case. PLoS Negl Trop Dis 2011 Jan 25;5(1):e840. [CrossRef] [Pubmed]   Back to citation no. 1  

41.

Cissé AM, Nassar I, Hammani L, Dafiri R, Imani F. Primary extensive hydatidosis of the thigh: Unusual radiological aspects. [Article in French]. J Radiol 2002 Nov;83(11):1778–80. [Pubmed]   Back to citation no. 1  

42.

Farzam RA, Ghaempanah A, Feizy A. Intramuscular hydatid cyst of the thigh: A case presentation. Journal of Zanjan university of medical sciences & health services 2010;18:73.   Back to citation no. 1  

43.

Mortazavi SM, Motamedi M, Asadollahi S, Haeri H, Moghtadaee M. Primary intermuscular hydatid cyst of the thigh. Orthopedics 2008 Jan;31(1):85. [Pubmed]   Back to citation no. 1  

44.

Bagatur AE, Ugur F, Zorer G. Primary giant hydatid cyst in the thigh. [Article in Turkish]. Acta Orthop Traumatol Turc 2002;36(1):72–5. [Pubmed]   Back to citation no. 1  

45.

Atmatzidis K, Koutelidakis I, Papaziogas B, Primary hydatid cyst of the thigh. Chirurgia (Bucur) 2006 Jul–Aug;101(4):419–21. [Pubmed]   Back to citation no. 1  

46.

Sipahioglu S, Koçarslan S, Zehir S. Primary hydatid cyst in the medial thigh: Case report. [Article in Turkish]. Eklem Hastalik Cerrahisi 2014;25(3):168–72. [CrossRef] [Pubmed]   Back to citation no. 1  

47.

Hammami T, Noomane F, Ketata M, et al. Hydatid cyst of the thigh: Three cases. [Article in French]. Rev Chir Orthop Reparatrice Appar Mot 2002 Apr;88 (2):193–6. [Pubmed]   Back to citation no. 1  

48.

Ekinci Y, Duygulu F, Vatansever F, Gürbüz K. A giant hydatid cyst localized in pelvis and thigh. [Article in Turkish]. Eklem Hastalik Cerrahisi 2014;25 (2):121–4. [CrossRef] [Pubmed]   Back to citation no. 1  

49.

Sakka SA. Primary hydatid cyst in the thigh with pain manifestation at the hip level. [Article in French]. Rev Chir Orthop Reparatrice Appar Mot 1993;79 (3):226–8. [Pubmed]   Back to citation no. 1  

50.

Ghoroobi J, Mohajerzadeh L, Mirshemirani A, Mahdavi A. Primary hydatid cyst of thigh: A case report in child. Journal of Krishna Institute of Medical Sciences (JKIMSU) 2017;6(4):111–3.   Back to citation no. 1  

51.

Yalavarthi S, Satya NV, Ramamurti T, Supriya M. Intermuscular hydatid cyst in the thigh: An unusual presentation. Medical Journal of Dr. DY Patil Vidyapeeth 2013;6(2):191. [CrossRef]   Back to citation no. 1  

52.

Lotfi SR, Hamidi A, Araba Z. Primary intramuscular hydatid cyst of thigh, a case report. Police Medicine 2012;1(2):137–41.   Back to citation no. 1  

53.

Salih AM, Kakamad FH, Salih RQ, et al. Hydatid cyst of the thigh; a case report with literature review. Int J Surg Case Rep 2018 Aug 11;51:8–10. [CrossRef] [Pubmed]   Back to citation no. 1  

SUPPORTING INFORMATION


Author Contributions

Abdulwahid M. Salih - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Fahmi H. Kakamad - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Shvan H. Mohammed - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Hiwa O. Baba - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Karukh K. Mohammed - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Dlshad R. Ahmmad - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Suhaib H. Kakamad - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Guaranter of Submission

The corresponding author is the guarantor of submission.

Source Of Support

None

Data Availability

All relevant data are within the paper and its Supporting Information files.

Conflict of Interest

Authors declare no conflict of interest.

Copyright

© 2018 Abdulwahid M. Salih et al. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.


Comment on Article