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Multiple hydatid cysts in rare locations treated with albendazole: A case report
Somayeh Sadeghi1, Farzin Ghiasi1, Amir Aria2, Mahnaz Momenzadeh3, Elahe Nasri4, Shadi Reisizadeh Mobarakeh2
1 Acquired Immunodeficiency Research Center, Al-Zahra Hospital; Department of Internal Medicine, Alzahra Hospital, Isfahan University of Medical Sciences, Isfahan, Iran
2 Department of Internal Medicine, Alzahra Hospital, Isfahan University of Medical Sciences, Isfahan, Iran
3 Department of Clinical Pharmacy and Pharmacy Practice, School of Pharmacy and Pharmaceutical Sciences, Isfahan, Iran
4 Infectious Diseases and Tropical Medicine Research Center, Isfahan University of Medical Sciences, Isfahan, Iran
| Date of Submission | 23-Nov-2022 |
| Date of Acceptance | 04-Jan-2023 |
| Date of Web Publication | 20-Jul-2023 |
Correspondence Address:
Dr. Shadi Reisizadeh Mobarakeh
Department of Internal Medicine, Alzahra Hospital, Isfahan University of Medical Sciences, Isfahan
Iran
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/abr.abr_400_22
Hydatid cyst is a common disease between humans and animals. Foods that are contaminated with eggs of echinococcal species are the cause of hydatid disease. Although this parasite affects different organs, the liver and lungs are very susceptible but the cardiac hydatid disease is very rare. We report the case of a 33-year-old man admitted because of chest pain. Echocardiography had shown suspected round cyst in the myocardium. More diagnostic managements had demonstrated cystic lesion in the liver, spleen and lungs. There were also multiple cystic lesions in both lungs. He refused surgery and was taken to medical therapy. Myocardial involvement by hydatid cysts occurs very rarely. The main treatment approach for cardiac hydatid disease in patients should be surgery. The alternative treatment was done to those who refused surgical removal, adjuvant therapy such as albendazole for at least 1–6 months. Because myocardial involvement by hydatid cysts is uncommon, we decided to report this rare case.
Keywords: Chest pain, cystic lesion, hydatid cyst
How to cite this article:
Sadeghi S, Ghiasi F, Aria A, Momenzadeh M, Nasri E, Reisizadeh Mobarakeh S. Multiple hydatid cysts in rare locations treated with albendazole: A case report. Adv Biomed Res 2023;12:179 |
How to cite this URL:
Sadeghi S, Ghiasi F, Aria A, Momenzadeh M, Nasri E, Reisizadeh Mobarakeh S. Multiple hydatid cysts in rare locations treated with albendazole: A case report. Adv Biomed Res [serial online] 2023 [cited 2023 Sep 26];12:179. Available from: https://www.advbiores.net/text.asp?2023/12/1/179/382072 |
| Introduction | |  |
Hydatid cyst is a common disease between humans and animals, caused by adult or larval stages of tapeworms of the genus Echinococcus granulosus.[1] The main host in the life cycle of this parasite is dogs and sheeps that are the usual intermediate hosts. Human is infected unintentionally by eating contaminated food with eggs shed by animals such as dogs or foxes.[2],[3] The most common organs that are encountered by hydatid cyst disease are the liver and lungs. Hydatidosis of the kidney, as a single organ, is not usual and is accompanied by the involvement of the other organs, mostly.[4],[5] It has more prevalence in sheep and cattle-raising regions of the world like Australia, New Zealand, South Africa, South America, and the Mediterranean countries of Europe and Asia.[6] The presentation of cardiac hydatidosis is very rare and constitutes about 0.5–2% of hydatid disease locations that are classified in this group.[4],[7] The clinical presentations of myocardial hydatid cyst are not specific, such as atypical chest pain and shortness of breath. Cardiac cysts can rupture and cause critical events like anaphylactic reaction, systemic or pulmonary embolization, or pericardial tamponade.[8]
We report the case of a young man with multivisceral hydatidosis which revealed disseminated hydatid cyst that refused cardiac surgery and used medical therapy.
| Case Report | | |
A 33-year-old man was seen at the emergency department of a university hospital for complaints of sharp and continuous chest pain and a lack of appetite last over 2 weeks. His chest pain was in the left hemithorax and localized at the middle axillary line in about ribs 4–7. It was reproduced by palpating the area and did not increase with excursion and different positions. Additionally, he complained of frequent nonproductive cough. Not any other symptoms including fever, weight loss, or breathlessness were elucidated.
He was opioid-addicted and uses methadone syrup every day. Also, he was smoking about 7–10 packs per year. Prior to this admission, he never had had a problem that made him admit.
His priory job was working as a worker but lost it. He did not take part in any recreational activity and did not work or play with animals.
At the time of admission to our clinic, this patient had a normal vital sign without fever. Oxygen saturation was 96% without oxygen supply. He looked mildly ill and in pain but generally the appearance was normal. Head and neck examination expect of poor oral hygiene and several dental caries was normal; no lymphadenopathy or abnormality in neck and supraclavicular area was detected. Examination of chest also was normal; not any additional sound was heard. Palpating the painful area increased the pain. Cardiac auscultation in all fields was normal; S1 and S2 clearly heard and no murmur existed. Abdominal examination was normal. No hepatomegaly, splenomegaly, or tenderness existed. Genital exam did not reveal any abnormality.
His electrocardiogram showed sinus rhythm with no abnormal changes. In the chest X-ray of patient, diffuse round masses were visible in all lung fields. Some of them had air-fluid levels [Figure 1].
This pattern made us ask for a multidetector computed tomography (MDCT) scan with contrast, a modality with high accuracy for diagnosis of mediastinal and lung parenchymal abnormalities.
There were multiple intraparenchymal cystic lesions with three different types as simple thin-wall cyst, thick-wall cyst with peripheral alveolar consolidation (complicated cyst), and thin-wall cyst that contains air (cyst that communicated with bronchiole). Also, there was one cystic lesion in size of 25 mm in the myocardium. These lesions mostly suggested hydatid cysts. No mediastinal or hilar lymphadenopathy was seen [Figure 2].
To consider other organs, we take brain CT and abdominopelvic MDCT scan with contrast. There was no lesion in the brain CT scan.
The results of abdominopelvic MDCT scan reported at least three thin-wall hepatic cysts with the largest size of 38 mm; also there were two cysts in the spleen with a maximum size of 26 mm. Other organs were normal [Figure 3].
The transthoracic echocardiogram was normal and so we did transesophageal echocardiogram that was reported as one round cystic mass, 20 × 19 mm in RV myocardium [Figure 4]. | Figure 4: Transesophageal echocardiogram showing a round cystic mass in RV myocardium
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Because hydatidosis was the first diagnosis, we tested the serology of hydatid disease in ELISA method which was positive.
Following the diagnosis of the cardiac hydatid cyst, the patient was referred to a cardiac surgeon but he refused surgery, so we decided to start albendazole 400 mg twice a day as the second choice and following up, but the patient came back to our clinic after 15 months and he used albendazole in this duration. To consider the situation of the cysts, we performed an MDCT scan of the thorax and abdominopelvic with contrast. The thorax MDCT scan showed multiple cystic lesions in both lungs up to 22 mm in the right upper lobe and also some of them had cavitary appearance up to 30 mm in the left lower lobe which had decreased in size and number in comparison to the previous study. There was no cyst in the myocardium.
The abdominopelvic MDCT scan with contrast reported three hepatic hypodense lesions with a diameter of 25 mm in the 8th segment, 23 mm in the 2nd segment, and 18 mm in the 7th segment, which showed decrease in size in comparison to the previous study.
There were two hypodense lesions up to 18 mm in the upper pole of the spleen which showed a slight decrease in size in comparison to the previous study. Other organs were reported as normal.
| Discussion | | |
Hydatidosis is a zoonosis infection disease caused by the species of E. granulosus.[9] The ratio of multiple hydatid cysts is reported about 60%, and disseminated hydatidosis mentioned is significantly higher, as the early diagnosis.[10] The most frequent infected organ in humans is the liver 65% and lungs 25% and just 0.5–2% of cases include the heart.[11]
The most common location of cardiac hydatid cysts is the left ventricle (55–60%). Other locations are presented; right ventricle (15%), interventricular septum (9%), left atrium (4%), and interatrial septum (2%).[4] Pericardial involvement with the cysts is also common. They may rupture partially and spread around the pericardial space making more cystic lesions.[12]
It is believed that the involvement of the left side of the heart is caused by reaching via the coronary circulation. Cysts that pass through the liver reach the right side via the venous system and infect the right atrium, right ventricle, and interatrial septum.
There is a possible theory that in systemic circulation, some larvae may pass into the coronary pathway and penetrate into a subepicardial site of the left ventricle and interventricular septum. This may explain how cavitary hydatidosis is seen in the left ventricle, significantly more.[13]
Hydatidosis does not have a particular symptom. When the cysts reach a large size, they cause mass effect to the involved organs. The growth rate of the cysts is affected by the type of the infected organ. Many reports cleared that the growth speed of hydatid cysts in humans begins by approximately 1 cm in a year, whereas others reported that they can grow up to 4–5 cm a year.[14]
In spite of the other encountered organs where patients frequently appear asymptomatic, almost all cases with cardiac hydatid cysts develop some symptoms. Since cardiac echinococcosis can induce life-threatening complications, detection of liver and/or lung involvement during cardiac echinococcosis should always lead to screening for cardiac hydatidosis in those patients.[15],[16],[17]
The clinical manifestations of cardiac hydatidosis are affected by the specific location of the cyst within the heart and the function of the cardiac structures. When it becomes symptomatic, the presentations include anaphylaxis; manifestations of low cardiac output or nonspecific chest pain.
While the multiple cysts during hydatidosis often only allow a palliative medical therapy,[15] the main treatment approach for cardiac hydatid disease in patients, whether symptomatic or asymptomatic, should be the surgery pathway, since the cysts bear a high risk of a spontaneous rupture.[15],[16]
The gold standard drugs to use in adjuvant therapy are benzimidazoles, as albendazole with 10–15 mg/kg/day dosage, for at least 1–6 months.[17],[18]
Albendazole has been proposed as a treatment for hydatid cysts. This drug is used alone or in combination with surgery. Treatment of hydatid cyst with albendazole may be associated with prevention of recurrence and reduction of cyst size.[19]
Also, there is no definite guideline about the duration of pharmacological treatment,[4] and even in some researches, it is recommended that albendazole treatment be given to the patient for at least 2 years.[20] In addition, this anthelmintic should be prescribed in some spatial patients as the alternative treatment to those who refused a surgical removal or to cases with inoperable cardiac hydatid cysts, although guidelines do not exist to date. So we decided to continue medical therapy and follow up the patient at our clinic regularly.
| Conclusion | | |
Myocardial involvement by hydatid cysts occurs very rarely. The main treatment approach for cardiac hydatid disease in patients, whether symptomatic or asymptomatic, should be the surgery pathway. The alternative treatment was given to those who refused a surgical removal or to cases with inoperable cardiac hydatid cysts use in adjuvant therapy such as Albendazole for at least 1–6 months. Because myocardial involvement by hydatid cysts is uncommon, we decided to report this rare case.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Geramizadeh B. Unusual locations of the hydatid cyst: A review from Iran. Iran J Med Sci 2013;38:2-14.  |
| 2. | Das T, Özer M, Yagmur G, Yildirim M, Özgün A, Demirel H. Hydatid disease involved in the heart, liver, and kidney that caused sudden death: Case report. Am J Forensic Med Pathol 2015;36:265-7. |
| 3. | Zhang W, Li J, McManus DP. Concepts in immunology and diagnosis of hydatid disease. Clin Microbiol Rev 2003;16:18-36. |
| 4. | Bogdanovic A, Radojkovic M, Tomasevic RJ, Pesic I, Petkovic TR, Kovacevic P, et al. Presentation of pericardial hydatid cyst as acute cardiac tamponade. Asian J Surg 2017;40:175-7. |
| 5. | Charfeddine S, Mallek S, Gueldiche M, Triki F, Jmâa HB, Frikha I, et al. A huge cardiac hydatid cyst: An unusual cause of chest pain revealing multivisceral hydatidosis in a young woman. J Saudi Heart Assoc 2015;27:286-91. |
| 6. | Ünlü Y, Ceviz M, Karaoğlanoğlu N, Becit N, Koçak H. Arterial embolism caused by a ruptured hydatid cyst in the heart: Report of a case. Surg Today 2002;32:989-91. |
| 7. | Ulgen MS, Alan S, Karadede A, Aydinalp O, Toprak N. Cardiac hydatid cysts located in both the left ventricular apex and the intraventricular septum: Case report. Heart Vessels 2000;15:243-4. |
| 8. | Ipek G, Omeroglu SN, Goksedef D, Balkanay OO, Kanbur E, Engin E, et al. Large cardiac hydatid cyst in the interventricular septum. Tex Heart Instit J 2011;38:719-22. |
| 9. | Seth HS, Mishra P, Khandekar JV, Raut C, Mohapatra CK, Ammannaya GK. A concomitant intramyocardial and pulmonary hydatid cyst: A rare case report. Braz J Cardiovasc Surg 2017;32:138-40. |
| 10. | Cappello E, Cacopardo B, Caltabiano E, Volsi SL, Chiara R, Sapienza M, et al. Epidemiology and clinical features of cystic hydatidosis in Western Sicily: A ten-year review. World J Gastroenterol 2013;19:9351-8. |
| 11. | Tandon S, Darbari A. Hydatid cyst of the right atrium: A rare presentation. Asian Cardiovasc Thor Ann 2006;14:e43-4. |
| 12. | Noaman H, Rawaf S, Majeed A, Salmasi AM. Hydatid cyst of the heart. Angiology 2017;68:765-8. |
| 13. | Kocabay G, Yilmazer S, Korkmaz Y, Karadeniz A, Duran NE. Primary cardiac hydatid cyst presenting as noncompaction cardiomyopathy. Echocardiography 2017;34:141-2. |
| 14. | Çakir M, Balasar M, Küçükkartallar T, Tekin A, Kartal A, Karahan Ö, et al. Management of extra-hepatopulmonary hydatid cysts (157 cases). Türkiye Parazitol Derg 2016;40:72-6. |
| 15. | Brunetti E, Kern P, Vuitton DA. Expert consensus for the diagnosis and treatment of cystic and alveolar echinococcosis in humans. Acta Trop 2010;114:1-6. |
| 16. | Kern P, Grüner B, Wahlers K. Diagnosis and course of echinococcocal diseases in the transplant setting. Transpl Infect Dis 2011;13:217-21. |
| 17. | Salamone G, Licari L, Randisi B, Falco N, Tutino R, Vaglica A, et al. Uncommon localizations of hydatid cyst. Review of the literature. G Chir 2016;37:180-5. |
| 18. | Eckert J, Gemmell MA, Meslin FX, Pawlowski ZS, World Health Organization. WHO/OIE Manual on Echinococcosis in Humans and Animals: A Public Health Problem of Global Concern. World Organisation for Animal Health; 2001. |
| 19. | Dehkordi AB, Sanei B, Yousefi M, Sharafi SM, Safarnezhad F, Jafari R, et al. Albendazole and treatment of hydatid cyst: Review of the literature. Infect Disord Drug Targets 2019;19:101-4. |
| 20. | Gungor S, Yalcinsoy M, Akkan O, Altinsoy B, Ozseker ZF, Misirlioglu A, et al. Disseminated hydatid disease treated with albendazole: 15-year experience. Med 2018;7:935-9. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4]
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