This is a preprint. Preprints are preliminary reports that have not undergone peer review. They should not be considered conclusive, used to inform clinical practice, or referenced by the media as validated information.
Case Report
Long-term drug treatment of a giant liver hydatid cyst in a child
Valeri Velev
Medical University of Sofia
Corresponding Author
ORCiD: https://orcid.org/0000-0003-0161-6993
License:
This work is licensed under a Creative Commons Attribution 4.0 International License. Read Full License
Background
Cystic еchinococcosis is a chronic parasitosis caused by the larvae of the tapeworm Echinococcus granulosus. Humans act as intermediate hosts and the larvae can affect any organ in the form of cysts. They are most often found incidentally by diagnostic imaging, where they have become large in size, with treatment being primarily invasive. Conservative treatment with albendazole is one of the appropriate treatment options, but it features a low success rate in cysts over 5 cm.
Case presentation
We describe a case of an 14-year-old girl with an active 12 cm cyst in the liver. Two consecutive courses with albendazole, with duration 3 months each, were conducted. The cyst reached a safe inactive stage and no relapses have been observed for 1 year since the last administration of the medication.
Conclusions
Our experience shows that with large cysts albendazole courses may be extended including in childhood. Ultrasonography has clearly demonstrated its ability to follow-up patient in a cost effective manner and avoiding ionizing radiations.
Keywords
Echinococcosis, Albendazole, Ultrasonography
Figure 1
Cystic echinococcosis (CE) is a severe parasitic zoonosis caused by the larvae of the tapeworm Echinococcus granulosus. It is spread almost globally - endemic areas in Europe are the Balkans and the Mediterranean. Definitive hosts are mainly dogs and intermediate hosts are ruminants (sheep). Because of their proximity to these animals, humans may be accidental intermediate hosts. The larval form called hydatid cyst can affect any tissue or organ, but the liver is most commonly attacked. The course of the disease is slow and asymptomatic, and the disease is most often diagnosed incidentally at an advanced stage. The pediatric population accounts for 10–20% of all cases [1, 2]. Depending on the duration of the disease, liver cysts have different sonographic characteristics and dimensions (Table 1), based on which, one of four approaches is recommended - medication treatment, open surgery, cyst puncture or “watch and wait” for cysts in a transitional stage [3].
| Stage Echographic image according to WHO-IWGE Classification |
|---|
| CL Anechogenic uniloculated cyst, with no echoes or internal sepsis |
| CE 1 Anechogenic cyst, with fine echoes inside, representing the hydatic sand - active cyst |
| CE 2 Cyst with multiple septums at the interior, or “honeycomb” aspect,with a uniloculated primary cyst - active cyst |
| CE 3 Uniloculated cyst with decolated membrane (“waterlily sign”) (CE3a) or daughter vesicles type hypo/hyperechogene images (CE3b) - cyst in transition phase |
| CE 4 Cyst with mixed content, hypo/hyperechogenic, without daughter vesicles - degenerative phase |
| CE 5 Cyst with partial or totally calcified wall - inactive cyst |
We report a case of an 14-year-old girl with an active 12 cm cyst in the liver. Diagnosis was obtained by ultrasonography (US) (Samsung Medison Convex Array Ultrasound Transducer 3–7 MHz.) and serologically with enzyme-linked immunosorbent assay [ELISA] immunoglobulin G [IgG]; BulBio-National Centre of Infectious and Parasitic Diseases, Sofia, Bulgaria). The present study was approved by the ethical committee of University Hospital “Prof. Iv. Kirov” and Medical University of Sofia (number 12/2018).
A 14-year-old girl living in a rural area in southern Bulgaria visited her family doctor because of complaints of fatigue and heaviness in the right hypochondrium. On examination, the doctor found mild tenderness upon palpation in the liver area and elevated eosinophil level in the peripheral blood (11.4%). Due to eosinophilia, the patient was referred to our clinic with suspected parasitosis. Abdominal sonography showed an active echinococcal cyst in the right lobe of the liver, 12 cm in diameter. By the WHO classification, the cyst was in stage CE2 [4]. Serological testing of the patient was positive for echinococcosis. Due to patient’s advanced age, concomitant diseases, and refusal to undergo surgery, long-term albendazole treatment (15 mg/kg for 3 months, without interruption) was initiated. The follow-up sonography, after the 3-month treatment course, showed that the cyst had reached a safer stage CE3b - transitional cyst. The medication treatment was discontinued for one month, and then re-initiated, with albendazole at the same dose for 3 months. Upon completion of the second course of medication treatment, the sonographic examination showed that the cyst had reached CE4 stage - inactive cyst (Fig. 1). Liver transaminase levels were observed throughout the course of medication treatment, and they did not rise above normal. Presently, the patient has been having an inactive cyst for 1 year, and sonographic monitoring is performed using the “watch and wait” approach. Serological testing is still positive.
CE is most often found incidentally during prophylactic examination or due to non-specific complaints. At the time of diagnosis, the cyst is most often relatively large, active, in some cases, two or more cysts are found - multiple echinococcosis. Treatment is very important to avoid complications [4]. Conservative treatment with albendazole is one of the appropriate treatment options, but it features a low success rate in cysts over 5 cm in diameter, probably due to the more difficult penetration of the active substance into the cyst [5, 6]. In such cases, surgery or puncture method are preferred. Despite their good results, these two invasive methods often have contraindications or patients refuse to have them administered because they would like to try medication. The WHO informal echinococcosis group recommends continuous medication treatment for 1 month [7, 8]. Some authors accept that albendazole therapy for < 3 months is associated with suboptimal outcomes [9]; and some authorities propose even longer therapies (> 6 months), although this approach requires further validation [9, 10]. In the case described, we administered longer courses because of the size of the cyst and the risk of rupture (active and superficial). Our experience shows that in some cases albendazole courses may be extended if the patient has good tolerability to the medication. In inoperable cases, conservative treatment may also be applied to larger cysts, including in childhood. Adverse reactions have been associated with non-adherence and discontinuation of therapy and have been found to affect adherence to visits in conditions requiring prolonged therapy [11]. Despite CT (Computed tomography) or MR (Magnetic resonance) are often used, in this case US has clearly demonstrated its ability to follow-up patient in a cost effective manner and avoiding ionizing radiations [5].
CE remains a serious problem in some endemic areas of Europe, such as the Balkans and the Mediterranean. Along with extensive sheep breeding and many stray dogs, in some of these areas, veterinary measures for control of sheep and deparasitation of dogs as a major preventive measure against parasitosis are underestimated.
Acknowledgements
“Not applicable”
Availability of data and materials
Data sharing is not applicable to this article as no datasets were generated or analyzed during the current study.
Authors’ contributions
VV wrote the manuscript; GD designed Tab. 1 and helped to
write the manuscript; VV diagnosed and treated the patient; YMM and GD helped to epidemiological study and serologycal testing. VV make a design to Figs. 1; YMM revised manuscript critically for important intellectual content; all authors read and approved the final manuscript.
Ethics approval and consent to participate
Ethics approval was obtained from University Hospital “Prof. Iv. Kirov”. Written
informed consent to participate in the study from the patient's parents was obtained.
Consent for publication
Written informed consent to publication on the study from the patient's parents was obtained.
Competing interests
The authors declare that they have no competing interests.
“Not applicable”
- Thompson RC. Biology and Systematics of Echinococcus. Adv Parasitol. 2017;95:65–109. doi: 10.1016/bs.apar.2016.07.001.
- Jordanova DP, Harizanov RN, Kaftandjiev IT, et al. Cystic echinococcosis in Bulgaria 1996–2013, with emphasis on childhood infections. Eur J Clin Microbiol Infect Dis. 2015;34(7):1423–8.
- Musaev GK, Fatyanova AS, Levkin VV. Principles and modern trends in liver echinococcosis treatment. Khirurgiia (Mosk). 2017;(12):90–94. doi: 10.17116/hirurgia20171290-94.
- Greco S, Cannella R, Giambelluca D, Pecoraro G, Battaglia E, Midiri M. et. al. Complications of Hepatic Echinococcosis: Multimodality Imaging Approach. Insights Imaging. 2019;10(1):113. doi:10.1186/s13244-019-0805-8.
- Pendse HA, Nawale AJ, Deshpande SS, Merchant SA. Radiologic features of hydatid disease: the importance of sonography. J Ultrasound Med. 2015;34(5):895–905. 10.7863/ultra.34.5.895.. ; ). doi.
- Jee KN. Hepatic hydatid cyst. Korean J Intern Med. 2015 Jul;30(4):554-5. 10.3904/kjim.2015.30.4.554.. ; ). doi.
- Todorov T, Vutova K, Donev S, Ivanov A, Katzarov K, Takov D. The types and timing of the degenerative changes seen in the cysts during and after benzimidazole treatment of cystic echinococcosis. Ann Trop Med Parasitol. 2005;99(7):649–59.
- Agudelo Higuita NI, Brunetti E, McCloskey C. Cystic Echinococcosis. J Clin Microbiol. 2016;54(3):518 – 23. doi: 10.1128/JCM.02420-15.
- Stojković M, Weber T, Junghanss T.. Clinical Management of Cystic Echinococcosis: State of the Art and Perspectives. Curr Opin Infect Dis. 2018; (5):383–92. doi:10.1097/QCO.0000000000000485.
- Legonkov Y, Bronshtein A. Experience with Albendazole treatment in children with cystic echinococcosis. Med Parazitol (Mosk). 2017 Apr;(2):9–13. (article in Russian).
- Salinas J, Gonzales H, Astuvilca J, Arce-Villavicencio Y, Carbajal-Gonzalez D, Talledo L, Willig J. Long-Term Albendazole Effectiveness for Hepatic Cystic Echinococcosis. Am J Trop Med Hyg. 2011;85(6):1075–9. doi:10.4269/ajtmh.2011.11-0382.
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Case Report
Long-term drug treatment of a giant liver hydatid cyst in a child
Valeri Velev
Medical University of Sofia
Corresponding Author
ORCiD: https://orcid.org/0000-0003-0161-6993
Badges
Prescreen
This manuscript passed our Prescreen assessment
Complete author information
Appropriate declaration statements
Potential risks to human health
Prescreen
History
CURRENT STATUS: Posted
Version 1
Posted 13 Jul, 2020
No community comments so far
Metrics
Comments: 0
PDF Downloads: ...
HTML Views: ...
License:
This work is licensed under a Creative Commons Attribution 4.0 International License. Read Full License
Background
Cystic еchinococcosis is a chronic parasitosis caused by the larvae of the tapeworm Echinococcus granulosus. Humans act as intermediate hosts and the larvae can affect any organ in the form of cysts. They are most often found incidentally by diagnostic imaging, where they have become large in size, with treatment being primarily invasive. Conservative treatment with albendazole is one of the appropriate treatment options, but it features a low success rate in cysts over 5 cm.
Case presentation
We describe a case of an 14-year-old girl with an active 12 cm cyst in the liver. Two consecutive courses with albendazole, with duration 3 months each, were conducted. The cyst reached a safe inactive stage and no relapses have been observed for 1 year since the last administration of the medication.
Conclusions
Our experience shows that with large cysts albendazole courses may be extended including in childhood. Ultrasonography has clearly demonstrated its ability to follow-up patient in a cost effective manner and avoiding ionizing radiations.
Figure 1
Cystic echinococcosis (CE) is a severe parasitic zoonosis caused by the larvae of the tapeworm Echinococcus granulosus. It is spread almost globally - endemic areas in Europe are the Balkans and the Mediterranean. Definitive hosts are mainly dogs and intermediate hosts are ruminants (sheep). Because of their proximity to these animals, humans may be accidental intermediate hosts. The larval form called hydatid cyst can affect any tissue or organ, but the liver is most commonly attacked. The course of the disease is slow and asymptomatic, and the disease is most often diagnosed incidentally at an advanced stage. The pediatric population accounts for 10–20% of all cases [1, 2]. Depending on the duration of the disease, liver cysts have different sonographic characteristics and dimensions (Table 1), based on which, one of four approaches is recommended - medication treatment, open surgery, cyst puncture or “watch and wait” for cysts in a transitional stage [3].
| Stage Echographic image according to WHO-IWGE Classification |
|---|
| CL Anechogenic uniloculated cyst, with no echoes or internal sepsis |
| CE 1 Anechogenic cyst, with fine echoes inside, representing the hydatic sand - active cyst |
| CE 2 Cyst with multiple septums at the interior, or “honeycomb” aspect,with a uniloculated primary cyst - active cyst |
| CE 3 Uniloculated cyst with decolated membrane (“waterlily sign”) (CE3a) or daughter vesicles type hypo/hyperechogene images (CE3b) - cyst in transition phase |
| CE 4 Cyst with mixed content, hypo/hyperechogenic, without daughter vesicles - degenerative phase |
| CE 5 Cyst with partial or totally calcified wall - inactive cyst |
We report a case of an 14-year-old girl with an active 12 cm cyst in the liver. Diagnosis was obtained by ultrasonography (US) (Samsung Medison Convex Array Ultrasound Transducer 3–7 MHz.) and serologically with enzyme-linked immunosorbent assay [ELISA] immunoglobulin G [IgG]; BulBio-National Centre of Infectious and Parasitic Diseases, Sofia, Bulgaria). The present study was approved by the ethical committee of University Hospital “Prof. Iv. Kirov” and Medical University of Sofia (number 12/2018).
A 14-year-old girl living in a rural area in southern Bulgaria visited her family doctor because of complaints of fatigue and heaviness in the right hypochondrium. On examination, the doctor found mild tenderness upon palpation in the liver area and elevated eosinophil level in the peripheral blood (11.4%). Due to eosinophilia, the patient was referred to our clinic with suspected parasitosis. Abdominal sonography showed an active echinococcal cyst in the right lobe of the liver, 12 cm in diameter. By the WHO classification, the cyst was in stage CE2 [4]. Serological testing of the patient was positive for echinococcosis. Due to patient’s advanced age, concomitant diseases, and refusal to undergo surgery, long-term albendazole treatment (15 mg/kg for 3 months, without interruption) was initiated. The follow-up sonography, after the 3-month treatment course, showed that the cyst had reached a safer stage CE3b - transitional cyst. The medication treatment was discontinued for one month, and then re-initiated, with albendazole at the same dose for 3 months. Upon completion of the second course of medication treatment, the sonographic examination showed that the cyst had reached CE4 stage - inactive cyst (Fig. 1). Liver transaminase levels were observed throughout the course of medication treatment, and they did not rise above normal. Presently, the patient has been having an inactive cyst for 1 year, and sonographic monitoring is performed using the “watch and wait” approach. Serological testing is still positive.
CE is most often found incidentally during prophylactic examination or due to non-specific complaints. At the time of diagnosis, the cyst is most often relatively large, active, in some cases, two or more cysts are found - multiple echinococcosis. Treatment is very important to avoid complications [4]. Conservative treatment with albendazole is one of the appropriate treatment options, but it features a low success rate in cysts over 5 cm in diameter, probably due to the more difficult penetration of the active substance into the cyst [5, 6]. In such cases, surgery or puncture method are preferred. Despite their good results, these two invasive methods often have contraindications or patients refuse to have them administered because they would like to try medication. The WHO informal echinococcosis group recommends continuous medication treatment for 1 month [7, 8]. Some authors accept that albendazole therapy for < 3 months is associated with suboptimal outcomes [9]; and some authorities propose even longer therapies (> 6 months), although this approach requires further validation [9, 10]. In the case described, we administered longer courses because of the size of the cyst and the risk of rupture (active and superficial). Our experience shows that in some cases albendazole courses may be extended if the patient has good tolerability to the medication. In inoperable cases, conservative treatment may also be applied to larger cysts, including in childhood. Adverse reactions have been associated with non-adherence and discontinuation of therapy and have been found to affect adherence to visits in conditions requiring prolonged therapy [11]. Despite CT (Computed tomography) or MR (Magnetic resonance) are often used, in this case US has clearly demonstrated its ability to follow-up patient in a cost effective manner and avoiding ionizing radiations [5].
CE remains a serious problem in some endemic areas of Europe, such as the Balkans and the Mediterranean. Along with extensive sheep breeding and many stray dogs, in some of these areas, veterinary measures for control of sheep and deparasitation of dogs as a major preventive measure against parasitosis are underestimated.
Acknowledgements
“Not applicable”
Availability of data and materials
Data sharing is not applicable to this article as no datasets were generated or analyzed during the current study.
Authors’ contributions
VV wrote the manuscript; GD designed Tab. 1 and helped to
write the manuscript; VV diagnosed and treated the patient; YMM and GD helped to epidemiological study and serologycal testing. VV make a design to Figs. 1; YMM revised manuscript critically for important intellectual content; all authors read and approved the final manuscript.
Ethics approval and consent to participate
Ethics approval was obtained from University Hospital “Prof. Iv. Kirov”. Written
informed consent to participate in the study from the patient's parents was obtained.
Consent for publication
Written informed consent to publication on the study from the patient's parents was obtained.
Competing interests
The authors declare that they have no competing interests.
“Not applicable”
- Thompson RC. Biology and Systematics of Echinococcus. Adv Parasitol. 2017;95:65–109. doi: 10.1016/bs.apar.2016.07.001.
- Jordanova DP, Harizanov RN, Kaftandjiev IT, et al. Cystic echinococcosis in Bulgaria 1996–2013, with emphasis on childhood infections. Eur J Clin Microbiol Infect Dis. 2015;34(7):1423–8.
- Musaev GK, Fatyanova AS, Levkin VV. Principles and modern trends in liver echinococcosis treatment. Khirurgiia (Mosk). 2017;(12):90–94. doi: 10.17116/hirurgia20171290-94.
- Greco S, Cannella R, Giambelluca D, Pecoraro G, Battaglia E, Midiri M. et. al. Complications of Hepatic Echinococcosis: Multimodality Imaging Approach. Insights Imaging. 2019;10(1):113. doi:10.1186/s13244-019-0805-8.
- Pendse HA, Nawale AJ, Deshpande SS, Merchant SA. Radiologic features of hydatid disease: the importance of sonography. J Ultrasound Med. 2015;34(5):895–905. 10.7863/ultra.34.5.895.. ; ). doi.
- Jee KN. Hepatic hydatid cyst. Korean J Intern Med. 2015 Jul;30(4):554-5. 10.3904/kjim.2015.30.4.554.. ; ). doi.
- Todorov T, Vutova K, Donev S, Ivanov A, Katzarov K, Takov D. The types and timing of the degenerative changes seen in the cysts during and after benzimidazole treatment of cystic echinococcosis. Ann Trop Med Parasitol. 2005;99(7):649–59.
- Agudelo Higuita NI, Brunetti E, McCloskey C. Cystic Echinococcosis. J Clin Microbiol. 2016;54(3):518 – 23. doi: 10.1128/JCM.02420-15.
- Stojković M, Weber T, Junghanss T.. Clinical Management of Cystic Echinococcosis: State of the Art and Perspectives. Curr Opin Infect Dis. 2018; (5):383–92. doi:10.1097/QCO.0000000000000485.
- Legonkov Y, Bronshtein A. Experience with Albendazole treatment in children with cystic echinococcosis. Med Parazitol (Mosk). 2017 Apr;(2):9–13. (article in Russian).
- Salinas J, Gonzales H, Astuvilca J, Arce-Villavicencio Y, Carbajal-Gonzalez D, Talledo L, Willig J. Long-Term Albendazole Effectiveness for Hepatic Cystic Echinococcosis. Am J Trop Med Hyg. 2011;85(6):1075–9. doi:10.4269/ajtmh.2011.11-0382.