Cystic Echinococcosis of the Bone: A European Multicenter Study

Letizia Cattaneo Department of Clinical, Surgical, Diagnostic and Pediatric Science, University of Pavia, Pavia, Italy;

Search for other papers by Letizia Cattaneo in
Current site
Google Scholar
PubMed Close
,
Tommaso Manciulli Department of Clinical, Surgical, Diagnostic and Pediatric Science, University of Pavia, Pavia, Italy;
PhD School of Experimental Medicine, University of Pavia, Pavia, Italy;

Search for other papers by Tommaso Manciulli in
Current site
Google Scholar
PubMed Close
,
Carmen-Michaela Cretu Parasitology Department, Colentina Clinical Hospital, Carol Davila University of Medicine and Pharmacy, Bucharest, Romania;

Search for other papers by Carmen-Michaela Cretu in
Current site
Google Scholar
PubMed Close
,
Maria Teresa Giordani Division of Infectious and Tropical Diseases, San Bortolo Hospital, Vicenza, Italy;

Search for other papers by Maria Teresa Giordani in
Current site
Google Scholar
PubMed Close
,
Andrea Angheben Centre for Tropical Diseases, Istituto di Ricovero e Cura a Carattere Scientifico (IRCCS) Sacro Cuore-Don Calabria Hospital, Verona, Italy;

Search for other papers by Andrea Angheben in
Current site
Google Scholar
PubMed Close
,
Alessandro Bartoloni Infectious and Tropical Diseases Unit, Careggi University Hospital, University of Florence, Florence, Italy;

Search for other papers by Alessandro Bartoloni in
Current site
Google Scholar
PubMed Close
,
Lorenzo Zammarchi Infectious and Tropical Diseases Unit, Careggi University Hospital, University of Florence, Florence, Italy;

Search for other papers by Lorenzo Zammarchi in
Current site
Google Scholar
PubMed Close
,
Filippo Bartalesi Infectious and Tropical Diseases Unit, Careggi University Hospital, University of Florence, Florence, Italy;

Search for other papers by Filippo Bartalesi in
Current site
Google Scholar
PubMed Close
,
Joachim Richter Institute of Tropical Medicine and International Health, Charité - Universitätsmedizin, Berlin, Germany;

Search for other papers by Joachim Richter in
Current site
Google Scholar
PubMed Close
,
Peter Chiodini Department of Clinical Parasitology, Hospital for Tropical Diseases, London, United Kingdom;

Search for other papers by Peter Chiodini in
Current site
Google Scholar
PubMed Close
,
Gauri Godbole Department of Clinical Parasitology, Hospital for Tropical Diseases, London, United Kingdom;

Search for other papers by Gauri Godbole in
Current site
Google Scholar
PubMed Close
,
Thomas Junghanss Section of Clinical Tropical Medicine, University Hospital, Heidelberg, Germany;

Search for other papers by Thomas Junghanss in
Current site
Google Scholar
PubMed Close
,
Marija Stojkovic Section of Clinical Tropical Medicine, University Hospital, Heidelberg, Germany;

Search for other papers by Marija Stojkovic in
Current site
Google Scholar
PubMed Close
,
Luigi Sammarchi Division of Radiology, IRCCS San Matteo Hospital Foundation, Pavia, Italy;

Search for other papers by Luigi Sammarchi in
Current site
Google Scholar
PubMed Close
,
Roberto Dore Department of Radiology, Istituti Clinici di Pavia e Vigevano, University Hospital, Pavia, Italy;

Search for other papers by Roberto Dore in
Current site
Google Scholar
PubMed Close
,
Alessandro Vercelli Department of Radiology, Istituti Clinici di Pavia e Vigevano, University Hospital, Pavia, Italy;

Search for other papers by Alessandro Vercelli in
Current site
Google Scholar
PubMed Close
,
Francesco Benazzo Department of Clinical, Surgical, Diagnostic and Pediatric Science, University of Pavia, Pavia, Italy;
Division of Orthopedics and Traumatology, IRCCS San Matteo Hospital Foundation, Pavia, Italy;

Search for other papers by Francesco Benazzo in
Current site
Google Scholar
PubMed Close
,
Fabrizio Cuzzocrea Division of Orthopedics and Traumatology, IRCCS San Matteo Hospital Foundation, Pavia, Italy;

Search for other papers by Fabrizio Cuzzocrea in
Current site
Google Scholar
PubMed Close
,
Francesca Tamarozzi Centre for Tropical Diseases, Istituto di Ricovero e Cura a Carattere Scientifico (IRCCS) Sacro Cuore-Don Calabria Hospital, Verona, Italy;

Search for other papers by Francesca Tamarozzi in
Current site
Google Scholar
PubMed Close
, and
Enrico Brunetti Department of Clinical, Surgical, Diagnostic and Pediatric Science, University of Pavia, Pavia, Italy;
Unit of Infectious and Tropical Diseases, IRCCS San Matteo Hospital Foundation, Pavia, Italy

Search for other papers by Enrico Brunetti in
Current site
Google Scholar
PubMed Close
DOI:
https://doi.org/10.4269/ajtmh.18-0758
Volume/Issue:
Volume 100: Issue 3
Page(s):
617–621
Restricted access
Get Permissions

Cystic echinococcosis (CE) is a zoonosis caused by the larval stage of the tapeworm Echinococcus granulosus. In humans, the infection induces the formation of parasitic cysts mostly in the liver and lungs, but virtually any organ can be affected. CE of the bone is one of the rarest forms of the disease, yet it is also extremely debilitating for patients and hard to manage for clinicians. Unlike abdominal CE, there is currently no expert consensus on the management of bone CE. In this study, we conducted a survey of the clinical records of seven European referral centers for the management of patients with CE and retrieved data on the clinical management of 32 patients with a diagnosis of bone CE. Our survey confirmed that the patients endured chronic debilitating disease with a high rate of complications (84%). We also found that diagnostic approaches were highly heterogeneous. Surgery was extensively used to treat these patients, as well as albendazole, occasionally combined with praziquantel or nitaxozanide. Treatment was curative only for two patients, with one requiring amputation of the involved bone. Our survey highlights the need to conduct systematic studies on bone CE, both retrospectively and prospectively.

Author Notes

Address correspondence to Enrico Brunetti, Department of Clinical, Surgical, Diagnostic and Pediatric Sciences, University of Pavia, Viale Brambilla 74, Pavia 27100, Italy or Unit of Infectious and Tropical Diseases, IRCCS San Matteo Hospital Foundation, Viale Taramelli 5, Pavia 27100, Italy. E-mail: enrico.brunetti@unipv.it

Authors’ addresses: Letizia Cattaneo, Department of Clinical, Surgical, Diagnostic and Pediatric Sciences, University of Pavia, Pavia, Italy, E-mail: letizia.cattaneo01@ateneopv.it. Tommaso Manciulli, PhD School of Experimental Medicine, Universita degli Studi di Pavia, Pavia, Italy, and Department of Clinical, Surgical, Diagnostic and Pediatric Sciences, Universita degli Studi di Pavia, Pavia, Italy, E-mail: tommaso.manciulli01@ateneopv.it. Carmen-Michaela Cretu, Parasitology Department, Colentina Clinical Hospital, University of Medicine and Pharmacy, Bucharest, Romania, and Parasitology Department, Bucharest, Romania, E-mail: michaelacarmen.cretu@gmail.com. Maria Teresa Giordani, Infectious and Tropical Diseases Unit, San Bortolo Hospital, Vicenza, Italy, E-mail: giordanimt@libero.it. Andrea Angheben, Centre of Tropical Diseases, Ospedale Sacro Cuore Don Calabria, Verona, Italy, E-mail: andrea.angheben@sacrocuore.it. Alessandro Bartoloni, Infectious and Tropical Diseases, University of Florence, Florence, Italy, E-mail: alessandro.bartoloni@unifi.it. Lorenzo Zammarchi, Dipartimento di Medicina Sperimentale e Clinica, Clinica Malattie Infettive, Università di Firenze, Florence, Italy, E-mail: lorenzo.zammarchi@unifi.it. Filippo Bartalesi, SOD Malattie Infettive e Tropicali, Azienda Ospedaliero-Universitaria Careggi, Firenze, Italy, E-mail: bartalesif@aou-careggi.toscana.it. Joachim Richter, Institute of Tropical Medicine and International Health, Charité - Universitätsmedizin, Berlin, Germany, E-mail: joachim.richter@charite.de. Peter Chiodini and Gauri Godbole, Department of Clinical Parasitology, Hospital for Tropical Diseases, London, United Kingdom, E-mails: peter.chiodini@uclh.nhs.uk and gauri.godbole@phe.gov.uk. Thomas Junghanss, Section of Clinical Tropical Medicine, University Hospital Heidelberg, Heidelberg, Germany, E-mail: thomas.junghanss@urz.uni-heidelberg.de. Marija Stojkovic, Clinical tropical medicine, UniversitatsKlinikum Heidelberg, Heidelberg, Germany, E-mail: marija.stojkovic@med.uni-heidelberg.de. Luigi Sammarchi, Department of Radiology, IRCCS San Matteo Hospital Foundation, Pavia, Italy, E-mail: sammarchi@smatteo.pv.it. Roberto Dore and Alessandro Vercelli, Department of Radiology, Istituti Clinici di Pavia e Vigevano, University Hospital, Pavia, Italy, E-mail: r.dore@smatteo.pv.it and aleverce@gmail.com. Francesco Benazzo, Department of Clinical, Surgical, Diagnostic and Pediatric Sciences, Universita degli Studi di Pavia, Pavia, Italy, and Division of Orthopedics and Traumatology, IRCCS San Matteo Hospital Foundation, Pavia, Italy, E-mail: francesco.benazzo@unipv.it. Fabrizio Cuzzocrea, Division of Orthopedics and Traumatology, IRCCS San Matteo Hospital Foundation, Pavia, Italy, E-mail: f.cuzzocrea@smatteo.pv.it. Francesca Tamarozzi, Centre for Tropical Diseases, Sacro Cuore - Don Calabria Hospital - Negrar (Verona), Italy, and WHO collaborating centre for clinical management of cystic echinococcosis, Pavia, Italy, E-mail: f_tamarozzi@yahoo.com. Enrico Brunetti, Department of Clinical, Surgical, Diagnostic and Pediatric Science, University of Pavia, Pavia, Italy, and Division of Infectious and Tropical Diseases, IRCCS S Matteo Hospital Foundation, Univeristy of Pavia, Pavia, Italy, E-mail: enrico.brunetti@unipv.it.

These authors contributed equally to this work.

Copyright:
© The American Society of Tropical Medicine and Hygiene 2019
Received:
16 Sep 2018
|
Accepted:
26 Nov 2018
|
Published Online:
28 Jan 2019
Cover The American Journal of Tropical Medicine and Hygiene
The American Journal of Tropical Medicine and Hygiene
Print ISSN:
0002-9637
Online ISSN:
1476-1645
  • 1.

    Romig T, Deplazes P, Jenkins D, Giraudoux P, Massolo A, Craig PS, Wassermann M, Takahashi K, de la Rue M, 2017. Ecology and life cycle patterns of Echinococcus species. Adv Parasitol 95: 213–314.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 2.

    Budke CM, Deplazes P, Torgerson PR, 2006. Global socioeconomic impact of cystic echinococcosis. Emerg Infect Dis 12: 296–303.

  • 3.

    Polat P, Kantarci M, Alper F, Suma S, Koruyucu MB, Okur A, 2003. Hydatid disease from head to toe. Radiographics 23: 475–494; quiz 536–537.

  • 4.

    Zlitni M, Ezzaouia K, Lebib H, Karray M, Kooli M, Mestiri M, 2001. Hydatid cyst of bone: diagnosis and treatment. World J Surg 25: 75–82.

  • 5.

    Togral G, Arkan S, Ekiz T, Kekec A, Eksioglu M, 2016. Musculoskeletal hydatid cysts resembling tumors: a report of five cases. Orthop Surg 8: 246–252.

  • 6.

    Neumayr A, Tamarozzi F, Goblirsch S, Blum J, Brunetti E, 2013. Spinal cystic echinococcosis–a systematic analysis and review of the literature: part 1. Epidemiology and anatomy. PLoS Negl Trop Dis 7: 1–11.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7.

    Monge-Maillo B, Chamorro Tojeiro S, López-Vélez R, 2017. Management of osseous cystic echinococcosis. Expert Rev Anti Infect Ther 15: 1075–1082.

  • 8.

    Steinmetz S, Racloz G, Stern R, Dominguez D, Al-mayahi M, Schibler M, Lew D, Hoffmeyer P, Uçkay I, 2014. Treatment challenges associated with bone echinococcosis. J Antimicrob Chemother 69: 821–826.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 9.

    Kireşi DA, Karabacakoğlu A, Odev K, Karaköse S, 2003. Uncommon locations of hydatid cysts. Acta Radiol 44: 622–636.

  • 10.

    Neumayr A, Tamarozzi F, Goblirsch S, Blum J, Brunetti E, 2013. Spinal cystic echinococcosis–a systematic analysis and review of the literature: part 2. Treatment, follow-up and outcome. PLoS Negl Trop Dis 7: e2458.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 11.

    Karray S, Zlitni M, Fowles JV, Slimane N, Kassab T, Rosset P, 1990. Vertebral hydatidosis and paraplegia. J Bone Joint Surg Br 72: 84–88.

  • 12.

    Loudiye H, Aktaou S, Hassikou H, El Bardouni A, El Manouar M, Fizazi M, Tazi A, Hajjaj-Hassouni N, 2003. Hydatid disease of bone: review of 11 cases. Joint Bone Spine 70: 352–355.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 13.

    Papanikolaou A, 2008. Osseous hydatid disease. Iran J Parasitol 3: 60–64.

  • 14.

    Kalinova K, Proichev V, Stefanova P, Tokmakova K, Poriazova E, 2005. Hydatid bone disease: a case report and review of the literature. J Orthop Surg (Hong Kong) 13: 323–325.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 15.

    Bracanovic D, Djuric M, Sopta J, Djonic D, Lujic N, 2013. Skeletal manifestations of hydatid disease in Serbia: demographic distribution, site involvement, radiological findings, and complications. Korean J Parasitol 51: 453–459.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 16.

    Brunetti E, Garcia HH, Junghanss T, 2011. Cystic echinococcosis: chronic, complex, and still neglected. PLoS Negl Trop Dis 5: 3–7.

  • 17.

    Barbieri M, Fernandez V, Gonzalez G, Luaces VM, Nieto A, 1998. Diagnostic evaluation of a synthetic peptide derived from a novel antigen B subunit as related to other available peptides and native antigens used for serology of cystic hydatidosis. Parasite Immunol 20: 51–61.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 18.

    Delunardo F, Ortona E, Margutti P, Perdicchio M, Vacirca D, Teggi A, Sorice M, Siracusano A, 2010. Identification of a novel 19 kDa Echinococcus granulosus antigen. Acta Trop 113: 42–47.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 19.

    Schweiger A, Grimm F, Tanner I, Müllhaupt B, Bertogg K, Müller N, Deplazes P, 2012. Serological diagnosis of echinococcosis: the diagnostic potential of native antigens. Infection 40: 139–152.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 20.

    Lissandrin R, Tamarozzi F, Piccoli L, Tinelli C, De Silvestri A, Mariconti M, Meroni V, Genco F, Brunetti E, 2016. Factors influencing the serological response in hepatic Echinococcus granulosus infection. Am J Trop Med Hyg 94: 166–171.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 21.

    Carmena D, Benito A, Eraso E, 2006. Antigens for the immunodiagnosis of Echinococcus granulosus infection: an update. Acta Trop 98: 74–86.

  • 22.

    Perez-Molina JA, Diaz-Menendez M, Gallego JI, Norman F, Monge-Maillo B, Ayala AP, Lopez-Velez R, 2011. Evaluation of nitazoxanide for the treatment of disseminated cystic echinococcosis: report of five cases and literature review. Am J Trop Med Hyg 84: 351–356.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 23.

    Stettler M et al. 2004. Secondary and primary murine alveolar echinococcosis : combined albendazole/nitazoxanide chemotherapy exhibits profound anti-parasitic activity. Int J Parasitol 34: 615–624.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 24.

    Prabhakar MM, Acharya AJ, Modi DR, Jadav B, 2005. Spinal hydatid disease: a case series. J Spinal Cord Med 28: 426–431.

  • 25.

    Lam KS, Faraj A, Mulholland RC, Finch RG, 1997. Medical decompression of vertebral hydatidosis. Spine (Phila Pa 1976) 22: 2050–2055.

  • 26.

    Yildiz Y, Bayrakci K, Altay M, Saglik Y, 2001. The use of polymethylmethacrylate in the management of hydatid disease of bone. J Bone Joint Surg Br 83: 1005–1008.

  • 27.

    Brunetti E, Kern P, Vuitton DA, 2010. Expert consensus for the diagnosis and treatment of cystic and alveolar echinococcosis in humans. Acta Trop 114: 1–16.

  • 28.

    Rossi P, Tamarozzi F, Galati F, Pozio E, Akhan O, Cretu CM, Vutova K, Siles-Lucas M, Brunetti E, Casulli A; HERACLES extended network, 2016. The first meeting of the European Register of Cystic Echinococcosis (ERCE). Parasit Vectors 9: 243.

    • PubMed
    • Search Google Scholar
    • Export Citation

Expert Committee on Hepatitis

First Report. World Health Organization Technical Report Series No. 62 (Available also in French). 26 pp. Geneva, W.H.O., Palais des Nations. March, 1953. Price 1/6, $0.20, Fr. fr. 65.-; Sw. fr. 0.80

Past two years Past Year Past 30 Days
Abstract Views 1810 1583 237
Full Text Views 875 11 1
PDF Downloads 443 11 1
 

 

 

 
 
Affiliate Membership Banner
 
 
Research for Health Information Banner
 
 
CLOCKSS
 
 
 
Society Publishers Coalition Banner
Save