In this clinical pearl an ectopic human Dioctophyma renale infection in the abdominal cavity is reported for the first time. The patient presented with a gastric perforation and the release of an adult Dioctophyma renale through an abdominal drain and three co-infections (Plasmodium malariae, Strongyloides stercoralis and Mansonella perstans).</p

Boerekamps, Anne

De Man, Peter

Janmaat, Vincent T

Koelewijn, Rob

Mendonça Melo, Mariana

Schrama, Yvonne C

Van Hellemond, Jaap J

Verdijk, Rob M

Westerman, Michiel

English

EUR Research Repository

EUR Research Information PortalFirst reported case of an ectopic renal giant worm (Dioctophyme renale) infection inthe abdominal cavityPublished in:Journal of Travel MedicinePublication status and date:Published: 01/05/2022DOI (link to publisher):10.1093/jtm/taac015Document VersionPublisher's PDF, also known as Version of recordDocument License/Available under:CC BYCitation for the published version (APA):Boerekamps, A., Janmaat, V. T., Schrama, Y. C., Westerman, M., Verdijk, R. M., Koelewijn, R., De Man, P., Mendonça Melo,M., & Van Hellemond, J. J. (2022). First reported case of an ectopic renal giant worm (Dioctophyme renale) infection in theabdominal cavity. Journal of Travel Medicine, 29(4), Article taac015. https://doi.org/10.1093/jtm/taac015Link to publication on the EUR Research Information PortalTerms and Conditions of UseExcept as permitted by the applicable copyright law, you may not reproduce or make this material available to any third partywithout the prior written permission from the copyright holder(s). Copyright law allows the following uses of this materialwithout prior permission:            • you may download, save and print a copy of this material for your personal use only;            • you may share the EUR portal link to this material.In case the material is published with an open access license (e.g. a Creative Commons (CC) license), other uses may beallowed. Please check the terms and conditions of the specific license.Take-down policyIf you believe that this material infringes your copyright and/or any other intellectual property rights, you may request itsremoval by contacting us at the following email address: openaccess.library@eur.nl. Please provide us with all the relevantinformation, including the reasons why you believe any of your rights have been infringed. In case of a legitimate complaint,we will make the material inaccessible and/or remove it from the website.© The Author(s) 2022. Published by Oxford University Press on behalf of International Society of Travel Medicine. All rights reserved. For permissions, pleasee-mail: journals.permissions@oup.comJournal of Travel Medicine, 2022, 1–2https://doi.org/10.1093/jtm/taac015Clinical PearlsClinical PearlsFirst reported case of an ectopic renal giant worm(Dioctophyme renale) infection in the abdominal cavityAnne Boerekamps, MD, PhD1,†*, Vincent T. Janmaat, MD, PhD1,2,†,Yvonne C. Schrama, MD, PhD1, Michiel Westerman, MD, PhD1,Rob M. Verdijk, MD, PhD3, Rob Koelewijn4,6, Peter De Man, MD, PhD5,Mariana de Mendonça Melo, MD4,6 and Jaap J. Van Hellemond, MD, PhD4,61Department of Internal Medicine, Franciscus Gasthuis Vlietland Hospital Rotterdam, Kleiweg 500, 3045 PM, Rotterdam,the Netherlands, 2Department of Gastroenterology and Hepatology, Erasmus MC University Medical Center Rotterdam,Doctor Molewaterplein 40, 3015 GD Rotterdam, the Netherlands, 3Department of Pathology, Erasmus MC UniversityMedical Center Rotterdam, Doctor Molewaterplein 40, 3015 GD Rotterdam, the Netherlands, 4Department of MedicalMicrobiology & Infectious Diseases, Erasmus MC University Medical Center Rotterdam, Doctor Molewaterplein 40, 3015GD Rotterdam, the Netherlands, 5Department of Medical Microbiology, Franciscus Gasthuis Vlietland Hospital Rotterdam,Kleiweg 500, 3045 PM, Rotterdam, the Netherlands and 6Institute for Tropical Diseases, Erasmus MC University MedicalCenter, Doctor Molewaterplein 40, 3015 GD Rotterdam, the Netherlands*To whom correspondence should be addressed. Anne Boerekamps, M.D., Ph.D., Franciscus Gasthuis Vlietland Hospital, Kleiweg 500, 3045 PMRotterdam, the Netherlands. Tel: 0616286189; Email: a.boerekamps@franciscus.nl†Contributed equally to this manuscript.Submitted 9 December 2021; Revised 13 January 2022; Editorial Decision 3 February 2022; Accepted 3 February 2022Key words: Dioctophyma renale, giant kidney worm, Dioctophymiasis, gastric perforationA 42-year-old HIV-negative man from Guinee-Bissau hadrecently travelled through Mali Mauretania, Algeria, Libya,and Italy to the Netherlands. He presented at the emergencydepartment with complaints for 1 week of nausea, vomiting andabdominal pain. His condition had worsened and he sufferedfrom partial loss of consciousness and signs of dehydration atthe time of presentation. The patient was diagnosed with acuterenal insufficiency, hospitalized and rehydrated with intravenousfluids. Initially, his condition improved, but after 3 days hedeveloped abdominal pain with acute onset. Within hours hedeveloped a septic profile despite conservative treatment withbroad spectrum antibiotics and intravenous fluids. A laparotomywas performed during which a stomach perforation wasidentified. The surgery went successful without complicationsand a drain was left behind in the upper abdomen. The patientrecovered quickly. After 4 days the abdominal drain wasremoved. During removal, a piece of a large worm, lodged in thedistal tip of the drain, was extracted from the patient (Figure 1).The worm in the drain was identified with certainty as agiant kidney worm (Dioctophyma renale), based on the follow-ing macroscopic morphology; the colour of the freshly isolatedworm, the length and shape of the worm (round and taperedoff to the end).1,2 Furthermore, our patient turned out to haveFigure 1. (A) The freshly released worm before fixation in formalin. (B)The worm after formalin fixation (bar in cm C, HE stained histologicalcross-section shows the contour of the worm (25× magnification). (D)HE stained histological cross-section shows the extensive neutrophilinfiltration with destruction of the internal structures of the worm (200×magnification)three microscopically proven co-infections (Plasmodium malar-iae, Strongyloides stercoralis and Mansonella perstans) for whichhe was successfully treated.D. renale is a large nematode that parasitizes terrestrial,piscivorous mammals, including humans that are sporadicallyinfected with only circa 40 reported cases.2 Female worms canDownloaded from https://academic.oup.com/jtm/advance-article/doi/10.1093/jtm/taac015/6524084 by Erasmus Universiteit Rotterdam user on 01 June 20222 Journal of Travel Medicinegrow up to 100 cm in length, males up to 35 cm. Adult D.renale normally reside in the kidney, where the worm sheds eggsin the urine. These eggs are consumed by aquatic oligochaeteworms where they develop to third-stage larvae. Fish and frogsare common paratenic hosts where the third-stage larvae residein the muscles until the paratenic host is predated upon by thedefinitive host. The larva penetrates the intestinal lining andmigrates to the liver (where the parasite is considered to maturefor ∼50 days) before finally lodging in the kidney. If an adult D.renale is found outside the kidneys in the final host, the infectionis considered ectopic. Effectiveness of anti-parasitic treatmentis unknown and surgical removal is the treatment of choice inmost cases.Our patient denied having eaten raw or undercooked fish,frogs of molluscs and an ultrasound image of his kidneys showedno sign of destruction or infection. Molecular identificationof the worm was unsuccessful,3 which might be explained byformalin fixation and DNA degeneration by the extensive leuko-cyte infiltration in the worm (Figure 1C and D). This indicatesdeath of the worm long before prior to its removal from theabdomen. Because no indications for other causes of stomachperforations were found in gastric biopsies (e.g. Helicobacterpylori infection, malignancy, acidic ulceration or ischemia ongastroscopy), it seems likely that the adult worm induced thegastric perforation either by penetration of the stomach wallfrom the abdominal side by the living worm (which was sub-sequently killed by the resulting inflammatory response), or bydeath of the adult worm in the abdominal cavity in the vicinityof the stomach (which subsequently provoked an inflammatoryresponse that weakened the stomach wall such that perforationwas caused).Our patient is the first reported case with a human D. renaleinfection in the abdominal cavity, whom presented with a gastricperforation and the release of an adult D. renale through anabdominal drain. Clinicians should be aware of rare parasiticinfections in a vulnerable migrant population.Authors’ contributionsB.A.: Data analysis, literature search, writing of manuscript;J.V.T.: Data analysis, literature search, writing of manuscript;S.Y.C.: Data collection, writing of manuscript; W.M.: Data col-lection, writing of manuscript; V.R.M.: Data analysis, figuredesigning and captions; K.R.: Data analysis, figure designing andcaptions; D.MP.: Data analysis, figure designing and captions; de.M.M.M.: Writing of manuscript, data interpretation, literaturesearch; Van H.J.J.: Writing of manuscript, data interpretation,literature search.AcknowledgementsWe thank Dr Corné Klaassen and Maarten Sarink, MD, for theirexpert attempt to identify D. renale by molecular methods.FundingThis work was not supported by any funding.Conflicts of interest: None declared.References1. Yang F, Zhang W, Gong B, Yao L, Liu A, Ling H. A human caseof Dioctophyma renale (giant kidney worm) accompanied by renalcancer and a retrospective study of dioctophymiasis. Un cas humainde Dioctophyma renale (le ver géant du rein) accompagné d’un cancerdu rein, et étude rétrospective de la dioctophymiose. Parasite 2019;26:22.2. Carra Perera S, Silveira Mascarenhas C, Brum Cleff M, Müller G, daSilva Rappeti JC. Dioctophimosis: a parasitic zoonosis of public healthimportance. Adv Exp Med Biol 2021; 1306:129–42.3. Tokiwa T, Ueda W, Takatsuka S et al. The first genetically con-firmed case of Dioctophyme renale (Nematoda: Dioctophymatida)in a patient with a subcutaneous nodule. Parasitol Int 2014; 63:143–7.Downloaded from https://academic.oup.com/jtm/advance-article/doi/10.1093/jtm/taac015/6524084 by Erasmus Universiteit Rotterdam user on 01 June 2022

First reported case of an ectopic renal giant worm (Dioctophyme renale) infection in the abdominal cavity

© The Author(s) 2022. Published by Oxford University Press on behalf of International Society of Travel Medicine. All rights reserved. For permissions, pleasee-mail: journals.permissions@oup.comJournal of Travel Medicine, 2022, 1–2https://doi.org/10.1093/jtm/taac015Clinical PearlsClinical PearlsFirst reported case of an ectopic renal giant worm(Dioctophyme renale) infection in the abdominal cavityAnne Boerekamps, MD, PhD1,†*, Vincent T. Janmaat, MD, PhD1,2,†,Yvonne C. Schrama, MD, PhD1, Michiel Westerman, MD, PhD1,Rob M. Verdijk, MD, PhD3, Rob Koelewijn4,6, Peter De Man, MD, PhD5,Mariana de Mendonça Melo, MD4,6 and Jaap J. Van Hellemond, MD, PhD4,61Department of Internal Medicine, Franciscus Gasthuis Vlietland Hospital Rotterdam, Kleiweg 500, 3045 PM, Rotterdam,the Netherlands, 2Department of Gastroenterology and Hepatology, Erasmus MC University Medical Center Rotterdam,Doctor Molewaterplein 40, 3015 GD Rotterdam, the Netherlands, 3Department of Pathology, Erasmus MC UniversityMedical Center Rotterdam, Doctor Molewaterplein 40, 3015 GD Rotterdam, the Netherlands, 4Department of MedicalMicrobiology & Infectious Diseases, Erasmus MC University Medical Center Rotterdam, Doctor Molewaterplein 40, 3015GD Rotterdam, the Netherlands, 5Department of Medical Microbiology, Franciscus Gasthuis Vlietland Hospital Rotterdam,Kleiweg 500, 3045 PM, Rotterdam, the Netherlands and 6Institute for Tropical Diseases, Erasmus MC University MedicalCenter, Doctor Molewaterplein 40, 3015 GD Rotterdam, the Netherlands*To whom correspondence should be addressed. Anne Boerekamps, M.D., Ph.D., Franciscus Gasthuis Vlietland Hospital, Kleiweg 500, 3045 PMRotterdam, the Netherlands. Tel: 0616286189; Email: a.boerekamps@franciscus.nl†Contributed equally to this manuscript.Submitted 9 December 2021; Revised 13 January 2022; Editorial Decision 3 February 2022; Accepted 3 February 2022Key words: Dioctophyma renale, giant kidney worm, Dioctophymiasis, gastric perforationA 42-year-old HIV-negative man from Guinee-Bissau hadrecently travelled through Mali Mauretania, Algeria, Libya,and Italy to the Netherlands. He presented at the emergencydepartment with complaints for 1 week of nausea, vomiting andabdominal pain. His condition had worsened and he sufferedfrom partial loss of consciousness and signs of dehydration atthe time of presentation. The patient was diagnosed with acuterenal insufficiency, hospitalized and rehydrated with intravenousfluids. Initially, his condition improved, but after 3 days hedeveloped abdominal pain with acute onset. Within hours hedeveloped a septic profile despite conservative treatment withbroad spectrum antibiotics and intravenous fluids. A laparotomywas performed during which a stomach perforation wasidentified. The surgery went successful without complicationsand a drain was left behind in the upper abdomen. The patientrecovered quickly. After 4 days the abdominal drain wasremoved. During removal, a piece of a large worm, lodged in thedistal tip of the drain, was extracted from the patient (Figure 1).The worm in the drain was identified with certainty as agiant kidney worm (Dioctophyma renale), based on the follow-ing macroscopic morphology; the colour of the freshly isolatedworm, the length and shape of the worm (round and taperedoff to the end).1,2 Furthermore, our patient turned out to haveFigure 1. (A) The freshly released worm before fixation in formalin. (B)The worm after formalin fixation (bar in cm C, HE stained histologicalcross-section shows the contour of the worm (25× magnification). (D)HE stained histological cross-section shows the extensive neutrophilinfiltration with destruction of the internal structures of the worm (200×magnification)three microscopically proven co-infections (Plasmodium malar-iae, Strongyloides stercoralis and Mansonella perstans) for whichhe was successfully treated.D. renale is a large nematode that parasitizes terrestrial,piscivorous mammals, including humans that are sporadicallyinfected with only circa 40 reported cases.2 Female worms canDownloaded from https://academic.oup.com/jtm/advance-article/doi/10.1093/jtm/taac015/6524084 by Erasmus Universiteit Rotterdam user on 01 June 20222 Journal of Travel Medicinegrow up to 100 cm in length, males up to 35 cm. Adult D.renale normally reside in the kidney, where the worm sheds eggsin the urine. These eggs are consumed by aquatic oligochaeteworms where they develop to third-stage larvae. Fish and frogsare common paratenic hosts where the third-stage larvae residein the muscles until the paratenic host is predated upon by thedefinitive host. The larva penetrates the intestinal lining andmigrates to the liver (where the parasite is considered to maturefor ∼50 days) before finally lodging in the kidney. If an adult D.renale is found outside the kidneys in the final host, the infectionis considered ectopic. Effectiveness of anti-parasitic treatmentis unknown and surgical removal is the treatment of choice inmost cases.Our patient denied having eaten raw or undercooked fish,frogs of molluscs and an ultrasound image of his kidneys showedno sign of destruction or infection. Molecular identificationof the worm was unsuccessful,3 which might be explained byformalin fixation and DNA degeneration by the extensive leuko-cyte infiltration in the worm (Figure 1C and D). This indicatesdeath of the worm long before prior to its removal from theabdomen. Because no indications for other causes of stomachperforations were found in gastric biopsies (e.g. Helicobacterpylori infection, malignancy, acidic ulceration or ischemia ongastroscopy), it seems likely that the adult worm induced thegastric perforation either by penetration of the stomach wallfrom the abdominal side by the living worm (which was sub-sequently killed by the resulting inflammatory response), or bydeath of the adult worm in the abdominal cavity in the vicinityof the stomach (which subsequently provoked an inflammatoryresponse that weakened the stomach wall such that perforationwas caused).Our patient is the first reported case with a human D. renaleinfection in the abdominal cavity, whom presented with a gastricperforation and the release of an adult D. renale through anabdominal drain. Clinicians should be aware of rare parasiticinfections in a vulnerable migrant population.Authors’ contributionsB.A.: Data analysis, literature search, writing of manuscript;J.V.T.: Data analysis, literature search, writing of manuscript;S.Y.C.: Data collection, writing of manuscript; W.M.: Data col-lection, writing of manuscript; V.R.M.: Data analysis, figuredesigning and captions; K.R.: Data analysis, figure designing andcaptions; D.MP.: Data analysis, figure designing and captions; de.M.M.M.: Writing of manuscript, data interpretation, literaturesearch; Van H.J.J.: Writing of manuscript, data interpretation,literature search.AcknowledgementsWe thank Dr Corné Klaassen and Maarten Sarink, MD, for theirexpert attempt to identify D. renale by molecular methods.FundingThis work was not supported by any funding.Conflicts of interest: None declared.References1. Yang F, Zhang W, Gong B, Yao L, Liu A, Ling H. A human caseof Dioctophyma renale (giant kidney worm) accompanied by renalcancer and a retrospective study of dioctophymiasis. Un cas humainde Dioctophyma renale (le ver géant du rein) accompagné d’un cancerdu rein, et étude rétrospective de la dioctophymiose. Parasite 2019;26:22.2. Carra Perera S, Silveira Mascarenhas C, Brum Cleff M, Müller G, daSilva Rappeti JC. Dioctophimosis: a parasitic zoonosis of public healthimportance. Adv Exp Med Biol 2021; 1306:129–42.3. Tokiwa T, Ueda W, Takatsuka S et al. The first genetically con-firmed case of Dioctophyme renale (Nematoda: Dioctophymatida)in a patient with a subcutaneous nodule. Parasitol Int 2014; 63:143–7.Downloaded from https://academic.oup.com/jtm/advance-article/doi/10.1093/jtm/taac015/6524084 by Erasmus Universiteit Rotterdam user on 01 June 2022

https://pure.eur.nl/files/60720334/First_reported_case_of_an_ectopic_renal_giant_worm_Dioctophyme_renale_infection_in_the_abdominal_cavity.pdf

First reported case of an ectopic renal giant worm (Dioctophyme renale) infection in the abdominal cavity

Abstract

Similar works

Full text

Available Versions

EUR Research Repository

EUR Research Repository

EUR Research Repository