Pathogenetic phenotypes of bone cement implantation syndrome in pediatric oncology patients: Case reports
- Authors: Leonov N.P.1, Leonova V.A.1, Schukin V.V.1, Shcherbakov A.P.1, Madonov P.G.2, Lazarev V.V.1,3, Spiridonova E.A.4,5, Grachev N.S.1
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Affiliations:
- Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology
- Novosibirsk State Medical University
- Pirogov Russian National Research Medical University
- Russian University of Medicine
- Federal Research and Clinical Center of Intensive Care Medicine and Rehabilitology
- Issue: Vol 15, No 1 (2025)
- Pages: 91-100
- Section: Case reports
- URL: https://journals.rcsi.science/2219-4061/article/view/312986
- DOI: https://doi.org/10.17816/psaic1885
- ID: 312986
Cite item
Abstract
Bone cement implantation syndrome in pediatric oncology patients remains poorly understood. A multimodal pathogenetic model determines the existence of two distinct clinical and pathophysiological phenotypes of this condition: the anaphylactic phenotype (distributive shock) and the embolic phenotype (obstructive shock). Both phenotypes are associated with coagulopathy, with thrombotic catastrophes representing their most severe manifestation. The pathways of thrombotic complications depend on the clinical and pathophysiological phenotype of this critical condition: in anaphylactic bone cement implantation syndrome, they are primarily driven by microthrombogenesis, whereas in embolic bone cement implantation syndrome, both microthrombogenesis and fibrinogenesis contribute to thrombosis. In the first clinical case, bone cement implantation syndrome developed through an anaphylactic mechanism. A boy with femoral osteosarcoma underwent bone cement spacer implantation following tumor resection. We assume that sensitization occurred during this period. This is supported by the presence of a periosteal reaction in the upper third of the right femur, as revealed by computed tomography (CT). During knee endoprosthesis implantation, the patient developed severe hemodynamic instability, cardiac rhythm disturbances, and oxygenation impairment. Despite the characteristic microthrombogenesis mechanism of this pathophysiological phenotype, multiorgan failure and life-threatening thrombotic complications were successfully averted due to effective anti-shock measures and early initiation of heparin therapy. Second clinical case illustrates the embolic phenotype of bone cement implantation syndrome. A boy with tibial osteosarcoma experienced hypotension, tachyarrhythmia, desaturation, and hypocapnia following bone cement application during endoprosthetic surgery. Postoperatively, desaturation persisted. CT revealed a mural defect in the left pulmonary artery and segmental obstructions in multiple branches of both lungs, along with elevated D-dimer levels and echocardiographic evidence of increased right heart pressure. By postoperative day 20, oxygen saturation normalized (the patient was breathing ambient air). CT imaging showed resolution of the filling defect in the left pulmonary artery; however, signs of segmental pulmonary artery obstruction remained in the upper and middle lobes of the right lung and the lower lobe of the left lung. Thus, the hemodynamic catastrophe in the embolic phenotype of bone cement implantation syndrome represents a classic presentation of obstructive shock with subsequent thrombotic complications, driven by the combined mechanisms of microthrombogenesis and fibrinogenesis. The proposed pathogenetic phenotyping of bone cement implantation syndrome allows for a targeted approach to the prevention and treatment of hemodynamic and thrombotic complications in affected patients. This approach appears to be relevant and has the potential to reduce the incidence of adverse outcomes and complications.
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##article.viewOnOriginalSite##About the authors
Nikolai P. Leonov
Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology
Author for correspondence.
Email: NikoLeonov@ya.ru
ORCID iD: 0000-0002-4364-8937
SPIN-code: 2128-9110
MD, Cand. Sci. (Medicine)
Russian Federation, MoscowViktoria A. Leonova
Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology
Email: NikoLeonov@ya.ru
ORCID iD: 0009-0008-7200-8278
Russian Federation, Moscow
Vladislav V. Schukin
Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology
Email: schukin.vv@ya.ru
ORCID iD: 0000-0002-7945-2565
SPIN-code: 4572-8611
MD, Cand. Sci. (Medicine)
Russian Federation, MoscowAlexey P. Shcherbakov
Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology
Email: alexey.shcherbakov@dgoi.ru
ORCID iD: 0000-0001-8129-0545
Russian Federation, Moscow
Pavel G. Madonov
Novosibirsk State Medical University
Email: pmadonov@yandex.ru
ORCID iD: 0000-0002-1093-8938
SPIN-code: 9457-4580
MD, Dr. Sci. (Medicine), Professor
Russian Federation, NovosibirskVladimir V. Lazarev
Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology; Pirogov Russian National Research Medical University
Email: lazarev_vv@inbox.ru
ORCID iD: 0000-0001-8417-3555
SPIN-code: 4414-0677
MD, Dr. Sci. (Medicine), Professor
Russian Federation, Moscow; MoscowElena A. Spiridonova
Russian University of Medicine; Federal Research and Clinical Center of Intensive Care Medicine and Rehabilitology
Email: spiridonova.e.a@gmail.com
ORCID iD: 0000-0002-5230-5725
SPIN-code: 1729-8002
MD, Dr. Sci. (Medicine), Professor
Russian Federation, Moscow; MoscowNikolai S. Grachev
Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology
Email: nick-grachev@yandex.ru
ORCID iD: 0000-0002-4451-3233
SPIN-code: 2836-2349
MD, Dr. Sci. (Medicine)
Russian Federation, MoscowReferences
- Vaishya R, Chauhan M, Vaish A. Bone cement. J Clin Orthop Trauma. 2013;4(4):157–163. doi: 10.1016/j.jcot.2013.11.005
- Bonfait H, Delaunay C, De Thomasson E, et al. Bone cement implantation syndrome in hip arthroplasty: Frequency, severity and prevention. Orthop Traumatol Surg Res. 2022;108(2):103139. doi: 10.1016/j.otsr.2021.103139 EDN: VLXUNG
- Al-Husinat L, Jouryyeh B, Al Sharie S, et al. Bone cement and its anesthetic complications: a narrative review. J Clin Med. 2023;12(6):2105. doi: 10.3390/jcm12062105 EDN: FYOEKP
- Schwarzkopf E, Sachdev R, Flynn J, et al. Occurrence, risk factors, and outcomes of bone cement implantation syndrome after hemi and total hip arthroplasty in cancer patients. J Surg Oncol. 2019;120(6):1008–1015. doi: 10.1002/jso.25675
- Yang TH, Yang RS, Lin CP, et al. Bone cement implantation syndrome in bone tumor surgeries: incidence, risk factors, and clinical experience. Orthop Surg. 2021;13(1):109–115. doi: 10.1111/os.12842 EDN: WFRYVE
- Donaldson AJ, Thomson HE, Harper NJ, et al. Bone cement implantation syndrome. Br J Anaesth. 2009;102(1):12–22. doi: 10.1093/bja/aen328
- Kalra A, Sharma A, Palaniswamy C, et al. Diagnosis and management of bone cement implantation syndrome: case report and brief review. Am J Ther. 2013;20(1):121–125. doi: 10.1097/MJT.0b013e31820b3de3
- Segerstad MHA. The bone cement implantation syndrome — epidemiology, pathophysiology and prevention [Doctoral thesis]. University of Gothenburg: Sweden; 2019. Available from: https://gupea.ub.gu.se/handle/2077/60777
- Chang JC. Disseminated intravascular coagulation: new identity as endotheliopathy-associated vascular microthrombotic disease based on in vivo hemostasis and endothelial molecular pathogenesis. Thrombosis J. 2020;18(1):1–21. doi: 10.1186/s12959-020-00231-0 EDN: KLNHDV
- Dahl OE, Pripp AH, Jaradeh M, et al. The bone cement hypercoagulation syndrome: pathophysiology, mortality, and prevention. Clin Appl Thromb Hemost. 2023;29. doi: 10.1177/10760296231198036 EDN: VYMNRA
- Reber LL, Hernandez JD, Galli SJ. The pathophysiology of anaphylaxis. J Allergy Clin Immunol. 2017;140(2):335–348. doi: 10.1016/j.jaci.2017.06.003
- Abbas M, Moussa M, Akel H. Type I hypersensitivity reaction. In: StatPearls. Treasure Island (FL): StatPearls Publishing; 2023.
- García-Mansilla A, Castro Lalín A, Holc F, et al. Intraoperative unfractionated heparin before femoral component cementation should be avoided in femoral neck fracture treated with hybrid total hip arthroplasty. Eur J Orthop Surg Traumatol. 2023;33(6):2547–2554. doi: 10.1007/s00590-023-03472-7 EDN: UTWZVN
- Hitti WA, Wali RK, Weinman EJ, et al. Cholesterol embolization syndrome induced by thrombolytic therapy. Am J Cardiovasc Drugs. 2008;8(1):27–34. doi: 10.2165/00129784-200808010-00004 EDN: LLJGUK
- Berkun Y, Haviv YS, Schwartz LB, et al. Heparin-induced recurrent anaphylaxis. Clin Exp Allergy. 2004;34(12):1916–1918. doi: 10.1111/j.1365-2222.2004.02129.x
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