A rapidly advancing intraosseous angiosarcoma masquerading as an infected pathological periprosthetic fracture: a case report
DOI:
https://doi.org/10.18203/issn.2455-4510.IntJResOrthop20250466Keywords:
Angiosarcoma, Prosthetic Joint infection, Musculoskeletal tumours, Periprosthetic fracture, Hypervascular lesionAbstract
We present a case of a rapidly progressive angiosarcoma that presented initially as a pathological fracture in our institution after a fall. Mdm A, a 74-year-old Chinese lady initially presented with non-specific thigh pain prior to an unwitnessed fall, following a recent bipolar hemiarthroplasty 3 months before current presentation. Blood markers were grossly within acceptable limits and computed tomography (CT) scan revealed displaced greater and lesser trochanteric periprosthetic fractures with subsidence of femoral stem, and findings suggestive of intramuscular haematomas. She subsequently turned septic on day of planned surgery for a revision arthroplasty with markedly raised inflammatory markers. Revision surgery was postponed, with repeat imaging noting evolving fluid collections suspicious of an infected prosthetic joint infection (PJI). Joint washout for presumptive PJI performed 1-week later yielded negative histopathology and cultures. She developed respiratory compromise a few days later. CT-thorax revealed bilateral pleural effusions with small spontaneous haemo-pneumothoraces which again yielded negative cultures and cytology. Eventually a repeat CT-hip 1-month post admission showed new soft tissue lesions in the superficial muscle layers. Biopsies performed returned as intermediate grade angiosarcoma with positive CD31 and ERG stainings. This is the first case study to describe an aggressive metastatic angiosarcoma mimicking a PJI on initial presentation. Angiosarcomas are very rare and presentations may also be highly varied. Diagnosis of angiosarcoma may be made difficult by the absence of clear lesions to biopsy, as in this particular case. A high clinical suspicion of a pathological periprosthetic fracture is needed when diagnosing patients who present atypically for PJIs.
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References
Cao J, Wang J, He C, Fang M. Angiosarcoma: a review of diagnosis and current treatment. Am J Cancer Res. 2019;9(11):2303-13.
Chan JY, Tan GF, Yeong J, Ong CW, Ng DYX, Lee E, et al. Clinical implications of systemic and local immune responses in human angiosarcoma. NPJ Precis Onc. 2021;5(1):11. DOI: https://doi.org/10.1038/s41698-021-00150-x
Gaballah AH, Jensen CT, Palmquist S, Pickhardt PJ, Duran A, Broering G, et al. Angiosarcoma: clinical and imaging features from head to toe. Br J Radiol. 2017;90(1075):20170039. DOI: https://doi.org/10.1259/bjr.20170039
Chen TW, Pang A, Puhaindran ME, Maw MM, Loong HH, Sriuranpong V, et al. The treatment landscape of advanced angiosarcoma in Asia—A multi‐national collaboration from the Asian Sarcoma Consortium. Cancer Sci. 2021;112(3):1095-104. DOI: https://doi.org/10.1111/cas.14793
Palmerini E, Maki RG, Staals EL, Alberghini M, Antonescu CR, Ferrari C, et al. Primary angiosarcoma of bone: A retrospective analysis of 60 patients from two institutions. Am J Clin Oncol. 2014;37(6):528-34. DOI: https://doi.org/10.1097/COC.0b013e31827defa1
Bhaludin BN, Thway K, Adejolu M, Renn A, Kelly-Morland C, Fisher C, et al. Imaging features of primary sites and metastatic patterns of angiosarcoma. Insights Imaging. 2021;12(1):189. DOI: https://doi.org/10.1186/s13244-021-01129-9
Jeyaraman M, Muthu S, Prabhakar M, Jeyaraman N, Agarwal G, Jain R. Primary Angiosarcoma of Humerus – A Case Report and Literature Review. J Orthop Case Rep. 2021;11(5):12-7. DOI: https://doi.org/10.13107/jocr.2021.v11.i05.2186
Shukla S, Acharya S, Bhola N, Chaudhary M, Borle Mr, Grover M. Central Haemangioma (Intraosseous) of Jaw. J Med. 2012;13(2):222-6. DOI: https://doi.org/10.3329/jom.v13i2.12762
Baliaka A, Balis G, Michalopoulou-Manoloutsiou E, Papanikolaou A, Nikolaidou A. Primary angiosarcoma of bone. A case report. Hippokratia. 2013;17(2):180-2.
Romanò CL, Petrosillo N, Argento G, Sconfienza LM, Treglia G, Alavi A, et al. The Role of Imaging Techniques to Define a Peri-Prosthetic Hip and Knee Joint Infection: Multidisciplinary Consensus Statements. J Clin Med. 2020;9(8):2548. DOI: https://doi.org/10.3390/jcm9082548
Zajonz D, Brand A, Lycke C, Özkurtul O, Theopold J, Spiegl UJA, et al. Risk factors for early infection following hemiarthroplasty in elderly patients with a femoral neck fracture. Eur J Trauma Emerg Surg. 2019;45(2):207-12. DOI: https://doi.org/10.1007/s00068-018-0909-8
Palmerini E, Leithner A, Windhager R, Gosheger G, Boye K, Laitinen M, et al. Angiosarcoma of bone: a retrospective study of the European Musculoskeletal Oncology Society (EMSOS). Sci Rep. 2020;10(1):10853. DOI: https://doi.org/10.1038/s41598-020-66579-5
Murphey MD, Fairbairn KJ, Parman LM, Baxter KG, Parsa MB, Smith WS. From the archives of the AFIP. Musculoskeletal angiomatous lesions: radiologic-pathologic correlation. RadioGraphics. 1995;15(4):893-917. DOI: https://doi.org/10.1148/radiographics.15.4.7569134
Vermaat M, Vanel D, Kroon HM, Verbeke SLJ, Alberghini M, Bovee JVMG, et al. Vascular tumors of bone: Imaging findings. Eur J Radiol. 2011;77(1):13-8. DOI: https://doi.org/10.1016/j.ejrad.2010.06.052
Vasireddi N, Neitzke CC, Chandi SK, Cororaton AD, Driscoll DA, Sculco PK, et al. Early Periprosthetic Femur Fractures After Primary Cementless Total Hip Arthroplasty: High Risk of Periprosthetic Joint Infection and Subsequent Reoperation. J Arthroplast. 2024;39(4):1083-7. DOI: https://doi.org/10.1016/j.arth.2023.10.037
Tateishi U, Hasegawa T, Kusumoto M, Yamazaki N, Iinuma G, Muramatsu Y, et al. Metastatic Angiosarcoma of the Lung: Spectrum of CT Findings. Am J Roentgenol. 2003;180(6):1671-4. DOI: https://doi.org/10.2214/ajr.180.6.1801671
Geller RL, Hookim K, Sullivan HC, Stuart LN, Edgar MA, Reid MD. Cytologic features of angiosarcoma: A review of 26 cases diagnosed on FNA. Cancer Cytopathol. 2016;124(9):659-68. DOI: https://doi.org/10.1002/cncy.21726
Buehler D, Rice SR, Moody JS, Rush P, Hafez GR, Attia S, et al. Angiosarcoma Outcomes and Prognostic Factors: A 25-Year Single Institution Experience. Am J Clin Oncol. 2014;37(5):473-9. DOI: https://doi.org/10.1097/COC.0b013e31827e4e7b
Liu K, Layfield LJ. Cytomorphologic features of angiosarcoma on fine needle aspiration biopsy. Acta Cytol. 1999;43(3):407-15. DOI: https://doi.org/10.1159/000331089
Naka N, Ohsawa M, Tomita Y, Kanno H, Uchida A, Myoui A, et al. Prognostic factors in angiosarcoma: A multivariate analysis of 55 cases. J Surg Oncol. 1996;61(3):170-6. DOI: https://doi.org/10.1002/(SICI)1096-9098(199603)61:3<170::AID-JSO2>3.0.CO;2-8