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Osteoid osteoma appearing after bony fracture in a girl with osteogenesis imperfecta

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Abstract

Osteoid osteoma (OO) is a common, benign bone tumor. However, there are no case reports of OO associated with osteogenesis imperfecta (OI), or pathological fractures in OO. A 3-year-old girl with OI sustained a complete right tibial diaphyseal fracture. Bony fusion was completed after 4 months of conservative therapy; nevertheless, 18 months later spontaneous pain appeared at the fracture site, without any cause. Plain radiographs showed a newly apparent, rounded area of translucency 1 cm in diameter, just overlapping the previous fracture. Images obtained using three-dimensional time-resolved contrast-enhanced magnetic resonance angiography showed strong central enhancement in the early phase, with an apparent nidus, suggesting the diagnosis of OO. Nineteen months after the first fracture, while skipping, the patient refractured her tibial diaphysis at the site of the previous fracture. This is a very rare case of OO, apparently co-existing with OI and leading to a bony fracture. In our case, the combination of bone fragility in OI and a recent fracture at the site of the OO may have caused the re-fracture.

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The data is available from the corresponding author upon reasonable request.

References

  1. Tepelenis K, Skandalakis GP, Papathanakos G, et al. Osteoid osteoma: an updated review of epidemiology, pathogenesis, clinical presentation, radiological features, and treatment options. In Vivo. 2021;35(4):1929–38.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  2. Carneiro BC, Da Cruz IAN, Ormond Filho AG, et al. Osteoid osteoma: the great mimicker. Insights Imaging. 2021;12(1):32.

    Article  PubMed  PubMed Central  Google Scholar 

  3. Goto T, Shinoda Y, Okuma T, Ogura K, Tsuda Y, Yamakawa K, et al. Administration of nonsteroidal anti-inflammatory drugs accelerates spontaneous healing of osteoid osteoma. Arch Orthop Trauma Surg. 2011;131(5):619–25.

    Article  PubMed  Google Scholar 

  4. Kneisl JS, Simon MA. Medical management compared with operative treatment for osteoid-osteoma. J Bone Joint Surg Am. 1992;74(2):179–85.

    Article  CAS  PubMed  Google Scholar 

  5. Orth P, Kohn D. Diagnostics and treatment of osteoid osteoma. Orthopade. 2017;46(6):510–21.

    Article  CAS  PubMed  Google Scholar 

  6. Talawadekar GD, Muller M, Zahn H. Benign self-limiting cystic lesion after lower end radius fracture in a child. Indian J Orthop. 2009;43(1):99–101.

    Article  PubMed  PubMed Central  Google Scholar 

  7. Dahapute AA, Gala RB, Dhar SB, Viranii S, Vaishnav A. Radial artery pseudoaneurysm in a post-operative case of midshaft radius fracture. J Orthop Case Rep. 2017;7(6):3–5.

    PubMed  PubMed Central  Google Scholar 

  8. Weatherall PT, Maale GE, Mendelsohn DB, et al. Chondroblastoma: classic and confusing appearance at MR imaging. Radiology. 1994;190(2):467–74.

    Article  CAS  PubMed  Google Scholar 

  9. Desimpel J, Posadzy M, Vanhoenacker F. The many faces of osteomyelitis: a pictorial review. J Belg Soc Radiol. 2017;101(1):24.

    Article  PubMed  PubMed Central  Google Scholar 

  10. Costelloe CM, Murphy WA, Madewell JE. Imaging chronic sclerosing osteitis of the diaphysis of tubular bones. AJR Am J Roentgenol. 2009;192(3):736–42.

    Article  PubMed  Google Scholar 

  11. Maygarden SJ, Askin FB, Siegal GP, et al. Ewing sarcoma of bone in infants and toddlers. A clinicopathologic report from the Intergroup Ewing’s Study. Cancer. 1993;71(6):2109–18.

  12. Duda SH, Laniado M, Schick F, Claussen CD. The double-line sign of osteonecrosis: evaluation on chemical shift MR images. Eur J Radiol. 1993;16(3):233–8.

    Article  CAS  PubMed  Google Scholar 

  13. Martí-Bonmatí L, Aparisi F, Poyatos C, Vilar J. Brodie abscess: MR imaging appearance in 10 patients. J Magn Reson Imaging. 1993;3(3):543–6.

    Article  PubMed  Google Scholar 

  14. Davies AM, Grimer R. The penumbra sign in subacute osteomyelitis. Eur Radiol. 2005;15(6):1268–70.

    Article  CAS  PubMed  Google Scholar 

  15. Greenspan A. Bone island (enostosis): current concept—a review. Skelet Radiol. 1995;24(2):111–5.

    Article  CAS  Google Scholar 

  16. Van Ruyssevelt CEA, Vranckx P. Subungual glomus tumor: emphasis on MR angiography. AJR Am J Roentgenol. 2004;182(1):263–4.

    Article  PubMed  Google Scholar 

  17. Goosens V, Vanhoenacker FM, Samson I, Brys P. Longitudinal cortical split sign as a potential diagnostic feature for cortical osteitis. JBR-BTR. 2010;93(2):77–80.

    CAS  PubMed  Google Scholar 

  18. Bilreiro C, Bahia C, Castro MOE. Longitudinal stress fracture of the femur: a rare presentation. Eur J Radiol Open. 2016;3:31–4.

    Article  PubMed  PubMed Central  Google Scholar 

  19. Barlow E, Davies AM, Cool WP, Barlow D, Mangham DC. Osteoid osteoma and osteoblastoma: novel histological and immunohistochemical observations as evidence for a single entity. J Clin Pathol. 2013;66(9):768–74.

    Article  CAS  PubMed  Google Scholar 

  20. Noordin S, Allana S, Hilal K, et al. Osteoid osteoma: contemporary management. Orthop Rev (Pavia). 2018;10(3):7496.

    Article  PubMed  Google Scholar 

  21. Adil A, Hoeffel C, Fikry T. Osteoid osteoma after a fracture of the distal radius. Am J Roentgenol. 1996;167(1):145–6.

    Article  CAS  Google Scholar 

  22. Vancamp E, Vanhoenacker FM, Vanderschueren G. Post-traumatic osteoid osteoma in an 18-year-old adolescent. BJR Case Rep. 2015;1(2):20150141. https://doi.org/10.1259/bjrcr.20150141.

  23. Renaud A, Aucourt J, Weill J, et al. Radiographic features of osteogenesis imperfecta. Insights Imaging. 2013;4(4):417–29.

    Article  PubMed  PubMed Central  Google Scholar 

  24. Samaila EM, Ditta A, Lugani G, Regis D, Leigheb M, Leigheb M, et al. Post-traumatic cystic lesion following radius fracture: a case report and literature review. Acta Biomed. 2019;90(12-S):162–6.

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Acknowledgements

We thank Drs. Kentaro Hayashi and Yoshitsugu Fukuda as the attending orthopedic physicians and Dr. Kosuke Nakano as an attending pathologist for this particular case. This case was presented at the 33rd annual meeting of the Japanese Society of Musculoskeletal Radiology, Kitakyushu, Japan.

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Authors and Affiliations

  1. Department of Radiology, National Center for Child Health and Development, 2-10-1 Okura, Setagaya, Tokyo, Japan

    Kei Sakamoto, Osamu Miyazaki, Ayako Imai, Reiko Okamoto, Yoshiyuki Tsutsumi, Mikiko Miyasaka & Shunsuke Nosaka

  2. Division of Orthopaedic Surgery, National Center for Child Health and Development, 2-10-1 Okura, Setagaya, Tokyo, Japan

    Atsuhito Seki

  3. Department of Pathology, National Center for Child Health and Development, 2-10-1 Okura, Setagaya, Tokyo, Japan

    Takako Yoshioka

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  1. Kei Sakamoto
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  2. Osamu Miyazaki
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  5. Yoshiyuki Tsutsumi
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  9. Shunsuke Nosaka
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Correspondence to Osamu Miyazaki.

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Sakamoto, K., Miyazaki, O., Imai, A. et al. Osteoid osteoma appearing after bony fracture in a girl with osteogenesis imperfecta. Skeletal Radiol (2024). https://doi.org/10.1007/s00256-024-04672-w

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  • DOI: https://doi.org/10.1007/s00256-024-04672-w

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