Pediatric Hematology/Oncology and Immunopathology

Вопросы гематологии/онкологии и иммунопатологии в педиатрии

1726-17082414-9314

Fund Doctors, Innovations, Science for Children

431

10.24287/1726-1708-2020-19-4-150-157

CLINICAL OBSERVATIONS

КЛИНИЧЕСКИЕ НАБЛЮДЕНИЯ

The transformation of aneurysmal bone cyst into telangiectatic osteosarcoma: the development of a second tumour or a wrong initial diagnosis?

Трансформация аневризмальной костной кисты в телеангиэктатическую остеосаркому: развитие второй опухоли или ошибочный первоначальный диагноз?

https://orcid.org/0000-0002-9054-5068

Zhukov

N. V.

Жуков

Н. В.

Russian Federation

Nikolay V. Zhukov, Dr. med. sci, Professor, Head of the Department of Interdisciplinary Oncology; Assistant Professor at the Department of Oncology, Hematology and Radiotherapy

1 Samory Mashela St., Moscow 117997

Жуков Николай Владимирович, д-р мед. наук, профессор, заведующий отделом междисциплинарной онкологии; доцент кафедры онкологии, гематологии и лучевой терапии

117997, Москва, ул. Саморы Машела, 1

zhukov.nikolay@rambler.ru

https://orcid.org/0000-0001-9450-125X

Kazakova

L. L.

Казакова

Л. Л.

Russian Federation

Moscow

Москва

Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology of Ministry of Healthcare of the Russian FederationФГБУ «Национальный медицинский исследовательский центр детской гематологии, онкологии и иммунологии им. Дмитрия Рогачева» Минздрава России

N.I. Pirogov Russian National Research Medical University of Ministry of Healthcare of the Russian FederationФГБОУ ВО «Российский национальный исследовательский медицинский университет им. Н.И. Пирогова» Минздрава России

08122020

194

1501572112202021122020

2025

«D. Rogachev NMRCPHOI»

ФГБУ «НМИЦ ДГОИ им. Дмитрия Рогачева» Минздрава России

https://creativecommons.org/licenses/by/4.0

https://hemoncim.com/jour/article/view/431

Aneurysmal bone cyst (ABC) and telangiectatic osteosarcoma (TOS) share a lot of similarities in terms of the initial clinical presentation, the radiological semiotics and even the morphological pattern, however, they are completely different nosological entities requiring absolutely different treatment approaches. This article reports a clinical case of TOS of the femur which developed 1.5 years after the treatment of ABC of the same location and presents an analysis of literature on the differential diagnosis of these diseases, the modern concept of the pathogenesis of ABC and the possibility of the transformation of ABC into TOS. The patient's parents gave consent to the use of their child's data, including photographs, for research purposes and in publications.

Аневризмальная костная киста (АКК) и телеангиэктатическая остеосаркома (ТОС) обладают крайне высоким сходством по инициальной клинической картине, рентгеносемиотике и даже морфологической картине, но при этом являются совершенно разными нозологическими единицами, требующими абсолютно разного лечения. В статье приводится клинический случай развития ТОС бедренной кости через 1,5 года после лечения АКК той же локализации, а также анализ литературных данных о дифференциальной диагностике этих заболеваний, современной концепции патогенеза АКК и возможности трансформации АКК в ТОС. Родители дали согласие на использование информации, в том числе фотографий ребенка, в научных исследованиях и публикациях.

aneurysmal bone cysttelangiectatic osteosarcomatransformationpathogenesisdifferential diagnosisUSP6 oncogene

аневризмальная костная кистателеангиэктатическая остеосаркоматрансформацияпатогенездифференциальный диагнозонкоген USP6

1. Fletcher C.D.M., Bridge J.A., Hogendoorn P.C.W., Mertens F. WHO Classification of Tumors of Soft Tissue and Bone. 4th Ed. 2013. Рр. 302–304, 281–296.

Fletcher C.D.M., Bridge J.A., Hogendoorn P.C.W., Mertens F. WHO Classification of Tumors of Soft Tissue and Bone. 4th Ed. 2013. Рр. 302–304, 281–296.

2. Cottalorda J., Bourelle S. Modern concepts of primary aneurysmal bone cyst. Arch Orthop Trauma Surg 2007; 127 (2): 105–14. DOI: 10.1007/s00402-006-0223-5

Cottalorda J., Bourelle S. Modern concepts of primary aneurysmal bone cyst. Arch Orthop Trauma Surg 2007; 127 (2): 105–14. DOI: 10.1007/s00402-006-0223-5

3. van de Luijtgaarden A.C.M., Veth R.P.H., Slootweg P.J., Wijers-Koster P.M., Schultze Kool L.J., Bovee J.V.M.G., et al. Metastatic potential of an aneurysmal bone cyst. Virchows Arch 2009; 455 (5): 455–9, 2009. DOI: 10.1007/s00428-009-0845-4

van de Luijtgaarden A.C.M., Veth R.P.H., Slootweg P.J., Wijers-Koster P.M., Schultze Kool L.J., Bovee J.V.M.G., et al. Metastatic potential of an aneurysmal bone cyst. Virchows Arch 2009; 455 (5): 455–9, 2009. DOI: 10.1007/s00428-009-0845-4

4. Rădulescu R., Bădilă A., Manolescu R., Sajin M., Japie I. Aneurysmal bone cyst – clinical and morphological aspects. Rom J Morphol Embryol 2014; 55 (3): 977–81.

Rădulescu R., Bădilă A., Manolescu R., Sajin M., Japie I. Aneurysmal bone cyst – clinical and morphological aspects. Rom J Morphol Embryol 2014; 55 (3): 977–81.

5. Hakim D.N., Pelly T., Kulendran M., Caris J.A. Benign tumors of the bone: A review. J Bon Oncol 2015; 4 (2): 37–41. DOI: 10.1016/j.jbo.2015.02.001

Hakim D.N., Pelly T., Kulendran M., Caris J.A. Benign tumors of the bone: A review. J Bon Oncol 2015; 4 (2): 37–41. DOI: 10.1016/j.jbo.2015.02.001

6. Yin J.Q., Fu Y.W., Xie X.B., Cheng X.Y., Yang X.Y., Liu W.H., et al. Telangiectatic osteosarcoma: Outcome analyses and a diagnostic model for differentiation from aneurysmal bone cyst. J Bone Oncol 2017; 11: 10–6. DOI: 10.1016/j.jbo.2017.11.003

Yin J.Q., Fu Y.W., Xie X.B., Cheng X.Y., Yang X.Y., Liu W.H., et al. Telangiectatic osteosarcoma: Outcome analyses and a diagnostic model for differentiation from aneurysmal bone cyst. J Bone Oncol 2017; 11: 10–6. DOI: 10.1016/j.jbo.2017.11.003

7. Vanel D., Tcheng S., Contesso G., Zafrani B., Kalifa C., Dubousset J., et al. The radiological appearances of telangiectatic osteosarcoma. A study of 14 cases. Skelet Radiol 1987; 16 (3): 196–200. DOI: 10.1007/BF00356952

Vanel D., Tcheng S., Contesso G., Zafrani B., Kalifa C., Dubousset J., et al. The radiological appearances of telangiectatic osteosarcoma. A study of 14 cases. Skelet Radiol 1987; 16 (3): 196–200. DOI: 10.1007/BF00356952

8. Gao Z.-H., Yin J.-Q., Liu D.-W., Meng Q.-F., Li J.-P. Preoperative easily misdiagnosed telangiectatic osteosarcoma: clinical-radiologicpathologic correlations. Cancer Imaging 2013; 13 (4): 520– 6. DOI: 10.1102/1470-7330.2013.0042

Gao Z.-H., Yin J.-Q., Liu D.-W., Meng Q.-F., Li J.-P. Preoperative easily misdiagnosed telangiectatic osteosarcoma: clinical-radiologicpathologic correlations. Cancer Imaging 2013; 13 (4): 520– 6. DOI: 10.1102/1470-7330.2013.0042

9. Scully S.P., Temple H.T., O’Keefe R.J., Gebhardt M.C. Case report 830: Aneurysmal bone cyst. Skeletal Radiol 1994; 23 (2): 157–60. DOI: 10.1007/BF00563215

Scully S.P., Temple H.T., O’Keefe R.J., Gebhardt M.C. Case report 830: Aneurysmal bone cyst. Skeletal Radiol 1994; 23 (2): 157–60. DOI: 10.1007/BF00563215

10.

10. Struthers P.J., Shear M. Aneurysmal bone cyst of the jaws. (II). Pathogenesis. Int J Oral Surg 1984; 13 (2): 92–100. DOI: 10.1016/s0300-9785(84)80078-2

Struthers P.J., Shear M. Aneurysmal bone cyst of the jaws. (II). Pathogenesis. Int J Oral Surg 1984; 13 (2): 92–100. DOI: 10.1016/s0300-9785(84)80078-2

11.

11. Szendroi M., Cser I., Konya A., Renyi-Vamos A. Aneurysmal bonecyst. A review of 52 primary and 16 secondary cases. Arch Orthop Trauma Surg 1992; 111 (6): 318– 22. DOI: 10.1007/BF00420058

Szendroi M., Cser I., Konya A., Renyi-Vamos A. Aneurysmal bonecyst. A review of 52 primary and 16 secondary cases. Arch Orthop Trauma Surg 1992; 111 (6): 318– 22. DOI: 10.1007/BF00420058

12.

12. Clough J.R., Price C.H. Aneurysmal bone cysts: Review of twelve cases. J Bone Joint Surg 1968; 50 (1): 116–27.

Clough J.R., Price C.H. Aneurysmal bone cysts: Review of twelve cases. J Bone Joint Surg 1968; 50 (1): 116–27.

13.

13. Saito T., Oda Y., Kawaguchi K., Tanaka K., Matsuda S., Sakamoto A., et al. Five-year evolution of a telangiectatic osteosarcoma initially managed as an aneurysmal bone cyst. Skeletal Radiol 2005; 34 (5): 290–4. DOI: 10.1007/s00256-004-0865-8

Saito T., Oda Y., Kawaguchi K., Tanaka K., Matsuda S., Sakamoto A., et al. Five-year evolution of a telangiectatic osteosarcoma initially managed as an aneurysmal bone cyst. Skeletal Radiol 2005; 34 (5): 290–4. DOI: 10.1007/s00256-004-0865-8

14.

14. Kyriakos M., Hardy D. Malignant transformation of aneurysmal bone cyst, with an analysis of the literature. Cancer 1991; 68 (8): 1770–80. DOI: 10.1002/1097-0142(19911015)68:8<1770::aid-cncr2820680821>3.0.co;2-7

Kyriakos M., Hardy D. Malignant transformation of aneurysmal bone cyst, with an analysis of the literature. Cancer 1991; 68 (8): 1770–80. DOI: 10.1002/1097-0142(19911015)68:8<1770::aid-cncr2820680821>3.0.co;2-7

15.

15. Anract P., de Pinieux G., Jeanrot C., Babinet A., Forest M., Tomeno B. Malignant fibrous histiocytoma at the site of a previously treated aneurysmal bone cyst: a case report. J Bone Joint Surg 2002; 84 (1): 106–11. DOI: 10.2106/00004623-200201000-00016

Anract P., de Pinieux G., Jeanrot C., Babinet A., Forest M., Tomeno B. Malignant fibrous histiocytoma at the site of a previously treated aneurysmal bone cyst: a case report. J Bone Joint Surg 2002; 84 (1): 106–11. DOI: 10.2106/00004623-200201000-00016

16.

16. Hsu C.C., Wang J.W., Huang C.H., Chen W.J. Osteosarcoma at the site of a previously treated aneurysmal bone cyst. A case report. J Bone Joint Surg 2005; 87 (2): 395–8. DOI: 10.2106/JBJS.C.01625

Hsu C.C., Wang J.W., Huang C.H., Chen W.J. Osteosarcoma at the site of a previously treated aneurysmal bone cyst. A case report. J Bone Joint Surg 2005; 87 (2): 395–8. DOI: 10.2106/JBJS.C.01625

17.

17. Wuisman P., Roessner A., Blasius S., Grünert J., Vestering T., Winkelmann W. High malignant surface osteosarcoma arising at the site of a previously treated aneurysmal bone cyst. J Cancer Res Clin Oncol 1993; 119 (7): 375–8. DOI: 10.1007/BF01218416

Wuisman P., Roessner A., Blasius S., Grünert J., Vestering T., Winkelmann W. High malignant surface osteosarcoma arising at the site of a previously treated aneurysmal bone cyst. J Cancer Res Clin Oncol 1993; 119 (7): 375–8. DOI: 10.1007/BF01218416

18.

18. Schreuder H.W., Veth R.P., Pruszczynski M., Lemmens J.A., Koops H.S., Molenaar W.M. Aneurysmal bone cysts are treated with curettage, cryotherapy and bone grafting. J Bone Joint Surg Br 1997; 79 (1): 20–5. DOI: 10.1302/0301-620x.79b1.7097

Schreuder H.W., Veth R.P., Pruszczynski M., Lemmens J.A., Koops H.S., Molenaar W.M. Aneurysmal bone cysts are treated with curettage, cryotherapy and bone grafting. J Bone Joint Surg Br 1997; 79 (1): 20–5. DOI: 10.1302/0301-620x.79b1.7097

19.

19. Atlas of Genetics and Cytogenetics in Oncology and Haematology: Aneurysmal bone cysts. Jean-Loup Huret, Genetics, Dept Medical Information, University Hospital, France. [Электронный ресурс]. URL: http://atlasgeneticsoncology.org/Tumors/AneurBoneCystID5133.html (Дата обращения 18.11.2020).

Atlas of Genetics and Cytogenetics in Oncology and Haematology: Aneurysmal bone cysts. Jean-Loup Huret, Genetics, Dept Medical Information, University Hospital, France. [Электронный ресурс]. URL: http://atlasgeneticsoncology.org/Tumors/AneurBoneCystID5133.html (Дата обращения 18.11.2020).

20.

20. Park H.Y., Young S.K., Sheppard V.L., Hegde V., Zoller S.D., Nelson S.D., et al. Ongoing treatment of aneurysmal bone cysts. Curr Rev Musculoskelet Med 2016; 9 (4): 435–44. DOI: 10.1007/s12178-016-9371-6

Park H.Y., Young S.K., Sheppard V.L., Hegde V., Zoller S.D., Nelson S.D., et al. Ongoing treatment of aneurysmal bone cysts. Curr Rev Musculoskelet Med 2016; 9 (4): 435–44. DOI: 10.1007/s12178-016-9371-6

21.

21. Lau A.W., Pringle L.M., Quick L., Riquelme D.N., Ye Y., Oliveira A.M,. Chou M.M. TRE17 /ubiquitin-specific protease 6 (USP6), an oncogene translocated in an aneurysmal bone cyst, blocks osteoblastic maturation through an autocrine mechanism involving bone marrow morphogenetic protein dysregulation. J Biol Chem 2010; 285 (47): 37111–20.

Lau A.W., Pringle L.M., Quick L., Riquelme D.N., Ye Y., Oliveira A.M,. Chou M.M. TRE17 /ubiquitin-specific protease 6 (USP6), an oncogene translocated in an aneurysmal bone cyst, blocks osteoblastic maturation through an autocrine mechanism involving bone marrow morphogenetic protein dysregulation. J Biol Chem 2010; 285 (47): 37111–20.

22.

22. Zishan U.S., Pressney I., Khoo M., Saifuddin A. The differentiation between aneurysmal bone cyst and telangiectatic osteosarcoma: A clinical, radiographic and MRI study. Skeletal Radiol 2020; 49 (9): 1375– 86. DOI: 10.1007/s00256-020-03432-w

Zishan U.S., Pressney I., Khoo M., Saifuddin A. The differentiation between aneurysmal bone cyst and telangiectatic osteosarcoma: A clinical, radiographic and MRI study. Skeletal Radiol 2020; 49 (9): 1375– 86. DOI: 10.1007/s00256-020-03432-w

23.

23. Deyrup A., Siegal G. Practical Orthopedic Pathology: A Diagnostic Approach. Chapter 13, 15. Philadelphia, PA: Elsevier; 2016.

Deyrup A., Siegal G. Practical Orthopedic Pathology: A Diagnostic Approach. Chapter 13, 15. Philadelphia, PA: Elsevier; 2016.

24.

24. Bullough P. Orthopaedic pathology. Maryland: Mosby, Elsevier; 2010. Рр. 453–456.

Bullough P. Orthopaedic pathology. Maryland: Mosby, Elsevier; 2010. Рр. 453–456.

25.

25. Folpe А., Inwards C., eds. Bone and soft tissue pathology. Philadelphia: Saunders, Elsevier; 2010. Рр. 428–432.

Folpe А., Inwards C., eds. Bone and soft tissue pathology. Philadelphia: Saunders, Elsevier; 2010. Рр. 428–432.

26.

26. Rossi G., Rimondi E., Bartalena T., Gerardi A., Alberghini M., Staals E.L., et al. Selective arterial embolization of 36 aneurysmal bone cysts of the skeleton with N-2-butylcyanoacrylate. Skeletal Radiol 2010; 39 (2): 161–7. DOI: 10.1007/s00256-009-0757-z

Rossi G., Rimondi E., Bartalena T., Gerardi A., Alberghini M., Staals E.L., et al. Selective arterial embolization of 36 aneurysmal bone cysts of the skeleton with N-2-butylcyanoacrylate. Skeletal Radiol 2010; 39 (2): 161–7. DOI: 10.1007/s00256-009-0757-z

27.

27. Papagelopoulos P.J., Currier B.L., Shaughnessy W.J., Sim F.H., Ebsersold M.J., Bond J.R., Unni K.K. Aneurysmal bone cyst of the spine. Management and results. Spine (Phila Pa 1976) 1998; 23 (5): 621–8. DOI: 10.1097/00007632-199803010-00018

Papagelopoulos P.J., Currier B.L., Shaughnessy W.J., Sim F.H., Ebsersold M.J., Bond J.R., Unni K.K. Aneurysmal bone cyst of the spine. Management and results. Spine (Phila Pa 1976) 1998; 23 (5): 621–8. DOI: 10.1097/00007632-199803010-00018

28.

28. Dürr H.R., Grahneis F., Baur-Melnyk A., Knösel T., Birkenmaier C., Jansson V., Klein A. Aneurysmal bone cyst: results of an off label treatment with Denosumab. BMC Musculoskeletal Disorders 2019; 20 (1): 456. DOI: 10.1186/s12891-019-2855-y

Dürr H.R., Grahneis F., Baur-Melnyk A., Knösel T., Birkenmaier C., Jansson V., Klein A. Aneurysmal bone cyst: results of an off label treatment with Denosumab. BMC Musculoskeletal Disorders 2019; 20 (1): 456. DOI: 10.1186/s12891-019-2855-y