Article Data

  • Views 863
  • Dowloads 148

Case Reports

Open Access

Vanishing Mandible in a 7-year Old Child: Response to Radiation Therapy

  • Mounabati Mohapatra1
  • Ashok Kumar Jena1,*,
  • Arun Kumar Dandapat1
  • Sombir Singh2

1Department of Dental Surgery, All India Institute of Medical Sciences, Sijua, Dumduma, Bhubaneswar Odisha, India

2Institute of Medical Education and ResearchSector-12, Chandigarh, UT India.

DOI: 10.17796/1053-4628-41.6.10 Vol.41,Issue 6,November 2017 pp.472-477

Published: 01 November 2017

*Corresponding Author(s): Ashok Kumar Jena E-mail: ashokkjena@yahoo.co.in

Abstract

Massive osteolysis in a 7-year old child is a rare condition. The etiology of massive osteolysis is unknown and it results in the progressive destruction of bony structures. There is no standard therapy available in the literature. Conservative treatment is often used for its management. Radiotherapy is considered as an accepted form of treatment with greater chance of success when it is used in the early course of disease. There are few case reports in the literature in which radiotherapy has been used for the treatment. This article highlights the literature update on various treatment modalities and a case managed by radiation therapy.

Keywords

Vanishing bone disease, Gorham’s disease, mandible, radiation therapy.

Cite and Share

Mounabati Mohapatra,Ashok Kumar Jena,Arun Kumar Dandapat,Sombir Singh. Vanishing Mandible in a 7-year Old Child: Response to Radiation Therapy. Journal of Clinical Pediatric Dentistry. 2017. 41(6);472-477.

References

1. Aviv RI, McHugh K, Hunt J. Angiomatosis of bone and soft tissue: A spectrum of disease from diffuse lymphangiomatosis to vanishing bone disease in young patients. Clin Radiol 56: 184-90, 2001.

2. Hardegger F, Simpson LA, Segmueller G. The syndrome of idiopathic osteolysis: Classification, review, and case report. J Bone Joint Surg Br 67: 89-93, 1985.

3. Yoo SY, Hong SH, Chung HW, Choi JA, Kim CJ, Kang HS. MRI of Gorham’s disease: findings in two cases. Skeletal Radiol 31: 301-6, 2002.

4. Fisher KL, Pogrel MA. Gorham’s syndrome: a case report. J Oral Maxillofac Surg 48: 1222–5, 1990.

5. Tilling G, Skobowytsh B. Disappearing bone disease, Morbus Gorham: report of a case. Acta Orthop Scand 39: 398-406, 1968.

6. Jackson JBS. A boneless arm. Boston Med Surg J 18: 368–9, 1838.

7. Jackson JBS. Absorption of humerus after fracture. Boston Med Surg J 10: 245–7, 1872.

8. Gorham LW, Stout AP. Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone): its relation to hemangiomatosis. J Bone Joint Surg 37-A: 985-1004, 1955.

9. Gowin W, Rahmanzadeh R. Radiologic diagnosis of massive idiopathic osteolysis (Gorham-Stout Syndrome). Rontgenpraxis 38: 128-34, 1985.

10. Horst M, Zsernaviczky J, Delling G. A rare case of so-called idiopathic osteolysis associated with a lymphangioma of the fibula. Z Orthop 117: 88-95, 1979.

11. Florchinger A, Bottger E, Claass-Bottger F, Georgi M, Harms J. Gorham Stout syndrome of the spine: case report and review of the literature. Rofo 168: 68-76, 1998.

12. Romer O. Die pathologic der Zahne. In: von Henke F, Lubarsch O (eds). Handbuch der speziellen pathologischen anatomie und histologie. Vol 4. Berlin: Springer Verlag, 1924, pp 135–499.

13. Frederikson NL, Wesley RK, Sciubba JJ, Helfrick J. Massive osteolysis of the maxillofacial skeleton: a clinical, radiographic, histologic and ultrastructural study. Oral Surg Oral Med Oral Pathol 55: 470-80, 1983.

14. Escande C, Schouman T, Franc¸oise G, Haroche J, Ménard P, Piette JC, et al. Histological features and management of a mandibular Gorham disease: a case report and review of maxillofacial cases in the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 106: e30-7, 2008.

15. Patel DV. Gorham’s disease or Massive osteolysis. Clin Med Res 3: 65-74, 2005.

16. Ricalde P, Ord RA, Sun CC. Vanishing bone disease in a five year old: report of a case and review of the literature. Int J Oral Maxillofac Surg 32: 222-62, 2003.

17. Moizan H, Talbi M, Devauchelle B. Massive mandibular osteolysis: a case with non-contributive histology. J Oral Maxillofac Surg 65: 772-6, 2007.

18. Anavi Y, Sabes WR, Mintz S. Gorham’s disease affecting the maxillofacial skeleton. Head Neck 11: 550-7, 1989.

19. Benhalima H, Lazrak A, Boulaich M, Mezahi M, Ourlai A, Kzadri M, et al. Massive osteolysis of the maxillo-facial bones: case report and review of the literature. Odontostomatol Trop 24: 35-40, 2001.

20. Raghuveer HP, Jayaleshmy R. Gorham’s massive osteolysis of the mandible–a progressive radiographic presentation. Dentomaxillofac Radiol 38: 292-5, 2009.

21. Klein M, Metelmann, HR, Gross U. Massive osteolysis (Gorham-Stout syndrome) in the maxillofacial region: an unusual manifestation. Int J Oral Maxillofac Surg 25: 376-8, 1996.

22. Ohya T, Shibata S, Takeda Y. Massive osteolysis of the maxillofacial bones: report of two cases. Oral Surg Oral Med Oral Pathol 70: 698-703, 1990.

23. Schiel H, Prien J. Seven-year follow-up of vanishing bone disease in a 14-year-old girl. Head Neck 15: 352-6, 1993.

24. Rubel IF, Carrer A, Gilles JJ, Howard R, Cohen G. Progressive Gorham disease of the forearm. Orthopedics 31: 284, 2008.

25. Poirier H. Massive osteolysis of the humerus treated by resection and prosthetic replacement. J Bone Joint Surg 50: 158–60, 1968.

26. Dunbar SF, Rosenberg A, Mankin H, Rosenthal D, Suit HD. Gorham’s massive osteolysis: the role of radiation therapy and a review of the literature. Int J Radiat Oncol Biol Phys 26: 491–7, 1993.

27. Fontanesi J. Radiation therapy in the treatment of Gorham disease. J Pediat Hematol Oncol 25: 816–7, 2003.

28. Avelar RL, Martins VB, Antunes AA, de Oliveira Neto PJ, de Souza Andrade ES. Use of zoledronic acid in the treatment of Gorham’s disease. Int J Pediat Otorhinolaryngol 74: 319–22, 2010.

29. Hagberg H, Lamberg K, Astrom G. α-2b interferon and oral clodronate for Gorham’s disease. Lancet 350: 1822–3, 1997.

30. Devlin RD, Bone HG, Roodman GD. Interleukin-6: a potential mediator of the massive osteolysis in patients with Gorham-Stout disease. J Clin Endocrinol Metabol 81:1893–7, 1996.

31. Hammer F, Kenn W, Wesselmann U, Hofbauer LC, Delling G, Allolio B, et al. Gorham-stout disease—stabilization during bisphosphonate treatment. J Bone Miner Res 20: 350-3, 2005.

32. Papadakis BSA, Khaldi L, Babourda EC, Papadakis S, Mitsitsikas T, Sapkas G. Vanishing bone disease: review and case reports. Orthopedics 31: 278, 2008.

33. Tsang WM, Tong AC, Chow LT, Ng IO. Massive osteolysis (Gorham disease) of the maxillofacial skeleton: report of 2 cases. J Oral Maxillofac Surg 62: 225-30, 2004.

34. Bruch-Gerharz D, Gerharz CD, Stege H, Krutmann J, Pohl M, Koester R et al. Cutaneous lymphatic malformations in disappearing bone (gorhamstout) disease: A novel clue to the pathogenesis of a rare syndrome. J Am Acad Dermatol 56: S21-5, 2007.

35. Radhakrishnan K, Rockson SG. Gorham’s disease: An osseous disease of lymphangiogenesis? Ann NY Acad Sci 1131: 203-5, 2008.

36. Skidmore TB, Lee CM, Abbott TM, Wiggins RH, Anderson GE, Tward JD et al. Gorham disease of the mandible: Radiographic findings and radiotherapy response. Ear Nose Throat J 87: E4-7, 2008.

37. Gondivkar SM, Gadbail AR. Gorham-Stout syndrome: a rare clinical entity and review of literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 109: e41-e48, 2010.

38. Hanly JG, Walsh NM, Bresnihan B. Massive osteolysis in the hand and response to radiotherapy. J Rheumatol 12: 580-2, 1985.

39. Campbell J, Almond HG, Johnson R. Massive osteolysis of the humerous with spontaneous recovery. J Bone Joint Surg Am 57: 238-40, 1975.

40. Bek V, Halcl Z, Kolar J, Bednar B. Radiotherapy of the Gorham-Stout syndrome. Cesk Radiol 35: 291-8, 1981.

41. Prasanna R, Sankar J, Ramachandran P. Gorham’s disease: Vanishing bone syndrome. Indian Pediatr 46: 255-6, 2009.

42. Kulenkampff HA, Richter GM, Hasse WE, Adler CP. Massive pelvic osteolysis in the Gorham-Stout syndrome. Int Orthop 14: 361-6, 1990.

43. Turra S, Gigante C, Scapinelli R. A 20-year follow-up study of a case of surgically treated massive osteolysis. Clin Orthop Relat Res 250: 297-302, 1990.

44. Woodward HR, Chan DP, Lee J. Massive osteolysis of the cervical spine: a case report of bone graft failure. Spine 6: 545-9, 1981.

45. Cannon SR. Massive osteolysis: a review of seven cases. J Bone Joint Surg Br 68: 24-8, 1998.

46. Sharma A, Iyer N, Mittal A, Das D, Sharma S. Vanishing mandible. J Oral Science 52: 513-6, 2010.

47. Resnick D. Osteolysis and chondrolysis. In: Resnick D, editor. Diagnosis of bone and joint disorders. 4th ed. Philadelphia: WB Saunders; 2002. p. 4920-44.

48. Mendez AA, Keret D, Robertson W, MacEwen GD. Massive osteolysis of the femur (Gorham’s disease): a case report and review of the literature. J Pediat Orthop 9: 604–8, 1989.

49. Pazzaglia UE, Andrini L, Bonato M, Leutner M. Pathology of disappearing bone disease: A case report with immunohistochemical study. Int Orthop 21: 303-7, 1997.

50. Hirayama T, Sabokbar A, Itonaga I, Watt-Smith S, Athanasou NA. Cellular and humoral mechanisms of osteoclast formation and bone resorption in Gorham-Stout disease. J Pathol 195: 624-30, 2001.

51. Tong ACK, Leung TM, Cheung PT. Management of massive osteolysis of the mandible: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 109: 238-41, 2009.

52. Assoun J, Richardi G, Railhac JJ, Le Guennec P, Caulier M, Dromer C, et al. CT and MRI of massive osteolysis of Gorham. J Comput Assist Tomogr 18: 981–4, 1994.

53. Speith ME, Greenspan A, Forrester DM, Ansari AN, Kimura RL, Gleason-Jordan I. Gorham’s disease of the radius: radiographic, scintigraphic and MRI findings with pathologic correlation. A case report and review of literature. Skeletal Radiol 26: 659–63, 1997.

54. Heffez L, Doku HC, Carter BL, Feeney JE. Perspectives on massive osteolysis: report of a case and review of literature. Oral Surg Oral Med Oral Pathol 55: 331-43, 1983.

55. Kiran DN, Anupama A. Vanishing Bone Disease: A Review. J Oral Maxillofac Surg 69: 199-203, 2011.

56. Landesberg R, Eisig S, Fennoy I, Siris E. Alternative indications of bisphosphonate therapy. J Oral Maxillofac Surg 67(Suppl 1): 27-34, 2009.

57. Migliorati CA. Commentary: massive osteolysis or Gorham-Stout disease. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 109: 242-3, 2010.

58. Abdulai AE, Nuamah IK, Avogo D, Konadu Akua B. Vanishing Bone (Gorham’s) Disease of the Mandible: A Case Report. Sch J Med Case Rep 2: 183-7, 2014.

59. Kontio R. Update on mandibular reconstruction: computer-aided design, imaging, stem cells and future applications. Curr Opin Otolaryngol Head Neck Surg 22: 307-15, 2014.

60. D’Aquino R, De Rosa A, Lanza V, Tirino V, Laino L, Graziano A, et al. Human mandible bone defect repair by the grafting of dental pulp stem/ progenitor cells and collagen sponge biocomplexes. Eur Cell Mater 18: 75–83, 2009.

61. Xu H, Han D, Dong JS, Shen GX, Chai G, Yu ZY, et al. Rapid prototyped PGA/PLA scaffolds in the reconstruction of mandibular condyle bone defects. Int J Med Robot 6: 66–72, 2010.

62. Yamada Y, Ito K, Nakamura S, Ueda M, Nagasaka T. Promising cellbased therapy for bone regeneration using stem cells from deciduous teeth, dental pulp, and bone marrow. Cell Transplant 20: 1003–13, 2011.

63. Warnke PH, Springer IN, Acil Y, Julga G, Wiltfang J, Ludwig K, et al. The mechanical integrity of in vivo engineered heterotopic bone. Biomaterials 27: 1081–7, 2006.

64. Abukawa H, Zhang W, Young CS, Asrican R, Vacanti JP, Kaban LB, et al. Reconstructing mandibular defects using autologous tissue-engineered tooth and bone constructs. J Oral Maxillofac Surg 67: 335–47, 2009.

65. Grayson WL, Fro¨ hlich M, Yeager K, Bhumiratana S, Chan ME, Cnnizzaro C, et al. Engineering anatomically shaped human bone grafts. Proc Natl Acad Sci USA 107: 3299–304, 2010.


Abstracted / indexed in

Science Citation Index Expanded (SciSearch) Created as SCI in 1964, Science Citation Index Expanded now indexes over 9,500 of the world’s most impactful journals across 178 scientific disciplines. More than 53 million records and 1.18 billion cited references date back from 1900 to present.

Biological Abstracts Easily discover critical journal coverage of the life sciences with Biological Abstracts, produced by the Web of Science Group, with topics ranging from botany to microbiology to pharmacology. Including BIOSIS indexing and MeSH terms, specialized indexing in Biological Abstracts helps you to discover more accurate, context-sensitive results.

Google Scholar Google Scholar is a freely accessible web search engine that indexes the full text or metadata of scholarly literature across an array of publishing formats and disciplines.

JournalSeek Genamics JournalSeek is the largest completely categorized database of freely available journal information available on the internet. The database presently contains 39226 titles. Journal information includes the description (aims and scope), journal abbreviation, journal homepage link, subject category and ISSN.

Current Contents - Clinical Medicine Current Contents - Clinical Medicine provides easy access to complete tables of contents, abstracts, bibliographic information and all other significant items in recently published issues from over 1,000 leading journals in clinical medicine.

BIOSIS Previews BIOSIS Previews is an English-language, bibliographic database service, with abstracts and citation indexing. It is part of Clarivate Analytics Web of Science suite. BIOSIS Previews indexes data from 1926 to the present.

Journal Citation Reports/Science Edition Journal Citation Reports/Science Edition aims to evaluate a journal’s value from multiple perspectives including the journal impact factor, descriptive data about a journal’s open access content as well as contributing authors, and provide readers a transparent and publisher-neutral data & statistics information about the journal.

Scopus: CiteScore 1.8 (2023) Scopus is Elsevier's abstract and citation database launched in 2004. Scopus covers nearly 36,377 titles (22,794 active titles and 13,583 Inactive titles) from approximately 11,678 publishers, of which 34,346 are peer-reviewed journals in top-level subject fields: life sciences, social sciences, physical sciences and health sciences.

Submission Turnaround Time

Conferences

Top