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Massive osteolysis of the mandible: a case report

2003; Elsevier BV; Volume: 61; Issue: 8 Linguagem: Inglês

10.1016/s0278-2391(03)00235-0

1531-5053

Mohammad Hosein Kalantar Motamedi, Seyyed Mohammad Homauni, Hossein Behnia,

IgG4-Related and Inflammatory Diseases

Massive osteolysis (MO), also known as Gorham’s disease, 1 Gorham L. Stout A. Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone) Its relation to hemangiomatosis. J Bone Joint Surg Am. 1955; 37: 985 PubMed Google Scholar vanishing bone disease, disappearing bone disease; progressive osteolysis, and phantom bone disease, is an extremely rare disease characterized by spontaneous and usually progressive destruction of one or more bones. 1 Gorham L. Stout A. Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone) Its relation to hemangiomatosis. J Bone Joint Surg Am. 1955; 37: 985 PubMed Google Scholar , 2 Choma N. Biscotti C. Bauer P. et al. Gorham’s syndrome A case report and review of the literature. Am J Med. 1981; 83: 1151 Abstract Full Text PDF Scopus (129) Google Scholar , 3 Waldron C.A. Bone pathology. in: Neville B.W. Damm D.D. Allen C.M. Oral and Maxillofacial Pathology. Saunders, Philadelphia, PA1995: 448-449 Google Scholar , 4 Frederiksen N.L. Massive osteolysis of the maxillofacial skeleton A clinical, radiographic, histologic and ultrastructural study. Oral Surg Oral Med Oral Pathol. 1983; 55: 470 Abstract Full Text PDF PubMed Scopus (37) Google Scholar , 5 Heffez L. Perspective of massive osteolysis Report of a case and review of the literature. Oral Surg Oral Med Oral Pathol. 1983; 55: 331 Abstract Full Text PDF PubMed Scopus (166) Google Scholar , 6 Holroyd I. Dillon M. Roberts G.J. et al. Gorham’s disease A case (including dental presentation) of vanishing bone disease. Oral Surg Oral Med Oral Pathol. 2000; 89: 125 Abstract Full Text Full Text PDF Scopus (39) Google Scholar The bone destroyed by osteolysis does not regenerate or repair and instead is replaced by dense fibrous tissue with the ultimate disappearance of the bone. The cause of MO remains obscure, and no evidence of underlying metabolic or endocrine disorders has yet been found. However, increased osteoclast activity and elevated levels of interleukin-6 (IL-6) have been reported. 7 Jackson J.B.S. A boneless arm. Boston Med Surg J. 1838; 10: 368 Google Scholar The majority of reported cases have occurred subsequent to trauma; thus many authors 1 Gorham L. Stout A. Massive osteolysis (acute spontaneous absorption of bone, phantom bone, disappearing bone) Its relation to hemangiomatosis. J Bone Joint Surg Am. 1955; 37: 985 PubMed Google Scholar , 2 Choma N. Biscotti C. Bauer P. et al. Gorham’s syndrome A case report and review of the literature. Am J Med. 1981; 83: 1151 Abstract Full Text PDF Scopus (129) Google Scholar , 3 Waldron C.A. Bone pathology. in: Neville B.W. Damm D.D. Allen C.M. Oral and Maxillofacial Pathology. Saunders, Philadelphia, PA1995: 448-449 Google Scholar , 4 Frederiksen N.L. Massive osteolysis of the maxillofacial skeleton A clinical, radiographic, histologic and ultrastructural study. Oral Surg Oral Med Oral Pathol. 1983; 55: 470 Abstract Full Text PDF PubMed Scopus (37) Google Scholar , 5 Heffez L. Perspective of massive osteolysis Report of a case and review of the literature. Oral Surg Oral Med Oral Pathol. 1983; 55: 331 Abstract Full Text PDF PubMed Scopus (166) Google Scholar , 6 Holroyd I. Dillon M. Roberts G.J. et al. Gorham’s disease A case (including dental presentation) of vanishing bone disease. Oral Surg Oral Med Oral Pathol. 2000; 89: 125 Abstract Full Text Full Text PDF Scopus (39) Google Scholar have implicated this as an initiating factor. The involvement of almost all bones and combinations of bones has been reported. Fewer than 140 cases have been reported since 1838. 6 Holroyd I. Dillon M. Roberts G.J. et al. Gorham’s disease A case (including dental presentation) of vanishing bone disease. Oral Surg Oral Med Oral Pathol. 2000; 89: 125 Abstract Full Text Full Text PDF Scopus (39) Google Scholar In the maxillofacial bones, the mandible in particular is affected and may be involved simultaneously with the maxilla. 3 Waldron C.A. Bone pathology. in: Neville B.W. Damm D.D. Allen C.M. Oral and Maxillofacial Pathology. Saunders, Philadelphia, PA1995: 448-449 Google Scholar , 4 Frederiksen N.L. Massive osteolysis of the maxillofacial skeleton A clinical, radiographic, histologic and ultrastructural study. Oral Surg Oral Med Oral Pathol. 1983; 55: 470 Abstract Full Text PDF PubMed Scopus (37) Google Scholar , 5 Heffez L. Perspective of massive osteolysis Report of a case and review of the literature. Oral Surg Oral Med Oral Pathol. 1983; 55: 331 Abstract Full Text PDF PubMed Scopus (166) Google Scholar , 6 Holroyd I. Dillon M. Roberts G.J. et al. Gorham’s disease A case (including dental presentation) of vanishing bone disease. Oral Surg Oral Med Oral Pathol. 2000; 89: 125 Abstract Full Text Full Text PDF Scopus (39) Google Scholar Only 21 mandibular cases have been reported in the literature up to 2002 6 Holroyd I. Dillon M. Roberts G.J. et al. Gorham’s disease A case (including dental presentation) of vanishing bone disease. Oral Surg Oral Med Oral Pathol. 2000; 89: 125 Abstract Full Text Full Text PDF Scopus (39) Google Scholar ; our report adds to this list. The prevention of progression is a main concern in MO because progression from the mandible to the maxilla, skull, and spine has been reported. 7 Jackson J.B.S. A boneless arm. Boston Med Surg J. 1838; 10: 368 Google Scholar In our case, progression stopped only after surgical intervention. In addition to halting progression of the disease, the technique preserved remaining bone both distal and proximal to the resection site as well as the alveolar dental nerve.