BACKGROUNDOsteofbrous dysplasia （OFD） is a developmental ske-letal disorder, and cases with a giant affected area in the pelvis are rare.CASE SUMMARYIn this case report, a 48-year-old man presented with a large tumo...BACKGROUNDOsteofbrous dysplasia （OFD） is a developmental ske-letal disorder, and cases with a giant affected area in the pelvis are rare.CASE SUMMARYIn this case report, a 48-year-old man presented with a large tumor in the right iliac region that turned out to be OFD. The patient had rebound tenderness in his right hip. After radiography examination, magnetic resonance imaging examinations and some physical examination, extensive bone destruction in the right ilium was confirmed. Moreover, changes in bone mi-neral density and peripheral cortical bone sclerosis with surrounding soft tissue swelling were observed. Thus, this patient was considered to have giant monostoticOFD of the ilium. The tumor-related area was removed completely by surgery, and the remaining cavity was filled by artificial bones from the opposite ilium. According to the results of follow-up, the patient had normal fexion and extension activities of the right hip joint, and there was no evidence of recurrence of the tumor.CONCLUSION Suture of iliopsoas and gluteus medius muscle following focus curettage and bone grafting is a promising and effective method to treat giant OFD of the ilium. It is a feasible way to fll a large cavity after removing a lesion like the one is this case.展开更多
Background: It is generally accepted that there is a spectrum ranging from Osteofibrous Dysplasia (OFD) to Osteofibrous dysplasia-like Adamantinoma (OFD-AD) to “classic” Adamantinoma (AD). However, it is controversi...Background: It is generally accepted that there is a spectrum ranging from Osteofibrous Dysplasia (OFD) to Osteofibrous dysplasia-like Adamantinoma (OFD-AD) to “classic” Adamantinoma (AD). However, it is controversial whether OFD may progress into OFD-AD and AD or these pathologic changes reflect sampling issues. We herein report the fifth case of late development of AD from OFD-AD to underline the importance of long-term follow-up. Case: At 6 years bone biopsy of the tibia revealed the diagnosis of OFD-AD, confirmed by repeat biopsies until the age of 18 years. At age 45 years a progressive focal osteolysis was biopsied and showed histologically a classic AD. The affected tibial diaphysis was resected and pasteurized. Reconstruction was performed adapting the “Capanna technique” of inserting the ipsilateral mobilized fibula into the replanted autologous, pasteurized tibial segment as biologic stabilizer. At 1.5 years follow-up, the reconstruction has healed allowing full weight bearing.展开更多
Purposes: We would like to inform the characteristics of recurred osteo-fibrous dysplasia (OFD), and also the possible use of vascularized composite autograft and allografts (VCA) combined with live fibular graft (LFG...Purposes: We would like to inform the characteristics of recurred osteo-fibrous dysplasia (OFD), and also the possible use of vascularized composite autograft and allografts (VCA) combined with live fibular graft (LFG) for the treatment of aggressive benign bone tumor and osteogenic sarcomas in children. Materials and Methods: We reviewed one boy with recurred OFD after LFG, and other four children with osteogenic sarcoma in long bone which was treated with LFG and VCA, and followed them for average 9 years (3 - 14 years). Survival of the LFG and VCA was estimated by the ISOLS Functional Radiologic Scoring System, but not with Kaplans-Meier’s scoring system because of a small series of case reports. Results: All succeed after surgeries initially, but the cause of recurrence of OFD was still unclear, and one girl with osteogenic sarcoma on distal femur died with skip and lung metastasis, 4 years after surgeries. Conclusions: The causes of recurred OFD are not found thru pathologic studies of our patient, but we believe the multifocal origin of tumor cells even on the adjacent soft tissues in OFD is one of the causes. The excised long bone tumor is recycled by pasteurization or autoclaving, or allograft, then LFG, and neo-adjuvant chemotherapy would be one of elective surgery for the treatment of malignant long bone tumors in children. The LFG into recycled autograft or allograft bone is difficult to perform simultaneously, but very effective to increase more earlier regenerative vascularities and also the stabilities of the dead bones in children.展开更多
文摘BACKGROUNDOsteofbrous dysplasia （OFD） is a developmental ske-letal disorder, and cases with a giant affected area in the pelvis are rare.CASE SUMMARYIn this case report, a 48-year-old man presented with a large tumor in the right iliac region that turned out to be OFD. The patient had rebound tenderness in his right hip. After radiography examination, magnetic resonance imaging examinations and some physical examination, extensive bone destruction in the right ilium was confirmed. Moreover, changes in bone mi-neral density and peripheral cortical bone sclerosis with surrounding soft tissue swelling were observed. Thus, this patient was considered to have giant monostoticOFD of the ilium. The tumor-related area was removed completely by surgery, and the remaining cavity was filled by artificial bones from the opposite ilium. According to the results of follow-up, the patient had normal fexion and extension activities of the right hip joint, and there was no evidence of recurrence of the tumor.CONCLUSION Suture of iliopsoas and gluteus medius muscle following focus curettage and bone grafting is a promising and effective method to treat giant OFD of the ilium. It is a feasible way to fll a large cavity after removing a lesion like the one is this case.
文摘Background: It is generally accepted that there is a spectrum ranging from Osteofibrous Dysplasia (OFD) to Osteofibrous dysplasia-like Adamantinoma (OFD-AD) to “classic” Adamantinoma (AD). However, it is controversial whether OFD may progress into OFD-AD and AD or these pathologic changes reflect sampling issues. We herein report the fifth case of late development of AD from OFD-AD to underline the importance of long-term follow-up. Case: At 6 years bone biopsy of the tibia revealed the diagnosis of OFD-AD, confirmed by repeat biopsies until the age of 18 years. At age 45 years a progressive focal osteolysis was biopsied and showed histologically a classic AD. The affected tibial diaphysis was resected and pasteurized. Reconstruction was performed adapting the “Capanna technique” of inserting the ipsilateral mobilized fibula into the replanted autologous, pasteurized tibial segment as biologic stabilizer. At 1.5 years follow-up, the reconstruction has healed allowing full weight bearing.
文摘Purposes: We would like to inform the characteristics of recurred osteo-fibrous dysplasia (OFD), and also the possible use of vascularized composite autograft and allografts (VCA) combined with live fibular graft (LFG) for the treatment of aggressive benign bone tumor and osteogenic sarcomas in children. Materials and Methods: We reviewed one boy with recurred OFD after LFG, and other four children with osteogenic sarcoma in long bone which was treated with LFG and VCA, and followed them for average 9 years (3 - 14 years). Survival of the LFG and VCA was estimated by the ISOLS Functional Radiologic Scoring System, but not with Kaplans-Meier’s scoring system because of a small series of case reports. Results: All succeed after surgeries initially, but the cause of recurrence of OFD was still unclear, and one girl with osteogenic sarcoma on distal femur died with skip and lung metastasis, 4 years after surgeries. Conclusions: The causes of recurred OFD are not found thru pathologic studies of our patient, but we believe the multifocal origin of tumor cells even on the adjacent soft tissues in OFD is one of the causes. The excised long bone tumor is recycled by pasteurization or autoclaving, or allograft, then LFG, and neo-adjuvant chemotherapy would be one of elective surgery for the treatment of malignant long bone tumors in children. The LFG into recycled autograft or allograft bone is difficult to perform simultaneously, but very effective to increase more earlier regenerative vascularities and also the stabilities of the dead bones in children.
