osteofibrous dysplasia is a rare benign self - limiting fibro - osseous lesion most commonly seen in the diaphysis of the tibia .
there are no case reports mentioning the use of synthetic bone graft to fill the defect following extraperiosteal excision .
a 13 year old girl presented with pain and swelling of the ( r ) leg since 2 months following a trivial injury at school .
examination revealed a 53 cm tender swelling on the anteromedial aspect of the middle third tibia .
radiographs and mri , revealed an eccentric expansile lytic lesion , which was multilocular and was present at the junction of the metaphysis and diaphysis on the antero -medial aspect of tibia .
we did an extraperiosteal excision of the lesion . to fill the cavity we harvested 10 cm of the contralateral fibula and since there was still space in the cavity , we packed bone graft substitute ( hydroxyapatite crystals ) into the defect .
the use of synthetic graft is an option in these patients as it reduces morbidity ; and in our case we had good graft incorporation with this method .
extraperiosteal excision of osteofibrous dysplasia combined with autologous free fibular graft and bone graft substitute is a good surgical option to prevent recurrence and mange bone defects in this rare lesion .
osteofibrous dysplasia is a rare benign self - limiting fibro osseous lesion most commonly seen in the diaphysis of the tibia .
the etiology of osteofibrous dysplasia , as well as the cell of origin , is unknown .
due to high incidence ( 60 - 100% ) of recurrence , any surgical procedure is recommended only after skeletal maturity .
a 13 year old girl presented with pain and swelling of the ( r ) leg since 2 months following a trivial injury at school .
pain was aggravated by running and standing for a long duration and was relieved with rest and medication .
examination revealed a 53 cm swelling on the anteromedial aspect of the middle third tibia .
radiographs - revealed an eccentric expansile lytic lesion , which was multilocular and was present at the junction of the metaphysis and diaphysis on the antero -medial aspect of tibia .
the cortex had ballooned out and there was a possibility of an impending fracture ( fig1 ) .
blood investigations reported hemogobin to be 9.3 g% and alkaline phosphatase to be 453u / l , serum calcium was 8.0 mg / dl and phosphorous was 5.2mg / dl .
radiographs of tibia revealed an eccentric expansile lytic lesion , which was multilocular and was present at the junction of the metaphysis and diaphysis on the antero -medial aspect of tibia first a biopsy from the lesion was done and greyish white bits of tissue were sent for histopathology .
biopsy reported bony trabeculae interspersed with fibrous tissue , some bony trabeculae showed curvilinear arrangement with osteoblastic rimming confirming the diagnosis of osteofibrous dysplasia .
after about a month , since the patient still had pain , we planned to do a extraperiosteal excision of the lesion with autologous fibular graft ( fig 2a ) . through an anteromedial approach we exposed the lesion
, we did an extraperiosteal excision of the lesion ( fig 2b ) . to fill the cavity we harvested 10 cm of the contra lateral fibula and
since there was still space in the cavity , we packed bone graft substitute ( hydroxyapatite crystals ) into the defect ( fig . 2c ) .
post operatively the wound healed and good radiographic filling of the cavity was seen ( fig 2d ) .
patient was kept non - weight bearing for a month and then started on partial weight bearing mobilization .
two years follow up radiograph showed good healing with no recurrence ( fig 3 ) and patient was symptom free .
c- autogenous non vascularised fibula graft in situ and d- immediate post operative radiographic appearance .
osteofibrous dysplasia ( ofd ) is a rare benign fibro - osseous lesion , the patient usually presents with swelling of the leg with or without pain , or with anterior bowing .
some authors mention it as a precursor to adamantinoma   while other authors conclude that it is a variant of fibrous dysplasia .
campanacci in 1976 named this lesion as  osteofibrous dysplasia of the tibia and fibula  in reference to its anatomic location , developmental origin , and histologic resemblance to fibrous dysplasia  .
sakamoto et al . found that gsa mutation at the arg201 was seen in fibrous dysplasia but this did not occur in osteofibrous dysplasia , and they concluded that this could be useful for distinguishing between the two lesions .
radiologically the lesion usually involves the anterior cortex and may cause anterior bowing of the tibia .
it usually manifests as an intracortical lytic lesion , which is generally well marginated and is often surrounded by a zone of sclerosis .
our patient also had involvement of anterior cortex of the tibia with mild bowing , there were areas of multiple lucencies with intervening sclerotic areas ( fig 1 ) .
histologically ofd is characterized by a loose , often storiform fibrous background containing spicules of woven bony trabeculae that are lined by a layer of osteoblasts .
this histologic appearance is quite similar to that of fibrous dysplasia but fibrous dysplasia typically lacks the distinctive osteoblastic rimming of the bony trabeculae .
also ofd has a zonal architecture , in which more immature woven bony trabeculae are located centrally ; however , as we move to the periphery of the lesion , the trabeculae become more numerous , larger , and more mature and lamellar .
a biopsy is very important to establish the diagnosis and differentiate it from early adamantinoma .
the different options of treatment include , observation without surgical intervention , bracing to prevent fracture and minimize deformity , surgical option which includes en bloc resection , extraperiosteal resection and filling the defect with autogenous fibular graft , vascularized fibular graft or iliac crest bone graft .
most authors advocate excision of the tumor after skeletal maturity as it reduces the chance of recurrence .
studied 35 patients with this disease and have advocated surgery in patients with extensive disease , but it should be delayed as much as possible .
studied 16 patients with osteofibrous dysplasia and have advocated a more aggressive approach for this lesion .
5 patients underwent sharkbite extraperiosteal excision , 6 patients with larger lesions underwent segmental excision and fibula autograft .
two year follow tip radiographs showed good healing and no recurrence in our patient we did an extraperiosteal shark bite excision of the tumor . following the excision we had a large defect , so we packed the defect with contralateral autologous free fibular graft .
we wanted to avoid harvesting graft from the iliac crest , so we used bone graft substitute to completely fill the defect .
now at 2 year follow up the graft has incorporated well , ( fig.11 ) the child has no pain and there is no evidence of recurrence .
autologous fibular graft , along with bone graft substitute is a good alternative in treating this benign lesion to fill up the defect that arises after extraperiosteal excision in large lesions .
on doing a literature search we could not find any article using synthetic bone grafts along with autologous fibular graft as a mode of treatment of this lesion .
extraperiosteal excision of osteofibrous dysplasia combined with autologous free fibular graft and bone graft substitute is a good surgical option to mange bone defects in this rare lesion .
we believe this will reduce the chances of recurrence , however a larger series of cases will be needed to prove this , which would be difficult in this rare disorder .
after skeletal maturity extraperiosteal excision of the tumor combined with autologous fibular graft and bone graft substitute may be done to prevent recurrence .
 introduction : osteofibrous dysplasia is a rare benign self - limiting fibro - osseous lesion most commonly seen in the diaphysis of the tibia . 
 its incidence is reported to be 0.2% of all primary bone tumors . 
 it occurs in the first two decades of life with a slight male preponderance . 
 surgical options include extra periosteal resection , autologous graft , limb lengthening procedures etc . 
 there are no case reports mentioning the use of synthetic bone graft to fill the defect following extraperiosteal excision.case report : a 13 year old girl presented with pain and swelling of the ( r ) leg since 2 months following a trivial injury at school . examination revealed a 53 cm tender swelling on the anteromedial aspect of the middle third tibia . 
 radiographs and mri , revealed an eccentric expansile lytic lesion , which was multilocular and was present at the junction of the metaphysis and diaphysis on the antero -medial aspect of tibia . 
 the cortex had ballooned out and there was a possibility of an impending fracture . 
 biopsy was done which revealed osteofibrous dysplasia . 
 we did an extraperiosteal excision of the lesion . to fill the cavity we harvested 10 cm of the contralateral fibula and since there was still space in the cavity , we packed bone graft substitute ( hydroxyapatite crystals ) into the defect . 
 the surgical management of osteofibrous dysplasia is controversial . 
 various methods of treatment of such cases have been described in literature . 
 the use of synthetic graft is an option in these patients as it reduces morbidity ; and in our case we had good graft incorporation with this method.conclusion:extraperiosteal excision of osteofibrous dysplasia combined with autologous free fibular graft and bone graft substitute is a good surgical option to prevent recurrence and mange bone defects in this rare lesion . 
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Introduction
Case Report
Discussion
Conclusion