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Case Reports
. 2020 Aug 13;18(1):203.
doi: 10.1186/s12957-020-01983-9.

Osteofibrous dysplasia-like adamantinoma treated via intercalary segmental resection with partial cortex preservation using pedicled vascularized fibula graft: a case report

Yuji Yamamura  1 , Makoto Emori  2 , Nobuyuki Takahashi  1 , Mitsumasa Chiba  1 , Junya Shimizu  1 , Yasutaka Murahashi  1 , Shintaro Sugita  3 , Kousuke Iba  1 , Tadashi Hasegawa  3 , Toshihiko Yamashita  1
Affiliations
Case Reports

Osteofibrous dysplasia-like adamantinoma treated via intercalary segmental resection with partial cortex preservation using pedicled vascularized fibula graft: a case report

Yuji Yamamura et al. World J Surg Oncol. .

Abstract

Background: Morphologically, osteofibrous dysplasia-like adamantinoma is thought to be intermediate between osteofibrous dysplasia and adamantinoma. Its treatment is not well established owing to its rarity.

Case presentation: We report about of a 10-year-old girl with osteofibrous dysplasia-like adamantinoma initially diagnosed as osteofibrous dysplasia and treated via intercalary segmental resection with partial cortex preservation using a pedicled vascularized fibula graft for reconstruction. Bone union was observed 9 weeks after surgery. Twenty-two months after the definitive surgery, no recurrence was observed.

Conclusion: This case illustrates the upgrade from osteofibrous dysplasia to osteofibrous dysplasia-like adamantinoma. The surgical method may aid the treatment of osteofibrous dysplasia-like adamantinoma with incomplete cortex involvement of the tumor.

Keywords: Bone tumor; Osteofibrous dysplasia; Vascularized fibula graft.

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Conflict of interest statement

The authors declare that they have no conflict of interest.

Figures

Fig. 1
Fig. 1
The X-ray, CT image, and MR images of the tumor. a Anteroposterior radiograph. b Lateral radiograph. c Axial CT image. d T1-weighted magnetic resonance sagittal image. e T2-weighted magnetic resonance sagittal image
Fig. 2
Fig. 2
The patient was diagnosed with osteofibrous dysplasia because there was no epithelial component in the biopsy specimen
Fig. 3
Fig. 3
The mass was a heterogeneously hypointense and isointense on a T1-weighted magnetic resonance image and b heterogeneously hyperintense on short TI inversion recovery magnetic resonance image. No pathological fracture was confirmed
Fig. 4
Fig. 4
a A locking plate was used to mechanically align and stabilize the lower leg. A pedicled vascularized fibula graft was inserted into the medullary cavity of the tibia and locked in place with cortical screws. b Bone union was observed 9 weeks after surgery, and the patient began to partially bear weight. c Complete bone union was observed 8 months after surgery. d The locking plate and screws were removed 19 months after surgery
Fig. 5
Fig. 5
The final diagnosis was osteofibrous dysplasia-like adamantinoma because the surgical specimen contained an epithelial component (a) with positive AE1/AE3 immunostaining (b)
See this image and copyright information in PMC

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