Femoral_Reconstruction_Using_Long_Tibial_Autograft.24.pdf

Femoral Reconstruction Using Long Tibial Autograft After
Resection of Giant Aneurysmal Bone Cyst
Mohammed S. Alisi, MD,*†‡ Freih Abu Hassan, FRCS,*
Mohammad N. Alswerki, MD,* Ahmed A. Abdulelah, MD,§
and Mohammad A. Alshrouf, MD§
Introduction: Management of giant aneurysmal bone cyst in skeletally
immature patients is challenging. The huge size can be destructive and
can lead to significant deformity, disability, and limb-length discrepancy.
Hence, reconstruction is warranted as a treatment option.
Materials and Methods: Here, we present a case of giant aneurysmal
bone cyst in the distal femur of a 9 year old obese child. After complete
resection, we reconstructed the large distal femur defect by a 15 cm
long-tibia autograft of the same limb. Fixation was done using hybrid
(monoplanar and circular) external fixation. Detailed surgical techni-
ques and several clinical and technical challenges have been discussed.
Results: After 13 years of follow-up, the distal femur is well formed
with preservation of the articular surface, and the site of the tibia
autograft is completely regenerated.
Conclusion: The tibia can be used as an autograft for the reconstruction
of large-sized defects in skeletally immature patients.
Key Words: aneurysmal bone cyst—giant—reconstruction—
autograft—tibia.
(Tech Orthop 2022;00: 000–000)
The aneurysmal bone cyst (ABC) is an expanding osteolytic
lesion, consisting of blood-filled cavernous channels of
variable size, surrounded by a spindle cell stroma separated by
connective tissue septa containing trabeculae or osteoid tissue
and osteoclast-like giant cells.1 It accounts for 1% of benign
bone tumors, and 30% of aneurysmal bone cysts (ABCs) are
secondary tumors. The majority of ABCs (80%) were observed
in skeletally immature individuals during the first 2 decades of
life.2
ABC can virtually arise in any bone, and more commonly
affects the metaphysis of the tubular bones (femur, tibia, and
fibula) and flat bones such as the spine and pelvis.3 It can cause
pain, swelling, deformity, fracture, disruption of the growth
plates, or neurological symptoms.
Giant ABC is an uncommon lesion that has a more
destructive effect on the bones and causes pressure on the sur-
rounding structures.4 The transverse diameter of the giant ABC
equals 3 times or more than the transverse diameter of the normal
bone. Giant ABC has been described in flat and small tubular
bones such as the mandible, rib, pelvis, fibula, metacarpal,
metatarsal, clavicle, sacrum, spine, and humerus.5–9
The treatment modality proposed for giant ABC is
resection with tibia allograft for weight-bearing bones or non-
vascularized fibular autograft for small tubular or upper limb
bones.5,6,10
Here, we describe a case of a 9-year-old boy with an
unusual ABC located to the distal femur, which became giant
and aggressive over 14 months, and caused significant walking
disability.
Our surgical strategy depended on staged total resection
and reconstructive surgery using a tibia structural strut autograft
stabilized by hybrid external fixation.
The patient and his family signed a written consent to
publish their data and images.
MATERIAL AND METHODS
This study has been waived from approval by our institutional
review board.
Case Presentation
A 9-year-old boy, morbidly obese with a body mass index
of 45, presented to our clinic complaining of slowly growing
painless swelling of the right distal thigh of 14 months duration.
He had a history of twice-right femur fractures (4 mo apart)
after simple trauma that was treated conservatively by casting
(Fig. 1). Incidentally, a lytic lesion seemed on the radiographs
at the site of the fracture, for which an open curettage was done
later on at the peripheral hospital with no documented evidence
of the lesion type.
Upon physical examination, he had large, diffuse, non-
tender, nonpulsatile, and firm swelling of the right knee and
distal thigh. There was a 6 cm scar on the lateral side of the
thigh indicating the site of the previous surgery (Fig. 2).
The right knee range of motion is limited due to swelling. The
neurovascular status of the right lower limb was intact.
Laboratory investigations such as complete blood count,
kidney function tests, erythrocyte sedimentation rate, C-reactive
protein, and alkaline phosphatase were within normal ranges.
Plain radiographs of the right femur showed a large
multilocular expansile osteolytic “blow-out” lesion, outlined by
a paper-thin shell of bone in the distal third femur, with loss of
normal bone architecture (Fig. 1E).
Magnetic resonance image (MRI) demonstrated the presence
of a 15 ×12 cm large multilocular cystic expansile lesion in the
distal third of the femur with multiple fluid levels, with no signs of
soft tissue mass or nodularity (Fig. 3).
From the *Department of Special Surgery, Division of Orthopaedics;
§School of Medicine, The University of Jordan, AMM, Jordan; †Faculty of
Medicine, Islamic University of Gaza; and ‡Faculty of Medicine, Al-Azhar
University, Gaza, Palestine.
The authors declare that they have nothing to disclose.
For reprint requests, or additional information and guidance on the
techniques described in the article, please contact Mohammed S. Alisi,
MD, at m.elessi2007@hotmail.com or by mail at The University of
Jordan, School of Medicine, Queen Rania Al Abdullah Street 266 Al
Jubaiha Amman, 11942, Jordan. You may inquire whether the author
(s) will agree to phone conferences and/or visits regarding these
techniques.
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Operative Procedure
A trucut core biopsy conﬁrmed the diagnosis of the ABC.
One week later, we applied 2 Ilizarov rings connected by
rods around the knee. Then, we did local curettage through the
previous lateral incision. Because of the extensive bleeding, the
cavity was packed with surgical Gamgee gauzes for 48 hours.
This was followed by the application of a monoplanar external
ﬁxator connected to the rings of Ilizarov to stabilize the femur
(Fig. 4).
Four weeks later, under proximal thigh-tourniquet pressure
control (220 mm Hg), we did a midline anterior distal thigh
incision to evacuate hematoma and seroma. Multiple samples
were sent for cultures and histopathology. We irrigated the
surgical site with 3 L of normal saline, followed by total exci-
sion of the remnants of the cyst with the careful clearing of the
remnants from the surface of the growth plate by peanuts swabs.
The tibia strut autograft was harvested in 1 piece by
subperiosteal resection involving all the cortices without leav-
ing a strut of normal bone behind (Fig. 5). A longitudinal
incision was made over the anteromedial aspect of the tibia
down to the periosteum. The periosteal sheath was incised
longitudinally and raised gently with a sharp periosteal elevator
from all around the tibia. Multiple 2.5 mm diameter drill holes
were made at the tibia proximally and distally, the bone ends
cut by a small sharp osteotome, followed by removal of
the graft.
The medullary canal proximally and distally was opened
using a 3 mm curette to allow the free communication of the
bone marrow with the tibia defect. The remaining periosteal
sheath was sutured by continuous closure using 2/0 vicryl
suture to obtain a tight closure to form a periosteal tube. The
skin was closed by subcuticular sutures using 2/0 vicryl, with
no wound drainage.
About 40 mL of iliac crest bone marrow was injected into
the periosteal tube through plastic green cannula size G16 to
prevent the collapse of the periosteal tube, and allow for the
organized hematoma to initiate the future tibia bone
regeneration.
One Ilizarov ring was added to the distal tibia and con-
nected by rods to the proximal tibia ring to stabilize the donor
site until the regeneration of the tibia (Fig. 4).
The nonvascularized tibia strut autograft was implanted to
bridge the defect gap in the distal third of the right femur. The
proximal end of the graft passed for 2 cm through the femoral
FIGURE 1. Radiograph of the right knee showing the evolution from simple aneurysmal bone cyst (ABC) to giant ABC. A, AP and lateral
views plain radiograph demonstrate a radiolucent lesion involving the supracondylar area of the right femur with very thin cortices associated
with long oblique fissure fracture. B, The same lesion 3 months after casting showed healing of the fracture with more septations and
thickening of the proximal cortices. C, Comminuted fracture in the cyst seen on AP and lateral views of plain radiograph in the supracondylar
area, 3 months after removal of the cast with posterior angulation of the distal femur. D, The same lesion 3 months after the second fracture
showing progressive expansion with more septations at the distal femur. E, AP and lateral views plain radiograph at the first presentation to
our clinic, showing metaphyseal giant, expansile, centric, and lytic lesion with bony septae “bubbly appearance,” thin rim of periosteal new
bone surrounding at the proximal end of the lesion, and no matrix mineralization. AP indicates Anteroposterior.
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canal and was stabilized anteriorly by two 4 mm Schanz screws
connected to the proximal Ilizarov ring by the Ilizarov plate.
The distal end of the graft was fixed by a 2 mm Ilizarov wire to
the middle of the distal femoral epiphysis (Fig. 4).
The patient was mobilized on a walking frame nonweight-
bearing after 3 days for 4 weeks, then partial weight-bearing on
a walking frame for another 4 weeks. Then, he was allowed full
weight-bearing as tolerated.
After 10 weeks, we removed the rods between the proximal
2 Ilizarov rings for knee exercises and exchanged the mono-
planar lateral fixator with a special long Ilizarov plate as it was
attached to the proximal 2 rings (Fig. 6B and C).
Two months later, we removed the loose proximal anterior
Schanz screw and the other distal screw with the plates from the
distal femur (Fig. 7A).
Unfortunately, the graft had a fracture at the site of the
previous distal anterior Schanz screw after 2 weeks of removal
(Fig. 7B). This was treated conservatively.
All remaining metalwork was removed 15 months post-
operatively, followed by insertion of locked retrograde femoral
nailing 1 month later due to nonunion of the fractured site of the
graft (Fig. 7F and G). The nail was removed after 3.5 years
(Fig. 7H).
The 2 Ilizarov rings on the tibia were removed 6 months
postoperatively after confirmation of the regeneration of the
donor site (Fig. 8A).
The early signs of the graft union with the femur were
noticed by the formation of callus at the proximal end 1 month
after implantation and distally as new bone formation from the
remnant growth plate after 2 months of implantation (Fig. 6B).
The early signs of new bone formation at the donor site
were noticed after 4 weeks of harvesting as a thin new bone
filling the defect. The obvious regeneration of the tibia was
noticed at the donor site after 3 months (Fig. 6C).
Table 1 summarizes the sequence of events in a timetable
manner from the initial presentation to the final follow-up.
Of note, the results of intraoperative cultures were neg-
ative for infection and multiple histopathology specimens
emphasized the initial diagnosis of ABC.
DISCUSSION
The main concern in the management of the ABC is the
presence of an existing bone tumor, mostly telangiectatic
osteosarcoma (TOS). Preexisting benign bone lesions such as
fibrous dysplasia, giant cell tumors, chondroblastoma, and
nonossifying fibromas also have been reported.11 We did not
found these concerns in our case after multiple biopsies were
sent for histopathology.
High recurrence rates have been reported after the surgical
treatment of ABCs, ranging from 10% to 59%, especially in
young patients with local curettage.12,13 We overcome this
problem by complete resection of the giant ABC.
As most ABCs are encountered in the metaphysis of an
immature skeleton, permanent limb deformities or growth arrest
FIGURE 3. Magnetic resonance image of the distal third of the right thigh showing the characteristic fluid levels and septations (A), and
low signal on T1 weighted images (B) and high signal on T2 weighted images (C), with no solid components.
FIGURE 2. Clinical photograph showing large diffuse swelling of
the right knee and distal thigh seen from the lateral (A) and
medial (B) sides with the scar of previous surgery on the lateral
side of the thigh.
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FIGURE 5. Steps of tibia autograft harvesting. A, Exposure of the tibia through a longitudinal incision, with opening the periosteum
longitudinally and raising it with periosteal elevator from all around the tibia, protected by small blunt bone levers inserted inside the
periosteal sheath around the proximal and distal end of the proposed osteotomy. B, Removal of the tibia autograft in 1 piece. C, The
intact periosteum sheath after removal of the graft with early hematoma formation. D, The periosteum was closed continuously with
Vicryl suture to form a tube.
FIGURE 4. Postoperative plain radiograph and photograph. The tibia graft stabilized at the femur proximally by Schanz screw (A) and
distally by K wire (B). The defect of the right tibia (C) and the whole limb stabilized by Hybrid external fixation (D).
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are of great concern. In this way, it is of importance to preserve
the growth plate, and a future follow-up for bone length is
encouraged to maintain the adequate function of the involved
limb and neighboring joints.
It has been estimated that 7% to 15% of patients develop
growth disturbance in cysts directly adjacent to the growth plate
due to the pressure effect of the cyst.14 We made our efforts to
preserve the growth plate by gently sweeping the remnants of the
cyst from the growth plate with peanuts swabs. The viability of the
growth plate was noticed by the prominent bone formation around
the distal end of the graft (Fig. 7E). At a later stage, we were
forced to insert locked retrograde nailing of the femur through the
graft to overcome the nonunion, which compromised the growth
as the patient had a 6 cm leg-length discrepancy (Fig. 7J).
The plain radiograph is essential for early investigation
and it did reveal in our case, the progressive features of ABC as
a large radiolucent multilocular osteolytic lesion with thin
sclerotic borders “soap bubble appearance” in the distal third of
the right femur (Fig. 1).
Magnetic resonance imaging (MRI) is required to sup-
plement the radiographs as it reveals the expansive lesion with
double density fluid level and septations. Low signals on T1
images and high signals on T2 images are suggestive of ABC.15
All the MRI features were found in our case besides its giant
cystic form.
TOS has occasionally been misdiagnosed as an ABC.2 MRI
can help differentiate the 2 by demonstrating a corresponding soft
tissue mass with a nodular appearance and necrosis to the tissue
surrounding the cavities in TOS. TOS will also reveal cortical
destruction and thick septal enhancement.1,16 We did not observe
any soft tissue lesions, nodularity, necrosis, or thick septal
enhancement on all MRI sequence images in the reported case.
The cortical destruction in our case was attributed to the
long-standing aggressive nature of the nontreated ABC, how-
ever, histopathology examination of tissue biopsy remains
crucial for the definitive diagnosis of malignant tumor cells.17
The histopathology suggestive of ABC shows proliferative
fibroblasts, spindle cells, areas of osteoid formation, and an
uneven distribution of multinucleated giant cells that tend to
surround the fluid‑filled cavities in a “pigs at the trough”
formation.18 This was confirmed 3 times and revealed the
benign nature of the giant ABC.
Despite the wide range of treatment modalities, the man-
agement of ABCs should be tailored and individualized
according to cyst size, location, type of bone involved, aggres-
siveness, and extent of the lesion to prevent local recurrence.
Curettage with or without an adjuvant such as phenol,
hydrogen peroxide, liquid nitrogen, or polymethylmethacrylate
remains the standard procedure in localized contained ABC.19
Embolization may be a primary treatment in large lesions
to minimize intraoperative bleeding, as in pelvic ABC.20 We
may be benefited from embolization if the service was found at
our institution.
Bone transport after tumor resection encountered side
effects of pin tract infection, nonunion, and limitation of the
transport up to 10 cm.21 This technique was not applicable in
our case as the defect length was more than 10 cm. Besides, the
large size of the patient thigh due to obesity may preclude the
applicability of this option.
Cortical strut allografts have an important role in the
treatment of large benign bone lesions of the upper limb after
resection. It brings the advantage of unlimited supply without
additional donor site morbidity.22 The vascularized bone grafts
could replace large bone defects with faster incorporation and
remodeling. They are technically demanding procedures with a
high failure rate for those with limited experience.23,24
Successful long-term results of surgical resection and
nonvascularized autologous fibular graft for small tubular and
upper limb giant ABC are technically much easier to use and
provide excellent structural bone support at the recipient side.5
The nonvascularized autologous fibular graft was not applicable
in our case of giant ABC of the distal femur, because we need a
large size graft comparable to femur diaphysis.
Giant ABC of large weight-bearing bones is scarcely found
in the orthopedic literature. En-bloc excision is the only treatment
modality associated with the lowest rate of recurrence 0%.4,5,14,25
The vascularized and nonvascularized autografts have been used
as a reconstructive method after the resection of giant ABCs.
To the best of our knowledge, no description in the literature
examines the use of tibia strut autograft for the reconstruction of
the defects after major resection of bone pathology. Only 1 article
has been published recently describing the same technique but for
malignant bone tumors.26 Interestingly, this article is also from
our center.
FIGURE 6. Early follow-up images. A, AP view plain radiograph of the proximal femur showing new callus formation at the proximal end of the
graft with the femur after 6 weeks (open white arrows). B, AP view plain radiograph of the knee joint with the distal third femur and proximal
tibia showing new bone formation from the remaining growth plate around the distal end of the graft with femur distal epiphysis after 8 weeks
(closed red arrows). C, AP view plain radiograph of the right leg showing the well-formed new bone regeneration at the donor site (yellow
arrows) with small defect distally (open white arrow) after 3 months. D, A standing photograph of the patient 3 months postoperatively showing
the gross obesity of the right thigh and the construct of the hybrid external fixator. AP indicates Anteroposterior.
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FIGURE 7. Long-term follow-up radiographs demonstrating the serial progression of graft incorporation. A, AP view plain radiograph of
the right femur showing loosening of the proximal Schanz screw 18 weeks after surgery. B, AP view plain radiograph of the right femur
showing transverse fracture of the graft at the site of distal Schanz screw 20 weeks after surgery. C, AP view plain radiograph the right
femur showing mild valgus angulation with early callus formation at the fracture site 2 months after the fracture. We inserted a new
Schanz screw proximally for more stability. D, AP view plain radiograph the right femur after removal of the external fixator, showing well-
incorporated graft proximally and good callus on the medial side of the fractured graft but poor callus laterally (closed red arrow), which
indicates instability at this site. E, AP view plain radiograph of the knee and distal femur 1 month after removal of the fixator showing
prominent new bone formation around the distal end of the graft which indicative of viable growth plate (open yellow arrows). F and G,
AP and lateral views plain radiograph of the right femur 6 months after nailing. showing broadening of the graft to be a new femur. H, AP
view plain radiograph after nail removal (3.5 y after insertion). I, AP view plain radiograph of hips and both femurs 13 years after surgery
showing normal shape femur with leg-length discrepancy. AP indicates Anteroposterior.
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FIGURE 8. Progression of regeneration of bone at the donor site. A, Oblique view plain radiograph of the right tibia donor site 6 months
after harvesting the graft showing regeneration of the tibia with mild distal defect (open white arrow). B, AP view plain radiograph of the
right tibia 18 months after harvesting showing complete regeneration of the donor site with early increase density of cortices and around
15-degree valgus deformity. C, AP view plain radiographs of the right tibia 40 months after harvesting showing complete regeneration of the
donor site with prominent medullary canal and cortical thickening with improvement in valgus deformity to 10 degrees. D, AP view standing
plain radiographs of both tibias 13 years after harvesting showing complete remodeling of the right tibia. AP indicates Anteroposterior.
TABLE 1. Summarize the Sequence of Events From the Initial Presentation to the Final Follow-up
Past history Two times of right distal femur fracture; treated conservatively.
On follow-up for the second fracture Lytic lesion was observed at the site of fracture, which was managed with open curettage at another hospital
with no documentation of lesion type (Fig. 1).
Presentation Right distal thigh painless swelling of 14 mo duration.
Radiograph showed large multilocular expansile osteolytic “blow-out” lesion.
Magnetic resonance image demonstrated the presence of a 15 × 12 cm large multilocular cystic expansile lesion
(Fig. 3).
Biopsy of the lesion (at the time of
presentation)
Aneurysmal bone cyst was confirmed by histopathology examination.
First surgery (1 wk later) Local curettage, Ilizarov fixation, and addition of monoplanar external fixator.
Second surgery (4 wk later) Complete excision of the lesion and curettage of cyst remnants from the growth plate.
Harvesting of tibia strut autograft with injection of iliac crest bone marrow in the periosteal sleeve at the site of
graft (Fig. 5).
Implantation at the bone defect at distal femur.
Stabilization and fixation of the donor and recipient sites by Ilizarov ring fixator (Fig. 4).
Postoperative course
(4-10 wk later) Nonweight-bearing ambulation for 4 wk, then partial weight-bearing for 2 wk, then weight-bearing as
tolerated.
Outpatient follow-up (10 wk later) Ambulation with full weight-bearing, removal of 2 Ilizarov rings for knee range of motion exercises, exchange
of monoplanar lateral fixator with specialized long Ilizarov plate (Figs. 6B, C).
Follow-up at (18 wk) Loosening of proximal anterior Schanz screw; we removed it with the other distal screw and plate from the
distal femur (Fig. 7A).
Follow-up at (20 wk) Fracture was noticed at the graft, at the site of previous distal anterior Schanz screw; was treated conservatively
(Fig. 7B).
Follow-up at (6 mo) The 2 Ilizarov rings on the tibia were removed after confirmation of the regeneration of the donor site
(Fig. 8A).
Follow-up at (15 mo) All remaining metalwork around the femur was removed.
Follow-up at (16 mo) Nonunion at the graft site was noticed, followed by insertion of locked retrograde femoral nailing.
Follow-up at (18 mo) Complete regeneration of the donor site (tibia) with early increase density of cortices and around 15-degree
valgus deformity (Fig. 8B).
Follow-up at (22 mo) 6 mo after femoral nailing; radiographs showed well-formed femur and complete incorporation of the graft
(Figs. 7F, G).
Follow-up at (3.5 y) The femoral nail was removed (Fig. 7H).
Follow-up at (13 y) Well remodeled donor and recipient sites with residual limb shortening about 6 cm (Figs. 7J, 8D).
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Subperiosteal resection of long tubular bone cyst is a
successful surgical modality without using a bone graft as the
remaining periosteal sac will cause regeneration of the new
bone within weeks.5 With the previous idea in our mind, we
used the subperiosteal harvest of the middle ipsilateral tibia and
closure of the periosteum to form a tube followed by bone
marrow filling. Then, we implanted the tibia strut graft to bridge
the remaining defect in the distal femur.
The tibia completely regenerated after harvest, with early
signs of new bone formation at 1 month, full regeneration at
3 months, and progressed to complete remodeling (Fig. 8)
To the best of our knowledge, giant ABC in the distal
femur of an immature skeleton has not been described before.
We had the following challenges during dealing with this
case. The giant ABC requires total resection, with considerable
risk of bleeding. The resection was performed in 2 stages,
stabilization with hybrid external fixation first then curettage as
much as we can from the ABC followed by packing with
Gamgee gauzes for 48 hours. The definitive surgery was done
after 4 weeks.
The length of the defect was quite large (about 15 cm)
after resection, which was difficult to reconstruct. We used the
nonvascularized tibia autograft graft as it matches the
femur width.
The patient developed a 6 cm leg-length discrepancy at
maturity due to the fracture in the implanted graft and the
locked retrograde nailing. This issue was planned to be
addressed after finishing his university due to financial issues.
The final problem was the previous lateral incision scar,
which was not ideal for reconstruction, and the presence of a
monoplanar fixator on the lateral thigh. We used the anterior
approach to complete the resection and application of the strut
tibia graft.
CONCLUSION
Despite the major challenges and obstacles during the
management of such giant ABC in an immature skeleton
patient, we successfully managed the resection and recon-
struction using a unique new technique of reconstruction. This
technique ended with complete regeneration of the donor site
and incorporation of the graft in the recipient site with the final
remodeling of the distal femur.
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