Aneurysmal Bone Cyst of the Fifth Metacarpal

Abstract

Aneurysmal bone cyst is a rare, rapidly growing, and destructive benign bone tumor that even more rarely involves the bones of the hand. Various treatment options for aneurysmal bone cyst have been reported in the literature, but controversy exists regarding optimal treatment. Due to its rarity in the hand, no evidence-based treatment regimen has been established.
A 21-year-old man presented with a history of pain and local swelling over his fifth metacarpal of 5 months' duration. Physical and radiographic examination of the hand was consistent with aneurysmal bone cyst. After biopsy, pathologic examination confirmed the diagnosis of aneurysmal bone cyst. En-block resection of the tumor and autologous bicortical strut graft fixation with Kirschner wires was performed. The hand was immobilized in a short arm cast for 3 weeks after the patient received 3 weeks of physiotherapy. Kirschner wires were removed 6 weeks postoperatively. Excellent clinical and functional results were obtained with no recurrence after 3 years of follow-up with en-block resection and reconstruction with iliac crest graft. Radiographic examination demonstrated the osseous integration of the graft with no signs of recurrence.
Although treatment should be planned individually according to lesion site and size and to patient age, we suggest en-block resection to prevent recurrence and secondary surgical interventions particularly in cases with no articular involvement.

Full text

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■ Case Report
abstract
Full article available online at OrthoSuperSite.com/view.asp?rID=41932
extremity
SPOTLIGHT ON
upper
Aneurysmal Bone Cyst of the Fifth
Metacarpal
SELAHATTIN OZYUREK, MD; OSMAN RODOP, MD; OZKAN KOSE, MD; FERIDUN CILLI, MD;
MAHIR MAHIROGULLARI, MD
Aneurysmal bone cyst is a rare, rapidly growing, and destructive benign bone tumor
that even more rarely involves the bones of the hand. Various treatment options for
aneurysmal bone cyst have been reported in the literature, but controversy exists re-
garding optimal treatment. Due to its rarity in the hand, no evidence-based treatment
regimen has been established.
A 21-year-old man presented with a history of pain and local swelling over his fi fth meta-
carpal of 5 months’ duration. Physical and radiographic examination of the hand was
consistent with aneurysmal bone cyst. After biopsy, pathologic examination confi rmed
the diagnosis of aneurysmal bone cyst. En-block resection of the tumor and autologous
bicortical strut graft fi xation with Kirschner wires was performed. The hand was immobi-
lized in a short arm cast for 3 weeks after the patient received 3 weeks of physiotherapy.
Kirschner wires were removed 6 weeks postoperatively. Excellent clinical and functional
results were obtained with no recurrence after 3 years of follow-up with en-block resec-
tion and reconstruction with iliac crest graft. Radiographic examination demonstrated
the osseous integration of the graft with no signs of recurrence.
Although treatment should be planned individually according to lesion site and size
and to patient age, we suggest en-block resection to prevent recurrence and secondary
surgical interventions particularly in cases with no articular involvement.
Dr Ozyurek is from the Department of Orthopedics, Izmir Military Hospital, Izmir, Drs Rodop, Cilli,
and Mahirogullari are from GATA Haydarpasa Training Hospital, Istanbul, and Dr Kose is from Diyar-
bakir Education and Research Hospital, Diyarbakir, Turkey.
Drs Ozyurek, Rodop, Kose, Cilli, and Mahirogullari have no relevant fi nancial relationships to disclose.
Correspondence should be addressed to: Selahattin Ozyurek, MD, Department of Orthopedics,
Izmir Military Hospital, Inonu Caddesi, Hatay, Izmir, Turkey.
DOI: 10.3928/01477447-20090624-25
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ANEURYSMAL BONE CYST | OZYUREK ET AL
Aneurysmal bone cyst is a rare, rap-
idly growing, and destructive be-
nign bone tumor. Jaffe and Lich-
tenstein1 described aneurysmal bone cyst
in 1942 as a distinct pathological entity by
clearly separating it from hemangiomas of
the bone and from other tumors in which
giant cells were also a prominent feature.
It is a rare tumor and accounts for 1% to
2% of all primary bone tumors.2 Aneurys-
mal bone cysts usually occur in the fi rst 2
decades of life and exhibit a slight female
preponderance.2,3 Aneurysmal bone cyst
shows an evident predilection for long
bones and the vertebral column, particu-
larly the femur, humerus, tibia, and fi bula.
However, aneurysmal bone cysts arising
from long bones of the hand occur rarely.
Less than 5% of all aneurysmal bone cysts
involve long bones of the hand.4
The pathogenesis of aneurysmal bone
cyst is obscure. Lichtenstein5 suggested
that persistent local disturbance in hemo-
dynamics (venous thromboses or arteriove-
nous aneurysm) causes marked increase in
venous pressure and leads to development
of a dilated engorged vascular bed. Some
authors proposed that aneurysmal bone
cysts arise on a preexisting bone lesion as
a secondary reaction.6,7 Trauma has been
implicated as an initiative factor due to the
fact that aneurysmal bone cyst is preceded
by trauma with fracture or subperiosteal
hematoma in some cases.8 However, most
authors agree that trauma draws attention to
a preexisting lesion.9,10 Recently, the genet-
ic basis of aneurysmal bone cyst has been
investigated, and specifi c chromosomal
translocations have been reported.2,11,12
Furthermore, overexpression of insulin-
like growth factor 1 is postulated to play a
role in the pathogenesis.2,13
The natural history of aneurysmal
bone cyst has been described as evolv-
ing through 4 radiologic stages: initial,
active, stabilization, and healing.14 In the
initial phase, the lesion is characterized
by a well-defi ned area of osteolysis with
discrete elevation of the periosteum. This
is followed by a growth phase, in which
the lesion grows rapidly with progressive
destruction of bone and development of
the characteristic blown-out radiologic
appearance. The growth phase is succeed-
ed by a period of stabilization, in which
the characteristic soap bubble appearance
develops as a result of maturation of the
bony shell. Diagnosis generally occurs
during the active or stabilization phase.
Final healing results in progressive calci-
fi cation and ossifi cation, with the lesion
transformed into a dense bony mass.
Histologically, aneurysmal bone cyst is
composed of cavernous or slit-like hem-
orrhagic spaces surrounded and traversed
by fi brous septa containing spindled cells,
infl ammatory cells, and a lesser number of
osteoclast-like multinucleated giant cells
that are often distributed around the hem-
orrhagic, cystic spaces. Typically, osteoid
formation with or without osteoblastic
rimming is observed.2,15
Various options for the treatment of
aneurysmal bone cyst have been reported
in the literature,16 but controversy exists
regarding optimal treatment. Due to its
rarity in the hand, there is no established
evidence-based treatment regimen. This
article presents a case of aneurysmal bone
cyst affecting the fi fth metacarpal that was
treated by en-block resection and recon-
struction with bicortical iliac crest graft.
CASE REPORT
A 21-year-old man presented with a his-
tory of pain and local swelling over his fi fth
metacarpal of 5 months’ duration. On physical
examination, the lesion was fi rm and immobile
and there was slight tenderness with palpation.
Active range of motion of his fi fth metacarpo-
phalangeal joint was slightly restricted, and
pain was aggravated with movement. There
was no history of trauma. His past medical his-
tory revealed no abnormality.
Radiographic examination of the hand
showed a marked increase in diameter along
the fi fth metacarpal and widening of the med-
ullary canal. The cortex was uniformly thin and
the metacarpal head was spared (Figure 1A).
The characteristics of the lesion were consis-
tent with aneurysmal bone cyst. After biopsy,
pathologic examination confi rmed the diagno-
sis of aneurysmal bone cyst (Figure 1B).
En-block resection of the tumor was per-
formed through a dorsal longitudinal incision
over the fi fth metacarpal. The metacarpal head,
together with its carpometacarpal joint capsule,
was left intact. Autologous bicortical strut graft
was harvested from left iliac crest. The graft was
molded into its defi nitive shape and inserted into
the created bony defect. Multiple K-wires were
used for graft fi xation (Figure 2). The hand was
immobilized in a short arm cast for 3 weeks af-
ter the patient received 3 weeks of physiothera-
py consisting of progressive active range of mo-
tion exercises. Kirschner wires were removed 6
weeks postoperatively.
At fi nal follow-up 3 years postoperatively,
the patient had gained full range of hand mo-
tion with no pain. The patient was satisfi ed
with the functional and cosmetic results. Ra-
diographic examination demonstrated osseous
integration of the graft with no signs of recur-
rence (Figure 3).
Figure 1: Radiograph of the hand at admission (A). Pathologic appearence of the tumor (B; hematoxylin-
eosin, magnifi cation ⫻40).
1A 1B
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ORTHOPEDICS | ORTHOSuperSite.com
■ Case Report
DISCUSSION
The main goals in the treatment of
aneurysmal bone cyst of the hand are
eradication of the lesion, prevention of
recurrence, and preservation of hand
function. The literature contains confl ict-
ing knowledge about the optimal treat-
ment method.
Currently, curettage and bone grafting
is the most common operative procedure
used. However, recurrence rate is high af-
ter this procedure. Basarir et al17 reported
that 2 of 3 cases that were initially treated
with curettage and grafting recurred. Sim-
ilarly, in a case series by Frassica et al,18
curettage and bone grafting in 7 cases was
associated with 4 recurrences.
However, contrary reports are also
found in the literature. Ropars et al19 sug-
gested that curettage and grafting is suf-
fi cient for treatment, and aggressive meth-
ods such as cryotherapy or resection with
reconstruction should only be used in case
of recurrences and articular involvement.
Other authors have reported parallel suc-
cessful outcomes with no relapse after
simple curettage and grafting.20,21 A prob-
lem with this method is that osteoclastic
activity can reabsorb the graft material,
depending on the aggressiveness of the
lesion. Another limitation of this method
is that if graft incorporation occurs, the
original size of the lesion is present and
can take years to remodel. Possible re-
currence after insuffi cient primary treat-
ment will increase the size of the defect;
the tumor may reach joint structures, and
consequent bone grafting must include the
epiphysis and even complete joints.
Due to the high risk of recurrence af-
ter curettage and grafting alone, various
forms of adjunctive therapy have been
used to decrease the rate of local recur-
rence.16 There are 2 cases in which cryo-
surgery and sclerotherapy were used as an
adjuvant intralesional treatment for aneu-
rysmal bone cyst arising in the hand.22,23
These treatments are diffi cult to use in the
small bones of the hand and may damage
surrounding intact tissue and cause seri-
ous complications such as neurapraxia,
postoperative fracture, burn, infection,
and wound necrosis, which may happen
more easily in distal lesions. Although a
3.7% local recurrence rate was reported
with cryosurgery, there is a potential risk
of amputation of small bones.22,24
En-block resection and reconstruction
with strut grafting is another operative
treatment option. Given the aggressive
nature of aneurysmal bone cysts with the
tendency to develop local recurrence, en-
block resection seems to be the therapy
of choice. No recurrences have been re-
ported after en-block resection in the rel-
evant literature.10,17,18,25-29 Despite it being
a curative method of treatment, its use is
limited, particularly in cases where the le-
sion is close to articular surfaces. Articular
surface reconstruction and preservation of
hand function need further advanced op-
erative techniques such as nonvascular-
ized or vascularized toe phalanx trans-
plantation.23,30,31 Long operation time, the
need for microsurgical skills, and donor
site complications are major problems
associated with these techniques. Other-
wise, reconstruction can only be achieved
with arthrodesis, which may impair hand
function.
In our case, excellent clinical and
functional results were obtained with en-
block resection and reconstruction with
iliac crest graft with no recurrence after
3-year follow-up. The metacarpal head
was spared; therefore, articular surface
was left intact.
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