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VOL. 91-B, No. 9, SEPTEMBER 2009 1227
Subperiosteal resection of aneurysmal bone
cysts of the distal fibula
F. O. Abuhassan,
A. O. Shannak
From Jordan
University Hospital,
School of Medicine,
Queens Rania Street,
Amman 11942,
Jordan
„ F
. O. Abuhassan, FRCS,
FRCS(Trauma & Orth),
Professor
„ A. O. Shannak, FRCS(G),
FRCS(Orth), Professor
Department of Orthopaedic
Surgery
Jordan University, School of
Medicine, Queens Rania Street,
Amman 11942, Jordan.
Correspondence should be sent
to Mr F
. O. Abuhassan; e-mail:
freih@ju.edu.jo
©2009 British Editorial Society
of Bone and Joint Surgery
doi:10.1302/0301-620X.91B9.
22395 $2.00
J Bone Joint Surg [Br]
2009;91-B:1227-31.
Received 4 February 2009;
Accepted after revision 27 May
2009
We describe the treatment by subperiosteal resection of an aneurysmal bone cyst in the
distal fibula in eight patients and highlight the role of the periosteum in the regeneration of
bone defects. The mean age of the patients was 13.5 years (12 to 17). Seven had an open
growth plate. The mean size of the resected specimen was 5.12 cm (3.5 to 8.0). None of the
patients received instillation of bone marrow, autogenous bone graft, allograft or any
synthetic bone substitutes.
All had complete regeneration of the bone defect within three to nine months, with no
joint instability or recurrence.
The mean length of follow-up was 11.5 years (2 to 18). At the final follow-up there was no
difference in the range of movement, alignment or stability of the ankle when compared
with the opposite side. The periosteum played a major role in the complete filling of the
bone defects and avoided the morbidity of other techniques.
The distal fibula is an uncommon site for an
aneurysmal bone cyst. Total resection of the
lateral malleolus may produce a deformity and
compromise the stability of the ankle.1
The
main aims of treatment are to eradicate the cyst,
avoid local recurrence, prevent damage to the
growth plate, avoid subsequent valgus defor-
mity and maintain the stability of the ankle.2-5
Curettage and bone grafting have been the usual
methods of treatment4
for accessible cystic
lesions.6,7
Various methods have been proposed
for the reconstruction of the ankle after total
resection of the distal fibula.1,4,5,8
The complications of curettage and grafting
include a high rate of recurrence, injury to the
growth plate, limb-length discrepancy and
joint instability.9
Resection of the distal fibula
for benign bone lesions is an accepted practice,
but necessitates reconstruction of the distal
tibiofibular joint. It should be reserved for
aggressive or recurrent juxtaphyseal lesions,
since there is an increased morbidity following
multiple procedures, problems with implants
and prolonged immobilisation.4
We describe our experience of the treatment
by subperiosteal resection of an aneurysmal
bone cyst of the distal fibula in eight young
patients without a bone graft.
Patients and Methods
There were five girls and three boys with a
mean age of 13.5 years (12 to 17) who were
treated between 1988 and 2006. The lesions
were located on the right side in five and on the
left in three.
Local pain was the main symptom in all
the patients and was associated with unre-
lated previous injuries in three. Two had a
limp and a localised tender mass was
palpable in three. The mean duration of
symptoms before diagnosis was 4.9 months
(2 to 7). When first seen none of the patients
had abnormal alignment or limitation of
movement of the ankle. Seven had a visible
open growth plate on plain radiographs and
in one the growth plate was closed.
Anteroposterior and lateral plain radio-
graphs of the ankle were taken in all the
patients. None had MRI or CT. The clinical
and radiological details are given in Table I.
Radiological evaluation described the rela-
tionship of the cysts to the growth plate and
was classified according to the system of
Campanacci, Capanna and Picci,10
and
Capanna et al11
based on the anatomical loca-
tion. There were five types of lesion with
types 1 and 2 corresponding to central forms
and types 3 to 5 to peripheral forms. The
activity was graded by the system of
Capanna et al,11
which included three stages,
namely aggressive, active and inactive.
The diagnosis was made by the appearance
of the fluid drained from the cyst after pre-
operative aspiration, the typical radiological
1228 F. O. ABUHASSAN, A. O. SHANNAK
THE JOURNAL OF BONE AND JOINT SURGERY
findings of ballooning of the distal fibula and by histo-
logical examination of the resected lesion.
The mean follow-up was 11.5 years (2 to 18). All the
patients had the same surgical procedure of subperiosteal
resection without reconstruction. None received instilla-
tion of bone marrow, autogenous bone graft, an allograft
or any synthetic bone substitutes.
At follow-up clinical and radiological evaluation of the
extent of regeneration at the site of resected lesion, phy-
seal growth, alignment, the range of movement of both
ankles and recurrence was recorded. Conventional radi-
ography was carried out at four, eight and 12 weeks, at
six months and then annually.
Operative technique. Under general anaesthesia with a
tourniquet applied to the thigh, the cyst was removed in
one piece by subperiosteal resection involving all the corti-
ces without leaving a strut of normal bone.
A lateral longitudinal incision was made over the centre
of the lesion down to the periosteum. The periosteal sheath
was incised longitudinally and raised gently with a
periosteal elevator from the underlying cystic lesion and
marked by silk structures (Fig. 1). If the end of the cyst was
more than 0.5 cm from the growth plate, multiple 2.5 mm
diameter drill holes were made away from the growth plate
and the bone ends cut by a fine osteotome. The edge of the
cyst near the growth plate was removed by a bone nibbler
Table I. Clinical and radiological details of the eight patients
Case
Age
(yrs) Gender
Relation to growth
plate
Size of the resected
specimen (cm) Type of lesion
Follow-up
(yrs)
1 12 F Metaphyseal > 5 mm 7.5 Active 18
2 12 F Juxta-epiphyseal 3.5 Active 15
3 17 F Closed physis 3.5 Active 2
4 14 M Juxta-epiphyseal 6.0 Active 16
5 13 M Juxta-epiphyseal 8.0 Active 18
6 13 M Metaphyseal > 5 mm 3.5 Active 8
7 14 F Juxta-epiphyseal 4.6 Active 6
8 13 F Juxta-epiphyseal 5.0 Active 9
Fig. 1e
Case 1. A 12-year-old girl with an aneurysmal bone cyst of the distal fibula. (a) Anteroposterior and lateral plain radiographs showing an expansile
lytic lesion of the distal fibula less than 5 mm from the open growth plate. b) Intra-operative photograph showing the longitudinal incision in the thick
periosteum and c) complete peeling of the periosteal sheath with an osteotomy of the lesion proximally and distally and d) the residual defect after
complete removal of the lesion. e) Photograph of the resected specimen which measured 7.5 cm in length. f) Intra-operative photograph showing the
Kirschner wire in the distal part of the remaining fibula and the sutures after tight closure of the periosteal sheath.
Fig. 1a Fig. 1b Fig. 1c
Fig. 1f
Fig. 1d
SUBPERIOSTEAL RESECTION OF ANEURYSMAL BONE CYSTS OF THE DISTAL FIBULA 1229
VOL. 91-B, No. 9, SEPTEMBER 2009
and a fine curette, with careful preservation of the growth
plate. The subperiosteal shell of the bone with the cyst was
removed in one piece as far as its attachment to the margins
of the remaining normal bone without leaving any bony
fragments in the defect. The medullary canal proximal to
the remaining bone defect was opened using a 3 mm
Kirschner (K)-wire to allow free communication with the
defect. The thick periosteal sheath around the defect was
then sutured using 3/0 Vicryl to obtain a tight closure. The
remaining distal part of the fibula was fixed to the tibia by
a 1.5 mm K-wire in all cases in which there was an open
growth plate (Fig. 2a).
The skin was closed by subcuticular sutures, with no
wound drainage. The limb was immobilised in a plaster
cast with partial weight-bearing allowed. After two months
the cast and K-wires were removed and the patients were
allowed full movement and weight-bearing as tolerated.
They were able to participate in sports after six months.
Results
The mean size of the resected cysts was 5.2 cm (3.5 to 8.0).
Using the classification of Campanacci et al10
all the lesions
were type 2 and were active. At the final follow-up there
was no difference in the mean range of movement in the
ankle compared with that of the opposite side. No lesions
recurred and there was no injury to the growth plate or evi-
dence of joint instability. Shortening of 2 mm to 3 mm was
seen at the distal fibula in comparison with the normal side,
but this did not affect the range of movement or the stabil-
ity of the ankle.
Bone began to regenerate three weeks after resection fol-
lowing the course of the closed periosteal tube (Fig. 2b).
Complete healing of the defect occurred at a mean of
4.1 months (3 to 9). In one patient with a closed growth
plate bony consolidation was not achieved for nine months
but in those with an open growth plate this occurred at a
mean of 3.3 months (3 to 4).
None of the defects showed evidence of delayed or non-
union. One patient had delayed incorporation of the regen-
erate at the site of proximal resection after an electric saw
had been used to cut the bone ends. No patient had a frac-
ture of the regenerative bone.
At the final follow-up all patients had normal function of
the treated limb without discomfort and had resumed their
normal activities. Healing was obtained by filling and ossi-
fication of the defect.
Discussion
Aneurysmal bone cysts are benign, non-neoplastic, expansile,
osteolytic lesions of multifactorial aetiology, composed of
blood-filled spaces separated by connective tissue septa con-
taining fibroblasts, osteoclast-type giant cells and reactive
woven bone.12
Their precise pathogenesis is unclear, although
suggestions include a post-traumatic reaction to reactive vas-
cular malformation or a genetic predisposition.13,14
The incidence is 1.4 per 100 000 individuals, with 80%
of cases seen before the age of 20 years. The peak incidence
is in the second decade of life.15,16
They occur more com-
monly in the metaphysis of long bones, especially the tibia,
humerus and femur.6
The reported incidence of lesions in the distal fibula var-
ies between 7.1% and 16.4%.1,4,17
The management of an
aneurysmal bone cyst depends on the age of the patient, the
location, the size and the aggressiveness of the lesion.9
The
clinical and pathological behaviour in younger patients
does not seem to be more aggressive than that in older chil-
dren, with no statistically significant differences between
the age groups and the rate of recurrence.18,19
Since the origin and growth of aneurysmal bone cysts are
controversial, a variety of treatments has been described,
but a relatively high rate of local recurrence has been
recorded depending on the method used.7,9,13,19-22
The
most common treatments are curettage, resection, intra-
cystic injections and embolisation.13
Recurrence is more
common in active and aggressive lesions and occurs at a
mean of 7.6 months.11,18
This has been attributed to incom-
plete curettage of the primary soft-tissue component
because of the reluctance of the surgeon to perform a suffi-
ciently extensive procedure near an open growth plate,
especially in the leg.10,21,23
A mitotic index of seven or more
per 50 fields (× 750) is associated with a higher rate of
recurrence than a lower mitotic index.21
In aggressive lesions the rate of recurrence using curet-
tage with or without bone grafting varies between 18% and
59%.17-19,24
Adjunctive therapy such as liquid nitrogen,
phenol, the use of polymethylmethacrylate and irradiation
Fig. 2b
Case 1. Anteroposterior radiographs showing a) the Kirschner wire hold-
ing the lateral malleolus to the tibia with early faint regeneration of new
bone in the periosteal tube at three weeks after surgery and b) regenera-
tion two months after surgery before removal of the Kirschner wire.
Fig. 2a
1230 F. O. ABUHASSAN, A. O. SHANNAK
THE JOURNAL OF BONE AND JOINT SURGERY
has been used, but is associated with increased morbidity
and no apparent decrease in the rate of local recurrence
compared with curettage alone.9,23
Marginal en bloc resection may be adopted in an eccen-
tric lesion or in an expandable bone such as the fibula, rib,
pubic ramus, metacarpal or metatarsal. This procedure has
little morbidity and a minimal risk of recurrence.3,6,22,25,26
Resection of the lesion has the lowest association with
recurrence while excision through the margin has consis-
tently decreased the rate of local recurrence.10,27-29
Despite
the rate of recurrence in various methods of treatments,
more aggressive operative intervention does not appear to
be indicated and recurrence can be successfully treated by
repeated extended curettage and bone graft.19,30
It is important to preserve the growth plate, especially in
young children, even if the lesions are likely to recur.25
When growth of the lower fibular physis is arrested, a val-
gus deformity of the ankle may develop, the degree of
which depends on how many years of potential growth
remain.31
The risk of secondary valgus deformity is insig-
nificant between ten and 12 years.31
If curettage and bone
grafting have been performed carefully, the growth plate is
unlikely to be affected.2,24
In children with an open growth
plate, the main problems are at the level of the distal cut
with the risk of iatrogenic damage or incomplete resection
and the subsequent need for reconstruction.3
In younger
children, the lesion should be resected at the edge of the
growth plate.5
All our patients were treated meticulously by
resection of the cyst near the edge of the physis without any
residual damage.
Recurrence of distal fibular lesions after curettage and
bone grafting may occur for which resection is the only
successful method of treatment.8
An increased morbidity,
however, must be considered after such a procedure.9,18
The technique used in our study avoided the use of bone
graft from the iliac crest, allograft bone or synthetic bone
substitutes. Subperiosteal resection has been previously
described as a primary method of treatment of lesions of
the distal fibula, but there was no regeneration of the bone
defect.32
Autologous tibial graft has been suggested after
total subperiosteal resection of solitary bone cysts of the
humerus in order to bridge the defect and to decrease local
recurrence.33
No previous study has highlighted the effec-
tiveness of the periosteum alone in forming new bone after
resection of lesions of the distal fibula.27,32
We assume that
the inner layer of the periosteum has an osteoblastic capa-
bility which allows invasion of the haematoma in the
tightly sutured periosteal tube by osteoprogenitor cells.
Healing of the remaining defect occurred in a progressive
manner which suggests that osteogenesis occurred initially
at the margins of the cavity and moved toward its centre
over the following weeks. Progressive calcification and
ossification followed and the lesion was transformed into a
solid bony mass.
The location of the fibular lesion near the ankle may
cause instability, recurrence or growth arrest,32
but we did
not encounter these problems. Although age, the location
of the lesion, and its size have been suggested as risk factors
for local recurrence in aneurysmal bone cysts,9,22
the sub-
periosteal resection totally removed the pathological lesion.
The mean healing time after traditional surgical treatment
is 11.6 months (8 to 15) whereas after injection with
steroids or bone marrow it is 13.9 months (15 to 18).33
In
our patients the healing time was much shorter within a
mean of 4.12 months (3 to 9).
The main advantage of subperiosteal resection is the
complete regeneration of the bone defect and the absence of
local recurrence. This can be attributed to the presence of a
thick periosteum with substantial regenerative capacity.
Preservation of the integrity of the periosteum after sub-
periosteal resection constitutes a valuable matrix for bone
regeneration in children and young adolescents. It plays a
major role in the complete filling of the bone defects and
avoids the morbidity associated with other techniques.
No benefits in any form have been received or will be received from a commer-
cial party related directly or indirectly to the subject of this article.
References
1. Capanna R, van Horn JR, Biagini R, et al. Reconstruction after resection of the
distal fibula for bone tumor. Acta Orthop Scand 1986;57:290-4.
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young children: a multicenter study. J Pediatr Orthop B 2005;14:212-18.
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Subperiosteal resection of aneurysmal bone .pdf

  • 1. VOL. 91-B, No. 9, SEPTEMBER 2009 1227 Subperiosteal resection of aneurysmal bone cysts of the distal fibula F. O. Abuhassan, A. O. Shannak From Jordan University Hospital, School of Medicine, Queens Rania Street, Amman 11942, Jordan „ F . O. Abuhassan, FRCS, FRCS(Trauma & Orth), Professor „ A. O. Shannak, FRCS(G), FRCS(Orth), Professor Department of Orthopaedic Surgery Jordan University, School of Medicine, Queens Rania Street, Amman 11942, Jordan. Correspondence should be sent to Mr F . O. Abuhassan; e-mail: freih@ju.edu.jo ©2009 British Editorial Society of Bone and Joint Surgery doi:10.1302/0301-620X.91B9. 22395 $2.00 J Bone Joint Surg [Br] 2009;91-B:1227-31. Received 4 February 2009; Accepted after revision 27 May 2009 We describe the treatment by subperiosteal resection of an aneurysmal bone cyst in the distal fibula in eight patients and highlight the role of the periosteum in the regeneration of bone defects. The mean age of the patients was 13.5 years (12 to 17). Seven had an open growth plate. The mean size of the resected specimen was 5.12 cm (3.5 to 8.0). None of the patients received instillation of bone marrow, autogenous bone graft, allograft or any synthetic bone substitutes. All had complete regeneration of the bone defect within three to nine months, with no joint instability or recurrence. The mean length of follow-up was 11.5 years (2 to 18). At the final follow-up there was no difference in the range of movement, alignment or stability of the ankle when compared with the opposite side. The periosteum played a major role in the complete filling of the bone defects and avoided the morbidity of other techniques. The distal fibula is an uncommon site for an aneurysmal bone cyst. Total resection of the lateral malleolus may produce a deformity and compromise the stability of the ankle.1 The main aims of treatment are to eradicate the cyst, avoid local recurrence, prevent damage to the growth plate, avoid subsequent valgus defor- mity and maintain the stability of the ankle.2-5 Curettage and bone grafting have been the usual methods of treatment4 for accessible cystic lesions.6,7 Various methods have been proposed for the reconstruction of the ankle after total resection of the distal fibula.1,4,5,8 The complications of curettage and grafting include a high rate of recurrence, injury to the growth plate, limb-length discrepancy and joint instability.9 Resection of the distal fibula for benign bone lesions is an accepted practice, but necessitates reconstruction of the distal tibiofibular joint. It should be reserved for aggressive or recurrent juxtaphyseal lesions, since there is an increased morbidity following multiple procedures, problems with implants and prolonged immobilisation.4 We describe our experience of the treatment by subperiosteal resection of an aneurysmal bone cyst of the distal fibula in eight young patients without a bone graft. Patients and Methods There were five girls and three boys with a mean age of 13.5 years (12 to 17) who were treated between 1988 and 2006. The lesions were located on the right side in five and on the left in three. Local pain was the main symptom in all the patients and was associated with unre- lated previous injuries in three. Two had a limp and a localised tender mass was palpable in three. The mean duration of symptoms before diagnosis was 4.9 months (2 to 7). When first seen none of the patients had abnormal alignment or limitation of movement of the ankle. Seven had a visible open growth plate on plain radiographs and in one the growth plate was closed. Anteroposterior and lateral plain radio- graphs of the ankle were taken in all the patients. None had MRI or CT. The clinical and radiological details are given in Table I. Radiological evaluation described the rela- tionship of the cysts to the growth plate and was classified according to the system of Campanacci, Capanna and Picci,10 and Capanna et al11 based on the anatomical loca- tion. There were five types of lesion with types 1 and 2 corresponding to central forms and types 3 to 5 to peripheral forms. The activity was graded by the system of Capanna et al,11 which included three stages, namely aggressive, active and inactive. The diagnosis was made by the appearance of the fluid drained from the cyst after pre- operative aspiration, the typical radiological
  • 2. 1228 F. O. ABUHASSAN, A. O. SHANNAK THE JOURNAL OF BONE AND JOINT SURGERY findings of ballooning of the distal fibula and by histo- logical examination of the resected lesion. The mean follow-up was 11.5 years (2 to 18). All the patients had the same surgical procedure of subperiosteal resection without reconstruction. None received instilla- tion of bone marrow, autogenous bone graft, an allograft or any synthetic bone substitutes. At follow-up clinical and radiological evaluation of the extent of regeneration at the site of resected lesion, phy- seal growth, alignment, the range of movement of both ankles and recurrence was recorded. Conventional radi- ography was carried out at four, eight and 12 weeks, at six months and then annually. Operative technique. Under general anaesthesia with a tourniquet applied to the thigh, the cyst was removed in one piece by subperiosteal resection involving all the corti- ces without leaving a strut of normal bone. A lateral longitudinal incision was made over the centre of the lesion down to the periosteum. The periosteal sheath was incised longitudinally and raised gently with a periosteal elevator from the underlying cystic lesion and marked by silk structures (Fig. 1). If the end of the cyst was more than 0.5 cm from the growth plate, multiple 2.5 mm diameter drill holes were made away from the growth plate and the bone ends cut by a fine osteotome. The edge of the cyst near the growth plate was removed by a bone nibbler Table I. Clinical and radiological details of the eight patients Case Age (yrs) Gender Relation to growth plate Size of the resected specimen (cm) Type of lesion Follow-up (yrs) 1 12 F Metaphyseal > 5 mm 7.5 Active 18 2 12 F Juxta-epiphyseal 3.5 Active 15 3 17 F Closed physis 3.5 Active 2 4 14 M Juxta-epiphyseal 6.0 Active 16 5 13 M Juxta-epiphyseal 8.0 Active 18 6 13 M Metaphyseal > 5 mm 3.5 Active 8 7 14 F Juxta-epiphyseal 4.6 Active 6 8 13 F Juxta-epiphyseal 5.0 Active 9 Fig. 1e Case 1. A 12-year-old girl with an aneurysmal bone cyst of the distal fibula. (a) Anteroposterior and lateral plain radiographs showing an expansile lytic lesion of the distal fibula less than 5 mm from the open growth plate. b) Intra-operative photograph showing the longitudinal incision in the thick periosteum and c) complete peeling of the periosteal sheath with an osteotomy of the lesion proximally and distally and d) the residual defect after complete removal of the lesion. e) Photograph of the resected specimen which measured 7.5 cm in length. f) Intra-operative photograph showing the Kirschner wire in the distal part of the remaining fibula and the sutures after tight closure of the periosteal sheath. Fig. 1a Fig. 1b Fig. 1c Fig. 1f Fig. 1d
  • 3. SUBPERIOSTEAL RESECTION OF ANEURYSMAL BONE CYSTS OF THE DISTAL FIBULA 1229 VOL. 91-B, No. 9, SEPTEMBER 2009 and a fine curette, with careful preservation of the growth plate. The subperiosteal shell of the bone with the cyst was removed in one piece as far as its attachment to the margins of the remaining normal bone without leaving any bony fragments in the defect. The medullary canal proximal to the remaining bone defect was opened using a 3 mm Kirschner (K)-wire to allow free communication with the defect. The thick periosteal sheath around the defect was then sutured using 3/0 Vicryl to obtain a tight closure. The remaining distal part of the fibula was fixed to the tibia by a 1.5 mm K-wire in all cases in which there was an open growth plate (Fig. 2a). The skin was closed by subcuticular sutures, with no wound drainage. The limb was immobilised in a plaster cast with partial weight-bearing allowed. After two months the cast and K-wires were removed and the patients were allowed full movement and weight-bearing as tolerated. They were able to participate in sports after six months. Results The mean size of the resected cysts was 5.2 cm (3.5 to 8.0). Using the classification of Campanacci et al10 all the lesions were type 2 and were active. At the final follow-up there was no difference in the mean range of movement in the ankle compared with that of the opposite side. No lesions recurred and there was no injury to the growth plate or evi- dence of joint instability. Shortening of 2 mm to 3 mm was seen at the distal fibula in comparison with the normal side, but this did not affect the range of movement or the stabil- ity of the ankle. Bone began to regenerate three weeks after resection fol- lowing the course of the closed periosteal tube (Fig. 2b). Complete healing of the defect occurred at a mean of 4.1 months (3 to 9). In one patient with a closed growth plate bony consolidation was not achieved for nine months but in those with an open growth plate this occurred at a mean of 3.3 months (3 to 4). None of the defects showed evidence of delayed or non- union. One patient had delayed incorporation of the regen- erate at the site of proximal resection after an electric saw had been used to cut the bone ends. No patient had a frac- ture of the regenerative bone. At the final follow-up all patients had normal function of the treated limb without discomfort and had resumed their normal activities. Healing was obtained by filling and ossi- fication of the defect. Discussion Aneurysmal bone cysts are benign, non-neoplastic, expansile, osteolytic lesions of multifactorial aetiology, composed of blood-filled spaces separated by connective tissue septa con- taining fibroblasts, osteoclast-type giant cells and reactive woven bone.12 Their precise pathogenesis is unclear, although suggestions include a post-traumatic reaction to reactive vas- cular malformation or a genetic predisposition.13,14 The incidence is 1.4 per 100 000 individuals, with 80% of cases seen before the age of 20 years. The peak incidence is in the second decade of life.15,16 They occur more com- monly in the metaphysis of long bones, especially the tibia, humerus and femur.6 The reported incidence of lesions in the distal fibula var- ies between 7.1% and 16.4%.1,4,17 The management of an aneurysmal bone cyst depends on the age of the patient, the location, the size and the aggressiveness of the lesion.9 The clinical and pathological behaviour in younger patients does not seem to be more aggressive than that in older chil- dren, with no statistically significant differences between the age groups and the rate of recurrence.18,19 Since the origin and growth of aneurysmal bone cysts are controversial, a variety of treatments has been described, but a relatively high rate of local recurrence has been recorded depending on the method used.7,9,13,19-22 The most common treatments are curettage, resection, intra- cystic injections and embolisation.13 Recurrence is more common in active and aggressive lesions and occurs at a mean of 7.6 months.11,18 This has been attributed to incom- plete curettage of the primary soft-tissue component because of the reluctance of the surgeon to perform a suffi- ciently extensive procedure near an open growth plate, especially in the leg.10,21,23 A mitotic index of seven or more per 50 fields (× 750) is associated with a higher rate of recurrence than a lower mitotic index.21 In aggressive lesions the rate of recurrence using curet- tage with or without bone grafting varies between 18% and 59%.17-19,24 Adjunctive therapy such as liquid nitrogen, phenol, the use of polymethylmethacrylate and irradiation Fig. 2b Case 1. Anteroposterior radiographs showing a) the Kirschner wire hold- ing the lateral malleolus to the tibia with early faint regeneration of new bone in the periosteal tube at three weeks after surgery and b) regenera- tion two months after surgery before removal of the Kirschner wire. Fig. 2a
  • 4. 1230 F. O. ABUHASSAN, A. O. SHANNAK THE JOURNAL OF BONE AND JOINT SURGERY has been used, but is associated with increased morbidity and no apparent decrease in the rate of local recurrence compared with curettage alone.9,23 Marginal en bloc resection may be adopted in an eccen- tric lesion or in an expandable bone such as the fibula, rib, pubic ramus, metacarpal or metatarsal. This procedure has little morbidity and a minimal risk of recurrence.3,6,22,25,26 Resection of the lesion has the lowest association with recurrence while excision through the margin has consis- tently decreased the rate of local recurrence.10,27-29 Despite the rate of recurrence in various methods of treatments, more aggressive operative intervention does not appear to be indicated and recurrence can be successfully treated by repeated extended curettage and bone graft.19,30 It is important to preserve the growth plate, especially in young children, even if the lesions are likely to recur.25 When growth of the lower fibular physis is arrested, a val- gus deformity of the ankle may develop, the degree of which depends on how many years of potential growth remain.31 The risk of secondary valgus deformity is insig- nificant between ten and 12 years.31 If curettage and bone grafting have been performed carefully, the growth plate is unlikely to be affected.2,24 In children with an open growth plate, the main problems are at the level of the distal cut with the risk of iatrogenic damage or incomplete resection and the subsequent need for reconstruction.3 In younger children, the lesion should be resected at the edge of the growth plate.5 All our patients were treated meticulously by resection of the cyst near the edge of the physis without any residual damage. Recurrence of distal fibular lesions after curettage and bone grafting may occur for which resection is the only successful method of treatment.8 An increased morbidity, however, must be considered after such a procedure.9,18 The technique used in our study avoided the use of bone graft from the iliac crest, allograft bone or synthetic bone substitutes. Subperiosteal resection has been previously described as a primary method of treatment of lesions of the distal fibula, but there was no regeneration of the bone defect.32 Autologous tibial graft has been suggested after total subperiosteal resection of solitary bone cysts of the humerus in order to bridge the defect and to decrease local recurrence.33 No previous study has highlighted the effec- tiveness of the periosteum alone in forming new bone after resection of lesions of the distal fibula.27,32 We assume that the inner layer of the periosteum has an osteoblastic capa- bility which allows invasion of the haematoma in the tightly sutured periosteal tube by osteoprogenitor cells. Healing of the remaining defect occurred in a progressive manner which suggests that osteogenesis occurred initially at the margins of the cavity and moved toward its centre over the following weeks. Progressive calcification and ossification followed and the lesion was transformed into a solid bony mass. The location of the fibular lesion near the ankle may cause instability, recurrence or growth arrest,32 but we did not encounter these problems. Although age, the location of the lesion, and its size have been suggested as risk factors for local recurrence in aneurysmal bone cysts,9,22 the sub- periosteal resection totally removed the pathological lesion. The mean healing time after traditional surgical treatment is 11.6 months (8 to 15) whereas after injection with steroids or bone marrow it is 13.9 months (15 to 18).33 In our patients the healing time was much shorter within a mean of 4.12 months (3 to 9). The main advantage of subperiosteal resection is the complete regeneration of the bone defect and the absence of local recurrence. This can be attributed to the presence of a thick periosteum with substantial regenerative capacity. Preservation of the integrity of the periosteum after sub- periosteal resection constitutes a valuable matrix for bone regeneration in children and young adolescents. It plays a major role in the complete filling of the bone defects and avoids the morbidity associated with other techniques. No benefits in any form have been received or will be received from a commer- cial party related directly or indirectly to the subject of this article. References 1. Capanna R, van Horn JR, Biagini R, et al. Reconstruction after resection of the distal fibula for bone tumor. Acta Orthop Scand 1986;57:290-4. 2. Rizzo M, Dellaero DT, Harrelson JM, Scully SP. Juxtaphyseal aneurysmal bone cysts. Clin Orthop 1993;64:205-12. 3. Rizzo M, Scully SP, Harrelson JM. Ipsilateral fibular slide grafts in the manage- ment of distal fibula lesions. Foot Ankle Int 1999;20:135-6. 4. Lampasi M, Magnani M, Donzelli O. Aneurysmal bone cysts of the distal fibula in children: long-term results of curettage and resection in nine patients. J Bone Joint Surg [Br] 2007;89-B:1356-62. 5. Ramirez N, Perez C, Rivera YC. Distal third fibular aneurysmal bone cyst: en bloc resection and proximal third fibular reconstruction. Am J Orthop 2001;30:237-40. 6. Ramirez AR, Stanton RP. Aneurysmal bone cyst in 29 children. J Pediatr Orthop 2002;22:533-9. 7. Marcove RC, Sheth DS, Takemoto S, Healey JH. The treatment of aneurysmal bone cyst. Clin Orthop 1995;311:157-63. 8. Lubliner JA, Robbins H, Lewis MM, Present D. Aneurysmal bone cyst of the fib- ula: en bloc resection with allograft reconstruction. Bull Hosp Jt Dis Orthop Inst 1985;45:80-6. 9. Cottalorda J, Bourelle S. Current treatments of primary aneurysmal bone cysts. J Pediatr Orthop B 2006;15:155-67. 10. Campanacci M, Capanna R, Picci P. Unicameral and aneurysmal bone cysts. Clin Orthop 1986;204:25-36. 11. Capanna R, Betteli G, Biaginia R, et al. Aneurysmal bone cysts of long bones. Ital J Orthop Traumatol 1985;11:409-17 (in Italian). 12. Rosenberg AE, Nielsen GP, Fletcher JA. Aneurysmal bone cyst. In: Fletcher CDM, Unni KK, Mertens F, eds. World Health Organization: classification of tumours: pathology and genetics of tumours of soft tissue and bone. Lyon: IARC Press, 2002:338-9. 13. Cottalorda J, Bourelle S. Modern concepts of primary aneurysmal bone cyst. Arch Orthop Trauma Surg 2007;127:105-14. 14. Althof PA, Ohmori K, Zhou M, et al. Cytogenetic and molecular cytogenetic find- ings in 43 aneurysmal bone cysts: aberrations of 17p mapped to 17p13.2 by fluores- ence in situ hybridization. Mod Pathol 2004;17:518-25. 15. Ozaki T, Hillmann A, Linder N. Cementation of primary aneurysmal bone cysts. Clin Orthop 1997;337:240-8. 16. Leithner A, Windhager R, Lang S, et al. Aneurysmal bone cyst: a population based epidemiologic study and literature review. Clin Orthop 1999;363:176-9. 17. 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