Neuroscience & Medicine, 2013, 4, 280-283
Published Online December 2013 (http://www.scirp.org/journal/nm)
http://dx.doi.org/10.4236/nm.2013.44041
Open Access NM
Secondary Rec onstruction of Recurrent Primary
Intraosseous Meningioma of the Calvarium Using a
Fasciocutaneous Anterolateral Thigh Free Flap
Raakhi Mistry1, Chris Tsimiklis2, Marcus Wagstaff1, Amal Abou-Hamden2, Yugesh Caplash1
1Department of Plastic & Reconstructive Surgery, Royal Adelaide Hospital, Adelaide, South Australia; 2Department of Neurosurgery,
Royal Adelaide Hospital, Adelaide, South Australia.
Email: raakhimistry@gmail.com
Received October 20th, 2013; revised November 15th, 2013; accepted December 5th, 2013
Copyright © 2013 Raakhi Mistry et al. This is an open access article distributed under the Creative Commons Attribution License,
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
ABSTRACT
We present a patient with a primary intraosseous meningioma of the calvarium that recurred three years after initial
resection and reconstruction with a free anterolateral thigh (ALT) flap. The patient re-presented with progressive swell-
ing in the same area. Imaging confirmed aggressive tumour recurrence with both intra- and extra-cranial diseases and
also extension to the ipsilateral orbit. The original anterolateral free flap was raised on anterior pedicle and further de-
bulking of the tumour was performed. Histology confirmed widely infiltrative atypical meningioma (WHO grade II).
This case highlights the benefit of using a fasciocutaneous free flap for reconstruction given the potential for tumour
recurrence.
Keywords: Primary Intraosseous Meningioma; Fasciocutaneous Free Flap
1. Introduction
A 76-year-old man was initially presented with a large
mass involving his right frontal scalp which had been
growing over a period of five years (Figure 1). The mass
was firm and non-tender. He had no lymphadenopathy.
He had no significant comorbidities, however, he was re-
ported to have sustained a traumatic injury to his head
when he was a teenager. CT scan showed an exophytic
lesion measuring 9 × 5 × 10 cm arising from the right
frontal bone. The lesion appeared to be composed predo-
minantly of soft tissue with a sunburst pattern of ossifica-
tion centrally. There was hyperostosis of the frontal bone
which extended to involve the roof and lateral wall of the
ipsilateral orbit. Post-contrast imaging revealed avid and
fairly uniform enhancement. On MRI, dural thickening
and enhancement were seen suggestive of dural involve-
ment but there was no convincing evidence of intra-axial
disease (Figure 2). Initial differential diagnosis based on
radiological features was a malignant process such as
osteosarcoma. Staging CT showed no evidence of dis-
ease elsewhere.
Pre-operative embolization was performed given the
highly vascular nature of the tumour, and the patient
subsequently underwent primary resection. The tumour
was resected down to the frontal bone and the underlying
skull was burred until normal calvarial bone was en-
countered macroscopically. The temporalis muscle also
appeared to be grossly involved and thus was partially
excised (Figure 3). A free anterolateral thigh flap was
raised to reconstruct the soft tissue defect.
Histology demonstrated a WHO grade I meningioma
Figure 1. Pre-operative photograph showing the mass on his
right frontal scalp on initial presentation.
Secondary Reconstruction of Recurrent Primary Intraosseous Meningioma of the
Calvarium Using a Fasciocutaneous Anterolateral Thigh Free Flap
281
Figure 2. Pre-operative T1 post-Gadolinium coronal (up)
and axial (down) slices demonstrating the extent of the tu-
mour and its avid enhancement.
Figure 3. Intra-operative photograph following initial tu-
mour resection.
with some features of atypia. Tumour was present at all of
the surgical margins, but the patient elected for surveillance
with serial MRI scans rather than for adjuvant therapy.
Three years after his initial surgery, the patient devel-
oped swelling under the free flap as well as progressive
ptosis of the right upper eyelid. Repeat MRI showed
prominent recurrent disease with an intra-cranial com-
ponent, and extension through the calvarium and diploic
space and with a larger extra-cranial soft tissue compo-
nent extending to involve the superior aspect of the right
orbit (Figure 4). Following multidisciplinary discussion,
it was felt that the combined treatment with radical sur-
gery and post-operative radiotherapy would improve his
quality of life.
Repeat surgery involved carefully raising the flap
based on an anterior pedicle to preserve the previously
anastomosed vessels. The tumour, which was firmly ad-
herent to the underlying bone and surrounding soft tissue,
was debulked. Further resection of the remnant tempo-
ralis muscle was necessary. A skull flap was raised and
the diseased bone was removed en bloc. The intracranial
component of tumour was then resected noting areas of
localised pial invasion. The skull defect was recon-
structed with titanium mesh. Finally, the orbital roof was
raised and the intraorbital component of disease was de-
bulked. This was then reconstructed with titanium plates
and screws. The ALT flap was then re-inset. Histology
demonstrated an extensively infiltrating atypical men-
ingioma (WHO grade II). The patient made a good post-
operative recovery and was subsequently referred to ad-
juvant radiotherapy (Figure 5).
Figure 4. T1 post-gadolinium coronal (up) and axial (down)
MRI slices three years after initial resection showing tu-
mour recurrence with involvement of the ipsilateral orbit
and extension through the dura.
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Secondary Reconstruction of Recurrent Primary Intraosseous Meningioma of the
Calvarium Using a Fasciocutaneous Anterolateral Thigh Free Flap
282
Figure 5. Photograph six weeks post resection of recurrent
tumour.
2. Discussion
Primary intraosseous meningioma of the calvarium is a
subtype of primary extradural meningioma (PEM). In
this case, we have demonstrated the potentially aggres-
sive nature of this tumour and possibility for early recur-
rence.
Meningiomas that arise from locations other than the
meninges are known as PEM’s [1]. They are a rare type
of meningioma, comprising 1% - 2% of all meningiomas
[2]. A number of extradural sites have been reported in
the literature, however the most common is within the
calvaria [3]. Several classification systems of PEM’s
have been described in the literature. Lang et al. recently
undertook a retrospective review of PEM’s and proposed
a classification scheme based on the tumour’s relation-
ship to the cranium [3]. Using the schema demonstrated
in Table 1 our patient initially had a type IIIC primary
intraosseous meningioma.
The aetiology of PEMs is not completely understood
but there is evidence to support their development from
arachnoid cap cells via several potential mechanisms,
including displacement during embryologic development
or secondary to a traumatic event [4]. Interestingly, our
patient reported having had trauma to the site as a teen-
ager.
In the data reviewed by Lang et al. consisting of a co-
hort of 142 patients with PEMs, the majority (67%) of
tumours were histologically benign [3,5]. Although
fewer PEMs were malignant or atypical (33%), their in-
cidence was higher than observed for intracranial men-
ingiomas. Furthermore recurrence was more likely to
occur in type IIB and IIIB tumours compared with type
IIC or type IIIC. This may reflect the difficulty in
achieving complete macroscopic resection in such cases.
The most important factor in prevention of recurrence is
the extent of macroscopic resection and the use of the
Simpson grading system is commonplace (Table 2) [5].
In our case, a Simpson grade III resection was initially
achieved as baseline MRI indicated underlying dural
Table 1. Classification of PEM’s by Lang et al. [3].
Type Tumour relationship to the cranium
I Purely extracalvarial with no attachment to bone
II Located entirely within the calvarial bone
III Calvarial tumours with extracalvarial extension
*Type II and III tumours are then further subdivided as being skull based (B)
or convexity (C) lesions depending on their anatomical location.
Table 2. Simpson grading system for surgical excision of
meningiomas [5].
GradeDegree of removal
I
Macroscopically complete removal with excision of dural
attachment and abnormal bone
(including sinus resection when involved)
II Macroscopically complete with endothermy coagulation o
f
dural attachment
III
Macroscopically complete without resection or coagulation
of dural attachment or of its extradural extensions
(eg. hyperostotic bone)
IV Partial removal leaving tumour in situ
V Simple decompression (+/ biopsy)
enhancement and thus likely disease involvement.
Other important factors in tumour recurrence are the
histological subtype and whether or not adjuvant therapy
was undertaken. In one particular study by Barbaro et al.,
there appeared to be benefit in radiotherapy following
partial resection in a series of patients with non-malig-
nant meningiomas [6]. In our case, adjuvant radiotherapy
was not undertaken initially given the patient’s prefer-
ence for surveillance.
The ALT free flap is a popular workhorse for recon-
struction of head and neck defects. It is based on the lat-
eral circumflex femoral artery and can be raised as a cu-
taneous, fasciocutaneous or musculocutaneous flap. The
advantages of this flap include no need for change in
patient position, simultaneous flap harvest, relatively
straightforward harvest, adequate pedicle length and
calibre and negligible donor site morbidity [7,8]. In this
case, a fasciocutaneous ALT free flap was raised and
microvascular anastomoses were performed end-to-end
to the right superficial temporal vessels. The advantage
of using this flap was the ability to raise it on a pedicle
and perform further tumour resection when it recurred.
Because the flap had well-established vascularity from
the wound edges we were able to raise it on an anterior
pedicle without threatening its viability. This meant that
the patient did not require further soft tissue reconstruc-
tion, resulting in faster recovery and sooner commence-
ment of radiotherapy.
Open Access NM
Secondary Reconstruction of Recurrent Primary Intraosseous Meningioma of the
Calvarium Using a Fasciocutaneous Anterolateral Thigh Free Flap
Open Access NM
283
Over the past two decades primary microsurgical re-
construction has become the treatment of choice for head
and neck tumours. We are now seeing more patients with
recurrent tumours who were initially treated with surgical
resection and free flap reconstruction. While the aims of
primary reconstruction following tumour resection are to
achieve functional and aesthetically acceptable results, it
is also important to consider the risk of tumour recur-
rence when choosing a flap for reconstruction.
3. Conclusion
In this case, we have demonstrated a patient with aggres-
sive recurrence of a primary intraosseous meningioma of
the calvarium. Although rare, there are increasing reports
of such tumours in the literature and they can represent a
major challenge in achieving complete resection. When
this patient developed tumour recurrence, we were able
to raise the original fasciocutaneous flap on a pedicle and
perform further resection without threatening the flap’s
viability.
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