CASE
REPORT
–
OPEN
ACCESS
International
Journal
of
Surgery
Case
Reports
3 (2012) 407–
411
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at
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International
Journal
of
Surgery
Case
Reports
j
ourna
l
ho
me
page:
www.elsevier.com/locate/ijscr
Intraneural
lipomatous
tumor
of
the
median
nerve:
Three
case
reports
with
a
review
of
literature
Taketo
Okubo
a,b,∗
, Tsuyoshi
Saito
b
, Hiroyuki
Mitomi
b
, Tatsuya
Takagi
a
, Tomoaki
Torigoe
a
,
Yoshiyuki
Suehara
a
,
Hirohisa
Katagiri
c
,
Hideki
Murata
c
,
Mitsuru
Takahashi
c
,
Ichiro
Ito
d
,
Takashi
Yao
b
,
Kazuo
Kaneko
a
a
Department
of
Orthopaedic
Surgery,
Juntendo
University
School
of
Medicine,
Tokyo,
Japan
b
Department
of
Human
Pathology,
Juntendo
University
School
of
Medicine,
Tokyo,
Japan
c
Department
of
Orthopaedic
Surgery,
Shizuoka
Cancer
Center,
Shizuoka,
Japan
d
Department
of
Diagnostic
Pathology,
Shizuoka
Cancer
Center,
Shizuoka,
Japan
a
r
t
i
c
l
e
i
n
f
o
Article
history:
Received
15
April
2012
Received
in
revised
form
11
May
2012
Accepted
16
May
2012
Available online 23 May 2012
Keywords:
Intraneural
lipoma
Fibrolipomatous
hamartoma
of
the
nerve
a
b
s
t
r
a
c
t
INTRODUCTION:
Intraneural
lipoma
and
fibrolipomatous
hamartoma
of
the
nerve
are
rare
soft
tissue
tumors
that
most
commonly
occur
in
the
forearm
and
the
wrist,
and
particularly
within
the
median
nerve.
When
the
lesions
are
large
enough,
they
may
cause
progressive
compression
neuropathy.
They
are
distinct
entities
each
other
with
different
clinical
and
radiological
findings
and
thereby
need
different
surgical
treatments.
PRESENTATION
OF
CASE:
We
report
here
3
cases
of
intraneural
lipomatous
tumors
of
the
median
nerve
(1
case
of
intraneural
lipoma
and
2
cases
of
fibrolipomatous
hamartoma).
DISCUSSION:
All
patients
were
surgically
treated
successfully
with
complete
excision
for
intraneural
lipoma
and
with
carpal
tunnel
releases
for
the
both
fibrolipomatous
hamartomas.
CONCLUSION:
A
careful
preoperative
planning
is
necessary
for
the
optimal
treatment
by
distinguishing
whether
it
is
a
resectable
or
non-resectable
tumor
based
on
the
clinical
and
radiological
findings,
because
they
have
characteristic
findings
each
other.
© 2012 Surgical Associates Ltd. Published by Elsevier Ltd. All rights reserved.
1.
Introduction
Intraneural
lipoma
and
fibrolipomatous
hamartoma
of
the
nerve
are
rare
soft
tissue
tumors
that
most
commonly
occur
in
the
fore-
arm
and
the
wrist,
and
particularly
within
the
median
nerve.
When
the
lesions
are
large
enough,
they
may
cause
progres-
sive
compression
neuropathy.
The
term
intraneural
lipoma
means
encapsulated
lipoma,
which
can
be
shelled
out
or
enucleated
from
the
surrounding
structures,
and
microscopic
examination
reveals
that
these
lesions
are
composed
of
adipose
tissue
without
neu-
ral
elements.
On
the
other
hand,
various
terms
have
been
used
to
describe
fibrolipomatous
hamartoma
of
the
nerve,
including
fatty
infiltration,
lipofibroma,
fibrofatty
proliferation,
and
intraneural
lipofibroma.
1
It
is
pathologically
composed
of
a
fatty
and
fibrous
tissue
with
intermingling
nerve
fibers.
Significant
differences
do
exist
between
intraneural
lipoma
and
fibrolipomatous
hamartoma
of
the
nerve.
2
Intraneural
lipomas
are
usually
well
encapsulated
with
nerve
fibers
that
run
on
the
outer
surface
of
the
mass;
thus,
complete
excision
without
damage
to
∗
Corresponding
author
at:
Department
of
Human
Pathology,
Juntendo
University
School
of
Medicine,
Hongo
2-1-1,
Bunkyo-ku,
Tokyo
113-8421,
Japan.
Tel.:
+81
3
3813
3111;
fax:
+81
3
3813
3428.
E-mail
address:
(T.
Okubo).
the
adjoining
nerve
is
possible.
On
the
other
hand,
fibrolipomatous
hamartoma
of
the
nerve
is
composed
of
fibrous
tissues,
fatty
tis-
sues,
and
normal
nerve
fibers,
making
complete
excision
without
nerve
damage
difficult.
Although
the
World
Health
Organization
(WHO)
grouped
intraneural
lipoma,
fibrolipomatous
hamartoma
of
the
nerve,
fatty
infiltration,
and
neural
fibrolipoma
under
lipo-
matosis
of
the
nerve
in
2002,
3
the
clinical
findings
and
treatments
of
these
conditions
differ.
We
report
3
cases
of
intraneural
lipo-
matous
tumor:
1
intraneural
lipoma
of
the
median
nerve
and
2
neural
fibrolipomatous
hamartoma
of
the
median
nerve,
all
of
which
caused
carpal
tunnel
syndrome.
We
present
these
3
cases
with
special
emphasis
on
the
clinical
and
radiological
findings
and
surgical
treatments.
2.
Presentation
of
cases
2.1.
Case
1
A
56-year-old
woman
presented
with
progressive
swelling
of
the
right
forearm
over
a
6-month
period.
Physical
examination
revealed
a
10
× 8-cm
soft
elastic
mass
at
the
palmar
aspect
of
the
right
forearm.
There
was
no
history
of
trauma.
She
did
not
have
any
motor
symptoms,
hypoesthesia,
or
pain.
There
was
no
tenderness
on
palpation
and
no
Tinel-like
sign.
Magnetic
resonance
imaging
2210-2612/$
–
see
front
matter ©
2012 Surgical Associates Ltd. Published by Elsevier Ltd. All rights reserved.
http://dx.doi.org/10.1016/j.ijscr.2012.05.007
CASE
REPORT
–
OPEN
ACCESS
408 T.
Okubo
et
al.
/
International
Journal
of
Surgery
Case
Reports
3 (2012) 407–
411
Fig.
1.
MRI,
macro-
and
microscopic
findings
in
Case
1.
The
mass
shows
high
signal
intensities
on
T1
(A)-
and
T2
(B)-weighted
images,
and
the
intensities
changes
to
low
on
short
tau
inversion
recovery
(STIR)
images
(C).
The
sagittal
image
also
shows
high-intensity
mass
(D).
Well-encapsulated
lipoma
by
which
the
median
nerve
fibers
were
spread
(E).
The
resected
specimen
shows
a
proliferation
of
mature
adipocytes
without
nerve
fibers
(F).
(MRI)
demonstrated
the
presence
of
a
well-circumscribed
soft
tis-
sue
mass
measuring
4
cm
on
the
radial
side
of
the
right
forearm.
On
T1-
and
T2-weighted
images,
the
signal
intensities
of
the
mass
were
found
to
be
markedly
high,
and
the
mass
intensities
on
short
tau
inversion
recovery
(STIR)
images
changed
to
low
(Fig.
1A–D).
We
performed
an
open
biopsy
and
made
a
pathological
diagnosis
of
lipoma.
At
the
time
of
the
biopsy,
the
patient
felt
some
numb-
ness
and
pain
at
the
median
nerve
area.
Exploration
revealed
a
well-encapsulated
lipomatous
tumor
spreading
the
fibers
of
the
easily
identifiable
median
nerve
(Fig.
1E).
Complete
excision
of
the
tumor
was
comparatively
easy.
Histological
examination
revealed
a
lipomatous
tumor
composed
of
mature
adipocytes
without
inter-
mingling
nerve
fibers
(Fig.
1F).
At
6
months
after
the
excision,
the
numbness
and
pain
had
disappeared.
We
diagnosed
this
patient
as
intraneural
lipoma
based
on
the
clinical
and
pathological
findings.
2.2.
Case
2
A
17-year-old
boy
had
a
mass
on
his
right
hand
since
child-
hood,
and
he
had
received
no
treatment
for
it.
He
had
felt
continual
numbness
on
the
volar
side
of
his
right
hand
for
the
past
year.
The
numbness
had
been
gradually
getting
worse;
therefore,
he
had
vis-
ited
our
hospital.
He
had
no
history
of
trauma.
Physical
examination
revealed
a
4
×
2-cm
elastic
soft
mass
at
the
volar
aspect
of
the
right
distal
forearm
and
hand.
On
palpation,
decreased
perception
was
noted
in
the
middle
finger
in
addition
to
disordered
thumb
opposi-
tion
movement.
Digital
enlargement
was
not
evident
in
this
patient.
On
MRI,
the
mass
measured
4
cm
on
the
center
of
the
hand.
On
T1-
and
T2-weighted
images,
the
mass
had
an
alveolar
structure
in
which
thickened
nerve
bundles
appeared
as
hypointensive
tubular
structures
(Fig.
2A–C).
On
sagittal
view,
bowing
of
the
flexor
reti-
naculum
and
displacement
of
the
flexor
tendons
were
observed
in
the
carpal
tunnel.
On
exploration,
there
was
a
large
lipomatous
tumor
under
the
flexor
retinaculum
that
was
enlarging
the
median
nerve
(Fig.
2D).
We
judged
total
excision
of
the
tumor
impossible,
and
thus,
performed
carpal
tunnel
release
and
a
biopsy.
Histologi-
cal
analysis
showed
a
mixture
of
collagenous
fiber
and
fatty
tissue
(Fig.
2E).
Numbness
was
improved
and
the
patient’s
condition
had
not
relapsed
at
the
time
of
6-months
follow-up.
Although
the
inter-
mingled
nerve
fascicles
were
not
observed
in
the
biopsy
sample,
CASE
REPORT
–
OPEN
ACCESS
T.
Okubo
et
al.
/
International
Journal
of
Surgery
Case
Reports
3 (2012) 407–
411 409
Fig.
2.
MRI,
macro-
and
microscopic
findings
in
Case
2.
The
mass
showed
alveolar
structures
on
both
T1
(A)-
and
T2
(B)-weighted
images.
The
“spaghetti-like”
appearance
in
sagittal
section
(C).
Large
lipomatous
lesions
under
the
flexor
retinaculum
look
like
hypertrophic
nerve
(D).
Biopsy
specimen
shows
a
mixture
of
collagen
fibers
and
fatty
tissue
without
intermingling
nerve
fibers
(E).
we
diagnosed
this
patient
with
fibrolipomatous
hamartoma
of
the
median
nerve.
2.3.
Case
3
A
15-year-old
boy
felt
numbness
in
the
middle
finger
of
his
right
hand
while
playing
baseball.
After
6
months,
he
visited
our
hospital
because
he
noticed
a
mass
on
his
right
hand.
On
exami-
nation,
diffuse
swelling
around
the
muscles
and
slight
tenderness
was
observed.
On
palpation,
there
was
no
decreased
perception
or
finger
movement.
MRI
showed
a
3-cm
mass
at
the
center
of
the
hand,
and
the
other
findings
were
similar
to
those
of
Case
2
(Fig.
3A–D).
On
coronal
view,
tubular
structures
were
seen
in
the
carpal
tunnel.
Because
his
chief
complaint
was
numbness,
he
had
been
under
observation
for
3
months;
however,
because
his
symp-
toms
had
been
getting
worse,
we
performed
a
carpal
tunnel
release
and
biopsy
for
diagnosis.
Operative
and
histological
findings
resem-
bled
those
of
Case
2
(Fig.
3E
and
F).
Numbness
was
improved
and
the
patient’s
symptoms
had
not
relapsed
at
time
of
4-years
follow-
up.
We
diagnosed
this
patient
with
fibrolipomatous
hamartoma
of
the
median
nerve.
3.
Discussion
Lipomatous
tumors
arising
in
the
peripheral
nerves
are
rare.
Morley
reported
the
first
case
of
intraneural
lipoma
in
1964.
4
The
tumors
that
could
be
separated
from
the
neural
elements
and
showed
the
pathological
characteristics
of
a
benign
lipoma
without
intermingling
with
neural
elements
were
termed
as
“true
intraneu-
ral
lipoma”
by
Rusko
and
Larson.
5
To
our
knowledge,
not
so
many
cases
have
been
reported
in
the
English
literature
as
intraneural
lipomas.
2,4–9
The
intraneural
lipoma
tends
to
occur
in
the
fourth
and
fifth
decades
and
has
a
female
predominance.
Most
of
the
intraneural
lipoma
occurs
in
the
median
nerve,
while
also
reported
to
occur
in
the
radial
nerve,
2
sciatic
nerve,
7
ulnar
nerve,
8
and
posterior
interosseous
nerve.
9
The
maximum
size
of
the
intraneural
lipoma
was
reported
to
be
200
×
100
mm
9
and
the
minimum
to
be
25
×
40
mm.
4
A
history
of
trauma
was
reported
in
only
1
case.
5
Pathological
findings
of
lipomas
include
proliferation
of
mature
adipocytes
without
inter-
mingled
nerve
fibers.
The
patient
in
Case
1
of
this
manuscript
is
older
as
one
with
fibrolipomatous
hamartoma
which
arises
in
a
younger
age
group.
Therefore,
clinical
information
is
also
important
CASE
REPORT
–
OPEN
ACCESS
410 T.
Okubo
et
al.
/
International
Journal
of
Surgery
Case
Reports
3 (2012) 407–
411
Fig.
3.
MRI,
macro-
and
microscopic
findings
in
Case
3.
The
mass
showed
alveolar
structures
on
both
T1
(A)-
and
T2
(B)-weighted
images.
The
“coaxial
cable-like”
appearance
in
cross-section
(C)
and
“spaghetti-like”
appearance
in
longitudinal
section
(D),
respectively.
Operative
and
histological
findings
in
Case
3
(E
and
F)
resembled
those
of
Case
2.
The
feature
was
not
enough
informative
for
the
diagnosis
of
fibrolipomatous
hamartoma.
for
the
diagnosis,
because
even
the
biopsy
sample
cannot
always
be
obtained
from
the
appropriate
lesion
for
a
diagnosis
where
the
involved
nerve
fibers
exist
if
it
is
an
infiltrative
fibrolipomatous
hamartoma.
On
the
other
hand,
fibrolipomatous
hamartoma
was
first
described
in
1953
by
Mason.
10
Various
terms
describe
fibrolipo-
matous
hamartoma
of
the
nerve
such
as
fatty
infiltration,
fibrofatty
proliferation,
intraneural
fibrolipoma,
lipofibroma,
and
fibrolipo-
matosis.
The
pathology
of
fibrolipomatous
hamartoma
of
the
nerve
usually
shows
a
mixture
of
collagenous
fibers
and
fatty
tissue,
and
nerve
fibers
are
noticed
in
cases
of
partial
resection.
The
etiol-
ogy
of
fibrolipomatous
hamartoma
of
the
nerve
remains
unclear,
however,
several
potential
etiologic
factors
have
been
described
such
as
abnormal
development
of
flexor
retinaculum
in
children,
11
history
of
trauma,
12
and
chronic
nerve
inflammation.
13
It
has
been
reported
that
fibrolipomatous
hamartoma
tend
to
arise
in
younger
age
groups
before
the
third
decade
and
have
no
sexual
predominance.
2
Two
patients
(Cases
2
and
3)
in
this
manuscript
had
suffered
a
mass
on
his
hand
since
childhood.
However,
the
digital
enlargement
was
not
evident
in
both
cases,
although
it
has
been
reported
that
patients
with
fibrolipomatous
hamartoma
often
had
digital
enlargement
due
to
an
increase
in
perineural
soft
tissue
and
skin.
14,15
Especially,
the
it
has
been
demonstrated
that
patients
early
in
their
childhoods
present
with
macrodactyly,
and
later
in
adolescence
present
with
a
volar
forearm
mass.
14,15
In
addition
to
the
clinicopathological
difference,
it
would
be
also
very
important
to
note
that
intraneural
lipoma
and
fibrolipo-
matous
hamartoma
of
the
nerve
have
crucial
differences
in
MR
imaging
since
their
surgical
treatments
differ
as
well.
On
T1-
and
T2-weighted
images,
the
intraneural
lipoma
intensities
are
markedly
high.
However,
on
STIR
images,
intensities
change
to
low,
a
level
that
is
equal
to
that
of
normal
lipoma.
These
findings
sug-
gest
that
intraneural
lipoma
is
a
pure
lipoma
occurring
only
in
the
nerve
bundles
and
that
the
nerve
fibers
run
on
the
outer
surface
of
the
mass.
On
the
other
hand,
MR
imaging
of
fibrolipomatous
hamartoma
of
the
nerve
has
a
characteristic
“coaxial
cable-like”
appearance
in
cross-section
or
“spaghetti-like”
appearance
in
lon-
gitudinal
section.
9
MR
imaging
show
serpentine
nerve
bundles
with
low
signal
intensity
embedded
within
excessive
fatty
tis-
sue,
which
appears
hyperintense
on
both
T1-
and
T2-weighted
images.
16
Transverse
sections
show
thin
hypointense
septa
within
the
fat
tissue
that
separate
some
nerve
bundles,
16
what
we
refer
CASE
REPORT
–
OPEN
ACCESS
T.
Okubo
et
al.
/
International
Journal
of
Surgery
Case
Reports
3 (2012) 407–
411 411
to
as
the
“lotus
sign.”
These
septa
may
represent
thickened
per-
ineurium,
a
characteristic
pathological
finding.
16
These
differences
in
MR
imaging
would
be
useful
for
the
differential
diagnosis
between
intraneural
lipoma
and
fibrolipomatous
hamartoma
of
the
nerve,
because
even
the
biopsy
data
might
be
sometimes
not
informative.
Treatment
of
intraneural
lipoma
involves
total
excision,
which
was
also
performed
in
our
Case
1.
Cut
of
the
nerve
fibers
is
also
reported
in
case
they
were
involved
in
the
huge
intraneu-
ral
lipoma.
8
On
the
other
hand,
the
gold
standard
treatments
for
fibrolipomatous
hamartoma
of
the
nerve
are
usually
conservative
approaches
including
decompression
and
debulking
of
the
fibro-
fatty
sheath,
microsurgical
dissection
of
the
neural
elements
and
observation
for
asymptomatic
patients.
2
In
case
of
fibrolipoma-
tous
hamartoma
associated
with
macrodactyly,
excision
of
the
involved
nerve
segment
with
or
without
nerve
grafting,
or
ampu-
tation
of
the
finger
is
recommended.
2,16
Treatment
should
focus
on
alleviating
the
patients’
symptoms,
and
complete
excision
of
these
tumors
is
not
recommended
if
clinically
significant
motor
or
sensory
deficits
are
anticipated.
17
Objective
evaluations
of
pre-
operative
and
post-operative
hand
functions
using
some
kinds
of
scoring
have
not
performed
for
either
of
3
cases.
However,
the
symptoms
of
3
patients
reported
here
improved
or
disappeared
after
surgery,
and
no
complication
was
observed
after
surgery
in
all
cases.
Furthermore,
relapse
of
tumor
and
patients’
symptoms
have
not
been
observed
so
far
at
6-months
(Cases
1
and
2)
and
4-years
(Case
3)
follow-up.
In
contrast,
Louis
et
al.
reported
in
1985
long-
term
follow-up
of
4
cases
in
which
decompression
was
performed,
although
their
symptoms
were
described
to
be
deteriorating.
18
In
cases
in
which
the
mass
has
already
led
to
major
motor
distur-
bance,
simple
decompression
or
decreasing
the
bulk
of
the
mass
may
not
improve
nerve
function,
and
it
may
be
necessary
to
per-
form
a
resection
of
the
involved
nerves
and
then
reconstruct
them
as
necessary
to
restore
function.
4.
Conclusion
Finally,
when
MRI
findings
suggest
a
lipoma
in
the
forearm,
the
possibility
of
intraneural
lipoma
should
be
considered
and
treated
with
care.
When
MRI
findings
indicate
fibrolipomatous
hamartoma
of
the
nerve,
careful
preoperative
planning
of
reconstruction
for
long-term
reactivation
of
motor
or
sensory
deficits
should
be
per-
formed.
Conflict
of
interest
statement
None.
Funding
This
work
was
supported
in
part
by
a
Grant-in-Aid
for
Gen-
eral
Scientific
Research
from
the
Ministry
of
Education,
Science,
Sports
and
Culture,
Tokyo,
Japan
(#23590434
to
Tsuyoshi
Saito
and
#24590429
to
Hiroyuki
Mitomi).
Ethical
approval
Written
informed
consent
was
obtained
from
each
patient
for
publication
of
this
case
report
and
accompanying
images.
A
copy
of
the
written
consents
is
available
for
review
by
the
Editor-in-Chief
of
this
journal
on
request.
Author
contributions
All
authors
have
contributed
significantly,
and
that
all
authors
are
in
agreement
with
the
content
of
the
manuscript.
Tatsuya
Takagi,
Tomoaki
Torigoe,
Yoshiyuki
Suehara,
Hirohisa
Katagiri,
Hideki
Murata,
Mitsuru
Takahashi,
and
Kazuo
Kaneko
per-
formed
operation
at
least
one
of
these
3
cases;
Tsuyoshi
Saito,
Hiroyuki
Mitomi,
and
Ichiro
Ito
diagnosed
these
3
cases;
and
Taketo
Okubo,
Tsuyoshi
Saito,
and
Takashi
Yao
wrote
the
paper.
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