CASE
REPORT
–
OPEN
ACCESS
International
Journal
of
Surgery
Case
Reports
17
(2015)
128–132
Contents
lists
available
at
ScienceDirect
International
Journal
of
Surgery
Case
Reports
j
ourna
l
h
om
epage:
www.casereports.com
Focal
anatomic
resurfacing
implantation
for
bilateral
humeral
and
femoral
heads’
avascular
necrosis
in
a
patient
with
Hodgkin’s
lymphoma
and
literature
review
Onur
Bilge
a,∗
,
Mahmut
Nedim
Doral
b
,
Anthony
Miniaci
c
a
Department
of
Orthopaedics
and
Traumatology,
Konya
Necmettin
Erbakan
University,
Meram
Faculty
of
Medicine,
Meram,
42080
Konya,
Turkey
b
Department
of
Orthopaedics
and
Traumatology,
Hacettepe
University,
Faculty
of
Medicine,
06230
Ankara,
Turkey
c
Cleveland
Clinic
Sports
Health
Center,
5555
Transportation
Blvd,
Garfield
Heights,
OH
44125,
USA
a
r
t
i
c
l
e
i
n
f
o
Article
history:
Received
30
June
2015
Received
in
revised
form
22
October
2015
Accepted
25
October
2015
Available
online
25
November
2015
Keywords:
Femoral
head
Humeral
head
Avascular
necrosis
Resurfacing
arthroplasty
HemiCAP
Hodgkin’s
lymphoma
a
b
s
t
r
a
c
t
INTRODUCTION:
The
femoral
and
humeral
heads
are
among
the
most
common
sites
of
osteonecrosis.
The
aims
of
this
case
report
was
to
report
three
years’
results
for
sequential
treatment
of
bilateral,
concomitant
involvement
of
humeral
and
femoral
heads
with
focal
anatomic
resurfacing
implantation
in
a
single
patient
with
Hodgkin’s
lymphoma
and
to
review
the
relevant
literature,
which
is
relatively
scarce.
PRESENTATION
OF
CASE:
We
present
a
48-year-old
male
patient
with
concomitant,
bilateral
femoral
and
humeral
head
avascular
necrosis.
He
was
diagnosed
as
Hodgkin’s
lymphoma
in
1984.
He
had
bilateral
groin
and
shoulder
pain,
lasting
for
three
years
and
aggravated
by
joint
motions.
Radiological
evaluations
demonstrated
bilateral
focal
osteonecrosis
of
femoral
heads
and
humeral
heads,
respectively.
Despite
conservative
treatment,
he
did
not
obtain
any
symptomatic
relief.
Following
the
common
decision,
he
was
treated
with
sequential
implantations
with
the
HemiCAP
®
device
for
both
bilateral
pathologies,
by
a
single
surgeon
and
standard
surgical
approaches.
Neither
intraoperative
nor
postoperative
complication
was
encountered.
After
the
follow-up
period
of
36
months
after
the
last
surgery,
he
was
symptomless
and
with
normal
range
of
motion
for
all
four
joints.
DISCUSSION:
The
bilateral,
concomitant
involvement
of
humeral
and
femoral
head
in
the
setting
of
avas-
cular
necrosis
is
relatively
rare.
Moreover,
the
optimal
treatment
method
at
earlier
stages,
in
young
patients
has
not
been
established
yet.
CONCLUSION:
This
study
is
the
first
report
to
present
the
three-years’
clinical
result
of
a
single,
relevant
case,
who
was
treated
with
sequential
focal
anatomic
resurfacing
implantations
(HemiCAP
®
)
in
four
aforementioned
joints.
©
2015
The
Authors.
Published
by
Elsevier
Ltd.
on
behalf
of
IJS
Publishing
Group
Ltd.
This
is
an
open
access
article
under
the
CC
BY-NC-ND
license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
1.
Introduction
The
femoral
head
and
humeral
head
are
the
first
and
second
most
common
sites
of
osteonecrosis
in
the
human
body,
respec-
tively
[1,2].
But,
bilateral
and
concomitant
involvement
is
rare.
The
risk
factors
are
commonly
corticosteroid
use
and
trauma
[3].
The
management
should
start
with
a
high
index
of
suspicion
in
order
to
diagnose
earlier
and
to
prevent
further
arthritic
process.
Although
there
are
non-operative
and
operative
options,
the
opti-
mal
treatment
method
has
not
been
established
yet
[4,5].
Partial
or
total
resurfacing
options
have
the
advantage
of
preserving
the
patient’s
anatomy
to
a
maximum
extent,
under
“joint-preserving”
surgeries,
which
have
recently
gained
importance
especially
for
young
and
middle-aged,
symptomatic
patients
[6].
There
are
only
∗
Corresponding
author.
Fax:
+90
332
223
61
82.
E-mail
addresses:
(O.
Bilge),
(M.N.
Doral),
(A.
Miniaci).
a
few
studies
related
with
the
treatment
of
osteonecrosis
of
the
femoral
or
humeral
heads
with
resurfacing
[7,8].
To
the
best
of
our
knowledge,
there
was
no
previous
report
of
a
case—who
had
symp-
tomatic,
concomitant
and
bilateral
osteonecrosis
of
femoral
head
and
humeral
head—who
was
treated
with
the
joint-preserving
focal
anatomic
resurfacing
implant
(HemiCAP
®
)
with
a
three
years
of
follow-up.
The
aims
of
this
study
was
to
report
a
case
of
a
48-year-old
male
patient
with
Hodgkin’s
lymphoma,
who
was
diagnosed
with
bilat-
eral
femoral
head
and
humeral
head
osteonecrosis
and
who
was
treated
successfully
with
the
HemiCAP
®
and
to
review
the
relevant
literature,
which
is
relatively
scarce.
2.
Presentation
of
case
A
48-year-old
male
patient
with
a
history
of
Hodgkin’s
lym-
phoma
admitted
to
our
department
of
adult
reconstructive
surgery
service
with
complaints
of
bilateral
hip
and
shoulder
pain
and
limitation
in
range
of
motion
of
all
four
joints
in
2011.
He
was
http://dx.doi.org/10.1016/j.ijscr.2015.10.034
2210-2612/©
2015
The
Authors.
Published
by
Elsevier
Ltd.
on
behalf
of
IJS
Publishing
Group
Ltd.
This
is
an
open
access
article
under
the
CC
BY-NC-ND
license
(http://
creativecommons.org/licenses/by-nc-nd/4.0/
).
O.
Bilge
et
al.
/
International
Journal
of
Surgery
Case
Reports
17
(2015)
128–132
129
diagnosed
as
Hodgkin’s
lymphoma
in
1984.
He
was
treated
with
chemotherapy,
corticosteroids
and
finally
cured
with
bone
marrow
transplantation
in
2007.
The
hip
pain
was
localized
to
bilateral
groins,
aggravating
with
joint
movements—especially
with
internal
rotation-
and
during
weight
bearing.
The
shoulder
pain
was
getting
worse
with
joint
movements,
especially
with
overhead
activities.
The
duration
of
his
complaints
was
lasting
approximately
for
3
years,
before
his
admis-
sion.
There
was
no
history
of
trauma
to
both
joints.
He
was
treated
non-surgically
with
non-steroidal
anti-inflammatory
drugs
and
use
of
crutches
to
limit
weight
bearing,
over
the
last
year.
But
this
non-
operative
management
provided
very
limited
symptomatic
relief.
The
physical
examination
revealed
an
antalgic
gait
without
Trendelenburg
sign
and
limp
length
inequality.
The
range
of
motion
(ROM)
of
the
right
and
left
hips
was
painful
and
limited
as
follows,
respectively:
full
extension—full
extension,
100–90
◦
of
flexion,
15–10
◦
of
internal
rotation,
40–30
◦
of
external
rotation,
30–30
◦
of
abduction
and
20–15
◦
of
adduction.
On
physical
examination
of
the
shoulder,
the
range
of
motion
of
the
right
and
left
sides
were
painful
and
limited
as
follows,
respectively:
100–90
◦
of
active
forward
elevation
and
30–30
◦
of
external
rotation.
Magnetic
Resonance
Imaging
(MRI)
evaluations
of
both
hips
and
shoulders
demonstrated
bilateral
focal
osteonecrosis
of
femoral
heads
(Ficat-Arlet
Stage
III)
and
humeral
heads
(Cruess
Stage
III),
respectively
(Fig.
1)
[9,10].
The
largest
anteroposterior
and
medio-
lateral
sizes
(mm)
of
the
lesions
measured
on
MRI
were
recorded
as
follows:
19.8–30.3
(right
femur),
17.8–29.5
(left
femur),
22.8–31.5
(right
humerus),
31.7–36.2
(left
humerus).
Based
on
the
symptomatology
and
age
of
the
patient
and
his
unresponsiveness
to
non-operative
management,
surgical
options
were
discussed
with
the
patient.
The
common
decision
was
made
to
perform
initial
focal
anatomical
resurfacing
implantation
for
the
relevant
joints.
Following
perioperative
antibiotic
prophylaxis
and
sterile
preparation
of
the
surgical
sites;
standard
surgical
approaches
were
performed
by
a
single
surgeon
(O.B.);
firstly
for
both
hips
with
one-month
interval
and
then
two
months
later,
sequentially
for
both
shoulders
(with
two-months
inter-
val).
Neither
intraoperative
nor
postoperative
complication
was
encountered.
2.1.
Surgery
of
the
hip
Under
spinal
anesthesia,
safe
surgical
dislocation
was
per-
formed
according
to
the
technique
described
by
Ganz
et
al.
[11].
After
proper
debridement
of
the
osteonecrotic
lesion,
appropriate-
sized
focal
resurfacing
implant
(HemiCAP
®
)
matching
patient’s
femoral
head
curvature
was
impacted
securely
over
the
tapered
titanium
screw.
The
range
of
motion
and
the
impingement
of
the
hip
joint
were
checked
after
relocation
of
the
hip.
The
intraopera-
tive
control
of
radiography
was
done
routinely
in
anteroposterior
and
lateral
planes.
The
osteotomy
site
and
surgical
planes
were
closed
accordingly.
2.2.
Surgery
of
the
shoulder
Following
interscalene
brachial
plexus
blockage;
standard
del-
topectoral
approach
was
performed
in
the
half-sitting
position
with
the
head
elevated
45
◦
.
After
the
incision
of
the
subscapularis
ten-
don
and
the
capsule
underneath
–
leaving
a
cuff
tissue
on
the
lesser
tuberosity
for
reattachment
–,
the
humeral
head
was
dislocated
anteriorly
by
externally
rotating
the
arm,
with
full-exposure
of
the
humeral
head.
After
debridement
of
the
lesion,
appropriate-sized
implant
(HemiCAP
®
)
matching
patient’s
humeral
head
curvature
was
impacted
securely
over
the
tapered
titanium
screw.
The
35
mm
and
40
mm
diameter
final
implants
were
used
for
right
and
left
sides,
respectively.
After
relocation
of
the
shoulder,
the
range
of
Fig.
1.
MRI
images.
(A)
Preoperative
T1-weighted
MRI
scans
of
both
hips
demon-
strating
Ficat-Arlet
Stage
III
avascular
necrosis.
(B)
Right
and
(C)
left
shoulders’
preoperative
T1-weighted
MRI
scans,
demonstrating
Cruess
Stage
III
avascular
necrosis.
130
O.
Bilge
et
al.
/
International
Journal
of
Surgery
Case
Reports
17
(2015)
128–132
Fig.
2.
Full
range
of
motion
of
patient’s
both
hips
and
shoulders
at
36th
months
(A–C).
motion
was
checked
for
surface
congruency.
The
layers
were
closed
accordingly.
The
intraoperative
control
of
radiography
was
done
routinely
in
anteroposterior
and
lateral
planes.
A
sling
was
used
postoperatively.
On
one
hand,
thromboprophylaxis
was
used
for
6
weeks
post-
operatively
following
only
the
hip
surgeries.
The
patient
was
mobilized
toe-touch
weight
bearing
during
the
first
four
weeks
in
order
to
allow
healing
of
trochanteric
osteotomy
and
sufficient
implant-bone
integration.
Then
the
mobilization
was
progressed
to
full-weight
bearing
as
tolerated.
The
osteotomies
were
healed
at
postoperative
2nd
months.
Thereafter,
the
screws
were
removed
within
the
first
year
due
to
irritation.
On
the
other
hand,
shoulder
joint
rehabilitation
followed
as
described
in
the
study
by
Uribe
and
Botto-van
Bemden
[8].
The
follow-up
period
after
the
last
surgery
was
36
months.
At
the
end
of
this
period,
the
patient
was
with-
out
symptom
and
with
normal
range
of
motion
for
all
four
joints
(
Fig.
2A–C).
Fig.
3
shows
the
last
radiograph
at
36th
months
since
the
last
surgery.
The
patient
was
informed
that
data
from
the
case
would
be
sub-
mitted
for
publication,
and
gave
his
consent
for
both
surgeries
and
gave
permission
for
publication
of
the
data
from
this
case,
including
photographs.
Our
work
has
been
reported
in
line
the
CARE
criteria
[12].
3.
Discussion
To
the
best
of
our
knowledge,
this
study
is
the
first
report
to
present
represents
the
mid-term
successful
clinical
results
of
focal,
anatomic
resurfacing
implantation
for
the
treatment
of
a
middle-
aged
patient
with
avascular
necrosis
of
bilateral
femur
and
bilateral
humeral
heads,
firstly
in
the
literature.
The
femoral
head
and
humeral
head
are
the
two
common
sites,
in
which
osteonecrosis
are
encountered.
But,
their
simultaneous,
bilateral
involvement
and
their
sequential
treatment
with
focal,
anatomic
resurfacing
implants
was
not
reported
before.
In
general,
the
management
of
the
osteonecrosis,
non-operative
and
operative
options
are
present.
The
early
diagnosis
with
a
high
index
of
sus-
picion
is
of
utmost
importance,
in
order
to
have
the
opportunity
to
apply
a
“joint-preserving”
treatment
modality
–
especially
for
a
weight
bearing
joint
like
hip-,
which
have
regained
popularity
in
the
recent
years,
as
stressed
by
Leuning
and
Ganz
[6].
We
think
that
HemiCAP
®
implants
have
offered
a
variety
of
advantages
since
their
first
launch
into
the
market
in
2002.
The
preoperative
planning
is
much
more
easier
than
any
total
arthro-
plasty.
Moreover,
intraoperative
diameters
and
contour
shapes
allow
the
surgeon
to
not
only
cover
the
lesion
effectively,
but
also
fit
the
implant
to
the
patient
while
preserving
healthy
bone
and
cartilage
[13].
HemiCAP
®
implant
constitutes
one
of
the
new
alter-
native
options
of
“joint-preserving”
surgeries,
while
protecting
the
patients’
anatomy
with
little
bone
bony
resection
[14].
In
addition,
the
implant
matches
the
patient’s
anatomy
and
the
related
contour
of
the
joint
surface.
As
they
are
suitable
for
young
and
middle-
aged
patients,
the
chance
for
their
revision
with
total
hip
or
total
shoulder
arthroplasty
is
always
possible,
in
case
of
implant
failure,
fracture
or
further
progression
of
arthritis,
if
any.
O.
Bilge
et
al.
/
International
Journal
of
Surgery
Case
Reports
17
(2015)
128–132
131
Fig.
3.
Final
radiograph
of
all
four
focal
anatomic
resurfacing
implants
(HemiCAP
®
)
in
bilateral
femoral
and
humeral
heads,
at
postoperative
36th
months
since
the
last
surgery.
The
longest
follow-up
periods
of
patients
with
its
clinical
use
were
3
years,
and
6
years
for
shoulder
[2,8,15–17]
and
hip
[18–21]
surgeries,
respectively.
The
clinical
experience
reported
till
now
was
limited
with
only
four
case
reports
and
a
case
series
related
with
the
use
in
hip
pathologies
of
the
femoral
head
[7,18–21].
4.
Conclusion
In
summary,
the
alternative
focal,
anatomic
resurfacing
implan-
tation
with
HemiCAP
®
in
this
particular
case,
having
bilateral,
focal
osteonecrosis
of
the
femoral
and
humeral
heads
has
functioned
well
in
mid-term.
To
the
best
of
our
knowledge,
a
single
patient
with
Hodgkin’s
lymphoma
–
who
was
operated
by
sequential
focal,
anatomic
resurfacing
implantation
on
both
femoral
and
humeral
heads
due
to
focal
avascular
necrosis-
was
firstly
presented
in
the
relevant
literature.
Although
this
study
demonstrated
successful
clinical
results
as
an
alternative
modality
for
the
sequential
treat-
ment
of
avascular
necrosis
in
the
relevant
joints;
in
order
to
make
concrete
conclusions
for
the
long-term
efficacy
and
the
routine
use
of
this
implant
as
an
alternative
option
for
the
treatment
of
avascular
necrosis
of
the
relevant
joints;
additional
prospective,
randomized
studies
with
longer
follow-up
period
and
higher
num-
ber
of
patients
are
warranted.
Conflict
of
interest
A.M.
has
royalties
with
Arthrosurface.
Other
authors
have
no
relevant
conflicts
of
interest
to
declare.
Source
of
funding
The
study
had
no
sponsors.
Ethical
approval
The
present
study
was
approved
by
the
local
ethical
commit-
tee
of
the
Konya
Necmettin
Erbakan
University
Meram
Faculty
of
Medicine
(Reference
number:
2014-709).
Consent
The
patient
was
informed
that
data
from
the
case
would
be
sub-
mitted
for
publication,
and
gave
his
written
and
signed
consent
for
all
surgeries
and
gave
permission
for
publication
of
the
data
from
this
case,
including
photographs.
Author
contribution
O.
Bilge
designed
the
study
concept,
carried
out
the
surg-
eries,
collected
data,
analysed
data,
wrote
the
paper,
revised
the
manuscript
critically,
gave
approval
for
the
final
version.
M.N.
Doral
revised
the
manuscript
critically
for
content,
gave
approval
for
the
final
version.
A.
Miniaci
revised
the
manuscript
critically,
partici-
pated
in
its
design
and
coordination,
and
gave
approval
for
the
final
version.
Guarantor
The
guarantor
is
O.
Bilge.
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