Vol.2, No.9, 562-567 (2013) Case Reports in Clinical Medicine
Long term treatment of recurring pathological
fractures due to Mccune Albright Syndrome: Case
report and literature review
Yoshvin Sunnassee, Yuhui Shen, Rong Wan, Jianqiang Xu, Weibin Zhang*
Department of Orthopedics, Shanghai Ruijin Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai Institute of
Traumatology and Orthopedics, Shangha i, China; *Corresponding Author: weibin@medmail.com.cn
Received 17 June 2013; revised 15 July 2013; accepted 10 August 2013
Copyright © 2013 Yoshvin Sunnassee et al. This is an open access article distributed under the Creative Commons Attribution Li-
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McCune Albright syndrome is a rare genetic
disorder which is characterized by café au lait
skin pigment ation, precocious pubert y and poly -
ostotic fibrous dysplasia. Treating recurring pa-
thological fractures due to Albright syndrome is
a very challenging endeavor, and more so w hen
it is accompanied by poor bone quality and de-
formity. We hereby presen t the case of a 23-year-
old male patient who is treated several times for
recurrent pathological fractures of the femur at
our center. We analyze the difficulties associ-
ated with treating a patient w ith poor bone qual-
ity over several years, discuss our treatment
options, review the literature for similar cases
and look at what we could have done differently.
We weigh in on the difficulties in treating a se-
verely deformed shepherd’s crook, the ways of
achieving proper internal fixation and the dan-
gers of using plating instead of an IM nail as
suggested in the literature. Our main goal in
reporting this case is to bring forth the unusual
challenges encountered when treating patients
with Albright syndrome and discussing the op-
tions of the orthopedic surgeons when treating
these types of patients.
Keywords: McCune Albright Syndrome; Surgical
Treatment; Polyostotic Fibrous Dysplasia
Mccune Albright syndrome (MAS) is a rare genetic
disorder which is characterized by café au lait skin pig-
mentation, precocious puberty and polyostotic fibrous
dysplasia (PFD) [1,2]. Patients with PFD have poor bone
quality and are liable to pathological fractures. We here-
by report the case of a male patient who is treated for 7
years in our center for recurring pathological fractures of
the femur due to MAS. When the patient was first seen,
he presented with a shepherd’s crook deformity of the
proximal femur. The condition was associated with un-
equal lower limbs’ length, inability to ambulate and pain
on motion. He had already undergone several surgeries
for recurring femoral neck fractures at another centre. We
discuss the challenges of treating this patient, review the
literature on the subject and anticip ate the challeng es that
future pathological fracture s might pose.
The patient suffered a fracture of the femur while
swimming at the age of 10 in 1997. The fracture did not
occur as a result of an impact or a fall. He was treated
surgically two times in the pediatrics orthopedics depart-
ment of another hospital. The first time, bone grafts and a
flexible intramedullary nail were used. The fracture did
not unite and a subsequent surgery was carried out in
1999 to remedy to the non-united fracture. The exact
information of both surgeries and of the post-surgical
protocol was not available. The patien t still had a painful
right lower limb and was dependent on crutches to be
able to ambulate. Following the surgery, the patient wit-
nessed weakness in his right lower limb, with pain on
ambulation. In 2005, at 16 years of age the patient was
first seen at our outpatient department. He presented with
a shepherd’s crook deformity of the proximal femur, with
a fracture in the subtrochanteric region. Physical exami-
nation revealed unequaled lower limbs length, difficulty
Copyright © 2013 SciRes. OPEN ACCESS
Y. Sunnasse e et al. / Case Reports in Clinical Medicine 2 (2013) 562-567 563
ambulating, pain in th e right thigh, deformity of th e right
thigh and dispersed café au lait spots on the abdomen.
Roentgenographic examination showed that the patient
had several fibrous lesions of the bone in the right pro-
ximal femur (Figure 1), craniofacial bone, ribs and bil-
ateral tibia and fibula. The initial diagnosis was Mccune
Albright’s syndrome with PFD and Type 1 diabetes. He
was placed on a course of oral biphosphonates and moni-
tored on a monthly basis. After two years of treatment
the patient’s dysplastic lesions did not resolve signifi-
cantly and he was admitted for surgery. Pre-operative
tests showed that the patient’s blood sugar oscillated
between 13 mmol/L and 18 mmol/L; so we opted for the
patient to be discharged. After a course of treatment in
our endocrinology department, the daily dose of insulin
was re-adjusted. He was re-admitted to our center in
2009 for surgical treatment.
The treatment protocol was limited by several factors
which rest r i c t e d our options:
1. The patient had an unusually small and dysplastic
right actebula, when compared to the left side, in which
the femoral head could not fit completely.
2. MRI scans showed that a large part of the femoral
head was also affected by fibrous dysplasia. This raised
the question of improper screw purchase in dysplastic
bone. We did not consider the femoral head to be ne-
crotic because the fracture was largely subtrochanteric
and would not have affected the blood supply to the fe-
moral head.
3. The bone strip at the point of the fracture, along the
shepherd’s crook deformity, was very thin which ren-
dered the use of an intramedullary nail very difficult.
4. The diameter of the femur and of the medullary
canal was unusually small which would have rendered
reaming and the implantation of an intramedullary nail
very difficult, with the smallest IM nail available to us
being 9 mm in diameter. A CT examination showed the
Figure 1. Shows an anterior-posterior view of the pelvis. The
right proximal femur has fibrous dysplasia lesions.
medullary canal to be also affected by fibrous lesions and
was less than 5 mm in diameter at the point of the lesion
and less than 7 mm diameter distally.
5. Due to the fact that the patient exhibited fibrous
bony lesions in the tibia and fibula, we were reluctant to
make use of a fibular strut for the corrective osteotomy,
as has been su gg ested by ot her authors.
6. The patient’s severe diabetes could negatively affect
bone healing, prompting us to use plating and screws
which ensured absolute fixation.
After re-admission, blood sugar level was lower than
14 mmol/L and a corrective osteotomy with internal fix-
ation and autologous bone grafting was carried out. The
intertrochanteric fracture was treated with a DCS plate
(Smith & Nephew, Memphis, Tennessee, United States)c,
through a Hardinge approach to the proximal femur.
Autologous bone grafts was obtained from the femoral
condyles and was used to make up for the considerable
bone deficiency at the site of fracture. The patient was
given prophylactic antibiotics as post-surgical treatment
and was advised to refrain from ambulation. Following
the surgery, he was followed at our outpatient depart-
ment, continued using oral biphosphonates and was able
to achieve complete weight-bearing 3 months after the
surgery. Subsequent X-rays showed that the fracture
united completely after one year and the patien t was able
to ambulate normally. However, he still had weakness in
the affected limb and was only able to walk for a dis-
tance of around 50 meters, without resting. The manual
muscle test was a grade 4. Continuou s use of biphospho-
nates did not decrease pain level s.
In November 2011, the patient suffered a fall and he
was unable to ambulate. In January 2012, he was once
again admitted to our center after roentgenograph ic scans
showed a fatigue fracture of the femur at the distal part
of the DCS plate with bending deformity of the femur
(Figure 2). The patient was once more treated with an
open reduction and internal fixation of the femur. We still
faced the same obstacles as during the first surgery but
with the added difficulty of having a structurally com-
promised femur, severe bone deficiency along the fibrous
lesions and a femoral head which was both dysplastic
and bone deficient. We opted to use a longer plate (DCS,
Synthes) and placed the proximal nails in the lower part
of the femoral head to allow for better purchase of the
nails in normal bone and made up for bony deficiency
through bone grafting (Figure 3). The patient was dis-
charged with advice to refrain from ambulation and to
undergo rehabilitation under the guidance of the main
surgeon. Following the surgery, the patient restarted am-
bulation after one month and slowly regained the ability
to ambulate without walking aids.
The patient was admitted again in April 2012 after he
suffered a fracture after another fall. The fracture was
Copyright © 2013 SciRes. OPEN ACCESS
Y. Sunnasse e et al. / Case Reports in Clinical Medicine 2 (2013) 562-567
Figure 2. Shows the anterior-posterior
view of the femur with bending defor-
mity of the femur at the distal part of
the plate.
Figure 3. Shows two anterior posterior views of the femur. The
left X-ray shows the femur after use of a DCS plate with the
proximal nails placed in the lower part of the femoral head. The
right X-ray shows the femur after placement of a second plate
at the distal part of the femur.
located in the distal part of the femur and was considered
to be a result of stress shielding due to the distal part of
the DCS plate. We fixed the distal fracture with plating
and screws. The fracture line involved a screw which had
to be removed and could not be fixed again. The LC-
DCP plate was placed in the antero-medial part of the
femur due to the fact that there was already another plate
placed laterally (Figure 3).
At the latest check-up, in June 2012, at 3 months after
the last surgery, the patient had regained ambulation, had
unequal lower limbs length with the right leg being
shorter than the left leg by 3 cm. The patient is still wit-
nessing weakness in the affected limb and is unwilling to
make use of corrective shoes. MMT was grade 4. The
fracture healed partially as was seen on follow-up post-
operative X-rays.
PFD is a benign bony lesion but can however lead to
severe deformity which eventually requires surgery. The
shepherd’s crook presentation develops as a result of the
mechanical load bearing acting on the structurally
weakened proximal femoral bone, is a well documented
phenomenon [3-5]. Intracapsular fracture may occur due
to a shearing force against severe valgus deformity [5]
coupled with the fact that the proximal femur has poor
bone quality. According to Russell and Chandler, who
reported on the surgical treatment of PFD, the indications
for surgery in fibrous dysplasia are continued pain in the
region of a localized bone lesion, fracture through a le-
sion, or severe deformity [6]. The medical literature offer
very few reports of long term treatment of patient with
this disease. Studies have noted that bone dysplasia have
normal bone healing capacities [7,8]. Surgical options
should take into account the quality of the bone affected
and the deformity present. Monostotic fibrous dysplasia,
once treated surgically, is unlikely to reoccur while poly-
ostotic fibrous dysplasia, whether it is a presentation of
MAS or not, is likely to progress with the disease reach-
ing a quasi-static phase once adulthood is reached [7,9].
Several ways of treating PFD of the proximal femur
have been previously discussed in the medical literature,
with methods such as intralesional resection and bone
grafting or corrective osteotomy with internal fixation
which can either be plating or an intramedullary nail. The
medical literature supports the idea that in cases of fi-
brous dysplasia of the proximal femur, an intramedullary
device should be used with a screw fixed in the femoral
head [10]. The idea of fixing the screw in the femoral
head is based on the premise that the femoral head is not
affected or rarely affected by fibrous dysplasia and as
such can provide proper purchase for screws [9,11,12].
The IM nail can be very useful when dealing with cases
of proximal femur PFD with a shepherd’s crook deform-
ity, where a wedge osteotomy can be performed to re-
store the natural geometry of the bone [3,13,14]. Correc-
tive osteotomy is particularly useful in cases where there
is no fracture of the proximal femur. Success of correc-
tive osteotomy resides on the presence of healthy sub-
trochanteric bone or in cases where bone grafting is used,
autologous healthy bone grafts might be preferred. Jung
et al. reported on cases where multiple osteotomies were
performed to restore the neck shaft angle using an in-
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Y. Sunnasse e et al. / Case Reports in Clinical Medicine 2 (2013) 562-567 565
tramedullary nail with neck cross-pinning [3]. Further-
more, when we reviewed the literature we found that
most instances where IM nails were used were cases with
comparatively mild shepherd’s crook deformity. Use of
plating and screws has been met with mixed success.
Chen et al. reported on a case of an intracapsular fracture
with shepherd’s crook deformity which was treated with
corrective osteotomy and internal fixation with a dy-
namic hip screw. The patient in the report healed com-
pletely and did not suffer subsequent fractures [4].
However, other reports have either underlined the risk of
bending deformity of the femur or fatigue fracture [3].
According Guile et al., metal plate removal will nor-
mally lead to refracture.
The success of plating depends on proper purchase of
the screws in healthy bone, which in this case will de-
pend on the state of the femoral head bone and femoral
stem bone. Proper purchase guarantees stability of the
fixation. It has been noted that restoring the mechanical
axis reduces the deforming forced on the bone, with me-
dial displacement varus osteotomy being a possible solu-
tion [12]. Decreased neck fracture in PFD patient is a
concern and is associated with decreased functional mo-
bility for sports [15]. However, these considerations are
at times a luxury in patients with PFD. Restoring ambu-
lation ability remains the goal in most severe cases of
polyostotic fibrous dysplasia. Surgical treatments that
aim at restoring the proper neck-shaft angle, especially in
patients with lower extremity deformity [10] or even in
patients with unequal lower limbs length is questionable,
because the mechanical axis is not properly restored. An
important part of treating PFD induced fracture of the
proximal femur is dealing with a structurally compro-
mised stability [7]. Therefore, our choice of surgical
protocol mainly focused on allo wing the patient to retain
ambulation capacity while assuring that the fracture
would unite, while having to contour the several restric-
tions posed by this patien t’s case.
Intralesional resection and bone grafting was popular-
ized by Enneking [16]. He obtained positive results with
this method. However, subsequent studies by other au-
thors revealed that the bone grafts subsequently resolved
leaving place to dysplastic lesions. Treatment of PFD
with bone grafting, independently of the type of bone
strut used or the age of the patient, is unlikely to offer
positive results in PFD [7,17]. After the initial surgical
treatment in this case, following fractures were all due to
trauma. However, even if we were left with no other
choices, the use of plating and screws is normally not
advised in the treatment of PFD of the proximal femur.
As seen in this case, following treatment with plating and
screws there was a bending deformity of the proximal
femur. It is a controversy in certain cases whether sur-
gery would be the best course of actions, especially in
patients with severe bone deformity and deficiency. It
has been noted by other authors that in certain cases of
PFD, the best approach is to not operate [10], however,
we also think that allowing the patient to retain ambula-
tion without walking aids should be a primary goal of
PFD surgery of the lower limbs.
The use of biphosphonates in the treatment of PFD has
proven its merits with no harmful side-effects, especially
when it comes to the ability of bone growth in younger
patients [10]. Oral or intravenous biphosphonates both
have a positive effect on pain and bone metabolism
markers [18,19]. However, we noticed that the patient’s
bone was hard during internal fixation; we cannot con-
firm whether this change in the quality of bone is directly
related to the use of biphosphon a tes.
Other considerations are important in the peri-opera-
tive management of a PFD patient. It is especially im-
portant to check bone markers metabolism in the build-
up to the surgery. A state of hyperparathyroidism would
be an indication against surgical treatment. High levels
of parathyroid hormones are likely to signal negative
post-surgical results. MAS has been known to develop
with endocrinopathies, with patients exhibiting hyper-
parathyroidism or diabetes with MAS. Even if this pa-
tient has type 1-diabetes and his blood sugar level cannot
be controlled properly, we chose not to discuss this as-
pect of the patient’s condition as being related to MAS.
Even though there has been reports of MAS causing en-
docrinopathies and a few reports of MAS being related to
type 1 diabetes, we could not ascertain that our patient’s
diabetic condition was a direct resu lt of MAS. It must be
noted that the type 1 diabetes represented an obstacle in
the peri-operative management of the patient; we think
that it should not affect treatment protocol but as is
known high blood sugar level can severely affect bone
3.1. What Would We Do Differently?
When surgeons face this type recurrent fracture they
always need to review their treatment protocol in light of
new advances and assess whether they made the right
decisions throughout the treatment. These cases of MAS
with severe deformity of the proximal femur are always
difficult to treat, however, we think that there are a few
things that we could c on si der doing dif ferently.
1. Using a longer plate with fewer screws in the shaft
of the femur and placing a cortical screw at the distal end
of the plate so as to decrease the stress shielding on the
bone would be an operative option that could have bene-
fited the patient. Placing a co rtical screw at the distal end
has been shown to be beneficial towards decreasing the
risk of stress fracture [20]. Using a 3.5 inch pediatric
plate instead of a 4.5 inch would also may be have been a
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Y. Sunnasse e et al. / Case Reports in Clinical Medicine 2 (2013) 562-567
better fit since the patient’s bone was small in size.
2. Placing the femoral neck at an increased valgus
angle instead of 90˚ varus is also another option but it
would have been difficult to achieve due to the very thin
strip of bone left along the shepherd’s hook. When the
patient was operated we placed the femoral neck at a
varus angle of 90˚, which exposed the non-weight bear-
ing part of the articular cartilage of the femoral head (the
more lateral side of the femoral head) to greater loading.
Furthermore, the weight from the hip was transferred
onto a 90˚ varus femoral neck which could have caused
instability at the site of fracture, rendering the internal
fixation unstable and thus unlikely to heal. The patient
should have been advised to refrain from weight bearing
until the fracture heals completely. Furthermore, placing
the femoral neck at a valgus angle could have helped to
slightly lengthen the leg; however this would be limited
by the atrophied muscles and by the sciatic nerve.
3. The degree of aggressivity of the treatment in early
childhood is an important question when dealing with
these types of patients. Funk et al. noted that the progr ess
of the disease subsides, therefore aggressive surgical
intervention must be practiced in children to prevent the
possibility of difficult procedures in adults [7]. In this
specific case, it is difficult to assess whether a more
aggressive course of treatment in early childhood would
have made any difference in the prognosis.
3.2. Future Considerations
Even if at the last follow-up the patient had regained
ambulation, we must consider the surgical protocol in
cases of subsequent fractures. The quality of bone of the
proximal femur is poor, coupled with the fact that plates
were used; it is highly likely that the patient will suffer
more fatigue fractures at the distal part of the plate.
Treating these fractures will be increasingly difficult. He
is still at the very beg inning of adulthood, and we do not
know yet if the fibrous dysplasia will advance or cease to
progress. Furthermore, in old age, when osteopenic
changes occur, the patient is likely to suffer more frac-
tures and the subsequent treatment protocol will have to
take into consideration the impoverished bone quality.
This patient’s condition will not improve and is likely to
become worse in case of more fractures of the proximal
femur, unless major reconstructive surgery of the ace-
tabula and of the femur is undertaken, with the use of
total length femoral pro sthesis. And even then, the diffe-
rence of 3 cm between the lower limbs will not be
corrected because a drastic increase in the right lower
limb length might damage nerves and blood vessels
which are already being taxed by the patient’s diabetic
condition. We hope to be able to continue fixing future
fractures with open reduction and internal fixation until
the bone quality is too poor or deformity too severe to
continue doing so at which point major reconstructive
surgery will be required.
The treatment of polyostotic fibrous dysplasia asso-
ciated with shepherd’s crook deformity can represent a
considerable challen g e to th e o r thoped ic su rgeon. Ev en if
common knowledge had indicated the use of IM nails, in
our patient’s case, the use of an IM nail would have been
much more difficult. However, our primary goal of sup-
plying an absolute fixation at the proximal femur to
allow for fracture union and allow the patient to re-
acquire his ambulating capacities in his right leg was
attained. In su ch cases of PFD, the d ifficulty of treatment
lies in the fact that eventual fatigue fractures are d ifficult
to treat and the end-goal is allowing the patient to retain
ambulation and remain pain free.
We would like to thank Dr Edward P. Southern for his insight and
invaluable help in the conception of this work.
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