Insufficiency fractures: A rare cause of foot and ankle pain in three patients with rheumatoid arthritis

Abstract

Insufficiency fractures are recognized but rare complications in patients with longstanding rheumatoid arthritis. Such fractures are typically solitary and are rarely seen to affect the foot and ankle. We describe 3 women with longstanding rheumatoid arthritis, treated with one, or a combination of, corticosteroids, DMARDs and anti-TNF, presenting with foot and ankle pain with no prior history of trauma. MRIs showed rare multiple florid insufficiency fractures of the foot and ankle, in 2 cases bilaterally, which were managed conservatively. These cases highlight the importance of considering insufficiency fractures in similar patients presenting with foot and ankle pain. Radiographs may fail to demonstrate these lesions, delaying diagnosis, and worsening patient outcome, therefore in such cases MRI is a valuable modality.

Full text

Radiology Case Reports 13 (2018) 855–861
Available online at www.sciencedirect.com
journal homepage: www.elsevier.com/locate/radcr
Case Report
Insufciency fractures: A rare cause of foot and
ankle pain in three patients with rheumatoid
arthritis
R , RR
Katherine C.L. Hillyard
∗
, Shabnam Shabbir , Udawattage Nadeeka Sirisena ,
Maxine Hogarth, Ajay Sahu
Radiology Department, Ealing Hospital, Uxbridge Rd, Southall, London UB1 3HW, UK
          
Article history:
Received 16 February 2018
Revised 24 May 2018
Accepted 26 May 2018
Keywords:
Insufciency
Fracture
Fragility
Rheumatoid
Foot
Ankle
       
Insufciency fractures are recognized but rare complications in patients with longstanding
rheumatoid arthritis. Such fractures are typically solitary and are rarely seen to affect the
foot and ankle. We describe 3 women with longstanding rheumatoid arthritis, treated with
one, or a combination of, corticosteroids, DMARDs and anti-TNF, presenting with foot and
ankle pain with no prior history of trauma. MRIs showed rare multiple orid insufciency
fractures of the foot and ankle, in 2 cases bilaterally, which were managed conservatively.
These cases highlight the importance of considering insufciency fractures in similar pa-
tients presenting with foot and ankle pain. Radiographs may fail to demonstrate these le-
sions, delaying diagnosis, and worsening patient outcome, therefore in such cases MRI is a
valuable modality.
© 2018 The Authors. Published by Elsevier Inc. on behalf of University of Washington.
This is an open access article under the CC BY-NC-ND license.
( http://creativecommons.org/licenses/by-nc-nd/4.0/ )
Introduction
Rheumatoid arthritis predisposes patients to insufciency
fractures secondary to osteoporosis, this is in part due to the
disease process, but also due to environmental factors such
as reduced weight bearing and as a side effect of medications
used to treat the disease. In the medical literature, single in-
sufciency fractures of the hip and pelvic girdle are more com-
monly reported. This case series presents 3 cases of insuf-
R Acknowledgments: This case report had no source of funding or relevant nancial interest.
RR Conict of interests: The authors have declared there is no known potential conict of interest.
∗Corresponding author.
E-mail addresses: katehillyard@hotmail.co.uk (K.C.L. Hillyard), shabnam.shabbir@nhs.net (S. Shabbir), n.sirisena@gmail.com (U.N.
Sirisena), maxine.hogarth@nhs.net (M. Hogarth), asahu@nhs.net (A. Sahu).
ciency fractures that are unusual in a number of ways. First,
the site of the fractures is unusual in affecting the foot and an-
kle, in 2 cases bilaterally. Second, the fractures are also orid
rather than solitary in nature. Third, clinical suspicion of frac-
ture was low with few signs of inammation. And nally, in
1 case plain lms of the extremities failed to detect any frac-
ture, with MRI providing the nal diagnosis. This paper high-
lights the importance of including insufciency fracture in
one’s differential, in patients with longterm RA presenting
with pain.
https://doi.org/10.1016/j.radcr.2018.05.016
1930-0433/© 2018 The Authors. Published by Elsevier Inc. on behalf of University of Washington. This is an open access article under the
CC BY-NC-ND license. ( http://creativecommons.org/licenses/by-nc-nd/4.0/ )

856 Radiology Case Reports 13 (2018) 855–861
Case 1
Presentation
Case 1 is a 66-year-old lady with seropositive RA treated with
methotrexate for over 20 years. In September 2015, her arthri-
tis became active and she had synovitis affecting her ankles
and feet. She received a steroid injection and the methotrex-
ate was increased. She was also commenced on alendronate,
as she had osteoporosis with a T score of −2.6 in the femoral
neck and −2.7 in the spine, with a 16% 10 year probability
of major fracture using fracture risk assessment tool assess-
ment. Calcium and vitamin D levels were normal. The in-
ammation improved, but the pain in the ankles and feet in-
creased. X-rays of the right foot in February 2016 showed os-
teopenia and 4th and 5th metatarsophalangeal (MTP) sublux-
ation and degenerative change and she was referred for podia-
try assessment for presumed mechanical foot pain. In March
2016, she was complaining of severe pain in the feet, worse
on the right with difculty weight bearing and was seen by
an orthopaedic consultant in April. On examination, she had
tenderness particularly over the ankle and subtalar joints and
Achilles tendons but the joints were not hot, red, or swollen.
Investigation
She was referred to radiology for MRI of both feet and/or ul-
trasound guided steroid injections. An MRI of both feet was
performed. MRI of the right foot showed multiple fractures in
the ankle and foot with extensive high signal change noted
within the distal tibia, posterior talus, cuboid, and calcaneus
with corresponding serpentine low signal lines noted in these
respective bones ( Figs. 1a ,2,3 ). MRI of the left foot showed ex-
tensive marrow oedema in the distal tibia, calcaneus, talar
neck, navicular, and proximal phalanx of the great toe with
corresponding low signal lines in the distal tibia, calcaneus,
and navicular ( Fig. 2 ). Further high signal changes were noted
within the lateral cuneiform. In short, she had orid bilateral
insufciency fractures of the foot and ankle ( Fig. 1b ).
Fig. 1a –Case 1—Sagittal T1 image of right foot and ankle.
Serpentine low signal changes within the distal tibial
metaphysis (white arrowhead) and navicular (white arrow).
Fig. 1b –Case 1—Sagittal T1 images of left foot and ankle
showing low signal serpentine line in keeping with
fractures in the distal tibial metaphysis (white arrow),
posterior calcaneus (black arrow), and talar neck (white
arrowhead).
Treatment
From an orthopaedic perspective, she was managed conser-
vatively using an air-cast boot for 3 months. Repeat MRI
at 3 months showed healing fractures, following which she
was discharged by orthopaedics. She remains under regular
rheumatology follow up.
Case 2
Presentation
Case 2 is a 75-year-old lady who was diagnosed with seroneg-
ative rheumatoid arthritis in 2003 at a center elsewhere and
was seen in our department in March 2017. She was taking
methotrexate 20 mg once weekly and had not received any
corticosteroid treatment to our knowledge (previous clinical
records were unavailable). Tw o months previously she had de-
veloped increased right ankle pain and swelling, and had re-
ceived a course of antibiotics from the GP for an associated
infection and had recently been started on furosemide for on-
going swelling. On examination she had pitting oedema at the
ankle with reduced movement and mild tenderness but there
was no erythema. The furosemide was discontinued and an
ankle MRI was requested.
Investigation
MRI showed a complete healing insufciency fracture in the
lower one-third of the tibia as evidenced by high signal
change, periosteal reaction, callus formation, and subtle val-

Radiology Case Reports 13 (2018) 855–861 857
Fig. 2 –Case 1—Coronal STIR image of the right ankle
showing high signal changes within the distal tibial
metaphysis (thin arrow), lateral malleolus (thick arrow), and
orid oedematous changes within the calcaneous
(arrowhead) in keeping with the acute insufciency
fractures.
gus angulation ( Fig. 4 ). In addition, there were multilevel insuf-
ciency fractures of the lower third of the distal tibia, bula
and posterior talus, and the upper calcaneus ( Fig. 5 ). Several
days later she went on to have an ultrasound and a plain ra-
diograph of the right foot. Ultrasound noted ankle joint effu-
sion or the appearance of synovitis as well as gross subcu-
taneous oedematous changes. X-ray showed healing insuf-
ciency fracture of the right lower one-third of the distal tibia
with some callus formation on the lateral aspect as well as
healing fractures of the distal one-third of the bula and sub-
chondral bone of the right distal tibia. While generalized os-
Fig. 3 –Case 1—Long axis axial T1 image showing low
signal changes within the right calcaneous (white arrow),
and proximal phalanx of the great toe showing multiple
fractures (arrowhead) in keeping with oedematous change
associated with serpentine fracture lines.
teopenia of the bones was noted, the orid insufciency frac-
tures of the hindfoot were not identiable ( Fig. 6 ).
Treatment
She was commenced on oral bisphosphonate treatment at di-
agnosis of her insufciency fractures. She was treated conser-
vatively in an air-cast boot. She has been discharged by or-
thopaedics and remains under rheumatology follow up.

858 Radiology Case Reports 13 (2018) 855–861
Fig. 4 –Case 2—Coronal STIR image of right foot and ankle
showing a complete healing insufciency fracture in the
lower one-third of the tibia as evident by high signal
change, periosteal reaction, callus formation, and subtle
valgus angulation (white arrow). Insufciency type fracture
within the distal tibia as evident by serpentine high signal
line (arrowhead).
Case 3
Presentation
Case 3 is a 74-year-old woman diagnosed with seropositive
rheumatoid arthritis in 2009. She also had bilateral knee re-
Fig. 5 –Case 2—Sagittal T1 image of right foot showing
serpentine low signal line in the inferior talus to the
subtalar joint (thin arrow) with a similar line in the
posterosuperior aspect of the calcaneous (arrowhead), and
distal tibia (thick arrow) suggestive of further pathological
fractures.
placements for degenerative joint disease and a history of me-
chanical foot pain for which she had seen a podiatrist. Her
arthritis was controlled on methotrexate 15 mg once weekly
but in 2016 she started to have ares affecting the ankles and
feet, the methotrexate was increased, hydroxychloroquine
and sulfasalazine added and she received several steroid in-
jections and a course of oral steroids. In March 2017, her dis-
ease activity had improved but she was still complaining of
pain in the feet with synovitis at 2 metacarpal phalangeal
joints and the ankles were noted to be warm. X-rays and an
MRI of the feet were requested.
Investigation
She went on to have plain radiographs of both feet. While
generalized osteopenia and mild-to-moderate degenerative
changes in the bilateral rst MTPJs and mid foot joints were
noted there was no sign of fracture ( Fig. 7 ). She went on to have
an MRI. Left foot MRI showed extensive high signal changes
within the posterior aspect of the calcaneus, with subchon-
dral collapse and corresponding low signal line on T1 imaging
in keeping with pathological fracture of the calcaneus ( Fig. 8 ).
Less orid high signal changes were noted within the nav-
icular, accompanied by a low signal line noted on T1 imag-
ing in keeping with a second insufciency fracture. Further
oedematous changes were noted within the proximal aspect
of the cuboid in keeping with microtrabecular fracture. Fur-

Radiology Case Reports 13 (2018) 855–861 859
Fig. 6 –Case 2—Anteroposterior and lateral plain
radiographs of right ankle showing healing insufciency
fracture of the right lower one-third of the distal tibia with
some callus formation on the lateral aspect (thin arrow).
Alongside healing fractures of the distal one-third of the
bula (thick arrow) and subchondral bone of the right distal
tibia (arrowhead). Generalized osteopenia of the bones
noted. Multiple fractures of the hindfoot bones are
unidentiable.
Fig. 7 –Case 3—plain radiographs of both feet showing
generalized osteopenia and no erosive arthropathy. There
is increased sclerosis of bilateral navicular bones
(arrowheads).
Fig. 8 –Case 3—left foot long axis axial T1 image showing
low signal line in keeping with pathological fracture of the
calcaneous (white arrow). Oedematous changes noted
within the proximal aspect of the cuboid in keeping with
microtrabecular fracture and proximal aspect of the fth
metatarsal base (arrowhead).
ther areas of patchy oedematous changes in the bone marrow
were noted within the distal tibia, lateral malleolus, proximal
aspect of the fth metatarsal base, and in the intermediate
cuneiform.
Treatment
She went on to have a dual energy X-ray absorptiometry scan
that surprisingly showed a T score of −1.3 at the hip (os-

860 Radiology Case Reports 13 (2018) 855–861
teopenia) and T score of −0.7 at the spine (normal). Vitamin
D was borderline low at 54 with normal calcium levels. Vita-
min D was replaced and alendronic acid commenced. She was
managed conservatively in an air-cast boot following which
she was discharged from orthopaedics. She remains under
rheumatology follow up.
Discussion
Rheumatoid arthritis is a chronic autoimmune inammatory
condition of unclear aetiology. It is thought to be a product
of genetic susceptibility and environmental triggers [1,2] . A
conservative estimate for RA prevalence in the UK is 1.16% in
women and 0.44% in men [3] . RA primarily affects the joints.
Synovitis causes a symmetrical destruction of the articular
cartilage leading to pain and loss of movement. Chronic joint
inammation associated with RA is known to cause local os-
teoporosis in subchondral bone. However RA sufferers are at
increased risk of osteoporosis at sites distant to joint inam-
mation. The reason behind this is thought to be due to the
systemic nature of inammation present in RA. In healthy
bones there is a balance between bone resorption and forma-
tion. The inammatory changes associated with RA decouple
these forces, leading to an overall bone loss with cancellous
bone being most affected [4–6] . Inammatory cytokines in-
cluding TNF αand interleukins 1, 7, 11, and 17 have been im-
plicated in this pathophysiology [4] . These cytokines increase
expression of receptor activator of nuclear factor kappa-B lig-
and which binds to RANK initiating bone resorption [7] . The
rate of bone formation cannot keep pace with the rate of bone
resorption leading to generalized weakening of the bone, pre-
disposing RA patients to insufciency fractures secondary to
osteoporosis [8] . In addition pain, reduced range of movement
and the increased basal metabolic rate associated with RA
contribute to reduced loading of the bone and muscle atro-
phy further predisposing patients to osteoporosis [7] . Tre a t -
ments for RA also increase bone loss. Glucocorticoids which
are used long-term to suppress immune mediated inamma-
tion reduce bone strength and stiffness [4,9] . While high level
evidence is lacking, methotrexate osteopathy has also been
implicated in increasing susceptibility to insufciency frac-
tures in RA patients via osteoblast inhibition [10] .
Insufciency fractures are a type of stress fracture caused
by subthreshold trauma to diseased bone. Loss of trabecular
bone reduces the elastic resistance of the bone, predisposing
it to fracture [11] . Insufciency fractures are commonly seen
in bones with a high proportion of trabecular bone such as the
vertebrae, femur, pelvis, and irregular bones of the foot [5,12] .
This type of fracture of the foot and ankle usually involves
the second metatarsal and the calcaneus, with the navicular
and talus less commonly involved [13] . If early insufciency
fractures are suspected, MRI and bone scans are preferable
imaging modalities. On plain radiograph insufciency frac-
tures may show as radiolucent lines in osteopenic bone, but
in cases of recent fracture appearances may be normal. MRI
has equal sensitivity to bone scans but higher specicity for
locating the exact site and aetiology of the abnormality [14] .
MRI is adept at identifying soft tissue abnormalities, for ex-
ample in the bone marrow [15] . Features suggestive of insuf-
ciency fracture on MRI include ill-dened low bone marrow
signal on T1-weighted images with a hypointense serpentine
intramedullary fracture line which may extend to the cortex
[11,16] . T2-weighted images show high bone marrow signal
and may be accompanied by a hypointense periosteal callus
[13] . Changes in bone marrow signal are caused by oedema
or haemorrhage, in either case the borders are poorly dened
[13] . The fracture line and surrounding oedema become more
apparent several weeks after injury [13] . Contrast enhance-
ment, typically with gadolinium, can help delineate lesions.
There are case reports documenting single insufciency
fractures, for example, Straaton et al.‘s case series reported
3 patients with RA and distal tibial fracture. Similarly, atyp-
ical fractures have been identied at the subtrochanteric
and/or femoral diaphysis in patient with RA, taking glucocor-
ticoids, methotrexate, and alendronate. Multiple fractures of
the pelvis and lumbrosacral region were reported by Fukun-
ishi et al. [17] . There are infrequent case reports in the medical
literature describing insufciency foot fractures in more than
1 bone in patients with RA. Spina et al. report 1 case of talar
and calcaneal insufciency fracture in a patient with RA [13] .
Plain radiograph suggested a fracture in the foot, which was
conrmed by MRI and bone scintigraphy. The cases we present
are unusual in that they show multiple fractures in numerous
bones in the foot and ankle, in 2 cases bilaterally. As far as the
authors are aware, no other case series has reported similar
presentations. A further salient feature is that plain lm did
not detect any fracture, despite MRI ndings showing orid
and multiple insufciency fractures. From this we can appre-
ciate the importance of investigating patients with MRI, which
is superior at identifying early changes associated with frac-
ture, such as bone marrow oedema.
Informed consent statement
Written informed consent was obtained from all 3 patients for
publication of this case report, including accompanying im-
ages.
Learning points
• Clinicians need to consider insufciency fractures in their
differential diagnosis when patients present with reduced
functional status or joint or bone pain, with a background
of RA and corticosteroid use.
• Complaints of new or worsening symptoms need to be in-
vestigated as symptoms of insufciency fracture are dif-
cult to differentiate from an exacerbation of RA. Delay in
diagnosis can lead to worse outcomes.
• Plain radiograph may fail to elucidated pathology, so con-
sideration of MRI is strongly advised for patients with RA
complaining of pain or loss of functional status without a
history of trauma.

Radiology Case Reports 13 (2018) 855–861 861
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