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CASE REPORT (AUTHOR'S SERIES)
A 26-year-old Caucasian woman, para 0 gravida 1,
developed the rapid onset of severe bilateral hip and
groin pain at 34 weeks of gestation. She was known to
have mild OI without dentinogenesis imperfecta (type
IA). Her father had type I OI and had died at age 25 fol-
lowing an automobile accident. Her sister and paternal
aunts and uncles also have type I OI.
A cesarean section was performed at 36 weeks' ges-
tation. Her son who weighed 6 lb 12 oz had a fractured
clavicle at delivery.
Bilateral hip pain radiating to the groin which
increased with hip motion persisted and progressed dur-
ing the immediate postpartum period. At 3 weeks post-
partum she was confined to bed and a wheelchair and
could not bear weight on her lower extremities. She was
unable to climb stairs. Standard X-rays of the pelvis and
hips were negative for fracture.
Physical examination at 3 weeks postpartum did not
show skeletal deformities. She had blue sclerae and nor-
mal dentition. There was marked pain on palpation lat-
erally over each hip, more marked on the right. Pain
markedly restricted right hip flexion or internal and exter-
nal rotation. Erythrocyte sedimentation rate (ESR) was
105 mm/hr, serum total alkaline phosphatase 152 IU/L
(normal 30-120), a second void urine for n-telopeptide
(NTx) excretion was 133 nMBCE/nM creatinine (normal
14-87). Magnetic resonance imaging (MRI) T1 images
showed a small right hip effusion. MRI T2-weighted
images demonstrated increased signal consistent with
bone marrow edema in the right and left femoral heads,
the right anterior pubis and superior sacrum.
Pamidronate was administered intravenously at a
dose of 60 mg twice at 3-week intervals, but due to vein
irritation following the second infusion, treatment was
changed to oral alendronate, 10 mg daily. At 4 weeks
from the start of treatment she had experienced a rapid
and substantial decrease in hip pain and was weight
bearing by 5 weeks at which time sedimentation rate had
decreased to 32 mm/hr. She was able to walk upstairs by
the sixth week.
Weitzel et al., in 2000, described a 30-year-old woman
with type I OI who presented with pain in her left groin
and difficulty in walking 5 days after undergoing a
cesarean section at 29 weeks. Plain radiographs showed
“osteopenia” at the left hip. Symptoms resolved at 6
months. In this person, at 33 weeks in a second preg-
nancy pain occurred in the right hip. Both sintigraphic
scan and MRI confirmed the presence of transient osteo-
porosis now affecting the right hip.
4
The previously
involved left hip was normal by MRI. Symptoms again
resolved in 6 months with conservative treatment.
A 32-year-old woman with OI type I developed tran-
sient osteoporosis involving her right hip during the late
third trimester.
26
Hip pain responded to conservative
treatment. Following weaning, transient osteoporosis of
the contralateral hip developed that did not respond to
conservative treatment. Subsequent treatment with pami-
dronate and a prostaglandin I(2) agonist, iloprost, led to
resolution of the hip pain.
Thus, each of these cases illustrates recurrent and
bilateral hip involvement complicating pregnancy and
the postpartum period in OI. It is possible that the occur-
rence of transient osteoporosis surrounding pregnancy
may be secondary to pregnancy-related demineralization
or the occurrence of microfractures occurring at the later
stages of gestation.
3
Core Decompression in Transient Osteoporosis
In a series of nine patients with transient osteoporo-
sis involving ten hips aggregate of 4 months duration,
bone marrow pressure measurements and intraosse-
ous venography showed pathological values rang-
ing from 20 to 80 mm Hg at the trochanter-neck-head
of the femur and intraosseous venography was posi-
tive. Following core decompression pain was relieved,
patients returned to work by 6 weeks and MRI of the
involved hip returned to normal at 3 months.
21
the onset of conservative treatment, the patient had no
more pain.
Illustrative of a less than satisfactory result is the
report of Weitzel, who reported a case of a 30-year-
old woman with type I OI who experienced tran-
sient osteoporosis 5 days postpartum.
4
Treatment
was restricted weight bearing and analgesics. By 3
months, her symptoms were only slightly improved.
At 6 months, however, her symptoms had completely
resolved. Sixteen months later, the patient was 33
weeks into a second pregnancy and developed the
same symptoms in the opposite hip. Blood and radio-
graphs were normal. MRI showed characteristic find-
ings of TMO. Again, she was treated with analgesics
and restricted weight bearing. By 6 months her symp-
toms had much improved.
Pharmacological Therapy
Proposed interventions include the use of various
analgesics and non-steroidal anti-inflammatory agents.
