|
?2LL 1eEe2t0tTtt0eTer7rE tsoR t Soth TtehO Ee dT EiHtodErit EoDr ITOR Dercum’s disease (adiposis
dolorosa): a report of improvement
with infliximab and methotrexate
Editor
A 50 year-old, Caucasian woman with a history of
ankylosing spondylitis and insulin-dependent diabetes
had a 5-year history of increasingly painful bulky tumours.
Surgical excision of a tumour from her right buttock in
2002 revealed a lipoma. In 2003 she was diagnosed with
Dercum’s disease. In July 2004 the patient weighed 362
lbs, with a body mass index of 54.5. Physical examination
revealed multiple, tender, subcutaneous tumours on her
trunk and extremities.
The patient was started on infliximab infusions in
September 2004 at a dose of 3 mg/kg to control her ankylosing
spondylitis. The intervals between infusions were
gradually increased from 2 to 6 weeks. In November 2004
the patient reported improvement in her polyarthritis, an
incidental improvement in the pain from her tumours,
and an 18-lb weight loss. Methotrexate was added to her
regimen, starting at 10 mg/week and then increasing
to 20 mg/week. A month later, the patient reported an
additional 10-lb weight loss. Over the next 6 months the
redness and size of her lipomas continued to regress, and
there was a decrease in subjective pain and in insulin
requirements.
Infliximab and methotrexate were discontinued in May
2005 because of bilateral cellulitis of the lower extremities.
Since this time her pain has recurred, she has gained
15 lb in weight and there has been an increase in her
insulin requirements.
Treatment options for Dercum’s disease are few, and are
often ineffective. Weight loss and common analgesics
have been tried with little to no benefit. Intravenous lidocaine
and low-dose, daily prednisone have helped in only
some patients.1,2 Surgery and liposuction may temporarily
reduce the number and size of the lipomas as well as
the associated pain, but the tumours usually recur over
time.3
Our patient is the first reported case of improvement in
Dercum’s disease with infliximab therapy. It is unclear
whether the patient’s improvement derived solely from
the infliximab, or if methotrexate played an additive role.
However, we do know her initial improvement occurred
on infliximab alone. TNF-alpha has recently been reported
to play a role in the release of circulating free fatty acids,
leading to a decrease in peripheral insulin sensitivity.4 Infliximab,
as a TNF-alpha inhibitor, may improve Dercum’s
disease by inhibiting this process. Metabolic studies have
shown both alterations in fatty acid metabolism and a
peripheral insulin resistance in Dercum’s patients, suggesting
Hits: 2025
|