OBSERVATIONS
Implantable Pump
Therapy Restores
Metabolic Control
and Quality of Life in
Type 1 Diabetic
Patients With
Buschke’s
Nonsystemic
Scleroderma
B
uschke’s nonsystemic scleroderma
is an uncommon dermatosis charac-
terized by thickened and indurate
skin of unknown origin, mostly affecting
upper parts of the body but also the ab-
dominal area. While diabetes is rarely as-
sociated, subcutaneous insulin treatment
may be hardly feasible and effective be-
cause of incomplete absorption of insulin
(1).
After approval of our institutional
ethical committees, four type 1 diabetic
patients (one male and three female sub-
jects) affected by this condition received
Medtronic MiniMed implantable pumps
(Northridge, CA) for intraperitoneal insu-
lin delivery (2) between 1994 and 2004.
The patients, aged 45.2 2.9 years, had a
mean duration of diabetes of 33.5 1.3
years, including multiple microvascular
complications. Biopsies of the affected
skin, performed in three patients, con-
firmed expanded dermis with enlarged
collagen bundles and, in one case, visible
mucin. None had evidence of systemic
sclerosis by biopsy and other investiga-
tions at baseline or during follow-up. Skin
status was clinically assessed on a yearly
basis, diabetes complications surveyed as
required, and HbA
1c
(A1C) measured us-
ing a high-performance liquid chroma-
tography method (normal values 5.6%)
on a quarterly basis.
From implantation time, improve-
ment of glucose control was sustained as
shown by mean yearly A1C moving from
9.3 0.7% (baseline) to 7.9 1.3%
(2005). Microvascular complications re-
mained stable or decreased. All patients
experienced a dramatic decrease of skin
induration as early as a few months after
implantation. We found clinical improve-
ment of the skin aspect in terms of red-
ness, swelling, and induration, even if we
did not have tools at our disposal to mea-
sure skin elasticity.
Several published case reports de-
scribe either poorly effective therapeutic
options for Buschke’s scleroderma (3,4)
or the effective, albeit rather extreme, ra-
diation therapy (5). Poor glycemic control
may play a role in worsening of the skin
condition, resulting in a vicious cycle of
worsening control and worsening disease
driven by poor absorption of insulin (6).
Although not documented by pathologi-
cal or biochemical skin analyses, our find-
ings support previous hypotheses that
glucose excess causes impairment
through increased glycation alterations of
tissue collagen, which are involved in
Buschke’s scleroderma, and that this pro-
cess can be reversed with improved glu-
cose control. We suggest implantable
pump therapy as an option in patients
with type 1 diabetes affected by this un-
common skin condition when optimized
subcutaneous insulin treatment fails to
achieve acceptable blood glucose control.
SABINE BAILLOT-RUDONI, MD
1
DOMINIQUE APOSTOL, MD
2
GENEVIE ` VE VAILLANT, MD
1
JEAN-MARCEL BRUN, MD
1
ERIC RENARD, MD, PHD
2
ON BEHALF OF THE EVADIAC
STUDY GROUP
From the
1
Department of Endocrinology and Meta-
bolic Diseases, Bocage Hospital, Dijon, France; and
the
2
Endocrinology Department, Lapeyronie Hospi-
tal, Montpellier, France.
Address correspondence to Sabine Baillot-
Rudoni, MD, Department of Endocrinology and
Metabolic Diseases, Bocage Hospital, 21079 Dijon,
France. E-mail: sabine.rudoni@dijon.fr.
DOI: 10.2337/dc06-0582
© 2006 by the American Diabetes Association.
●●●●●●●●●●●●●●●●●●●●●●●
References
1. Jabbour SA: Cutaneous manifestations of
endocrine disorders: a guide for derma-
tologists. Am J Clin Dermatol 4:315–331,
2003
2. Broussolle C, Jeandidier N, Hanaire-
Broutin H, the EVADIAC Study Group:
French multicenter experience of im-
plantable insulin pumps. Lancet 343:514 –
515, 1994
3. Seyger MM, Van Den Hoogen FH, De
Mare S, Van Haelst U, De Jong EM: A pa-
tient with a severe scleroedema diabetico-
rum, partially responding to low-dose
methotrexate. Dermatology 198:177–179,
1999
4. Krasagakis K, Hettmannsperger U, Traut-
mann C, Tebbe B, Garbe C: Persistent
scleredema of Buschke in a diabetic: im-
provement with high-dose penicillin (Let-
ter). Br J Dermatol 134 :597–598, 1996
5. Bowen AR, Smith L, Zone JJ: Scleredema
adultorum of Buschke treated with radia-
tion. Arch Dermatol 139:780 –784, 2003
6. Buckingham BA, Uitto J, Sandborg C,
Keens T, Roe T, Costin G, Kaufman F,
Bernstein B, Landing B, Castellano A:
Scleroderma-like changes in insulin-de-
pendent diabetes mellitus: clinical and
biochemical studies. Diabetes Care 7:163–
169, 1984
Possible Problem
With Optipen Pro-1
Should diabetic patients continue
to use this product?
I
nsulin glargine is a modified basal in-
sulin analog that has been recently in-
troduced and is now available
worldwide. Insulin glargine is available in
3-ml cartridges that can be used with the
OptiPen Pro-1 (Sanofi-Aventis). The
Medicines and Healthcare Products Reg-
ulatory Agency (MHRA; executive agency
of the Department of Health in the U.K.,
which enhances and safeguards the health
of the public by ensuring the effectiveness
of medicines and medical devices) has re-
ceived a significant number of reports
concerning difficulties in the operation
and use of the OptiPen Pro-1 insulin pen
injection system. A fault was found with
some batches of the OptiPen Pro-1 sys-
tem, whereby the dosage button failed to
engage at the end of an injection (1).
A total of 32 type 1 diabetic patients
(age 17.0 4.4 years [mean SD]) on
multiple daily injection regimens, who
had been treated with insulin for at least
for 6 months and were insulin glargine
naive, were transferred from NPH insulin
to insulin glargine between May 2004 and
March 2005. Two patients without any
evidence of infection or of skipped insulin
doses had ketosis on the 3rd and 4th
month of insulin glargine treatment, re-
spectively. When the patients were ques-
tioned about the reasons for ketosis, they
stated leakage of insulin from the sides of
the pen during injection.
An inquiry form regarding OptiPen
Pro-1 was given to all subjects on the 6th
month of therapy. Leakage from the sides
of the pen was noted by 58.8% of sub-
jects, and a problem with the dosage but-
ton not locking when it was fully
depressed following injection of the de-
sired dose of insulin was reported by
38.2%. The patients were asked to rate
L E T T E R S
1710 DIABETES CARE, VOLUME 29, NUMBER 7, JULY 2006