SCLEREDEMA DIABETICORUM WITH UNUSUAL PRESENTATION AND FATAL OUTCOME

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Abstract

We present a case of Scleredema Diabeticorum (SD) in a patient with diabetic neuropathy and restrictive respiratory disease with unusual skin lesion distribution. The onset of dermatologic symptoms heralded a progressive respiratory disease with constrictive component. Painful diabetic neuropathy was noteworthy and difficult to relieve. Predominantly, distribution of the skin lesions on the thighs makes the case exceptional. T2-weighted MRI showed abnormal hyperintensities along the muscles of the thighs in correspondence with the skin lesions. Gait and respiratory symptoms progressively worsened. After a transient remitting period, he developed sudden shortening of breath, arrested and expired at home.The atypical distribution of the skin lesions with further involvement of underlying muscles plus concomitant polyneuropathy and respiratory constrictive disease with sudden death is quite unusual and aggressive presentation of SD.

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Most cited references 9

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The spectrum of neuropathy in diabetes and impaired glucose tolerance.

D Cornblath, Rachel Sumner, M Polydefkis … (2003)

To compare the neuropathy associated with impaired glucose tolerance (IGT) and diabetes mellitus (DM) determined by oral glucose tolerance testing (OGTT). Patients with peripheral neuropathy of unknown cause were prescribed OGTT. Duration of neuropathic symptoms, neuropathic pain, neuropathy classification, nerve conduction test results, and intraepidermal nerve fiber densities (IENFD) were compared between IGT and DM groups. Seventy-three patients completed OGTT; 41 (56%) had abnormal results. Of these 41 patients, 26 had IGT and 15 had DM. Patients with IGT had less severe neuropathy than patients with diabetes, as measured by sural nerve amplitudes (p = 0.056), sural nerve conduction velocities (p = 0.03), and distal leg IENFD (p = 0.01). Patients with IGT had predominantly small fiber neuropathy, compared to patients with DM (p = 0.05), who had more involvement of large nerve fibers. The neuropathy associated with IGT is milder than the neuropathy associated with DM. Small nerve fibers are prominently affected and may be the earliest detectable sign of neuropathy in glucose dysmetabolism. OGTT is appropriate in patients with idiopathic neuropathy.

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Multicenter study of the incidence of and predictive risk factors for diabetic neuropathic foot ulceration.

A L Carrington, L Vileikyte, Simon J. Boulton … (1998)

To investigate longitudinally prognostic factors for foot ulceration in a large population of diabetic patients with established neuropathy. A double-blind multicenter study of a potential new agent for diabetic neuropathy provided the opportunity for this 1-year investigation since intervention demonstrated no efficacy in the condition. A total of 1,035 patients with NIDDM and IDDM were included. Inclusion criteria were vibration perception threshold (VPT) at the great toe > or = 25 V in at least one foot and < or = 50 V in both feet, normal peripheral circulation, and no previous foot ulceration. VPT and clinical components of the Michigan diabetic polyneuropathy (DPN) score were assessed at baseline and subsequent visits. After 1 year, the incidence of first foot ulcers for the total population was 7.2%. Neuropathy parameters were the same between the treatment and placebo groups at baseline and were unchanged at 1 year; therefore, baseline data were combined for multiple regression analysis. VPT, age, and Michigan DPN scores for muscle strength and reflexes were significant independent predictors for first foot ulceration (P < 0.01). For each 1-U increase in VPT values at baseline, the hazard of the first foot ulcer increased by 5.6%. Similarly, for each 1-U increase in muscle strength and reflex components of the Michigan DPN scores, the hazard of the first foot ulcer increased by 5.0%. Tests of VPT and Michigan DPN scores for muscle strength and reflexes are useful clinical predictors for foot ulceration in diabetic patients with established neuropathy. The rate of subsequent ulceration in the following year was alarmingly high, however, despite standardized foot care education at baseline and regular follow-up visits.

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Amyloidosis: recognition, confirmation, prognosis, and therapy.

M A Gertz, A Dispenzieri, M Q Lacy (1999)

Amyloidosis should be considered in any patient older than 40 years who has nephrotic syndrome, congestive heart failure (not on an ischemic basis), idiopathic peripheral neuropathy, or unexplained hepatomegaly. When a patient has one of these problems, immunoelectrophoresis and immunofixation of the serum and urine should be done for the detection of a monoclonal light chain. If a monoclonal light chain is found, a diagnosis usually can be established by amyloid stains performed on a bone marrow biopsy specimen or a subcutaneous fat aspirate. The presence or absence of cardiac involvement with amyloid is the most important prognostic factor. Treatment can range from observation to oral chemotherapy to hematopoietic stem cell transplantation. A practical understanding of the mechanisms underlying this disease can lead to prompt diagnosis and early therapeutic intervention.

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Author and article information

Journal

Journal ID (nlm-ta): Indian J Dermatol

Journal ID (publisher-id): IJD

Title: Indian Journal of Dermatology

Publisher: Medknow Publications (India )

ISSN (Print): 0019-5154

ISSN (Electronic): 1998-3611

Publication date (Print): 2008

Volume: 53

Issue: 4

Pages: 217-219

Affiliations

From Department of Neurology, Hamad General Hospital, Doha, State of Qatar

[1 ] From Department of Radiology, Hamad General Hospital, Doha, State of Qatar

Author notes

Address for correspondence: Dr. Francisco J Ruiz Miyares, Department of Neurology, HMC, POB 3050, Doha, State of Qatar, Dr. Francisco Ruiz Miyares, E-mail: fruizelo@ 123456gmail.com

Article

Publisher ID: IJD-53-217

DOI: 10.4103/0019-5154.44798

PMC ID: 2763766

PubMed ID: 19882043

SO-VID: bf345495-45c9-41bf-bb93-974fd2857c33

License:

This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

SCLEREDEMA DIABETICORUM WITH UNUSUAL PRESENTATION AND FATAL OUTCOME

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Abstract

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Karger: Dermatology

Most cited references 9

The spectrum of neuropathy in diabetes and impaired glucose tolerance.

Multicenter study of the incidence of and predictive risk factors for diabetic neuropathic foot ulceration.

Amyloidosis: recognition, confirmation, prognosis, and therapy.

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