Skip to main content
Key Specialties
Cardiology Diabetology Endocrinology Gastroenterology Geriatrics and Gerontology Gynecology and Obstetrics Hematology Infectious Disease Internal Medicine Nephrology Neurology Oncology Pediatrics Respiratory Medicine Rheumatology Surgery
Congress Coverage
2024 ACC Congress 2023 ESMO Congress 2023 EASD Congress 2023 ERS Congress 2023 ESC Congress
Clinical Collections
Advances in Alzheimer’s

At a glance: The STEP trials

A round-up of the STEP phase 3 clinical trials evaluating semaglutide for weight loss in people with overweight or obesity.
ADVANCED SEARCH
Log in
Top
Clinical Rheumatology
Published in: Clinical Rheumatology 1/2012

01-01-2012 | Case Based Review

D-penicillamine in the treatment of eosinophilic fasciitis: case reports and review of the literature

Authors: CU Manzini, M Sebastiani, D Giuggioli, A Manfredi, M Colaci, AM Cesinaro, C Ferri

Published in: Clinical Rheumatology | Issue 1/2012

Login to get access

Abstract

Eosinophilic fasciitis (EF) is a rare disease characterized by symmetrical thickness and hardening of the skin, especially localized to forearms and thorax, with eosinophilia. Corticosteroids represent the first-line therapy, even if some patients are scarcely responsive and/or may develop important side effects due to long-term treatment. Here, we describe three cases of EF, two of them refractory to previous steroid therapy, successfully treated with D-penicillamine. The present clinical observations together with the updated review of the literature suggest usefulness of D-penicillamine in EF patients, as well as its potential steroid-sparing value.
Literature
1.
Shulman LE (1974) Diffuse fasciitis with hypergammaglobulinemia and eosinophilia: a new syndrome? J Rheumatol 1:S46
2.
Miret C, Nonell F, Cervera R, Rodrigues FE, Torres M (2003) IgA nephropathy associated with eosinophilic fasciitis: report a case. Clin Exp Rheum 21:268
3.
Kirschstein M, Helmchen U, Jensen R, Kuster RM, Lehmann H (1999) Kidney involvement in a 17-year old boy with eosiniphilic fasciitis. Clin Nephrol 52:183–187PubMed
4.
Takeda SI, Takarakura E, Fukui Y (1995) Tubulointerstitial nephritis in a patient with eosinophilic fasciitis and IgA nephropathy. Nephron 69:314–317PubMedCrossRef
5.
Rizzo S (2002) Eosinophilic pleuropericarditis and fasciitis. A new case Monaldi Arch Chest Dis 57:311–313
6.
Killen JW, Swift GL, White RG (2000) Eosinophilic fasciitis with pulmonary and pleural involvement. Postgrad Med 76:36–37CrossRef
7.
Cayla J, Rondier J, Thibierge M, Emerit I, Malherbe M, Guiraudon C (1981) Un cas de fasciite à eosinophiles avec péricardite. Rev Rhum Mal Osteoartic 48:809–812PubMed
8.
Giordano M, Ara M, Cicala C, Valentini G, Chianese U, Vatti M (1980) Eosinophilic fasciitis. Am J Med 93:645–646
9.
Rosenthal J, Benson MD (1980) Diffuse fasciitis and eosinophilia with symmetric polyathritis. Ann Int Med 92:507–509PubMed
10.
Moriguchi M, Terai C, Kuroki S, Tanaka E, Someya N, Tsunoda Y, Kashivazaki S (1998) Eosinophilic fasciitis complicated with peripheral polyneuropathy. Intern Med 37:417–420PubMedCrossRef
11.
Farrell AM, Ross JS, Bunker CB (1999) Eosinophilic fasciitis associated with autoimmune thyroid disease and myelodisplasia treated with pulsed methylprednisolone and antihistamines. Br J Dermatol 140:1185–1188PubMed
12.
Khanna D, Verity A, Grossman JM (2002) Eosinophilic fasciitis with multiple myeloma. Ann Rheum Dis 61:1111–1112PubMedCrossRef
13.
Masuoka H, Kikuchi K, Takahasi S, Hayashi N, Farue M (1998) Eosinophilic fasciitis associated with low-grade T cell Lymphoma. Br J Dermatol 139:928–930PubMedCrossRef
14.
Junca J, Cuxart A, Tural C, Ojanguren I, Flores A (1994) Eosinophilic fasciitis and non-Hodgkin lymphoma. Eur J Haematol 52:34–36
15.
Garcia VP, de Quiros JF, Caminal L (1998) Autoimmune hemolitic anemia with eosinophilic fasciitis. J Rheumatol 25:1864–1865PubMed
16.
Backmayer C, Monge M, Dhote R, Sanguina M, Aractingi S, Mougeot-Martin M (1999) Eosinophilic fasciitis following idiopathyc trombocytopenic purpura, autoimmune hemolytic anemia and Hashimoto’s disease. Dermatology 199:282CrossRef
17.
Gallardo F, Vadello M, Mitjavila F, Servitje O (1998) Systemic lupus erithematosus after eosinophilic fasciitis:a case report. J Am Acad Dermatol 39:283–285PubMedCrossRef
18.
Cervini C, Scuppa L, Grassi W, Piergiacomi G (1982) La fasciite à eosinophiles: description d’un cas clinique avec syndrome de Sjogren et syndrome du tunnel carpien. Rev Rhum 25:1180–1185
19.
Manzini CU, Dotoli R, Villani M, Mascia MT, Manzini E (1986) La fascite eosinofila. Contributo casistico (Considerazioni patogenetiche e nosografiche). Boll Soc Med Chir Modena 1–2:53–62
20.
Liou CH, Huang GS, Taylor JA, Juan CJ, Gao HW, Chen CY (2003) Eosinophilic fasciitis in a military recruit: MRI evaluation with clinical correlation. Skeletal Radiol 32:152–157CrossRef
21.
Velasquez X, Gutierrez MA, Rosenberg H, Figueroa F, Bronstein E, Jacobelli S (2002) Eosinophilic fasciitis; report of 3 cases. Rev Med Chil 130:206–14
22.
Barnes L, Rodnan GP, Medsger TA Jr, Short D (1979) Eosinophilic fasciitis. A pathologic study of twenty cases. Am J Pathol 96:493–517PubMed
23.
Romero AG, Fernandez JG, Calataynd JC (2001) Eosinophilic fasciitis associated with simple traumatism. Acta Dermatovenereol Croat 9:287–290
24.
Hamilton M (1991) Eosinophilic fasciitis associated with l-tryptofan ingestion. J Rheum Dis 50:55–56CrossRef
25.
Choquet-Kastylevvski G, Kanikatis J, Dumas V, Descotes J, Faure M, Claudy AR (2001) Eosinophilic fasciitis and simvastatin. Arch Int Med 161:1456–1457CrossRef
26.
Cantini F, Salvarani C, Olivieri I, Padula A, Senesi C, Bellandi F, Truglia et al (1998) Possible association between fasciitis and subcutaneous heparin use. J Rheumatol 25:383–385PubMed
27.
Smith JD, Chang KL, Gums JG (1988) Possible lansoprazole-induced eosinoplic sindrome. Ann Pharmacother 32:196–200
28.
Solomon G, Barland P, Rifkin H (1982) Eosinophilic fasciitis responsive to cimetidine. Ann Intern Med 97:547–549PubMed
29.
Hayashi N, Igarashi A, Matsuyama T, Harada T (2000) Eosinophilic fasciitis following exposure to trichloroethylene: successful treatment with cyclosporin. Brit J Dermatol 142:830–832CrossRef
30.
Valencia IC, Chang A, Kirsner RS, Kerdel FA (1999) Eosinophilic fasciitis responsive to treatment with pulsed steroids and cyclosporine. Int J Dermatol 38:369–372PubMed
31.
Kato Y, Naito Y, Narita Y, Kuzuhara S (1997) D-penicillamine-induced myasthenia gravis in a case of eosinophilic fasciitis. J Neurol Sci 146:85–86PubMedCrossRef
32.
Caspi D, Fishel R, Varon M, Yona E, Baratz M, Yaron M (1982) Multisystem presentation of eosinophilic fasciitis. Rheumatol Rehabil 21:218–221PubMedCrossRef
33.
Martínez-Osuna P, Wallach PM, Seleznick MJ, Levin RW, Silveira LH, Jara LJ, Espinoza LR (1991) Treatment of the eosinophilia-myalgia syndrome. Semin Arthritis Rheum 21:110–121PubMedCrossRef
34.
Stork J, Nĕmcová D, Hoza J, Kodetová D (1996) Eosinophilic fasciitis in an adolescent girl with lymphadenopathy and vitiligo-like and linear scleroderma-like changes. A case report Clin Exp Rheumatol 14:337–344
35.
Bischoff L, Derk CT (2008) Eosinophilic fasciitis: demographics, disease pattern and response to treatment: report of 12 cases and review of the literature. Int J Dermatol 47:29–35PubMedCrossRef
36.
Epler GR, Snider GL, Gaensler EA, Cathcart ES, FitzGerald MX, Carrington CB (1979) Bronchiolitis and bronchitis in connective tissue disease. A possible relationship to the use of penicillamine. JAMA 242:528–532PubMedCrossRef
37.
Kovalev VM, Krivenko ZF New aspects of the pathogenesis and treatment of Schulmann's eosinophilic fasciitis. Vestn Dermatol Venerol 1989;12:13–5
38.
Farrington ML, Haas JE, Nazar-Stewart V, Mellins ED (1993) Eosinophilic fasciitis in children frequently progresses to scleroderma-like cutaneous fibrosis. J Rheumatol 20:128–132PubMed
39.
Coyle HE, Chapman RS (1980) Eosinophilic fasciitis (Shulman syndrome) in association with morphoea and systemic sclerosis. Acta Derm Venereol 60:181–182PubMed
40.
Pouplin S, Daragon A, Le Loet X (1998) Treatment of eosinophilic fasciitis with methotrexate. J Rheumatol 25:606–607PubMed
41.
Uckum A, Sipahi T, Akgun D, Oksai A (2002) Eosinophilis fasciitis successfully treated with oral hydroxyzine: a new therapeutic use of an old drug? Eur J Pediatr 161:118–119CrossRef
42.
Boni-Sadr F, Leautez S, El Kouri D, Hamidou M, Barrier JH, Raffi F (2000) Intéret des immunoglobulines au cours de la fasciite de Shulman. La Presse Méd 6:307
43.
Romano C, Rubegni P, De Aloe G, Stanghellini E, D’Ascenzo G, Andreassi L, Fimiani M (2003) Extracorporeal photochemotherapy in the treatment of eosinophlic fasciitis. Eur Acad Dermatol Venereol 17:10–13CrossRef
44.
Schiener R, Behrens-Williams SC, Gottlober P, Pillekamp H, Peter RU, Kerscher M (2000) Eosinophilic fasciitis treated with psoralen-ultraviolet. A bath photochemotherapy. Br J Dermatol 142:804PubMedCrossRef
45.
Cetkovsky P, Koza V, Cetkovska P, Svojgrova M (1998) Successful treatment of severe Shulman’ syndrome by allogenic bone marrow transplantion. Bone Marrow Transpl 21:637CrossRef
46.
Neumeister MW, Robertson GA (1998) Therapeutic fasciectomy for eosinophilic fasciitis. Ann Plasturgery 41:208–210CrossRef
47.
Naschitz JE, Boss JH, Misselevich I, Yeshurun D, Rosner I (1996) The fasciitis-panniculitis syndromes. Clinical and pathologic features. Medicine 75:6–16PubMedCrossRef
48.
Gardner H, Strehlow D, Bradley L, Widom R, Farina A, de Fougerolles A et al (2004) Global expression analysis of the fibroblast transcriptional response to TGFγά. Clin Exp Rheumatol 22:S47PubMed
49.
Gabrielli A, De Nictolis M, Campanati G, Cinti S (1983) Eosinophilic fasciitis: a mast cell disorder? Clin Exp Rheumatol 1:75–80PubMed
50.
Toquet C, Hamidou MA, Renaudin K, Jarry A, Foule P, Barbarot S, Laboisse C, Mussini J (2003) In situ immunophenotype of the inflammatory infiltrate in eosinophilic fascitis. J Rheumatol 30:1811–1815PubMed
51.
Mekori YA (2004) The mastocyte: the « other » inflammatory cell in immunopathogenesis. J Allergy Clin Immunol 114:15–21CrossRef
Metadata
Title
D-penicillamine in the treatment of eosinophilic fasciitis: case reports and review of the literature
Authors
CU Manzini
M Sebastiani
D Giuggioli
A Manfredi
M Colaci
AM Cesinaro
C Ferri
Publication date
01-01-2012
Publisher
Springer-Verlag
Published in
Clinical Rheumatology / Issue 1/2012
Print ISSN: 0770-3198
Electronic ISSN: 1434-9949
DOI
https://doi.org/10.1007/s10067-011-1866-3

Other articles of this Issue 1/2012

Clinical Rheumatology 1/2012 Go to the issue

Brief Report

Efficacy of leflunomide addition in relation to prognostic factors for patients with active early rheumatoid arthritis failing to methotrexate in daily practice

Original Article

Indirect comparison of etanercept, infliximab, and adalimumab for psoriatic arthritis: mixed treatment comparison using placebo as common comparator

Original Article

An overview of systematic reviews of complementary and alternative medicine for fibromyalgia

Brief Report

Re-challenge with Etanercept in patients with Etanercept-induced Neutropenia

Original Article

How common is methotrexate pneumonitis? A large prospective study investigates

Original Article

No effect of physiotherapy on the serum levels of adipocytokines in patients with ankylosing spondylitis
Obesity Clinical Trial Summary

At a glance: The STEP trials

Read more

A round-up of the STEP phase 3 clinical trials evaluating semaglutide for weight loss in people with overweight or obesity.

Developed by: Springer Medicine

Highlights from the ACC 2024 Congress

Year in Review: Pediatric cardiology

Pediatric Cardiology Video

Watch Dr. Anne Marie Valente present the last year's highlights in pediatric and congenital heart disease in the official ACC.24 Year in Review session.

Year in Review: Pulmonary vascular disease

Cardiopulmonary Comorbidity Video

The last year's highlights in pulmonary vascular disease are presented by Dr. Jane Leopold in this official video from ACC.24.

Year in Review: Valvular heart disease

Valvular Heart Disease Video

Watch Prof. William Zoghbi present the last year's highlights in valvular heart disease from the official ACC.24 Year in Review session.

Year in Review: Heart failure and cardiomyopathies

Heart Failure Video

Watch this official video from ACC.24. Dr. Biykem Bozkurt discuss last year's major advances in heart failure and cardiomyopathies.

ACC 2024 Congress Coverage

Heart Failure Video

Year in Review: Heart failure and cardiomyopathies

Watch now

Watch this official video from ACC.24. Dr. Biykem Bozkurt discuss last year's major advances in heart failure and cardiomyopathies.

Semaglutide benefits people with type 2 diabetes and obesity-related heart failure

16-04-2024 Type 2 Diabetes News

Incentivised to exercise

11-04-2024 Lifestyle Modifications News

New intervention for STEMI-related cardiogenic shock

10-04-2024 Myocardial Infarction News

» View more highlights on the ACC 2024 congress hub page

Image Credits