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

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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.

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References

  1. Shulman LE (1974) Diffuse fasciitis with hypergammaglobulinemia and eosinophilia: a new syndrome? J Rheumatol 1:S46

    Google Scholar 

  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

    CAS  Google Scholar 

  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 52:183–187

    PubMed  CAS  Google Scholar 

  4. Takeda SI, Takarakura E, Fukui Y (1995) Tubulointerstitial nephritis in a patient with eosinophilic fasciitis and IgA nephropathy. Nephron 69:314–317

    Article  PubMed  CAS  Google Scholar 

  5. Rizzo S (2002) Eosinophilic pleuropericarditis and fasciitis. A new case Monaldi Arch Chest Dis 57:311–313

    CAS  Google Scholar 

  6. Killen JW, Swift GL, White RG (2000) Eosinophilic fasciitis with pulmonary and pleural involvement. Postgrad Med 76:36–37

    Article  CAS  Google Scholar 

  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 48:809–812

    PubMed  CAS  Google Scholar 

  8. Giordano M, Ara M, Cicala C, Valentini G, Chianese U, Vatti M (1980) Eosinophilic fasciitis. Am J Med 93:645–646

    CAS  Google Scholar 

  9. Rosenthal J, Benson MD (1980) Diffuse fasciitis and eosinophilia with symmetric polyathritis. Ann Int Med 92:507–509

    PubMed  CAS  Google Scholar 

  10. Moriguchi M, Terai C, Kuroki S, Tanaka E, Someya N, Tsunoda Y, Kashivazaki S (1998) Eosinophilic fasciitis complicated with peripheral polyneuropathy. Intern Med 37:417–420

    Article  PubMed  CAS  Google Scholar 

  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 140:1185–1188

    PubMed  CAS  Google Scholar 

  12. Khanna D, Verity A, Grossman JM (2002) Eosinophilic fasciitis with multiple myeloma. Ann Rheum Dis 61:1111–1112

    Article  PubMed  CAS  Google Scholar 

  13. Masuoka H, Kikuchi K, Takahasi S, Hayashi N, Farue M (1998) Eosinophilic fasciitis associated with low-grade T cell Lymphoma. Br J Dermatol 139:928–930

    Article  PubMed  CAS  Google Scholar 

  14. Junca J, Cuxart A, Tural C, Ojanguren I, Flores A (1994) Eosinophilic fasciitis and non-Hodgkin lymphoma. Eur J Haematol 52:34–36

    Google Scholar 

  15. Garcia VP, de Quiros JF, Caminal L (1998) Autoimmune hemolitic anemia with eosinophilic fasciitis. J Rheumatol 25:1864–1865

    PubMed  CAS  Google Scholar 

  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 199:282

    Article  Google Scholar 

  17. Gallardo F, Vadello M, Mitjavila F, Servitje O (1998) Systemic lupus erithematosus after eosinophilic fasciitis:a case report. J Am Acad Dermatol 39:283–285

    Article  PubMed  CAS  Google Scholar 

  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

    Google Scholar 

  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

    Google Scholar 

  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 32:152–157

    Article  Google Scholar 

  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

    Google Scholar 

  22. Barnes L, Rodnan GP, Medsger TA Jr, Short D (1979) Eosinophilic fasciitis. A pathologic study of twenty cases. Am J Pathol 96:493–517

    PubMed  CAS  Google Scholar 

  23. Romero AG, Fernandez JG, Calataynd JC (2001) Eosinophilic fasciitis associated with simple traumatism. Acta Dermatovenereol Croat 9:287–290

    CAS  Google Scholar 

  24. Hamilton M (1991) Eosinophilic fasciitis associated with l-tryptofan ingestion. J Rheum Dis 50:55–56

    Article  CAS  Google Scholar 

  25. Choquet-Kastylevvski G, Kanikatis J, Dumas V, Descotes J, Faure M, Claudy AR (2001) Eosinophilic fasciitis and simvastatin. Arch Int Med 161:1456–1457

    Article  Google Scholar 

  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 25:383–385

    PubMed  CAS  Google Scholar 

  27. Smith JD, Chang KL, Gums JG (1988) Possible lansoprazole-induced eosinoplic sindrome. Ann Pharmacother 32:196–200

    Google Scholar 

  28. Solomon G, Barland P, Rifkin H (1982) Eosinophilic fasciitis responsive to cimetidine. Ann Intern Med 97:547–549

    PubMed  CAS  Google Scholar 

  29. Hayashi N, Igarashi A, Matsuyama T, Harada T (2000) Eosinophilic fasciitis following exposure to trichloroethylene: successful treatment with cyclosporin. Brit J Dermatol 142:830–832

    Article  CAS  Google Scholar 

  30. Valencia IC, Chang A, Kirsner RS, Kerdel FA (1999) Eosinophilic fasciitis responsive to treatment with pulsed steroids and cyclosporine. Int J Dermatol 38:369–372

    PubMed  CAS  Google Scholar 

  31. Kato Y, Naito Y, Narita Y, Kuzuhara S (1997) D-penicillamine-induced myasthenia gravis in a case of eosinophilic fasciitis. J Neurol Sci 146:85–86

    Article  PubMed  CAS  Google Scholar 

  32. Caspi D, Fishel R, Varon M, Yona E, Baratz M, Yaron M (1982) Multisystem presentation of eosinophilic fasciitis. Rheumatol Rehabil 21:218–221

    Article  PubMed  CAS  Google Scholar 

  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 21:110–121

    Article  PubMed  Google Scholar 

  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

    CAS  Google Scholar 

  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 47:29–35

    Article  PubMed  Google Scholar 

  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 242:528–532

    Article  PubMed  CAS  Google Scholar 

  37. Kovalev VM, Krivenko ZF New aspects of the pathogenesis and treatment of Schulmann's eosinophilic fasciitis. Vestn Dermatol Venerol 1989;12:13–5

    Google Scholar 

  38. Farrington ML, Haas JE, Nazar-Stewart V, Mellins ED (1993) Eosinophilic fasciitis in children frequently progresses to scleroderma-like cutaneous fibrosis. J Rheumatol 20:128–132

    PubMed  CAS  Google Scholar 

  39. Coyle HE, Chapman RS (1980) Eosinophilic fasciitis (Shulman syndrome) in association with morphoea and systemic sclerosis. Acta Derm Venereol 60:181–182

    PubMed  CAS  Google Scholar 

  40. Pouplin S, Daragon A, Le Loet X (1998) Treatment of eosinophilic fasciitis with methotrexate. J Rheumatol 25:606–607

    PubMed  CAS  Google Scholar 

  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 161:118–119

    Article  Google Scholar 

  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

    Google Scholar 

  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 17:10–13

    Article  CAS  Google Scholar 

  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 142:804

    Article  PubMed  CAS  Google Scholar 

  45. Cetkovsky P, Koza V, Cetkovska P, Svojgrova M (1998) Successful treatment of severe Shulman’ syndrome by allogenic bone marrow transplantion. Bone Marrow Transpl 21:637

    Article  CAS  Google Scholar 

  46. Neumeister MW, Robertson GA (1998) Therapeutic fasciectomy for eosinophilic fasciitis. Ann Plasturgery 41:208–210

    Article  CAS  Google Scholar 

  47. Naschitz JE, Boss JH, Misselevich I, Yeshurun D, Rosner I (1996) The fasciitis-panniculitis syndromes. Clinical and pathologic features. Medicine 75:6–16

    Article  PubMed  CAS  Google Scholar 

  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:S47

    PubMed  CAS  Google Scholar 

  49. Gabrielli A, De Nictolis M, Campanati G, Cinti S (1983) Eosinophilic fasciitis: a mast cell disorder? Clin Exp Rheumatol 1:75–80

    PubMed  CAS  Google Scholar 

  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 30:1811–1815

    PubMed  Google Scholar 

  51. Mekori YA (2004) The mastocyte: the « other » inflammatory cell in immunopathogenesis. J Allergy Clin Immunol 114:15–21

    Article  Google Scholar 

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Manzini, C., Sebastiani, M., Giuggioli, D. et al. D-penicillamine in the treatment of eosinophilic fasciitis: case reports and review of the literature. Clin Rheumatol 31, 183–187 (2012). https://doi.org/10.1007/s10067-011-1866-3

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  • DOI: https://doi.org/10.1007/s10067-011-1866-3

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