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2012, Number 1

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Med Cutan Iber Lat Am 2012; 40 (1)

Morphea in childhood: a retrospective study

Martínez MML, Azaña DJM, Rodríguez VM, Faura BC, López VMT, Iñiguez OL

Language: Spanish
References: 37
Page: 11-17
PDF size: 92.01 Kb.


ABSTRACT

Morphea is an uncommon disease, whose prevalence is not well established. The aim of our studty was to determine the clinical features of morphea in pediatric patients diagnosed in our department.
Methods: We retrospectively analized pediatric patients with clinical and histological diagnosis of morphea from Jannuary 1996 to April 2010.
Results: Thirteen pediatric patients were diagnosed clinically and histologically of morphea, 9 women and 4 men. The mean age was 12,6 years. The time from the first sign of morphea to diagnosis was 5,69 months. The most frequent clinical presentation was plaque morphea (61,54%), followed by generalized (23,07%) and linear (15,38%). ANA were found in 38,46% and eosinophilia in 23,07% of patients. Extracutaneous manifestations were observed in four patients (30,77%); three cases (23,07%) were accompanied by neurological symptoms, and two of autoimmune diseases (15,38%). The outcome was good except in 3 patients (23,08%) who presented persistence of the disease with episodes of exacerbation.
Conclusions: Morphea in childhood does not always have a benign course to complete resolution. Most patients have a mild and self-limited outcome, but this disorder can cause permanent dissability and noncutaneous manifestations.


REFERENCES

  1. Fett N, Werth VP. Update on morphea: part I. Epidemiology, clinical presentation, and pathogenesis. J Am Acad Dermatol 2011; 64: 217-28.

  2. Peterson LS, Nelson AM, Su WP, Mason T, O’Fallon WM, Gabriel SE. The epidemiology of morphea (localized scleroderma) in Olmsted County 1960-1993. J Rheumatol 1997; 24: 73-80.

  3. Murray KJ, Laxer RM. Scleroderma in children and adolescents. Rheum Dis Clin North Am 2002; 28: 603-24.

  4. Christen-Zaech S, Hakim MD, Afsar FS, Paller AS. Pediatric morphea (localized scleroderma): review of 136 patients. J Am Acad Dermatol 2008; 59: 385-96.

  5. Zulian F, Athreya BH, Laxer R, Nelson AM, Feitosa de Oliveira SK, Punaro MG et al. Juvenile localized scleroderma: clinical and epidemiological features in 750 children. An international study. Rheumatology (Oxford) 2006; 45: 614-20.

  6. Leitenberger JJ, Cayce RL, Haley RW, Adams-Huet B, Bergstresser PR, Jacobe HT. Distinct autoimmune syndromes in morphea: a review of 245 adult and pediatric cases. Arch Dermatol 2009; 145: 545-50.

  7. Laguna Argente C, García-Melgares Linares ML, Febrer Bosch I. Morfea infantil. Piel 2009; 24: 28-34.

  8. Hom C, Ilowite NT. Other rheumatic diseases in adolescence: dermatomyositis, scleroderma, overlap syndromes, systemic vasculitis, and panniculitis. Adolesc Med 1998; 9: 69-83.

  9. Isenberg DA, Black C. ABC of rheumatology: Raynaud’s phenomenon, scleroderma, and overlap syndromes. BMJ 1995; 310: 795-8.

  10. Steen VD, Oddis CV, Conte CG, Janoski J, Casterline GZ, Medsger TA Jr. Incidence of systemic sclerosis in Allegheny County, Pennsylvania: a twenty-year study of hospital-diagnosed cases, 1963-1982. Arthritis Rheum 1997; 40: 441-5.

  11. Peterson LS, Nelson AM, Su WPD. Classification of morphea (localized scleroderma). Mayo Clin Proc 1995; 70: 1068-76.

  12. Beltramelli M, Vercellesi P, Frasin A, Gelmetti C, Corona F. Localized Severe Scleroderma: A Retrospective Study of 26 Pediatric Patients. Pediatr Dermatol 2010; 27: 476-80.

  13. Toledano C, Rabhi S, Kettaneh A, Fabre B, Fardet L, Tiev KP et al. Localized scleroderma: a series of 52 patients. Eur J Intern Med 2009; 20: 331-6.

  14. Saxton-Daniels S, Jacobe HT. An Evaluation of Long-term Outcomes in Adults with Pediatric Onset Morphea. Arch Dermatol 2010; 146: 1044-5.

  15. Marzano AV, Menni S, Borghi A, Fuligni A, Fabbri P, Caputo R. Localized scleroderma in adults and children. Clinical and laboratory investigations on 239 cases. Eur J Dermatol 2003; 13: 171-6.

  16. Torrelo A, Suárez J, Colmenero I, Azorin D, Perera A, Zambrano. A. Deep morphea after vaccination in two young children. Pediatr Dermatol 2006; 23: 484-7.

  17. Herrmann T, Gunther C, Csere P. Localized morphea—a rare but significant secondary complication following breast cancer radiotherapy. Case report and review of the literature on radiation reaction among patients with scleroderma/morphea. Strahlenther Onkol 2009; 185: 603-7.

  18. Peroni A, Zini A, Braga V, Colato C, Adami S, Girolomoni G. Druginduced morphea: report of a case induced by balicatib and review of the literature. J Am Acad Dermatol 2008; 59: 125-9.

  19. Kraigher O, Brenner S, Tur E. Anti-doublestranded DNApositive unilateral generalized morphea in an adult, possibly exacerbated by ibuprofen. Arch Dermatol 2009; 145: 844-6.

  20. Prinz JC, Kutasi Z, Weisenseel P, Poto L, Battyani Z, Ruzicka T. ‘‘Borrelia-associated early-onset morphea’’: a particular type of scleroderma in childhood and adolescence with high titer antinuclear antibodies? Results of a cohort analysis and presentation of three cases. J Am Acad Dermatol 2009; 60: 248-55.

  21. Arkachaisri T, Fertig N, Pino S, Medsger TA Jr. Serum autoantibodies and their clinical associations in patients with childhood- and adult-onset linear scleroderma. A singlecenter study. J Rheumatol 2008; 35: 2439-44.

  22. Takehara K, Sato S. Localized scleroderma is an autoimmune disorder. Rheumatology (Oxford) 2005; 44: 274-9.

  23. Hayakawa I, Hasegawa M, Takehara K, Sato S. Anti-DNA topoisomerase II alpha autoantibodies in localized scleroderma. Arthritis Rheum 2004; 50: 227-32.

  24. Zulian F, Vallongo C, Woo P, Russo R, Ruperto N, Harper J et al. Localized scleroderma in childhood is not just a skin disease. Arthritis Rheum 2005; 52: 2873-81.

  25. Zannin ME, Martini G, Athreya BH, Russo R, Higgins G, Vittadello F et al. Ocular involvement in children with localised scleroderma: a multi-centre study. Br J Ophthalmol 2007; 91: 1311-4.

  26. Birdi N, Laxer RM, Thorner P Fritzler MJ, Silverman ED. Localized sclerodermia progressing to systemic disease: Case report and review of the literature. Arthritis Rheum 1993; 36: 410-5.

  27. Fett N, Werth VP. Update on morphea: part II. Outcome measures and treatment. J Am Acad Dermatol 2011; 64: 231-42.

  28. Dytoc MT, Kossintseva I, Ting PT. First case series on the use of calcipotriol-betamethasone dipropionate for morphoea. Br J Dermatol 2007; 157: 615-8.

  29. Dytoc M, Ting PT, Man J, Sawyer D, Fiorillo L. First case series on the use of imiquimod for morphoea. Br J Dermatol 2005; 153: 815-20.

  30. Kroft EB, Groeneveld TJ, Seyger MM, de Jong EM. Efficacy of topical tacrolimus 0.1% in active plaque morphea: randomized, double- blind, emollient-controlled pilot study. Am J Clin Dermatol 2009; 10: 181-7.

  31. Sator PG, Radakovic S, Schulmeister K, Honigsmann H, Tanew A. Medium-dose is more effective than low-dose ultraviolet A1phototherapy for localized scleroderma as shown by 20-MHz ultrasound assessment. J Am Acad Dermatol 2009; 60: 786-91.

  32. Kreuter A, Hyun J, Stucker M, Sommer A, Altmeyer P, Gambichler T. A randomized controlled study of low-dose UVA1, mediumdose UVA1, and narrowband UVB phototherapy in the treatment of localized scleroderma. J Am Acad Dermatol 2006; 54: 440-7.

  33. Cox D, O’Regan G, Collins S, Byrne A, Irvine A, Watson R. Juvenile localised scleroderma: a retrospective review of response to systemic treatment. Ir J Med Sci 2008; 177: 343-6.

  34. Kroft EB, Creemers MC, van den Hoogen FH, Boezeman JB, De Jong EM. Effectiveness, side-effects and period of remission after treatment with methotrexate in localized scleroderma and related sclerotic skin diseases: an inception cohort study. Br J Dermatol 2009; 160: 1075-82.

  35. Weibel L, Sampaio MC, Visentin MT, Howell KJ, Woo P, Harper JI. Evaluation of methotrexate and corticosteroids for the treatment of localized scleroderma (morphoea) in children. Br J Dermatol 2006; 155: 1013-20.

  36. Fitch PG, Rettig P, Burnham JM, Finkel TH, Yan AC, Akin E et al. Treatment of pediatric localized scleroderma with methotrexate. J Rheumatol 2006; 33: 609-14.

  37. Martini G, Ramanan AV, Falcini F, Girschick H, Goldsmith DP, Zulian F. Successful treatment of severe or methotrexate-resistant juvenile localized scleroderma with mycophenolate mofetil. Rheumatology (Oxford) 2009; 48: 1410-3.




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