Coronel-Martínez DL, Maldonado-Velázquez R, Carreño-Manjarrez R, Gamboa-Marrufo JD

Language: Spanish
References: 18
Page: 256-260
PDF size: 87.01 Kb.

ABSTRACT

Introduction. Progressive myositis ossificans is a rare cause of myositis in childhood, is an autosomal dominant inflammatory disorder that results in swelling of muscle followed by fibrosis and calcification.
Material and methods. We present 2 cases in which, the main clinical manifestations were congenitally short great toes and thumbs; and hardening of the soft tissues; in both cases calcifications of the neck and the thorax were demonstrated by roentgenograms. The 2 patients received conventional management with diphosphonates and isotretinoine, with poor response to these therapies; because of this we decided to use 3 initial doses of intravenous methylprednisolone (30 mg/kg/doses), and after that, we continued the treatment with monthly applications of methylprednisolone.
Results. With this therapy our patients improved; and this was evidenciated by diminishing of calcifications and by improvement in their functional status.
Conclusion. In this paper we propose an alternative pharmacological therapy with intravenous methylprednisolone, looking for its antiinflammatory and immunosuppressive effects of steroids; mainly in patients with poor response to conventional treatments.

REFERENCES

1. Cassidy JT, Petty RE. Juvenile dermatomyositis. En: Cassidy JT, Petty RE, editores. Textbook of pediatric rheumatology. 4th ed. Philadelphia: WB Saunders; 2001. p. 465-504.

2. Cook WE, Rosen FS, Banker BQ. Dermatomyositis and focal scleroderma. Pediatr Clin North Am. 1963; 10: 979.

3. Dua T, Kabra M, Kalra V. Myositis ossificans progressive. Indian Pediatr. 2001; 38: 1305-9.

4. Smith R, Russell RG, Woods CG. Myositis ossificans progressive: clinical features of eight patients and their response to treatment. J Bone Joint Surg. 1976; 58B: 48-53.

5. Hentzer B, Jacobsen HH, Asboe G. Fibrodysplasia ossificans progressive. Scand J Rheumatol. 1977; 6: 161-7.

6. Bruni L, Giammaria P, Tozzi MC, Camparcola D, Scopinaro F, Imperato C. Fibrodysplasia ossificans progressive. An 11 year old boy treated with diphosphonate. Acta Pediatr Scand. 1990; 79: 994-8.

7. Brantus JF, Meunier PJ. Effects of intra-venous etidronate and oral corticosteroids in fibrodysplasia ossificans progressive. Clin Orthop. 1998; 346: 117-20.

8. Yazici M, Etensel B, Gursoy MH, Aydogdu A, Erkus M. Nontraumatic myositis ossificans with an unusual location: case report. J Pediatr Surg. 2002; 37: 1621-2.

9. Karapinar H, Yagdi S. A case of myositis ossificans as a complication of tetanus treated by surgical excision. Acta Orthop Belg. 2003; 69: 285-8.

10. Moratis JM, Gannon FH, Shore EM, Bilker W, Zasloff MA, Kaplan FS. Limb swelling in patients who have fibrodysplasia ossificans progressive. Clin Orthop Rel Res. 1997; 336: 247-53.

11. Brandao L, Liphaus B, Almeida da Silva C. Fibrodysplasia ossificans progressive. Indian Pediatr. 2003; 40: 786-8.

12. Blaszcyck M, Majewski S, Brzenzinska-Waislo L, Jablonska S. Fibrodysplasia ossificans progressive. Eur J Dermatol. 2003; 13: 234-7.

13. Palhares DB, Leme LM. A perspective on the control of myositis ossificans progressive. J Pediatr (Rio J). 2001; 77: 431-4.

14. Jones G, Rocke DM. Multivariate survival analysis with doubly-censored data: application to the assessment of acute treatment for fibrodysplasia ossificans progressive. Stat Med. 2002; 21: 2547-62.

15. Hochberg MC, Chang RW, Dwosh I. The American College of Rheumatology 1991 revised criteria for the classification of global functional status in rheumatoid arthritis. Arthritis Rheum. 1992; 35: 498.

16. Buttgereit F, Wehling M, Burmester G. A new hypothesis of modular glucocorticoid actions. Arthritis Rheum. 1998; 41: 761-7.

17. Buttgereit F, Brand MD, Müller M. Effects of methylprednisolone on the energy metabolism of quiescent and concanavalin-A-stimulated thymocytes of the rat. Bioscience Rep. 1993; 13: 41-52.

18. Andreae J, Tripmaches R, Weltrich R. Effect of glucocorticoid therapy on glucocorticoid receptors in children with autoimmune diseases. Pediatr Res. 2001; 49: 130-5.