Macrophagic myofasciitis:: description and hypotheses

被引:17
作者
Chérin, P [1 ]
Laforet, P [1 ]
Ghérardi, RK [1 ]
Authier, FJ [1 ]
Coquet, M [1 ]
Maisonobe, T [1 ]
Mussini, JM [1 ]
Pellissier, JF [1 ]
Herson, S [1 ]
机构
[1] Hop La Pitie Salpetriere, Serv Med Interne 1, F-75651 Paris 13, France
来源
REVUE DE MEDECINE INTERNE | 1999年 / 20卷 / 06期
关键词
myofasciitis; inflammatory myopathy; fasciitis; macrophage; histiocytosis;
D O I
10.1016/S0248-8663(99)80083-6
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
Purpose. - A new type of inflammatory myopathy of unknown etiology has recently been described in France. The myopathy, called macrophagic myofasciitis, had never been described in the literature. Methods. - In December 1998, 35 cases of macrophagic myofasciitis were reported, showing an increase in its incidence since the description of the first case in 1993. The first 22 cases are described. Results. - The 22 patients were each referred with a presumptive diagnosis of either polymyositis (11 patients), polymyalgia rheumatica (5 patients), mitochondrial cytopathy (4 patients), or congenital myopathy or muscle dystrophy (1 patient for each). Clinical symptoms included myalgias (91%), arthralgias (68%), marked asthenia (55%), muscle weakness (45%), and fever (32%). Laboratory findings included elevated CK levels (50%) and a marked increased in the erythrocyte sedimentation rate (37%). Electromyographic recordings showed the existence of myopathy (35%). Muscle biopsy showed a unique pattern characterized by: (i) centripetal infiltration of the epimysium, perimysium and perifascicular endomysium by non epitheloid, cells of the monocyte/macrophage lineage (CD68+, CD1a-, S100-) with both large cytoplasm and PAS-positive content; (ii) absence of necrosis, of both epitheloid and giant cells, and of mitotic figures; (iii) occasional CD8+ T-cells; and, (iiii) minimal myocyte suffering. The disease symptoms were easily distinguishable from those of sarcoid myopathy and fasciitis-panniculitis syndromes. Infectious diseases known to be associated with reactive histiocytosis, including Whipple's disease, Mycobacterium avium intracellulare infection and malakoplakia, could not be documented. Patients' condition improved under corticosteroid therapy, associated or not with non-specific antibiotic therapy. Conclusion. - A new inflammatory muscle disorder of unknown etiology, characterized by a distinctive pathological pattern of macrophagic myofasciitis, is emerging in France. Diagnosis is based on muscular biopsy. Numerous clinical, epidemiological and etiopathologic studies initiated by the GERMMAD (Groupe d'etudes et de recherche sur les maladies musculaires acquises) are in progress. (C) 1999 Elsevier, Paris.
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页码:483 / 489
页数:7
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