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Fasciitis in Chronic Graft-versus-Host Disease: A Clinicopathologic Study of 14 Cases

Anne Janin, MD; Gerard Socie, MD; Agnes Devergie, MD; Selim Aractingi, MD; Helene Esperou, MD; Olivier Verola, MD; and Eliane Gluckman, MD
[+] Article and Author Information

From Hopital Calmette, Lile, France, and Hopital Saint Louis, Paris, France. Requests for Reprints: Anne Janin, MD, Department of Pathology C, Hopital Calmette, Bd J. Leclercq, 59800 Lille Cedex, France. Acknowledgments: The authors thank R. Lafyatis, MD, for critical review of the manuscript and A. Plockyn for help with the photographs.


Copyright ©2004 by the American College of Physicians


Ann Intern Med. 1994;120(12):993-998. doi:10.7326/0003-4819-120-12-199406150-00004
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Objective: To describe the clinicopathologic features of fasciitis in patients with chronic graft-versus-host disease (GVHD) after allogeneic bone marrow transplantation from human leukocyte antigen (HLA)-identical donors.

Design: A retrospective cohort study.

Setting: Tertiary care center.

Patients: Patients who had allogeneic bone marrow transplantation and developed chronic GVHD with clinical and pathologic signs of fasciitis.

Main Outcome Measure: Analysis of clinical presentations and of deep cutaneo-muscular biopsy specimens.

Results: Between January 1974 and January 1991, 14 of 475 patients who had allogeneic bone marrow transplantation developed chronic GVHD that began with the sicca syndrome and liver or digestive tract involvement, or both, 60 to 170 days after the graft was received. Sudden and painful skin swelling was reported 350 to 3745 days after the graft was received. Follow-up over 2 to 7 years showed failure of the fasciitis to respond to steroid therapy or to any conventional treatment of chronic GVHD. Although 7 patients showed moderate improvement, the others remained functionally disabled because of skin tightness, joint stiffness, contractures, and sores. Patients with fasciitis in chronic GVHD had no specific immunogenetic profile and no history of L-tryptophan intake or phytonadione injections.

Conclusion: Among alloimmune syndromes, fasciitis is a distinct entity that leads to functional disability. This rare form of chronic GVHD may provide clues to understanding the mechanisms involved in fasciitis from other causes.

Figures

Grahic Jump Location
Figure 1.
A.B.arrowheadsarrowsC.D.arrowarrowheadsE.F.G.

Clinical aspect of the skin, with multiple small depressed areas. Intermediate septa ( ) and muscular fascia ( ) are thickened in a skin-to-muscle biopsy specimen obtained from the flank 11 days after the onset of edema (original magnification, × 10). Higher magnification of the edematous and thickened intermediate septa (original magnification, × 250). Presence of a lymphocytic infiltrate around capillaries ( ), and small nerves ( ) (original magnification, × 400). Perivascular lymphocytic infiltrate is dense, but no evidence of necrotizing vasculitis is seen (original magnification, × 400). Biopsy specimen from an older sclerotic lesion. The fascia is thickened, with lymphocytic infiltrate and sclerosis extending to the subcutaneous fat. (Original magnification, × 250.) The epidermis and superficial dermis are devoid of lymphocytic infiltrate. No “satellite cell necrosis” is found in the basal layer of the epidermis. (Original magnification, × 250). All specimens were stained with Hematein-eosin.

Grahic Jump Location

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