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Resolution of Severe Pyoderma Gangrenosum in a Patient with Streaking Leukocyte Factor Disease after Treatment with Tacrolimus (FK 506)

Kareem Abu-Elmagd, MD; David H. Van Thiel, MD; Brian V. Jegasothy, MD; Jerry C. Jacobs, MD; Patricia Carroll, MD; Horacio Rodriquez-Rilo, MD; Cheryl D. Ackerman, MD; John J. Fung, MD, PhD; and Thomas E. Starzl, MD, PhD
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From the Pittsburgh Transplant Institute and the University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania; Columbia University College of Physicians and Surgeons, New York, New York. Requests for Reprints: Kareem Abu-Elmagd, MD, Pittsburgh Transplant Institute, 3601 Fifth Avenue, Falk Clinic 5C, Pittsburgh, PA 15213. Acknowledgments: The authors thank Drs. Ramadan Sha'afi and Kozo Yasui for their help and technical assistance in measuring the serum factor. They also thank Nancy Johnson for patient care and Sharon Wesolowski for preparing the manuscript. Grant Support: In part by research grants from the Veterans Affairs and grant DK29961 from the National Institutes of Health, Bethesda, Maryland

Copyright 2004 by the American College of Physicians

Ann Intern Med. 1993;119(7_Part_1):595-598. doi:10.7326/0003-4819-119-7_Part_1-199310010-00008
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Severe, lifelong, unresolving pyoderma gangrenosum occurs in association with recurrent episodes of sterile pyoarthrosis and the presence of a serum factor (called streaking leukocyte factor) [1] responsible for enhancing random migration of purified human neutrophils and mononuclear leukocytes in vitro. Pyoderma gangrenosum is only one feature of this unusual disease. Minor trauma of any sort leads to an excessive accumulation of both mononuclear and polymorphonuclear leukocytes in tissue. This causes subcutaneous induration, sterile abscesses, sterile pyoarthrosis, and extensive areas of skin necrosis similar to those occurring in classical pyoderma gangrenosum, except that the lesions are larger and more confluent. The arthritic lesions are characterized by synovial fluid leukocyte counts greater than 100 000/mm3 and a severe synovitis [1]. We describe a patient with the streaking leukocyte factor syndrome who has been treated successfully with tacrolimus (FK 506, Prograf, Fujisawa Pharmaceutical Co., Osaka, Japan).

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Figure 1.
Ulcer in patient with streaking leukocyte factor disease.Left.Right.

A large necrotic ulcer on the front of the lower right thigh region. Marked improvement and healing of the ulcer was noted during 12 weeks of tacrolimus (FK 506) treatment.

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