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Letters to the Editor to:

Scientific Articles:
Frederick S. Kaplan, David L. Glaser, Eileen M. Shore, Robert J. Pignolo, Meiqi Xu, Yi Zhang, David Senitzer, Stephen J. Forman, and Stephen G. Emerson
Hematopoietic Stem-Cell Contribution to Ectopic Skeletogenesis
J Bone Joint Surg Am 2007; 89: 347-357 [Abstract] [Full text] [PDF]
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Electronic letters published:

[Read Letter to the Editor] Bone marrow Transplantation Does Not Cure FOP
Frederick S. Kaplan, M.D., David L. Glaser, M.D., Eileen . Shore, PH.D., Robert J. Pignolo, M.D., Ph.D., Meiqi Xu, Bs, Yi Zhang, M.D., Ph.D., David Senitzer, Ph.D., Stephen JK.Forman, M.D., Stephen G. Emerson, M.D., Ph.D.   (24 April 2007)
[Read Letter to the Editor] Appreciation of a Bone Marrow Transplant Patient with Fibrodysplasia Ossificans Progressiva
Eric L. Altschuler, M.D., Ph.D.   (17 April 2007)

Bone marrow Transplantation Does Not Cure FOP 24 April 2007
Previous Letter to the Editor Top
Frederick S. Kaplan, M.D.,
Professor of Orthopaedic Molecular Medicine
The University of Pennsylvania School of Medicine, Philadelphia, PA,
David L. Glaser, M.D., Eileen . Shore, PH.D., Robert J. Pignolo, M.D., Ph.D., Meiqi Xu, Bs, Yi Zhang, M.D., Ph.D., David Senitzer, Ph.D., Stephen JK.Forman, M.D., Stephen G. Emerson, M.D., Ph.D.

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Re: Bone marrow Transplantation Does Not Cure FOP

Frederick.Kaplan{at}uphs.upenn.edu Frederick S. Kaplan, M.D., et al.

We did indeed cite Dr. Altschuler's contribution(1) in alerting the fibrodysplasia ossificans progressiva (FOP) community to this patient. However, the important theoretical point which we explored and highlighted in our article(2), is that bone marrow transplantation did not cure FOP in this patient, while immunosuppression effectively ameliorated symptoms.

References:

1. Altschuler EL. Letters to the editor:Bone marrow transplantation in a patient with fibrodysplasia. Clin Orthop Rel Res.2004;422:277-278.

2. Kaplan FS, Glaser DL, Shore EM, Pignolo RJ, Xu M, Zhang Y, Senitzer D, Forman SJ, Emerson SG. Hematopoietic stem cell contribution to ectopic skeletogenesis. J Bone Joint Surg Am.2007;89:347-357.
Appreciation of a Bone Marrow Transplant Patient with Fibrodysplasia Ossificans Progressiva 17 April 2007
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Eric L. Altschuler, M.D., Ph.D.
Department of Physical Medicine & Rehabilitation, University of Medicine & Dentistry of New Jersey

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Re: Appreciation of a Bone Marrow Transplant Patient with Fibrodysplasia Ossificans Progressiva

eric.altschuler{at}umdnj.edu Eric L. Altschuler, M.D., Ph.D.

To The Editor:

I read with great interest the recent paper by Kaplan et al.(1) describing the disease course of a patient with fibrodysplasia ossificans progressiva (FOP) who had a bone marrow transplant(2) twenty-five years previously for aplastic anemia. However, I was quite surprised at the incomplete (and erroneus) manner in which Kaplan et al. noted that the patient “first came to the attention of the fibrodysplasia ossificans progressiva community at the age of thirty-five”, and so I write to set the record straight.

The patient came to the attention of the fibrodysplasia ossificans community due initially and crucially to my efforts. The patient was lost for a decade, and from a research point of view, neither the patient’s hematologists, nor any one in the FOP community, or anyone else(3), cited the report(2) until I did(4). I went to considerable lengths to alert and exhort the transplant physicians to follow-up this case.

After transplantation the patient had both acute and chronic graft-versus-host disease which was successfully treated with prednisone, cyclosporine and methotrexate for 14 years. This time period of treatment of graft-versus-host disease coincided with a cessation of FOP disease activity—sui generis(1) and returned after the drugs were stopped. These medicines have significant side effects and toxicities; also, as Kaplan et al.(1) note, the patient had a “non-FOP” hematopoietic system so it may not be possible to generalize treatment with this drug regimen to other afflicted patients.

Nevertheless, we may be cautiously and guardedly optimistic that FOP has been moved out of the untreatable column.

The author did not receive any outside funding or grants in support of his research for or preparation of this work. Neither he nor a member of his immediate family received payments or other benefits or a commitment or agreement to provide such benefits from a commercial entity. No commercial entity paid or directed, or agreed to pay or direct, any benefits to any research fund, foundation, division, center, clinical practice, or other charitable or nonprofit organization with which the author, or a member of his immediate family, is affiliated or associated .

References:

1. Kaplan FS, Glaser DL, Shore EM, Pignolo RJ, Xu M, Zhang Y, Senitzer D, Forman SJ, Emerson SG. Hematopoietic stem-cell contribution to ectopic skeletogenesis. J Bone Joint Surg Am. 2007;89:347-57.

2. Spruce WE, Forman SJ, Blume KG, Farbstein MJ, Scott EP, Wolf JL, Krance R. Successful second bone marrow transplantation in a patient with myositis ossificans progressiva and aplastic anemia. Am J Pediatr Hematol Oncol. 1983;5:337 -40.

3. http://scientific.thomson.com/products/sci/ (accessed April 6, 2007).

4. Altschuler EL. Letters to the editor: Bone marrow transplantation in a patient with fibrodysplasia. Clin Orthop Relat Res.2004; 422:277 -8.