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Successful use of autologous bone marrow transplantation in treating a patient with POEMS syndrome

Bone Marrow Transplantation volume 30, pages 61–62 (2002)Cite this article

In their article, Hogan et al1 report the successful use of autologous bone marrow transplantation in treating a patient with POEMS syndrome who did not respond to conventional chemotherapy. We have used the same therapeutic approach in two patients with similar success.

A 43-year-old male was admitted to hospital in January 1989, with abdominal pain. The pain had begun in May 1988 and was followed by diarrhea and weight loss (7 kg in 2 months). On admission the patient reported paresthesiae in the lower limbs, and impotence. Neurologic examination revealed diffuse areflexia, without sensorimotor deficit, and papillary edema. Electromyography showed a demyelinating neuropathy. Cerebrospinal fluid analysis showed high protein levels (1.02 g/l) without cells. The sensorimotor deficit worsened. It was decided to perform plasmapheresis and to initiate corticosteroid therapy (1 mg/kg). The sensorimotor deficit subsquently regressed in the upper limbs. In December 1989, other lesions developed (sclerodactyly, thoraco-abdominal hypertrichosis and cutaneous angiomas), and the neuropathy again worsened. Electrophoresis was normal, and immuno- electrophoresis revealed IgAλ. These findings prompted the diagnosis of POEMS syndrome, and a plasma cell dyscrasia was sought. Bone marrow smears were normal. Skeletal survey revealed a band of osteosclerosis in the left ilium, which was confirmed by tomodensitometry. Bone scintigraphy showed no areas of increased uptake. The different classes of immunoglobulin were normal in value: IgG 8 g/l, IgM 1.9 g/l, IgA 2.3 g/l. Hemoglobin was normal at 15.3 g/l. Thrombocytosis was present at 511 000/mm3. Endocrinologic investigations showed low testosterone levels with normal FSH and LH values, hyperestrogenia and hyperprolactinemia. There was no hepatosplenomegaly or lymphoadenopathy. Radiotherapy – 45 Gy to the ilium – failed to improve his clinical condition, and monoclonal Ig was still present. Chemotherapy with cyclophosphamide and prednisone was initiated in early 1990. Four cycles of treatment produced no beneficial clinical effect. Examinations performed in November 1990 showed new osteosclerotic lesions in the left sacrum and ilium and the left ischiopubic ramus. Five further cycles of chemotherapy (adriamycin, melphalan and prednisone) were unsuccessful. Autologous transplantation was performed in June 1991 after collection of bone marrow stem cells and administration of melphalan (140 mg/m2). Gradual improvement was observed. Ten years later, the only sequellae of the neuropathy were sensorimotor deficit in both feet. There was no hepatosplenomegaly. Endocrine results were normal. There were no new bone lesions. Immuno-electrophoresis showed no monoclonal component. The serum vascular endothelial growth factor (VEGF), which was raised at onset (44604 pg/ml (N: 62–707)), had returned to normal levels (747 pg/ml).

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References

  1. Hogan WJ, Lacy MQ, Wiseman GA et al. Successful treatment of POEMS syndrome with autologous hematopoietic progenitor cell transplantation Bone Marrow Transplant 2001 28: 305 309

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  2. Miralles DG, O'Fallon JR, Talley NJ . Plasma-cell dyscrasia with polyneuropathy. The spectrum of POEMS Syndrome N Engl J Med 1992 327: 1919 1923

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  3. Soubrier M, Dubost JJ, Sauvezie B The French Study Group on POEMS Syndrome. POEMS Syndrome: a study of 25 cases and a review of the literature Am J Med 1994 97: 543 552

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  4. Soubrier M, Dubost JJ, Serre AF et al. Growth factors in POEMS syndrome : evidence for a marked increase in circulating vascular endothelial growth factor Arthritis Rheum 1997 40: 786 787

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  5. Nakano A, Mitsui T, Endo I et al. Solitary plasmocytoma with VEGF overproduction: report of a patient with polyneuropathy Neurology 2001 56: 818 819

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Authors and Affiliations

  1. Unité d'Immunologie Clinique, Rhumatologie, Clermont-Ferrand, CHU 63000, France

    M Soubrier, JJ Dubost & B Sauvezie

  2. Médecine Interne, Hôtel Dieu, Clermont-Ferrand, France

    M Ruivard & P Philippe

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  1. M Soubrier
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  2. M Ruivard
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  4. B Sauvezie
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Soubrier, M., Ruivard, M., Dubost, J. et al. Successful use of autologous bone marrow transplantation in treating a patient with POEMS syndrome. Bone Marrow Transplant 30, 61–62 (2002). https://doi.org/10.1038/sj.bmt.1703600

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