Transfer of autoimmune thyroiditis and resolution of palmoplantar pustular psoriasis following allogeneic bone marrow transplantation

Abstract

Summary:Case reportA 40-year-old man was diagnosed with AML in January We report an unusual case of a patient who was cured of one autoimmune disease (palmoplantar pustular pso-1992. Bone marrow cytology revealed M6 by the FAB classification with trilineage myelodysplasia. The patient riasis (PPP)) but developed another autoimmune disease (autoimmune thyroiditis) after allogeneic BMT. A 40-went into complete remission following induction chemotherapy with daunorubicin, 6-mercaptopurine, and behenoyl year-old man suffering from AML with PPP underwent allogeneic BMT from his HLA-identical sister for the cytosine arabinoside. After receiving three courses of consolidation chemotherapy, he developed skin lesions on the treatment of AML. The patient experienced complete clearance of the cutaneous PPP despite the cessation of palms and soles ( Figure 1). The skin lesions were diagnosed as PPP by histological examination in February 1993. immunosuppressive therapy for over 2 years. However, he developed hyperthyroidism with anti-thyroglobulin They were treated with combinations of topical corticosteroids and etretinate, but showed only partial response. antibodies 5 months after BMT, although he had showed normal thyroid functions without anti-thyrogloIn November 1993, after myeloablation with BU (16 mg/kg) and CY (120 mg/kg), the patient received an bulin antibodies before BMT. The donor had no history of thyroid diseases and showed normal thyroid funcallogeneic BMT from his HLA-identical younger sister, who did not suffer from PPP. The mixed lymphocyte reactions but was positive for anti-thyroglobulin antibodies. Thus, even when the donor is in a subclinical state, autotion was negative. Although the patient's elder sister had died of hyperthyroidism, his younger sister had no history immune thyroiditis may be transferred from donors to recipients by BMT.of thyroid diseases and showed normal thyroid functions, but was positive for anti-thyroglobulin antibodies. Keywords: autoimmune thyroiditis; palmoplantar pustular psoriasis; bone marrow transplantation; autoimmunity CYA and MTX were used for GVHD prophylaxis. The hematological follow-up showed stable engraftment with complete hematopoietic recovery and sustained complete chimerism. There were no signs of acute GVHD. After his It is well known that the hematopoietic and immune system of recipients is eventually replaced by donor-derived cells after allogeneic BMT. Experimental work in animal models has demonstrated that BMT can be used to treat systemic and organ-specific autoimmune diseases. 1 In humans, the resolution of immune-mediated diseases after BMT has recently been reported. [2][3][4] On the other hand, autoimmune diseases have also been transferred from donors to recipients in both mice 5 and humans 6-8 by BMT. We report the unusual case of a patient who was cured of palmoplantar pustular psoriasis (PPP) but developed autoimmune thyroiditis after allogeneic BMT. This is the first case report in which a pre-existing immune-mediated disease was cured while ...

Resolution of psoriasis following allogeneic bone marrow transplantation for chronic myelogenous leukemia: Case report and review of the literature

“…Experimental study in animal models has demonstrated that various autoimmune diseases can be transferred to healthy animals by BMT after total body irradiation [17]. Additionally, transfer of other autoimmune diseases has been reported in human BMT [7,18]. These findings indicate that a clone of donor lymphocytes programmed to induce autoantibodies and/or a cytotoxic response may be transferred from donor to recipient, and they may support the hypothesis that immune diseases are "stem cell disorders.…”

“…Some patients with coincidental autoimmune disorder who underwent allogeneic BMT for a malignancy have been cured of their autoimmune disease [4][5][6][7][8][9]; however, allogeneic BMT is not routinely considered for treatment of autoimmune disease because of its significant mortality and morbidity. In this paper, we have described a patient with CML whose psoriasis resolved completely following allogeneic BMT, and reviewed similar cases [4,5,[7][8][9][10][11] (Table I).…”

“…In this paper, we have described a patient with CML whose psoriasis resolved completely following allogeneic BMT, and reviewed similar cases [4,5,[7][8][9][10][11] (Table I).…”

“…Therefore, it has been suspected that the course of autoimmune disease can be modified by allogeneic BMT. Recently, the resolution of coincidental immunemediated diseases following BMT has been reported [4][5][6][7][8][9]. In the present report, we describe a patient with chronic myelogenous leukemia (CML) whose psoriasis resolved completely over 2.5 years of follow-up period after allogeneic BMT.…”

Clinically demonstrable anti-autoimmunity mediated by allogeneic immune cells favorably affects outcome after stem cell transplantation in human autoimmune diseases

“…13,16,[23][24][25][26][27][28][29][30][31][32][33][34] The AID were detected 0.5 to 33 (median 8) years before transplant. Twenty-three patients received bone marrow transplants, which were T cell-depleted in two cases.…”