Resolution of atopic dermatitis following allogeneic bone marrow transplantation for chronic myelogenous leukemia

Koharazawa, Hideyuki; Kanamori, Heiwa; Takabayashi, Maki; Yamaji, Satoshi; Taguchi, Jun; Fujimaki, Katsumichi; Ishigatsubo, Yoshiaki

doi:10.1038/sj.bmt.1704990

Cited by 14 publications

(13 citation statements)

References 8 publications

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“…However, it is difficult to determine whether the remission of RS3PE was due to replacement of the donor immune system after CBT or due to elimination of concomitant MDS clone after the conditioning regimen. Similar to our case, it has been reported that concomitant autoimmune diseases such as rheumatoid arthritis, Behcet's disease, psoriasis, and atopic dermatitis were resolved in patients who received allogeneic HSCT to treat conventional hematological diseases [3,[8][9][10]. Although the efficacy of allogeneic HSCT in treating severe autoimmune disease is under further investigation, our findings indicate that allogeneic HSCT could be effective in patients with paraneoplastic RS3PE.…”

supporting

confidence: 84%

Remission of remitting seronegative symmetrical synovitis with pitting edema after unrelated cord blood transplantation for myelodysplastic syndrome

et al. 2015

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Dear Editor, Allogeneic hematopoietic stem cell transplantation (HSCT) has been reported to induce remission of coincidental or sole autoimmune disease [1][2][3]. We report on a patient with myelodysplastic syndrome (MDS) whose remitting seronegative symmetrical synovitis with pitting edema (RS3PE) achieved remission without any treatment after unrelated cord blood transplantation (CBT).A 60-year-old man with a two-month history of morning stiffness in the hand joints, polyarthralgia, and edema of the dorsum of both the hands and feet was admitted to our hospital. Laboratory findings revealed an elevated matrix metalloproteinaise-3 (MMP-3) of 824.1 ng/mL (<121), along with macrocytic anemia. Rheumatoid factor, antinuclear antibody, and anti-cyclic citrullinated peptide antibody were negative. Magnetic resonance imaging (MRI) of the hands demonstrated soft tissue swelling without bone erosion. Based on these data, he was diagnosed with RS3PE. Bone marrow examination revealed dysplasia of the erythroid series and chromosomal abnormality including dup(1)(q21;q42), indicating that macrocytic anemia was due to MDS refractory anemia according to the WHO classification, and with the IPSS intermediate-1 risk group. He was treated with prednisolone, salazosulfapyridine, and etanercept and showed mild improvement of RS3PE symptoms. Eighteen months after diagnosis of MDS, pancytopenia progressed, and the proportion of myeloblasts in the bone marrow increased, which was consistent with the disease progression to refractory anemia with excess blasts-2 (RAEB-2). He received seven courses of azacitidine for MDS RAEB-2 and achieved marrow complete remission with hematological improvement according to the International Working Group (IWG) response criteria [4]. Symptoms of RS3PE improved in parallel with treatment response to MDS. However, since the response to azacitidine was transient, he received an HLA-mismatched CBT for MDS. One month before CBT, MRI showed the presence of fluid in the tenosynovial sheaths of the metacarpal phalangeal (MCP) joint and flexor and extensor tendons of both the hands (Fig. 1a). The number of cord blood nucleated cells was 2.43×10 7 /kg, and the number of CD34-positive cells was 0.97×10 5 /kg. The conditioning regimen consisted of 4 Gy of total body irradiation, intravenous busulfan 9.6 mg/kg, fludarabine 180 mg/m 2 , and cytarabine 12 g/m 2 . Prophylaxis for graft-versus-host disease (GVHD) consisted of intravenous cyclosporine and oral mycophenolate mofetil. His hand dorsum edema and arthralgia disappeared rapidly following the conditioning regimen. Limited chronic GVHD involving the skin occurred, but resolved spontaneously. Immunosuppressive treatment was discontinued 10 months after CBT. Bone marrow examination 12 months after CBT revealed complete remission and complete donor chimerism. Fifteen months after CBT, an MRI of his hands revealed no findings of RS3PE (Fig. 1b), and the serum MMP-3 level had decreased to 48.5 ng/mL.

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confidence: 84%

Remission of remitting seronegative symmetrical synovitis with pitting edema after unrelated cord blood transplantation for myelodysplastic syndrome

et al. 2015

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“…This event was confirmed in a number of investigations, and recently a follow-up study demonstrated that T and B cell clones with allergen-specific memory influencing IgE production, rhinitis and asthma extend their activity for up to 14 years after BMT [5]. Conversely, BMT from non-allergic donor to atopic recipient leads to resolution of atopic dermatitis [6]. These data indicate clearly that BMT gives rise to both local and systemic effects on allergic disease.…”

mentioning

confidence: 50%

Allergy as an organ and a systemic disease

Pucci

Incorvaia²

2008

Clinical and Experimental Immunology

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SummaryAllergic disorders are viewed generally as organ diseases and thus referred to organ specialists, such as the ear, nose and throat specialist for rhinitis, the pulmonologist for asthma, the dermatologist for dermatitis, and so on. Indeed, the systemic nature of allergy is made evident by the fact that the same individual may develop during the life different manifestations to a given allergen. This is true for example in sensitisation to house dust mites, which may start in childhood as atopic dermatitis and later express as asthma or rhinitis.The major player in driving the immune response is the T lymphocyte, and the T helper subpopulations -Th1 and Th2 -as well as the T regulatory cells, are involved in orienting tolerance or reactivity to allergens. Interesting observations on the systemic or organ-specific actions of T cells were obtained by transplantations from allergic donors to non-allergic recipients. Bone marrow is able to transfer all allergic manifestations, while lung transplantation transfers only asthma. A number of factors are involved in the expression of allergy as a systemic or organ disease and deserve deeper investigations. They include the antigen presenting cells, the homing of T cells, the cytokine and chemokine pattern, and the adhesion molecules.

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“…Improvement of the immunosuppression caused by HTLV-1 infection might influence this clinical course. On the other hand, an association between AD and HTLV-1 infection has also been suggested [22], and there have been reports on the resolution of AD after allogeneic HSCT [23] and the transfer of AD from a donor who had AD after allogeneic HSCT [24]. Since her donor did not have AD, the clinical course of her skin lesions was also compatible with refractory AD.…”

Section: Discussionmentioning

confidence: 92%

Successful treatment of young-onset adult T cell leukemia/lymphoma and preceding chronic refractory eczema and corneal injury by allogeneic hematopoietic stem cell transplantation

et al. 2009

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Only some carriers of human T cell lymphotropic virus type I (HTLV-1) develop adult T cell leukemia/lymphoma (ATLL) after a long latency period, and an association has been reported between chronic refractory eczema, known as infective dermatitis, and young-onset ATLL. A 25-year-old female developed ATLL and underwent allogeneic hematopoietic stem cell transplantation (HSCT) in non-remission. She had chronic refractory eczema and corneal injury at the onset of ATLL. Remission of ATLL was achieved, and the HTLV-1 proviral load decreased after HSCT. In addition, her pre-existing eczema and corneal injuries almost disappeared. More than a year has passed since the transplantation was performed, and she has had no recurrence of either ATLL or lesions in the skin and eye. Her clinical course suggests a possible association between skin and eye lesions and HTLV-1 infection. Changes in the immunological condition after HSCT might play a key role. Special attention is needed when HTLV-1 carriers develop eye or skin lesions.

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Resolution of atopic dermatitis following allogeneic bone marrow transplantation for chronic myelogenous leukemia

Cited by 14 publications

References 8 publications

Remission of remitting seronegative symmetrical synovitis with pitting edema after unrelated cord blood transplantation for myelodysplastic syndrome

Remission of remitting seronegative symmetrical synovitis with pitting edema after unrelated cord blood transplantation for myelodysplastic syndrome

Allergy as an organ and a systemic disease

Successful treatment of young-onset adult T cell leukemia/lymphoma and preceding chronic refractory eczema and corneal injury by allogeneic hematopoietic stem cell transplantation

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