Behçet's disease complicated with myelodysplastic syndrome a report of two cases and review of the literature

Yano, Koji; Eguchi, Katsumi; Migita, Kiyoshi; Takashima, Hidetoshi; Tamura, Masahito; Izumino, Kiyohiro; I, Sasagawa; Sadamori, Naoki; Nagataki, Shigenobu

doi:10.1007/bf02231696

Cited by 33 publications

(19 citation statements)

References 4 publications

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“…Recently, it is also used as a new therapy for the treatment of severe autoimmune diseases such as multiple sclerosis, systemic sclerosis and systemic lupus erythematosus (13)(14)(15). Autologous HSCT has also been performed on three patients with Behçet's disease (16)(17)(18) (2,5,11,12 …”

Section: Discussionmentioning

confidence: 99%

“…Interestingly, it is speculated that this is not a simple coexistence but that the aetiology of intestinal Behçet's disease is at least partly derived from MDS itself (2,5,11,12 (4,6,9 CT s c a n s s h o we d ma r k e d e d e ma o f t h e i l e o c e c u m r e g i o n ( a r r o ws ) a n d i n f l a mma t i o n o f t h e s u r r o u n d i n g f a t t i s s u e . dup (1) …”

Section: Myelodysplastic Syndrome (Mds) Is a Heterogeneous Group Of Smentioning

confidence: 99%

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Successful Treatment of Myelodysplastic Syndrome (MDS)-related intestinal Behcet's Disease by Up-front Cord Blood Transplantation

et al. 2007

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Section: Discussionmentioning

confidence: 99%

Section: Myelodysplastic Syndrome (Mds) Is a Heterogeneous Group Of Smentioning

confidence: 99%

Successful Treatment of Myelodysplastic Syndrome (MDS)-related intestinal Behcet's Disease by Up-front Cord Blood Transplantation

et al. 2007

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“…Previous reports revealed several clinical characteristics such as a low prevalence rate of eye lesions and a high prevalence rate of intestinal lesions in BD patients with MDS involving trisomy 8 [14]. As shown in table 1, only 5.1% of BD patients with MDS involving trisomy 8 had eye lesions, whereas 74.4% had intestinal lesions [2,8,9,10,15,16,17,18,19,20,21,22,23,24,25,26,27,28,29,30,31,32,33,34]. These data showed a tendency similar to previous reports.…”

Section: Discussionmentioning

confidence: 99%

Refractoriness of Intestinal Behçet's Disease with Myelodysplastic Syndrome Involving Trisomy 8 to Medical Therapies - Our Case Experience and Review of the Literature

et al. 2013

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Background/Aims: Gastrointestinal lesions of Behçet's disease (BD) are often refractory to medical therapy and sometimes result in serious comorbidities such as gastrointestinal perforation and massive bleeding. There are several reports of patients with BD comorbid with myelodysplastic syndrome (MDS) involving trisomy 8 that frequently have intestinal lesions refractory to conventional medical therapy. Little is known about the efficacy of infliximab (IFX) for these intestinal lesions. Methods: We present 2 cases of intestinal BD with MDS involving trisomy 8 who did not respond to IFX, and review previous reports of BD with MDS involving trisomy 8 concerning their responsiveness to conventional medical therapy. Results: Among 31 previously reported cases that received medical treatment for BD, 19 (61.3%) showed temporary improvement of the BD symptoms, 9 (29.0%) deteriorated, and 3 (9.7%) showed no change. All of the 9 cases that showed deterioration had intestinal lesions. Our 2 cases failed to respond to IFX, resulting in a poor prognosis. Conclusions: IFX might not be effective for improving intestinal BD comorbid with MDS involving trisomy 8. Trisomy 8 is associated with the BD prognosis and refractoriness to conventional medical therapy.

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“…[16][17][18][19][20][21][22][23][24][25][26][27][28][29][30] Their clinical characteristics are summarized in Table 4. The median age of the patients was 48 years (range 23-74 years).…”

Section: Discussionmentioning

confidence: 99%

Beh�et?s disease associated with myelodysplastic syndrome with elevated levels of inflammatory cytokines

Hasegawa

Iwamasa

Hatta³

et al. 2003

Modern Rheumatology

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We report the case of a 56-year-old Japanese woman with Behçet's disease and myelodysplastic syndrome (MDS), who had a history of episodic high-grade fever, recurrent oral and genital ulcers, and erythema nodosum, during a 13-year period from 1989 to 2002. Bone marrow aspirates obtained in January 1995 showed refractory anemia with trisomy 8, a subtype of MDS. Her serum levels of soluble interleukin-2 receptor (IL-2R), interferon-γ, IL-1 , IL-6, IL-8, and granulocyte-macrophage colony stimulating factor in the active state were higher than those in the inactive state, whereas those of tumor necrosis factor-α and IL-10 did not increase even in the active state. In this case, it was speculated that a T-cell immune response might have been involved in the disease pathogenesis, and that the repeated febrile episodes might have been a manifestation of neutrophil hyperfunction induced by increased serum levels of inflammatory cytokines.

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Behçet's disease complicated with myelodysplastic syndrome a report of two cases and review of the literature

Cited by 33 publications

References 4 publications

Successful Treatment of Myelodysplastic Syndrome (MDS)-related intestinal Behcet's Disease by Up-front Cord Blood Transplantation

Successful Treatment of Myelodysplastic Syndrome (MDS)-related intestinal Behcet's Disease by Up-front Cord Blood Transplantation

Refractoriness of Intestinal Behçet's Disease with Myelodysplastic Syndrome Involving Trisomy 8 to Medical Therapies - Our Case Experience and Review of the Literature

Beh�et?s disease associated with myelodysplastic syndrome with elevated levels of inflammatory cytokines

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