Masamichi Kimura; Yoshihisa Tsuji; Masako Iwai; Masahiro Inagaki; Ali Madian; Takuya Yoshino; Minoru Matsuura; Hiroshi Nakase

doi:10.5217/ir.2015.13.2.166

Articles

Page Path: HOME > Intest Res > Volume 13(2); 2015 > Article

Case Report Usefulness of Adalimumab for Treating a Case of Intestinal Behçet's Disease With Trisomy 8 Myelodysplastic Syndrome: Masamichi Kimura¹, Yoshihisa Tsuji¹, Masako Iwai¹, Masahiro Inagaki¹, Ali Madian², Takuya Yoshino¹, Minoru Matsuura¹, Hiroshi Nakase¹; Intestinal Research 2015;13(2):166-169.
DOI: https://doi.org/10.5217/ir.2015.13.2.166
Published online: April 27, 2015

¹Department of Gastroenterology and Hepatology, Kyoto University Hospital, Kyoto, Japan.

²Department of Internal Medicine, Al Azhar University, Assint, Egypt.

Correspondence to Yoshihisa Tsuji, Department of Gastroenterology and Hepatology, Kyoto University Hospital, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto 606-8507, Japan. Tel: +81-75-751-4302, Fax: +81-75-751-4303, ytsuji@kuhp.kyoto-u.ac.jp

• Received: December 15, 2014 • Revised: February 13, 2015 • Accepted: February 27, 2015

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

prev next

10,173 Views
86 Download
23 Web of Science
24 Crossref

Full Article

Download PDF

Abstract
INTRODUCTION
CASE REPORT
DISCUSSION
NOTES
REFERENCES

Abstract

Behçet's disease (BD) is a systemic vasculitis, while myelodysplastic syndrome (MDS) is a heterogeneous group of clonal hematologic disorders characterized by ineffective hematopoiesis. Some studies suggest a relationship between MDS and BD, especially intestinal BD, and trisomy 8 seems to play an important role in both diseases. There are several reports on patients with BD comorbid with MDS involving trisomy 8 that frequently have intestinal lesions refractory to conventional medical therapies. Tumor necrosis factor (TNF)-α is strongly involved in the pathophysiology of several autoimmune diseases such as rheumatoid arthritis, inflammatory bowel disease, and BD. In addition, TNF-α plays an important role in the pathophysiology of MDS by inhibiting normal hematopoiesis and inducing the programmed cell death of normal total bone marrow cells and normal CD34+ cells. Recent clinical reports demonstrate the favorable effect of TNF-α antagonists in patients with refractory intestinal BD and in those with MDS. We present the case of a patient with intestinal BD and MDS involving trisomy 8 who was successfully treated with adalimumab.
Keywords: Adalimumab; Behçet syndrome; Trisomy 8; Myelodysplastic syndromes

INTRODUCTION

Behçet's disease (BD) is a chronic, relapsing, multisystem inflammatory disorder of unknown etiology, which is classified among the vasculitides.1 The cause of BD remains unknown; however, an autoimmune reaction triggered by an infectious agent in a genetically predisposed individual has been suggested. Recurrent oral ulcers in combination with genital ulcers, ocular disease, cutaneous lesions, arthritis, and less frequently, involvement of the gastrointestinal (GI) tract, central nervous system, and vascular beds have been typically observed.1 The disease can affect both sexes, and although it has a worldwide distribution, it is more prevalent in the Mediterranean and Far East.

Intestinal BD is characterized by intestinal ulcerations and GI symptoms. The incidence of BD involving the GI tract varies by country, ranging from 3-60%. The GI lesions are often refractory to conventional medical therapies (e.g., corticosteroids [CS] and immunosuppressants),2 and they sometimes cause life-threatening comorbidities such as intestinal perforation and massive bleeding.

Myelodysplastic syndrome (MDS) is a heterogeneous group of clonal hematologic disorders characterized by ineffective hematopoiesis. Several reports suggest that patients with MDS and trisomy 8 are prone to having intestinal BD.3,4,5 In addition, intestinal BD patients with MDS involving trisomy 8 fail to respond to the aforementioned conventional medical therapies. Therefore, a new therapeutic strategy based on its pathogenesis is well expected for treating intestinal BD patients with MDS.

Tumor necrosis factor (TNF)-α is strongly involved in the pathophysiology of several autoimmune diseases such as rheumatoid arthritis, IBD, and BD. In addition, TNF-α plays an important role in the pathophysiology of MDS by inhibiting normal hematopoiesis and inducing the programmed cell death of normal total bone marrow cells and normal CD34+ cells.6 Recent clinical reports have demonstrated the favorable effect of TNF-α antagonists in patients with refractory intestinal BD and those with MDS.7

Considering the common pathophysiology of intestinal BD and MDS, the TNF-α antagonist may be an ideal drug for treating intestinal BD patients with MDS involving trisomy 8. However, there are no reports on treating intestinal BD and MDS successfully with TNF-α antagonist alone. Herein, we present the case of a patient with intestinal BD and MDS involving trisomy 8 who was successfully treated with adalimumab (ADA), a completely humanized IgG 1 monoclonal anti-TNF-α antibody.

CASE REPORT

A 79-year-old female underwent endoscopic submucosal dissection (ESD) for an ileocecal, laterally spreading tumor. After ESD, she had a cecal refractory ulcer at the ESD site. Surgical resection of the ileocecal area was performed. However, the wound at the anastomotic site failed to heal, resulting in an enterocutaneous fistula. In addition, new ileal ulcers appeared at the anastomotic site. Moreover, the patient had intractable oral and genital ulcerations during her disease course. Despite no involvement of eye lesions or oral or genital ulcerations at this time, she had recurrent ulcers of the intestines with positive HLA-B51 alleles; thus, she was diagnosed with BD. Three months of prednisolone (30 mg/day) treatment without any other immune modulators relieved her upper abdominal pain. However, endoscopic findings revealed deterioration of the colonic ulcer (Fig. 1). She was admitted to our hospital for further treatment.

On admission, the physical examination revealed oral ulcers, genital ulcer scarring, and enterocutaneous fistulas. Blood tests showed that the white blood cell (WBC), hemoglobin (Hb), and CRP levels were 6,500/µL, 11.4 g/dL, and 0.6 mg/dL, respectively. Firstly, surgical resection of the ileum and fistula closure was performed. However, her abdominal pain continued, and the laboratory data showed the following: leukocytopenia (WBC, 1,800/µL), anemia (Hb, 5.9 g/dL), and elevation of the CRP (18.4 mg/dL). She frequently required blood transfusion for severe anemia. A bone marrow biopsy was performed to further evaluate her continuous leukocytopenia and anemia. Bone marrow biopsy and aspiration revealed normocellular marrow with dysplasia. Conventional cytogenetic testing (G-banding) of her bone marrow demonstrated the presence of trisomy 8 in all the cells. These findings led to the diagnosis of MDS (refractory anemia with excess blasts-1) involving trisomy 8. The international prognostic scoring system of BD was 4.75.8

Because of her diagnosis (i.e., CS-refractory BD associated with MDS involving trisomy 8), we administered ADA. We decided to only use adalimumab to treat MDS instead of using drugs such as azacitidine or decitabine, which may cause myelo-suppression, and she was an elderly patient with a high risk of infection. After the induction and maintenance therapy of ADA, her symptoms subsided (Fig. 1), and there was a striking improvement in her CRP level. Of note, a gradual improvement in her leukocytopenia and anemia was observed at 4 months after starting ADA. Presently, she does not require additional blood transfusions.

DISCUSSION

In this case, ADA was effective for treating intestinal BD and also anemia related to MDS with trisomy 8. To our knowledge, this is the first case of an intestinal BD patient with MDS involving trisomy 8 who was successfully treated with ADA. In addition, our case emphasizes that TNF-α plays a critical role in the pathogenesis of intestinal BD with MDS.

Until recently, there have been 39 reported cases of BD associated with MDS involving trisomy 8. The trisomy 8 allele is one of the most frequent genetic abnormalities found in 6.5-16.3% of primary MDS patients.9 Furthermore, the prevalence rate of trisomy 8 is as high as 73.7% in MDS patients with BD.10 The majority of cases have incomplete types of BD (mainly the intestinal type) and the refractory anemia subtype of MDS. Generally, it has been reported that patients with intestinal BD with MDS involving trisomy 8 are refractory to conventional medical therapies.

Since extensive intramedullary cell death in MDS patients is related to TNF-α11,12 and the serum TNF-α concentrations are increased in BD patients,13 a TNF-α antagonist would theoretically be ideal for treating intestinal BD patients with MDS. Currently, there are several reports on the effectiveness of TNF-α antagonists such as infliximab and ADA in patients with intestinal BD. A previous case of intestinal BD with MDS involving trisomy 8 was successfully treated with a combination of infliximab and methotrexate for an intestinal lesion; however, infliximab monotherapy was not effective for improving intestinal BD comorbid with MDS and trisomy 8.14,15

When treating patients with BD who have severe symptoms related to deep ulcers of the intestine, CS are the firstline of induction therapy. However, in BD patients refractory to CS, the use of anti-TNF-α monoclonal antibodies would be considered, because there are many reports on the efficacy of anti-TNF-α monoclonal antibodies in managing intestinal BD; in addition, ADA was approved for intestinal BD in Japan in 2013.16 Moreover, many physicians avoid using a high dose of CS in elderly patients as much as possible. Therefore, in our case, we selected ADA as an alternative to increasing the dose of CS. We recommend anti-TNF-α therapy in patients with refractory BD, although more data regarding the efficacy and safety of anti-TNF-α antibodies on refractory BD are needed.

It remains unclear whether infliximab is effective for intestinal BD comorbid with MDS involving trisomy 8. However, there are no previous reports on intestinal BD patients with MDS involving trisomy 8 who were treated with ADA. In our case, ADA monotherapy distinctly improved the patient's intestinal lesion and anemia. It is possible that the healing of the intestinal ulcers and good control of MDS with ADA may have contributed to the improvement of her anemia.

In conclusion, our case strongly suggests that intestinal BD patients with MDS involving trisomy 8 require optimal control of TNF-α using TNF-α antagonists. In the future, clinical trials with a larger number of patients will be required to elucidate whether ADA monotherapy is optimal for intestinal BD patients with MDS involving trisomy 8.

NOTES

Financial support: This work was supported by the Japanese Society for the Promotion of Science 8 involving (grant nos. 24590941, 25860532, 26460967, and 26893122) and also in part by the Health and Labor Sciences Research Grants for research on intractable diseases from the Ministry of Health, Labor, and Welfare of Japan (Investigation and Research for Intractable Inflammatory Bowel Disease).

Conflict of interest: None.

REFERENCES

1. Perra D, Alba MA, Callejas JL, et al. Adalimumab for the treatment of Behçet's disease: experience in 19 patients. Rheumatology (Oxford) 2012;51:1825–1831.PMID: 22723596.Article PubMed PDF
2. Ketch LL, Buerk CA, Liechty D. Surgical implications of Behçet's disease. Arch Surg 1980;115:759–760.PMID: 6966918.Article PubMed
3. Ogawa H, Kuroda T, Inada M, et al. Intestinal Behçet's disease associated with myelodysplastic syndrome with chromosomal trisomy 8-a report of two cases and a review of the literature. Hepatogastroenterology 2001;48:416–420.PMID: 11379321.PubMed
4. Kimura S, Kuroda J, Akaogi T, Hayashi H, Kobayashi Y, Kondo M. Trisomy 8 involved in myelodysplastic syndromes as a risk factor for intestinal ulcers and thrombosis-Behcet's syndrome. Leuk Lymphoma 2001;42:115–121.PMID: 11699198.Article PubMed
5. Kawabata H, Sawaki T, Kawanami T, et al. Myelodysplastic syndrome complicated with inflammatory intestinal ulcers: significance of trisomy 8. Intern Med 2006;45:1309–1314.PMID: 17170506.Article PubMed
6. Mundle SD, Ali A, Cartlidge JD, et al. Evidence for involvement of tumor necrosis factor-α in apoptotic death of bone marrow cells in myelodysplastic syndromes. Am J Hematol 1999;60:36–47.PMID: 9883804.Article PubMed
7. Naganuma M, Sakuraba A, Hisamatsu T, et al. Efficacy of infliximab for induction and maintenance of remission in intestinal Behçet's disease. Inflamm Bowel Dis 2008;14:1259–1264.PMID: 18393375.Article PubMed
8. Greenberg P, Cox C, LeBeau MM, et al. International scoring system for evaluating prognosis in myelodysplastic syndromes. Blood 1997;89:2079–2088.PMID: 9058730.Article PubMed
9. Chen G, Zeng W, Miyazato A, et al. Distinctive gene expression profiles of CD34 cells from patients with myelodysplastic syndrome characterized by specific chromosomal abnormalities. Blood 2004;104:4210–4218.PMID: 15315976.Article PubMed
10. Lin YC, Liang TH, Chang HN, Lin JS, Lin HY. Behçet disease associated with myelodysplastic syndrome. J Clin Rheumatol 2008;14:169–174.PMID: 18525438.Article PubMed
11. Campioni D, Secchiero P, Corallini F, et al. Evidence for a role of TNF-related apoptosis-inducing ligand (TRAIL) in the anemia of myelodysplastic syndromes. Am J Pathol 2005;166:557–563.PMID: 15681838.Article PubMed PMC
12. Lee RW, Dick AD. Treat early and embrace the evidence in favour of anti-TNF-α therapy for Behçet's uveitis. Br J Ophthalmol 2010;94:269–270.PMID: 20215370.Article PubMed
13. Duzgun N, Ayaslioglu E, Tutkak H, Aydintug OT. Cytokine inhibitors: soluble tumor necrosis factor receptor 1 and interleukin-1 receptor antagonist in Behçet's disease. Rheumatol Int 2005;25:1–5.PMID: 14600787.Article PubMed
14. Toyonaga T, Nakase H, Matsuura M, et al. Refractoriness of intestinal Behçet's disease with myelodysplastic syndrome involving trisomy 8 to medical therapies - our case experience and review of the literature. Digestion 2013;88:217–221.PMID: 24247154.Article PubMed
15. Iwata S, Saito K, Yamaoka K, et al. Efficacy of combination therapy of anti-TNF-α antibody infliximab and methotrexate in refractory entero-Behçet's disease. Mod Rheumatol 2011;21:184–191.PMID: 21052764.Article PubMed
16. Hisamatsu T, Ueno F, Matsumoto T, et al. The 2nd edition of consensus statements for the diagnosis and management of intestinal Behçet's disease: indication of anti-TNFα monoclonal antibodies. J Gastroenterol 2014;49:156–162.PMID: 23955155.Article PubMed

Fig. 1

Endoscopic findings before and after adalimumab (ADA) treatment. (A and B) Endoscopic findings prior to ADA treatment revealing redness, edematous mucosa, and multiple punched-out ulcers located at the postoperative anastomosis and the ileum. (C and D) Endoscopic findings showing marked improvement of the inflamed mucosa 1 year after starting ADA treatment.

Figure & Data

REFERENCES

Citations

Citations to this article as recorded by

The Expanding Spectrum of Autoinflammatory Diseases
Kiyoshi Migita, Yuya Fujita, Tomoyuki Asano, Shuzo Sato
Internal Medicine.2023; 62(1): 43. CrossRef
Refractory Intestinal Behçet-Like Disease Associated with Trisomy 8 Myelodysplastic Syndrome Resolved by Parenteral Nutrition
Ryo Takahashi, Yasuo Matsubara, Satoshi Takahashi, Kazuaki Yokoyama, Lim Lay Ahyoung, Michiko Koga, Hiroyuki Sakamoto, Narikazu Boku, Dai Shida, Hiroshi Yotsuyanagi
Case Reports in Gastroenterology.2023; 17(1): 287. CrossRef
The Expanding Spectrum of Autoinflammatory Diseases
Kiyoshi Migita, Yuya Fujita, Tomoyuki Asano, Shuzo Sato
Internal Medicine.2022;[Epub] CrossRef
CAISC: A software to integrate copy number variations and single nucleotide mutations for genetic heterogeneity profiling and subclone detection by single-cell RNA sequencing
Jeerthi Kannan, Liza Mathews, Zhijie Wu, Neal S. Young, Shouguo Gao
BMC Bioinformatics.2022;[Epub] CrossRef
Autoimmune disorders associated with myelodysplastic syndromes: clinical, prognostic and therapeutic implications
Claudio Fozza, Andrea Murtas, Giovanni Caocci, Giorgio La Nasa
Leukemia Research.2022; 117: 106856. CrossRef
Advances in Management of Intestinal Behçet’s Disease: A Perspective From Gastroenterologists
Jae Hee Cheon
Journal of Rheumatic Diseases.2021; 28(1): 4. CrossRef
Syndrome de pseudo-Behçet et autres manifestations dysimmunitaires associées aux syndromes myélodysplasiques avec trisomie 8
N. Wesner, P. Fenaux, V. Jachiet, L. Ades, O. Fain, A. Mekinian
La Revue de Médecine Interne.2021; 42(3): 170. CrossRef
The Clinical Features of Intestinal Disease in Myelodysplastic Syndromes with Trisomy 8
Yusuke Honzawa
Nippon Daicho Komonbyo Gakkai Zasshi.2021; 74(10): 594. CrossRef
Update on the Treatment of Behcet’s Disease of the Small Bowel with Biologic Agents
Yehyun Park, Jae Hee Cheon
Current Gastroenterology Reports.2020;[Epub] CrossRef
How to treat myelodysplastic syndrome with clinical features resembling Behçet syndrome: a case-based systematic review
Umut Yilmaz, Muhlis Cem Ar, Sinem Nihal Esatoglu, Isil Bavunoglu, Yusuf Ziya Erzin, Ali Ibrahim Hatemi, Suleyman Yildirim, Gülen Hatemi, Aykut Ferhat Celik
Annals of Hematology.2020; 99(6): 1193. CrossRef
Evidence-based diagnosis and clinical practice guidelines for intestinal Behçet’s disease 2020 edited by Intractable Diseases, the Health and Labour Sciences Research Grants
Kenji Watanabe, Satoshi Tanida, Nagamu Inoue, Reiko Kunisaki, Kiyonori Kobayashi, Masakazu Nagahori, Katsuhiro Arai, Motoi Uchino, Kazutaka Koganei, Taku Kobayashi, Mitsuhiro Takeno, Fumiaki Ueno, Takayuki Matsumoto, Nobuhisa Mizuki, Yasuo Suzuki, Tadakaz
Journal of Gastroenterology.2020; 55(7): 679. CrossRef
Sonic hedgehog signaling pathway in Myelodysplastic Syndrome: Abnormal activation and jervine intervention
YuTing Qin, Ming Jiang, Nilupar Tuerxung, Huan Wang, Fang Zhao, Yin Zhen, Jianping Hao
Gene.2020; 754: 144881. CrossRef
Gastrointestinal Behcet’s-like disease with myelodysplastic neoplasms with trisomy 8: a French case series and literature review
Nadege Wesner, Louis Drevon, Alexis Guedon, Jean Baptiste Fraison, Benjamin Terrier, Salim Trad, Jean Emmanuel Kahn, Achille Aouba, Jerome Gillard, Matthieu Ponsoye, Thomas Hanslik, Clement Gourguechon, Eric Liozon, Kamel Laribi, Julien Rossignol, Olivier
Leukemia & Lymphoma.2019; 60(7): 1782. CrossRef
Usefulness of Ustekinumab for Treating a Case of Myelodysplastic Syndrome-associated Inflammatory Bowel Disease
Masashi Kono, Toshiharu Sakurai, Kazuki Okamoto, Tomoyuki Nagai, Yoriaki Komeda, Hiroshi Kashida, Kosuke Minaga, Ken Kamata, Mamoru Takenaka, Satoru Hagiwara, Tomohiro Watanabe, Naoshi Nishida, Eisuke Enoki, Hiroaki Inoue, Itaru Matsumura, Masatoshi Kudo
Internal Medicine.2019; 58(14): 2029. CrossRef
Intestinal Behçet disease associated with myelodysplastic syndrome accompanying trisomy 8 successfully treated with abdominal surgery followed by hematopoietic stem cell transplantation
Tomoyuki Asano, Shuzo Sato, Makiko Yashiro Furuya, Hiroshi Takahashi, Akiko Shichishima-Nakamura, Hiroshi Ohkawara, Tatsuo Fujiwara, Naohiko Gunji, Choichiro Hashimoto, Tomoyuki Momma, Motonobu Saito, Hiroshi Nakano, Guy Watanabe, Jumpei Temmoku, Yuya Fuj
Medicine.2019; 98(46): e17979. CrossRef
Long-term maintenance of the mucosal healing induced by azacitidine therapy in a patient with intestinal Behçet’s-like disease accompanied with myelodysplastic syndrome involving trisomy 8
Nahoko Tanaka, Hirotake Sakuraba, Hiroto Hiraga, Ko Mayama, Hidezumi Kikuchi, Dai Kishida, Yui Akemoto, Keisuke Hasui, Shinji Ota, Rina Watanabe, Yasuhisa Murai, Takato Maeda, Kosuke Kamata, Tetsuya Tatsuta, Manabu Sawaya, Daisuke Chinda, Tatsuya Mikami,
Immunological Medicine.2019; 42(3): 135. CrossRef
Updated treatment strategies for intestinal Behçet’s disease
Yong Eun Park, Jae Hee Cheon
The Korean Journal of Internal Medicine.2018; 33(1): 1. CrossRef
High Incidence of Gastrointestinal Ulceration and Cytogenetic Aberration of Trisomy 8 as Typical Features of Behçet’s Disease Associated with Myelodysplastic Syndrome: A Series of 16 Consecutive Chinese Patients from the Shanghai Behçet’s Disease Database
Yan Shen, Hai-Fen Ma, Dan Luo, Jian-Fei Cai, Jun Zou, Jian-Long Guan
BioMed Research International.2018; 2018: 1. CrossRef
Anti-Tumor Necrosis Factor Therapy in Intestinal Beh Ccedil;et rsquo;s Disease
Jihye Park, Jae Hee Cheon
Gut and Liver.2018; 12(6): 623. CrossRef
Update on the Medical Management of Gastrointestinal Behçet’s Disease
Giuseppe Lopalco, Donato Rigante, Vincenzo Venerito, Claudia Fabiani, Rossella Franceschini, Michele Barone, Giovanni Lapadula, Mauro Galeazzi, Bruno Frediani, Florenzo Iannone, Luca Cantarini
Mediators of Inflammation.2017; 2017: 1. CrossRef
Risk factors and outcomes of acute lower gastrointestinal bleeding in intestinal Behçet’s disease
Jihye Park, Jae Hee Cheon, Yong Eun Park, Yoon Jee Lee, Hyun Jung Lee, Soo Jung Park, Sung Pil Hong, Tae Il Kim, Won Ho Kim
International Journal of Colorectal Disease.2017; 32(5): 745. CrossRef
18F-FDG PET/CT Findings in Two Cases With Myelodysplastic Syndrome Accompanied by Behçet’s Disease
Kimiteru Ito, Kazuo Kubota
Clinical Nuclear Medicine.2016; 41(8): e392. CrossRef
Multiple colonic ulcers associated with trisomy 8: serial changes in colonoscopic findings
Shunichi Yanai, Shotaro Nakamura, Keisuke Kawasaki, Shigeki Ito, Tamotsu Sugai, Takayuki Matsumoto
Clinical Journal of Gastroenterology.2016; 9(5): 298. CrossRef
Acute Monocytic Leukemia Masquerading Behçet’s Disease-Like Illness at Onset in an Elderly Female
Shigeru Koba, Toshio Sekioka, Sorou Takeda, Aya Miyagawa-Hayashino, Keisuke Nishimura, Shinsaku Imashuku
Case Reports in Hematology.2016; 2016: 1. CrossRef

PubReader

Cite

CITE: export Copy Download Format; Close

Download Citation

Download a citation file in RIS format that can be imported by all major citation management software, including EndNote, ProCite, RefWorks, and Reference Manager.

Format:

RIS — For EndNote, ProCite, RefWorks, and most other reference management software
BibTeX — For JabRef, BibDesk, and other BibTeX-specific software

Include:

Citation for the content below
Citation and abstract for the content below

Usefulness of Adalimumab for Treating a Case of Intestinal Behçet's Disease With Trisomy 8 Myelodysplastic Syndrome

Intest Res. 2015;13(2):166-169. Published online April 27, 2015

DOI: https://doi.org/10.5217/ir.2015.13.2.166

XML Download

Figure

Usefulness of Adalimumab for Treating a Case of Intestinal Behçet's Disease With Trisomy 8 Myelodysplastic Syndrome

Fig. 1 Endoscopic findings before and after adalimumab (ADA) treatment. (A and B) Endoscopic findings prior to ADA treatment revealing redness, edematous mucosa, and multiple punched-out ulcers located at the postoperative anastomosis and the ileum. (C and D) Endoscopic findings showing marked improvement of the inflamed mucosa 1 year after starting ADA treatment.

Fig. 1

Usefulness of Adalimumab for Treating a Case of Intestinal Behçet's Disease With Trisomy 8 Myelodysplastic Syndrome