Clinically and radiologically successful treatment of spondylitis by guselkumab in a patient with pustulotic arthro-osteitis

IF 1.1 Q4 ALLERGY
Natsumi Ikumi MD, Hideki Fujita MD, PhD
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引用次数: 0

Abstract

A 29-year-old Japanese woman presented with a 6-month history of pustular eruptions on the palms and soles. She also suffered from pain and stiffness in the anterior chest and the lumbar spine for 3 months, which were treated with loxoprofen. Although she had no history of rheumatoid arthritis, psoriatic arthritis, or ankylosing spondylitis, she was diagnosed with endometrial uterine cancer 3 years before. After treatment with surgery and chemotherapy, she experienced no recurrence of the tumor. She had neither focal infection nor history of smoking. At presentation, the patient had erythema with scales and pustules on the palms and soles. Palmoplantar pustulosis (PPP) area and severity index (PPPASI) was 16.8. Serologically, C-reactive protein and rheumatoid factor were negative. Lumbar magnetic resonance imaging (MRI) revealed bone marrow edema in L4 and L5 under T2-weighted condition, corroborating the presence of spondylitis (Figure 1A). 99mTc bone scintigraphy showed increased uptakes in the right clavicle and sternoclavicular joint and the lumbar spines (Figure 1B). A diagnosis of PPP with pustulotic arthro-osteitis (PAO) was made. Three-month treatment with topical betamethasone butyrate propionate and maxacalcitol resulted in partial improvement of skin symptoms, and PPPASI decreased to 7.9 (Figure 1C). On the other hand, pain in the back and anterior chest was persistent. Her Bath Ankylosing Spondylitis Disease Activity Index (BASDAI) remained as high as 4.8. Because of insufficient improvement of skin symptoms and intractable back pain, treatment with anti-interleukin-23p19 subunit monoclonal antibody guselkumab was initiated. After 6-month guselkumab therapy, back pain visual analogue scale improved to 3 compared to 6 at the initiation of the treatment. In addition, BASDAI decreased to 4. Follow-up lumbar MRI demonstrated remarkable reduction of bone marrow edema in the vertebral bodies of L4 and L5 using short TI inversion recovery image (Figure 1D). Bone scintigraphy also confirmed decreased uptakes in the lumbar spines and the right clavicular region (Figure 1E). On the other hand, her anterior chest pain did not ameliorate even after the treatment. During the 6-month treatment, skin symptoms completely disappeared (Figure 1F).

The efficacy of guselkumab for PPP has been established,1, 2 and it is approved for PPP in Japan. However, very limited information is available on the efficacy of this drug for PAO.3 In particular, its effectiveness for spondylitis in PAO is largely unknown. We have recently reported that guselkumab improves joint pain of PAO patients as a whole.4 In a PPP clinical trial, 52-week guselkumab treatment showed beneficial outcomes for MRI scores as well as EQ-5D pain/discomfort scores in PPP patients with PAO, in which the spine, sacroiliac joint, and peripheral joints were considered together.3 It was also reported that guselkumab alleviated joint pain in a patient with ankylosing-spondylitis-type PAO,5 albeit follow-up imaging assessment was not performed after treatment. Therefore, to our knowledge, this is the first report showing the effectiveness of guselkumab for spondylitis due to PAO demonstrated not only by amelioration of clinical symptoms but also by the improvement in the imaging findings, which warrants further studies on the usefulness of guselkumab for PAO.

NI has received honoraria for speaker from AbbVie, Novartis, Kyowa-Kirin, Chugai Pharmaceutical, Taiho, and UCB. HF has received honoraria for speaker and consultancy from AbbVie, Eisai, Novartis, Janssen Pharmaceutical, Maruho, Taiho, Eli Lilly, Sun Pharma, and Mitsubishi Tanabe Pharma.

Approval of the research protocol: N/A.

Informed Consent: The patients provided informed consent for the publication of the images submitted with this article.

Registry and the registration No: N/A.

Animal Studies: N/A.

Abstract Image

guselkumab在脓疱性关节炎患者中成功治疗脊柱炎的临床和放射学研究
29岁日本女性,手掌和脚底有6个月的脓疱疹史。患者前胸腰椎疼痛僵直3个月,给予loxoprofen治疗。虽然她没有类风湿关节炎、银屑病关节炎或强直性脊柱炎病史,但她在3年前被诊断为子宫内膜子宫癌。经过手术和化疗治疗,她的肿瘤没有复发。她没有局灶性感染,也没有吸烟史。患者就诊时手掌和脚掌有红斑、鳞片和脓疱。掌跖脓疱病(PPP)面积及严重程度指数(PPPASI)为16.8。血清学上,活性蛋白和类风湿因子均为阴性。腰椎磁共振成像(MRI)显示t2weight条件下L4和L5骨髓水肿,证实脊柱炎的存在(图1A)。99mTc骨显像显示右锁骨、胸锁关节和腰椎摄取增加(图1B)。诊断为脓疱性关节骨炎(PAO)。外用丁酸丙酸倍他米松和麦卡骨糖醇治疗三个月后,皮肤症状部分改善,PPPASI降至7.9(图1C)。另一方面,背部和胸部前部的疼痛持续存在。她的浴缸强直性脊柱炎疾病活动指数(BASDAI)仍然高达4.8。由于皮肤症状和难治性背痛改善不足,开始使用抗白介素23p19亚基单克隆抗体guselkumab治疗。治疗6个月后,背痛视觉模拟评分从治疗开始时的6分提高到3分。此外,BASDAI下降到4。随访腰椎MRI显示L4和L5椎体骨髓水肿明显减少,使用短TI反演恢复图像(图1D)。骨显像也证实腰椎和右锁骨区域摄取减少(图1E)。另一方面,即使在治疗后,她的前胸痛也没有改善。治疗6个月后,皮肤症状完全消失(图1F)。guselkumab对PPP的疗效已经确立1,2,并且在日本被批准用于PPP。然而,关于该药对PAO的疗效的信息非常有限。3特别是其对PAO脊柱炎的疗效在很大程度上是未知的。我们最近报道了guselkumab整体上改善PAO患者的关节疼痛在一项PPP临床试验中,52周的guselkumab治疗在PPP合并PAO患者的MRI评分和EQ5D疼痛/不适评分方面显示出有利的结果,其中脊柱、骶髂关节和外周关节被一起考虑也有报道称,guselkumab缓解了强直性脊柱炎型PAO患者的关节疼痛5,尽管治疗后未进行随访影像学评估。因此,据我们所知,这是第一篇报道guselkumab治疗PAO引起的脊柱炎的有效性,不仅表现在临床症状的改善,而且表现在影像学表现的改善,这值得进一步研究guselkumab治疗PAO的有效性。
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来源期刊
CiteScore
0.60
自引率
10.00%
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69
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