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托法替布对一名儿童脱发患者的显著疗效:病例报告及文献复习

An Excellent Response to Tofacitinib in a Pediatric Alopecia Patient: A Case Report and Review.

作者信息

Brown Liza, Skopit Stanley

出版信息

J Drugs Dermatol. 2018 Aug 1;17(8):914-917.

Abstract

KD is an 8 year-old male patient who presented to our clinic in December 2016 with a history of patchy hair loss for many months duration that was worsening. KD's past medical history was notable for atopic dermatitis, and a positive family history of autoimmune thyroid disease. Upon examination he had well circumscribed areas of hair loss throughout his scalp, with exclamation mark hairs seen on dermoscopy. Eyebrows and eyelashes were intact, no epidermal changes of scale or erythema were noted on the scalp and no palpable lymph nodes were present. He was diagnosed with alopecia areata at this time and was treated with Clobetasol 0.05% solution QHS as well as Kenalog 2.5 mg/ml injections to the areas of hair loss. Patient followed up two months later with worsening of his alopecia at a rapid pace, presenting now with hair loss of the entire scalp and loss of the eyebrows. He was diagnosed with progression to alopecia universalis at that time, with a corresponding SALT (Severity of Alopecia Tool) score of 100. Both KD and his mother stated the hair loss was causing much distress in the patient's life both at school and at home. After a thorough discussion of treatment alternatives to include continued topical high dose steroids, intralesional injections, high dose oral methylprednisolone, topical irritation with anthralin, topical immunotherapy with diphenylcyclopropenone (DPCP) or squaric acid dibutylester (SADBE) and systemic immunosuppressives, both the mother, patient and clinician agreed to try tofacitinib 5 mg twice daily with continued usage of topical steroids. Patient and his family was counseled about support groups, and local meetings to ease the mental distress associated with this condition. After baseline labs were obtained and reported within normal limits, to include CBC, CMP, thyroid studies, lipids and Quanitferon gold, KD was started on tofacitinib 5 mg BID. Labs were repeated one month later, and 3 months ongoing thereafter. At KD's 3 month follow up, after starting tofacitinib 5 mg twice daily, KD showed complete regrowth of the eyebrows with minimal hair growth of the posterior occiput (Figure 1 a-d). At KD's 6 month follow up he had 100% regrowth of eyebrows and complete scalp regrowth, resulting in a SALT score of 0 (Figure 2). KD reported no side effects until month 6, after full hair regrowth, when patient started to report mild headaches. Drug holiday was offered but the patients family chose to discontinue treatment at this time as they were concerned side effects were secondary to medication usage. Unfortunately, patient was lost to follow-up after the discontinuation of treatment. From previous case reports we can postulate that his alopecia returned to baseline after discontinuation of tofacitinib. KD had an incredible response to treatment, as has been reported previously in literature of adolescents using these novel therapies. This is the youngest patient ever reported to be successfully treated with oral tofacitinib 5 mg twice daily for alopecia and its variants. J Drugs Dermatol. 2018;17(8):914-917.

摘要

KD是一名8岁男性患者,于2016年12月前来我院就诊,有数月斑秃病史且病情不断恶化。KD既往有特应性皮炎病史,家族中有自身免疫性甲状腺疾病阳性史。检查发现,他头皮上有界限清楚的脱发区,皮肤镜检查可见惊叹号样毛发。眉毛和睫毛完好无损,头皮未见鳞屑或红斑等表皮变化,未触及肿大淋巴结。此时他被诊断为斑秃,接受了0.05%氯倍他索溶液每晚一次外用治疗,以及在脱发区注射2.5mg/ml曲安奈德治疗。两个月后患者复诊,脱发迅速加重,现已发展为整个头皮脱发且眉毛脱落。当时他被诊断为进展为全秃,相应的SALT(脱发严重程度工具)评分为100分。KD及其母亲均表示,脱发给患者在学校和家中的生活带来了极大困扰。在全面讨论了包括继续外用高剂量类固醇、皮损内注射、高剂量口服甲泼尼龙、蒽林局部刺激、二苯环丙烯酮(DPCP)或二丁基硫酸 squarate(SADBE)局部免疫疗法以及全身免疫抑制剂等治疗方案后,母亲、患者和临床医生一致同意尝试每日两次服用5mg托法替布,并继续使用外用类固醇。向患者及其家人介绍了支持小组及当地会议,以缓解与该疾病相关的精神困扰。在获得包括血常规、生化全项、甲状腺检查、血脂和Quanitferon gold在内的基线实验室检查结果且报告均在正常范围内后,KD开始每日两次服用5mg托法替布。一个月后复查实验室检查,此后每3个月复查一次。在开始每日两次服用5mg托法替布3个月后的随访中,KD的眉毛完全重新生长,枕后部仅有少量头发生长(图1a - d)。在6个月的随访中,他的眉毛100%重新生长,头皮完全重新生长,SALT评分为0(图2)。直到第6个月头发完全重新生长后,KD才报告有轻微头痛,此前未报告有副作用。已建议停药,但患者家属此时选择停止治疗,因为他们担心副作用是药物使用所致。不幸的是,停药后患者失访。根据既往病例报告推测,停用托法替布后他的脱发情况恢复到了基线水平。KD对治疗有惊人的反应,正如之前文献中报道的青少年使用这些新型疗法的情况一样。这是有史以来报道的最年轻的成功接受每日两次口服5mg托法替布治疗斑秃及其变体的患者。《皮肤药物学杂志》。2018;17(8):914 - 917。

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