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病例漫谈11:斑秃毛发再生后发质的改变:附一病例报告 [复制链接]

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Alteration in Hair Texture Following Regrowth in Alopecia Areata: A Case Report
斑秃毛发再生后发质的改变:附一病例报告



Background
         Alopecia areata is a common cause of hair loss seen in 3.8% of patients in dermatology clinics and in 0.2% to 2.0% of the general US population. The pathology of the disease remains poorly understood. Hair loss in alopecia areata can range from a single patch to 100% loss of body hair. When hair regrowth occurs in alopecia areata, the new hair may demonstrate pigment alterations, but a change in hair texture (ie, curly or straight) has rarely been reported as a consequence of alopecia areata.
研究背景
    斑秃是常见的脱发性疾病,占皮肤科就诊患者的3.8%,占美国总人口的0.2%到2.0%。人们对该病的病理学仍然知之甚少。斑秃可以是孤立性的单个脱发斑,甚至全身所有体毛脱落。斑秃毛发再生后,新生的头发会发生色泽改变,但是目前很少有关于斑秃后发外形改变(曲或直)的报道。

Observations
   We report a case of a 13-year-old African American boy who experienced an alteration of hair shape following regrowth after alopecia areata. The new hair recapitulated his hair shape from early childhood.
临床观察
    我们报道了一例病例,患者为13岁非洲裔美国男性儿童,在斑秃毛发再生后发发外形发生了改变。新生的发质犹如儿童早期。

Conclusions
   The precipitating factor for a change in hair texture in alopecia areata may be a result of treatment, pathophysiologic changes, or a combination of both. Whether the change is triggered at the level of stem cell differentiation, by cytokine or hormonal influences, gene expression during hair follicle development, a combination of all of these, or an unknown cause is a question that remains to be answered.
结论
   斑秃发质改变可能的原因包括:治疗药物(或方法)和病理生理变化、或两者兼有。在干细胞分化水平上,这种改变是否受细胞因子或激素影响,毛囊发育期基因表达变化,或以上因素综合激发,或受其它某种未知因素影响,这一问题有待进一步探讨。

REPORT OF A CASE
    Alopecia areata (AA) is a common cause of hair loss seen in 3.8% of patients in dermatology clinics[1,2] and 0.2% to 2.0% of the general US population.[3,4] Usually up to 50% of patients recover within 1 year of treatment, but spontaneous remissions[5] and frequent relapses have been reported in up to 90% of cases.[6] Initially during regrowth, the new hair may have a smaller shaft size and may show pigment alterations ranging from total achromia to mild hypopigmentation, with a tendency toward total repigmentation over time.[7]
病例报告
   斑秃(AA)是导致脱发常见的病因,占皮肤科就诊患者的3.8%[1,2],占美国总人口的0.2%至2.0%[3,4]。通常,经一年治疗,治愈率高达50%。有文献报道,高达90%的病例会自然痊愈[5]但又频繁复发[6]。毛发再生的初期,新生毛发可能比较细,并会发生色泽变化,从完全色素缺乏到轻微色素减退,最终会趋向完全复色[7]。

   A review of the literature has shown that the shape (ie, curly or straight) of the new hair after regrowth is usually similar to the shape of the hair prior to the AA episode, although rare reports of hair shape changes have been documented [8]. Herein, we report a case of an alteration in hair shape following regrowth in a male adolescent patient with AA, where the regrowth resembles the patient’s hair shape from early childhood.
    文献回顾分析发现尽管有个别文献报道斑秃后新头发的外形(曲或直)会发生变化[8],但新生发的外形往往跟斑秃发作前的相似。在此,我们报道了一例患有斑秃的男性青少年患者,该患者头发再生后发生了发外形的变化。新生发与他儿童早期的发外形类似。

    A 13-year-old African American boy presented with “a few months of hair loss.” His medical history included seborrheic dermatitis limited to the scalp, self-treated with an over-the-counter medicated shampoo; and seasonal allergic rhinitis and hypersensitivity to cat allergens, both treated with a mometasone furoate monohydrate nasal spray as needed. His family history was significant for hypothyroidism diagnosed in his mother and maternal grandmother, and negative for AA.
    一名13岁的非洲裔美国男性儿童,因“脱发数月”就诊。他的病史包括:头皮的脂溢性皮炎,自行使用非处方药物的洗发水进行治疗;季节性过敏性鼻炎和对猫的过敏症,均使用糠酸莫米他松鼻喷雾剂治疗。他的母亲和外祖母有甲状腺功能减退病史,无斑秃史。

    Physical examination revealed a 5×3-cm patch of hair loss on the right supratemporal hairline and a 3.5×2.0-cm patch of hair loss on the vertex of the scalp, with evidence of partial regrowth. There was no evidence of cicatricial alopecia or skin lesions suggestive of discoid lupus erythematosus. In addition, nail pitting was not seen. Relevant laboratory findings included mild elevation in the serum level of free thyroxine.
    体格检查显示在右侧颞上发际线处有一5×3cm2以及顶部一3.5×2.0cm2的头发缺失斑。没有瘢痕性秃发或盘状红斑性狼疮性脱发的迹象。另外,没有发现指甲凹点。相关实验发现血清游离甲状腺素水平轻微升高。



Figure. The areas of prior alopecia displayed a different shape (ie, straight) in contrast to the curly shape seen on the rest of the patient’s scalp.
图. 与患者头皮的其他部位头发的卷曲外形相比,斑秃区域再生发表现为不同的外形(直)。




    After the clinical diagnosis of AA was made, the patient was treated with clobetasol propionate foam, 0.05%, once daily; triamcinolone acetonide cream, 0.1%, twice daily Monday through Friday; and clobetasol propionate cream, 0.05%, twice daily Saturday through Sunday.
    在临床诊断为斑秃后,患者使用0.05%丙酸氯倍他索洗发水,每日一次;周一到周五每日两次外用0.1%醋酸曲胺萘德乳膏;周六到周日每日两次外用0.05%丙酮氯倍他米松乳膏进行治疗。

    After 3 months, the patient returned for a follow-up visit and showed evidence of substantial hair regrowth in both areas of prior alopecia. In addition, the newly growing hair in the 2 areas of prior alopecia displayed a different shape (ie, straight), in contrast to the curly shape seen on the rest of the patient’s scalp (Figure). The patient’s mother confirmed that the hair shape displayed in those specific areas of prior AA was similar to the hair shape seen in the patient during childhood. At a 1-year follow-up visit, the patient had not developed any new alopecic patches, and the hair growing in the areas of prior AA continued to display a straight shape, in contrast to the rest of the hair on his scalp.
    三个月后随访显示以前脱发的两处区域有头发明显再生。此外,与患者头皮其他区域的卷曲的头发形成相比,两处斑秃区域里新长出的头发表现为不同的外形(直)。患者的母亲证实斑秃的再生发外形跟患者儿童时期的头发外形相似(直)。一年后随访,患者没有发生新斑秃,此前斑秃区域新生的头发继续表现为直线外形。


COMMENT
    Hair shape, or curliness, is a spectrum ranging from straight hair to tightly curled (frizzy) hair. [9] Prior classifications of hair shape included 3 categories based on ethnicity: African, Asian, and European. Recently, several morphologic parameters have been used to broaden the hair shape classification to 8 types, independent of ethnicity. These parameters include the curve diameter, the curl index (which measures the ratio of the stretched length of the hair related to its length at rest), the highest number of waves in the hair when it is pulled to four-fifths of its length, and the number of twists detected along the fiber.[9,10]
评论
    头发外形,或卷曲,是从直发到紧紧卷曲头发的变动范围[9]。头发外形曾基于种族分为3类:非洲人种、亚洲人种和欧洲人种。最近,根据几个形态学参数将头发外形分类扩大展到八种不依赖于种族的外形。这些参数包括曲线直径、旋度指数(头发拉伸长度与静止长度的比率)、头发的最高波数(当头发维持在它长度的五分之四时),以及顺着头发检测到的扭曲次数[9,10]。

    There are several theories as to what generates the degree of hair curvature. In mouse models, mutations in transforming growth factor α(TGF-α), TGF-α receptor, and ETS2 (transcription factor receptor) cause mice to develop wavy hair and a change in follicular structure. [11] Another theory suggests that morphologic parameters of the hair shaft, such as the diameter and geometric contour, determine hair shape and are frequently related to ethnicity. Examination of hair shafts in cross-section reveals that thicker, curly hair, such as that of some African Americans, is ellipsoid, whereas the hair of European Americans is thin, with a spheroid appearance (this correlates with a straight hair phenotype).
    关于导致产生头发弯曲程度的原因存在几个理论。在小鼠模型中,转化生长因子α(TGF-α),TGF-α受体,和ETS2(转录因子受体)的突变引起小鼠产生卷毛和毛囊结构的改变[11]。另一个理论表明头发外形的形态学参数,例如直径和几何轮廓,决定了头发的外形并且通常跟种族有关。检查揭示了更粗的卷曲头发,例如某些非裔美国人,毛干横截面呈椭圆体,然而欧裔美国人的头发较细的、毛干横截面呈圆形这跟直发表型相关联)。


   Immunohistochemical and in vitro studies performed by Thibaut et al [12] suggest that hair shape is more of a dynamic process, affected by the expression of certain proteins regulated by the hair bulb as well as by mechanical stress induced by myofibroblasts. Proliferative markers, specifically Ki-67, show increased expression in an asymmetrical manner on the convex side of curly hairs within the metrical cells, which may contribute to curving. [12,13]
    Thibaut等人进行的免疫组化和体外研究表明头发外形更多的是一个动态过程,受由毛球调节的某些蛋白质表达以及由肌纤维母细胞诱导的机械应力的影响。增生的标记物,特别是Ki-67,表现为在卷曲头发的突出处以不对称的方式过高表达,这可能有与毛发弯曲有关[12,13]。


    Similarly, α–smooth muscle actin expression, a marker for myofibroblasts, is increased on the concave surface creating a mechanical imbalance, which may contribute to kinking. [14] Interestingly, when the proximal portion of curly hair is cultured in vitro, it maintains its shape; this suggests that at the very least, hair shape can be maintained by the follicle with no input from other adnexal structures. [12]
    同样的,α–平滑肌动蛋白表达(肌纤维母细胞的标记物)在凹面处增多,产生一种机械不平衡,这样就促成了屈曲。有趣的是,当卷发的近端部分在体外培养时,它的外形得到保持;这至少表明头发的外形由毛囊维持,不需要其他附件结构[12]。


   Acquired straightening of the hair has been described patients with human immunodeficiency virus as well as in patients with chronic malnutrition [15,16] and has been ascribed to mineral deficiencies and hormonal dysfunction. Kinking of the hair (in addition to pigmentation changes) has also been reported with the use of acitretin and etretinate, which are thought to be associated with a change in keratinization patterns within the inner root sheath.[17,18]
    头发的后天矫直可存在于艾滋病患者以及长期营养不良患者中[15,16],并认为与微量元素缺乏和内分泌功能障碍有关。有报道阿维A酸和依曲替酯可促使的头发屈曲(除颜色改变之外),这被认为是与內毛根鞘部角质化模式的改变有关[17,18]。


    To our knowledge, this is the first report of a patient with AA who experienced a change in hair structure that recapitulated the patient’s hair texture as a child. The precipitating factor for this change may have been a result of treatment of AA, pathophysiologic changes associated with the AA, or a combination of both.
    据我们所知,这是第一次有关斑秃患者的头发结构发生改变,重现了患者儿童时期的头发结构的报道。这一变化的诱发因素可能是斑秃治疗的结果、有关斑秃病理生理的改变,或者两者皆有。


    The pathogenesis underlying AA is yet to be fully understood. It is clear that immune dysregulation consisting of cytokines, hormones, and T cells cause the hair cycle to become dysfunctional and come to a halt.[19] When the hair follicle reenters anagen, the same signaling molecules active during morphogenesis (ie, WNT and SHH) trigger hair growth.[11,20] In their work on hair follicle development, Legue´ et al [21] suggest that each hair cycle is a distinct morphogenetic event during which hair stem cells, under the influence of growth factors and cytokines, migrate from the bulge to promote new follicle growth, mostly maintaining their omnipotent properties.
    斑秃的发病机理尚未得到完全理解。明确的原因包括细胞因子、激素和T淋巴细胞在内的的免疫失调导致毛发周期功能障碍仍至停滞等[19]。当毛囊重新进入生长期时,同样的形态发生信号(即WNT和SHH)激发毛发生长[11,20]。Legue´等人提出每一次的头发周期都是一个不同的形态发生事件。在此周期中,在生长因子和细胞因子的作用下,头发干细胞从凸处(毛囊生长点)移出促进新的毛囊生长,并维持其全能性。


    In our patient, hair regrowth was achieved, but the pathologic changes induced by inflammation and/or treatment caused an alteration of hair texture. Whether the change was triggered at the level of stem cell differentiation, by cytokine or hormonal influences, by gene expression during hair follicle development, a combination of these, or an unknown cause is a question that remains to be answered. Elucidating triggers in hair texture change could help us further understand AA. Furthermore, it could have a major impact on the world of hair grooming, where countless hours are spent by people attempting to change their hair texture.
    我们报道的患者头发获得了重新生长,但是发质发生了改变,可能是由于炎症和/或治疗引起的病理变化。这种改变是否是在干细胞分化水平上,通过细胞因子或激素的作用、毛囊发育期基因表达变化,或二者的综合作用,抑或是某种未知的原因,这仍是一个有待解答的问题。阐明发质改变的原因能帮助我们进一步了解斑秃。此外,还能对头发美容领域产生重要的影响,在这个领域里,人们花了大量的时间试图改变他们的发质。


参考文献
1. Sharma VK, Dawn G, Kumar B. Profile of alopecia areata in Northern India. Int J Dermatol. 1996; 35(1):22-27.
2. Tan E, Tay YK, Goh CL, et al. The pattern and profile of alopecia areata in Singapore: a study of 219 Asians. Int J Dermatol. 2002;41(11):748-753.
3. Safavi K. Prevalence of alopecia areata in the First National Health and Nutrition Examination Survey. Arch Dermatol. 1992; 128(5):702.
4. National Alopecia Areata Foundation. About alopecia areata. Accessed on May 24, 2011.
5. Shapiro J, Madani S. Alopecia areata: diagnosis and management. Int J Dermatol. 1999; 38(suppl 1):19-24.
6. Muller SA, Winkelmann RK. Alopecia areata: an evaluation of 736 patients. Arch Dermatol. 1963; 88:290-297.
7. Berker DAR, Messenger AG, Sinclair RD. Disorders of hair. In: Burns DA, Breathnach SM, Cox N, Griffiths CE, eds. Rook’s Textbook of Dermatology. Vol 4. 7th ed. Oxford, England: Wiley-Blackwell; 2004:63.1-63.120.
8. Weinburg S, Pros NS, Kristal L. Color Atlas of Pediatric Dermatology. 3rd ed. New York, NY: McGraw-Hill Professional; 1997.
9. Loussouarn G, Garcel AL, Lozano I, et al. Worldwide diversity of hair curliness: a new method of assessment. Int J Dermatol. 2007;46 (suppl 1):2-6.
10. De la Mettrie R, Saint-Le´ger D, Loussouarn G, Garcel A, Porter C, Langaney A. Shape variability and classification of human hair: a worldwide approach. Hum Biol. 2007;79 (3):265-281.
11. Millar SE. Molecular mechanisms regulating hair follicle development. J Invest Dermatol. 2002;118 (2):216-225.
12. Thibaut S, Gaillard O, Bouhanna P, Cannell DW, Bernard BA. Human hair shape is programmed from the bulb. Br J Dermatol. 2005; 152(4):632-638.
13. Scholzen T, Gerdes J. The Ki-67 protein: from the known and the unknown. J Cell Physiol. 2000; 182(3):311-322.
14. Skalli O, Pelte MF, Peclet MC, et al. Alpha-smooth muscle actin, a differentiation marker of smooth muscle cells, is present in microfilamentous bundles of pericytes. J Histochem Cytochem. 1989; 37(3):315-321.
15. Smith KJ, Skelton HG, DeRusso D, et al. Clinical and histopathologic features of hair loss in patients with HIV-1 infection. J Am Acad Dermatol. 1996; 34(1):63-68.
16. Green SL, Nelson DL. Straightening of the hair is not pathognomonic for HIV infection. Clin Infect Dis. 2002; 35(10):1276-1277.
17. Seckin D, Yildiz A. Repigmentation and curling of hair after acitretin therapy. Australas J Dermatol. 2009; 50(3):214-216.
18. Graham RM, James MP, Ferguson DJ, Guerrier CW. Acquired kinking of the hair associated with etretinate therapy. Clin Exp Dermatol. 1985; 10(5):426-431.
19. Gregoriou S, Papafragkaki D, Kontochristopoulos G, Rallis E, Kalogeromitros D, Rigopoulos D. Cytokines and other mediators in alopecia areata. http://www.ncbi .nlm.nih.gov/pmc/articles/PMC2837895/?tool=pubmed. Accessed May 23, 2011.
20. Enshell-Seijffers D, Lindon C, Kashiwagi M, Morgan BA. Beta-catenin activity in the dermal papilla regulates morphogenesis and regeneration of hair. Dev Cell. 2010; 18(4):633-642.
21. Legue´ E, Sequeira I, Nicolas JF. Hair follicle renewal: authentic morphogenesis that depends on a complex progression of stem cell lineages. Development. 2010; 137(4):569-577.



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