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Dupilumab Efficacy and Safety in Severe Atopic Dermatitis in Children under 6 Years of Age: Two Clinical Cases Dupilumab治疗6岁以下儿童严重特应性皮炎的疗效和安全性:2例临床病例
Q4 Medicine Pub Date : 2023-11-06 DOI: 10.15690/vsp.v22i5.2622
Nikolay N. Murashkin, Leonid A. Opryatin, Ekaterina S. Pavlova, Roman V. Epishev, Alexandr I. Materikin, Roman A. Ivanov, Alena A. Savelova, Maria A. Leonova
Background . Atopic dermatitis (AD) manifests during the first year of life in majority of all cases. The early disease onset is associated with the development of comorbid atopic conditions within the «atopic march» phenomenon. The AD pathogenesis is associated with genetic predisposition, epidermal barrier dysfunction, and immune dysregulation. T2-inflammation specifically determines the entire immune cascade of inflammatory reactions, and, thus, dictates the need of early drug intervention to modify the disease course. Clinical case description. This article presents two clinical cases of severe AD in children under 6 years of age. The treatment of both cases included genetically engineered biologic drug dupilumab. Continuous therapy for 4–5 months made it possible to relieve the skin manifestations of the disease. Conclusion . AD, manifesting in infancy, is associated with high risk of developing other atopic spectrum diseases in older age. The timely onset of biological therapy allows us to affect immune dysregulation, and thereby to prevent the comorbid atopic conditions development.
背景。特应性皮炎(AD)在大多数情况下表现在生命的第一年。在“特应性行军”现象中,疾病的早期发病与共病特应性疾病的发展有关。AD的发病机制与遗传易感性、表皮屏障功能障碍和免疫失调有关。t2炎症特异性地决定了炎症反应的整个免疫级联反应,因此,需要早期药物干预来改变疾病进程。临床病例描述。本文报道两例6岁以下儿童重度AD的临床病例。这两个病例的治疗包括基因工程生物药物杜匹单抗。持续治疗4-5个月,可以缓解疾病的皮肤表现。结论。阿尔茨海默病表现于婴儿期,与老年时发生其他特应性谱系疾病的高风险相关。及时的生物治疗使我们能够影响免疫失调,从而防止共病的特应性疾病的发展。
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引用次数: 0
Linear IgA Dermatosis in a Child: Clinical Case 1例儿童线状IgA皮肤病临床病例
Q4 Medicine Pub Date : 2023-11-06 DOI: 10.15690/vsp.v22i5.2642
Nikolay N. Murashkin, Alexander I. Materikin, Roman V. Epishev, Maria A. Leonova, Leonid A. Opryatin, Roman A. Ivanov, Alena A. Savelova, Oksana R. Katunina, Kirill A. Kulikov, Daniel R. Danielyan
Background. Linear IgA dermatosis is a rare autoimmune bullous disease characterized by vesiculo-bullous subepidermal lesions (affecting the skin and mucous membranes) and by linear homogeneous IgA deposition in the epidermis basal membrane. Clinical case description. Clinical case of linear IgA dermatosis in children is presented. Clinical data assessment, histological examination of the skin biopsy (to determine the depth of bullous), immunofluorescent examination (to reveal IgA deposition in the epidermis basal membrane) are crucial for reliable disease diagnosis according to the studies. Altogether it helps to establish the final diagnosis and determine the patient's management. Dapsone is the first-line treatment for this disease, it has proven to be an effective and safe medication. Conclusion. This case of linear IgA dermatosis is of concern due to disease severity and its rarity in clinical practice. Differential diagnosis is rather complicated and clinically requires high-tech research methods. Only immunofluorescent examination allows to diagnose linear IgA dermatosis accurately. Lesions' regression was achieved due to systemic therapy with dapsone (1.8 mg/kg/day).
背景。线性IgA皮肤病是一种罕见的自身免疫性大疱性疾病,其特征是表皮下的囊泡大疱性病变(影响皮肤和粘膜)和表皮基膜的线性均匀IgA沉积。临床病例描述。报告1例儿童线状IgA皮肤病。临床资料评估、皮肤活检组织学检查(确定大疱深度)、免疫荧光检查(显示表皮基膜IgA沉积)对可靠的疾病诊断至关重要。总之,它有助于建立最终诊断和确定病人的管理。氨苯砜是这种疾病的一线治疗药物,它已被证明是一种有效和安全的药物。结论。本例线状IgA皮肤病因其严重程度和临床罕见而引起关注。鉴别诊断相当复杂,临床上需要高科技的研究方法。只有免疫荧光检查才能准确诊断线性IgA皮肤病。病变的消退是由于全身治疗氨苯砜(1.8 mg/kg/天)。
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引用次数: 0
TNF-α Inhibitor-Induced Psoriasis and Psoriatic Alopecia in Adolescent with Ulcerative Colitis: Clinical Case TNF-α抑制剂诱导的青少年溃疡性结肠炎银屑病和银屑病性脱发的临床研究
Q4 Medicine Pub Date : 2023-11-06 DOI: 10.15690/vsp.v22i5.2635
Eduard T. Ambarchyan, Vladislav V. Ivanchikov, Anna L. Arakelyan, Andrey N. Surkov, Anastasia D. Kuzminova, Evgeny E. Bessonov, Elena V. Komarova
Background . Genetically engineered biological therapy has revolutionized the treatment of many chronic inflammatory diseases. It often allows to achieve significant clinical effect and improve the patient's quality of life. However, sometimes it leads to adverse events, and physicians encounter them more often. One of such side effects is paradoxical psoriasis (PP) that can be revealed during the therapy with tumor necrosis factor alpha (TNF-α) inhibitors. PP is the debut or exacerbation of pre-existing psoriasis. Clinical case description . Patient A., 17 years old, has suffered from ulcerative colitis since 2020; biological therapy with infliximab has been initiated in March 2022. Numerous rashes were revealed, as well as development of confluent alopecia foci on the background of scalp psoriatic damage, during the next hospitalization in December 2022. The patient was examined by dermatologist; diagnosis of PP was established according to the clinical picture and medical history. Skin rashes progressed and ulcerative colitis worsened (diarrheal syndrome, fecal calprotectin levels increased up to 526 μg/g) after cessation of infliximab therapy. Biological therapy with the inhibitor IL-12/23 (ustekinumab) was initiated due to the aggressive cutaneous pathological process and the aggravation of inflammatory bowel disease (IBD) symptoms. The gradual regression of rashes with the restoration of scalp hair growth and ulcerative colitis clinical and laboratory remission were noted during the treatment. Conclusion. PP is a rare complication that develops during therapy with TNF-α inhibitors, and it is most often observed in patients with IBD. In our case there was aggressive course of psoriasis with severe scalp lesion and hair loss (it is specific type of lesion in such patients). The ustekinumab, inhibitor IL-12/23, treatment efficacy correlates with the literature data on this drug successful use in both nosologies. Ustekinumab can be a first-line therapy in such pediatric patients. This clinical case is the first case in the Russian literature on effective management of PP and ulcerative colitis with ustekinumab in children.
背景。基因工程生物疗法已经彻底改变了许多慢性炎症性疾病的治疗。往往可以取得显著的临床效果,提高患者的生活质量。然而,有时它会导致不良事件,医生更经常遇到它们。其中一个副作用是悖论性牛皮癣(PP),可以在肿瘤坏死因子α (TNF-α)抑制剂治疗期间显示。PP是原有牛皮癣的首发或加重。临床病例描述。患者A, 17岁,自2020年以来患有溃疡性结肠炎;英夫利昔单抗的生物治疗已于2022年3月开始。在2022年12月的下一次住院期间,发现了许多皮疹,并在头皮银屑病损伤的背景下发展为合流性斑秃。皮肤科医生对患者进行了检查;根据临床表现和病史确定PP的诊断。停止英夫利昔单抗治疗后,皮疹进展,溃疡性结肠炎恶化(腹泻综合征,粪便钙保护蛋白水平升高至526 μg/g)。由于侵袭性皮肤病理过程和炎症性肠病(IBD)症状加重,开始使用抑制剂IL-12/23 (ustekinumab)进行生物治疗。在治疗期间,随着头皮毛发生长的恢复,皮疹逐渐消退,溃疡性结肠炎的临床和实验室缓解。结论。PP是一种罕见的并发症,在TNF-α抑制剂治疗期间发生,最常见于IBD患者。在我们的病例中,银屑病病程严重,伴有严重的头皮损伤和脱发(这是此类患者的特殊病变类型)。ustekinumab,抑制剂IL-12/23,治疗效果与该药物在两种疾病中成功使用的文献数据相关。Ustekinumab可作为此类儿科患者的一线治疗。该临床病例是俄罗斯文献中第一例使用ustekinumab有效治疗儿童PP和溃疡性结肠炎的病例。
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引用次数: 0
Adalimumab Experience in the Management of Hidradenitis Suppurativa Burdened with Rapp-Hodgkin Syndrome 阿达木单抗治疗伴拉普-霍奇金综合征的化脓性汗腺炎的经验
Q4 Medicine Pub Date : 2023-11-06 DOI: 10.15690/vsp.v22i5.2623
Nikolay N. Murashkin, Alexander I. Materikin, Roman V. Epishev, Maria A. Leonova, Leonid A. Opryatin, Dmitri V. Fedorov, Roman A. Ivanov, Alena A. Savelova, Oksana R. Katunina, Kirill A. Kulikov, Anastasiya U. Ufimtseva
Background. Rapp-Hodgkin syndrome is a rare genetic disease from the ectodermal dysplasia group. It manifests with damage of ectodermal structures — layer of embrional tissue that provides the development of many organs and tissues in the body, such as: skin, sweat glands, hair, teeth, and nails. Disease symptoms may range from mild to severe and include dental abnormalities, fragile, thin or no hair, abnormal nails, hypohidrosis (due to the reduced number of sweat glands), cleft lips and palate. Hidradenitis suppurativa is a chronic inflammatory disease that predominantly affects apocrine glands areas. This nosology is characterized by painful nodes, abscesses, and fistulas, prone to relapses and leading to scarring.Clinical case description. The authors present a clinical case demonstrating the combination of Rapp-Hodgkin syndrome and severe hidradenitis suppurativa in 17-year-old male patient.Conclusion. The heterogeneous clinical findings of hidradenitis suppurativa and history of rare genetic disease result in patients' admissions to various medical specialists. Thus, it leads to delayed verification of the diagnosis and incorrect treatment methods. As a result, patients have persistent cosmetic skin defects, possible complications of irrational treatment, and decreased quality of life by the time of diagnosis.
背景。拉普-霍奇金综合征是一种罕见的遗传性疾病,来自外胚层发育不良组。它表现为外胚层结构的损伤,外胚层是胚胎组织,提供身体许多器官和组织的发育,如皮肤、汗腺、头发、牙齿和指甲。疾病症状从轻微到严重不等,包括牙齿异常、脆弱、毛发稀疏或无毛、指甲异常、汗腺减少(由于汗腺数量减少)、唇腭裂。化脓性汗腺炎是一种慢性炎症性疾病,主要影响大汗腺区域。这种疾病的特点是疼痛的淋巴结、脓肿和瘘管,容易复发并导致瘢痕形成。临床病例描述。作者提出了一个临床病例显示合并拉普-霍奇金综合征和严重化脓性汗腺炎在17岁的男性患者。结论。化脓性汗腺炎的不同临床表现和罕见遗传病史导致患者就诊于不同的医学专家。因此,它导致诊断的验证延迟和错误的治疗方法。因此,患者在诊断时存在持续的美容皮肤缺陷,可能出现不合理治疗的并发症,生活质量下降。
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引用次数: 0
Results of Combined Pathogenetic Therapy in Netherton Syndrome: Clinical Case 内瑟顿综合征综合病因治疗的临床效果
Q4 Medicine Pub Date : 2023-11-06 DOI: 10.15690/vsp.v22i5.2621
Nikolay N. Murashkin, Leonid A. Opryatin, Anna S. Bridan-Rostovskaya, Ekaterina S. Pavlova, Roman A. Ivanov, Karine O. Avetisyan, Kirill A. Kulikov
Background . Netherton syndrome is a severe autosomal recessive disease based not only on genetically determined keratinization disorders, but also on immune system dysregulation. Clinical case description . Boy K., 9 years old, diagnosed with severe atopic dermatitis and revealed Netherton syndrome during examinations. The dupilumab was administered due to the presence of severe polysensitization and clinical signs of allergic process. Wave-like course of the disease was observed during this therapy. Patient had acute exacerbation — generalized pustular rash — at the 7th month of treatment. The therapy with secukinumab was initiated according to the foreign literature on pathogenetic treatment of Netherton syndrome with interleukin (IL) 17 inhibitor. It has shown positive dynamics. Currently patient is administered with the combined therapy of IL-17 and IL-4/13 inhibitors with significant positive effect. Conclusion. Genetically engineered biologic drugs targeting both the Th17 and Th2 have shown their efficacy in Netherton syndrome management.
背景。内瑟顿综合征是一种严重的常染色体隐性遗传病,不仅基于遗传决定的角化疾病,而且基于免疫系统失调。临床病例描述。男童K., 9岁,诊断为严重特应性皮炎,检查时发现内瑟顿综合征。由于存在严重的多致敏和过敏过程的临床体征,给予杜匹单抗。在此治疗期间观察到疾病的波浪形病程。患者在治疗第7个月出现急性加重-广泛性脓疱性皮疹。根据国外关于白细胞介素(IL) 17抑制剂治疗内瑟顿综合征的发病文献,启动了secukinumab治疗。它已经显示出积极的动力。目前患者采用IL-17和IL-4/13抑制剂联合治疗,疗效显著。结论。针对Th17和Th2的基因工程生物药物在内瑟顿综合征的治疗中已显示出其疗效。
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引用次数: 0
Pemphigus Herpetiformis in Childhood: Clinical Case 儿童期疱疹样天疱疮临床一例
Q4 Medicine Pub Date : 2023-11-06 DOI: 10.15690/vsp.v22i5.2636
Nikolay N. Murashkin, Alexander I. Materikin, Roman V. Epishev, Maria A. Leonova, Leonid A. Opryatin, Dmitri V. Fedorov, Roman A. Ivanov, Alena A. Savelova, Oksana R. Katunina, Kirill A. Kulikov, Ilgiz I. Yarullin
Background. Pemphigus herpetiformis is a rare atypical bullous dermatosis of autoimmune nature. It is characterized by rashes in the form of plaques with papules and vesicles at peripheral areas, or in the form of grouped papules, vesicles or tense bubbles with clinical similarity to During's herpetiformis dermatitis. Pemphigus herpetiformis diagnosis in pediatric practice is difficult due to the similarity of clinical manifestations to bullous dermatoses of other etiology, the rarity of the disease in children, and small number of scientific publications. Clinical case description . Clinical case of pemphigus herpetiformis in a child is described. All necessary diagnostic algorithms for diagnosis and therapeutic tactics for patient management are presented. Dapsone and prednisolone were used as first-line drugs during the patient's treatment. They have shown its efficacy in management of such patients. Conclusion . Differential diagnosis of pemphigus herpetiformis and other bullous dermatoses in children is presented. The results of successful drug treatment are shown.
背景。疱疹样天疱疮是一种罕见的自身免疫性大疱性皮肤病。它的特点是在周围区域出现斑块、丘疹和小泡,或以成组丘疹、小泡或紧张气泡的形式出现,临床与During的疱疹样皮炎相似。由于临床表现与其他病因的大疱性皮肤病相似,儿童罕见,科学出版物少,因此在儿科实践中诊断疱疹样天疱疮是困难的。临床病例描述。描述了一个儿童疱疹样天疱疮的临床病例。提出了所有必要的诊断算法和患者管理的治疗策略。在患者治疗期间,应用氨苯砜和强的松龙作为一线药物。他们已经证明了它在治疗这类病人方面的有效性。结论。提出了儿童疱疹样天疱疮和其他大疱性皮肤病的鉴别诊断。显示了药物治疗成功的结果。
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引用次数: 0
To the Anniversary of Elena Nikolaevna Baibarina 埃琳娜·尼古拉耶夫娜·巴巴里娜的周年纪念
Q4 Medicine Pub Date : 2023-11-06 DOI: 10.15690/vsp.v22i5.2644
Article Editorial
.
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引用次数: 0
Features of Cell-Mediated Immunity in Children with Congenital Ichthyosis and Their Role in the Pathogenetic Management 先天性鱼鳞病患儿细胞免疫的特点及其在发病管理中的作用
Q4 Medicine Pub Date : 2023-11-05 DOI: 10.15690/vsp.v22i5.2645
Karine O. Avetisyan, Nikolay N. Murashkin, Svetlana G. Makarova, Svetlana S. Petrichuk, Daria G. Kuptsova
Background. Congenital ichthyoses (CIs) are a heterogeneous clinical-etiological group of genodermatoses. Typical clinical symptoms of this disease, regardless of the form, are generalized erythroderma, peeling, itching, hyperkeratosis, severe structural and functional disorders of the epidermal barrier, other organs and systems. Patients have an extremely low quality of life due to changes in appearance, discomfort, constant disease symptoms. Thus far, there are no effective treatment methods for ichthyosis. That is why scientific search for new therapies is the topical issue in pediatrics and pediatric dermatology. Objective . The aim of the study is to examine the cell-mediated immunity state in patients with CI via assessment of the pattern of lymphocyte subpopulations in peripheral blood. The research was conducted to study the content of the main and small lymphocyte subpopulations in 86 patients with established diagnosis of CI aged from 1 month to 18 years. The diagnosis was made according to the clinical data and the results of molecular genetic testing. Comparative analysis of blood immunological indicators in children with CI and in patients with other immunemediated chronic dermatoses: atopic dermatitis (AD; n = 68) and psoriasis vulgaris (n = 55). Methods. The level of T lymphocytes, T helpers (Th), cytotoxic T lymphocytes (Tc), B lymphocytes, NK cells, Treg-cells (Treg), activated T helpers (Thact), Th17 lymphocytes in peripheral blood was evaluated via flow cytometry using monoclonal antibodies. Statistical analysis was performed via Statistica 10.0. Differences between the groups were assessed via Mann-Whitney non-parametric test, differences were considered significant at p < 0.05. Results . A significant increase of activated T-helpers level in peripheral blood was revealed in patients with CI and psoriasis compared to children with AD (p < 0.001), as well as an increased levels of B-lymphocytes and Treg in children with CI (p < 0.05). Conclusion. Children with CI have shown some features of cell-mediated immunity such as: pathological activation of Th lymphocytes, impaired terminal differentiation of naive CD4+ cells to Thact, Treg, Th17 lymphocytes and their proliferation. Comparative analysis of mentioned immunological indicators in children with CI, psoriasis and AD has shown comparable results of increased Thact lymphocytes levels in patients in CI and psoriasis groups. This results open up potential of using immunobiological drugs of psoriasis target therapy within the new management strategy for children with CI.
背景。先天性鱼鳞病(CIs)是一种异质的临床病因组遗传性皮肤病。本病的典型临床症状,不论形式如何,均为全身性红皮病、脱皮、瘙痒、角化过度、严重的表皮屏障及其他器官和系统的结构和功能障碍。患者的生活质量极低,由于外观的变化,不适,持续的疾病症状。迄今为止,尚无有效的治疗鱼鳞病的方法。这就是为什么科学研究新疗法是儿科和儿科皮肤病学的热门问题。目标。该研究的目的是通过评估外周血淋巴细胞亚群的模式来检查CI患者的细胞介导免疫状态。本研究对86例年龄1个月~ 18岁确诊CI患者的主要淋巴细胞亚群和小淋巴细胞亚群的含量进行了研究。根据临床资料和分子基因检测结果作出诊断。CI患儿与其他免疫介导型慢性皮肤病患者血液免疫学指标的比较分析:特应性皮炎(AD;寻常型牛皮癣(N = 55)。方法。采用单克隆抗体,流式细胞术检测外周血T淋巴细胞、T辅助细胞(Th)、细胞毒性T淋巴细胞(Tc)、B淋巴细胞、NK细胞、Treg细胞(Treg)、活化T辅助细胞(Thact)、Th17淋巴细胞的水平。采用Statistica 10.0软件进行统计分析。通过Mann-Whitney非参数检验评估组间差异,p <认为差异显著;0.05. 结果。与AD患儿相比,CI和银屑病患者外周血中活化t辅助细胞水平显著升高(p <0.001),以及CI患儿b淋巴细胞和Treg水平升高(p <0.05)。结论。CI患儿表现出细胞免疫的一些特征,如:Th淋巴细胞病理性活化,初始CD4+细胞向Thact、Treg、Th17淋巴细胞的终末分化及增殖受损。对CI、牛皮癣和AD患儿上述免疫指标的比较分析显示,CI组和牛皮癣组患者的Thact淋巴细胞水平升高具有可比性。这一结果开辟了在CI患儿的新管理策略中使用免疫生物学药物靶向治疗牛皮癣的潜力。
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引用次数: 0
Modern Treatment Options for Epidermal Dysfunction at Atopic Dermatitis 特应性皮炎表皮功能障碍的现代治疗选择
Q4 Medicine Pub Date : 2023-11-05 DOI: 10.15690/vsp.v22i5.2619
Eduard T. Ambarchyan, Anastasiya D. Kuzminova, Vladislav V. Ivanchikov
Atopic dermatitis (AD) is a chronic inflammatory skin disease. Its crucial component of pathogenesis is malfunction of the epidermal barrier. Filaggrin protein and associated mutations in the filaggrin gene play one of the key roles in this problem. Nowadays new topical products (emollients) has been created and implemented into practice with the aim of more personalized approach and increased therapy efficacy in patients with AD. Such drugs would allow us to restore epidermal barrier function and to achieve elimination of disease symptoms.
特应性皮炎(AD)是一种慢性炎症性皮肤病。其发病机制的关键组成部分是表皮屏障功能障碍。聚丝蛋白和相关的聚丝蛋白基因突变在这一问题中起着关键作用。如今,新的局部产品(润肤剂)已经被创造出来并付诸实践,目的是为AD患者提供更个性化的治疗方法,提高治疗效果。这些药物将使我们能够恢复表皮屏障功能,实现疾病症状的消除。
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引用次数: 0
Features of Staphylococcus Aureus Antibiotic Sensitivity in Children with Atopic Dermatitis 特应性皮炎患儿金黄色葡萄球菌抗生素敏感性特点
Q4 Medicine Pub Date : 2023-11-05 DOI: 10.15690/vsp.v22i5.2640
Nikolay N. Murashkin, Alexander I. Materikin, Roman V. Epishev, Maria A. Leonova, Leonid A. Opryatin, Roman A. Ivanov, Alena A. Savelova
Background. Excessive colonization of the skin by various bacteria and fungi can be noted in patients with atopic dermatitis (AD), and the prevalence of secondary infection complications 30–48%. Several studies have shown that Staphylococcus aureus colonization is 60–100% in patients with AD compared to 5–30% in healthy persons from the control group. Moreover, the incidence of methicillinresistant Staphylococcus aureus (MRSA) isolates is up to 10–30% in skin cultures at AD, according to experts. Therapy of AD complicated by secondary infection is one of the crucial challenges of modern dermatology. Mupirocin can be considered as one of the most effective topical antibiotic among others used for etiotropic therapy of infectious complications in AD patients, and it has been confirmed by numerous clinical studies. Conclusion. Staphylococcus aureus is the most common trigger of AD aggravation. MRSA in AD complicated by secondary infection is difficult to manage as it becomes resistant to many types of antibiotics; however, it shows persistent sensitivity to mupirocin. Mupirocin-based (2%) external agent is the most effective, safe, and preferred therapy variant for AD complicated by secondary infection in pediatrics.
背景。特应性皮炎(AD)患者可注意到各种细菌和真菌在皮肤上的过度定植,继发感染并发症的发生率为30-48%。几项研究表明,金黄色葡萄球菌在AD患者中的定植率为60-100%,而对照组健康人群的定植率为5-30%。此外,据专家称,在AD患者的皮肤培养中,耐甲氧西林金黄色葡萄球菌(MRSA)分离株的发病率高达10-30%。阿尔茨海默病并发继发感染的治疗是现代皮肤病学的关键挑战之一。莫匹罗星是AD患者感染性并发症致病因治疗中最有效的外用抗生素之一,已被大量临床研究证实。结论。金黄色葡萄球菌是最常见的阿尔茨海默病加重诱因。由于耐甲氧西林金黄色葡萄球菌对许多类型的抗生素具有耐药性,因此在AD中并发继发感染是难以控制的;然而,它对莫匹罗星表现出持久的敏感性。以莫匹罗星为基础(2%)的外用药物是儿科AD并发继发感染最有效、最安全、最优选的治疗方案。
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引用次数: 0
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Voprosy Sovremennoi Pediatrii - Current Pediatrics
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