L. Spelman, A. E. Potter, C. Baker, S. Shumack, R. Sinclair, D. Christie, B. Wong, P. Foley, S. Hacker, C. C. Allison, the National Dermatology Radiation Oncology Registry (NDROR) investigators and sites
We read with interest the letter to the editor by Daly et al.1 in response to the 24-month National Dermatology Radiation Oncology Registry (NDROR; ACTRN12618000627257) publication,2 which reiterates the important considerations relating to the use of widefield radiation therapy (RT), such as Volumetric Modulated Arc Therapy (VMAT) to treat extensive skin field cancerisation (SFC) in patients with current or previous invasive in-field keratinocyte cancer (KC). The NDROR is a multidisciplinary collaboration between dermatologists, radiation oncologists, nurses, and other skin cancer specialists with the aim of collecting efficacy, cosmesis, toxicity, and QoL data for skin cancer patients receiving widefield RT. It is the largest prospective cohort of its kind, necessitating publication of analyses as available.
It is important to note that extensive SFC can produce KCs causing significant morbidity and mortality, whilst field-directed therapies have poor durability—particularly for severe disease.3 A history of KCs, as well as actinic keratoses number, thickness, and prior treatment failure, all correlate with risk of new disease.4-7 Extensive SFC should therefore be treated appropriately in line with the patient's disease presentation and treatment history. With the increasing KC incidence and the advent of new treatments, a multidisciplinary approach is required now more so than ever before. The range of specialists involved in this study, including patient assessment, is a testament to this new paradigm.
With respect to patient selection, 82% had received prior, sometimes multiple, interventions before consideration of widefield RT. Furthermore, >70% of patients had at least one co-morbidity, including surgical cautions. This confluence of factors was considered when determining appropriateness for widefield RT. We also agree that the concerns of widefield RT risks should not be diminished, which is why toxicity assessment has been a major focus of our data collections. Although longer follow-up is required, regular reporting is essential in the absence of any other contributions to the literature. This too is relevant for durability of clinical response. While we agree that the registry design precludes head-to-head comparisons with standard of care, the 10% new lesion rate reported in our study must be considered in the context of disease severity. Patients who fail prior treatment, and/or those with a history of multiple KCs have very high new lesion rates of 35%–90% within 2–4 years of treatment.4-7
Daly et al. rightly assert that ‘selected patients with a high burden of invasive and in situ disease who are exhausting efforts to treat invasive lesions may benefit from widefield RT, but the uncertain outcome, and the long-term effects, including the potential to undermine treatment of subsequent c
{"title":"A reply to ‘Efficacy and safety of widefield radiation therapy for extensive skin field cancerization remains unproven’","authors":"L. Spelman, A. E. Potter, C. Baker, S. Shumack, R. Sinclair, D. Christie, B. Wong, P. Foley, S. Hacker, C. C. Allison, the National Dermatology Radiation Oncology Registry (NDROR) investigators and sites","doi":"10.1002/jvc2.397","DOIUrl":"10.1002/jvc2.397","url":null,"abstract":"<p>We read with interest the letter to the editor by Daly et al.<span><sup>1</sup></span> in response to the 24-month National Dermatology Radiation Oncology Registry (NDROR; ACTRN12618000627257) publication,<span><sup>2</sup></span> which reiterates the important considerations relating to the use of widefield radiation therapy (RT), such as Volumetric Modulated Arc Therapy (VMAT) to treat extensive skin field cancerisation (SFC) in patients with current or previous invasive in-field keratinocyte cancer (KC). The NDROR is a multidisciplinary collaboration between dermatologists, radiation oncologists, nurses, and other skin cancer specialists with the aim of collecting efficacy, cosmesis, toxicity, and QoL data for skin cancer patients receiving widefield RT. It is the largest prospective cohort of its kind, necessitating publication of analyses as available.</p><p>It is important to note that extensive SFC can produce KCs causing significant morbidity and mortality, whilst field-directed therapies have poor durability—particularly for severe disease.<span><sup>3</sup></span> A history of KCs, as well as actinic keratoses number, thickness, and prior treatment failure, all correlate with risk of new disease.<span><sup>4-7</sup></span> Extensive SFC should therefore be treated appropriately in line with the patient's disease presentation and treatment history. With the increasing KC incidence and the advent of new treatments, a multidisciplinary approach is required now more so than ever before. The range of specialists involved in this study, including patient assessment, is a testament to this new paradigm.</p><p>With respect to patient selection, 82% had received prior, sometimes multiple, interventions before consideration of widefield RT. Furthermore, >70% of patients had at least one co-morbidity, including surgical cautions. This confluence of factors was considered when determining appropriateness for widefield RT. We also agree that the concerns of widefield RT risks should not be diminished, which is why toxicity assessment has been a major focus of our data collections. Although longer follow-up is required, regular reporting is essential in the absence of any other contributions to the literature. This too is relevant for durability of clinical response. While we agree that the registry design precludes head-to-head comparisons with standard of care, the 10% new lesion rate reported in our study must be considered in the context of disease severity. Patients who fail prior treatment, and/or those with a history of multiple KCs have very high new lesion rates of 35%–90% within 2–4 years of treatment.<span><sup>4-7</sup></span></p><p>Daly et al. rightly assert that ‘<i>selected patients with a high burden of invasive and in situ disease who are exhausting efforts to treat invasive lesions may benefit from widefield RT, but the uncertain outcome, and the long-term effects, including the potential to undermine treatment of subsequent c","PeriodicalId":94325,"journal":{"name":"JEADV clinical practice","volume":"3 2","pages":"749-750"},"PeriodicalIF":0.0,"publicationDate":"2024-03-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/jvc2.397","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140230992","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Mireia Seguí, Pedro Rodríguez-Jiménez, Aurora Fernández-Galván, Javier Fraga, Pablo Chicharro
A 48-year-old woman presented with a 4-year history of diffuse hyperpigmented papules beginning on the cleavage and progressing over the rest of the trunk and extremities. The lesions were mild pruritic. There were no family members with similar symptoms and both she and her family were from Spain and Caucasian. Her only significant past medical history was a papillary thyroid carcinoma that required total thyroidectomy. Clinical examination revealed generalized mottled hyper and hypopigmented macules on the trunk and extremities. Diffuse hyperpigmented small papules were also noted (Figure 1a−c). On dermoscopic examination, brownish areas with globular and dotted pigmentation alternating with round hypopigmented spots were observed (Figure 2). Laboratory investigations including a blood cell count, serum electrolytes and renal and liver function testing as well as protein serum electrophoresis revealed no abnormalities. Two skin biopsies were taken from the back of the patient (Figure 3).
Amyloidosis cutis dyschromica (ACD).
Two skin biopsies from the back showed deposition of amyloid in the papillary dermis confirmed by positive Congo red staining and immunohistochemical studies revealed that the amyloid expressed cytokeratins CK 5/6. There was also a mild inflammatory infiltrate, mostly composed of lymphocytes (Figure 3a,b). Based on the clinical and histopathological findings, the patient was diagnosed with ACD and due to the absence of symptoms scheduled for periodic follow-up.
ACD is a rare form of primary cutaneous amyloidosis, first described by Morishima in 1970.1 ACD is characterized by (i) dotted, reticular hyperpigmentation with hypopigmented macules distributed over nearly all of the body, (ii) no or little itch, (iii) usual onset before puberty and (iv) focal amyloid deposition under the epidermis.1, 2 Since this first description, both familiar and sporadic cases have been reported and most of the documented cases are from Asia. We herein present a sporadic case of a woman from Spain showing a late-onset of ACD.
Primary cutaneous amyloidosis is associated with the deposition of amyloid in the skin but not in internal organs. The most common variants of primary cutaneous amyloidosis include macular and lichen amyloidosis. ACD is a rare variant of primary cutaneous amyloidosis, characterized by reticular areas of hyperpigmentation with overlying hypopigmented macules and localized keratinocyte derived amyloid deposition within the papillary dermis. ACD has been most commonly reported in South and East Asian ethnic groups, having only few cases been reported in Caucasian ethnicity.2
Although most of the reported cases are familiar, sporadic cases have also been reported. Recent studies point out that most cases of ACD result from autosomal recessive mutations in GPNMB, encoding glycoprotein nonmetastatic gene B.3, 4
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W. C. A. M. Witkam, P. P. Buckers, S. E. Dal Belo, L. M. Pardo, T. Nijsten
<p>Acne vulgaris is a prevalent disease<span><sup>1</sup></span> with a high burden.<span><sup>2</sup></span> Despite the availability of effective treatment options,<span><sup>2</sup></span> undertreatment remains an issue.<span><sup>3, 4</sup></span> A possible explanation is access to care for minorities with less insurance coverage.<span><sup>5</sup></span> This study aims to describe acne prevalence and self-reported treatment exposures in adolescents and to assess whether socioeconomic status (SES) is associated with healthcare utilisation for acne.</p><p>This cross-sectional study from the population-based prospective study ‘Generation R’<span><sup>6</sup></span> consisted of adolescents around the age of 13 years and their parents in Rotterdam, the Netherlands. Parents received questionnaires with questions on acne in their child. When they reported a positive history of acne, they were subsequently asked about prior treatment. The outcome ‘treatment exposure’ was created by classifying these answers into three ordinal categories ranging from least to most healthcare utilisation: (1) ‘No’, (2) ‘Yes, bought over the counter’ or (3) ‘Yes, physician prescribed treatment’. The associations between SES-determinants ‘household income’ and ‘maternal education’<span><sup>6</sup></span> and the outcome were explored simultaneously while adjusting for potential relevant confounders (sex, ethnicity,<span><sup>6</sup></span> perceived skin colour and physician-graded acne severity<span><sup>7</sup></span>) using complete cases in ordinal logistic regression analyses. These resulted in adjusted odds ratios (AORs) with 95% confidence intervals (CIs) displaying the log-likelihood of utilising a higher level of healthcare for the treatment of acne.</p><p>Parents of 4698 adolescents responded to the acne-related questions (response rate 75.6%) (Table 1). While 45.8% of the parents indicated their children ever had acne, just 17.6% of them had ever used treatment (only 33% in the physician-graded moderate/severe acne group). More severe acne was positively associated with care (AOR 8.69 [95% CI 5.42–14.46] for moderate/severe versus. [almost] clear acne) (Table 2). SES-determinants were not associated with more healthcare utilisation (AORs: 1.47 [95% CI 0.71–2.86] low vs. intermediate maternal education and 0.98 [95% CI 0.63–1.51] low versus middle income levels). However, sex-stratified analyses showed that only boys from a low versus middle income used less care (AOR 0.30 [0.11–0.75]). Finally, Non-European ethnicity was associated with a higher level of care (AOR 1.96 [95% CI 1.23–3.12]), but sex-stratification showed this association merely in girls (AOR 2.43 [1.34–4.44]).</p><p>Our study confirms the gap between acne prevalence and treatment<span><sup>3-5</sup></span> among young adolescents—even in the most severe acne group—and shows that healthcare utilisation for acne in a country with a social healthcare system is a result of an interplay of SES
寻常痤疮是一种负担沉重的流行病1 。2 尽管目前已有有效的治疗方案，2 但治疗不足仍是一个问题3, 4 。本研究旨在描述青少年的痤疮患病率和自我报告的治疗情况，并评估社会经济地位（SES）是否与痤疮的医疗利用率相关。这项横断面研究来自基于人口的前瞻性研究 "R世代 "6 ，由荷兰鹿特丹 13 岁左右的青少年及其父母组成。家长们收到的调查问卷中包含有关孩子痤疮的问题。当他们报告有痤疮病史时，随后会被问及之前的治疗情况。结果 "接受治疗的情况 "是通过将这些答案分为三个从最少到最多的序数类别而得出的：(1) "没有"，(2) "有，非处方药 "或(3) "有，医生处方治疗"。在调整潜在的相关混杂因素（性别、种族6 、肤色和医生评定的痤疮严重程度7 ）的同时，我们还利用完整病例在序数逻辑回归分析中探讨了社会经济地位决定因素 "家庭收入 "和 "母亲教育程度 "6 与结果之间的关系。4698名青少年的家长回答了与痤疮相关的问题（回答率为75.6%）（表1）。虽然45.8%的家长表示他们的孩子曾经长过痤疮，但只有17.6%的家长曾经使用过治疗方法（在医生分级的中度/重度痤疮组中只有33%的家长使用过治疗方法）。更严重的痤疮与护理呈正相关（中度/严重痤疮与[几乎]透明痤疮的 AOR 值为 8.69 [95% CI 5.42-14.46]）。[几乎]无痤疮）（表 2）。社会经济地位决定因素与更多的医疗利用率无关（AORs：1.47[95%CI 0.71-2.86]低等与中等母亲教育程度，0.98[95%CI 0.63-1.51]低收入与中等收入水平）。然而，性别分层分析显示，只有低收入与中等收入的男孩使用的护理较少（AOR 0.30 [0.11-0.75]）。最后，非欧洲人种与较高的护理水平相关（AOR 1.96 [95% CI 1.23-3.12]），但性别分层分析表明，这种关联仅存在于女孩中（AOR 2.43 [1.34-4.44]）。我们的研究证实了青少年中痤疮发病率与治疗之间的差距3-5，即使在痤疮最严重的群体中也是如此，并表明在一个拥有社会医疗体系的国家中，痤疮的医疗使用是社会经济地位和性别差异相互作用的结果。更具体地说，性别改变了社会经济地位与医疗利用率之间的关系。我们的研究显示，在同一社会经济地位类别中，不同性别的求医行为不同，这可能是由于对痤疮严重程度3 或美容标准的认识不同。痤疮后遗症（疤痕或炎症后色素沉着）发病率较高不太可能是根本原因，因为分析已根据肤色和痤疮严重程度进行了调整。不同种族的妇女对痤疮的不同看法和态度8、9 也可能是一个原因。不过，这项研究的对象受过较好的教育，认为参加健康研究计划很有意义。她们可能对健康更感兴趣，也更了解治疗方案，从而高估了治疗暴露。其他局限性包括：参与者年龄较小，因此研究结果可能无法推断到年龄较大的人群；研究的横断面性质阻碍了因果关系的研究。这项研究的优势在于样本量大且来自多个种族。最后，痤疮发病率和治疗之间仍存在巨大差距，需要进一步研究性别和社会文化相关差异，以提高所有人的生活质量。Willemijn Witkam 和 Paul Buckers 分析了数据并起草了手稿。S. E. Dal Belo 是欧莱雅的员工。其他作者声明无利益冲突。欧莱雅研究与创新部（无限制研究基金）。R 代研究的总体设计、所有研究目的和具体测量方法均已获得鹿特丹大学医学中心 Erasmus MC 医学伦理委员会的批准。注册号：MEC 2015-749 NL55MEC 2015-749 NL55105.078.15。本手稿中的所有患者均已书面知情同意参与本研究，并同意将其去标识化、匿名化的汇总数据及其病例详情用于发表。
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Carmen Cantisani, Federica Trovato, Luca Gargano, Antonio Di Guardo, Alexandru Bâja Vasile, Emanuele Rovaldi, Giulia Azzella, Iolanda Speranza, Giuseppe Soda, Michela Roberto, Tiziana Lettera, Giovanni Pellacani
Breast cancer represents the most common malignancy in women worldwide. In most cases, metastasis is the leading cause of mortality. Early diagnosis is still challenging despite standardised, government-led screening programs, but is crucial in achieving improved survival rates. Additionally, a deeper understanding of the metastatic potential of breast cancer is critical for developing therapeutic interventions to combat widespread disease. Cutaneous metastasis from underlying breast carcinoma is uncommon as the first manifestation of visceral malignancies and is commonly observed in advanced-stage malignancies, often associated with poor prognosis, and a prompt, precise tissue diagnosis is mandatory. A high index of suspicion in oncologic patients is required to diagnose these lesions, as they can mimic benign skin manifestations and clinical findings may be subtle and going unnoticed. We report on a case of a 76-year-old female patient presenting to our non-invasive diagnostic outpatient clinic with an unusual cutaneous presentation, as an early sign of locally advanced invasive ductal carcinoma breast cancer recurrence. The aim of our article is to underline the importance of the dermatologist in the multi-disciplinary oncologic diagnostic process, including non-invasive imaging evaluation of cutaneous lesions, especially insidious breast carcinomas. Dynamic optical coherence tomography (D-OCT) is not routinely used due to its cost, therefore lesion's aspect has not been completely described, consequently, the correct evaluation of skin architecture appearance can add important information, especially in cutaneous oncology where it can help in early diagnosis or cancer recurrency. OCT may contribute to the detection of subclinical cutaneous manifestations of cancer or recurrence, in particular, when they are difficult to differentiate clinically from benign lesion. In the case we described, OCT of suspected lesion showed the loss of the DEJ with solid nests and irregular vessels.
乳腺癌是全世界妇女最常见的恶性肿瘤。在大多数情况下，转移是导致死亡的主要原因。尽管有政府主导的标准化筛查计划，但早期诊断仍然具有挑战性，但这对提高生存率至关重要。此外，更深入地了解乳腺癌的转移潜力对于开发治疗干预措施以应对广泛的疾病至关重要。潜在乳腺癌的皮肤转移作为内脏恶性肿瘤的首发表现并不常见，常见于晚期恶性肿瘤，通常与预后不良有关，因此必须进行及时、精确的组织诊断。肿瘤患者需要高度怀疑才能诊断出这些病变，因为它们可能会模仿良性皮肤表现，而且临床表现可能不明显而不被注意。我们报告了一例 76 岁女性患者的病例，她因不寻常的皮肤表现到我们的非侵入性诊断门诊就诊，这是局部晚期浸润性导管癌乳腺癌复发的早期征兆。本文旨在强调皮肤科医生在多学科肿瘤诊断过程中的重要性，包括对皮肤病变，尤其是隐匿性乳腺癌的无创成像评估。动态光学相干断层扫描（D-OCT）因其价格昂贵而未被常规使用，因此病变方面的描述并不完整，因此，对皮肤结构外观的正确评估可以增加重要的信息，尤其是在皮肤肿瘤学中，它可以帮助早期诊断或癌症复发。OCT 可能有助于发现癌症或复发的亚临床皮肤表现，尤其是在临床上难以与良性病变区分的情况下。在我们描述的病例中，疑似病变的 OCT 显示 DEJ 缺失，并伴有实性巢和不规则血管。
{"title":"A tricky singular papular peri-cicatricial mammarian lesion","authors":"Carmen Cantisani, Federica Trovato, Luca Gargano, Antonio Di Guardo, Alexandru Bâja Vasile, Emanuele Rovaldi, Giulia Azzella, Iolanda Speranza, Giuseppe Soda, Michela Roberto, Tiziana Lettera, Giovanni Pellacani","doi":"10.1002/jvc2.341","DOIUrl":"10.1002/jvc2.341","url":null,"abstract":"<p>Breast cancer represents the most common malignancy in women worldwide. In most cases, metastasis is the leading cause of mortality. Early diagnosis is still challenging despite standardised, government-led screening programs, but is crucial in achieving improved survival rates. Additionally, a deeper understanding of the metastatic potential of breast cancer is critical for developing therapeutic interventions to combat widespread disease. Cutaneous metastasis from underlying breast carcinoma is uncommon as the first manifestation of visceral malignancies and is commonly observed in advanced-stage malignancies, often associated with poor prognosis, and a prompt, precise tissue diagnosis is mandatory. A high index of suspicion in oncologic patients is required to diagnose these lesions, as they can mimic benign skin manifestations and clinical findings may be subtle and going unnoticed. We report on a case of a 76-year-old female patient presenting to our non-invasive diagnostic outpatient clinic with an unusual cutaneous presentation, as an early sign of locally advanced invasive ductal carcinoma breast cancer recurrence. The aim of our article is to underline the importance of the dermatologist in the multi-disciplinary oncologic diagnostic process, including non-invasive imaging evaluation of cutaneous lesions, especially insidious breast carcinomas. Dynamic optical coherence tomography (D-OCT) is not routinely used due to its cost, therefore lesion's aspect has not been completely described, consequently, the correct evaluation of skin architecture appearance can add important information, especially in cutaneous oncology where it can help in early diagnosis or cancer recurrency. OCT may contribute to the detection of subclinical cutaneous manifestations of cancer or recurrence, in particular, when they are difficult to differentiate clinically from benign lesion. In the case we described, OCT of suspected lesion showed the loss of the DEJ with solid nests and irregular vessels.</p>","PeriodicalId":94325,"journal":{"name":"JEADV clinical practice","volume":"3 2","pages":"687-692"},"PeriodicalIF":0.0,"publicationDate":"2024-03-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/jvc2.341","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140237006","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Body-focused repetitive behaviours (BFRBs), such as skin picking and trichotillomania, are conditions at the interface of dermatology and psychiatry.
Objectives
We asked individuals with various BFRBs about their habits and preferences preceding the onset of their BFRB(s). We also inquired about the emotions (positive, negative or mixed) accompanying the habit to explore predisposing and maintenance factors.
Methods
A sample of 201 individuals with mixed BFRBs were recruited online. We administered the Generic BFRB Scale (GBS-36) and the newly developed Somatic and Habitual Predisposition to BFRB Scale as well as the Ambivalence Towards BFRB Rating.
Results
Most participants reported both positive and negative feelings towards engaging in BFRBs, with only a minority (41.8%) indicating predominantly negative feelings. The study speaks to somatic and habitual predisposing factors that are topographically related to specific conditions (e.g., dislike of one's skin and skin impurities preceding skin picking, dislike of one's nails and brittle nails preceding nail biting, tendency to scarring and injuries preceding lip-cheek biting).
Conclusions
Our study speaks to important somatic and habitual predisposing factors in BFRBs. Positive feelings accompanying BFRBs may act as an important maintenance factor in BFRBs. Our results may inform new therapeutic approaches to treating or preventing BFRBs.
{"title":"Risk and maintenance factors in body-focused repetitive behaviours","authors":"Steffen Moritz, Danielle Penney, Luca Hoyer, Stella Schmotz","doi":"10.1002/jvc2.405","DOIUrl":"10.1002/jvc2.405","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>Body-focused repetitive behaviours (BFRBs), such as skin picking and trichotillomania, are conditions at the interface of dermatology and psychiatry.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Objectives</h3>\u0000 \u0000 <p>We asked individuals with various BFRBs about their habits and preferences preceding the onset of their BFRB(s). We also inquired about the emotions (positive, negative or mixed) accompanying the habit to explore predisposing and maintenance factors.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>A sample of 201 individuals with mixed BFRBs were recruited online. We administered the Generic BFRB Scale (GBS-36) and the newly developed Somatic and Habitual Predisposition to BFRB Scale as well as the Ambivalence Towards BFRB Rating.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>Most participants reported both positive and negative feelings towards engaging in BFRBs, with only a minority (41.8%) indicating predominantly negative feelings. The study speaks to somatic and habitual predisposing factors that are topographically related to specific conditions (e.g., dislike of one's skin and skin impurities preceding skin picking, dislike of one's nails and brittle nails preceding nail biting, tendency to scarring and injuries preceding lip-cheek biting).</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>Our study speaks to important somatic and habitual predisposing factors in BFRBs. Positive feelings accompanying BFRBs may act as an important maintenance factor in BFRBs. Our results may inform new therapeutic approaches to treating or preventing BFRBs.</p>\u0000 </section>\u0000 </div>","PeriodicalId":94325,"journal":{"name":"JEADV clinical practice","volume":"3 4","pages":"1183-1189"},"PeriodicalIF":0.0,"publicationDate":"2024-03-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/jvc2.405","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140240992","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Cutaneous manifestations in connective tissue diseases (CTDs) are often chronic and difficult to control.
Objectives
This study aims to review the use of combination systemic medications in CTDs and assess disease response.
Methods
An IRB-approved, retrospective chart review was conducted at a single large academic institution in New Orleans, Louisiana. Inclusion criteria were adult patients with CTDs on two or more systemic therapies.
Results
Out of 101 patients, 37.62% were taking combination systemic agents. Of these patients, 44% were diagnosed with lupus, 18% with dermatomyositis (DM), 8% with systemic scleroderma (SSc), 10% with morphea/limited scleroderma (M), 12% with mixed connective tissue disease (MCTD), and 8% with rheumatoid arthritis (RA). The most common regimen for lupus, MCTD, and SSc patients was hydroxychloroquine (HCQ) + prednisone (PRED) + mycophenolate mofetil (MMF). DM patients were most commonly taking combinations of the above regimen with intravenous immunoglobulin (IVIG). Morphea and RA patients were on multiple combinations of steroid and nonsteroidal agents including MMF, IVIG, azathioprine, methotrexate, HCQ, sulfasalazine, upadacitinib, apremilast, and tofacitinib.
Conclusions
This study aims to assess the efficacy of combination systemic medications and disease response in CTDs. Lupus was the most common CTD in our study, with patients taking 2–4 combination medications. HCQ, MMF, and PRED were the most common medications used within a combination regimen with minimal adverse effects. Though our study has several limitations including its retrospective nature and small sample size, it shows that CTDs can be resistant to monotherapy and that combination therapy might be required. More research in layering systemic agents is needed since this topic is currently limited to minimal case series and clinical trials.
{"title":"The utility of layering systemic therapies in connective tissue diseases","authors":"Kezia Surjanto, Lixin Ji, Erin Boh, Carole Bitar","doi":"10.1002/jvc2.401","DOIUrl":"10.1002/jvc2.401","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>Cutaneous manifestations in connective tissue diseases (CTDs) are often chronic and difficult to control.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Objectives</h3>\u0000 \u0000 <p>This study aims to review the use of combination systemic medications in CTDs and assess disease response.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>An IRB-approved, retrospective chart review was conducted at a single large academic institution in New Orleans, Louisiana. Inclusion criteria were adult patients with CTDs on two or more systemic therapies.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>Out of 101 patients, 37.62% were taking combination systemic agents. Of these patients, 44% were diagnosed with lupus, 18% with dermatomyositis (DM), 8% with systemic scleroderma (SSc), 10% with morphea/limited scleroderma (M), 12% with mixed connective tissue disease (MCTD), and 8% with rheumatoid arthritis (RA). The most common regimen for lupus, MCTD, and SSc patients was hydroxychloroquine (HCQ) + prednisone (PRED) + mycophenolate mofetil (MMF). DM patients were most commonly taking combinations of the above regimen with intravenous immunoglobulin (IVIG). Morphea and RA patients were on multiple combinations of steroid and nonsteroidal agents including MMF, IVIG, azathioprine, methotrexate, HCQ, sulfasalazine, upadacitinib, apremilast, and tofacitinib.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>This study aims to assess the efficacy of combination systemic medications and disease response in CTDs. Lupus was the most common CTD in our study, with patients taking 2–4 combination medications. HCQ, MMF, and PRED were the most common medications used within a combination regimen with minimal adverse effects. Though our study has several limitations including its retrospective nature and small sample size, it shows that CTDs can be resistant to monotherapy and that combination therapy might be required. More research in layering systemic agents is needed since this topic is currently limited to minimal case series and clinical trials.</p>\u0000 </section>\u0000 </div>","PeriodicalId":94325,"journal":{"name":"JEADV clinical practice","volume":"3 4","pages":"1179-1182"},"PeriodicalIF":0.0,"publicationDate":"2024-03-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/jvc2.401","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140244085","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pemphigus vulgaris (PV) and pemphigus foliaceus (PF) are autoimmune blistering diseases targeting desmoglein 3 (Dsg3) or desmoglein 1 (Dsg1). The current scoring system, pemphigus disease area index (PDAI), has limitations including inter- and intra-evaluator variability, as well as a time-consuming evaluation process.
Objectives
To identify objective and quantitative surrogate markers for assessing disease severity in PV and PF.
Methods
Eleven PV and PF patients were included. PDAI scores and candidate biomarkers [anti-Dsg3 antibody, anti-Dsg1 antibody, thymus and activation-regulated chemokine (TARC)/CCL17, immunoglobulin E (IgE) levels and eosinophil counts] were measured before oral corticosteroid treatment. Pearson's correlation coefficient was used for correlations between PDAI and candidate biomarkers.
Results
Serum TARC/CCL17 levels significantly correlated with PDAI scores in PV and PF (R = 0.72, p = 0.02), while anti-Dsg3 antibody levels correlated with PDAI scores for PV (R = 0.86, p = 0.012). No significant correlations were observed for anti-Dsg1 antibody, eosinophil counts or IgE levels.
Conclusions
Serum TARC/CCL17 may be a quantitative and unbiased disease biomarker in pemphigus patients, although further studies involving larger patient cohorts are needed.
{"title":"TARC/CCL17 reflects the severity of pemphigus (pemphigus vulgaris and pemphigus foliaceus) at the initial presentation","authors":"Tomoya Takegami, Satoru Yonekura, Takashi Nomura, Gyohei Egawa, Kenji Kabashima","doi":"10.1002/jvc2.396","DOIUrl":"10.1002/jvc2.396","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>Pemphigus vulgaris (PV) and pemphigus foliaceus (PF) are autoimmune blistering diseases targeting desmoglein 3 (Dsg3) or desmoglein 1 (Dsg1). The current scoring system, pemphigus disease area index (PDAI), has limitations including inter- and intra-evaluator variability, as well as a time-consuming evaluation process.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Objectives</h3>\u0000 \u0000 <p>To identify objective and quantitative surrogate markers for assessing disease severity in PV and PF.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>Eleven PV and PF patients were included. PDAI scores and candidate biomarkers [anti-Dsg3 antibody, anti-Dsg1 antibody, thymus and activation-regulated chemokine (TARC)/CCL17, immunoglobulin E (IgE) levels and eosinophil counts] were measured before oral corticosteroid treatment. Pearson's correlation coefficient was used for correlations between PDAI and candidate biomarkers.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>Serum TARC/CCL17 levels significantly correlated with PDAI scores in PV and PF (<i>R</i> = 0.72, <i>p</i> = 0.02), while anti-Dsg3 antibody levels correlated with PDAI scores for PV (<i>R</i> = 0.86, <i>p</i> = 0.012). No significant correlations were observed for anti-Dsg1 antibody, eosinophil counts or IgE levels.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>Serum TARC/CCL17 may be a quantitative and unbiased disease biomarker in pemphigus patients, although further studies involving larger patient cohorts are needed.</p>\u0000 </section>\u0000 </div>","PeriodicalId":94325,"journal":{"name":"JEADV clinical practice","volume":"3 4","pages":"1175-1178"},"PeriodicalIF":0.0,"publicationDate":"2024-03-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/jvc2.396","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140252988","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Deepika Roshith, Christopher Rawlingson, Timothy H. Clayton, Chitra Sethuraman, Peter D. Arkwright
Neutrophilic dermatoses are autoinflammatory disorders characterised by sterile cutaneous inflammatory neutrophilic infiltrates. This group of diseases is rare in infants and children, where sepsis is the diagnosis of exclusion. This report focuses on the clinical presentation, investigation and management of infants with idiopathic pyoderma gangrenosum. It should allow for a better appreciation of the disease and a more patient-focused approach to its diagnosis and management.
{"title":"Early-onset idiopathic pyoderma gangrenosum","authors":"Deepika Roshith, Christopher Rawlingson, Timothy H. Clayton, Chitra Sethuraman, Peter D. Arkwright","doi":"10.1002/jvc2.399","DOIUrl":"10.1002/jvc2.399","url":null,"abstract":"<p>Neutrophilic dermatoses are autoinflammatory disorders characterised by sterile cutaneous inflammatory neutrophilic infiltrates. This group of diseases is rare in infants and children, where sepsis is the diagnosis of exclusion. This report focuses on the clinical presentation, investigation and management of infants with idiopathic pyoderma gangrenosum. It should allow for a better appreciation of the disease and a more patient-focused approach to its diagnosis and management.</p>","PeriodicalId":94325,"journal":{"name":"JEADV clinical practice","volume":"3 4","pages":"1244-1247"},"PeriodicalIF":0.0,"publicationDate":"2024-03-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/jvc2.399","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140255117","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Sirkku Peltonen, Jørgen Serup, Mimmi Tang, Martin Gillstedt, Despoina Kantere, Noora Neittaanmäki, Peter Holmström, Jaishri O. Blakeley, Karli Rosner, Joshua Roberts, Torsten Bove, Katrine Elisabeth Karmisholt
Background
High-intensity focused ultrasound (HIFU) is widely used in the treatment of deep tumours, but clinical trials on skin tumours are not yet available. Neurofibromatosis Type I (NF1) is among the most common single-gene inherited conditions worldwide and predisposes to benign and malignant neoplasms of the nervous system. Multiple cutaneous neurofibromas (cNFs) often cause social and functional limitations, itching and pain.
Objectives
The objective of this study was to investigate the safety, local tolerability and efficacy of a novel method utilizing HIFU for the treatment of cNFs.
Methods
A 20 MHz HIFU-device with an integrated dermoscopic guidance and a handpiece with a focus depth of 2.3 mm below the skin surface was used. Doses of acoustic energy with 0.7 J/dose and pulse duration of 250 ms/dose were manually positioned with 1–2 mm distance between each applied dose. Number of applied doses depended on the size of the cNF. No anaesthetic was applied.
Results
Twenty patients with NF1 were recruited in two centres, and 147 cNFs were treated. There were no serious adverse events. Immediate and transient wheal-and-flare reactions occurred at treatment sites and occasionally there was minor epidermal damage which healed in 1–2 weeks. Dyspigmentation occurred in some tumours after 3–9 months but no scarring was observed at 9-month follow-up. During treatment, the patient-reported pain-score median was 3.5 (range 1–7) on a 0–10-point scale. Clinical rating of cNFs after 9 months showed 48.9% full or major tumour reduction. The median reduction in tumour thickness measured by ultrasound at 9 months was 0.53 mm (range: –100% to +19%).
Conclusions
HIFU treatment is a new noninvasive, rapid and tolerable treatment modality that with high precision targets intradermal lesions. This study demonstrates acceptable safety, local tolerance and efficacy of HIFU for the treatment of cNFs that may further be developed also for other skin tumours.
{"title":"High-intensity focused ultrasound: Safety and efficacy of a novel treatment modality for neurofibromatosis type 1 cutaneous neurofibroma","authors":"Sirkku Peltonen, Jørgen Serup, Mimmi Tang, Martin Gillstedt, Despoina Kantere, Noora Neittaanmäki, Peter Holmström, Jaishri O. Blakeley, Karli Rosner, Joshua Roberts, Torsten Bove, Katrine Elisabeth Karmisholt","doi":"10.1002/jvc2.398","DOIUrl":"10.1002/jvc2.398","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>High-intensity focused ultrasound (HIFU) is widely used in the treatment of deep tumours, but clinical trials on skin tumours are not yet available. Neurofibromatosis Type I (NF1) is among the most common single-gene inherited conditions worldwide and predisposes to benign and malignant neoplasms of the nervous system. Multiple cutaneous neurofibromas (cNFs) often cause social and functional limitations, itching and pain.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Objectives</h3>\u0000 \u0000 <p>The objective of this study was to investigate the safety, local tolerability and efficacy of a novel method utilizing HIFU for the treatment of cNFs.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>A 20 MHz HIFU-device with an integrated dermoscopic guidance and a handpiece with a focus depth of 2.3 mm below the skin surface was used. Doses of acoustic energy with 0.7 J/dose and pulse duration of 250 ms/dose were manually positioned with 1–2 mm distance between each applied dose. Number of applied doses depended on the size of the cNF. No anaesthetic was applied.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>Twenty patients with NF1 were recruited in two centres, and 147 cNFs were treated. There were no serious adverse events. Immediate and transient wheal-and-flare reactions occurred at treatment sites and occasionally there was minor epidermal damage which healed in 1–2 weeks. Dyspigmentation occurred in some tumours after 3–9 months but no scarring was observed at 9-month follow-up. During treatment, the patient-reported pain-score median was 3.5 (range 1–7) on a 0–10-point scale. Clinical rating of cNFs after 9 months showed 48.9% full or major tumour reduction. The median reduction in tumour thickness measured by ultrasound at 9 months was 0.53 mm (range: –100% to +19%).</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>HIFU treatment is a new noninvasive, rapid and tolerable treatment modality that with high precision targets intradermal lesions. This study demonstrates acceptable safety, local tolerance and efficacy of HIFU for the treatment of cNFs that may further be developed also for other skin tumours.</p>\u0000 </section>\u0000 </div>","PeriodicalId":94325,"journal":{"name":"JEADV clinical practice","volume":"3 4","pages":"1049-1060"},"PeriodicalIF":0.0,"publicationDate":"2024-03-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/jvc2.398","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140254938","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Astrid Chodziuk, Nikolaj Holgersen, Valdemar W. Nielsen, Jacob P. Thyssen, Alexander Egeberg, Simon F. Thomsen
Background
Patients with hidradenitis suppurativa (HS) experience fatigue, but no studies have yet examined the association between HS severity and fatigue intensity.
Objectives
To examine the possible association between HS severity and fatigue and identify fatigue subtypes present in patients with HS.
Methods
Adult patients with HS participated in a survey assessing fatigue using the Multidimensional Fatigue Inventory-20. Disease severity was determined by self-report.
Results
Data from 654 adult patients with HS were analysed and compared with 3,788 reference individuals from the general population. Total and subtype fatigue scores for patients with HS were significantly higher compared with the general population reference individuals and increased with higher HS severity, itch, pain, sleep, dermatology life quality index scores and lower socioeconomic status. The association between HS severity and increased fatigue scores remained statistically significant after adjustment for age, sex, and socioeconomic status.
Conclusions
Patients with HS had a significantly higher fatigue score compared with the general population reference individuals in a severity-dependent manner. A higher awareness of fatigue as a symptom in patients with HS is warranted.
{"title":"Fatigue is associated with disease severity in adult patients with hidradenitis suppurativa","authors":"Astrid Chodziuk, Nikolaj Holgersen, Valdemar W. Nielsen, Jacob P. Thyssen, Alexander Egeberg, Simon F. Thomsen","doi":"10.1002/jvc2.345","DOIUrl":"10.1002/jvc2.345","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>Patients with hidradenitis suppurativa (HS) experience fatigue, but no studies have yet examined the association between HS severity and fatigue intensity.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Objectives</h3>\u0000 \u0000 <p>To examine the possible association between HS severity and fatigue and identify fatigue subtypes present in patients with HS.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>Adult patients with HS participated in a survey assessing fatigue using the Multidimensional Fatigue Inventory-20. Disease severity was determined by self-report.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>Data from 654 adult patients with HS were analysed and compared with 3,788 reference individuals from the general population. Total and subtype fatigue scores for patients with HS were significantly higher compared with the general population reference individuals and increased with higher HS severity, itch, pain, sleep, dermatology life quality index scores and lower socioeconomic status. The association between HS severity and increased fatigue scores remained statistically significant after adjustment for age, sex, and socioeconomic status.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>Patients with HS had a significantly higher fatigue score compared with the general population reference individuals in a severity-dependent manner. A higher awareness of fatigue as a symptom in patients with HS is warranted.</p>\u0000 </section>\u0000 </div>","PeriodicalId":94325,"journal":{"name":"JEADV clinical practice","volume":"3 2","pages":"769-778"},"PeriodicalIF":0.0,"publicationDate":"2024-03-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/jvc2.345","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140266377","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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