Background: "Kaposi varicelliform eruption" (KVE), also known as "eczema herpeticum," refers to a rare widespread skin infection. The primary causal agent is thought to be the herpes simplex virus (HSV). Though common in patients with underlying skin dermatosis, systemic immunosuppression can at times lead to KVE. Case Presentation: A 27-years male, a renal transplant recipient, under systemic immunosuppressants, presented with lesions over the whole body for 2 weeks and fever for 10 days. Skin examination revealed multiple flaccid vesicles with hemorrhagic fluid over the face, trunk, and bilateral extremities. Multiple erythematous erosions over the chest and abdomen, multiple petechiae and purpura over bilateral legs, palms, soles, and abdomen, erosions over the hard palate along with thick crusts over the scalp. A tzanck smear showed plenty of acantholytic cells. With a diagnosis of KVE, he was managed with an injection of acyclovir that led to resolution of his symptoms. Conclusion: KVE is a serious condition that may have fatal outcomes. Early diagnosis and appropriate treatment of patients at risk for viral complications are very important medical considerations.
{"title":"Kaposi Varicelliform Eruption in a Chronic Kidney Disease Individual Under Tacrolimus: A Case Report.","authors":"Seema Sitaula, Suraj Shrestha, Elisha Poddar, Rabin Gosain","doi":"10.1155/2024/8373606","DOIUrl":"https://doi.org/10.1155/2024/8373606","url":null,"abstract":"<p><p><b>Background:</b> \"Kaposi varicelliform eruption\" (KVE), also known as \"eczema herpeticum,\" refers to a rare widespread skin infection. The primary causal agent is thought to be the herpes simplex virus (HSV). Though common in patients with underlying skin dermatosis, systemic immunosuppression can at times lead to KVE. <b>Case Presentation:</b> A 27-years male, a renal transplant recipient, under systemic immunosuppressants, presented with lesions over the whole body for 2 weeks and fever for 10 days. Skin examination revealed multiple flaccid vesicles with hemorrhagic fluid over the face, trunk, and bilateral extremities. Multiple erythematous erosions over the chest and abdomen, multiple petechiae and purpura over bilateral legs, palms, soles, and abdomen, erosions over the hard palate along with thick crusts over the scalp. A tzanck smear showed plenty of acantholytic cells. With a diagnosis of KVE, he was managed with an injection of acyclovir that led to resolution of his symptoms. <b>Conclusion:</b> KVE is a serious condition that may have fatal outcomes. Early diagnosis and appropriate treatment of patients at risk for viral complications are very important medical considerations.</p>","PeriodicalId":9630,"journal":{"name":"Case Reports in Dermatological Medicine","volume":"2024 ","pages":"8373606"},"PeriodicalIF":0.0,"publicationDate":"2024-10-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11540881/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142603360","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}
Scurvy, once prevalent among sailors, is now rare due to improved access to fresh fruits and vegetables yet persists in individuals with poor dietary habits. We report a case of a 35-year-old male presenting with month-long, nonpainful, nonitchy lower extremity lesions. A dermatological examination revealed follicular hyperkeratosis, perifollicular bleeding, corkscrew hairs, bleeding gums, and hemorrhagic purpura. Despite unavailable plasma vitamin C testing, scurvy was diagnosed based on clinical features and dietary history. Treatment with vitamin C resulted in significant improvement within 10 days. Inadequate intake of fruits and vegetables causes vitamin C deficiency, impairing collagen synthesis and leading to typical scurvy symptoms. This case underscores the importance of considering scurvy in patients with compatible symptoms, focusing on clinical diagnosis and treatment response when testing is unavailable. Management includes vitamin C supplementation and dietary changes, emphasizing healthcare providers' role in promoting sufficient fruit and vegetable consumptions to prevent nutritional deficiencies.
坏血病曾一度在水手中盛行,由于新鲜水果和蔬菜的供应量增加,现在已经很少见了,但在饮食习惯不良的人中却依然存在。我们报告了一例 35 岁男性的病例,他的下肢皮损长达一个月,不痛不痒。皮肤病检查发现毛囊角化过度、毛囊周围出血、开瓶器状毛发、牙龈出血和出血性紫癜。尽管无法进行血浆维生素 C 检测,但根据临床特征和饮食史诊断出了坏血病。使用维生素 C 治疗后,病情在 10 天内明显好转。水果和蔬菜摄入不足会导致维生素 C 缺乏,影响胶原蛋白的合成,从而引起典型的坏血病症状。本病例强调了对有类似症状的患者考虑坏血病的重要性,在无法进行检测的情况下,应重点关注临床诊断和治疗反应。治疗方法包括补充维生素 C 和改变饮食结构,强调医疗保健提供者在促进摄入足够的水果和蔬菜以防止营养缺乏方面的作用。
{"title":"Clinical Diagnosis and Treatment of Scurvy in a Young Adult With Poor Dietary Habits: A Case Report and Literature Review.","authors":"Lara Saeid, Moatasem Hussein Al-Janabi, Fouz Hassan","doi":"10.1155/2024/2779253","DOIUrl":"10.1155/2024/2779253","url":null,"abstract":"<p><p>Scurvy, once prevalent among sailors, is now rare due to improved access to fresh fruits and vegetables yet persists in individuals with poor dietary habits. We report a case of a 35-year-old male presenting with month-long, nonpainful, nonitchy lower extremity lesions. A dermatological examination revealed follicular hyperkeratosis, perifollicular bleeding, corkscrew hairs, bleeding gums, and hemorrhagic purpura. Despite unavailable plasma vitamin C testing, scurvy was diagnosed based on clinical features and dietary history. Treatment with vitamin C resulted in significant improvement within 10 days. Inadequate intake of fruits and vegetables causes vitamin C deficiency, impairing collagen synthesis and leading to typical scurvy symptoms. This case underscores the importance of considering scurvy in patients with compatible symptoms, focusing on clinical diagnosis and treatment response when testing is unavailable. Management includes vitamin C supplementation and dietary changes, emphasizing healthcare providers' role in promoting sufficient fruit and vegetable consumptions to prevent nutritional deficiencies.</p>","PeriodicalId":9630,"journal":{"name":"Case Reports in Dermatological Medicine","volume":"2024 ","pages":"2779253"},"PeriodicalIF":0.0,"publicationDate":"2024-10-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11530288/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142567398","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}
Pub Date : 2024-09-16eCollection Date: 2024-01-01DOI: 10.1155/2024/9775613
Giuseppe Lodi, Giovanni Cannarozzo, Irene Fusco, Tiziano Zingoni, Elena Campione, Mario Sannino
Background: Facial angiofibromas (FAs) are a dermatological characteristic which are typically linked to tuberous sclerosis (TS).
Aim: We discuss our experience, highlighting a rare occurrence of multiple FAs in a young patient, successfully treated with ablative CO2 laser combined with PDL therapy.
Methods: A 23-year-old male patient affected by TS who presents multiple erythematous and colored papules/nodules located on the face, mainly concentrated around nose, perinasal area, cheeks, and chin area, was treated with a combination of ablative CO2 laser and a pulsed dye laser. The patient underwent 3 sessions of combined treatment with CO2 and pulsed dye laser. The intralesional dye laser treatment was administered immediately after the CO2 laser session. The time interval between the combined laser treatments was approximately two months.
Results: After 4 months from the last laser treatment session, most of the facial erythematous and protruding lesions had improved. Following these procedures, the patient did not experience complications or severe adverse reaction.
Conclusion: The combined use of the CO2 and dye laser has been proved to be a safe and effective treatment for multiple FAs in the young patient affected by tuberous sclerosis.
{"title":"Effective Technique Using Combined CO<sub>2</sub> Laser and Pulsed Dye Laser for Facial Angiofibromas Management in Tuberous Sclerosis.","authors":"Giuseppe Lodi, Giovanni Cannarozzo, Irene Fusco, Tiziano Zingoni, Elena Campione, Mario Sannino","doi":"10.1155/2024/9775613","DOIUrl":"10.1155/2024/9775613","url":null,"abstract":"<p><strong>Background: </strong>Facial angiofibromas (FAs) are a dermatological characteristic which are typically linked to tuberous sclerosis (TS).</p><p><strong>Aim: </strong>We discuss our experience, highlighting a rare occurrence of multiple FAs in a young patient, successfully treated with ablative CO<sub>2</sub> laser combined with PDL therapy.</p><p><strong>Methods: </strong>A 23-year-old male patient affected by TS who presents multiple erythematous and colored papules/nodules located on the face, mainly concentrated around nose, perinasal area, cheeks, and chin area, was treated with a combination of ablative CO<sub>2</sub> laser and a pulsed dye laser. The patient underwent 3 sessions of combined treatment with CO<sub>2</sub> and pulsed dye laser. The intralesional dye laser treatment was administered immediately after the CO<sub>2</sub> laser session. The time interval between the combined laser treatments was approximately two months.</p><p><strong>Results: </strong>After 4 months from the last laser treatment session, most of the facial erythematous and protruding lesions had improved. Following these procedures, the patient did not experience complications or severe adverse reaction.</p><p><strong>Conclusion: </strong>The combined use of the CO<sub>2</sub> and dye laser has been proved to be a safe and effective treatment for multiple FAs in the young patient affected by tuberous sclerosis.</p>","PeriodicalId":9630,"journal":{"name":"Case Reports in Dermatological Medicine","volume":"2024 ","pages":"9775613"},"PeriodicalIF":0.0,"publicationDate":"2024-09-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11419839/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142307166","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}
Pub Date : 2024-09-11eCollection Date: 2024-01-01DOI: 10.1155/2024/1057957
Maiko Yamaura, Yuriko Iwahashi, Eri Hashimoto, Jun Miura, Yuri Murayama, Sachiko Koshikawa, Naoko Inomata
Carmine is an aluminium and/or calcium-chelated complex form of carminic acid (CA), which is derived from the Dactylopius coccus extract (cochineal), and is globally used as a red-colourant in foods and cosmetics. Although several allergens in carmine allergies, such as CC38K, have been reported, it remains unknown whether chelation affects the allergenicity of carmine. We report a case of Japanese fish sausage (Gyoniku Sausage) anaphylaxis induced by epicutaneous sensitization to carmine contained in eyeshadows. In addition, we report on the effect of chelation on carmine allergy. A 32-year-old woman had experienced itching, wheals, and swelling of her eyelids immediately after applying pink eyeshadows, which contained carmine, on several occasions for 3 years. Two months ago, she developed itching, wheals, and swelling on her whole body, especially her eyelids, and dyspnea immediately after ingesting fried pink fish sausages, which contained cochineal. In skin prick tests (SPTs) with all ingredients ingested in the two episodes of anaphylaxis, only fish sausage was positive. SPT was also positive for carmine. In IgE-immunoblotting using the eyeshadow and fish sausage, the patient serum IgE was bound to three protein bands at approximately 86, 114, and 130 kDa. In addition, IgE-immunoblotting using the carmine showed a broad band at 86-130 kDa, which were consistent with those using the eyeshadow and fish sausage, whereas there is no band using CA. Interestingly, the protein bands using the eyeshadow and carmine were diminished by preincubation in the presence of ethylenediaminetetraacetic acid (EDTA) as a chelating agent. The results indicated that the causative allergens of carmine contained in the eyeshadows could be dechelated by EDTA, reducing its allergenicity. In conclusion, carmine contained in cosmetics can cause epicutaneous sensitisation and consequently can induce food anaphylaxis. To prevent sensitisation in carmine allergy, the effect of chelation on allergenicity of carmine should be considered.
{"title":"A Case of Fish Sausage Anaphylaxis Induced by Epicutaneous Sensitization to Carmine Contained in Eyeshadows: The Effect of Chelation on Carmine Allergy.","authors":"Maiko Yamaura, Yuriko Iwahashi, Eri Hashimoto, Jun Miura, Yuri Murayama, Sachiko Koshikawa, Naoko Inomata","doi":"10.1155/2024/1057957","DOIUrl":"https://doi.org/10.1155/2024/1057957","url":null,"abstract":"<p><p>Carmine is an aluminium and/or calcium-chelated complex form of carminic acid (CA), which is derived from the <i>Dactylopius coccus</i> extract (cochineal), and is globally used as a red-colourant in foods and cosmetics. Although several allergens in carmine allergies, such as CC38K, have been reported, it remains unknown whether chelation affects the allergenicity of carmine. We report a case of Japanese fish sausage (<i>Gyoniku Sausage</i>) anaphylaxis induced by epicutaneous sensitization to carmine contained in eyeshadows. In addition, we report on the effect of chelation on carmine allergy. A 32-year-old woman had experienced itching, wheals, and swelling of her eyelids immediately after applying pink eyeshadows, which contained carmine, on several occasions for 3 years. Two months ago, she developed itching, wheals, and swelling on her whole body, especially her eyelids, and dyspnea immediately after ingesting fried pink fish sausages, which contained cochineal. In skin prick tests (SPTs) with all ingredients ingested in the two episodes of anaphylaxis, only fish sausage was positive. SPT was also positive for carmine. In IgE-immunoblotting using the eyeshadow and fish sausage, the patient serum IgE was bound to three protein bands at approximately 86, 114, and 130 kDa. In addition, IgE-immunoblotting using the carmine showed a broad band at 86-130 kDa, which were consistent with those using the eyeshadow and fish sausage, whereas there is no band using CA. Interestingly, the protein bands using the eyeshadow and carmine were diminished by preincubation in the presence of ethylenediaminetetraacetic acid (EDTA) as a chelating agent. The results indicated that the causative allergens of carmine contained in the eyeshadows could be dechelated by EDTA, reducing its allergenicity. In conclusion, carmine contained in cosmetics can cause epicutaneous sensitisation and consequently can induce food anaphylaxis. To prevent sensitisation in carmine allergy, the effect of chelation on allergenicity of carmine should be considered.</p>","PeriodicalId":9630,"journal":{"name":"Case Reports in Dermatological Medicine","volume":"2024 ","pages":"1057957"},"PeriodicalIF":0.0,"publicationDate":"2024-09-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11410408/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142280607","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}
Pub Date : 2024-09-06eCollection Date: 2024-01-01DOI: 10.1155/2024/1018971
Chengbei Bao, Zequn Tong, Qiuyun Xu, Zhixun Xiao, Bo Cheng, Ting Gong, Chao Ji
This study reported a woman with drug reaction with eosinophilia and systemic symptom (DRESS) syndrome induced by phloroglucinol who developed fulminant type 1 diabetes as sequelae. The literature review emphasized the necessity of at least seven months of follow-up for better management of DRESS syndrome.
{"title":"Phloroglucinol-Induced Drug Reaction with Eosinophilia and Systemic Symptoms (DRESS) Syndrome with Subsequent Fulminant Type 1 Diabetes (FT1D): A Rare Case and Literature Review.","authors":"Chengbei Bao, Zequn Tong, Qiuyun Xu, Zhixun Xiao, Bo Cheng, Ting Gong, Chao Ji","doi":"10.1155/2024/1018971","DOIUrl":"https://doi.org/10.1155/2024/1018971","url":null,"abstract":"<p><p>This study reported a woman with drug reaction with eosinophilia and systemic symptom (DRESS) syndrome induced by phloroglucinol who developed fulminant type 1 diabetes as sequelae. The literature review emphasized the necessity of at least seven months of follow-up for better management of DRESS syndrome.</p>","PeriodicalId":9630,"journal":{"name":"Case Reports in Dermatological Medicine","volume":"2024 ","pages":"1018971"},"PeriodicalIF":0.0,"publicationDate":"2024-09-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11398957/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142280608","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}
Pub Date : 2024-08-31eCollection Date: 2024-01-01DOI: 10.1155/2024/8879884
Ashley Wittmer, Katherine De Jong, Lauren Bolish, Lindsey Finklea
Nail changes are seen in some individuals with alopecia areata, with the most common variants including pitting and trachyonychia. The nail findings are presumed to be due to the same lymphocytic infiltration seen in hair bulbs in individuals with AA. Baricitinib is an immunomodulatory drug that acts as a selective and reversible inhibitor of JAK proteins and is indicated for adult patients with moderate to severe rheumatoid arthritis who have not responded to other disease-modifying antirheumatic drugs. The FDA has also approved baricitinib to treat patients hospitalized with COVID-19 and severe alopecia areata. In this report, we present a case of a patient with persistent AA-associated nail changes who has been successfully treated with baricitinib. The patient has been suffering from alopecia for several years. She presented with periungual inflammation in conjunction with persistent fingernail ridges and pitting of her right fourth digit. The nail dystrophy persisted despite treatment with tacrolimus ointment, clobetasol ointment, or oral fluconazole. Patient was started on a trial of baricitinib for alopecia areata, which was the suspected cause of the nail changes. After 4 months of treatment with baricitinib, the patient's nail showed mild improvement of nail dystrophy with some clubbing and pitting still present. Within 11 months of treatment, her nail was normalized in appearance and texture. There are no established guidelines to treat AA-associated nail changes. Our patient's AA-associated nail changes were normalized after 11 months of treatment with baricitinib. Further research is needed to determine which alopecia areata patients may benefit from treatment with baricitinib and when treatment should be initiated. Baricitinib may be an effective treatment option for AA-associated nail changes in some patients.
有些脱发症患者的指甲会发生变化,最常见的变异包括点状脱发和甲沟炎。据推测,指甲病变的原因与 AA 患者毛球中的淋巴细胞浸润相同。巴利替尼是一种免疫调节药物,是一种选择性和可逆性的JAK蛋白抑制剂,适用于对其他改善病情抗风湿药物无效的中重度类风湿关节炎成年患者。FDA 还批准巴利昔尼用于治疗 COVID-19 和重度斑秃住院患者。在本报告中,我们介绍了一例用巴利替尼成功治疗了持续性 AA 相关指甲变化的患者。该患者已被脱发困扰数年。她出现了甲周炎症,并伴有顽固性指(趾)甲脊和右侧第四个指(趾)甲凹陷。尽管使用他克莫司软膏、氯倍他索软膏或口服氟康唑进行治疗,但指甲营养不良症状依然存在。患者开始试用巴利昔尼治疗斑秃,因为怀疑斑秃是指甲变化的原因。使用巴利昔尼治疗 4 个月后,患者的指甲营养不良症状有了轻微改善,但仍存在一些畸形和凹陷。治疗 11 个月后,她的指甲外观和质地恢复正常。目前还没有治疗 AA 相关指甲变化的既定指南。我们的患者在接受巴利替尼治疗11个月后,AA相关的指甲变化已恢复正常。要确定哪些斑秃患者可从巴利替尼治疗中获益以及何时开始治疗,还需要进一步研究。巴利昔尼可能是治疗某些患者AA相关指甲变化的有效方法。
{"title":"Therapeutic Response of Alopecia Areata-Associated Nail Changes to Baricitinib.","authors":"Ashley Wittmer, Katherine De Jong, Lauren Bolish, Lindsey Finklea","doi":"10.1155/2024/8879884","DOIUrl":"10.1155/2024/8879884","url":null,"abstract":"<p><p>Nail changes are seen in some individuals with alopecia areata, with the most common variants including pitting and trachyonychia. The nail findings are presumed to be due to the same lymphocytic infiltration seen in hair bulbs in individuals with AA. Baricitinib is an immunomodulatory drug that acts as a selective and reversible inhibitor of JAK proteins and is indicated for adult patients with moderate to severe rheumatoid arthritis who have not responded to other disease-modifying antirheumatic drugs. The FDA has also approved baricitinib to treat patients hospitalized with COVID-19 and severe alopecia areata. In this report, we present a case of a patient with persistent AA-associated nail changes who has been successfully treated with baricitinib. The patient has been suffering from alopecia for several years. She presented with periungual inflammation in conjunction with persistent fingernail ridges and pitting of her right fourth digit. The nail dystrophy persisted despite treatment with tacrolimus ointment, clobetasol ointment, or oral fluconazole. Patient was started on a trial of baricitinib for alopecia areata, which was the suspected cause of the nail changes. After 4 months of treatment with baricitinib, the patient's nail showed mild improvement of nail dystrophy with some clubbing and pitting still present. Within 11 months of treatment, her nail was normalized in appearance and texture. There are no established guidelines to treat AA-associated nail changes. Our patient's AA-associated nail changes were normalized after 11 months of treatment with baricitinib. Further research is needed to determine which alopecia areata patients may benefit from treatment with baricitinib and when treatment should be initiated. Baricitinib may be an effective treatment option for AA-associated nail changes in some patients.</p>","PeriodicalId":9630,"journal":{"name":"Case Reports in Dermatological Medicine","volume":"2024 ","pages":"8879884"},"PeriodicalIF":0.0,"publicationDate":"2024-08-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11380711/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142153193","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}
Pub Date : 2024-07-31eCollection Date: 2024-01-01DOI: 10.1155/2024/9845501
Seda Altiner, Alper Ekinci
Periodic fever, aphthous stomatitis, pharyngitis, and cervical adenitis syndrome (PFAPA) is an autoinflammatory disease that is thought to occur with the contribution of genetic and environmental factors, but its etiology has not been clearly elucidated. It is characterized by recurrent attacks with fever, pharyngitis, oral aphthous lesions, and cervical lymphadenopathy, and an increase in the level of serum acute phase reactants is observed during the attacks. Although PFAPA usually begins in childhood, adult-onset cases are also reported in the literature. In the pathogenesis of PFAPA, an increase in the expression of various inflammatory cytokines, especially interleukin-1β (IL-1β), is observed as a result of the increase in inflammasome activity. Selective IgA deficiency (SIgAD) is the most prevalent primary immunodeficiency. Although most SIgAD cases remain asymptomatic and remain undiagnosed, it is known that the risk of mucosal infection is generally increased in SIgAD cases. In addition, the frequency of autoinflammatory diseases is increased in SIgAD cases compared with the general population. We aim to present a case of adult-onset PFAPA and SIgAD coexistence.
{"title":"Adult-Onset Periodic Fever, Aphthous Stomatitis, Pharyngitis, and Cervical Adenitis Syndrome on the Basis of Selective IgA Deficiency.","authors":"Seda Altiner, Alper Ekinci","doi":"10.1155/2024/9845501","DOIUrl":"10.1155/2024/9845501","url":null,"abstract":"<p><p>Periodic fever, aphthous stomatitis, pharyngitis, and cervical adenitis syndrome (PFAPA) is an autoinflammatory disease that is thought to occur with the contribution of genetic and environmental factors, but its etiology has not been clearly elucidated. It is characterized by recurrent attacks with fever, pharyngitis, oral aphthous lesions, and cervical lymphadenopathy, and an increase in the level of serum acute phase reactants is observed during the attacks. Although PFAPA usually begins in childhood, adult-onset cases are also reported in the literature. In the pathogenesis of PFAPA, an increase in the expression of various inflammatory cytokines, especially interleukin-1<i>β</i> (IL-1<i>β</i>), is observed as a result of the increase in inflammasome activity. Selective IgA deficiency (SIgAD) is the most prevalent primary immunodeficiency. Although most SIgAD cases remain asymptomatic and remain undiagnosed, it is known that the risk of mucosal infection is generally increased in SIgAD cases. In addition, the frequency of autoinflammatory diseases is increased in SIgAD cases compared with the general population. We aim to present a case of adult-onset PFAPA and SIgAD coexistence.</p>","PeriodicalId":9630,"journal":{"name":"Case Reports in Dermatological Medicine","volume":"2024 ","pages":"9845501"},"PeriodicalIF":0.0,"publicationDate":"2024-07-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11306723/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141906046","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}
Pub Date : 2024-06-19eCollection Date: 2024-01-01DOI: 10.1155/2024/6265608
Niccolò Gori, Eleonora De Luca, Andrea Chiricozzi, Stefania Sfregola, Alessandro Di Stefani, Ketty Peris
Acquired reactive perforating collagenosis is a rare cutaneous disorder characterised by the extrusion of abnormal connective tissue trough epidermidis and/or follicular units. Reactive perforating collagenosis is often associated with systemic diseases in which pruritus is a common symptom (e.g., diabetes and chronic kidney disease). Less commonly, it has been associated with chronic inflammatory dermatoses, including atopic dermatitis, as in this case. In this report, we describe the exceptional case of a 35-year-old man affected by acquired reactive perforating collagenosis associated with atopic dermatitis who was resistant to conventional topical and systemic treatment and experienced complete resolution of clinical signs and symptoms after 12 weeks of treatment with dupilumab. In our patient, the severe pruritus induced by atopic dermatitis likely contributed to the development of acquired perforating collagenosis lesions, which are thought to be a reactive response to chronic scratching and repetitive injury to the skin. Chronic pruritus in atopic dermatitis is known to be driven by type 2 cytokines, including IL-4 and IL-13, and dupilumab, a monoclonal antibody inhibiting IL-4 and IL-13 signalling, has been shown to be effective in the treatment of moderate to severe atopic dermatitis as well as other type 2-driven pruritic dermatological conditions. This case supports the potential use of dupilumab for the treatment of reactive perforating dermatosis.
{"title":"Successful Use of Dupilumab in the Treatment of Acquired Perforating Dermatosis Associated with Atopic Dermatitis.","authors":"Niccolò Gori, Eleonora De Luca, Andrea Chiricozzi, Stefania Sfregola, Alessandro Di Stefani, Ketty Peris","doi":"10.1155/2024/6265608","DOIUrl":"https://doi.org/10.1155/2024/6265608","url":null,"abstract":"<p><p>Acquired reactive perforating collagenosis is a rare cutaneous disorder characterised by the extrusion of abnormal connective tissue trough epidermidis and/or follicular units. Reactive perforating collagenosis is often associated with systemic diseases in which pruritus is a common symptom (e.g., diabetes and chronic kidney disease). Less commonly, it has been associated with chronic inflammatory dermatoses, including atopic dermatitis, as in this case. In this report, we describe the exceptional case of a 35-year-old man affected by acquired reactive perforating collagenosis associated with atopic dermatitis who was resistant to conventional topical and systemic treatment and experienced complete resolution of clinical signs and symptoms after 12 weeks of treatment with dupilumab. In our patient, the severe pruritus induced by atopic dermatitis likely contributed to the development of acquired perforating collagenosis lesions, which are thought to be a reactive response to chronic scratching and repetitive injury to the skin. Chronic pruritus in atopic dermatitis is known to be driven by type 2 cytokines, including IL-4 and IL-13, and dupilumab, a monoclonal antibody inhibiting IL-4 and IL-13 signalling, has been shown to be effective in the treatment of moderate to severe atopic dermatitis as well as other type 2-driven pruritic dermatological conditions. This case supports the potential use of dupilumab for the treatment of reactive perforating dermatosis.</p>","PeriodicalId":9630,"journal":{"name":"Case Reports in Dermatological Medicine","volume":"2024 ","pages":"6265608"},"PeriodicalIF":0.0,"publicationDate":"2024-06-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11208807/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141466412","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}
Granuloma annulare is a poorly understood dermatosis that, when generalized, can occur in up to 15 percent of patients. In these cases, treatment is frustrating and experimental. We report a case of a 60-year-old woman and a 41-year-old woman who demonstrated resolution of recalcitrant, generalized granuloma annulare (GA) following oral treatment with upadacitinib. After showing little to no response to other various treatments, such as steroids, antibiotic regimens, and systemic therapies, each patient was started on 15 mg of daily upadacitinib. At 2 months, one patient had complete clearance of all lesions while the other patient experienced noticeable improvement. Within 4 months, the other patient reached total resolution of her lesions. These cases provide evidence of a therapeutic option that may shorten disease duration and provide relief from cutaneous disease.
{"title":"Successful Treatment of Refractory Generalized Granuloma Annulare with Upadacitinib.","authors":"Alexis Coican, Abigail Meckley, Nathan Sagasser, Melinda Greenfield, Eingun James Song, Jessica El-Bahri","doi":"10.1155/2024/8859178","DOIUrl":"https://doi.org/10.1155/2024/8859178","url":null,"abstract":"<p><p>Granuloma annulare is a poorly understood dermatosis that, when generalized, can occur in up to 15 percent of patients. In these cases, treatment is frustrating and experimental. We report a case of a 60-year-old woman and a 41-year-old woman who demonstrated resolution of recalcitrant, generalized granuloma annulare (GA) following oral treatment with upadacitinib. After showing little to no response to other various treatments, such as steroids, antibiotic regimens, and systemic therapies, each patient was started on 15 mg of daily upadacitinib. At 2 months, one patient had complete clearance of all lesions while the other patient experienced noticeable improvement. Within 4 months, the other patient reached total resolution of her lesions. These cases provide evidence of a therapeutic option that may shorten disease duration and provide relief from cutaneous disease.</p>","PeriodicalId":9630,"journal":{"name":"Case Reports in Dermatological Medicine","volume":"2024 ","pages":"8859178"},"PeriodicalIF":0.0,"publicationDate":"2024-06-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11208790/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141466411","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}
Pub Date : 2024-05-29eCollection Date: 2024-01-01DOI: 10.1155/2024/6505274
R Afiouni, G Abadjian, F Stephan
Reactive angioendotheliomatosis (RAE) is a rare benign skin condition characterized histologically by the proliferation of dermal vessels and endothelial cells that occurs secondary to an underlying disease such as infections or lymphoproliferative disorders. To our knowledge, no previous cases of RAE associated with ulcerative colitis (UC) were reported in the literature. Therefore, we report the case of a 46-year-old man with a history of UC presenting with RAE confirmed on histopathology and immunostaining.
反应性血管内皮细胞瘤病(RAE)是一种罕见的良性皮肤病,其组织学特征是真皮血管和内皮细胞增生,继发于感染或淋巴增生性疾病等潜在疾病。据我们所知,文献中从未报道过 RAE 与溃疡性结肠炎(UC)相关的病例。因此,我们报告了一例经组织病理学和免疫染色证实伴有 RAE 的 46 岁男性 UC 病例。
{"title":"Reactive Angioendotheliomatosis in Association with Ulcerative Colitis.","authors":"R Afiouni, G Abadjian, F Stephan","doi":"10.1155/2024/6505274","DOIUrl":"10.1155/2024/6505274","url":null,"abstract":"<p><p>Reactive angioendotheliomatosis (RAE) is a rare benign skin condition characterized histologically by the proliferation of dermal vessels and endothelial cells that occurs secondary to an underlying disease such as infections or lymphoproliferative disorders. To our knowledge, no previous cases of RAE associated with ulcerative colitis (UC) were reported in the literature. Therefore, we report the case of a 46-year-old man with a history of UC presenting with RAE confirmed on histopathology and immunostaining.</p>","PeriodicalId":9630,"journal":{"name":"Case Reports in Dermatological Medicine","volume":"2024 ","pages":"6505274"},"PeriodicalIF":0.0,"publicationDate":"2024-05-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11152761/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141261240","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}