Hereditary angioedema (HAE) is a rare disease with acute attacks in the skin and mucosa throughout the body including life-threatening laryngeal edema and abdominal attacks with severe pain. Physicians, regardless of specialty, may encounter HAE patients in their daily practice; however, low disease awareness may attribute to a considerable number of undiagnosed HAE patients in Japan. This study aims to identify issues associated with the diagnosis processes of HAE and to determine levels of HAE awareness among Japanese physicians from various specialties.
Methods
A web-based quantitative survey was conducted using a physicians panel. Physicians from the following departments were included in the survey: internal medicine, dermatology, pediatrics, emergency medicine, and gastroenterological surgery.
Results
The proportions of physicians in dermatology, pediatrics, emergency medicine, internal medicine, and gastroenterological surgery who were able to select the C1-INH activity test as a diagnosis test for potential HAE patients were 71.8%, 59.7%, 57.1%, 40.3%, and 25.7%, respectively. Multivariate analysis showed significant association between physicians who selected “strongly suspected” AE based on the case-scenario and physicians who had knowledge of the essential HAE symptoms (laryngeal edema, swelling after tooth extraction, swelling of the tongue, and abdominal pain).
Conclusions
This study showed that disease awareness of HAE varied among medical specialties, suggesting the importance of educational activities in academic societies and specialist accreditation in raising HAE awareness. Proper knowledge of complement testing and HAE symptoms may help not only to diagnose patients with AE-like symptoms as AE but also to differentially diagnose HAE from AE.
{"title":"Physician awareness and understanding of hereditary angioedema: A web-based study in Japan","authors":"Atsushi Fukunaga MD, PhD, Miwa Kishimoto MD, PhD, Akinori Oh PhD, Takeshi Akiyama MBA, Ippei Kotera PhD, Yoichi Inoue MD, JD, Junichi Maehara MD","doi":"10.1002/cia2.12265","DOIUrl":"10.1002/cia2.12265","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Objectives</h3>\u0000 \u0000 <p>Hereditary angioedema (HAE) is a rare disease with acute attacks in the skin and mucosa throughout the body including life-threatening laryngeal edema and abdominal attacks with severe pain. Physicians, regardless of specialty, may encounter HAE patients in their daily practice; however, low disease awareness may attribute to a considerable number of undiagnosed HAE patients in Japan. This study aims to identify issues associated with the diagnosis processes of HAE and to determine levels of HAE awareness among Japanese physicians from various specialties.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>A web-based quantitative survey was conducted using a physicians panel. Physicians from the following departments were included in the survey: internal medicine, dermatology, pediatrics, emergency medicine, and gastroenterological surgery.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>The proportions of physicians in dermatology, pediatrics, emergency medicine, internal medicine, and gastroenterological surgery who were able to select the C1-INH activity test as a diagnosis test for potential HAE patients were 71.8%, 59.7%, 57.1%, 40.3%, and 25.7%, respectively. Multivariate analysis showed significant association between physicians who selected “strongly suspected” AE based on the case-scenario and physicians who had knowledge of the essential HAE symptoms (laryngeal edema, swelling after tooth extraction, swelling of the tongue, and abdominal pain).</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>This study showed that disease awareness of HAE varied among medical specialties, suggesting the importance of educational activities in academic societies and specialist accreditation in raising HAE awareness. Proper knowledge of complement testing and HAE symptoms may help not only to diagnose patients with AE-like symptoms as AE but also to differentially diagnose HAE from AE.</p>\u0000 </section>\u0000 </div>","PeriodicalId":15543,"journal":{"name":"Journal of Cutaneous Immunology and Allergy","volume":"5 5","pages":"158-169"},"PeriodicalIF":1.0,"publicationDate":"2022-06-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cia2.12265","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"41714951","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}
We report a case of 70-year-old woman with rectal malignant melanoma that recurred in the pelvic lymph node one year after surgery. Nivolumab was initiated and she achieved complete response after one year, but she discontinued nivolumab at her instance. At a follow-up 21 months after the discontinuation of nivolumab, a pelvic lymph node metastasis recurrence and a lung metastasis discovered. Nivolumab rechallenge was initiated, but it was not successful.
{"title":"Recurrent advanced rectal malignant melanoma that discontinued anti-PD-1 antibody after complete response and was refractory to rechallenge","authors":"Shintaro Saito MD, Masahito Yasuda MD, PhD, Takeshi Araki MD, Azusa Ida MD, Yuko Kuriyama MD, PhD, Akihito Uehara MD, PhD, Chikako Kishi MD, PhD, Yukie Endo MD, PhD, Hiroomi Ogawa MD, PhD, Sei-ichiro Motegi MD, PhD","doi":"10.1002/cia2.12264","DOIUrl":"10.1002/cia2.12264","url":null,"abstract":"<p>We report a case of 70-year-old woman with rectal malignant melanoma that recurred in the pelvic lymph node one year after surgery. Nivolumab was initiated and she achieved complete response after one year, but she discontinued nivolumab at her instance. At a follow-up 21 months after the discontinuation of nivolumab, a pelvic lymph node metastasis recurrence and a lung metastasis discovered. Nivolumab rechallenge was initiated, but it was not successful.\u0000 <figure>\u0000 <div><picture>\u0000 <source></source></picture><p></p>\u0000 </div>\u0000 </figure></p>","PeriodicalId":15543,"journal":{"name":"Journal of Cutaneous Immunology and Allergy","volume":"6 1","pages":"24-25"},"PeriodicalIF":1.0,"publicationDate":"2022-06-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cia2.12264","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"48713231","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}
Erythema exudativum multiforme (EM), Stevens-Johnson syndrome (SJS), and toxic epidermal necrolysis (TEN) are acute inflammatory diseases of the skin and mucous membranes. EM with mucosal eruptions is sometimes difficult to differentiate from SJS or TEN. This study aimed to understand the characteristics of these diseases by evaluating the backgrounds, clinical symptoms, and disease courses of EM/SJS/TEN patients treated at our hospital. It shows that persistent fevers and erosion are common in SJS/TEN. Therefore, we must pay attention to whether they become more severe.
{"title":"Evaluation of patients with erythema exudativum multiforme, Stevens-Johnson syndrome, and toxic epidermal necrolysis treated at our department during the previous 9-year period","authors":"Midori Kawasaki-Nagano MD, Risa Tamagawa-Mineoka MD, PhD, Koji Masuda MD, PhD, Mayumi Ueta MD, PhD, Chie Sotozono MD, PhD, Norito Katoh MD, PhD","doi":"10.1002/cia2.12259","DOIUrl":"10.1002/cia2.12259","url":null,"abstract":"<p>Erythema exudativum multiforme (EM), Stevens-Johnson syndrome (SJS), and toxic epidermal necrolysis (TEN) are acute inflammatory diseases of the skin and mucous membranes. EM with mucosal eruptions is sometimes difficult to differentiate from SJS or TEN. This study aimed to understand the characteristics of these diseases by evaluating the backgrounds, clinical symptoms, and disease courses of EM/SJS/TEN patients treated at our hospital. It shows that persistent fevers and erosion are common in SJS/TEN. Therefore, we must pay attention to whether they become more severe.</p>","PeriodicalId":15543,"journal":{"name":"Journal of Cutaneous Immunology and Allergy","volume":"5 5","pages":"174-178"},"PeriodicalIF":1.0,"publicationDate":"2022-06-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cia2.12259","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"47257295","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}
Excipient allergies are rare and difficult to diagnose. Carboxymethylcellulose (CMC, carmellose sodium) is an anionic water-soluble polymer derived from native cellulose, that is, used as an excipient. Here, we report a case of urticaria caused by the CMC in lidocaine jelly and dimethicone drops, which had used for upper gastrointestinal endoscopy. CMC is widely used in pharmaceutical preparations, food additives, and other pharmaceuticals, and its use is increasing. However, there are few reports on immediate hypersensitivity reactions because substances containing CMC. Previous reports and our case suggest that excipients, such as CMC, can be potential hidden allergens.
{"title":"Immediate hypersensitivity reaction to carboxymethylcellulose in lidocaine jelly and dimethicone drops: A case report and mini-review","authors":"Eri Hotta MD, PhD, Risa Tamagawa-Mineoka MD, PhD, Yuri Onishi MD, Ayaka Sotozono MD, Megumi Kusunoki MD, Junko Hattori MD, Natsue Ioka MD, Hiromi Mizutani MD, PhD, Koji Masuda MD, PhD, Norito Katoh MD, PhD","doi":"10.1002/cia2.12261","DOIUrl":"10.1002/cia2.12261","url":null,"abstract":"<p>Excipient allergies are rare and difficult to diagnose. Carboxymethylcellulose (CMC, carmellose sodium) is an anionic water-soluble polymer derived from native cellulose, that is, used as an excipient. Here, we report a case of urticaria caused by the CMC in lidocaine jelly and dimethicone drops, which had used for upper gastrointestinal endoscopy. CMC is widely used in pharmaceutical preparations, food additives, and other pharmaceuticals, and its use is increasing. However, there are few reports on immediate hypersensitivity reactions because substances containing CMC. Previous reports and our case suggest that excipients, such as CMC, can be potential hidden allergens.</p>","PeriodicalId":15543,"journal":{"name":"Journal of Cutaneous Immunology and Allergy","volume":"5 6","pages":"217-221"},"PeriodicalIF":1.0,"publicationDate":"2022-06-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cia2.12261","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"43820491","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}
Satoshi Yoshida MD, Kazuki Yatsuzuka MD, Yuta Kuroo MD, Ryo Utsunomiya MD, PhD, Rina Ando MD, PhD, Jun Muto MD, PhD, Koji Sayama MD, PhD
We describe a 73-year-old patient who presented with marked hemifacial swelling secondary to herpes zoster. Magnetic resonance imaging (MRI) revealed hyperintensity of the spinal trigeminal nucleus. In the case presented here, stimulation of the spinal trigeminal nucleus owing to herpes zoster can cause vasodilation of the facial skin.
{"title":"Involvement of the spinal trigeminal nucleus secondary to herpes zoster in a patient with hemifacial redness and swelling","authors":"Satoshi Yoshida MD, Kazuki Yatsuzuka MD, Yuta Kuroo MD, Ryo Utsunomiya MD, PhD, Rina Ando MD, PhD, Jun Muto MD, PhD, Koji Sayama MD, PhD","doi":"10.1002/cia2.12262","DOIUrl":"10.1002/cia2.12262","url":null,"abstract":"<p>We describe a 73-year-old patient who presented with marked hemifacial swelling secondary to herpes zoster. Magnetic resonance imaging (MRI) revealed hyperintensity of the spinal trigeminal nucleus. In the case presented here, stimulation of the spinal trigeminal nucleus owing to herpes zoster can cause vasodilation of the facial skin.\u0000 <figure>\u0000 <div><picture>\u0000 <source></source></picture><p></p>\u0000 </div>\u0000 </figure></p>","PeriodicalId":15543,"journal":{"name":"Journal of Cutaneous Immunology and Allergy","volume":"5 6","pages":"238-239"},"PeriodicalIF":1.0,"publicationDate":"2022-06-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cia2.12262","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"45476763","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}
Immune checkpoint inhibitors are currently developed for the treatment of cancers showing high efficacy even in the cases of advanced and persistent malignancies. Psoriasis has been reported as a rare irAE in both the exacerbation of preexisting psoriasis and the novel onset psoriasis during immunotherapy. Herein, we report a case of pre-existence psoriasis, which was exacerbated following the administration of immune checkpoint inhibitors. We also summarize case reports and conducted a review of the literature.
{"title":"Exacerbation of pre-existence psoriasis following immune checkpoint inhibitor treatment","authors":"Yoko Minokawa MD, Yu Sawada MD, PhD","doi":"10.1002/cia2.12244","DOIUrl":"10.1002/cia2.12244","url":null,"abstract":"<p>Immune checkpoint inhibitors are currently developed for the treatment of cancers showing high efficacy even in the cases of advanced and persistent malignancies. Psoriasis has been reported as a rare irAE in both the exacerbation of preexisting psoriasis and the novel onset psoriasis during immunotherapy. Herein, we report a case of pre-existence psoriasis, which was exacerbated following the administration of immune checkpoint inhibitors. We also summarize case reports and conducted a review of the literature.\u0000 <figure>\u0000 <div><picture>\u0000 <source></source></picture><p></p>\u0000 </div>\u0000 </figure></p>","PeriodicalId":15543,"journal":{"name":"Journal of Cutaneous Immunology and Allergy","volume":"5 5","pages":"196-198"},"PeriodicalIF":1.0,"publicationDate":"2022-06-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cia2.12244","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"47432443","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 : 2022-05-30DOI: 10.37191/mapsci-2582-6549-3(1)-029
H. B. Reinfeld
The novel coronavirus of 2019 has been present among us for decades, mainly confined to cattle and livestock where it is harmless and up until the end of 2019, did not infect human cells. Once it did, it produced a combination of mild to severe manifestations that guaranteed its spread across the globe. The combination of aerosol transmission alongside barely detectible and nonspecific symptoms allowed it to spread unmanageable. Finally, the ultimate onslaught of respiratory distress in a small proportion of unfortunate individuals solidified its deadliness. By the time a decent understanding of the situation was achieved, its already too late to contain it.
{"title":"Prevention Risks of Post Covid-19 Infection and Rebound Symptoms","authors":"H. B. Reinfeld","doi":"10.37191/mapsci-2582-6549-3(1)-029","DOIUrl":"https://doi.org/10.37191/mapsci-2582-6549-3(1)-029","url":null,"abstract":"The novel coronavirus of 2019 has been present among us for decades, mainly confined to cattle and livestock where it is harmless and up until the end of 2019, did not infect human cells. Once it did, it produced a combination of mild to severe manifestations that guaranteed its spread across the globe. The combination of aerosol transmission alongside barely detectible and nonspecific symptoms allowed it to spread unmanageable. Finally, the ultimate onslaught of respiratory distress in a small proportion of unfortunate individuals solidified its deadliness. By the time a decent understanding of the situation was achieved, its already too late to contain it.","PeriodicalId":15543,"journal":{"name":"Journal of Cutaneous Immunology and Allergy","volume":"368 1","pages":""},"PeriodicalIF":1.0,"publicationDate":"2022-05-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"73939795","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
<p>A 4-year-old girl with refractory nummular eczema with atopic dermatitis (AD) was reported successfully treated with narrowband ultraviolet B (NB-UVB) once a week (400 mJ/cm<sup>2</sup>) and topical delgocitinib for 8 weeks. The treatment of NB-UVB and topical delgocitinib improved the severe nummular lesions and strong pruritus, resulting in only brown postinflammatory hyperpigmentation without pruritus. The combination of NB-UVB and topical delgocitinib can be an alternative treatment for refractory nummular eczema in children.</p><p>A 4-year-old girl presented with a 2-year history of AD. She had impetigo contagiosum throughout her body due to methicillin-resistant <i>Staphylococcus aureus</i> (MRSA) infection. Subsequently, nummular eczema with elevated erythema, erosion, and brown pigmentation occurred over the former impetigo lesions on the shoulders, buttocks (Figure 1A), and thighs with severe pruritus. She was treated with topical steroids, oral antihistamines, and antimicrobials. However, the patient did not respond to these treatments. Thus, NB-UVB therapy (400 mJ/cm<sup>2</sup>) once a week and topical delgocitinib twice a day were administered. After 8 weeks, the nummular eczema remarkably improved, resulting in flat brown pigmentation (Figure 1B). Laboratory findings showed eosinophilia (22%) and high immunoglobulin (Ig) E levels (853 IU/ml). Radioallergosorbent test (RAST) had a score of 6 (House dust 1 and dust mite). Bacterial culture from nummular eczema was negative.</p><p>Topical corticosteroids, antihistamines, and antimicrobials were ineffective in our case. NB-UVB therapy is a tolerant and effective treatment for children with AD.<span><sup>1</sup></span> NB-UVB inhibits immunological reactions and has anti-inflammatory and anti-bacterial effects. It also recovers skin barrier defects.<span><sup>2</sup></span> Therefore, NB-UVB therapy is a tolerant and economical treatment for children with AD. Moreover, it inhibits immune reactions, cytotoxic effects, cis-urocanic induction, and decreases Langerhans cells, antigen presentation, NK cell activity, and apoptosis of T cells and keratinocytes.<span><sup>3</sup></span> However, the side effects of NB-UVB include erythema, reactivation of herpes simplex, and polymorphous light eruption.</p><p>Delgocitinib, a Janus kinase (JAK) inhibitor, is useful for treating AD.<span><sup>4</sup></span> It is available for children with AD with ages more than 2 years old.<span><sup>5</sup></span> It inhibits IL-4, IL-13, and IL-31,<span><sup>6</sup></span> resulting in the relief of pruritus.</p><p>In our case, the patient did not respond to topical corticosteroids, antihistamines, or oral antimicrobials. We preferred NB-UVB and topical delgocitinib treatments. We speculated that the synergistic effects of NB-UVB and delgocitinib improved the refractory nummular eczema.</p><p>In our case, to reduce the risk, we should have tried to use topical delgocitinib alone at first. Additionally, the
{"title":"Refractory nummular eczema in child successfully treated with NB-UVB and topical delgocitinib","authors":"Ichiro Kurokawa MD, Jun-Ichiro Ono MD","doi":"10.1002/cia2.12250","DOIUrl":"10.1002/cia2.12250","url":null,"abstract":"<p>A 4-year-old girl with refractory nummular eczema with atopic dermatitis (AD) was reported successfully treated with narrowband ultraviolet B (NB-UVB) once a week (400 mJ/cm<sup>2</sup>) and topical delgocitinib for 8 weeks. The treatment of NB-UVB and topical delgocitinib improved the severe nummular lesions and strong pruritus, resulting in only brown postinflammatory hyperpigmentation without pruritus. The combination of NB-UVB and topical delgocitinib can be an alternative treatment for refractory nummular eczema in children.</p><p>A 4-year-old girl presented with a 2-year history of AD. She had impetigo contagiosum throughout her body due to methicillin-resistant <i>Staphylococcus aureus</i> (MRSA) infection. Subsequently, nummular eczema with elevated erythema, erosion, and brown pigmentation occurred over the former impetigo lesions on the shoulders, buttocks (Figure 1A), and thighs with severe pruritus. She was treated with topical steroids, oral antihistamines, and antimicrobials. However, the patient did not respond to these treatments. Thus, NB-UVB therapy (400 mJ/cm<sup>2</sup>) once a week and topical delgocitinib twice a day were administered. After 8 weeks, the nummular eczema remarkably improved, resulting in flat brown pigmentation (Figure 1B). Laboratory findings showed eosinophilia (22%) and high immunoglobulin (Ig) E levels (853 IU/ml). Radioallergosorbent test (RAST) had a score of 6 (House dust 1 and dust mite). Bacterial culture from nummular eczema was negative.</p><p>Topical corticosteroids, antihistamines, and antimicrobials were ineffective in our case. NB-UVB therapy is a tolerant and effective treatment for children with AD.<span><sup>1</sup></span> NB-UVB inhibits immunological reactions and has anti-inflammatory and anti-bacterial effects. It also recovers skin barrier defects.<span><sup>2</sup></span> Therefore, NB-UVB therapy is a tolerant and economical treatment for children with AD. Moreover, it inhibits immune reactions, cytotoxic effects, cis-urocanic induction, and decreases Langerhans cells, antigen presentation, NK cell activity, and apoptosis of T cells and keratinocytes.<span><sup>3</sup></span> However, the side effects of NB-UVB include erythema, reactivation of herpes simplex, and polymorphous light eruption.</p><p>Delgocitinib, a Janus kinase (JAK) inhibitor, is useful for treating AD.<span><sup>4</sup></span> It is available for children with AD with ages more than 2 years old.<span><sup>5</sup></span> It inhibits IL-4, IL-13, and IL-31,<span><sup>6</sup></span> resulting in the relief of pruritus.</p><p>In our case, the patient did not respond to topical corticosteroids, antihistamines, or oral antimicrobials. We preferred NB-UVB and topical delgocitinib treatments. We speculated that the synergistic effects of NB-UVB and delgocitinib improved the refractory nummular eczema.</p><p>In our case, to reduce the risk, we should have tried to use topical delgocitinib alone at first. Additionally, the ","PeriodicalId":15543,"journal":{"name":"Journal of Cutaneous Immunology and Allergy","volume":"5 6","pages":"229-230"},"PeriodicalIF":1.0,"publicationDate":"2022-05-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cia2.12250","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"51154501","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}
<p>Various biologics and small-molecule compounds are continuously emerging for the novel treatment of atopic dermatitis (AD). Upadacitinib, an oral, selective Janus kinase (JAK) 1 inhibitor, was approved in August 2021 for moderate-to-severe AD in Japan. The efficacy and safety of upadacitinib have been demonstrated in clinical trials in Japan.<span><sup>1</sup></span> However, no reports of fungal infections were noted in patients with AD treated with upadacitinib. Herein, we report a rare case of a patient with AD who developed tinea corporis during upadacitinib treatment in a real-world clinical setting.</p><p>A 68-year-old Japanese man presented to our department with a 2-year history of whole-body rash. He had been treated with topical application of very strong steroid ointment and 5-15 mg of oral prednisolone for approximately half a year. Oral prednisolone had been discontinued for 3 months before visiting our hospital. Physical examination revealed erythema with scratch marks on his trunk and extremities including on his left lumber without annular plaque (Figure 1A-C). Investigator's Global Assessment and Eczema Area and Severity Index scores were 3 and 18, respectively. He was diagnosed with moderate AD and received 30 mg upadacitinib daily along with a very strong topical steroid. The eczema lesions and itching immediately improved after a few days, but the patient developed erythema with annular scaling on the left lumbar region 2 weeks later (Figure 1D-F). A potassium hydroxide test revealed fungal filaments and septate hyphae. He was diagnosed with tinea corporis and treated with topical application of terbinafine hydrochloride cream, and upadacitinib was continued. Then, tinea lesion was improved.</p><p>Recent studies demonstrated the heterogeneity of AD, and elevated Th22 and Th17 immunity are reported more in Asians than European and American patients.<span><sup>2</sup></span> Previous studies have shown that the interleukin (IL)-23/Th17 pathway is less expressed in AD than in psoriasis, but it is upregulated when compared to healthy controls.<span><sup>3</sup></span> The IL-23 heterodimer binds to the signaling receptors IL-12Rβ1 and IL-23R and activates downstream signaling via phosphorylation of JAK2/tyrosine kinase 2 (TYK2). Upadacitib is a selective JAK1 inhibitor, but its inhibitory effect on JAK2/2- or JAK/TYK2-dependent cytokines has also been reported.<span><sup>4</sup></span> Han et al.<span><sup>5</sup></span> reported that <i>Aspergillus fumigatus</i>–stimulated dendritic cells promoted a Th17 response in CD4<sup>+</sup> T cells via the JAK/STAT signaling pathway. Moreover, there is a care report, which demonstrated disseminated tinea corporis under baricitinib therapy for AD.<span><sup>6</sup></span> These findings suggest that the suppressive effect of Th17 immunity by upadacitinib possibly has contributed to the development of fungal infection, and that suppression of Th17 may be one of the mechanisms by which up
{"title":"Development of tinea corporis in a Japanese patient with atopic dermatitis under treatment with upadacitinib in a real-world clinical setting: Possible contribution of the suppression of Th17","authors":"Akihiko Uchiyama MD, PhD, Sei-ichiro Motegi MD, PhD","doi":"10.1002/cia2.12258","DOIUrl":"10.1002/cia2.12258","url":null,"abstract":"<p>Various biologics and small-molecule compounds are continuously emerging for the novel treatment of atopic dermatitis (AD). Upadacitinib, an oral, selective Janus kinase (JAK) 1 inhibitor, was approved in August 2021 for moderate-to-severe AD in Japan. The efficacy and safety of upadacitinib have been demonstrated in clinical trials in Japan.<span><sup>1</sup></span> However, no reports of fungal infections were noted in patients with AD treated with upadacitinib. Herein, we report a rare case of a patient with AD who developed tinea corporis during upadacitinib treatment in a real-world clinical setting.</p><p>A 68-year-old Japanese man presented to our department with a 2-year history of whole-body rash. He had been treated with topical application of very strong steroid ointment and 5-15 mg of oral prednisolone for approximately half a year. Oral prednisolone had been discontinued for 3 months before visiting our hospital. Physical examination revealed erythema with scratch marks on his trunk and extremities including on his left lumber without annular plaque (Figure 1A-C). Investigator's Global Assessment and Eczema Area and Severity Index scores were 3 and 18, respectively. He was diagnosed with moderate AD and received 30 mg upadacitinib daily along with a very strong topical steroid. The eczema lesions and itching immediately improved after a few days, but the patient developed erythema with annular scaling on the left lumbar region 2 weeks later (Figure 1D-F). A potassium hydroxide test revealed fungal filaments and septate hyphae. He was diagnosed with tinea corporis and treated with topical application of terbinafine hydrochloride cream, and upadacitinib was continued. Then, tinea lesion was improved.</p><p>Recent studies demonstrated the heterogeneity of AD, and elevated Th22 and Th17 immunity are reported more in Asians than European and American patients.<span><sup>2</sup></span> Previous studies have shown that the interleukin (IL)-23/Th17 pathway is less expressed in AD than in psoriasis, but it is upregulated when compared to healthy controls.<span><sup>3</sup></span> The IL-23 heterodimer binds to the signaling receptors IL-12Rβ1 and IL-23R and activates downstream signaling via phosphorylation of JAK2/tyrosine kinase 2 (TYK2). Upadacitib is a selective JAK1 inhibitor, but its inhibitory effect on JAK2/2- or JAK/TYK2-dependent cytokines has also been reported.<span><sup>4</sup></span> Han et al.<span><sup>5</sup></span> reported that <i>Aspergillus fumigatus</i>–stimulated dendritic cells promoted a Th17 response in CD4<sup>+</sup> T cells via the JAK/STAT signaling pathway. Moreover, there is a care report, which demonstrated disseminated tinea corporis under baricitinib therapy for AD.<span><sup>6</sup></span> These findings suggest that the suppressive effect of Th17 immunity by upadacitinib possibly has contributed to the development of fungal infection, and that suppression of Th17 may be one of the mechanisms by which up","PeriodicalId":15543,"journal":{"name":"Journal of Cutaneous Immunology and Allergy","volume":"5 6","pages":"233-235"},"PeriodicalIF":1.0,"publicationDate":"2022-05-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cia2.12258","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"42173747","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}
<p>Dupilumab is a human monoclonal antibody that binds to the alpha-subunit of interleukin (IL)-4 and IL-13 receptors that play a dominant role in the T-helper (Th)2 cytokine cascade related to atopic dermatitis (AD). Dupilumab is useful in treating moderate to severe AD by inhibiting the signaling of IL-4 and IL-13 without severe adverse effects.<span><sup>1</sup></span> Herein, we report a case of a patient with psoriasis-like eruptions, which developed during treatment with dupilumab.</p><p>The 49-year-old man had a history of AD since childhood. He also had bronchial asthma, allergic rhinitis, and allergic conjunctivitis. Although he was treated with topical medications, he experienced repeated flare-ups of eczema. At the time of presentation, erythema was distributed over the trunk and extremities, with some lichenification. There was also skin atrophy on the extremities. He was diagnosed as having severe AD to the Hanifin-Rajika criteria (eczema area and severity index = 19.2, patient-oriented eczema measure = 28, average numerical rating scale = 10, and dermatology life quality index = 29). After the diagnosis, the treatment with dupilumab started according to the dosage regimen for AD (600 mg as the first dose, followed by 300 mg every 2 weeks). Although overall symptoms showed improvement, the flare-up of erythema was observed with the discontinuation of topical steroids. After 16 months of treatment with dupilumab, the patient was referred to us for exacerbation on the scalp and back. Scattered erythematous and scaly plaques, whose morphology was suggestive of psoriasis, were found on the back and scalp (Figure 1A,B). Histopathological examination revealed parakeratosis and hyperkeratosis and a lack of stratum granulosum (Figure 1C). The diagnosis of psoriasis-like eruptions after dupilumab was made. Although the lesion once improved after topical steroids, narrowband UVB, and oral etretinate 50 mg, the eruptions repeated flare-ups. In addition, the AD lesion has also tended to flare up, and continuation of dupilumab has been necessary.</p><p>Psoriasis vulgaris is a disease considered to be driven by a Th17 cascade, with elevated levels of IL-17 A and IL-23. In contrast, AD is a Th2 cell-mediated disease with elevated IL-4 and IL-13 levels. Although there are some reports on the development of psoriasis-like eruptions during treatment with dupilumab for AD, no reports refer to Asians.<span><sup>2</sup></span> Recent studies have revealed that IL-4 negatively regulates Th1 and Th17 cells.<span><sup>3, 4</sup></span> A study on AD endotypes reported elevated levels of Th17-related cytokines in lesional and nonlesional skin of Asian AD patients compared to those of European AD patients.<span><sup>5</sup></span> Thus, we hypothesize that blocking IL-4 by dupilumab may have caused a relative increase in latent Th17-related inflammation, resulting in psoriasis-like eruptions in our case. Above all, the discontinuation of dupilumab was not nec
{"title":"Psoriasis-like eruptions developed in an atopic dermatitis patient treated with dupilumab","authors":"Rai Fujimoto MD, Yoko Kataoka MD","doi":"10.1002/cia2.12251","DOIUrl":"10.1002/cia2.12251","url":null,"abstract":"<p>Dupilumab is a human monoclonal antibody that binds to the alpha-subunit of interleukin (IL)-4 and IL-13 receptors that play a dominant role in the T-helper (Th)2 cytokine cascade related to atopic dermatitis (AD). Dupilumab is useful in treating moderate to severe AD by inhibiting the signaling of IL-4 and IL-13 without severe adverse effects.<span><sup>1</sup></span> Herein, we report a case of a patient with psoriasis-like eruptions, which developed during treatment with dupilumab.</p><p>The 49-year-old man had a history of AD since childhood. He also had bronchial asthma, allergic rhinitis, and allergic conjunctivitis. Although he was treated with topical medications, he experienced repeated flare-ups of eczema. At the time of presentation, erythema was distributed over the trunk and extremities, with some lichenification. There was also skin atrophy on the extremities. He was diagnosed as having severe AD to the Hanifin-Rajika criteria (eczema area and severity index = 19.2, patient-oriented eczema measure = 28, average numerical rating scale = 10, and dermatology life quality index = 29). After the diagnosis, the treatment with dupilumab started according to the dosage regimen for AD (600 mg as the first dose, followed by 300 mg every 2 weeks). Although overall symptoms showed improvement, the flare-up of erythema was observed with the discontinuation of topical steroids. After 16 months of treatment with dupilumab, the patient was referred to us for exacerbation on the scalp and back. Scattered erythematous and scaly plaques, whose morphology was suggestive of psoriasis, were found on the back and scalp (Figure 1A,B). Histopathological examination revealed parakeratosis and hyperkeratosis and a lack of stratum granulosum (Figure 1C). The diagnosis of psoriasis-like eruptions after dupilumab was made. Although the lesion once improved after topical steroids, narrowband UVB, and oral etretinate 50 mg, the eruptions repeated flare-ups. In addition, the AD lesion has also tended to flare up, and continuation of dupilumab has been necessary.</p><p>Psoriasis vulgaris is a disease considered to be driven by a Th17 cascade, with elevated levels of IL-17 A and IL-23. In contrast, AD is a Th2 cell-mediated disease with elevated IL-4 and IL-13 levels. Although there are some reports on the development of psoriasis-like eruptions during treatment with dupilumab for AD, no reports refer to Asians.<span><sup>2</sup></span> Recent studies have revealed that IL-4 negatively regulates Th1 and Th17 cells.<span><sup>3, 4</sup></span> A study on AD endotypes reported elevated levels of Th17-related cytokines in lesional and nonlesional skin of Asian AD patients compared to those of European AD patients.<span><sup>5</sup></span> Thus, we hypothesize that blocking IL-4 by dupilumab may have caused a relative increase in latent Th17-related inflammation, resulting in psoriasis-like eruptions in our case. Above all, the discontinuation of dupilumab was not nec","PeriodicalId":15543,"journal":{"name":"Journal of Cutaneous Immunology and Allergy","volume":"5 6","pages":"231-232"},"PeriodicalIF":1.0,"publicationDate":"2022-05-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/cia2.12251","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"47505820","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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