Nooshin Bagherani, Abolfazl Jokar, Golshan Mirmomeni, Bruce R Smoller, Alireza Ghanadan, Reza Shojaei, Alireza Firooz, Roxana Sahebnasagh, Gholamreza Tavoosidana
Introduction: Aging is a continuous and irreversible process which affects the skin. During the aging process, intrinsic progressive degenerative changes in the skin impair its structure, which makes it prone to different dermatoses, resulting in impaired quality of life in the elderly.
Objective: Carboxytherapy is considered as a safe, minimally invasive modality applied for skin rejuvenation, restoration, and recondition. Herein, we have assessed the efficacy of carboxytherapy in the treatment of skin aging through clinical and sonographic studies.
Materials and methods: Our study was a prospective 2-split clinical trial in which the efficacy of carboxytherapy was assessed in the treatment of abdominal skin aging through clinical and sonographic evaluations.
Results: Twenty-eight patients with skin-intrinsic aging manifestations in the abdomen completed the study. Their mean age was 44.13 years. The mean weight, BMI, and waist circumference of the subjects significantly decreased after the treatment. The clinical subjective and objective evaluations revealed statistically significant improvement of skin wrinkles, laxity, and skin pigmentation and overall satisfaction by carboxytherapy. Upon sonographic investigation, a significant increase in epidermis and dermis thickness was observed. No significant side-effect was reported by the subjects.
{"title":"Clinical and Sonographic Assessment of Carboxytherapy Efficacy in Treatment of Skin Aging: A 2-split Randomized Clinical Trial.","authors":"Nooshin Bagherani, Abolfazl Jokar, Golshan Mirmomeni, Bruce R Smoller, Alireza Ghanadan, Reza Shojaei, Alireza Firooz, Roxana Sahebnasagh, Gholamreza Tavoosidana","doi":"","DOIUrl":"","url":null,"abstract":"<p><strong>Introduction: </strong>Aging is a continuous and irreversible process which affects the skin. During the aging process, intrinsic progressive degenerative changes in the skin impair its structure, which makes it prone to different dermatoses, resulting in impaired quality of life in the elderly.</p><p><strong>Objective: </strong>Carboxytherapy is considered as a safe, minimally invasive modality applied for skin rejuvenation, restoration, and recondition. Herein, we have assessed the efficacy of carboxytherapy in the treatment of skin aging through clinical and sonographic studies.</p><p><strong>Materials and methods: </strong>Our study was a prospective 2-split clinical trial in which the efficacy of carboxytherapy was assessed in the treatment of abdominal skin aging through clinical and sonographic evaluations.</p><p><strong>Results: </strong>Twenty-eight patients with skin-intrinsic aging manifestations in the abdomen completed the study. Their mean age was 44.13 years. The mean weight, BMI, and waist circumference of the subjects significantly decreased after the treatment. The clinical subjective and objective evaluations revealed statistically significant improvement of skin wrinkles, laxity, and skin pigmentation and overall satisfaction by carboxytherapy. Upon sonographic investigation, a significant increase in epidermis and dermis thickness was observed. No significant side-effect was reported by the subjects.</p>","PeriodicalId":94367,"journal":{"name":"Acta dermatovenerologica Croatica : ADC","volume":"32 3","pages":"123-134"},"PeriodicalIF":0.0,"publicationDate":"2024-11-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144628802","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}
We report the case of an 18-month-old boy who developed a phototoxic skin reaction to terbinafine on his scalp, ears, and face in the form of disseminated erythematous plaques, which resembled subacute lupus erythematosus (SCLE) in their clinical presentation. Skin changes appeared a short time after the boy was exposed to sunlight during the period of time when he was treated with oral terbinafine due to Microsporum canis fungal scalp infection. Tinea capitis is a common dermatophyte infection primarily affecting prepubertal children (1). Microsporum canis remains the predominant causative organism in many countries of the Mediterranean basin, the most important dermatophyte carriers being stray cats and dogs. Systemic therapy is required for treatment because topical antifungal agents do not penetrate down to the deepest part of the hair follicle (2). Terbinafine is commonly used in the treatment of microsporosis, as its fungicidal action permits short periods of treatment (3,4). The first skin changes occurred in the parietal scalp region in the form of round scaly alopecia, with the presence of unevenly broken hairs and enlarged regional lymph nodes (Figure 1). Diagnosis of fungal infection included clinical assessment and Wood's light examination, which revealed green-yellow fluorescence on the lesional scalp region. Fungal culture identification was performed according to conventional methods, revealing fungal culture positive for dermatophytes from the genus Microsporum canis. The boy had a history of contact with a cat. Systemic therapy with the oral antifungal drug terbinafine was administered at a dose of 62.5 mg per day (5 mg/kg), with topical application of antifungal cream (miconazole), 10% Ichthyol cream in the evening, and antifungal shampoo (ketoconazole) twice a week. After two weeks of therapy, we observed initial regression of scalp lesions. Oral terbinafine was well-tolerated, and the patient did not experience any side-effects. Laboratory findings included liver function tests and were within normal ranges. At this point, the oral dose of terbinafine was increased to 125 mg per day (10 mg/kg) at a revised schedule according to body weight: 10-25 kg, 125 mg/day (5). Approximately five weeks after starting the treatment with oral terbinafine, after the boy was exposed to the sun, acute disseminated erythematosus lesions appeared on the face and scalp. Clinical presentation of the lesions and acute onset during exposure to sunlight raised the suspicion of a phototoxic reaction to terbinafine (Figure 2). The patient was not taking any other medication at that time, had no history of drug or food allergies, and had not previously experienced photosensitive skin reactions. Due to the inflamed skin changes resembling subacute lupus and photosensitivity, an immunological assay tests were also performed. Due to the young age of the patient, no skin biopsy or photo-patch test was performed. Despite the recent skin changes and sus
{"title":"Phototoxic reaction to oral terbinafine due to Tinea capitis in a child.","authors":"Ana Bakija-Konsuo, Lena Kotrulja, Matko Marlais","doi":"","DOIUrl":"","url":null,"abstract":"<p><p>We report the case of an 18-month-old boy who developed a phototoxic skin reaction to terbinafine on his scalp, ears, and face in the form of disseminated erythematous plaques, which resembled subacute lupus erythematosus (SCLE) in their clinical presentation. Skin changes appeared a short time after the boy was exposed to sunlight during the period of time when he was treated with oral terbinafine due to Microsporum canis fungal scalp infection. Tinea capitis is a common dermatophyte infection primarily affecting prepubertal children (1). Microsporum canis remains the predominant causative organism in many countries of the Mediterranean basin, the most important dermatophyte carriers being stray cats and dogs. Systemic therapy is required for treatment because topical antifungal agents do not penetrate down to the deepest part of the hair follicle (2). Terbinafine is commonly used in the treatment of microsporosis, as its fungicidal action permits short periods of treatment (3,4). The first skin changes occurred in the parietal scalp region in the form of round scaly alopecia, with the presence of unevenly broken hairs and enlarged regional lymph nodes (Figure 1). Diagnosis of fungal infection included clinical assessment and Wood's light examination, which revealed green-yellow fluorescence on the lesional scalp region. Fungal culture identification was performed according to conventional methods, revealing fungal culture positive for dermatophytes from the genus Microsporum canis. The boy had a history of contact with a cat. Systemic therapy with the oral antifungal drug terbinafine was administered at a dose of 62.5 mg per day (5 mg/kg), with topical application of antifungal cream (miconazole), 10% Ichthyol cream in the evening, and antifungal shampoo (ketoconazole) twice a week. After two weeks of therapy, we observed initial regression of scalp lesions. Oral terbinafine was well-tolerated, and the patient did not experience any side-effects. Laboratory findings included liver function tests and were within normal ranges. At this point, the oral dose of terbinafine was increased to 125 mg per day (10 mg/kg) at a revised schedule according to body weight: 10-25 kg, 125 mg/day (5). Approximately five weeks after starting the treatment with oral terbinafine, after the boy was exposed to the sun, acute disseminated erythematosus lesions appeared on the face and scalp. Clinical presentation of the lesions and acute onset during exposure to sunlight raised the suspicion of a phototoxic reaction to terbinafine (Figure 2). The patient was not taking any other medication at that time, had no history of drug or food allergies, and had not previously experienced photosensitive skin reactions. Due to the inflamed skin changes resembling subacute lupus and photosensitivity, an immunological assay tests were also performed. Due to the young age of the patient, no skin biopsy or photo-patch test was performed. Despite the recent skin changes and sus","PeriodicalId":94367,"journal":{"name":"Acta dermatovenerologica Croatica : ADC","volume":"32 2","pages":"113-114"},"PeriodicalIF":0.0,"publicationDate":"2024-11-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142974295","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}
Krzysztof Kanabaj, Barbara Bulińska, Małgorzata Sokołowska-Wojdyło
Schnitzler syndrome (SS) is an extremely rare acquired systemic disease that shares many similarities with various hereditary autoinflammatory syndromes. It presents as chronic non-pruritic urticarial rash, monoclonal gammopathy, and systemic symptoms, such as recurrent fever, arthralgia, myalgia, bone pain, bone lesions, and enlargement of the spleen and liver. The specific feature associated with SS is its spectacular response to treatment using anti-interleukin-1 (anti-IL-1) agents, such as anakinra or canakinumab. If it remains untreated, the disease can have a devastating effect on the patient's quality of life as well as increased mortality due to systemic complications. Herein, we will summarize the most recent findings in the pathogenesis, diagnosis, and management of SS.
{"title":"A Modern Look at Schnitzler Syndrome - A Literature Review.","authors":"Krzysztof Kanabaj, Barbara Bulińska, Małgorzata Sokołowska-Wojdyło","doi":"","DOIUrl":"","url":null,"abstract":"<p><p>Schnitzler syndrome (SS) is an extremely rare acquired systemic disease that shares many similarities with various hereditary autoinflammatory syndromes. It presents as chronic non-pruritic urticarial rash, monoclonal gammopathy, and systemic symptoms, such as recurrent fever, arthralgia, myalgia, bone pain, bone lesions, and enlargement of the spleen and liver. The specific feature associated with SS is its spectacular response to treatment using anti-interleukin-1 (anti-IL-1) agents, such as anakinra or canakinumab. If it remains untreated, the disease can have a devastating effect on the patient's quality of life as well as increased mortality due to systemic complications. Herein, we will summarize the most recent findings in the pathogenesis, diagnosis, and management of SS.</p>","PeriodicalId":94367,"journal":{"name":"Acta dermatovenerologica Croatica : ADC","volume":"32 3","pages":"154-158"},"PeriodicalIF":0.0,"publicationDate":"2024-11-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144628800","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}
Autosomal recessive congenital ichthyosis (ARCI) comprises a group of rare, clinically heterogeneous disorders of keratinization, characterized by hyperkeratosis, abnormal skin scaling, and a variable degree of erythroderma. Affected infants are most often born encased in a collodion membrane, which is usually shed within 2-4 weeks, revealing the underlying skin condition. To date, at least 14 genes have been identified as causative for ARCI, and phenotypes associated with mutation of different genes may overlap. Herein we report the case of an infant with ARCI due to heterozygous pathogenic mutations in the 12(R)-lipoxygenase (ALOX12B) gene.
{"title":"Autosomal Recessive Congenital Ichthyosis Due to Heterozygote Variants in the ALOX12B gene Presenting as Mild Nonbullous Congenital Ichthyosiform Erythroderma.","authors":"Iva Hižar Gašpar, Arnes Rešić, Nives Pustišek, Ljubica Odak","doi":"","DOIUrl":"","url":null,"abstract":"<p><p>Autosomal recessive congenital ichthyosis (ARCI) comprises a group of rare, clinically heterogeneous disorders of keratinization, characterized by hyperkeratosis, abnormal skin scaling, and a variable degree of erythroderma. Affected infants are most often born encased in a collodion membrane, which is usually shed within 2-4 weeks, revealing the underlying skin condition. To date, at least 14 genes have been identified as causative for ARCI, and phenotypes associated with mutation of different genes may overlap. Herein we report the case of an infant with ARCI due to heterozygous pathogenic mutations in the 12(R)-lipoxygenase (ALOX12B) gene.</p>","PeriodicalId":94367,"journal":{"name":"Acta dermatovenerologica Croatica : ADC","volume":"32 3","pages":"159-162"},"PeriodicalIF":0.0,"publicationDate":"2024-11-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144628801","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}
Eccrine angiomatous hamartoma (EAH) is a rare benign skin neoplasm characterized by an increased size and number of eccrine glands or ducts, along with proliferation of vascular structures in the dermis. This case is unique in its presentation of bilateral symmetrical nodules on both hands and the development of new nodules during puberty. It highlights the need for further research and understanding of this rare condition and its potential progression over time.
{"title":"A Pediatric Case of Multiple Bilateral Symmetric Eccrine Angiomatous Hamartoma.","authors":"Ilke Beyitler, Fikret Dirilenoglu","doi":"","DOIUrl":"","url":null,"abstract":"<p><p>Eccrine angiomatous hamartoma (EAH) is a rare benign skin neoplasm characterized by an increased size and number of eccrine glands or ducts, along with proliferation of vascular structures in the dermis. This case is unique in its presentation of bilateral symmetrical nodules on both hands and the development of new nodules during puberty. It highlights the need for further research and understanding of this rare condition and its potential progression over time.</p>","PeriodicalId":94367,"journal":{"name":"Acta dermatovenerologica Croatica : ADC","volume":"32 2","pages":"102-104"},"PeriodicalIF":0.0,"publicationDate":"2024-11-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142974289","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><p>Familial benign chronic pemphigus, also known as Hailey-Hailey disease, was first described by the Hailey brothers in 1939 (1). It represents a chronic autosomal-dominant genetic skin disorder with incomplete penetrance, usually diagnosed in children and young adults. As a result, family history of this disorder can be elicited in only about 66% of patients. We describe herein a patient with Hailey-Hailey disease who received treatment with hydroxyurea for a new diagnosis of polycythemia vera, with a surprising outcome. A 54-year-old man was diagnosed with Hailey-Hailey disease at age ten when he presented with several erythematous and blistering skin lesions involving the neck, wrist flexure surfaces, and the forearms. The patient underwent regular dermatology follow-up for this condition. His disease followed a relapsing-remitting pattern, with short disease-free intervals. It predominantly involved the neck, torso, and upper extremities (Figure 1), and only rarely buttocks and groin areas. Initially, mild-moderate potency topical steroid creams were tried, with only modest success. Topical antibiotics were required on several occasions due to secondary infections. Photodynamic therapy was only minimally helpful, as the disease continued to worsen. In his 40s, the disease became more difficult to manage, and several systemic options were tried, with very little if any success. Thus, the patient failed oral steroids, dapsone and azathioprine. He became anxious, depressed, and socially isolated. Other past medical history was significant for hypertension. The patient was a never-smoker, and denied alcohol or drug abuse. There was no family history of skin disorders of cancers in his immediate family members. In May 2019, the patient presented with elevated hemoglobin/hematocrit and moderate thrombocytosis. Further work-up identified JAK-2 V617F kinase mutated polycythemia vera, for which he was started on periodic phlebotomies and low-dose aspirin. Four months later, hydroxyurea was prescribed due to increased phlebotomy needs and worsening thrombocytosis. The hydroxyurea dose was subsequently titrated to 1000 mg orally per day, alternating with 1500 mg orally per day. The patient tolerated this agent well, without significant side-effects. He also achieved excellent control of hematocrit and normalization of platelet count. Pleasantly surprised, the patient also realized that he had not experienced any more relapsing Hailey-Hailey skin lesions 8 weeks after the commencement of hydroxyurea. Four years later, his polycythemia remains in excellent control. He also remains without any further evidence of skin lesions. The hallmark of Hailey-Hailey disease is believed to be the haploinsufficiency of the enzyme ATP2C1 (2). The ATP2C1 gene is located on chromosome 3 and encodes a Ca2+ ATPase protein. A mutation in one copy of the gene causes only half of this necessary protein to be synthesized. Consequently, impaired keratinocyte adhesion ensues
{"title":"Severe Relapsing Hailey-Hailey Disease Displaying a Durable Complete Response to Hydroxyurea.","authors":"Constantin A Dasanu","doi":"","DOIUrl":"","url":null,"abstract":"<p><p>Familial benign chronic pemphigus, also known as Hailey-Hailey disease, was first described by the Hailey brothers in 1939 (1). It represents a chronic autosomal-dominant genetic skin disorder with incomplete penetrance, usually diagnosed in children and young adults. As a result, family history of this disorder can be elicited in only about 66% of patients. We describe herein a patient with Hailey-Hailey disease who received treatment with hydroxyurea for a new diagnosis of polycythemia vera, with a surprising outcome. A 54-year-old man was diagnosed with Hailey-Hailey disease at age ten when he presented with several erythematous and blistering skin lesions involving the neck, wrist flexure surfaces, and the forearms. The patient underwent regular dermatology follow-up for this condition. His disease followed a relapsing-remitting pattern, with short disease-free intervals. It predominantly involved the neck, torso, and upper extremities (Figure 1), and only rarely buttocks and groin areas. Initially, mild-moderate potency topical steroid creams were tried, with only modest success. Topical antibiotics were required on several occasions due to secondary infections. Photodynamic therapy was only minimally helpful, as the disease continued to worsen. In his 40s, the disease became more difficult to manage, and several systemic options were tried, with very little if any success. Thus, the patient failed oral steroids, dapsone and azathioprine. He became anxious, depressed, and socially isolated. Other past medical history was significant for hypertension. The patient was a never-smoker, and denied alcohol or drug abuse. There was no family history of skin disorders of cancers in his immediate family members. In May 2019, the patient presented with elevated hemoglobin/hematocrit and moderate thrombocytosis. Further work-up identified JAK-2 V617F kinase mutated polycythemia vera, for which he was started on periodic phlebotomies and low-dose aspirin. Four months later, hydroxyurea was prescribed due to increased phlebotomy needs and worsening thrombocytosis. The hydroxyurea dose was subsequently titrated to 1000 mg orally per day, alternating with 1500 mg orally per day. The patient tolerated this agent well, without significant side-effects. He also achieved excellent control of hematocrit and normalization of platelet count. Pleasantly surprised, the patient also realized that he had not experienced any more relapsing Hailey-Hailey skin lesions 8 weeks after the commencement of hydroxyurea. Four years later, his polycythemia remains in excellent control. He also remains without any further evidence of skin lesions. The hallmark of Hailey-Hailey disease is believed to be the haploinsufficiency of the enzyme ATP2C1 (2). The ATP2C1 gene is located on chromosome 3 and encodes a Ca2+ ATPase protein. A mutation in one copy of the gene causes only half of this necessary protein to be synthesized. Consequently, impaired keratinocyte adhesion ensues","PeriodicalId":94367,"journal":{"name":"Acta dermatovenerologica Croatica : ADC","volume":"32 3","pages":"168-169"},"PeriodicalIF":0.0,"publicationDate":"2024-11-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144628714","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><p>Lupus erythematosus is a multisystem disease which frequently involves the skin. There are several variants of cutaneous lupus, which are defined and classified by the location and the depth of the inflammatory infiltrate, adnexal involvement, presence or absence of interphase dermatitis, and chronology (1). The most common clinical subtypes are acute, subacute and chronic cutaneous lupus erythematosus; however, other rare specific and non-specific cutaneous involvements also exist (2). Rowell syndrome is one of these rare specific variants and was originally described as the association of lupus erythematosus, erythema multiforme-like lesions without any known precipitating factors, and immunological abnormalities such as a speckled pattern of antinuclear antibody (ANA) staining, positive Anti-La antibody, and reactive rheumatoid factor (3). Subsequently, in order to enhance diagnostic specificity, the criteria were redefined as major (lupus erythematosus, erythema multiforme-like lesions, speckled pattern of ANA staining) and minor (chilblains, positive Anti-La or Anti-Ro antibodies, reactive rheumatoid factor); patients should present all three major criteria plus at least one minor criterion to be diagnosed with Rowell syndrome (4). First line treatment options for cutaneous lupus as well for Rowell syndrome comprise topical corticosteroids and calcineurin inhibitors, systemic anti-malarial therapy, and systemic corticosteroids (for active disease). In anti-malarial resistant disease, retinoids, dapsone, methotrexate, and other systemic immunosuppressive agents can be considered, though with a lower level of evidence (5). Herein, we present the case of a patient with Rowell syndrome with a therapeutic approach that is rarely included in the literature. Informed consent was obtained and signed from the patient regarding the use of the patient's information for the purposes of writing a case report publication. A 38-year-old woman who had been examined by the Rheumatology Department for connective tissue disease (CTD) because of her morning stiffness and peripheral arthritis was referred to us for consultation due to the new onset of a mild, itchy rash. The patient's lesions first appeared on her face, neck and upper trunk, subsequently becoming generalized. There was no previous history of recent infection or medication. The patient underwent follow-up under hydroxychloroquine therapy (400 mg/day) for CTD for 2 months. Dermatological physical examination showed violaceous-dark erythematous plaques with a prominent arcuate/targetoid shape at the periphery were present on her whole body, with oral mucosal erosions (Figure 1). In previous laboratory studies, ANA positivity with a speckled pattern and Anti-Ro positivity were observed. Rheumatoid factor was non-reactive. A punch biopsy was performed. Histopathological examination showed prominent interface dermatitis with basal vacuolar degeneration and apoptotic keratinocytes, which correspo
{"title":"A Case of Rowell Syndrome: Excellent Response to Oral Cyclosporine.","authors":"Ece Gokyayla, Sema Koç Yıldırım","doi":"","DOIUrl":"","url":null,"abstract":"<p><p>Lupus erythematosus is a multisystem disease which frequently involves the skin. There are several variants of cutaneous lupus, which are defined and classified by the location and the depth of the inflammatory infiltrate, adnexal involvement, presence or absence of interphase dermatitis, and chronology (1). The most common clinical subtypes are acute, subacute and chronic cutaneous lupus erythematosus; however, other rare specific and non-specific cutaneous involvements also exist (2). Rowell syndrome is one of these rare specific variants and was originally described as the association of lupus erythematosus, erythema multiforme-like lesions without any known precipitating factors, and immunological abnormalities such as a speckled pattern of antinuclear antibody (ANA) staining, positive Anti-La antibody, and reactive rheumatoid factor (3). Subsequently, in order to enhance diagnostic specificity, the criteria were redefined as major (lupus erythematosus, erythema multiforme-like lesions, speckled pattern of ANA staining) and minor (chilblains, positive Anti-La or Anti-Ro antibodies, reactive rheumatoid factor); patients should present all three major criteria plus at least one minor criterion to be diagnosed with Rowell syndrome (4). First line treatment options for cutaneous lupus as well for Rowell syndrome comprise topical corticosteroids and calcineurin inhibitors, systemic anti-malarial therapy, and systemic corticosteroids (for active disease). In anti-malarial resistant disease, retinoids, dapsone, methotrexate, and other systemic immunosuppressive agents can be considered, though with a lower level of evidence (5). Herein, we present the case of a patient with Rowell syndrome with a therapeutic approach that is rarely included in the literature. Informed consent was obtained and signed from the patient regarding the use of the patient's information for the purposes of writing a case report publication. A 38-year-old woman who had been examined by the Rheumatology Department for connective tissue disease (CTD) because of her morning stiffness and peripheral arthritis was referred to us for consultation due to the new onset of a mild, itchy rash. The patient's lesions first appeared on her face, neck and upper trunk, subsequently becoming generalized. There was no previous history of recent infection or medication. The patient underwent follow-up under hydroxychloroquine therapy (400 mg/day) for CTD for 2 months. Dermatological physical examination showed violaceous-dark erythematous plaques with a prominent arcuate/targetoid shape at the periphery were present on her whole body, with oral mucosal erosions (Figure 1). In previous laboratory studies, ANA positivity with a speckled pattern and Anti-Ro positivity were observed. Rheumatoid factor was non-reactive. A punch biopsy was performed. Histopathological examination showed prominent interface dermatitis with basal vacuolar degeneration and apoptotic keratinocytes, which correspo","PeriodicalId":94367,"journal":{"name":"Acta dermatovenerologica Croatica : ADC","volume":"32 3","pages":"170-171"},"PeriodicalIF":0.0,"publicationDate":"2024-11-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144628799","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}
Sapir Itzhaki Gabay, Barak Zlakishvili, Amir Horev
Background: An extensive body of literature has been published regarding alopecia areata (AA) in the past 50 years. The current paper used a bibliometric analysis (BA) to identify high-quality research articles using criteria such as annual citations (ACs) and journal impact factor.
Objectives: To identify and analyze the top 100 most cited articles in AA scientific literature over the past 50 years using BA methods.
Methods: Web of Science (webOS) citation indexing database was used, on April 4th, 2023, to identify the most cited articles on AA. Articles were ranked by their ACs. Data sets were then subdivided into corresponding and senior authors, year of publication, journal and impact factor, total citations according to webOS database, ACs, affiliation, country of origin, manuscript type, design, focus, and usage count since 2013.
Results: The extracted articles were published between 1975-2019. Mean total citations ranged between 67 and 578. The most cited paper was: "Tofacitinib for the treatment of severe alopecia areata and variants: A study of 90 patients" by Liu et al. with an AC of 26.5. Most publications were published between 1990 and 1999 (n=28). The Journal of the American Academy of Dermatology was the most published journal (25 articles). The research focus of original papers was treatment (36%), epidemiology (22%), pathogenesis (20%), basic science (16%), and diagnosis (6%).
Conclusion: This analysis is the first to provide detailed bibliometric characteristics, highlighting the worldwide burden and research trends in.
背景:在过去的 50 年中,发表了大量有关斑秃(AA)的文献。本文采用文献计量分析法(BA),以年度引文(AC)和期刊影响因子等标准来识别高质量的研究文章:采用文献计量学分析方法,确定并分析过去 50 年 AA 科学文献中被引用次数最多的前 100 篇文章:方法:使用 Web of Science(webOS)引文索引数据库(2023 年 4 月 4 日)来确定有关 AA 的被引用次数最多的文章。文章按其 AC 排序。然后将数据集细分为通讯作者和资深作者、发表年份、期刊和影响因子、webOS数据库的总引用次数、ACs、所属单位、原籍国、稿件类型、设计、重点以及自2013年以来的使用次数:提取的文章发表于 1975-2019 年间。平均总被引次数介于 67 与 578 之间。被引用次数最多的论文是"托法替尼治疗重度斑秃及其变异型:90名患者的研究",AC值为26.5。大多数论文发表于 1990 年至 1999 年(n=28)。美国皮肤病学会杂志》是发表文章最多的杂志(25 篇)。原创论文的研究重点是治疗(36%)、流行病学(22%)、发病机制(20%)、基础科学(16%)和诊断(6%):这项分析首次提供了详细的文献计量学特征,突出了该领域的全球负担和研究趋势。
{"title":"A Bibliometric Analysis of Alopecia Areata Literature over the Past 50 Years.","authors":"Sapir Itzhaki Gabay, Barak Zlakishvili, Amir Horev","doi":"","DOIUrl":"","url":null,"abstract":"<p><strong>Background: </strong>An extensive body of literature has been published regarding alopecia areata (AA) in the past 50 years. The current paper used a bibliometric analysis (BA) to identify high-quality research articles using criteria such as annual citations (ACs) and journal impact factor.</p><p><strong>Objectives: </strong>To identify and analyze the top 100 most cited articles in AA scientific literature over the past 50 years using BA methods.</p><p><strong>Methods: </strong>Web of Science (webOS) citation indexing database was used, on April 4th, 2023, to identify the most cited articles on AA. Articles were ranked by their ACs. Data sets were then subdivided into corresponding and senior authors, year of publication, journal and impact factor, total citations according to webOS database, ACs, affiliation, country of origin, manuscript type, design, focus, and usage count since 2013.</p><p><strong>Results: </strong>The extracted articles were published between 1975-2019. Mean total citations ranged between 67 and 578. The most cited paper was: \"Tofacitinib for the treatment of severe alopecia areata and variants: A study of 90 patients\" by Liu et al. with an AC of 26.5. Most publications were published between 1990 and 1999 (n=28). The Journal of the American Academy of Dermatology was the most published journal (25 articles). The research focus of original papers was treatment (36%), epidemiology (22%), pathogenesis (20%), basic science (16%), and diagnosis (6%).</p><p><strong>Conclusion: </strong>This analysis is the first to provide detailed bibliometric characteristics, highlighting the worldwide burden and research trends in.</p>","PeriodicalId":94367,"journal":{"name":"Acta dermatovenerologica Croatica : ADC","volume":"32 1","pages":"17-25"},"PeriodicalIF":0.0,"publicationDate":"2024-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141474355","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}
Mahnaz Fatahzadeh, Joseph Rinaggio, Robert A Schwartz
Plasma cell mucositis (PCM) is an unusual disorder most evident in the accessible mucosa and usually reported in the upper aerodigestive tract, although it is named according to its specific anatomical site of involvement such as plasma cell cheilitis, plasma cell gingivitis, plasma cell vulvitis, and Zoon's balanitis. PCM reflects a dense polyclonal rather than a monoclonal plasma cell proliferation of unclear and unknown etiology. This perplexing disorder tends to be treated by avoiding possible triggers and intralesional and/or systemic steroids. In this work, we provide a review and update on PCM, which often represents a clinical conundrum.
{"title":"Plasma Cell Mucositis: A Clinical Conundrum.","authors":"Mahnaz Fatahzadeh, Joseph Rinaggio, Robert A Schwartz","doi":"","DOIUrl":"","url":null,"abstract":"<p><p>Plasma cell mucositis (PCM) is an unusual disorder most evident in the accessible mucosa and usually reported in the upper aerodigestive tract, although it is named according to its specific anatomical site of involvement such as plasma cell cheilitis, plasma cell gingivitis, plasma cell vulvitis, and Zoon's balanitis. PCM reflects a dense polyclonal rather than a monoclonal plasma cell proliferation of unclear and unknown etiology. This perplexing disorder tends to be treated by avoiding possible triggers and intralesional and/or systemic steroids. In this work, we provide a review and update on PCM, which often represents a clinical conundrum.</p>","PeriodicalId":94367,"journal":{"name":"Acta dermatovenerologica Croatica : ADC","volume":"32 1","pages":"50-59"},"PeriodicalIF":0.0,"publicationDate":"2024-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141474361","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}
Natsuko Aoki, Hozumi Sano, Kimiko Nakajima, Shigetoshi Sano, Kozo Nakai
Acquired circumscribed hyperpigmented patches and plaques have various differential diagnoses, including post-inflammatory hyperpigmentation and mycosis fungoides (MF). Leukomelanoderma is an uncommon cutaneous condition in which the pathogenesis is not fully elucidated. It has been reported that leukomelanoderma occurs after allergic contact dermatitis from hydroquinone or acute cutaneous graft-versus-host disease (1,2). Hyperpigmented MF is a cutaneous T-cell lymphoma with a frequent CD8+ phenotype (3). Herein, we report a case of leukomelanoderma clinically and histologically resembling hyperpigmented MF. A 55-year-old Japanese woman was referred to our department for evaluation of reticulate pigmentation with pruritic erythema on the face. She had used commercially available depigmenting cosmetic reagents for 20 years and ointment containing 10% hydroquinone for 3 months. Physical examination revealed diffuse hyperpigmentation and demarcated hypopigmented macules on the face and neck (Figure 1, a). Dermoscopy showed depigmented spots and reticulated plus dotted hyperpigmentation; it presented a pseudo-pigment network (Figure 1, b). Histological examination of a tissue specimen biopsied from the lesion showed superficial band-like lymphocytic infiltration in dermis accompanying single cells or small clusters in epidermis (Figure 1, c). Interface changes were observed together with melanophages in the dermis. Melan-A-positive melanocytes were absent. Immunohistochemical analysis demonstrated that the epidermotropic lymphocytes were CD3+CD7-, and they had predominance of CD8+ cells (Figure 1, d). These immunohistochemical results mimicked MF. However, PCR analysis of the T-cell receptor g-gene rearrangement was negative. Closed patch test result with hydroquinone (5% pet.) was graded D2 (+?) and D3 (+). Ten months after discontinuing cosmetic reagents and hydroquinone, the pigmentary changes showed improvement. The pathomechanism of leukomelanoderma is unclear. Although post-inflammatory pigmentation due to allergic or contact dermatitis together with direct depigmenting effects from hydroquinone use has been suggested (1), the immunophenotype of T-cells has not been examined. As observed in our patient, interface changes with melanophages, in addition to frequent CD8+ phenotype of the epidermotropism and dermal infiltrate of lymphocytes, were characteristic for hyperpigmented MF (3). Moreover, minimal CD7 expression was a specific finding for MF (4). T-cell receptor clonality was negative in our patient, but the clonality appears to be detected by PCR in up to 50% of the patients with early MF (3). In contrast, the closed patch test was positive for hydroquinone in our patient, and it is reported that CD8+ T-cells are recruited to the interphase between the epidermis and the dermis of the patients with allergic contact dermatitis (5). CD8+ T-cells might contribute to acute cutaneous graft-versus-host disease-like interface changes and des
获得性环状色素沉着斑和斑块有多种鉴别诊断,包括炎症后色素沉着和真菌病(MF)。白斑病是一种不常见的皮肤病,其发病机制尚未完全阐明。有报道称,白斑病发生于对苯二酚过敏性接触性皮炎或急性皮肤移植物抗宿主病之后(1,2)。色素沉着性白皮病是一种皮肤 T 细胞淋巴瘤,多为 CD8+ 表型(3)。在此,我们报告了一例在临床和组织学上与色素沉着病相似的白斑病病例。一名 55 岁的日本妇女因网状色素沉着伴面部瘙痒性红斑转诊至我科。她使用市售脱色化妆品试剂已有 20 年,使用含 10% 氢醌的软膏已有 3 个月。体检发现面部和颈部有弥漫性色素沉着和分界不清的色素减退斑(图 1,a)。皮肤镜检查显示有色素减退斑和网状加点状色素沉着,呈现假性色素网络(图 1,b)。对病变组织标本的组织学检查显示,真皮层有浅表带状淋巴细胞浸润,表皮层有单细胞或小细胞群(图 1,c)。在真皮层观察到界面变化和噬黑色素细胞。Melan-A阳性黑素细胞缺失。免疫组化分析表明,表皮淋巴细胞为 CD3+CD7-,其中以 CD8+细胞为主(图 1,d)。这些免疫组化结果与 MF 相似。但 T 细胞受体 g 基因重排的 PCR 分析结果为阴性。氢醌(5%)封闭斑贴试验结果分为 D2(+?)和 D3(+)级。停用化妆品试剂和氢醌 10 个月后,色素变化有所改善。白斑病的病理机制尚不清楚。虽然有人认为过敏性或接触性皮炎导致的炎症后色素沉着,以及使用氢醌产生的直接脱色作用(1),但尚未对 T 细胞的免疫表型进行研究。正如在我们的患者身上观察到的那样,除了频繁出现的 CD8+ 表型表皮细胞和真皮浸润的淋巴细胞外,噬黑色素细胞的界面变化也是色素沉着性中耳炎的特征(3)。此外,CD7 表达极少也是 MF 的特异性发现(4)。在我们的患者中,T 细胞受体克隆呈阴性,但在多达 50% 的早期 MF 患者中,PCR 似乎可以检测到克隆(3)。与此相反,我们的患者在封闭斑贴试验中对苯二酚呈阳性,有报道称 CD8+ T 细胞被招募到过敏性接触性皮炎患者的表皮和真皮之间(5)。CD8+ T 细胞可能导致急性皮肤移植物抗宿主病样界面变化,并破坏白斑病皮损中的黑色素细胞。因此,我们的患者被认为是过敏性接触性皮炎表现为白斑病。然而,还需要更多的报告和研究来支持这一观点。因此,我们认为有必要对患者进行随访,因为 MF 并没有绝对消除。
{"title":"Leukomelanoderma Resembling Hyperpigmented Mycosis Fungoides.","authors":"Natsuko Aoki, Hozumi Sano, Kimiko Nakajima, Shigetoshi Sano, Kozo Nakai","doi":"","DOIUrl":"","url":null,"abstract":"<p><p>Acquired circumscribed hyperpigmented patches and plaques have various differential diagnoses, including post-inflammatory hyperpigmentation and mycosis fungoides (MF). Leukomelanoderma is an uncommon cutaneous condition in which the pathogenesis is not fully elucidated. It has been reported that leukomelanoderma occurs after allergic contact dermatitis from hydroquinone or acute cutaneous graft-versus-host disease (1,2). Hyperpigmented MF is a cutaneous T-cell lymphoma with a frequent CD8+ phenotype (3). Herein, we report a case of leukomelanoderma clinically and histologically resembling hyperpigmented MF. A 55-year-old Japanese woman was referred to our department for evaluation of reticulate pigmentation with pruritic erythema on the face. She had used commercially available depigmenting cosmetic reagents for 20 years and ointment containing 10% hydroquinone for 3 months. Physical examination revealed diffuse hyperpigmentation and demarcated hypopigmented macules on the face and neck (Figure 1, a). Dermoscopy showed depigmented spots and reticulated plus dotted hyperpigmentation; it presented a pseudo-pigment network (Figure 1, b). Histological examination of a tissue specimen biopsied from the lesion showed superficial band-like lymphocytic infiltration in dermis accompanying single cells or small clusters in epidermis (Figure 1, c). Interface changes were observed together with melanophages in the dermis. Melan-A-positive melanocytes were absent. Immunohistochemical analysis demonstrated that the epidermotropic lymphocytes were CD3+CD7-, and they had predominance of CD8+ cells (Figure 1, d). These immunohistochemical results mimicked MF. However, PCR analysis of the T-cell receptor g-gene rearrangement was negative. Closed patch test result with hydroquinone (5% pet.) was graded D2 (+?) and D3 (+). Ten months after discontinuing cosmetic reagents and hydroquinone, the pigmentary changes showed improvement. The pathomechanism of leukomelanoderma is unclear. Although post-inflammatory pigmentation due to allergic or contact dermatitis together with direct depigmenting effects from hydroquinone use has been suggested (1), the immunophenotype of T-cells has not been examined. As observed in our patient, interface changes with melanophages, in addition to frequent CD8+ phenotype of the epidermotropism and dermal infiltrate of lymphocytes, were characteristic for hyperpigmented MF (3). Moreover, minimal CD7 expression was a specific finding for MF (4). T-cell receptor clonality was negative in our patient, but the clonality appears to be detected by PCR in up to 50% of the patients with early MF (3). In contrast, the closed patch test was positive for hydroquinone in our patient, and it is reported that CD8+ T-cells are recruited to the interphase between the epidermis and the dermis of the patients with allergic contact dermatitis (5). CD8+ T-cells might contribute to acute cutaneous graft-versus-host disease-like interface changes and des","PeriodicalId":94367,"journal":{"name":"Acta dermatovenerologica Croatica : ADC","volume":"32 1","pages":"73-74"},"PeriodicalIF":0.0,"publicationDate":"2024-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141474359","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}