Kaveri Rusia, V. Saoji, Bhushan Madke, Adarshlata Singh
{"title":"Lucio Phenomenon: An Unusual Case of Skin Necrosis","authors":"Kaveri Rusia, V. Saoji, Bhushan Madke, Adarshlata Singh","doi":"10.1159/000536370","DOIUrl":"https://doi.org/10.1159/000536370","url":null,"abstract":"Abstract Introduction Lucio leprosy is a non-nodular diffuse type of lepromatous leprosy first described by Lucio and Alvarado. Lucio phenomenon is a rare vasculonecrotic reaction characterized by cutaneous necrosis with minimal constitutional features. Case Presentation We describe an unusual case of a 53-year-old man from Central India who had blisters, ulcers, and widespread erosions on his foot, forearms, and arms. The diagnosis of lepromatous leprosy with the Lucio phenomenon was established after thorough evaluation by clinical findings, histopathological findings, and slit-skin smear examination. Conclusion Lucio phenomenon is an uncommon cause of cutaneous infarction and necrosis. Primary care physicians should keep a high index of suspicion in patients with cutaneous necrosis and minimal constitution features. Since leprosy is a relatively curable disease, primary care physicians should think of a rare form of lepromatous leprosy presenting with cutaneous necrosis, especially in non-endemic zones.","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"30 5","pages":"47 - 54"},"PeriodicalIF":0.9,"publicationDate":"2024-02-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139957315","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}
Katya Kovacheva, Zornitza Kamburova, Preslav Vasilev, I. Yordanova
{"title":"Netherton Syndrome with a Novel Likely Pathogenic Variant c.420del (p.Ser141ProfsTer5) in SPINK5 Gene: A Case Report","authors":"Katya Kovacheva, Zornitza Kamburova, Preslav Vasilev, I. Yordanova","doi":"10.1159/000536083","DOIUrl":"https://doi.org/10.1159/000536083","url":null,"abstract":"Abstract Introduction Netherton syndrome (NS) is a rare autosomal recessive genodermatosis in the group of congenital ichthyosis. The clinical manifestations of the syndrome vary from a very mild clinical manifestation occurring with the picture of ichthyosis linearis circumflexa to exfoliative erythroderma. It can be fatal in the first days of a newborn’s life due to dehydration, hypothermia, weight loss, respiratory infections, and sepsis. A specific anomaly of the hair trichorrexis invaginata is considered pathognomonic for the syndrome. Genetic testing of SPINK5 gene is key to confirming the diagnosis and starting early treatment. Case Presentation We present a case report of NS in a 6-year-old boy who suffered from generalized erythroderma and desquamation of the skin from birth. The patient has atopic diathesis, recurrent skin infections, increased levels of IgE, and delayed physical development. Two genetic variants in SPINK5 gene with clinical significance were identified. The first detected variant is a nonsense mutation, predicted to cause loss of normal protein function either by protein truncation or by nonsense-mediated mRNA decay. The second variant is a likely pathogenic frameshift mutation that truncates the protein in 5 amino acids. The child was treated with acitretin, without satisfactory effect. Conclusion The genetic variant we have described correlates with a severe clinical phenotype of NS. The second genetic variant of the SPINK5 gene, inherited from the father in our case, is novel and has never been published in the literature.","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"3 3","pages":"47 - 47"},"PeriodicalIF":0.9,"publicationDate":"2024-02-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139957489","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}
{"title":"Atezolizumab-Induced Acrodermatitis and Pustular Psoriasis in a Patient with Non-Small Cell Lung Cancer: A Rare Case Report.","authors":"Davide Fattore, Gianluca Esposito, Ludovica Carangelo, Maria Antonietta Luciano, Matteo Megna","doi":"10.1159/000535168","DOIUrl":"10.1159/000535168","url":null,"abstract":"<p><strong>Introduction: </strong>Immune checkpoint inhibitors are new drugs approved for the treatment of many types of malignancies. Despite their wide use and unquestionable clinical benefits, these agents have also been associated with a unique spectrum of side effects known as immune-related adverse events. In this study, we report the first case of atezolizumab-induced pustular psoriasis and acrodermatitis.</p><p><strong>Case presentation: </strong>A 61-year-old woman presented to our department with erythematous-desquamative and pustular lesions involving all hands and feet fingers, inguinal region, and trunk, associated to severe psoriatic onychodystrophy. She was affected by non-small-cell lung carcinoma from 12 years, and 7 months before admission, she started a treatment with atezolizumab.</p><p><strong>Conclusion: </strong>Immune checkpoint inhibitors such as atezolizumab are linked to a plethora of adverse events. Identifying and treating certain adverse skin events, particularly in cancer patients, can be a challenge, leading oncologists to discontinue immunotherapy. Our case shows how it is necessary to have a shared therapeutic algorithm in order to manage serious skin reactions in cancer patients and avoid disruption of the oncotherapy.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"16 1","pages":"42-46"},"PeriodicalIF":0.9,"publicationDate":"2024-02-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10881193/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139930217","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}
{"title":"This “mite” Surprise You: Scabies Masquerading as Langerhans Cell Histiocytosis – A Case Report","authors":"Sara Al Janahi, Raghda Al Maashari, Tausif Saleem","doi":"10.1159/000536369","DOIUrl":"https://doi.org/10.1159/000536369","url":null,"abstract":"Abstract Introduction Scabies is a common parasitic infestation caused by the mite Sarcoptes scabiei. Scabies can mimic other entities clinically, resulting in misdiagnosis. The presence of a mite in the stratum corneum on biopsy specimens is diagnostic of scabies. However, there are instances when mites are not visible, and immunohistochemical (IHC) staining may be misleading. An example is when IHC demonstrates Cluster of Differentiation 1a and S100 positivity. The main differential diagnosis for this finding is Langerhans cell histiocytosis, a group of idiopathic disorders of bone marrow-derived Langerhans cells, with manifestations ranging from isolated to life-threatening multisystem disease. Case Presentation We present a case of a patient who was diagnosed with Langerhans cell histiocytosis based on histological findings, further review with a repeat reading and deeper sectioning of her biopsy revealed a mite in the stratum corneum, altering the diagnosis, course, and management. She subsequently developed persistent post-scabietic nodules, an underreported entity that may occur following infestation and persist for up to a year. These lesions are self-limiting and do not require repeated courses of treatment. Conclusion Langerhans cell hyperplasia may be seen in a multitude of entities, including scabies. Familiarity with this phenomenon is crucial to avoid unnecessary invasive investigations, aggressive management and alleviate patients’ concerns.","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"279 1","pages":"35 - 41"},"PeriodicalIF":0.9,"publicationDate":"2024-02-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139833508","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}
A. Pour Mohammad, Elahe Noroozi, M. Gholizadeh Mesgarha, N. Shayanfar, A. Goodarzi
{"title":"Interesting Mucocutaneous Manifestations in COVID-19 Infection or Vaccination Confirmed by Histopathology: A Case Series","authors":"A. Pour Mohammad, Elahe Noroozi, M. Gholizadeh Mesgarha, N. Shayanfar, A. Goodarzi","doi":"10.1159/000535739","DOIUrl":"https://doi.org/10.1159/000535739","url":null,"abstract":"Introduction: Mucocutaneous complications or adverse events due to SARS-CoV-2 infection or vaccination have been well delineated in the literature, respectively. Most eruptions are considered mild and self-limiting; however, for the atypical cases with a tentative clinical diagnosis, performing a biopsy and histopathological assessment is pivotal to confirm the diagnosis and subsequently prescribe a more tailored treatment. Despite the diverse reporting of such incidents globally, most studies restrict the rate of biopsied cases to less than 15%. Case Presentations: This case series elucidates 20 patients referred to the tertiary dermatology clinic, including 14 COVID-19 infection-related eruptions such as lichen planus (LP), cutaneous vasculitis, pityriasis rosea (PR), discoid lupus erythematosus, guttate psoriasis, sarcoidosis, Raynaud’s phenomenon, non-specific lesions resembling genital warts, Beau’s line, and one severe case of purpura fulminans with a promising outcome. Moreover, we presented six vaccine-induced cases comprising LP, urticarial vasculitis, PR, parapsoriasis, and localized morphea. The diagnosis of all challenging cases has been proven by histopathological evaluation. We included pertaining anamnesis details of each patient and vivid classifying images to pinpoint the morphologic features of each condition. Discussion: In line with our previous studies, the vaccine-induced eruptions were less severe compared to infection-related complications of COVID-19 and are mostly controllable by antihistamines and corticosteroid administration. Therefore, reporting such events should not impede COVID-19 vaccination in the general population.","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":" 3","pages":""},"PeriodicalIF":0.9,"publicationDate":"2024-01-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139624733","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}
{"title":"Dermoscopic Features of Heterotopic Salivary Gland Tissue in the Neck: Report of a Case.","authors":"Shinichiro Inoue, Takuya Maeda, Yuri Nagata, Teruki Yanagi, Hideyuki Ujiie","doi":"10.1159/000535740","DOIUrl":"10.1159/000535740","url":null,"abstract":"<p><strong>Introduction: </strong>Heterotopic salivary gland tissue (HSGT) is diagnosed when salivary gland tissue is found in areas other than the major or minor salivary glands. No dermoscopic findings of HSGT have been reported.</p><p><strong>Case presentation: </strong>This case report gives the dermoscopic findings of a 45-year-old woman with HSGT who presented with a longstanding pale red macule on her neck, characterized by repeated clear fluid discharge. Dermoscopic examination showed a red dot with a pale pink halo and telangiectasia throughout the lesion. An excisional biopsy confirmed the diagnosis of HSGT, revealing the presence of mucous and serous salivary glandular structures within subcutaneous fat tissue. It has been reported that HSGT is associated with periductal lymphocytic infiltration, which was also observed in this case.</p><p><strong>Conclusion: </strong>We think that the \"red dot with a pale pink halo\" is a unique dermoscopic finding of HSGT, which is rarely observed in branchial anomalies.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"16 1","pages":"17-20"},"PeriodicalIF":0.9,"publicationDate":"2024-01-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10776130/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139401944","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}
Saad Khayat, Nuria Bonsfills, Raúl Antúnez-Conde, Sara Álvarez-Mokthari, Hubert Aranibar, Manuel Tousidonis, Mario Fernández-Fernández, Alberto Díez-Montiel
{"title":"Parascapular Flap for Severe Hidradenitis Suppurativa.","authors":"Saad Khayat, Nuria Bonsfills, Raúl Antúnez-Conde, Sara Álvarez-Mokthari, Hubert Aranibar, Manuel Tousidonis, Mario Fernández-Fernández, Alberto Díez-Montiel","doi":"10.1159/000533387","DOIUrl":"10.1159/000533387","url":null,"abstract":"<p><p>Hidradenitis suppurativa is a chronic inflammatory disease which affects apocrine glands and hair follicles of the skin, primarily in the axillary and groin regions. This condition can be highly debilitating, causing painful lesions and a negative psychological impact on patients. While medical and minimally invasive treatments are available, surgical intervention may be necessary for severe cases. In cases involving axillary defects, the use of local flaps such as the parascapular flap is a viable option. In this case report, we present a 34-year-old woman who presented to our clinic with a history of recurrent abscesses and cutaneous infections in the axillary region. After thorough evaluation, we chose to use the parascapular flap for reconstruction. The parascapular flap is a one-stage procedure that allows for extensive resection of the axillary area without resulting in contractions or retractions over the long term. Additionally, this technique allows for preservation of the axilla's original shape with minimal donor site morbidity.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"16 1","pages":"8-16"},"PeriodicalIF":0.9,"publicationDate":"2024-01-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10769502/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139377235","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}
Enrico Bocchino, Simone Cappilli, Gerardo Palmisano, Andrea Paradisi, Alfredo Piccerillo, Alessandro Di Stefani, Ketty Peris
{"title":"Real-Life Experience with Sonidegib for Locally Advanced Basosquamous Carcinoma: A Case Series.","authors":"Enrico Bocchino, Simone Cappilli, Gerardo Palmisano, Andrea Paradisi, Alfredo Piccerillo, Alessandro Di Stefani, Ketty Peris","doi":"10.1159/000535506","DOIUrl":"10.1159/000535506","url":null,"abstract":"<p><strong>Introduction: </strong>Basosquamous carcinoma is an uncommon subtype of basal cell carcinoma (BCC), characterized by aggressive local growth and metastatic potential, that mainly develops on the nose, perinasal area, and ears, representing 1.2-2.7% of all head-neck keratinocyte carcinomas. Although systemic therapy with hedgehog inhibitors (HHIs) represents the first-line medical treatment in advanced BCC, to date, no standard therapy for advanced basosquamous carcinoma has been established. Herein, we reported a case series of patients affected by locally advanced basosquamous carcinomas, who were treated with HHIs.</p><p><strong>Case presentation: </strong>Data of 5 patients receiving HHIs for locally advanced basosquamous carcinomas were retrieved (2 women and 3 males, age range: 63-89 years, average age of 77 years). Skin lesions were located on the head-neck area; in particular, 4 tumors involved orbital and periorbital area and 1 tumor developed in the retro-auricular region. A clinical response was obtained in 3 out of 5 patients (2 partial responses and 1 complete response), while disease progression was observed in the remaining 2 patients. Hence, therapy was interrupted, switching to surgery or immunotherapy.</p><p><strong>Conclusion: </strong>Increasing evidence suggests considering HHIs for large skin tumors developing in functionally and cosmetically sensitive areas, in patients with multiple comorbidities, although their use for basosquamous carcinoma require more exploration, large cohort populations, and long follow-up assessment.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"16 1","pages":"1-7"},"PeriodicalIF":0.9,"publicationDate":"2024-01-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10764081/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139097397","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}
{"title":"Diagnosis, Management, and Outcome of Bart's Syndrome Observed in a Sub-Saharan African Country (Senegal, Dakar): 2 Case Reports.","authors":"Mame Tènè Ndiaye Diop, Maïmouna Bassoum, Khadim Diop, Yaye Diood Dieng, Birame Seck, Fatou Diasse Fall, Charles Tchibinda Delicat, Alassane Ndiaye, Assane Diop, Maodo Ndiaye, Pape Moctar Faye, Moussa Diallo, Ousmane Ndiaye, Fatimata Ly, Suzanne Oumou Niang","doi":"10.1159/000535038","DOIUrl":"10.1159/000535038","url":null,"abstract":"<p><strong>Introduction: </strong>Bart's syndrome is an uncommon inherited congenital disorder associating congenital cutaneous aplasia of the extremities and inherited epidermolysis bullosa. Bilateral and symmetrical involvement of the limbs is exceptionally described on black skin. In most cases, the diagnosis is clinical; however, the management remains very difficult and the extended forms are a real therapeutic challenge. We report 2 cases of Bart's syndrome observed in a sub-Saharan African country (Senegal, Dakar).</p><p><strong>Case presentation: </strong>It was about 2 premature female and male newborns. On physical examination, the girl presented with a total absence of skin on the limbs, associated with cutaneous detachment of the trunk representing a detached and detachable skin surface of 46%; the boy underwent a total absence of skin of more than 50% of the skin surface. The diagnosis of Bart's syndrome was set based on the typical clinical aspect. The blood count and CRP were normal for the girl whereas it revealed some disorders for the boy. The 2 newborns were urgently admitted to an incubator, and the intensive care was started with hyperhydration, anti-staphylococcal prophylaxis, and daily dermatological care with antiseptic baths and fatty dressings.</p><p><strong>Conclusion: </strong>Bart's syndrome is an uncommon genodermatosis characterized by a clinical triad associating congenital cutaneous aplasia of the extremities, inherited epidermolysis bullosa suspected in the presence of bubbles, and areas of cutaneous fragility and nail deformity. All types of which can be associated with this syndrome. The easy clinical diagnosis but the difficult management encumber the vital prognosis of our cases.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"15 1","pages":"225-230"},"PeriodicalIF":0.9,"publicationDate":"2023-12-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10695655/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138486817","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}
Terenzio Cosio, Rosalba Petruccelli, Roberta Gaziano, Carla Fontana, Marco Favaro, Paola Zampini, Enrico Salvatore Pistoia, Laura Diluvio, Flavia Lozzi, Luca Bianchi, Elena Campione
{"title":"Green Nail Syndrome Treated with Ozenoxacin: Two Case Reports.","authors":"Terenzio Cosio, Rosalba Petruccelli, Roberta Gaziano, Carla Fontana, Marco Favaro, Paola Zampini, Enrico Salvatore Pistoia, Laura Diluvio, Flavia Lozzi, Luca Bianchi, Elena Campione","doi":"10.1159/000533923","DOIUrl":"10.1159/000533923","url":null,"abstract":"<p><p>Green nail syndrome (GNS) is a persistent greenish pigmentation of the nail plate, originally described in 1944 by Goldman and Fox, due to <i>Pseudomonas aeruginosa</i> infection. Recently, pulmonary co-infection of <i>P. aeruginosa</i> and <i>Achromobacter</i> spp. has been described in patients with cystic fibrosis. <i>Achromobacter xylosoxidans</i> is a multidrug-resistant (MDR) pathogen involved in lung and soft tissue skin infections. Both <i>Achromobacter xylosoxidans</i> and <i>P. aeruginosa</i> are mainly found in humid environments or in water. There are no recognized co-infections due to <i>P. aeruginosa</i> and <i>A. xylosoxidans</i> in the skin and appendages. We describe two cases of GNS, the first due to <i>P. aeruginosa</i> associated with <i>Achromobacter xylosoxidans</i>; the other due to MDR <i>P. aeruginosa</i>, both successfully treated with topical ozenoxacin 1% cream daily for 12 weeks. The clinical management of GNS can be confusing, especially when the bacterial culture result is inconsistent or when non-<i>Pseudomonas</i> bacteria are isolated. In our case, due to the co-infection of <i>P. aeruginosa</i> and <i>Achromobacter</i> spp., local treatment with ozenoxacin - the first nonfluorinated quinolone - could be a safe and effective treatment in case of MDR nail infections. Further studies are required to evaluate clinical isolation from nail infections and the co-presence of <i>P. aeruginosa</i> and <i>A. xylosoxidans</i>.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"15 1","pages":"217-224"},"PeriodicalIF":0.9,"publicationDate":"2023-11-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10653707/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138458122","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}