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SAGE Open Medical Case Reports 2023Gianotti-Crosti syndrome, also known as papular acrodermatitis of childhood, is a common, self-limiting dermatosis often seen in children with triggers including viral...
Gianotti-Crosti syndrome, also known as papular acrodermatitis of childhood, is a common, self-limiting dermatosis often seen in children with triggers including viral and bacterial infections along with immunizations. Lesions are generally described as asymptomatic, skin colored to erythematous papules and papulovesicles that often spontaneously resolve within weeks. Here, we will discuss Gianotti-Crosti syndrome and present a rare case of chronic Gianotti-Crosti syndrome in an otherwise healthy 3-year-old male persisting for over 20 months. From this report, we aim to better educate the dermatologic community on the extremes of the Gianotti-Crosti syndrome disease course to improve diagnosis and treatment of symptomatic patients.
PubMed: 37077806
DOI: 10.1177/2050313X231164250 -
BMJ Case Reports Jun 2021A 3-month-old, full term female infant, adequate for gestational age, and exclusively breastfed, was admitted with a 10 day history of generalised scaling erythematous...
A 3-month-old, full term female infant, adequate for gestational age, and exclusively breastfed, was admitted with a 10 day history of generalised scaling erythematous dermatitis, affecting the face (perinasal, nasolabial folds and periauricular), acral and intertriginous areas, with irritability and failure to thrive. Her mother had been treated with isoniazid since the third trimester because of family contact with tuberculosis. Based on a diagnosis of suspected impetiginised eczema, the infant was treated with flucloxacillin and prednisolone, and maternal isoniazid was suspended, with no improvement. Investigations found low serum zinc levels in the infant (33 μg/dL; normal range (NR) >60 μg/dL), normal plasma zinc levels in the mother (111.3 μg/dL; NR 68-120 μg/dL) and lower than the normal range of zinc levels in breast milk (270μg/L; NR 1000-2500 μg/L), suggesting acrodermatitis caused by zinc deficiency. Oral zinc supplementation (3 mg/kg/day) was started with a marked improvement in skin lesions, as well as good weight gain. At the age of 6 months, after food diversification, supplementation was suspended, without any recurrence of symptoms.
Topics: Acrodermatitis; Breast Feeding; Female; Humans; Infant; Malnutrition; Milk, Human; Zinc
PubMed: 34193450
DOI: 10.1136/bcr-2021-241754 -
SAGE Open Medical Case Reports 2023Acrodermatitis continua of Hallopeau is a rare variant of localized pustular psoriasis characterized by the recurrent eruption of sterile pustules involving the distal...
Acrodermatitis continua of Hallopeau is a rare variant of localized pustular psoriasis characterized by the recurrent eruption of sterile pustules involving the distal portions of the fingers and toes that can lead to the destruction of the nail apparatus. Acrodermatitis continua of Hallopeau is a chronic, relapsing condition that is resistant to most topical and systemic psoriasis therapies, making it notoriously difficult to manage. Interleukin-36 and interleukin-17 are thought to play a pivotal role in the pathophysiology of pustular psoriasis, and evidence suggests that interleukin-17 inhibition can be an effective therapy for pustular psoriasis variants, including acrodermatitis continua of Hallopeau. Bimekizumab, a monoclonal antibody that inhibits the interleukin-17 pathway, may be a safe and effective treatment option for patients with acrodermatitis continua of Hallopeau. We present the first documented case of a patient with acrodermatitis continua of Hallopeau of the bilateral thumbnails who experienced an excellent response to bimekizumab treatment.
PubMed: 36968989
DOI: 10.1177/2050313X231160937 -
JAAD Case Reports Mar 2021
PubMed: 33665283
DOI: 10.1016/j.jdcr.2021.01.016 -
Dermatologic Therapy Nov 2021Since the emergence of the new coronavirus disease 19 (COVID-19) pandemic, there has been a concern for the patients with chronic autoimmune diseases including...
Since the emergence of the new coronavirus disease 19 (COVID-19) pandemic, there has been a concern for the patients with chronic autoimmune diseases including dermatological conditions over the potential exacerbation of these underlying conditions after infection with severe acute respiratory syndrome coronavirus-2 (SARS-CoV2). We performed a systematic review to evaluate presentations, postinfection change in the manifestation, diagnosis, and management of flare-ups of underlying dermatologic disease in patients with COVID-19. A total of 17 articles were recovered reporting on flare-ups of dermatological disease including pemphigus vulgaris, psoriasis, subacute cutaneous lupus erythematosus, acrodermatitis continua of Hallopeau, systemic sclerosis sine scleroderma, and Sézary syndrome (SS). Out of these, psoriasis and alopecia areata were the most common conditions. However, most cases of psoriasis could have been attributed to either antimalarial agents that were initially used for the treatment of COVID-19 or discontinuation of treatment following SARS-CoV2 infection.
Topics: COVID-19; Humans; Pandemics; Psoriasis; RNA, Viral; SARS-CoV-2
PubMed: 34453380
DOI: 10.1111/dth.15113 -
Frontiers in Immunology 2024Generalized pustular psoriasis (GPP) is a rare chronic inflammatory pustular dermatosis that presents as painful erythema with sterile pustules on nonacral skin. No...
Generalized pustular psoriasis (GPP) is a rare chronic inflammatory pustular dermatosis that presents as painful erythema with sterile pustules on nonacral skin. No unified standard and guideline for the treatment of GPP has been established. Several biologics have been tried for GPP, with varying success. Acrodermatitis continua of Hallopeau (ACH) is a very rare disabling variant of pustular psoriasis characterized by sterile pustules on the fingers and toes, including the nail bed. Comparatively, treating ACH is highly challenging due to its commonly therapy-resistant disease course. The pathogenic role of IL-36 signaling axis has been currently identified in GPP development. Spesolimab, the first anti-interleukin-36 receptor biologic, has been approved for treating GPP flares and shown promising results. In view of a shared pathogenesis between GPP and ACH, specolimab may be an effective treatment for ACH. Currently, there is no case and clinical trial data exist on this condition. Therefore, this case was aim to describe real-world experience of spesolimab use in ACH coexisting with GPP. We report an Asian patient with a 16-year-history of GPP and ACH with marked pustulosis on the nail bed and onychodystrophy. He received conventional systemic regimen acitretin, cyclosporine and biologics adalimumab and secukinumab, but experienced relapse for skin lesions and refractory for nail lesions. He was then treated with a single dose of spesolimab in combination with secukinumab, which resulted in skin clearance and nearly complete resolution of nail lesions over a 32-week period. Our observation suggests that spesolimab should be considered for the treatment of ACH, especially in the patients with intractable nail lesions and concomitant GPP.
Topics: Male; Humans; Acrodermatitis; Psoriasis; Skin; Biological Products; Antibodies, Monoclonal, Humanized
PubMed: 38464535
DOI: 10.3389/fimmu.2024.1338285 -
Journal of the American Academy of... Mar 2023
Topics: Humans; Mpox (monkeypox); Exanthema; Acrodermatitis; Blister; Emotions
PubMed: 36574862
DOI: 10.1016/j.jaad.2022.12.026 -
BMJ Case Reports Aug 2021
Topics: Acrodermatitis; Exanthema; Humans
PubMed: 34380692
DOI: 10.1136/bcr-2021-244988 -
American Journal of Human Genetics Sep 2020Generalized pustular psoriasis (GPP) is a severe multi-systemic inflammatory disease characterized by neutrophilic pustulosis and triggered by pro-inflammatory IL-36...
Generalized pustular psoriasis (GPP) is a severe multi-systemic inflammatory disease characterized by neutrophilic pustulosis and triggered by pro-inflammatory IL-36 cytokines in skin. While 19%-41% of affected individuals harbor bi-allelic mutations in IL36RN, the genetic cause is not known in most cases. To identify and characterize new pathways involved in the pathogenesis of GPP, we performed whole-exome sequencing in 31 individuals with GPP and demonstrated effects of mutations in MPO encoding the neutrophilic enzyme myeloperoxidase (MPO). We discovered eight MPO mutations resulting in MPO -deficiency in neutrophils and monocytes. MPO mutations, primarily those resulting in complete MPO deficiency, cumulatively associated with GPP (p = 1.85E-08; OR = 6.47). The number of mutant MPO alleles significantly differed between 82 affected individuals and >4,900 control subjects (p = 1.04E-09); this effect was stronger when including IL36RN mutations (1.48E-13) and correlated with a younger age of onset (p = 0.0018). The activity of four proteases, previously implicated as activating enzymes of IL-36 precursors, correlated with MPO deficiency. Phorbol-myristate-acetate-induced formation of neutrophil extracellular traps (NETs) was reduced in affected cells (p = 0.015), and phagocytosis assays in MPO-deficient mice and human cells revealed altered neutrophil function and impaired clearance of neutrophils by monocytes (efferocytosis) allowing prolonged neutrophil persistence in inflammatory skin. MPO mutations contribute significantly to GPP's pathogenesis. We implicate MPO as an inflammatory modulator in humans that regulates protease activity and NET formation and modifies efferocytosis. Our findings indicate possible implications for the application of MPO inhibitors in cardiovascular diseases. MPO and affected pathways represent attractive targets for inducing resolution of inflammation in neutrophil-mediated skin diseases.
Topics: Adult; Animals; Cytokines; Extracellular Traps; Female; Humans; Inflammation; Interleukin-1; Interleukins; Male; Mice; Mutation; Neutrophils; Peroxidase; Psoriasis; Rare Diseases; Skin; Skin Diseases
PubMed: 32758447
DOI: 10.1016/j.ajhg.2020.07.001 -
Clinical, Cosmetic and Investigational... 2023Acrodermatitis continua of Hallopeau (ACH) is a rare variant of pustular psoriasis that presents as a sterile pustular eruption of the periungual and subungual regions....
Acrodermatitis continua of Hallopeau (ACH) is a rare variant of pustular psoriasis that presents as a sterile pustular eruption of the periungual and subungual regions. It affects the skin and nail bed and can lead to distal phalangeal destruction as the disease progresses. ACH is an incurable disease that requires long-term maintenance therapy to prevent any complications. Because ACH is a variant of pustular psoriasis, it is commonly treated with anti-psoriatic therapies. Unfortunately, it is resistant to many available anti-psoriatic therapies, and there are no clinical guidelines for it; the treatment is therefore highly challenging. Current treatment strategies are mostly based on a few case reports and case series. In this study, we present a case of ACH in a 24-year-old man with a long history of severe skin lesions and overt onychodystrophy, successfully treated with Ustekinumab. In this patient, we observed rapid improvement in skin lesions and symptoms. Ustekinumab can considerably improve other symptoms besides plaque psoriasis. The treatment and excellent outcomes of Ustekinumab may provide clinical benefits to more patients and serve as a reference for other dermatologists.
PubMed: 37051587
DOI: 10.2147/CCID.S404860