Netherton syndrome (NS) is a rare autosomal recessive genodermatosis. In this article, we present two siblings with NS who harbour a novel variant in the SPINK5 gene and were treated with infliximab infusions. Both patients exhibited the characteristic clinical triad of NS, and their whole exome sequencing analysis revealed a homozygous variant, c.1820+53G>A, in the SPINK5 gene. Notably, this is the first documented instance of homozygosity for this particular variant. Despite the absence of a specific treatment, both patients achieved total clearance of the skin lesions, and a significant decrease in total IgE levels was documented.
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http://dx.doi.org/10.1111/ajd.14234 | DOI Listing |
Acta Dermatovenerol Alp Pannonica Adriat
December 2024
Department of Biochemistry, Faculty of Medicine and Health Sciences, Christian University of Krida Wacana, Jakarta, Indonesia.
Kallikrein proteases (KPs) are vital enzymes involved in the formation of dermatosomes and are regulated by the body's internal inhibitors. Maintaining a balance between KPs and their inhibitors is essential for promoting a healthy scalp. The scalp specifically contains two KPs: human kallikrein (hK) 5 and hK7, which are encoded by their respective genes.
View Article and Find Full Text PDFDiscov Oncol
November 2024
Department of Stomatology, Taizhou Central Hospital (Taizhou University Hospital), No.999, Donghai Avenue, Taizhou, 318000, Zhejiang, People's Republic of China.
Orphanet J Rare Dis
November 2024
Department of Dermatology, Maastricht University Medical Centre +, Maastricht, The Netherlands.
Background: The Mendelian Disorders of Cornification (MeDOC) comprise a large number of disorders that present with either localised (palmoplantar keratoderma, PPK) or generalised (ichthyoses) signs. The MeDOC are highly heterogenic in terms of genetics and phenotype. Consequently, diagnostic process is challenging and before implementation of the next generation sequencing, was mostly symptomatic, not causal, which limited research on those diseases.
View Article and Find Full Text PDFAllergol Select
October 2024
Center for Child and Adolescent Health, Helios Hospital Krefeld, Academic Hospital of RWTH Aachen, Krefeld.
Anal Cell Pathol (Amst)
October 2024
Research Center for Molecular Medicine, Hamadan University of Medical Sciences, Hamadan, Iran.
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