Oleogranulomatous response in lymph nodes associated with emollient use in Netherton's syndrome

Cockayne,; Lee, Myeong Soo; Harrington,

doi:10.1046/j.1365-2133.1999.03061.x

Cited by 9 publications

(4 citation statements)

References 9 publications

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“…After changing to a sole skin care treatment findings normalized completely. Increased transcutaneous absorption of paraffin‐based emollients and topical tacrolimus in NS has recently been reported 8,9 . Systemic administration of retinoids is discussed controversially in the literature, but might be beneficial in older age and particularly in milder forms with ILC 5,10 …”

Section: Discussionmentioning

confidence: 99%

Genetic analysis of a severe case of Netherton syndrome and application for prenatal testing

Haußer

Berg

et al. 2002

Br J Dermatol

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Netherton syndrome (NS) is a rare autosomal recessive disease with variable expression. It is defined by a triad of symptoms: congenital ichthyosiform erythroderma, trichorrhexis invaginata and atopy. Recently, genetic linkage has been established to the SPINK5 gene locus on chromosome 5q32 encoding the serine protease inhibitor LEKTI. In this study, we present a recurrent homozygous mononucleotide deletion (153delT) resulting in a severe case of NS exhibiting exfoliative erythroderma with lethal outcome at the age of 4 months and its application in prenatal testing in a subsequent pregnancy of the mother.

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Section: Discussionmentioning

confidence: 99%

Genetic analysis of a severe case of Netherton syndrome and application for prenatal testing

Haußer

Berg

et al. 2002

Br J Dermatol

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“…reactions to ingredients of topical creams can be observed and are facilitated by the skin barrier defect. For example, excessive use of emollients containing liquid paraffin can lead to penetration of paraffin into the skin, resulting in lymphadenopathy [77].…”

Section: Emollients Moisturizers Keratolyticsmentioning

confidence: 99%

Advances in understanding of Netherton syndrome and therapeutic implications

Petrova

Hovnanian

2020

Expert Opinion on Orphan Drugs

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Introduction: Netherton syndrome (NS) is a rare and severe ichthyosis characterized by superficial scaling, skin inflammation, a specific hair shaft defect, severe atopic manifestations and multisystemic complications. It is an orphan disease with currently no satisfactory treatment. NS is caused by loss-offunction mutations in SPINK5 encoding the serine protease inhibitor LEKTI. NS patients present with ichthyosiform erythroderma or ichthyosis linearis circumflexa and show considerable clinical variability. Areas covered: Uncontrolled serine protease activity leads to a profound skin barrier defect and the release of pro-inflammatory and pro-allergic mediators by keratinocytes and immune cells. Improved understanding of NS pathogenesis has led to the successful use of repurposed biologics such as intravenous immunoglobulins and anti-IL-17A blockers. Between April 1, 2020 and November 18, 2020, authors searched for NS-relevant information in the following databases: MEDLINE, DrugBank, ClinicalTrials.gov, and patent datasets accessed through lens.org. Expert opinion: Specific KLK5 and/or KLK7 inhibitors represent the most promising disease-modifying treatments. They are currently being developed by several companies. Comprehension of the determinants of NS variability, flares and modification over time will be the foundation for precision medicine. While improved knowledge of the inflammatory and allergic pathways involved is still needed, clinical trials using repurposed biologics have already begun.

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“…7 Mesenteric lymph node involvement is also well described and is often an incidental finding on routine autopsy in developed countries. 8 This is thought to be a result of dietary intake of mineral oil containing waxes used on fruit, vegetables, and meat, in addition to laxative use. 8 The most common sequelae is the production of chronic fibrotic reactions surrounding the mineral oil deposits that may be mistaken for a malignant neoplasm.…”

Section: Discussionmentioning

confidence: 99%

“…8 The most common sequelae is the production of chronic fibrotic reactions surrounding the mineral oil deposits that may be mistaken for a malignant neoplasm. 4,6,9,10 The ability of mineral oils to traverse fascial planes 10,11 and spread along the lymphatic system 8,11 allows further migration to other sites. Indeed, it is reasonable to speculate that disruption of lymphatic channels allowing chyle leakage into the tissues may similarly lead to an oleogranulomatous reaction.…”

Section: Discussionmentioning

confidence: 99%

Peritoneal Oleogranulomatosis After Laparoscopic Gastric Banding

Shen

Sharard³

et al. 2011

Surgical Laparoscopy, Endoscopy &Amp; Percutaneous Techniques

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Laparoscopic adjustable gastric banding is one of the most frequently performed bariatric operations worldwide owing to its low incidence of iatrogenic complications. Peritoneal oleogranulomatosis, a sequelae of mineral oil contact with tissues, has not been reported in the context of bariatric surgery. We present the case of a 52-year-old woman with an incidental finding of peritoneal oleogranulomatosis at laparoscopic cholecystectomy, who had previously undergone laparoscopic gastric banding. A potential mechanism linking the two is postulated and the importance of histological diagnosis highlighted, given the macroscopic resemblance to carcinomatosis peritonei.

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Oleogranulomatous response in lymph nodes associated with emollient use in Netherton's syndrome

Abstract: Paraffin-based emollients are widely used in dermatological practice and are not usually absorbed through the skin. We report a case where transcutaneous transfer did occur in the context of damaged skin in Netherton's syndrome, resulting in a reversible lymphadenopathy.

Cited by 9 publications

References 9 publications

Genetic analysis of a severe case of Netherton syndrome and application for prenatal testing

Genetic analysis of a severe case of Netherton syndrome and application for prenatal testing

Advances in understanding of Netherton syndrome and therapeutic implications

Peritoneal Oleogranulomatosis After Laparoscopic Gastric Banding

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