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Ichthyosis linearis circumflexa

MedGen UID:
78578
Concept ID:
C0265962
Disease or Syndrome
Synonym: ichthyosis linearis circumflexa
SNOMED CT: Ichthyosis linearis circumflexa (54336006)
 
Monarch Initiative: MONDO:0043106

Definition

Netherton syndrome is a disorder that affects the skin, hair, and immune system. Newborns with Netherton syndrome have skin that is red and scaly (ichthyosiform erythroderma), and the skin may leak fluid. Some affected infants are born with a tight, clear sheath covering their skin called a collodion membrane. This membrane is usually shed during the first few weeks of life. Because newborns with this disorder are missing the protection provided by normal skin, they are at risk of becoming dehydrated and developing infections in the skin or throughout the body (sepsis), which can be life-threatening. Affected babies may also fail to grow and gain weight at the expected rate (failure to thrive). The health of older children and adults with Netherton syndrome usually improves, although they often remain underweight and of short stature.

After infancy, the severity of the skin abnormalities varies among people with Netherton syndrome and can fluctuate over time. The skin may continue to be red and scaly, especially during the first few years of life. Some affected individuals have intermittent redness or experience outbreaks of a distinctive skin abnormality called ichthyosis linearis circumflexa, involving patches of multiple ring-like lesions. The triggers for the outbreaks are not known, but researchers suggest that stress or infections may be involved.

Itchiness is a common problem for affected individuals, and scratching can lead to frequent infections. Dead skin cells are shed at an abnormal rate and often accumulate in the ear canals, which can affect hearing if not removed regularly. The skin is abnormally absorbent of substances such as lotions and ointments, which can result in excessive blood levels of some topical medications. Because the ability of the skin to protect against heat and cold is impaired, affected individuals may have difficulty regulating their body temperature.

People with Netherton syndrome have hair that is fragile and breaks easily. Some strands of hair vary in diameter, with thicker and thinner spots. This feature is known as bamboo hair, trichorrhexis nodosa, or trichorrhexis invaginata. In addition to the hair on the scalp, the eyelashes and eyebrows may be affected. The hair abnormality in Netherton syndrome may not be noticed in infancy because babies often have sparse hair.

Most people with Netherton syndrome have immune system-related problems such as food allergies, hay fever, asthma, or an inflammatory skin disorder called eczema. [from MedlinePlus Genetics]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVIchthyosis linearis circumflexa
Follow this link to review classifications for Ichthyosis linearis circumflexa in Orphanet.

Professional guidelines

PubMed

Gan C, King E, Orchard D
Australas J Dermatol 2022 Aug;63(3):365-367. Epub 2022 May 27 doi: 10.1111/ajd.13880. PMID: 35622930
Nouwen AEM, Schappin R, Nguyen NT, Ragamin A, Bygum A, Bodemer C, Dalm VASH, Pasmans SGMA
Front Immunol 2022;13:864449. Epub 2022 Mar 30 doi: 10.3389/fimmu.2022.864449. PMID: 35464459Free PMC Article
DiGiovanna JJ, Robinson-Bostom L
Am J Clin Dermatol 2003;4(2):81-95. doi: 10.2165/00128071-200304020-00002. PMID: 12553849

Recent clinical studies

Etiology

Ennouri M, Zimmer AD, Bahloul E, Chaabouni R, Marrakchi S, Turki H, Fakhfakh F, Bougacha-Elleuch N, Fischer J
BMC Med Genomics 2022 Jan 5;15(1):4. doi: 10.1186/s12920-021-01154-z. PMID: 34983512Free PMC Article
Godic A, Dragos V
Eur J Dermatol 2004 Mar-Apr;14(2):115-7. PMID: 15197002
DiGiovanna JJ, Robinson-Bostom L
Am J Clin Dermatol 2003;4(2):81-95. doi: 10.2165/00128071-200304020-00002. PMID: 12553849
Milstone LM, Ellison AF, Insogna KL
Arch Dermatol 1992 Jul;128(7):926-30. PMID: 1320849
Happle R, van de Kerkhof PC, Traupe H
Dermatologica 1987;175 Suppl 1:107-24. doi: 10.1159/000248867. PMID: 2961628

Diagnosis

Schepis C, Siragusa M, Centofanti A, Vinci M, Calì F
Dermatol Online J 2019 Jul 15;25(7) PMID: 31450277
De Niear MA, Gigante J
J Pediatr 2018 Jan;192:262-262.e1. Epub 2017 Nov 6 doi: 10.1016/j.jpeds.2017.09.022. PMID: 29106927
Bittencourt Mde J, Moure ER, Pies OT, Mendes AD, Deprá MM, Mello AL
An Bras Dermatol 2015 Jan-Feb;90(1):114-6. doi: 10.1590/abd1806-4841.20153011. PMID: 25672309Free PMC Article
Meltem Akkurt Z, Tuncel T, Ayhan E, Uçmak D, Uluca U, Uçak H
J Cutan Med Surg 2014 Jul-Aug;18(4):280-2. doi: 10.2310/7750.2013.13106. PMID: 25008447
DiGiovanna JJ, Robinson-Bostom L
Am J Clin Dermatol 2003;4(2):81-95. doi: 10.2165/00128071-200304020-00002. PMID: 12553849

Therapy

Dev T, Raman Kumar M, Sethuraman G
BMJ Case Rep 2017 May 28;2017 doi: 10.1136/bcr-2017-220227. PMID: 28554884Free PMC Article
Kumar P, Gharami RC, Mondal A, Ghosh K
Indian Pediatr 2010 Dec;47(12):1052. PMID: 21220804
Godic A, Dragos V
Eur J Dermatol 2004 Mar-Apr;14(2):115-7. PMID: 15197002
DiGiovanna JJ, Robinson-Bostom L
Am J Clin Dermatol 2003;4(2):81-95. doi: 10.2165/00128071-200304020-00002. PMID: 12553849
Lazarous MC, Kerdel FA
Drugs Today (Barc) 2002 Jan;38(1):7-15. PMID: 12532181

Prognosis

Özkaya E, Günay MB, Babuna Kobaner G, Keskinkaya Z, Gökalp MO
Australas J Dermatol 2023 May;64(2):272-277. Epub 2023 Feb 6 doi: 10.1111/ajd.13986. PMID: 36745433
Takahashi M, Hagiya H, Tanaka S, Yamamoto K, Honda H, Hasegawa K, Otsuka F
J Infect Chemother 2022 Jul;28(7):978-981. Epub 2022 Mar 8 doi: 10.1016/j.jiac.2022.02.021. PMID: 35277342
DiGiovanna JJ, Robinson-Bostom L
Am J Clin Dermatol 2003;4(2):81-95. doi: 10.2165/00128071-200304020-00002. PMID: 12553849
Bitoun E, Chavanas S, Irvine AD, Lonie L, Bodemer C, Paradisi M, Hamel-Teillac D, Ansai S, Mitsuhashi Y, Taïeb A, de Prost Y, Zambruno G, Harper JI, Hovnanian A
J Invest Dermatol 2002 Feb;118(2):352-61. doi: 10.1046/j.1523-1747.2002.01603.x. PMID: 11841556
Happle R, van de Kerkhof PC, Traupe H
Dermatologica 1987;175 Suppl 1:107-24. doi: 10.1159/000248867. PMID: 2961628

Clinical prediction guides

Nouwen AEM, Schappin R, Nguyen NT, Ragamin A, Bygum A, Bodemer C, Dalm VASH, Pasmans SGMA
Front Immunol 2022;13:864449. Epub 2022 Mar 30 doi: 10.3389/fimmu.2022.864449. PMID: 35464459Free PMC Article
Zhang Z, Pan C, Wei R, Li H, Yang Y, Chen J, Li M, Yao Z
Mol Genet Genomic Med 2021 Mar;9(3):e1600. Epub 2021 Jan 16 doi: 10.1002/mgg3.1600. PMID: 33452875Free PMC Article
Bitoun E, Chavanas S, Irvine AD, Lonie L, Bodemer C, Paradisi M, Hamel-Teillac D, Ansai S, Mitsuhashi Y, Taïeb A, de Prost Y, Zambruno G, Harper JI, Hovnanian A
J Invest Dermatol 2002 Feb;118(2):352-61. doi: 10.1046/j.1523-1747.2002.01603.x. PMID: 11841556
Milstone LM, Ellison AF, Insogna KL
Arch Dermatol 1992 Jul;128(7):926-30. PMID: 1320849

Recent systematic reviews

Nouwen AEM, Schappin R, Nguyen NT, Ragamin A, Bygum A, Bodemer C, Dalm VASH, Pasmans SGMA
Front Immunol 2022;13:864449. Epub 2022 Mar 30 doi: 10.3389/fimmu.2022.864449. PMID: 35464459Free PMC Article

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