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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]

Clinical features

From HPO
Failure to thrive
MedGen UID:
746019
Concept ID:
C2315100
Disease or Syndrome
Failure to thrive (FTT) refers to a child whose physical growth is substantially below the norm.
Intestinal atresia
MedGen UID:
7129
Concept ID:
C0021828
Disease or Syndrome
An abnormal closure, or atresia of the tubular structure of the intestine.
Villous atrophy
MedGen UID:
154306
Concept ID:
C0554101
Finding
The enteric villi are atrophic or absent.
Abnormal intestine morphology
MedGen UID:
1388201
Concept ID:
C4316788
Finding
An abnormality of the intestine. The closely related term enteropathy is used to refer to any disease of the intestine.
Global developmental delay
MedGen UID:
107838
Concept ID:
C0557874
Finding
A delay in the achievement of motor or mental milestones in the domains of development of a child, including motor skills, speech and language, cognitive skills, and social and emotional skills. This term should only be used to describe children younger than five years of age.
Abnormality of the musculature
MedGen UID:
867380
Concept ID:
C4021745
Anatomical Abnormality
Abnormality originating in one or more muscles, i.e., of the set of muscles of body.
Asthma
MedGen UID:
2109
Concept ID:
C0004096
Disease or Syndrome
Asthma is characterized by increased responsiveness of the tracheobronchial tree to multiple stimuli, leading to narrowing of the air passages with resultant dyspnea, cough, and wheezing.
Erythroderma
MedGen UID:
3767
Concept ID:
C0011606
Disease or Syndrome
An inflammatory exfoliative dermatosis involving nearly all of the surface of the skin. Erythroderma develops suddenly. A patchy erythema may generalize and spread to affect most of the skin. Scaling may appear in 2-6 days and be accompanied by hot, red, dry skin, malaise, and fever.
Increased circulating IgE level
MedGen UID:
116018
Concept ID:
C0236175
Finding
An abnormally increased overall level of immunoglobulin E in blood.
Recurrent infections
MedGen UID:
65998
Concept ID:
C0239998
Finding
Increased susceptibility to infections.
Hypereosinophilia
MedGen UID:
148170
Concept ID:
C0745091
Disease or Syndrome
A severely increased count of eosinophils in the blood defined as a blood eosinophil count of at least 1.5 billion cells per liter.
Allergic rhinitis
MedGen UID:
382012
Concept ID:
C2607914
Disease or Syndrome
It is characterized by one or more symptoms including sneezing, itching, nasal congestion, and rhinorrhea.
Food allergy
MedGen UID:
1635115
Concept ID:
C4554344
Disease or Syndrome
Primary food allergies primarily occur as a result (most likely) of gastrointestinal sensitization to predominantly stable food allergens (glycoproteins). A secondary food allergy develops after primary sensitization to airborne allergens (e. g., pollen allergens) with subsequent reactions (due to cross-reactivity) to structurally related often labile allergens in (plant) foods.
Decreased circulating IgG level
MedGen UID:
1720114
Concept ID:
C5234937
Finding
An abnormally decreased level of immunoglobulin G (IgG) in blood.
Hypernatremic dehydration
MedGen UID:
340564
Concept ID:
C1850544
Finding
Dehydration resulting from abnormally high levels of sodium in the blood. Infants and individuals that are intubated are at highest risk.
Sparse eyebrow
MedGen UID:
371332
Concept ID:
C1832446
Finding
Decreased density/number of eyebrow hairs.
Sparse scalp hair
MedGen UID:
346499
Concept ID:
C1857042
Finding
Decreased number of hairs per unit area of skin of the scalp.
Angioedema
MedGen UID:
1543
Concept ID:
C0002994
Pathologic Function
Rapid swelling (edema) of the dermis, subcutaneous tissue, mucosa and submucosal tissues of the skin of the face, normally around the mouth, and the mucosa of the mouth and/or throat, as well as the tongue during a period of minutes to several hours. The swelling can also occur elsewhere, typically in the hands. Angioedema is similar to urticaria, but the swelling is subcutaneous rather than on the epidermis.
Parakeratosis
MedGen UID:
10572
Concept ID:
C0030436
Disease or Syndrome
Abnormal formation of the keratinocytes of the epidermis characterized by persistence of nuclei, incomplete formation of keratin, and moistness and swelling of the keratinocytes.
Urticaria
MedGen UID:
22587
Concept ID:
C0042109
Disease or Syndrome
Raised, well-circumscribed areas of erythema and edema involving the dermis and epidermis. Urticaria is intensely pruritic, and blanches completely with pressure.
Congenital nonbullous ichthyosiform erythroderma
MedGen UID:
38180
Concept ID:
C0079154
Disease or Syndrome
The term collodion baby applies to newborns who appear to have an extra layer of skin (known as a collodion membrane) that has a collodion-like quality. It is a descriptive term, not a specific diagnosis or disorder (as such, it is a syndrome). Affected babies are born in a collodion membrane, a shiny waxy outer layer to the skin. This is shed 10-14 days after birth, revealing the main symptom of the disease, extensive scaling of the skin caused by hyperkeratosis. With increasing age, the scaling tends to be concentrated around joints in areas such as the groin, the armpits, the inside of the elbow and the neck. The scales often tile the skin and may resemble fish scales.
Brittle hair
MedGen UID:
120480
Concept ID:
C0263490
Disease or Syndrome
Fragile, easily breakable hair, i.e., with reduced tensile strength.
Brittle scalp hair
MedGen UID:
870836
Concept ID:
C4025296
Finding
Fragile, easily breakable scalp hair.

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

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
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
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

Nartisa I, Kirsteina R, Neiburga KD, Zigure S, Ozola L, Grantina I, Micule I, Murmane D, Slisere B, Gailite L, Vilne B, Rots D, Taurina G, Kurjane N
Pediatr Allergy Immunol 2023 Apr;34(4):e13937. doi: 10.1111/pai.13937. PMID: 37102386
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
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

Bilgili SG, Karadag AS, Calka O, Onder S, Bayram I
Genet Couns 2016;27(3):353-356. PMID: 30204963
Kumar P, Gharami RC, Mondal A, Ghosh K
Indian Pediatr 2010 Dec;47(12):1052. PMID: 21220804
Mizuno Y, Suga Y, Muramatsu S, Hasegawa T, Shimizu T, Ogawa H
Br J Dermatol 2005 Sep;153(3):661-3. doi: 10.1111/j.1365-2133.2005.06770.x. PMID: 16120162
DiGiovanna JJ, Robinson-Bostom L
Am J Clin Dermatol 2003;4(2):81-95. doi: 10.2165/00128071-200304020-00002. PMID: 12553849
Coenraads PJ
Br J Dermatol 1980 Apr;102(4):484-5. PMID: 7387896

Therapy

Bilgili SG, Karadag AS, Calka O, Onder S, Bayram I
Genet Couns 2016;27(3):353-356. PMID: 30204963
Kumar P, Gharami RC, Mondal A, Ghosh K
Indian Pediatr 2010 Dec;47(12):1052. PMID: 21220804
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
Happle R, van de Kerkhof PC, Traupe H
Dermatologica 1987;175 Suppl 1:107-24. doi: 10.1159/000248867. PMID: 2961628

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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