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Bathing suit ichthyosis

MedGen UID:
1386460
Concept ID:
C4511230
Disease or Syndrome
Synonyms: bathing suit ichthyosis; BSI
SNOMED CT: Bathing suit ichthyosis (725588002)
Modes of inheritance:
Autosomal recessive inheritance
MedGen UID:
141025
Concept ID:
C0441748
Intellectual Product
Source: Orphanet
A mode of inheritance that is observed for traits related to a gene encoded on one of the autosomes (i.e., the human chromosomes 1-22) in which a trait manifests in individuals with two pathogenic alleles, either homozygotes (two copies of the same mutant allele) or compound heterozygotes (whereby each copy of a gene has a distinct mutant allele).
 
Monarch Initiative: MONDO:0015085
Orphanet: ORPHA100976

Definition

A rare variant of autosomal recessive congenital ichthyosis. Less than 20 patients are reported in the literature. Large dark scales are present on warmer skin areas such as the trunk, the scalp, and the axillary region. On affected areas, the scales are similar to those observed in lamellar ichthyosis. Caused by specific thermo-sensitive mutations in the TGM1 gene (encoding transglutaminase 1, involved in the cornification of the stratum corneum). Affected skin areas, show a clearly reduced enzyme activity in contrast to healthy skin areas that demonstrate an almost normal enzyme activity. Transmission is autosomal recessive. [from SNOMEDCT_US]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVBathing suit ichthyosis

Professional guidelines

PubMed

Chiramel MJ, Mathew L, Athirayath R, Chapla A, Sathishkumar D, Mani T, Danda S, George R
Pediatr Dermatol 2022 May;39(3):420-424. Epub 2022 Apr 12 doi: 10.1111/pde.14944. PMID: 35412663

Recent clinical studies

Etiology

Supsrisunjai C, Bunnag T, Chaowalit P, Boonpuen N, Kootiratrakarn T, Wessagowit V
Pediatr Dermatol 2023 Jan;40(1):107-112. Epub 2022 Oct 19 doi: 10.1111/pde.15156. PMID: 36262015
Takeichi T, Akiyama M
J Dermatol 2016 Mar;43(3):242-51. doi: 10.1111/1346-8138.13243. PMID: 26945532
Liu JJ, Yuan YY, Zhang XQ, Li ZM, Xu YS, Gao SM, Cai JF, Shao XH, Lin XH, Li BX
Clin Exp Dermatol 2015 Jan;40(1):56-62. Epub 2014 Aug 22 doi: 10.1111/ced.12410. PMID: 25154629
Rodríguez-Pazos L, Ginarte M, Vega A, Toribio J
Actas Dermosifiliogr 2013 May;104(4):270-84. Epub 2013 Apr 3 doi: 10.1016/j.adengl.2011.11.021. PMID: 23562412
Jacyk WK
Eur J Dermatol 2005 Nov-Dec;15(6):433-6. PMID: 16280294

Diagnosis

Li W, Oberlin KE, Wilson TE, Haggstrom AN
Pediatr Dermatol 2020 Jan;37(1):165-170. Epub 2019 Oct 20 doi: 10.1111/pde.14030. PMID: 31631373
Marukian NV, Hu RH, Craiglow BG, Milstone LM, Zhou J, Theos A, Kaymakcalan H, Akkaya DA, Uitto JJ, Vahidnezhad H, Youssefian L, Bayliss SJ, Paller AS, Boyden LM, Choate KA
JAMA Dermatol 2017 Jun 1;153(6):537-543. doi: 10.1001/jamadermatol.2017.0202. PMID: 28403434Free PMC Article
Takeichi T, Akiyama M
J Dermatol 2016 Mar;43(3):242-51. doi: 10.1111/1346-8138.13243. PMID: 26945532
Rodríguez-Pazos L, Ginarte M, Vega A, Toribio J
Actas Dermosifiliogr 2013 May;104(4):270-84. Epub 2013 Apr 3 doi: 10.1016/j.adengl.2011.11.021. PMID: 23562412
Trindade F, Fiadeiro T, Torrelo A, Hennies HC, Hausser I, Traupe H
Eur J Dermatol 2010 Jul-Aug;20(4):447-50. Epub 2010 Jun 3 doi: 10.1684/ejd.2010.1008. PMID: 20522418

Therapy

Benmously-Mlika R, Zaouak A, Mrad R, Laaroussi N, Abdelhak S, Hovnanian A, Mokhtar I
Int J Dermatol 2014 Dec;53(12):1478-80. Epub 2014 Sep 10 doi: 10.1111/ijd.12569. PMID: 25209454
Washio K, Fukunaga A, Terai M, Hitomi K, Yamanishi K, Nishigori C
Acta Derm Venereol 2014 May;94(3):349-50. doi: 10.2340/00015555-1739. PMID: 24158451

Prognosis

Marukian NV, Hu RH, Craiglow BG, Milstone LM, Zhou J, Theos A, Kaymakcalan H, Akkaya DA, Uitto JJ, Vahidnezhad H, Youssefian L, Bayliss SJ, Paller AS, Boyden LM, Choate KA
JAMA Dermatol 2017 Jun 1;153(6):537-543. doi: 10.1001/jamadermatol.2017.0202. PMID: 28403434Free PMC Article

Clinical prediction guides

Bourrat E, Blanchet-Bardon C, Derbois C, Cure S, Fischer J
Arch Dermatol 2012 Oct;148(10):1191-5. doi: 10.1001/archdermatol.2012.1947. PMID: 22801880

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