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Hypospadias 1, X-linked(HYSP1)

MedGen UID:
394735
Concept ID:
C2678098
Disease or Syndrome
Synonym: HYSP1
 
Gene (location): AR (Xq12)
 
Monarch Initiative: MONDO:0010384
OMIM®: 300633

Definition

Hypospadias is a common congenital malformation of the penis, affecting approximately 1 in 750 births in Europe. Due to developmental arrest of urethral fusion, the urethral opening is displaced along the ventral side of the penis. The opening can be located glanular, penile, or even more posterior in the scrotum or perineum. Although most children with this condition undergo surgery in their second year of life, serious medical, social, and sexual problems may still exist later in life (summary by van der Zanden et al., 2011). Hypospadias is a feature of several syndromic disorders, including the androgen insensitivity syndrome (300068) and Opitz syndrome (300000). Genetic Heterogeneity of Hypospadias See also HYSP2 (300758), caused by mutation in the MAMLD1 gene (300120) on chromosome Xq28; HYSP3 (146450), a familial form which has been mapped to chromosome 7q32.2-q36.1; and HYSP4 (300856), a susceptibility locus mapped to chromosome Xp11.22 and associated with variation in the DGKK gene (300837). [from OMIM]

Clinical features

From HPO
Perineal hypospadias
MedGen UID:
105292
Concept ID:
C0452148
Congenital Abnormality
Hypospadias with location of the urethral meatus in the perineal region.

Recent clinical studies

Etiology

Pinke LA, Rathbun SR, Husmann DA, Kramer SA
J Urol 2001 Nov;166(5):1865-8. doi: 10.1016/s0022-5347(05)65708-4. PMID: 11586250
Brinkmann A, Jenster G, Ris-Stalpers C, van der Korput H, Brüggenwirth H, Boehmer A, Trapman J
Steroids 1996 Apr;61(4):172-5. doi: 10.1016/0039-128x(96)00008-6. PMID: 8732995
Opitz JM, Richieri-da Costa A, Aase JM, Benke PJ
Am J Med Genet 1988 May-Jun;30(1-2):309-28. doi: 10.1002/ajmg.1320300132. PMID: 3052062
Roe TF, Alfi OS
Pediatrics 1977 Jul;60(1):55-9. PMID: 876735
Gonzalez CH, Herrmann J, Opitz JM
Eur J Pediatr 1977 Apr 26;125(1):1-13. doi: 10.1007/BF00470600. PMID: 870322

Diagnosis

Micale L, Russo F, Mascaro M, Morlino S, Nardella G, Fusco C, Bisceglia L, Meroni G, Castori M
Pediatr Res 2023 Apr;93(5):1208-1215. Epub 2022 Aug 11 doi: 10.1038/s41390-022-02237-y. PMID: 35953512
Finsterer J, Soraru G
J Mol Neurosci 2016 Mar;58(3):321-9. Epub 2015 Oct 19 doi: 10.1007/s12031-015-0663-x. PMID: 26482145
Suntharalingham JP, Buonocore F, Duncan AJ, Achermann JC
Best Pract Res Clin Endocrinol Metab 2015 Aug;29(4):607-19. Epub 2015 Jul 14 doi: 10.1016/j.beem.2015.07.004. PMID: 26303087Free PMC Article
Tian L, Chen M, Peng JH, Zhang JW, Li L
J Huazhong Univ Sci Technolog Med Sci 2014 Feb;34(1):81-86. Epub 2014 Feb 6 doi: 10.1007/s11596-014-1235-y. PMID: 24496683
El-Khairi R, Martinez-Aguayo A, Ferraz-de-Souza B, Lin L, Achermann JC
Endocr Dev 2011;20:38-46. Epub 2010 Dec 16 doi: 10.1159/000321213. PMID: 21164257

Therapy

Martínez-Frías ML
Am J Med Genet 1994 Aug 1;52(1):5-8. doi: 10.1002/ajmg.1320520103. PMID: 7977461
Grino PB, Isidro-Gutierrez RF, Griffin JE, Wilson JD
J Clin Endocrinol Metab 1989 Mar;68(3):578-84. doi: 10.1210/jcem-68-3-578. PMID: 2918059
Roe TF, Alfi OS
Pediatrics 1977 Jul;60(1):55-9. PMID: 876735

Prognosis

Micale L, Russo F, Mascaro M, Morlino S, Nardella G, Fusco C, Bisceglia L, Meroni G, Castori M
Pediatr Res 2023 Apr;93(5):1208-1215. Epub 2022 Aug 11 doi: 10.1038/s41390-022-02237-y. PMID: 35953512
Grino PB, Isidro-Gutierrez RF, Griffin JE, Wilson JD
J Clin Endocrinol Metab 1989 Mar;68(3):578-84. doi: 10.1210/jcem-68-3-578. PMID: 2918059

Clinical prediction guides

Frank S, Gabassi E, Käseberg S, Bertin M, Zografidou L, Pfeiffer D, Brennenstuhl H, Falk S, Karow M, Schweiger S
Life Sci Alliance 2024 Apr;7(4) Epub 2024 Jan 18 doi: 10.26508/lsa.202302288. PMID: 38238086Free PMC Article
Micale L, Russo F, Mascaro M, Morlino S, Nardella G, Fusco C, Bisceglia L, Meroni G, Castori M
Pediatr Res 2023 Apr;93(5):1208-1215. Epub 2022 Aug 11 doi: 10.1038/s41390-022-02237-y. PMID: 35953512
Battaglia A, Chines C, Carey JC
Am J Med Genet A 2006 Oct 1;140(19):2075-9. doi: 10.1002/ajmg.a.31302. PMID: 16691600
Willatt L, Cox J, Barber J, Cabanas ED, Collins A, Donnai D, FitzPatrick DR, Maher E, Martin H, Parnau J, Pindar L, Ramsay J, Shaw-Smith C, Sistermans EA, Tettenborn M, Trump D, de Vries BB, Walker K, Raymond FL
Am J Hum Genet 2005 Jul;77(1):154-60. Epub 2005 May 25 doi: 10.1086/431653. PMID: 15918153Free PMC Article
Proud VK, Levine C, Carpenter NJ
Am J Med Genet 1992 Apr 15-May 1;43(1-2):458-66. doi: 10.1002/ajmg.1320430169. PMID: 1605226

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