Am J Perinatol 2004; 21(3): 131-134
DOI: 10.1055/s-2004-823771
Copyright © 2004 by Thieme Medical Publishers, Inc., 333 Seventh Avenue, New York, NY 10001, USA.

Co-Occurrence of Coarctation of the Aorta and Hypospadias in Smaller Twins in Monochorionic Pregnancies: Two Case Reports

Kazushi Yasuda1 , Shigeru Ohki1 , Masashi Seguchi1
  • 1Department of Pediatrics, Seirei Hamamatsu General Hospital, Hamamatsu, Shizuoka, Japan
Further Information

Publication History

Publication Date:
14 April 2004 (online)

We reconstructed of the aortic arches of two unrelated boys, who were the smaller twin in monochorionic pregnancies, because they had coarctation of the aorta (CoA). They both had hypospadias. The co-twins, the larger twins, had neither CoA nor hypospadias. Although CoA and hypospadias both have been associated separately with monozygotic pregnancy, this is the first report of CoA and hypospadias occurring together. These cases indicate that epigenetic factors such as unbalanced fetal circulation in monochorionic pregnancy may be involved in co-occurrence of CoA and hypospadias.

REFERENCES

  • 1 Robertson E G, Neer K J. Placental injection studies in twin gestation.  Am J Obstet Gynecol. 1983;  147 170-174
  • 2 Bajoria R, Wigglesworth J, Fisk N M. Angioarchitecture of monochorionic placentas in relation to the twin-twin transfusion syndrome.  Am J Obstet Gynecol. 1995;  172 856-863
  • 3 Simpson L L, Marx G R, Elkadry E A, D'Alton M E. Cardiac dysfunction in twin-twin transfusion syndrome: a prospective, longitudinal study.  Obstet Gynecol. 1998;  92 557-562
  • 4 Zosmer N, Bajoria R, Weiner E, Rigby M, Vaughan J, Fisk N M. Clinical and echographic features of in utero cardiac dysfunction in the recipient twin in twin-twin transfusion syndrome.  Br Heart J. 1994;  72 74-79
  • 5 Murakoshi T, Yamamori K, Tojo Y et al.. Pulmonary stenosis in recipient twins in twin-to-twin transfusion syndrome: report on 3 cases and review of literature.  Croat Med J. 2000;  41 252-256
  • 6 Lougheed J, Sinclair B G, Fung Kee Fung K et al.. Acquired right ventricular outflow tract obstruction in the recipient twin in twin-twin transfusion syndrome.  J Am Coll Cardiol. 2001;  38 1533-1538
  • 7 Nakayama D, Masuzaki H, Yoshimura S, Moriyama S, Ishimaru T. Monozygotic twins discordant for single umbilical artery and congenital heart disease.  Am J Obstet Gynecol. 1998;  179 256-257
  • 8 Negishi H, Okuyama K, Sagawa T, Makinoda S, Fujimoto S. Two cases of monozygotic twins, in each of which one fetus was prenatally diagnosed as having a heart anomaly.  J Obstet Gynaecol. 1995;  21 293-298
  • 9 Driver C D, Suckling P V. Coarctation of the aorta in one of identical twins.  S Afr Med J. 1960;  34 592-593
  • 10 Sehested J. Coarctation of the aorta in monozygotic twins.  Br Heart J. 1982;  47 619-620
  • 11 Roberts C J, Lloyd S. Observations on the epidemiology of simple hypospadias.  BMJ. 1973;  1 768-770
  • 12 Källén B, Bertollini R, Castilla E et al.. A joint international study on the epidemiology of hypospadias.  Acta Paediatr Scand Suppl. 1986;  324 1-52
  • 13 Fredell L, Lichtenstein P, Pedersen N L, Svensson J, Nordenskjold A. Hypospadias is related to birth weight in discordant monozygotic twins.  J Urol. 1998;  160 2197-2199
  • 14 Rudolph A M, Heymann M A, Spitznas U. Hemodynamic considerations in the development of narrowing of the aorta.  Am J Cardiol. 1972;  30 514-525
  • 15 Machin G A. Some causes of genotypic and phenotypic discordance in monozygotic twin pairs.  Am J Med Genet. 1996;  61 216-228
  • 16 Nieuwint A, Van Zalen-Sprock R, Hummel P et al.. “Identical” twins with discordant karyotypes.  Prenat Diagn. 1999;  19 72-76
  • 17 Büyükgebiz A, Ören H, Ünsal E, Akçoral A, Özen E, Sakizli M. A case of mixed gonadal dysgenesis with 45 X/46 XY karyotype and aortic coarctation.  J Pediatr Endocrinol. 1993;  6 191-196
  • 18 Konrad D, Sossai R, Winklehner H L, Binkert F, Artan S, Scharli A F. Penoscrotal hypospadias and coarctation of the aorta with mixed gonadal dysgenesis.  Pediatr Surg Int. 2000;  16 226-228
  • 19 Cattolica E V, Solomon I L. The urologic manifestations of XO/XY mosaicism.  J Urol. 1978;  120 103-105
  • 20 McElhinney D B, Clark III B J, Weinberg P M et al.. Association of chromosome 22q11 deletion with isolated anomalies of aortic arch laterality and branching.  J Am Coll Cardiol. 2001;  37 2114-2119
  • 21 Wu H Y, Rusnack S L, Bellah R D et al.. Genitourinary malformations in chromosome 22q11.2 deletion.  J Urol. 2002;  168 2564-2565
  • 22 Jacobson Z, Glickstein J, Hensle T, Marion R W. Further delineation of the Opitz G/BBB syndrome: report of an infant with complex congenital heart disease and bladder exstrophy, and review of the literature.  Am J Med Genet. 1998;  78 294-299
  • 23 Stevens C A, Wilroy Jr R S. The telecanthus-hypospadias syndrome.  J Med Genet. 1988;  25 536-542

Kazushi YasudaM.D. 

Department of Pediatrics, Toyohashi Municipal Hospital

50 Hachiken-nishi, Aotake, Toyohashi

Aichi 441-8570, Japan

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