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At least 1,171 records · Page 65Linked to original sources
PubMed1980

Unusual case of urethral duplication.

We present a case of unique penile malformation, a child with common prostatic urethra with two normal, double urethras parallel to one another with a bifid glans penis. This case emphasizes how abnormal embryology can contribute to our knowledge of normal development.

Abnormalities, Multiple↗
PubMed1992

Hypoplasia of urinary bladder.

Three female patients with hypoplasia of the urinary bladder and associated congenital anomalies are described. All were first seen in early infancy with significant obstructive uropathy and were managed by early loop cutaneous ureterostomy. All have survived to adulthood and 2 patients have received a kidney transplant into an ileal conduit; 1 patient continues to do well with cutaneous ureterostomy only. Pathogenesis, management, and long-term outcome are discussed.

Abnormalities, Multiple↗
PubMed1992

Accessory and ectopic scrotum with VATER association.

Scrotal ectopia or accessory scrotum are both rare conditions. This report describes a neonate with both abnormalities, who also has elements of the VATER association. This constellation of anomalies has not been previously described.

Abnormalities, Multiple↗
PubMed1989

CHARGE association in two monozygous triplets.

In an unusual set of triplets, two monozygous girls presented with CHARGE association. Dissimilar surgical management of identical cardiovascular lesions has resulted in a disparate effect on their clinical state. Their case poses interesting questions concerning how CHARGE association may develop.

Abnormalities, Multiple↗
PubMed1985

Congenital absence of the scaphoid in the "VATER" association.

A rare carpal anomaly is reported in a patient with multiple congenital abnormalities. A literature search reveals few such cases, but a characteristic clinical picture emerges. Patients with this deformity should be carefully examined to exclude other congenital abnormalities.

Abnormalities, Multiple↗
PubMed1993

Qualitative abnormal fetal breathing movements, associated with tracheal atresia.

A case is reported in which qualitatively, grossly abnormal fetal breathing movements turned out to be indicative of complete tracheal atresia. Fetal breathing movements were vigorous and jerky and of large amplitude; similarly abnormal movements were observed after birth. At postmortem tracheal atresia was diagnosed, in combination with other congenital abnormalities, which pointed to a VATER-association. The combination of a VATER-association with tracheal atresia has only been reported once before. An absence of the normal Hering-Breuer reflex in this case of complete tracheal atresia, probably explains the abnormal fetal breathing pattern. This case is in line with a growing number of reports in literature in which qualitatively abnormal fetal movements were indicative of congenital malformations.

Abnormalities, Multiple↗
PubMed2003

Single umbilical artery and the VATER-animal model.

BACKGROUND/PURPOSE: Vascular disruption is recognized as a cause of congenital malformations. The authors analyzed the significance of single umbilical artery (SUA) in the Adriamycin-animal model to find out if it was associated with organ malformations. METHODS: Pregnant SD rats were injected with Adriamycin intraperitoneally at a dose of 2 mg/kg on days 6 through 9 of gestation. Serial transverse sections of full-term fetuses were analyzed by light microscopy. Embryos also were removed on different gestational days during organogenesis, and serial transverse histologic sections were examined and compared with suitable controls. RESULTS: In experimental embryos (n = 47), presence of a SUA resulted from either persistence of the primitive umbilical arteries that joined each other ventral to the hind gut to give rise to one umbilical artery or from secondary atrophy of one of the definitive umbilical arteries. Malformations such as intestinal atresia were associated with anomalous fusion between the dorsal aorta and the persistent primitive umbilical arteries. CONCLUSIONS: SUA is a prominent feature of the Adriamycin-animal model. No obvious association was found between malformations and SUA that resulted from atrophy of one of the definitive umbilical arteries; however, it was associated with anomalies such as intestinal atresia when it resulted from persistence of the primitive umbilical arteries.

Abnormalities, Drug-Induced↗
PubMed2004

Caudal duplication syndrome: more evidence for theory of caudal twinning.

Caudal duplication syndrome is a rare entity in which structures derived from the embryonic cloaca and notochord are duplicated to various extents. The term encompasses a spectrum and often is quoted as one type of incomplete separation of monovular twins. The authors present more evidence giving credence to caudal twining as the mechanism behind the syndrome. The authors report successful surgical management of a full-term infant with a constellation of anomalies of caudal duplication syndrome.

Abnormalities, Multiple↗
PubMed2004

Cecal volvulus: a report of two cases occurring after the antegrade colonic enema procedure.

BACKGROUND: Many children with chronic constipation and fecal incontinence have benefited from the antegrade colonic enema (ACE) procedure. Routine antegrade colonic lavage often allows such children to avoid daytime soiling. This report describes 2 children in whom the ACE procedure was complicated by a cecal volvulus. METHODS: A retrospective review of 164 children with an ACE procedure was conducted. Two instances of cecal volvulus were identified. RESULTS: The first child presented with abdominal pain and difficulty intubating the ACE site. Over the subsequent day, his pain worsened, and radiographs depicted a colonic obstruction. At laparotomy, a cecal volvulus resulting in bowel necrosis was observed, and resection of the affected bowel and appendix (in the right lower quadrant) and end ileostomy was required. He subsequently had the stoma closed and a new ACE constructed with a colon flap. The second child presented with shock and evidence of an acute abdomen. At laparotomy, a cecal volvulus was noted, and ileocolic resection including the ACE stoma (located at the umbilicus) and an ileostomy and Hartmann pouch was performed. He had a protracted hospital course requiring ventilator and inotropic support. He currently is well and still has an ileostomy stoma. CONCLUSIONS: A high index of suspicion for a potentially life-threatening cecal volvulus should be maintained in children undergoing an ACE procedure who present with abdominal pain, evidence of bowel obstruction, or difficulty in advancing the ACE irrigation catheter.

Anus, Imperforate↗