[Hydrocolpos in infants with urinary retention].
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Female↗
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Hydrocolpos and haematocolpos in childhood.
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Adolescent↗
[Mckusick Kaufman syndrome. Report of a case with hydrocolpos and polydactyly].
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Female↗
Continent urinary reconstruction in ischiopagus tripus conjoined twins.
The principle of the continent urinary reservoir involves use of a variety of intestinal segments providing continence, a mechanism of antireflux and a catheterizable stoma in either an abdominal or pelvic location. This concept was used to create a continent urinary reservoir in a 3-year-old former ischiopagus tripus conjoined twin. The solitary renal unit had been drained into a hydrocolpos with an antirefluxing ureteral reimplantation at separation. The patient remained incontinent through the urogenital sinus. At subsequent reconstruction the posterior aspect of the hydrocolpos was tubularized as a vagina, while the remainder of the hydrocolpos was augmented with ileum to create a urinary reservoir. The conduit was constructed with imbricated ileum, in which myectomy had been performed to allow for easier imbrication and tubularization for a narrow neourethra. A second set of conjoined twins have been separated similarly. Of these twins 1 will undergo creation of a continent urinary reservoir in an identical fashion. This represents the first report of the use of hydrocolpos in the creation of a urinary reservoir.
Child, Preschool↗
Pitfalls in the management of newborn cloacas.
Clinicians caring for newborns with persistent cloaca face significant challenges in the newborn period. Avoiding key pitfalls during this time can have dramatic implications. We reviewed the medical records of 361 patients with cloaca operated on at our institution and analyzed sequelae that resulted from incorrect management in the newborn period. Of 361 patients, 282 underwent primary operations at our institution, and 79 patients were referred to us after a failed repair at other institutions. Pitfalls in management during the newborn period included the following: (1) Failure to recognize and manage hydrocolpos, which occurred in 46 patients. Of these, three patients developed pyocolpos (two progressed to vaginal perforation), and 43 suffered from persistent bilateral hydronephrosis, megaureters, recurrent urinary tract infections, persistent acidosis, or failure to thrive due to undrained hydrocolpos. They underwent unnecessary urinary drainage procedures (nephrostomy, ureterostomy, cystostomy, or vesicostomy) in the newborn period. When the vagina was finally decompressed, all of these symptoms disappeared. (2) Colostomy or vesicostomy problems, which occurred in 50 patients. These included incorrect placement of the colostomy (too distal, which interfered with the pull-through) in 24 and colostomy prolapse in 23. Incompletely diverting loop colostomies led to urinary tract infections in 49 patients. Vesicostomy prolapse occurred in three patients. (3) Clinical misdiagnosis, which occurred in 42 patients. Six were incorrectly diagnosed as "intersex" and 36 as "rectovaginal fistula." In this group only the rectum was repaired, and the patients were left with a urogenital sinus that required reoperation. Proper management of a newborn with cloaca includes drainage of a hydrocolpos, which avoids unnecessary urinary diversions and pyocolpos. Our preferred colostomy is one with separated stomas, adequate distal bowel for the pull-through, and use of a proper technique to avoid prolapse. Correct clinical diagnosis of cloaca avoids problems during the definitive repair.
Cloaca↗
Hydrometrocolpos in neonate due to distal vaginal atresia.
Hydrocolpos is the result of vaginal obstruction and can become an emergency in the newborn period. The treatment of imperforate hymen is well defined, but the treatment of vaginal atresia is more complex. We encountered two cases of hydrocolpos secondary to distal vaginal atresia, that were operated on in the first days of life. One baby had distal atresia without persistance of urogenital sinus. Surgery combining abdominal perineal approaches and a posterior vaginoplasty was carried out. The second baby had hydrocolpos with persistance of urogenital sinus. Drainage through the sinus was unsuccessful because the baby developed sepsis by trapping urine in the uterus. Finally an abdominoperineal vaginal pull-through was successfully done. The embryology and literature are reviewed. The classification, indications and surgical technique are discussed.
Female↗
Pyocolpos: diagnosis and treatment.
Pyocolpos is a rare complication of hydrocolpos. Hydrocolpos usually presents during adolescence and is associated with an imperforate hymen. The following is a case of a 3-month-old girl with pyocolpos. Her history was significant for a urinary tract infection (UTI) at 7 weeks of age. The authors believe that her UTI was caused by urinary retention secondary to hydrocolpos. A complete evaluation may have prevented the complication of pyocolpos.
Drainage↗
An unusual case of urinary retention in a competitive gymnast.
Hydrocolpos is the accumulation of blood behind an imperforate hymen. Patients often present in early adolescence with cyclic abdominal pain. We present the case of an elite gymnast who, after 6 months of symptoms and a series of incorrect diagnoses, was diagnosed with hydrocolpos. It is likely that the patient's extraordinary training regimen contributed to the difficulty in diagnosis.
Adolescent↗
Hydrometrocolpos, postaxial polydactyly, congenital heart disease, and anomalies of the gastrointestinal and genitourinary tracts: a rare autosomal recessive syndrome.
Nine cases with the hydrometrocolpospolydactyly syndrome (4 males, 5 females) from four unrelated families are presented. Leading symptoms of this rare disorder were hydrocolpos and postaxial polydactyly. Three affected girls had urinary hydrocolpos without vaginal septum or imperforate hymen, one had partial vaginal atresia, and one had no hydrometrocolpos. Glandular hypospadias and prominent scrotal raphe are added to the spectrum of malformations in this disorder in males. The literature is reviewed and problems in genetic counseling in this autosomal recessive disorder are discussed.
Abnormalities, Multiple↗
Surgical management of cloacal malformations: a review of 339 patients.
BACKGROUND/PURPOSE: The aim of this study was to describe lessons learned from the authors' series of patients with cloaca and convey the improved understanding and surgical treatment of the condition's wide spectrum of complexity. METHODS: The medical records of 339 patients with cloaca operated on by the authors were retrospectively reviewed. RESULTS: A total of 265 patients underwent primary operations, and 74 were secondary. All patients were approached posterior sagittally; 111 of them also required a laparotomy. The average length of the common channel was 4.7 cm for patients that required a laparotomy and 2.3 cm for those that did not. Vaginal reconstruction involved a vaginal pull-through in 196 patients, a vaginal flap in 38, vaginal switch in 30, and vaginal replacement in 75 (36 with rectum, 31 with ileum, and 8 with colon). One hundred twenty-two patients underwent a total urogenital mobilization. Complications included vaginal stricture or atresia in 17, urethral strictures in 6, and urethro-vaginal fistula in 19, all of which occurred before the introduction of the total urogenital mobilization. A total of 54% of all evaluated patients were continent of urine and 24% remain dry with intermittent catheterization through their native urethra and 22% through a Mitrofanoff-type of conduit. Seventy-eight percent of the patients with a common channel longer than 3 cm require intermittent catheterization compared with 28% when their common channel was shorter than 3 cm. Sixty percent of all cases have voluntary bowel movements (28% of them never soiled, and 72% soiled occasionally). Forty percent are fecally incontinent but remain clean when subjected to a bowel management program. Forty-eight patients born at other institutions with hydrocolpos were not treated correctly during the neonatal period. The surgeons failed to drain the dilated vaginas, which interfered with the drainage of the ureters and provoked urinary tract infections, pyocolpos, and/or vaginal perforation. In 24 patients, the colostomy was created too distally, and it interfered with the pull-through. Twenty-three patients suffered from colostomy prolapse. All of these patients required a colostomy, revision before the main repair. Thirty-six patients underwent reoperation because they had a persistent urogenital sinus after an operation done at another institution, and 38 patients underwent reoperation because they suffered from atresia or stenosis of the vagina or urethra. The series was divided into 2 distinct groups of patients: group A were those with a common channel shorter than 3 cm (62%) and group B had a common channel longer than 3 cm (38%). CONCLUSIONS: The separation of these groups has important therapeutic and prognostic implications. Group A patients can be repaired posterior sagittally with a reproducible, relatively short operation. Because they represent the majority of patients, we believe that most well-trained pediatric surgeons can repair these type of malformations, and the prognosis is good. Group B patients (those with a common channel longer than 3 cm), usually require a laparotomy and have a much higher incidence of associated urologic problems. The surgeons who repair these malformations require special training in urology, and the operations are prolonged, technically demanding, and the functional results are not as good as in group A. It is extremely important to establish an accurate neonatal diagnosis, drain the hydrocolpos when present, and create an adequate, totally diverting colostomy, leaving enough distal colon available for the pull-through and fixing the colon to avoid prolapse. A correct diagnosis will allow the surgeon to repair the entire defect and avoid a persistent urogenital sinus. Cloacas comprise a spectrum of defects requiring a complex array of surgical decisions. The length of the common channel is an important determinant of the potential for urinary control, and predicts the extent of surgical repair.
Cloaca↗
Hydrometrocolpos: diagnosis and follow-up by ultrasound--a case report.
Hydrometrocolpos is accumulation of secretions in the vagina and uterus, caused by excessive intrauterine stimulation of the infant's cervical mucous glands by maternal oestrogen in the presence of an intact hymen (Wilson et al 1978). Hydrocolpos is dilatation of the vagina proximal to a congenital obstruction. If the uterus is also dilated, the condition is called hydrometrocolpos. Most cases of hydrocolpos are associated with an imperforate hymen that forms a thin bulging membrane between the labia. Though many cases of hydrometrocolpos have been reported in literature, there is no case report which shows classical ultrasound findings and follow up ultrasound scans to show the involuted uterus. We are presenting a case report whose diagnosis was established on ultrasound. This case highlights the value of ultrasound in diagnosing this condition and excluding other associated renal anomalies.
Body Water↗
Pallister-Hall syndrome and McKusick-Kaufmann syndrome: one entity?
The Pallister-Hall syndrome is characterised by specific facial anomalies, postaxial polydactyly, imperforate anus, and brain anomalies including a diencephalic hamartoblastoma. The hallmarks of the McKusick-Kaufmann syndrome are hydrocolpos owing to vaginal atresia, postaxial polydactyly, imperforate anus, and congenital heart defects. We report a patient with the unique features of hydrocolpos, postaxial polydactyly, and hypothalamic hamartoblastoma and discuss the different aetiological considerations of both syndromes and implications for clinical management.
Abnormalities, Multiple↗
Unilateral occlusion of duplicated uterus with ipsilateral renal anomaly in young girls: a study with MRI.
Twenty-four young girls (mean age 13.0 years) with unilateral occlusion of a duplicated uterus and ipsilateral renal agenesis, dysplasia or hypoplasia were studied with magnetic resonance imaging (MRI) following ultrasound examination. Hydrocolpos (n = 4), hydrometrocolpos (n = 2), hematocolpos (n = 11), hematometrocolpos (n = 5), hematocolpometra, hematosalpinx (n = 3) and hematometra, hematosalpinx (n = 1) were noted (two of these patients had presented with hydrocolpos and hematocolpos before and after the menarche). Twenty-two of these girls presented with ipsilateral renal agenesis (right 11, left 11) with ectopic ureters to Gartner's duct cysts (GDC) in two, in one renal hypoplasia and in one renal dysplasia with ectopic ureters to GDC. MRI offered specific images of the genital tract, showing the exact type of müllerian duct anomaly and providing high diagnostic accuracy. Such preoperative identification of a uterine anomaly, complemented with appropriate surgical intervention, can assist young girls in achieving normal fertility in the future.
Abnormalities, Multiple↗
McKusick-Kaufman syndrome: report of an instructive family.
We report on two sisters and their brother, all of whom are affected by McKusick-Kaufman syndrome. This sibship confirms autosomal recessive inheritance and the occurrence of the syndrome in Melanesians. It also demonstrates multisystem involvement in both sexes and illustrates difficulties in the diagnosis and management of hydrocolpos.
Abnormalities, Multiple↗
McKusik-Kaufman syndrome: prenatal diagnosis, genetics and follow up.
McKusick-Kaufman syndrome (MKKS) is a rare autosomal recessive genetic disease with classical hexadactyly and hydrocolpos in females and sometimes cardiac abnormality. We report such a case diagnosed just before birth with a favourable outcome. From this case we describe and discuss all the prenatal sonographic signs which are not always present. On the genetic side, the gene has recently been localized together with the mutation responsible for MKKS. The phenotypic relationship between MKKS which has a good prognosis and Bardet-Biedl syndrome (BBS) with a worse prognosis requires great caution before diagnosing MKKS and a long follow-up is necessary to recognize obesity, growth retardation and pigmentary retinitis.
Abnormalities, Multiple↗
The dawn of paediatric surgery: Johannes Fatio (1649-1691)--his life, his work and his horrible end.
Johannes Fatio was probably the first surgeon who systematically studied and treated surgical condition of children. Born in Basle in 1649, he never studied medicine at the university there but became a member of the barber's guild. Later he studied medicine and became a doctor of the University of Valence. He returned to Basle to practice surgery, midwifery and paediatric surgery. He wrote a number of medical books, but only one, The Helvetic Reasonable Midwife, still exists. In it he devotes a whole section to surgical afflictions of children. He describes ingenuous operations for such conditions as hypospadias, hydrocolpos, imperforate anus and many more. He was the first surgeon successfully to separate conjoined twins. He became involved in the turbulent politics of the city of Basle and was largely responsible for the overthrow and modern ratio of the Basle parliament and government. In the following counter-revolution he was captured, imprisoned, tortured and executed. All his written works were destroyed by the authorities; The Helvetic Reasonable Midwife was published 70 years after his death.
General Surgery↗
Risk for recombinants in pericentric inversions of the (p11 leads to q21) region of chromosome 18.
A child with female hypospadia complicated by bilateral hydronephrosis, hydroureter, and hydrocolpos was heterozygous for a pericentric inversion of chromosome 18, 46,XX,inv(18)(p11q21). The normal mother and her father had the same inversion. The abnormal phenotype of the girl could be due to undetectable recombination or to a position effect. She had a low level of the enzyme peptidase-A whose locus is on 18q, while her mother and grandfather had normal levels. The two other cases of familial inversions for chromosomes 18 in the literature both involve the same (p11 leads to q21) region. These three families give a tentative figure of at least 10% as the risk for a normal carrier of this pericentric inversion to have an affected offspring due to recombination.
Chromosome Aberrations↗
Successful separation of thoraco-omphalopagus and ischiopagus tetrapus twins in Korea.
A pair of male thoraco-omphalopagus twins with common liver, diaphragm, pericardium, and sternum was separated at the age of 59 days after a parasitic relationship had developed between them. Before separation one baby developed acute renal failure during which he had no edema and had normal serum electrolytes, urea nitrogen, and creatinine due to "autodialysis" by the other baby. The boys have now grown normally to the age of 6 years. A pair of female ischiopagus tetrapus twins who had a common terminal ileum and colon with imperforate anus was separated at the age of 20 h. The smaller baby had congenital multiple arthrogryposis of both lower extremities, a fracture at the middle of the left femur, and a double vagina with hydrocolpos due to an imperforate hymen of the right vagina and a rectovaginal fistula on the left. Posterior sagittal rectoplasties were performed at 7 months of age in both babies. They have normal bowel movements. All four children are alive and developing normally. These are the first two case reports of successfully separated conjoined twins in Korea.
Abnormalities, Multiple↗