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Pregnancy, delivery and puerperium in primigravida after surgical correction of vesical exstrophy and reconstruction of vagina with chronic renal insufficiency was presented. Both pregnancy and delivery were successful. Vesical exstrophy the rare congenital malformation in aspect of obstetrical-gynaecological treatment were discussed. Conduction of pregnancy with chronic renal disease was briefly reviewed.
PURPOSE: Variants of the bladder/cloacal exstrophy complex are rare. Different presentations and subsequent management and outcome are discussed. MATERIALS AND METHODS: We performed a retrospective review of our database of more than 815 patients with the exstrophy complex. Patients with variants of classic epispadias or bladder or cloacal exstrophy were identified. Anatomical presentation, surgical management, type of continence procedures and final outcome were evaluated. RESULTS: Of the 25 patients with variants 13 were treated primarily at our institution and 12 were referred. Time until primary bladder closure ranged from 1 day to 4 years. Followup after continence procedure ranged from 1 month to 39 years. Seven of the 25 patients are awaiting a continence procedure. Six patients are dry without a continence procedure, of whom 4 have superior vesical fistulas. A total of 11 patients underwent bladder neck reconstruction (BNR), of whom 3 are dry, 2 are dry during the day but are wet at night, 1 had a failed procedure and 5 are dry after continent diversion (CD). One additional patient underwent CD initially and is dry. Referred cases of epispadias with bladder prolapse were not recognized at birth and had delayed closure. Impaired bladder growth or failed BNR required CD in 4 patients, and 2 are awaiting a continence procedure. Skin covered and duplicate exstrophy had comparable outcomes to the classic presentations. Duplicated organs were used for reconstructive procedures. Of the 6 patients with cloacal variant 2 are continent of stool and 2 await a Pena procedure. One of these patients has an ileal stoma and 1 has a colostomy. CONCLUSIONS: The initial presentation of exstrophy variants can be confusing, often delaying initial treatment. Superior vesical fistulas permit continence without BNR due to an intact urinary sphincter. Variants such as epispadias with bladder prolapse and duplicate or skin covered exstrophy should be closed at birth with standardized techniques to promote bladder growth for later BNR. These cases are faced with the same long-term problems as the classic presentation. Cloacal variants can present with intact anal innervation, allowing a later Pena procedure.
OBJECTIVES: Intestinal metaplasia often coexists with adenocarcinoma of the urinary bladder, suggesting to some investigators that it is premalignant. However, the natural history and long-term outcome of intestinal metaplasia in isolation are unknown. We report 53 cases of intestinal metaplasia of the urinary bladder followed for more than 10 years. METHODS: We reviewed the Mayo Clinic surgical pathology files between 1926 and 1996 and all patients with exstrophic bladder recorded in the files of the Hospital for Sick Children (Toronto, Ontario, Canada) and Dallas Children's Hospital (Dallas, Texas) between 1953 and 1987, and identified all patients with intestinal metaplasia of the bladder. RESULTS: A total of 53 cases were identified from both series, and none of the patients developed adenocarcinoma of the bladder. The Mayo Clinic series consisted of 24 patients. Nineteen of the 24 (79.1%) were alive without evidence of cancer (median follow-up 14 years, range 0.9 to 53), and 5 patients died of intercurrent disease (at 0.9, 4, 8, 11, and 53 years after diagnosis) without evidence of bladder cancer. The Dallas Children's Hospital and the Hospital for Sick Children series consisted of 29 patients. Twenty-seven of the 29 (93.1%) were alive without evidence of cancer (median follow-up 13 years, range 3 to 23.9). Two patients died of trauma (at 10.9 and 12 years after diagnosis) and at autopsy had no evidence of bladder cancer. CONCLUSIONS: Intestinal metaplasia of the urinary bladder is not a strong risk factor for adenocarcinoma or urothelial cancer.
Four of five patients with cloacal exstrophy seen at the Massachusetts General Hospital since 1978 have had pelvic kidneys. This association is discussed and a possible embryological explanation considered.
Mid-ileal conduit stenosis is an unusual yet morbid complication following ileal conduit urinary diversion. We report the cases of four patients who developed mid-ileal stenosis at an average interval of 19.5 years after urinary diversion, with the longest interval being 25 years. This series emphasizes the importance of life-long follow-up of patients who have had urinary-intestinal diversion.
Pseudoexstrophy is a rare, mild exstrophy variant which involves the major musculoskeletal defects of the exstrophy complex without any associated defect in the urinary system. A case is reported presenting at birth as an umbilical positional anomaly. Differential diagnosis and management are reviewed.
We report a case of a male newborn with complete epispadias and an umbilical lesion. Histological examination of the surgically removed lesion revealed a colonic remnant with no communication to the underlying bowel. This entity is one of the exstrophy/epispadias complex variants known as covered exstrophy with sequestered bowel, and is case 4 in the world literature. Simple excision of the lesion was accomplished with staged correction of the epispadias planned.
We report a case of a male newborn with asymmetrical epispadic diphallia and congenital urethral fistula, and exomphalos minor. A segment of bowel was attached in the region of the pubis; with no communication to the underlying bowel. This was excised; histological examination revealed this to be a colonic remnant. The excision of the examphalos minor and approximation of the pubis and the rectus sheath over the bladder was also done; urethroplasty is planned at a later date. A comprehensive review of the available literature revealed only eight such cases before this; the possible embryology is discussed.
We describe a case of cloacal exstrophy what was diagnosed prenatally by ultrasound and confirmed at birth. We also review the literature on 15 previous prenatally diagnosed cloacal extrophy cases.
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OBJECTIVE: To compare the histology of gonads excised from males with cloacal exstrophy (the most severe variant of the exstrophy-epispadias complex) undergoing early gonadectomy and gender reassignment (to obviate difficulties in reconstructing male external genitalia) with those published previously. PATIENTS AND METHODS: Testicular tissue was obtained from males with cloacal exstrophy undergoing gender reassignment shortly after birth. Slides were stained for histological evaluation and reviewed by one author. The histology was compared with known normal features for newborn testicular tissue. The mean age at the time of gender reassignment was 6 months. RESULTS: There were no significant abnormalities in the architecture of the rete testis, epididymis or vas of the study specimens. There were more germ cells in some patients with cloacal exstrophy, but this finding was not specific and is of unknown clinical significance. CONCLUSIONS: Despite the severity of the malformations in cloacal exstrophy, testicular histology is preserved. In highly selected genotypic males who have adequate phallic structures, congruent gender assignment is appropriate and early orchidopexy is recommended.
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Mucinous adenocarcinoma is a rare entity within the group of primary adenocarcinoma of the bladder which represent 0.5-2% of all malignant epithelial bladder tumours. In spite of the rarity of this tumoral type; it is a poor prognosis entity mainly due to its diagnosis especially in advanced stage of the disease. There is no general agreement on the treatment of adenocarcinoma of bladder. Not withstanding surgery would be the only curative treatment, although unfortunately, it is curative in just a few cases. We report six cases with mucinous adenocarcinoma of the bladder attended in our Department in the last ten years (january 1991-december 2001). In one of them a radical cystectomy was performed, while transurethral resection with or without adjuvant treatment was practiced in the other one. Only one patient is alive today, namely, the one where the tumour not invade the muscular tissue. These findings show the discouraging results of this entity closely intertwined with the pathologic stage.
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The congenital defects in the pelvis and perineal area, which are derived from defective development of the cloacal membrane, the urorectal septum, the mesonephric and paramesonephric (müllerian) ducts and the urogenital sinus, have been described. These may be life threatening, are often complex, and are certainly devastating to the parents. With knowledge of their embryologic origin, of their anatomy in detail, and the effect on function and drainage, treatment can be planned so that a good quality of life usually can be expected. Improved reconstructive programs and techniques can usually achieve near-normal appearance and function, with the expectation of good social adjustment.