Postgrad Med J: first published as 10.1136/pgmj.48.562.486 on 1 August 1972. Downloaded from

Postgraduate Medical Journal (August 1972) 48, 486 -492.

The presentation and management of neonatal obstructive uropathies J. H. JOHNSTON F.R.C.S. Alder Hey Children's Hospital, Liverpool

Presentation urinary, alimentary and genital tracts share a The existence of a congenital urinary obstruction common evacuatory channel is similarly frequently may be suggested when routine examination of the associated with upper urinary tract lesions. newborn infant reveals such signs as enlargement of one or both kidneys, distension of the bladder or (3) Anomalies of the female genital tract slow, dribbling micturition. The paediatrician should Congenital absence of the vagina is associated also be alerted to the possibility of obstructive uro- with urinary tract anomalies in some 500 of cases pathy when there are present non-urological con- (Bryan, Nigro & Counsellor, 1949). A similar high genital anomalies which often secondarily involve incidence occurs with such conditions as duplication the urinary tract or which are known commonly to ofthe vagina and uterus but these may not be obvious co-exist with congenital urinary tract lesions. clinically in the young child. Secondary involvement, with obstruction, of the urinary tract occurs with space-occupying masses in Deficient abdominal mucsculature (4) by copyright. the pelvis such as hydrometrocolpos or a large intra- Urinary tract anomalies of various degrees of pelvic component of a sacrococcygeal teratoma. are The pelvic tumour, by displacing the bladder, leads severity always present. to chronic urinary retention and upper tract dilata- Cardiac anomalies tion. A congenital retroperitoneal teratoma may dis- (5) place laterally, and obstruct, one or both ureters. Mehrizi (1962) found that 23% of 279 cases of Examples of congenital anomalies which fre- malformation of the urinary tract had congenital quently coincide with congenital urological lesions heart disease. Ventricular septal defect was the most include the following. common lesion; of eighty-nine examples, twenty- two had urinary tract pathology. (1) Anomalies of the umbilicus and umbilical cord http://pmj.bmj.com/ A giant umbilical cord results from the accumula- (6) Limb deformities tion of urine within it as a consequence of a uracho- Hanke & Weicher (1965) reported that twenty-five umbilical fistula. The condition may be an isolated of 100 children with phocomelia due to thalidomide anomaly but the possibility ofan infravesical obstruc- embryopathy had urinary tract abnormalities. In tion must be investigated. Absence of one umbilical the Marfan syndrome, arachnodactyly is commonly artery is strongly suggestive of serious pathology in associated with cardiac and urinary anomalies; the alimentary and urinary tracts; Faierman (1960) Loughridge (1959) recorded eight cases with the on September 30, 2021 by guest. Protected found it to be present in 82% of cases of single um- latter. bilical artery. The baby with an exomphalos is very likely to have a urinary anomaly (McKeown, (7) Exstrophy McMahon & Record, 1953). The more severe forms of exstrophy, particularly the cloacal type, carry a 50/0 incidence of upper (2) Abnormalities of the rectum and anus urinary tract pathology (Soper & Kilger, 1964). In the Alder Hey series (Johnston, 1969) urinary tract pathology was present in 51% of cases of high (8) Gonadal dysgenesis rectal anomaly and in 430o of cases with a low, anal Turner's syndrome, which is suggested when the lesion; these did not include examples of cloacal newborn infant exhibits neck webbing, a low hair- exstrophy. The corresponding figures in Smith's line on the neck and oedema of the extremities, was (1968) series were 38% and 24%. Persistence of the associated with urinary lesions in 72% of cases in original cloacal state in the female in which the the series of Jeune et al. (1963). Postgrad Med J: first published as 10.1136/pgmj.48.562.486 on 1 August 1972. Downloaded from

Neonatal obstructive uropathies 487

In a child with one of the above congenital I_ _··· anomalies urographic investigation of the urinary .:.J tract is advisable even in the absence of symptoms .1. suggestive of urologic disease. Hypospadias, especi-

ally in its more severe forms, and undescent of the C. lieI- testis both a incidence of .clcl carry higher-than-average *1-.I rlcU* coincident urinary tract pathology but this is not 11*1

sufficiently high to warrant routine urological investi- ---·· ··uurrrr*llrLUI gation. Ascites in the newborn infant, particularly II, when associated with absent or abnormal voiding _ II, should arouse suspicion of the extravasation of urine wmr into the peritoneal cavity as a consequence of a C3 C. "- severe urinary obstruction; because of dialysis across . the peritoneal membrane the urea content of the _...... _ ,1. . ascitic fluid is generally much lower than that of the --F. _II 111 urine. o 11. Pathology . FIG. 1. Cystic, dysplastic, functionless (aplastic) kidney Urinary obstruction which originates post-natally from newborn boy with posterior urethral valves. characteristically leads to dilatation of the urinary tract above the obstructing lesion and to progressive Very often the first therapeutic needs of such thinning and destruction of the renal parenchyma as seriously ill babies are the correction of severe a result of back-pressure. Obstructions active during oligaemic dehydration and of acidaemia by the prenatal life may produce similar effects but often appropriate intravenous infusions and the control other pathological features exist. Prenatally ob- of infection by chemotherapy. Pending the results

structed ureters in addition to being dilated are often of urine, and possibly of blood, culture, the most by copyright. lengthened and tortuous and on occasions the result- effective drug is likely to be ampicillin which can be ing ureteral angulations may cause secondary ob- given by intramuscular injection in a dosage of 50 structions, especially at the pelvi-ureteric junction. mg/kg body weight daily in four divided doses. The kidneys may show, instead of, or in addition to, Peritoneal dialysis may be needed when the clinical hydronephrotic atrophy, parenchymal changes indi- or biochemical response to intravenous therapy is cative of failure of maturation; cysts of various sizes unsatisfactory and, in particular, in the presence of may be visible macroscopically, and on histological dangerous hypernatraemia or, less often, of hyper- examination primitive glomeruli and tubules and kalaemia. Commercially available packs (Dialafex, nests of cartilage may be seen. The degree and extent Allen and Hanbury; Dianeal, Baxter) are suitable for of such developmental parenchymal dysplasia vary the newborn. Up to 50 ml fluid/lb body weight is http://pmj.bmj.com/ according to the severity of the obstruction and instilled into the peritoneal cavity at each cycle and probably also according to its time of onset in foetal siphoned out after ~-j hr. life. Withsevere obstructions of early onset the entire When the child's condition has sufficiently im- parenchyma may be grossly dysplastic and the kid- proved, the appropriate urological investigations ney is then functionless (Fig. 1). Lesser degrees of can be performed. In the presence of severely im- dysplasia may, however, be found in kidneys show- paired renal function, intravenous urography re- useful functional ing activity. quires a relatively high dose of contrast medium and on September 30, 2021 by guest. Protected delayed exposures for several hours after injection; Clinical features and general management prior dehydration of the infant by withholding feeds The most severe effects of urinary obstruction in is unnecessary and may be dangerous. Endoscopy, the newborn infant are seen when the obstructive with or without retrograde pyelography, and cysto- lesion is infravesical so that both kidneys are graphy with micturating or expression cysto- affected or when upper tract disease is bilateral or urethrography are commonly needed for diagnosis involves a solitary, or solitary functioning kidney. and full assessment. Such infants generally present with manifestations of azotaemia, acidaemia and, frequently, of urinary Posterior urethral valves infection, often with complicating septicaemia. In the male, the most severe form of congenital Anorexia, vomiting and diarrhoea are usually the lower urinary tract obstruction is that caused by initial symptoms; convulsions may occur and jaun- posterior urethral valves. In the commonest variety dice and a bleeding diathesis may develop as a result of this anomaly (Type 1 of Young & McKay, 1929) of septicaemia. the mucosal valve cusps diverge downwards and Postgrad Med J: first published as 10.1136/pgmj.48.562.486 on 1 August 1972. Downloaded from

488 J. H. Johnston laterally from the lower end of the verumontanum neous ureterostomy. Through a small incision in the and meet on the anterior wall of the urethra at a flank the ureter is brought to the surface in con- lower level. As the name implies, the valves cause a tinuity, opened and sutured to the skin edges. The one-way obstruction; there is no hindrance to retro- method provides free, tubeless urinary drainage and, grade urethral instrumentation but the cusps fall in addition, allows the defunctioned ureter below together during micturition and obstruct the flow of the stoma to regain its muscular tone and contrac- urine through the urethra. Since the obstruction has tility. When the child's general condition has im- existed prenatally, the child is often born with a proved sufficiently, which may require weeks or urinary tract and kidneys already severely damaged months, and when descending ureterography shows by back-pressure. effective ureteral peristalsis and free emptying of the As a rule, the bladder is persistently distended and ureter into the bladder, the ureterostomies can be micturition is slow and dribbling. However, some closed, one side at a time. On occasions, ureter- infants can produce quite a forceful stream. The ostomy reveals that one kidney is functionless and in diagnosis is confirmed by micturating or expression these circumstances nephrectomy is indicated. urethrography which shows the typical distension of An alternative method of dealing with the de- the posterior urethra above the valves (Fig. 2a). compensated ureter has been described by Hendren Surgical treatment of urethral valves should (1970) who advocates a three or four-staged pro- generally be directed firstly to the destruction of the cedure by which both ends of both ureters are valves themselves. This is best done by endoscopic straightened and narrowed and the ureters are re- diathermy coagulation; in the infant the endoscope implanted into the bladder, employing an antireflux must be inserted through a perineal urethrotomy. technique. In general, temporary loop ureterostomy After operation, the urethral outline rapidly returns is preferable in the seriously ill infant; Hendren's to normal (Fig. 2b) and complete bladder emptying method is applicable to the less acute case. The occurs. In many instances, destruction of the valves indication for either type of ureteral surgery must, is the only surgery required but, in others, there is however, be based on the clinical state of the child

impaired emptying of the dilated ureters into the and not merely on the radiological appearance of by copyright. bladder because of decompensation of the ureteral the urinary tract since severely dilated and tortuous musculature. The resulting urinary stasis, with infec- ureters can straighten remarkably with time simply tion, is the commonest cause of failure of the child's by destruction of the valves (Johnston & Kulatilake, condition to improve after the coagulation of the 1972). valves and requires urgent relief. This is best achieved Less common causes of infravesical obstruction by performing temporary, bilateral, high-loop cuta- in the male infant include anterior urethral valve,

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'':i;':::: iC" ~ I ~~~E1O~:ll~~'i:~4~i!i~i!,~!11~.~ ~ il. :0 FIG. 2. Cysto-urethrograms in infant with posterior urethral valves. (a) Pre-operative. Severe dilatation of prostatic urethra. Reflux to very dilated ureter. (b) Six months after endoscopic diathermy coagulation of valves. No residual urethral obstruction. Postgrad Med J: first published as 10.1136/pgmj.48.562.486 on 1 August 1972. Downloaded from

Neonatal obstructive uropathies 489 posterior urethral diaphragmatic stenosis (Young's a very high vaginal obstruction, this may necessitate Type 3 valves) and posterior urethral polyp. In laparotomy; the distended vagina is opened on its general, the management of such cases is similar to anterior wall and the membrane is incised from that of posterior urethral valves; that is, the primary above, with guidance from below. As a rule, the pathology is treated initially and secondary patho- urinary tract obstruction in these babies has de- logical changes in the ureters are dealt with, if neces- veloped fairly late in prenatal life and the dilated sary,either by temporary ureterostomy or by ureteral upper urinary tract generally shows good and prompt reconstruction. improvement when the vaginal obstruction has been relieved. Hydrocolpos In the female infant, severe degrees of intrinsic Ectopic ureterocele infravesical obstruction are rare. More often, im- An ectopic ureterocele is a cystic dilatation of the paired bladder emptying results from extrinsic com- intravesical and intraurethral portions of a ureter pression. The commonestcause of this is hydrocolpos draining the upper moiety of a double kidney. The resulting from an imperforate hymen or from an twin ureters both penetrate the bladder musculature occluding vaginal septum at a higher level. The at about the usual site. The ureter from the lower normal prenatal mucoid secretions which result from hemikidney opens into the bladder; the other con- stimulation of the cervical and vaginal glands by tinues under the vesical mucosa to open ectopically maternal hormones, accumulate above the obstruct- in the proximal urethra. The ectopic ureter dilates as ing lesion so that the vagina and, to a lesser degree, a consequence of stenosis of its orifice or as a result the uterus become distended with mucinous fluid. of compression by the enclosing vesico-urethral The pelvic mass displaces the bladder upwards into sphincter musculature. The condition affects both the abdomen leading to incomplete bladder emptying males and females but is commoner in the latter. and upper urinary tract dilatation. When the ob- In some instances the pathological changes are structing membrane is low in the vagina the diag- restricted to the ectopic ureter and its related renal nosis is usually readily made by inspection of the parenchyma but frequently the ureterocele itself by copyright. vulva which reveals a bulging septum (Fig. 3); with causes secondary obstructive effects. The twin ureter a higher vaginal occlusion, vaginoscopy may be may be obstructed by the ureterocele encroaching needed. The obstruction of the genital and urinary upon and compressing its intravesical part (Fig. 4) tracts is relieved by incision of the obstructing mem- or the upper urinary tract may be affected bilaterally brane and releaseof theaccumulated secretions. With when the ureterocele obstructs the urethra and inter- feres with bladder emptying (Fig. 5). Occasionally, Y"' :·:;'"' ·· ::··..···.'·:::::id::I··,:'i-"::'i;;iiiiil6:i:iii:i,:.,i;idi:i;i:::·':·::'il:· ;'' i·· r... *·::··

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::e :::· :: ::.':' :s::·.·i :;··::: ""i'·ii:·::..;:i···:::: ·i·· .i:·.:.·· .:·".:ii ·' '·· ::·:i ...... ··:··;··:·.;:::· i:· ..r: ·:::;:'r:tii'....8;.r.p..... FIG. 4. Operation photograph showing large left ectopic ureterocele in bladder of baby girl. The probe is in the FIG. 3. Vulva ofnewborn girl with hydrocolpos. Bulging orifice of the twin ureter which was obstructed by the membrane at vaginal introitus. ureterocele. Postgrad Med J: first published as 10.1136/pgmj.48.562.486 on 1 August 1972. Downloaded from

490 J. H. Johnston and rarely shows significant improvement after the ureterocele has been removed; as a rule, removal of this hemikidney and its ureter are required. Secon- darily obstructed twin and contralateral ureters and renal segments are, however, capable of considerable ii~i -:::::,:i:I:.....:..,.... anatomical and functional improvement.

*...... Obstructed mega-ureter : :.:: . In this condition there is a functional obstruction :.,' . i .... in the lower portion of the ureter. There is, as a rule, E...... : no anatomical ureteral narrowing but an aperistaltic :::4j ::::::::"·::' ,:::i segment exists which interferes with the flow of urine. The precise pathogenesis of the condition remains ...... ~~i iii:'!ji,':.. obscure. There is an obvious similarity to Hirsch- sprung's disease of the large bowel but ganglion cells are stated to be present normally throughout the ii :: : :: ======ureter (Bischoff & Busch, 1961). Murnaghan (1957) found an abnormality in the ureteral musculature in that there is an excessive number of circular fibres in proportion to longitudinal ones. The obstruction leads to ureteral dilatation and back-pressure atrophy of the renal parenchyma. Infection commonly com- plicates the lesion and calculi may form in the dilated ureter and kidney. The obstructed mega- ureter is not commonly encountered in the neonatal

period but it may be when the condition involves a by copyright. FIG. 5. Intravenous pyelogram in baby girl with left solitary kidney or, less often, when it is bilateral. ectopic ureterocele which obstructed the twin ureter and Surgical relief of the obstruction requires that the the vesical outlet. The left upper hemikidney draining ureter above the is into the ureterocele does not concentrate the contrast obstructing segment re-implanted medium. The left lower hemikidney is severely hydro- into the bladder. Very often ureteral tortuosities nephrotic, and the right side shows moderate hydro- must be freed and a redundant portion of the ureter ureteronephrosis. The bladder is distended and the resected. The extremity of the remaining ureter is ureterocele is seen as a filling defect in the base. then narrowed and re-implanted into the bladder to lie in a submucous tunnel so that subsequent vesico- in girls, acute urinary retention may be precipitated ureteric reflux will be prevented. by the ureterocele prolapsing through the urethra to The obstructed mega-ureter has great capacity for http://pmj.bmj.com/ the exterior and becoming strangulated. improvement when the obstruction has been relieved As with other forms of congenital urinary obstruc- and quite severely dilated pyelons can return to tion, the more severe the degree of obstruction the virtually normal calibre with a corresponding im- earlier the child presents, and as a rule it is cases in provement in renal function (Fig. 6). which the ureterocele blocks the urethra that are seen most frequently in the neonatal period. Pelvic hydronephrosis Relief of the urinary obstruction is obtained by obstruction of the Congenital pelvi-ureteric junc- on September 30, 2021 by guest. Protected decompression of the ureterocele. Simple endoscopic tion may result from a variety of causes. The most incision of the ureterocele wall may rarely be re- common lesions are firstly, an angulation of the quired in the seriously ill baby with severe infection ureter on the pelvis resulting from a high ureteral but definitive surgery requires an open, transvesical origin and secondly, a ureteral kinking caused by an operation and uncapping or de-roofing ofthe uretero- accessory or aberrant artery crossing to the lower cele. The ureterocele floor remains in situ. It is im- pole of the kidney. Less frequently encountered con- portant to remove the projecting part of the uretero- ditions include a mucosal flap-valve covering the cele completely, especially in its urethral portion, orifice of the ureter in the pelvis and the persistence, otherwise folds remain which may obstruct micturi- in exaggerated form, of foetal ureteral folds. It has tion. Surgery to the ipsilateral twin ureteral orifice often been contended that the gross pathological or to the contralateral orifice may be needed in order changes at the pelvi-ureteral junction, although in to cure vesico-ureteric reflux. The parenchyma of the themselves obstructive, are in most instances secon- upper hemikidney draining into the ureterocele has, dary to a functional disorder of peristaltic conti- in the majority of cases, been irreparably damaged nuity; pressure-flow studies of hydronephrotic Postgrad Med J: first published as 10.1136/pgmj.48.562.486 on 1 August 1972. Downloaded from

Neonatal obstructive uropathies 491

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FIG. 6. Intravenous pyelograms of baby boy with obstructed mega-ureter affecting a solitary right kidney. (a) Before surgery. Ureter dilated and tortuous. Severely dilated calyces. (b) Two years after ureteral re-implantation into bladder. Ureter of normal calibre. Improvement in calyceal dilatation.

kidneys in children have, however, indicated that a consequent upon renal insufficiency, the prompt by copyright. functional obstruction is an unusual occurrence relief of obstruction and the control of complicating (Johnston, 1969). infection are of obvious immediate prognostic im- When pelvic hydronephrosis presents in early portance in cases of neonatal urinary obstruction infancy, the condition commonly proves to be bi- but the long-term outlook for the child with a con- lateral or to affect a solitary or solitarily functioning genital obstruction affecting all his renal tissue kidney. In the series of twenty-six infantile cases of Williams & Karlaftis (1966), ten were bilateral and :·:·:··: i' :i:iiiiiii:: in five the opposite kidney was afunctional. Contra- :·:'·· :s.ln; lateral renal agenesis may be present (Fig. 7) but a ·· ·'.: iii ::·.·:: ·:···:: http://pmj.bmj.com/ more common contralateral lesion is multicystic "':'"": "'"""''''';;iiiii;· ··:'i renal dysplasia associated with ureteral atresia. In i:·i,::·:ii. i· :::I::.' .·' i. five of the author's series of twenty-two cases of "·:;:' ·.:; :. multicystic dysplasia the contralateral kidney was

the site of a hydronephrosis. In such cases the multi- .:····· ..··;:. ·::.:;.i ·i cystic kidney is usually the presenting clinical feature, i:i:· '······ :····'(:; a lobulated,freely movable and often transillumin- "·';':

able loin swelling being found on routine neonatal on September 30, 2021 by guest. Protected i:l· examination. often the kidney :::ic.i·:· Very hydronephrotic ::·:··i ,··''.. ::r::i itself forms a palpable mass and urinary infection is I'... ::: .i·:···i. :·... commonly present. ::·. ::::··: :·· In the seriously ill infant with an infected hydro- ii ::···::·:·i·'··· ··:':· :i' i::: i.l.::i::······· ·:··i·:i nephrosis involving a solitary functioning kidney, i:::: nephrostomy may be needed as an emergency mea- sure (Fig. 7). Definitive reconstructive surgery for the relief of the obstruction does not differ from that performed in the older child. In most instances the Anderson-Hynes (Anderson, 1953) dismembered pyelo-ureteroplasty is very effective. FIG. 7. Pelvic hydronephrosis affecting solitary left Prognosis kidney in infant boy. Emergency nephrostomy has been The correction of fluid and electrolyte disorders performed. Postgrad Med J: first published as 10.1136/pgmj.48.562.486 on 1 August 1972. Downloaded from

492 J. H. Johnston depends mainly upon the severity of the damage in- FAIERMAN, E. (1960) The significance of one umbilical flicted upon the kidney parenchyma before birth. artery. Archives of Disease in Childhood, 35, 285. HANKE, H. & WEICKER (1965) Thalidomide embryopathies. The degree of this varies with the degree of obstruc- Renal malformations. Deutsche medizinische Wochen- tion and with its time of onset in foetal life. With an schrift, 90, 2200. early, severe obstruction such as often occurs with HENDREN, W.H. (1970) A new approach to infants with urethral the be in- severe obstructive uropathy: Early complete reconstruc- posterior valves, kidneys may tion. Journal of Pediatric Surgery, 5, 184. capable of sustaining life. In the author's series of JEUNE, M., BERTRAND, J., DEFFRERNE, R. & FORGET, M. sixty-two cases of posterior urethral valves, twenty- (1963) Frequency of renal anomalies in Turner's syndrome four boys presented in the first month of life; the in children. Annals of Endocardiology, 24, 856. mortality rate with these, over a maximal follow-up JOHNSTON, J.H. (1969) In: Neonatal Surgery (Ed. by P. P. Rickham and J. H. Johnston). Butterworths, London. of 12 years, has been 50%. At the outset, however, JOHNSTON, J.H. (1969) The pathogenesis of hydronephrosis prognostication can be extremely difficult. The initial in children. British Journal of Urology, 41, 724. azotaemia and acidaemia in the infant are in part JOHNSTON, J.H. & KULATILAKE, A.E. (1971) The sequelae of result of and infection and of posterior urethral valves. British Journal of Urology, 43, the dehydration are, 743. course, exaggerated by the immaturity of function in LOUGHRIDGE, L.W. (1959) Renal abnormalities in Marfan the newborn kidney. All infants with urinary obstruc- syndrome. Quarterly Journal of Medicine, 28, 531. tion are entitled, therefore, to an optimistic thera- MCKEOWN, T., MCMAHON, B. & RECORD, B.G. (1953) An peutic approach. Even though one must be aware investigation in sixty-nine cases of exomphalos. American Journal of Human Genetics, 5, 168. that a considerable proportion have irreparable renal MEHRIZI, A. (1962) Congenital malformation of the heart damage, many others just as ill at the time of presen- associated with congenital anomalies of the urinary tract. tation have kidneys which can respond to the relief Journal of Pediatrics, 61, 582. of obstruction and these have a MURNAGHAN, G.F. (1957) Experimental investigation of the urinary good long- dynamics of the normal and dilated ureter. British Journal term outlook. of Urology, 29, 403. SMITH, E.D. (1968) Urinary anomalies and complications in References imperforate anus and rectum. Journal ofPediatric Surgery,

3, 337. by copyright. ANDERSON, J.C. (1953) Hydronephrosis. In: Modern Trends SOPER, R.T. & KILGER, K. (1964) Vesico-intestinal fissure. in Urology (Ed. by E. Riches). Butterworths, London. Journal of Urology, 92, 490. BISCHOFF, P.F. & BUSCH, H.G. (1961) Origin, clinical ex- WILLIAMS, D.I. & KARLAFTIS, C.M. Hydronephrosis due to periences and treatment of urinary obstructions of the pelvi-ureteric obstruction in the newborn. British Journal lower ureter in childhood. Journal of Urology, 85, 739. of Urology, 38, 138. BRYAN, A.L., NIGRO, J.A. & COUNSELLOR, V.S. (1949) One YOUNG, H.H. & McKAY, R.W. (1929) Congenital valvular hundred cases of congenital absence of vagina. Surgery, obstruction of prostatic urethra. Surgery, Gynecology and Gynecology and Obstetrics, 88, 79. Obstetrics, 48, 509. http://pmj.bmj.com/ on September 30, 2021 by guest. Protected