PERIURETERITIS PLASTICA: A REPORT OF A CASE, WITH INDICATIONS OF THE PROBABLE PATHOLOGY

By WILLIAM HOUSTON, M.D., M.Ch., F.R.C.S.

Honorary Surgeon to the Bulawayo General Hospital

THE condition described as periureteritis plastica by Vest and Barclare (1 953) does not appear to have been reported in British urological literature, and as it appears to be a rare finding of unknown atiology I report a case which suggests the possible cause.

CASE REPORT The patient, a European female, then aged 45 years, had nephropexy performed in December 1951 to relieve

‘’ Dietl’s crises,” diagnosed on the basis of a thirty-year history of attacks of pain in the right loin, with occasional haematuria and vomiting. Clinical examination had shown “ visceroptosis and enlarged right kidney,” and excretion urography showed “ no organic lesion of the urinary tract ; the right kidney is ptosed and rotated on its longitudinal axis; there is no evidence of a true hydronephrosis.” The surgeon had performed a simple nephropexy, without exploration of the pelvi-ureteric region or complete mobilisation of the kidney, by reflection of a flap of posterior renal capsule which was sutured to the fascia overlying quadratus lumborum.

Excretion urography on 10th January 1952 (Fig. 1) showed the kidney in fairly good position, and the patient remained well and symptom-free until April 1952, when she had two attacks of right renal colic, with haematuria. Excretion urography at this time showed “ good function of both kidneys (Fig. 2). The right kidney remains fixed in satisfactory position. There is no evidence of obstruction or stone.” It was assumed that she had passed a small ureteric calculus, a fairly common event in these warm regions.

The patient first came under my care in November 1953, when she was referred for opinion because of occasional attacks of pain in the right loin, accompanied by the finding of coliform organisms in the urine. These attacks cleared up when the urinary infection was overcome, and this had usually been accomplished with one of the antibiotics, streptomycin, chloromycetin, and aureomycin having been used on different occasions : sulphamezathine had been used in the earlier attacks, but had been superseded later as it appeared to have lost its effectiveness. There had been no hmnaturia since April 1952, and she had no frequency or dysuria.

Clinical examination at this time showed no more than an easily palpable and tender right kidney, rather low in position, and possibly a little enlarged.

At cystoscopy, on 20th November 1953, I found a normal bladder and ureteric orifices and a 6F catheter passed easily up the right ureter for a distance of 15 cm. Ascending pyelography (Fig. 3) showed a mal-rotated kidney, with mild hydronephrosis. There was obstruction and kinking at the pelvi-ureteric junction, with dilatation of the ureter below this level almost to the level of the bladder. Attempts to pass dilators up to the renal pelvis were unsuccessful. A specimen of urine from the right ureter showed a moderate growth of coliform organisms.

Operation was advised, but the patient was not willing to have this done. However, several further attacks of right loin pain, also treated with antibiotics by her local physician, caused her to change her mind.

On 19th February 1954 I explored her right kidney through an incision in the bed of the twelfth rib; a Mayo incision had been used for the previous operation. The kidney was very adherent posteriorly and at the lower pole, and the ureter was difficult to identify. The renal pelvis was cleared, denervation as described by Oldham (1950) performed, and the ureter exposed at some distance below the kidney pelvis. As the ureter was traced upwards, it was found to become encased in a sheath of greyish tissue, rather like the cut surface of a tuberculous lymph gland before caseation has occurred. This grey tissue formed a fusiform investment of the upper 8 cm. of the ureter and pelvi-ureteric region, fading away over the pelvis proper and blending elsewhere with the somewhat indurated perinephric fat.

Delicate longitudinal incision of this tissue revealed the ureter lying in its depths; the ureter appeared normal, and was easily teased out of its bed in this unusual tissue, to which it was only loosely attached. The investment of the ureter was about 3 mm. thick in a radial direction. Further dissection allowed the abnormal tissue to be removed en bloc when it was seen to form a cast of the upper 8 cm. of ureter and pelvi-ureteric junction. This manceuvre revealed a ‘ I congenital ” stricture at the pelvi-ureteric junction with adhesions binding the ureter to the pelvis and causing it to follow an S-shaped path. There was minimal dilatation of the pelvis, and so a Mikulicz type of repair of the stricture was undertaken, using a No. 4 polythene tube as a combined splint and pyelostomy drain.

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4 R E P O R T O F A C A S E O F P E R I U R E T E R I T I S P L A S T I C A

FIGS. I to 4

Fig I : -Post-operative excretion pyelogram. one month after original nephropexy, showing rotation of kidney, but no evidence of hydronephrosis o r deformity of the upper ureter o r

pelvi-ureteric stricture. (5th January 1952.) Fig. 2.--Excretion pyelogram after renal colic four months after nephropexy. showing niininiiil dilatation of upper ureter. and suggesting. in retrospect. some kinking of the upper

ureter. (10th April 1952.) Fig. 3.--Ascending pyelogram two years after nephropexy. There is some overdistension, hut hydronephrosis. hydro-ureter. and the marked deformity of the pelvi-ureteric junction

Fig. 5.-Pos(-operative excretion pyelogram. two months after plastic operation to pelvi- ureteric junction showing no evidence of hydronephrosis o r obstruction. (29th April 1954.)

are well shown. (20th November 1953.)

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40 B R I T I S H J O U R N A L O F U R O L O G Y

She had been given caps. terramycin, 250 mg. six-hourly for twenty-four hours pre-operatively, and these were continued for five days post-operatively. She made an uneventful recovery, the polythene tube being removed on the fourth day, and she was discharged from hospital on the tenth post-operative day with the wound well healed.

A post-operative excretion urogram on 26th April 1954 (Fig. 4) showed “ good function of both kidneys. The hydronephrosis has diminished. There is no evidence of stricture at the pelvi-ureteric junction.”

Histological examination of the excised tissue showed “ a portion of fibro-fatty connective tissue, showing no specific pathological features.” Review of the sections recently has not revealed any more precise pathology.

DISCUSSION

The present case differs considerably from those originally described by Vest and Barclare (l953), and even more from the case of “ perinephritis plastica ” reported by Paul1 et al. (1959, yet presents sufficient points of resemblance to make it reasonable to assume that they are all stages in the same pathological process, whatever that may finally prove to be. All these writers suggest that the basic process is infective, but are unable to find much pathological or bacteriological evidence for this. Vest and Barclare state that “ from pathological examination it is reasonable to assume that infection of some unknown type, probably occurring at some remote period, is the underlying ztiological mechanism.” However, they were never able to demonstrate gross infection at the time when their four cases came under their care. They further state that “ in view of the foregoing findings it is impossible to establish any single bacteriological common denominator, although from the above data one might consider an ascending streptococcal periureteral infection from lower tract infection as the possible aetiologic process.” And later, that “ one must consider in a process of this sort the possibility of the so-called ‘ abortive infections ’ which have been well described following administration of various chemotherapeutic agents. We recently encountered a case of marked perirenal fibrosis in which an acute perirenal infection had evidently been aborted by treatment with a number of antibiotics. This mechanism, however, does not account for the type of tissue in these cases, because Cases 2 and 3 did not receive any chemotherapy. The first and third patients in our series did receive sulpha drugs for a short period early in their medical treatment, but not during any acute phase of urinary tract infection.”

Paul1 et al. state that “ an exhaustive history failed to reveal anything suggesting a perirenal haematoma or a perinephric abscess ” in their case, and later, that “ the present case gave no history of urinary tract disease except for one episode of gonorrheal urethritis treated with sulpha drugs fifteen years previously. . . . On the other hand, the patients with periureteritis plastica gave past histories of urinary tract disease. Of questionable significance is the fact that all Vest and Barclare’s cases involved the right side, while in ours the pathological process was present on the left.”

In the case which I am reporting, one of the main differences from the cases of Vest and Barclare is that an intrinsic obstruction at the pelvi-ureteric junction was discovered, whereas in theirs the obstruction was purely extrinsic. However, this did not appear on the excretion pyelogram prior to her nephropexy, and it seems reasonable, therefore, to assume that it was at least aggravated by the later development of the extrinsic obstruction. Moreover, although it may well have been overlooked at the time of this operation, the surgeon concerned, a man of considerable experience, did not remark any unusual process at that time, which suggests that it was not present then ; but it was definitely present later when there was a definite history of repeated severe urinary tract infection, with energetic treatment with sulphonamides, and more particularly, with various antibiotics.

The appearances at operation strongly suggested to me the type of inflammatory connective tissue seen in infective lesions, which finally come to surgery after treatment with antibiotics, such as mammary abscess or cervical adenitis. The naked-eye appearances would have been consistent with tubercle or other granulomatous process.

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Although the sulphonamide group of drugs have been in use a long time, antibiotics, especially those effective against the organisms usually found in infections of the urinary tract, have been generally available only for a much shorter period. The fact that the type of infection described is beginning to appear only now, after a considerable interval of time, supports the theory that this type of lesion is due to infection more or less controlled by antibiotics (or sulphonamides), but in effect, a chronic infection, producing the changes which we associate with granulomatous infection.

SUMMARY

1. A case is described of a condition not previously reported in British urological literature,

2. It is suggested that the lesion is due to infection partially controlled by antibiotics, leading resembling the “ periureteritis plastica ” of Vest and Barclare (1953).

to pathological changes resembling those associated with the infective granulomata.

ADDENDUM

Since the preparation of this report, the patient has undergone right nephrectomy for calculus impacted in the lowest minor calyx.

Histological examination of the kidney showed only the presence of “ marked interstitial nephritis,” and sections from the pelvis and ureter showed only chronic inflammatory changes. As would be expected, there were very dense adhesions between the lower half of the kidney and the perinephric tissues, but there was no evidence, at this time, of the abnormal tissue found previously.

REFERENCES

OLDHAM, J. B. (1950). Ann. R. Coll. Surg. Engl., 7, 222. PAULL, D. P., CAUSEY, J. C., and HODGES, C. V. (1955). J. Urol., 73, 212. VEST, S. A., and BARCLARE, B., jun. (1953). J. Urol., 70, 38.