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Gas in the urinary tract
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BY II. P. KENT RADIOLOGIST~ MEDICAL ARTS CLINICj REGINA, SASKATCFIEWAN
PNEUMATURIAis usually defined as the presence of air or gas in the voided urine. The gas may come from the kidneys, the ureters, or the bladder. Senator (I89i) first classified pneumaturia as : - I. Following instrumental introduction into the urogenital tract. 2. Resulting from fistulae. 3. The result of fermentation, i.e., b y the actmn of organisms u p o n the urine. This group, which many writers classify as primary pneumaturia, is now subdivided into : (a) fermentation with glycosuria ; and (b) fermentation without glycosuria.
Fig. 36.--Case L Straight film of abdomen, ill-defined mass in left renal region, surrounded by colon inferlorly a~d laterally,
Fag. 37.--Case I. 4 ° rain. I.V.P. Homugeneous collection ot gas in left renal region. Colon essentially in position as in ,Fig. 36.
Pneumaturia is also a valuable radiological sign. Gas in the urinary tract is rarely recognized on a straight radiograph of the abdomen, partly because it may be mistaken for intestinal gas, partly because it may be missed unless the observer is aware that gas can be present in the urinary tract. It is the purpose of this paper to draw" attention to the significance of gas in the urinary tract, and to illustrate some of the problems arising from this finding by the case histories of 4 patients seen in the last few years.
CASE REPORTS Case i . - - R e n o c o l i c
fistula.
M r s . L . S., w i d o w , a g e d 72, w a s a d m i t t e d t o H a c k n e y H o s p i t a l o n M a y 5, I 9 5 2 ,
complaining of frequency of micturition, h~ematuria, and dysuria of one week's duration. She had suffered repeated attacks of pain in the left loin over the last twenty-five years, but had never passed any stones. During the last week she had typical left renal pain. She did not vomit and there had been no pneumatttria. On examination she was found to be ill and pyrexial. In the chest there were signs of bilateral basal bronchopncmnonia. In the distended and tender abdomen a doubtful mass was felt in the left upper quadrant.
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T h e u r i n e c o n t a i n e d a l b u m i n , b u t c u l t u r e was sterile. T h e w h i t e b l o o d - c e l l c o u n t w a s 12,ooo, of w h i c h 85 p e r c e n t w e r e p o l y m o r p h s . A r a d i o g r a p h of t h e a b d o m e n (Fig. 36) s h o w e d a n ill-defined m a s s in the left renal region, skirted b y the c o l o n i n f c r i o r l y a n d laterally. F o l l o w i n g a d m i s s i o n she r e m a i n e d p y r e x i a l a n d h a d i n t e r m i t t e n t s h i v e r i n g attacks. O n M a y ~8 she was f o u n d in a pool ot u r i n e in h e r b e d twice w i t h i n t w e n t y f o u r h o u r s , a n d a l t h o u g h t h o u g h t to be i n c o n t i n e n t b y the n u r s i n g staff was a d a m a n t t h a t she v o i d e d t h e u r i n e p e r r e c t u m . F o l l o w i n g this significant event h e r t e m p e r a t u r e settled g r a d u a l l y . A n i n t r a v e n o u s p y e l o g r a m w a s p e r f o r m e d o n M a y 28. T h r o u g h o u t t h e film series a large oval loculus of gas w i t h w e l l - d e f i n e d m a r g i n a n d of u m f o r m a p p e a r a n c e was n o t e d in t h e r e g i o n p r e v i o u s l y o c c u p i e d b y t h e
Fig. 38.--Case I. Circumscribed collection of gas anterior to and overlapping the lumbar spine in the lateral view.
Fig. 39. Case t. Barium enema. Loculus ofgaspersists in same position, almost surrounded by colon, with slight distornon of colon at one point. Fistula not shown.
ill-defined m a s s (Fig. 37). N o excretion t o o k place o n t h e left side a n d v e r y p o o r excretion o n t h e right. T h e b l a d d e r yeas opacified at 20 m i n u t e s . L o c a l i z a t i o n of t h e loculus of gas s h o w e d this to be l y i n g anterolateral to t h e l u m b a r spine (L.~, 2, 3) (Fig. 38). Clinically she was t h o u g h t to be suffering f r o m either a perinepl~ric abscess or a closed p y o n e p h r o s i s . W i t h the h i s t o r y of p a s s i n g u r i n e p e r r e c t u m in large quantities a n d the a p p e a r a n c e s o n the I.V.P., a radiological diagnosis of a renocolic fistula w a s considered, a n d a b a r i u m e n e m a s u g g e s t e d . T h i s was d o n e on M a y 3 o, b u t a p a r t f r o m a v e r y slight d i s t o r t i o n of t h e distal t r a n s v e r s e colon w h e r e it skirted t h e still visible loeulus of gas, n o a b n o r m a l i t y c o u l d b e f o u n d . T h e p o s t - e v a c u a t i o n film was u n s a t i s f a c t o r y (Fig. 39), the p a t i e n t hardly- v o i d i n g a n y b a r i u m , a n d s h o w e d n o evidence of a fistula. I n view of this essentially negative e x a m i n a t i o n it wa~ c o n s i d e r e d m o r e likely t h a t t h e p a t i e n t w a s suffering f r o m a p y o p n e u m o n e p h r o s i s . D u r i n g t h e n e x t five days t h e p a t i e n t ' s t e m p e r a t u r e steadily rose a n d t h e surgical i n d i c a t i o n for d r a i n a g e was clear-cut. P r i o r to incision a f u r t h e r film was r e q u e s t e d - - a n d this s h o w e d b a r i u m in a v e r y large a n d o b v i o u s l y h y d r o n e p h r o t i e k i d n e y (Fig. 40), giving u n d i s p u t e d p r o o f of a renocolie fistula_ A s p i r a t i o n of t h e left k i d n e y yielded gas, pus, b a r i u m , a n d f o u l - s m e l l i n g urine. A m p l e d r a i n a g e w a s instituted. P o s t - o p e l ~ t i v e l y h e r p r o g r e s s was slow b u t satisfactory, so t h a t b y S e p t e m b e r n e p h r e e t o m y c o u l d b e p e r f o r m e d , at w h i c h t i m e t h i c k adhesions to t h e eolon w e r e f o u n d . A small p o r t i o n of t h e pelvis of t h e left k i d n e y r e m a i n e d a d h e r e n t to t h e colon at t h e p r e s u m e d site of t h e fistula. T h e fistula, was n o t p a t e n t . T h e r e m o v e d k i d n e y s h o w e d gross h y d r o n e p h r o t i c changes_ T h e p a t i e n t m a d e a n u n i n t e r r u p t e d recovery.
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C o r n m e n t . - - T h i s p a t i e n t ' s clinical features indicated a l o n g - s t a n d i n g 1eft u r i n a r y infection, and surgical o p i n i o n f a v o u r e d a p e r i n e p h r i c abscess or a p y o n e p h r o t i c kidney. The demonstration of the loculus of gas in the left renal region, in an area w h e r e p r e v i o u s l y an indefinite m a s s had been present, raised t h e differential diagnosis of a renoc01ic fistula a n d a p y o p n e u m o nephrosis. T h e h i s t o r y of the passage of u r i n e p e r r e c t u m , followed b y the a p p e a r a n c e of the loculus of gas, clearly indicated drainage of the left kidney into t h e gut, either t h r o u g h a p r e existing fistula or by the f o r m a t i o n of a fistula. It was therefore particularly d i s a p p o i n t i n g to iail to d e m o n s t r a t e this fistula at first b y the b a r i u m enema. E v e n m o r e s u r p r i s i n g was the virtual lack of distortion of the colon at the site of the fistula, a p o i n t particularly s t r e s s e d b y F e l d m a n (1937). N o d o u b t the explanation in this p a t i e n t was t h a t t h e fistula was very small in size a n d t h a t it closed after drainage of the kidney, since t h e p a t i e n t at no t i m e following the first operation p a s s e d any intestinal c o n t e n t s f r o m the drainage site. Case 2 . ~ e o v e s i c a l fistula. Mr. W. H., aged 23, was admitted to Hackney Hospital in June, 195 i, with a 3-year history of recurrent attacks of intestinal colic, watery diarrhoea, anorexia, general malaise, drenching night sweats, and fever. A perineal abscess was opened two years previously, and a pelvic abscess d r a i n e d s p o n t a n e o u s l y p e r r e c t u m . A barium Fig. 4 o. C'ase ~. Five days later, Barium in ~rossly hydromeal and enema at another hospital proved ineonncphrotic kidney. elusive. Pneumaturia was first noted three months before admission, associated with the passage of intestinal contents per urethram, frequency, and dysuria. On examination the only significant findings were a tender fixed mass felt per rectum in the rectovesical pouch, and pneumaturia at the end of micturition, the urine containing flecks of solid material. A provisional clinical diagnosis of a vesico-intestinal fistula probably due to Crohn's disease was made. Laboratory investigations showed protein in the urine, pus cells, and Bact. col~ communis on culture. The h~emoglobin was 12'5 g., the total white cells amounted to e i , 2 o e , of which 92 per cent were polynlorphs. A straight film of the abdomen showed some gas of uniform translucency in a normal-sized bladderRenal function and outlines were seen to be normal on both sides on intravenous pyelography. An erect view of the bladder demonstrated a gas and fluid level (F~'. 4I), but no opaque medium was seen outside the renal tract. A barium meal, followed through, showed a normal stomach and duodenum. The csecum was reached at 4 hours, with the patient fasting. There was gross distortion of the terminal ileum, which was ragged in appearance, and there was a suggestion of several sinuses. The c~ecum was pulled medially, but the ileocsecal valve could not be identified. The bladder filled wlth barium before the descending colon had filled, but the actual fistula was not shown (Fig. 42). With a barium enema no overflow into the small intestine occurred. The deformity of the c~ecum was confirmed. No vesiooealie fistula was demonstrated. On these radiological findings Crohn's disease with an ileovesical fistula was thought to be the most likely diagnosis. The patient developed an anterior perineal abscess requiring drainage. On culture of the pus B a s t . cell communis and B. proteus were obtained. This abscess prevented eystoscopy, but sigmoidoscopy revealed no significant feature. Laparotomy to close the fistula showed a somewhat dilated and kinked loop of terminal small gut, which was densely adherent to the bladder. The csecum was pulled medially, and the appendix could not be idannfied. There was no macroscopical evidence of Crohn's disease. The fistula between the bladder and terminal jleum was identified and closed. A further lice-ileal fistula was closed. An ileotransverse colostomy was performed because of the distortion of the ileum following the closure of the fistula. Biopsy from the edge of the fistula showed purulent granulation tissue.
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Fig. 4I.--Case z.
E r e c t v i e w of b l a d d c r at end of I . V . P . examination, d e m u n ~ t r a t i n g gas a n d fluid level.
Fig. 43.--Case 3. a o - m i n u t e I.V.P. film. H o m o g e n e o u s o u t l i n i n g of b l a d d e r a n d left u r e t e r w i t h gas. Bilateral h y d r o n e p h r o t i c change~.
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[qg. 42.---Cose ~. B a r i u m m e a l ftnllcw-through, 4 hr. p.o. D i s t o r t e d ileocmcal region, m e d i a l dq~pl,neement of ea~'cum, a n d p r o b a b l e sinuses. A c t u a l fistula w i t h b l a d d e r n o t s h o w n , b u t b m ' i u m present ila t h e bladder.
Ielg. 44.--Case 4-
S t r a i g h t filnl Gf abdolzmn. M o d e r a t e l y large bladder, completely filIed w i t h gas.
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Post-operatively an intraperitoneal abscess developed in the ileocmeal region, and a faecal fistula developed following drainage. This fistula closed spontaneously. Soon after laparotomy the urine cleared, the patient gained weight, and felt fit. On follow-up examination he stated that he still occasionally suffered from slight pneumaturia but no other symptom. His urine, however, remained clear, and he passed no further intestinal contents per urethram. Comment.--This m a n w a s t h e o n l y p a t i e n t in t h i s series w h o c o m p l a i n e d of p n e u m a t u r i a . Clinical e v i d e n c e gave a s t r o n g lead to t h e c a u s e of t h e p n c u m a t u r i a , a n d t h e pl-cscnce of a n i n t e s t i n o vesical fistula w a s a s s u m e d , in v i e w of t h e p a s s a g e of i n t e s t i n a l c o n t e n t s p e r u r e t h r a m . Radiological i n v e s t i g a t i o n s were p r i m a r i l y called for to d e t e r m i n e t h e site a n d c a u s e o f t h e fistula. F o l l o w i n g these, C r o h n ' s d i s e a s e w a s t h o u g h t to be t h e m o s t r e a s o n a b l e p r e - o p e r a t i v e d i a g n o s i s for t h e f o r m a tion of t h e p r o v e n fistula, b u t o p e r a t i o n s h o w e d n o t h i n g to s u p p o r t this. A s j u d g e d f r o m t h e b i o p s y s p e c i m e n a b e n i g n i n f l a m m a t o r y lesion m u s t be a c c e p t e d as t h e final diagnosis, and, s i n c e t h e a p p e n d i x could n o t be identified, it w o u l d a p p e a r likely t h a t a c h r o n i c s u p p u r a t i v e a p p e n d i c i t i s was t h e u n d e r lying cause. T h e difficulty in d e m o n s t r a t i n g the actual fistula is well s h o w n in this case, a l m o s t certainly o w i n g to t h e g r o s s d i s t o r t i o n o f t h e a n a t o m y of t h e t e r m i n a l s m a l l i n t e s t i n e . C a s e 3 , - - U r e t e r o - r e e t o - v a g i n a l fistula. Mrs. L_ P., aged 59, was first seen at Hackney Hospital in September, ~95 o, and presented the typical symptoms and signs of a carcinoma of the cervix. An intravenous pyelogxam at this time was normal. In October of that year a Wertheim's operation was performed. During the operation the left ureter appeared infiltrated. Part of the anterior wall of the rectum was removed accidentally during removal of the pelvic viscera. This rent was repaired with continuous cbronnc catgut sutures. T h e biopsy report was " solid, trabecular, squamous-celled carcinoma of the cervix, grade z ". T h e general condition post-operatively was satisfactory, but nine days after operation she passed stools per vaginam. This gradually cleared, and she was discharged on Jan. I9, I95I. She was re-admitted five days later becauae " urine just ran away from h e r " and at times it was " thick ". There was no faecal incontinence. At this time her blood-urea was 69 my. per cent. An intravenous pyelogram was performed on Feb. IS. Throughout the series the bladder was outlined by gas. In addition the left ureter wasMso clearly outlined by gas up to the renal pelvis (Big_ 43)- Excretion on both sides was poor early on, but at 20 minutes both kidneys showed hydronephrotlc changes. T h e radiologlcal conclusion was that a fistula existed between the left ureter and either the gut or the vagina, allowing air to enter the urinary tract. T h e site of the fistula was not established. Cystoscopy was performed twice after the radiological investigation. On the first occasion the bladder was dirty and exhibited cystitis. Both ureteric catheters were held up in the bladder wall. T h e vagina contained urine and faeces. On the second examination a large hole was seen between the upper vagina and rectum, and urine was present m the rectum, Again the catheters would not pass up into the ureters. Surgical opinion favoured a fistula between the lower left ureter and the rectum, the urine entering the vagina via the rectovaginal fistula. On April 5, the rectovaginai fistula was excised and closed, the vagina being readily separated from the rectmn. Apart from a urinary infection the patient did well post-operatively. T h e repaired fistula remained elosed. It was considered that the left ureter drained into the r e c t u m - - a m o u n t i n g to a ureterie transplantation. eemrnent.--Several p o i n t s r e m a i n s p e c u l a t i v e in this patient. I t is n o w c o n s i d e r e d t h a t a barium enema probably would have demonstrated the fistulous communication between the upper r e c t u m a n d t h e left u r e t e r . T h i s fistula w a s a s s u m e d in v i e w of t h e p r e s e n c e of u r i n e in t h e r e c t u m ; its cause is u n c e r t a i n , b u t it w a s p r o b a b l y n e o p l a s t i c in origin r a t h e r t h a n t r a u m a t i c , s i n c e t h e r e was n e o p l a s t i c infiltration of t h e l o w e r e n d o f t h e left u r e t e r at t h e t i m e o f t h e o p e r a t i o n . T h i s fistula w o u l d e x p l a i n t h e p r e s e n c e o f gas i n t h e left u r e t e r as s h o w n o n t h e I,V.P. T h e r e c t o v a g i n a l fistula w a s m o s t likely t r a u m a t i c in origin, in v i e w o f t h e a c c i d e n t a l d a m a g e to t h e a n t e r i o r rectal wall. T h i s s e e m s c o r r o b o r a t e d b y t h e a b s e n c e of o b v i o u s infiltration of t h e r e c t a wall as well as b y t h e fact t h a t t h e r c c t o v a g i n a l fistula d i d n o t r e c u r f o l l o w i n g repair. T h e p r e s e n c e of gas in t h e b l a d d e r r e m a i n s u n e x p l a i n e d . C y s t o s c o p i c e v i d e n c e r u l e s o u t a fistula b e t w e e n t h e b l a d d e r a n d a g a s - c o n t a i n i n g v i s c u s . S o m e air m i g h t h a v e e n t e r e d t h e b l a d d e r f r o m t h e left u r e t e r , w h i c h was filled b y colonic gas, b u t t h i s is n o t w h o l l y c o n v i n c i n g , s i n c e n e i t h e r of t h e u r e t e r i c orifices c o u l d be p a s s e d b y c a t h e t e r s . N e v e r t h e l e s s t h i s p o s s i b i l i t y c a n n o t be r u l e d out. It is m o r e likely t h a t air w a s i n t r o d u c e d into t h e b l a d d e r f r o m w i t h o u t b y u r e t h r a l c a t h e t e r i z a t i o n at a n u n k n o w n
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time. I n the absence of autopsy control this case, therefore, is tentatively classified as one of a uretero-recto-vaginal fistula. Case 4---"Primary p n e u m a t u r l a " of t h e Madder. Mr. L K., aged 40, was first seen at the Medical Arts Clinic in Regina in April, I95o, when he complaincd of dizzy spells, nocturia, cloudy urinc, and loss of energy. He thought he had passed a stone the previous winter_ Following trauma to the right kidney in I939 a right nephrectomy had been performed. Examination of the urine revealed glycosuria, as well as pyuria. An intravenous pyelogram demonstrated poor function and moderate dilatation of the calicine system in the remaining left kidney. No opaque urinary calculi were seen. Throughout the examination the bladder was seen to be filled by gas (Fig. 44). Cystoscopy two weeks later demonstrated a double stricture of the urethra, and a foul, greyish slough covering the major portion of the floor and part of the lateral walls of the bladder. It was impossible to identify the ureteral orifices. The subsequent course of the patient is not pertinent to this paper, except that films of the abdomen obtained in July 195o, and again in the summer of 1953, did not reveal recurrence of gas in the bladder~ the patient requiring extensive treatment fur repeated urinsry infections. On tw0 occasions coliform bacilli were cultured from the urine. C o m m e n t . - - T h e patient was not fully investigated radiologically, b u t he seems to fit rather well with the criteria laid down by A r t h u r and Johnson (i948). A diabetic, the subject of two urethral strictures, he did not volunteer whether he passed gas during micturition. T h e I.V.P. showed gas in the bladder in such amounts that the condition could hardly be overlooked, or mistaken for gas in the intestine. A fistula with one of the n e i g h b o u r m g gas-containing viscera was not excluded radiologically, b u t no history of a fistula was elicited, and cystoscopy failed to reveal one. Although a fistula could not be ruled out completely, in view of the extensive greyish slough covering a large part of the bladder wall, tile subsequent course, repeated negative cystoscopies, and the absence of gas in the bladder on later films of the a b d o m e n would exclude it. At the original examination urine culture was not done, b u t it seems reasonable to assume that Bact. coE communis, found on cultures at a later stage, was the organism responsible for the fermentation of the urine in this diabetic patient.
THE L I T E R A T U R E Although a very extensive medical and surgical literature exists on the subject of pneumaturia, little is said about the radiological manifestations of gas in the urinary tract in text-books of radiology. Only Wesson (I946), in his book, mentions spontaneous gas formation in the kidney and gives an excellent example of a pneumonephrosis (the case of Math~ and E. de la Pena (i933)). I n s t r u m e n t a l pneumopyelography, however, is occasionally mentioned. Some recent papers have described the radiological demonstration of gas in part of the urinary tract. Spontaneous gas formation in the kidneys was first diagnosed by radiography by Randall (i927) , and was confirmed by operation. A most interesting case was presentcd by Surraco (I932). I n his patient s u p p u r a t i o n within the cavity of a huge renal hydatid cyst resulted in gas formation, shown radiographically as a gas and fluid level. Math& and E. de la Pena (T933) reported a case of p n e u m o n e p h r o s i s - - a pyonephrosis in the u p p e r pole of a duplex left kidney, with gas formation. Alexander (i94 z) described a further case of pneumonephrosis in a diabetic woman. Excellent radiographs became available during the course of the illness, showing first a spontaneous gas pyelogram on t h e left side three days after the passage of a calculus, later that the gas extended and eventually surrounded the whole kidney. Bassalleck (i95~) described a case of bilateral renal caleuh, those o n t h e left being surrounded by gas. He considered this to be due to fermentation in the infected kidney. T h e spontaneous presence of gas in the bladder was first diagnosed by X rays by Mulsow and Gillies (~934) in a non-diabetic patient. Riley and Bragdon (I937) mention the demonstration 0~ gas in the bladder in a diabetic suffering From cystitis. A r t h u r and Johnson (i948), Danzer (~95o), and lately Fineman, Ferher, and Roginsky (1952), have described the presence of gas in the bladder
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shown radiographically under the temls ' spontaneous pneumaturia ', ' pneumaturia ', and ' primary pneumaturia '. In view of the large literature on urinary-intestinal fistula:, it is surprising how rarely gas has been noted or commented upon in these cases. Parks (1951) described intermittent spontaneous pneumo-urography following end-to-end ureterosigmoid transplantation in 6 out of 33 cases. Faingold, Hansen, and Rigler (1953) recently reviewed the literature on cystitis emphyscmatosa and reported 4 cases diagnosed radiologically. Pathology.--Fistul~e, irrespective of their underlying cause, can connect almost any part of the u r i n a l - tract with adjacent viscera, if these contain air or gas it may escape into the urinary tract and so become radiologically demonstrable. By far the commonest of the urogenital tract fistulae are the vesico-intestinal fistula. Higgins (r936) analysed the aetiological factors in 328 cases described in the literature_ The majority were of an inflammatory basis (5 ° per cent), and of these two thirds were due to diverticulitis of the colon. Next came neoplasms, followed by traumatic and congenital fistulae. Renovisceral and ureteroviscerai fistula~ are uncommon. They are not a disease sui generis, but a complication of an already existing disease. Wesson (1938) considered that rer~ocolic fistul~e, the commonest renovisceral type of fistula, were more frequent than generally believed, but Ratcliff and Barnes (1939) only found 38 cases at the tinle of their report. In addition to these authors Hirsch and Bass (i937) , Weiser (1929) , Higgins and Hicken (1933) , Deuticke (i935) , and Feldman (1937) stress the fact that it is generally primary renal pathology in the form of pyelonephritis, pyonephrosis, tuberculosis, or nephrolithiasis with infection which causes the formation of a renocolic fistula, once a perinephric extension of the infection has been established. Fistul~ between the kidney and the stomach have been described by Wildbolz (1937-8), the duodenum by Pulvertaft (1935) , the small intestine by Friedrich (~935), and the lungs and bronchi by Friedrich (i935) , Deuticke (1935) and Crenshaw (~932)_ Conversely, viscerurenal fistulae may occur, as described by Weiser (i9z 9 a, b) in his exhaustive survey oF the literalure. It is interesting to nole that most of the uncommon fistulae were described in the older literature, Their rarity nowadays is nu doubt due to the fact that modern diagnostic facilities and treatment methods have improved to such extent that few patients now reach that stage in the course of their disease, where fistuI~e with the kidneys or ureters can furm. Spontaneous gas formation may occur in either the kidneys or the bladder. In the kidney this is a distinct rarity, exemplified by the cases of Randall (19z7), Mathd and E. de la Pena (1933), Alexander (I941), and Bassalleck (1951), all cases of longstanding renal pathology. Where mentioned, it was generally Bact. coll communis, an aerobic gas-forming organism, which was cultured. In the bladder spontaneous gas formation is usually found in diabetic patients, but it can also occur in the absence of glycosuria, in non-diabetic patients. Urinary rctentmn with severe cystitis has usually been associated in these patients suffering from pneumaturia. Both aerobic gas-forming organisms may cause the fermentation of the urine, as well as, very rarely, anaerobic gas-forming organisms, as described by Wetser (i9~ 9 a, b) in a patient with a fulminating gas-gangrene infection. C l i n i c a l Aspects. Clinical aspects of the various conditions under discussion vary considerably, and can only be touched upon briefly. I n renovisceral fistulae the symptomatology is divided into those symptoms and signs due to the usually longstanding primary underlying condition, and those due to the establishment and persistence of the abnormal communication. The former are clearly outside the scope of this paper. Formation of the fistula with the intestinal tract may result in the passage of stones, pus, and urine per rectum or even in vomit, mad similarly renal contents may be coughed up if the fistula communicates with the bronchi. Bizarre clinical features may occur. A reduction in the size of a mass in the upper abdomen has not infrequently been noted, after the fistula has formed. The
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symptoms of vesico-intestinal fistulae are again separated into those of the underlying condition, and those of fistula formation. The latter classically include the passage of faeces, food, and gas per urethram ; urine may also be passed per rectum. The cases of pneumonephrosis quoted gave essentially a history of longstanding renal pathology. Two of these patients also complained of pncumaturia. In so-called ' primary pneumaturia ' the symptoms are those of cystitis, frequently recurrent in nature, with or without those of lower urinary tract obstruction. The symptomatology of diabetes mellitus in its various forms was present in approximately 5o per cent of these cases. Fineman and others (i952) state " the gas is passed at thc end of voiding, but the patient is not always aware of the passage of gas from the bladder ". Arthur and Johnson (i948) felt that the pneumaturia is of little consequence to the patient, but that its presence should suggest the possibility of glycosuria as the underlying cause. Radiological D i a g n o s i s . - - W h e n gas is noticed in the urinary tract on a film of the abdomen, it should arouse immediate interest as to its cause. If on inquiry it is found that recent urological instrumentation has not been performed on the patient, further radiological investigation is called for, always allowing for clinical considerations. Similarly, the complaint of pneumaturia should lead to radiological investigation in a high proportion of cases, in order to ascertain the cause of this symptom. It may be very difficult to be certain about small amounts of gas in either kidney, ureter, or bladder, in view of overlying intestinal gas shadows, larger amounts of gas within the confines of a renal pelvis, or calices in a hydronephrotie kidney, or surrounding calculi can be diagnosed, as well as large amounts of gas in the bladder. In addition there may be evidence of a renal mass, or of nephrolithiasis. Very rarely gas can also extend into the perirenal tissues, so that the kidney appears surrounded by gas, as in Alexander's case_ It must be understood that in probably most cases of urinary fistulae no gas may be visible in the urinary tract, even in the presence of a clinically symptomatic pneumaturia. When gas has been found in the urinary tract the fundamental diagnostic problem rests on a decision whether it is due to fermentation, i.e., spontaneous gas formation, or secondary to a fistula, instrumental introduction having been excluded. Knowledge and consideration of the clinical findings is essential. Intravenous pyelography, and in chosen cases retrograde pyelography, will have to be considered as a further diagnostic aid. Intestinal contrast studies, both as a barium meal followed through and as a barium enema, may be essential ; and in the case of the bladder cystography may have to be performed. Each patient must be considered on his own merits. It must not be assumed that a fistula is easily demonstrable. The literature abounds in references to the difficulty in demonstrating the actual fistula. This is partly due to the fact that an abnormal communication is often traversed by a contrast medium only in one direction, and not in the other, and partly because of the associated distortion of the normal anatomy. From a practical point of view the demonstration of the fistula itself is of secondary importance in many cases, the diagnosis of the cause of the fistula being of paramount importance to the patient as regards treatment. Interpretation of all the findings has to be cautious, and a diagnosis of spontaneous gas-formation in any part of the urinary tract should be offered only if the diagnosis of fistula formation can be reasonably excluded. Primary pneumaturia of the bladder has to be differentiated from the interesting condition variously called 'cystitis emphysematosa ', ' pneumatosis of the bladder', 'cystitis pneumatosis cystica ', etc., in which all the layers of the bladder wall may be studded with minute gas-containing vesicles i - 6 mm. in size. This condition has been described by Ravich and Katzen (i932), Redewill (~934), Burrell (I936), Herbut (1952), and others, and lately from the radiological point of view by Faingold and others (~953)- Certain similarities exist in this condition in comparison with
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primary pneumaturia, but the pathognomonic feature is the demonstration of the gas-filled vesicles on cystoscopy. Faingold and his colleagues demonstrated a most valuable radiological sign in the e o n d i t i o n - - a concentric ring of translucence outlining the bladder wall on a straight film of the abdomen. T h i s was seen in 3 of their 4 patients, and was quite unlike the gas-filled l u m e n of the bladder in primary pneurn~turia. According to Faingold sufficient gas may escape from the vesicles to fill and outline the l u m e n of the bladder, this being present in the film of their first patient. This latter appearance is t h e n indistinguishable from a case of primary pneumaturia, or indeed any case where the bladder is filled with gas. Faingold and his colleagues do not discuss the differentiation of cystitis emphysernatosa from primary pneumaturia. T h e relationship, or otherwise, between the two conditions is at present undecided, and depends on future work.
SUMMARY i. P n e u m a t u r i a as a radiologieal sign is rarely noted in the urinary tract on straight radiographs of the abdomen. I t is a most valuable sign requiring further radiological investigation in chosen cases. 2. Four case reports are given to illustrate the radiological aspects and problems arising from this finding. 3. T h e pertinent literature, pathology, and clinical aspects have been reviewed. 4. A radiologieal diagnostic approach to patients found to have p n e u m a t u r i a has been outlined.
Acknowledgements.--I am indebted to m y former colleagues at Hackney Hospital, M r . J. R. St.G. Stead and Miss Gladys Dodds, for permission to use their case records of the first 3 cases ; to Dr. Lloyd Bower for his records of'Case 4 ; and to Dr. T . H. C. Barclay and Dr. A. J. L o n g m o r e for valuable advice. REFERENCES ALEXANDER,J. C. (I94I), J_ Urol., 45, 57a. ARTHUR, L- M. and JorrNsoN, H. W. (I948), Ibid., 6o, 659. BASSALLeCK, H. (1951), Z. ~¥ol., 44, z3. BIJRRELL, N. L. (1'936), J. Urol., 36, 690. CnENSHAW, J. L. (I932), Ibid., 28, 427 . DANZER, J. T. (I95O), Radiology, 5~, 244. DEUTICKE, P. (1935) , Arch. klin. Chit., 182, 69. FAtNGOLD, J. E., HANSEN, C. O., and RIGLER, L. G. (I953) , Radiology, 61, 346. FISLDMAN, M. (I937), Arner.~. dig. Dis., 4, iio. FINEMAN, S., FERBER, W. L., and RO~INSKY, D. N. (195a), ,Radiology, 59, 63. FP,EIDI~IClt, I-I. (1935) ' Chlrurg., 7, 745H~I~EUT, P. A. (I952), Urological Pathology. London: Kimpton. HIGGII~S, C. C. (I936), J. Urd., 36, 694. - - - - and HIeKEN, N. F. (I933), Arch. Surg_, Chicago, 27, 817. Hmscn, E. W., and BAss, H. (i937) , J. Urol., 38, 371. MATHI~, C. P., and DE LA PENA, E. (I933), Vrol. cutan. Rev., 37, 732. MULSOW, F. W., and GILLIES, C. L. (I934),J. b~rol., 32, ~6I. PARKS, R. E. (I95I), Amer. J: Roentgenol., 66, 222. PI:LVERTA~T, R. J. V. (1935), Lancet, I, 24. RANDALL, A. (I927), Trans. Amer. Ass. gen_-urin. Surg., 2o, 261. RATCLIEV', R. K., and BARNES, A. C. (I939),J. [J¥ol., 42, 311RAVlCH, A., and KATZEN, P. (I932), 7. Amer. reed. Ass., 98, 1256. REDEWILL, F. H. (I934), Urol. cutan. Rev., 38, 537RILEY, F. G., and BRAGDOI,¢,F. H. (i937),J. Amer. rned. Ass., IO8, 1596. SENATOR, H. (1891), quoted by ALEXANI)I~R,J. C. (1941). SumtAco, L. A. (i93a), Anal. d. l. Fac. d. 1Fled., Montevideo, 17, 668. WEISER, A. (I929 a), Z. urol. Chit., 28, 113. (1929 b), Ihid_, 28, Izo. WESSON, M. B. (I938), J. Urol., 39, 589 . - - - - (i946), Urologic Roentgcnology. London : Kimpton. WILDBOLZ, H. (i937-8), "7. M t Sinai Hosp., 4, 579-
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PNEUMATURIAis usually defined as the presence of air or gas in the voided urine. The gas may come from the kidneys, the ureters, or the bladder. Senator (I89i) first classified pneumaturia as : - I. Following instrumental introduction into the urogenital tract. 2. Resulting from fistulae. 3. The result of fermentation, i.e., b y the actmn of organisms u p o n the urine. This group, which many writers classify as primary pneumaturia, is now subdivided into : (a) fermentation with glycosuria ; and (b) fermentation without glycosuria.
Fig. 36.--Case L Straight film of abdomen, ill-defined mass in left renal region, surrounded by colon inferlorly a~d laterally,
Fag. 37.--Case I. 4 ° rain. I.V.P. Homugeneous collection ot gas in left renal region. Colon essentially in position as in ,Fig. 36.
Pneumaturia is also a valuable radiological sign. Gas in the urinary tract is rarely recognized on a straight radiograph of the abdomen, partly because it may be mistaken for intestinal gas, partly because it may be missed unless the observer is aware that gas can be present in the urinary tract. It is the purpose of this paper to draw" attention to the significance of gas in the urinary tract, and to illustrate some of the problems arising from this finding by the case histories of 4 patients seen in the last few years.
CASE REPORTS Case i . - - R e n o c o l i c
fistula.
M r s . L . S., w i d o w , a g e d 72, w a s a d m i t t e d t o H a c k n e y H o s p i t a l o n M a y 5, I 9 5 2 ,
complaining of frequency of micturition, h~ematuria, and dysuria of one week's duration. She had suffered repeated attacks of pain in the left loin over the last twenty-five years, but had never passed any stones. During the last week she had typical left renal pain. She did not vomit and there had been no pneumatttria. On examination she was found to be ill and pyrexial. In the chest there were signs of bilateral basal bronchopncmnonia. In the distended and tender abdomen a doubtful mass was felt in the left upper quadrant.
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T h e u r i n e c o n t a i n e d a l b u m i n , b u t c u l t u r e was sterile. T h e w h i t e b l o o d - c e l l c o u n t w a s 12,ooo, of w h i c h 85 p e r c e n t w e r e p o l y m o r p h s . A r a d i o g r a p h of t h e a b d o m e n (Fig. 36) s h o w e d a n ill-defined m a s s in the left renal region, skirted b y the c o l o n i n f c r i o r l y a n d laterally. F o l l o w i n g a d m i s s i o n she r e m a i n e d p y r e x i a l a n d h a d i n t e r m i t t e n t s h i v e r i n g attacks. O n M a y ~8 she was f o u n d in a pool ot u r i n e in h e r b e d twice w i t h i n t w e n t y f o u r h o u r s , a n d a l t h o u g h t h o u g h t to be i n c o n t i n e n t b y the n u r s i n g staff was a d a m a n t t h a t she v o i d e d t h e u r i n e p e r r e c t u m . F o l l o w i n g this significant event h e r t e m p e r a t u r e settled g r a d u a l l y . A n i n t r a v e n o u s p y e l o g r a m w a s p e r f o r m e d o n M a y 28. T h r o u g h o u t t h e film series a large oval loculus of gas w i t h w e l l - d e f i n e d m a r g i n a n d of u m f o r m a p p e a r a n c e was n o t e d in t h e r e g i o n p r e v i o u s l y o c c u p i e d b y t h e
Fig. 38.--Case I. Circumscribed collection of gas anterior to and overlapping the lumbar spine in the lateral view.
Fig. 39. Case t. Barium enema. Loculus ofgaspersists in same position, almost surrounded by colon, with slight distornon of colon at one point. Fistula not shown.
ill-defined m a s s (Fig. 37). N o excretion t o o k place o n t h e left side a n d v e r y p o o r excretion o n t h e right. T h e b l a d d e r yeas opacified at 20 m i n u t e s . L o c a l i z a t i o n of t h e loculus of gas s h o w e d this to be l y i n g anterolateral to t h e l u m b a r spine (L.~, 2, 3) (Fig. 38). Clinically she was t h o u g h t to be suffering f r o m either a perinepl~ric abscess or a closed p y o n e p h r o s i s . W i t h the h i s t o r y of p a s s i n g u r i n e p e r r e c t u m in large quantities a n d the a p p e a r a n c e s o n the I.V.P., a radiological diagnosis of a renocolic fistula w a s considered, a n d a b a r i u m e n e m a s u g g e s t e d . T h i s was d o n e on M a y 3 o, b u t a p a r t f r o m a v e r y slight d i s t o r t i o n of t h e distal t r a n s v e r s e colon w h e r e it skirted t h e still visible loeulus of gas, n o a b n o r m a l i t y c o u l d b e f o u n d . T h e p o s t - e v a c u a t i o n film was u n s a t i s f a c t o r y (Fig. 39), the p a t i e n t hardly- v o i d i n g a n y b a r i u m , a n d s h o w e d n o evidence of a fistula. I n view of this essentially negative e x a m i n a t i o n it wa~ c o n s i d e r e d m o r e likely t h a t t h e p a t i e n t w a s suffering f r o m a p y o p n e u m o n e p h r o s i s . D u r i n g t h e n e x t five days t h e p a t i e n t ' s t e m p e r a t u r e steadily rose a n d t h e surgical i n d i c a t i o n for d r a i n a g e was clear-cut. P r i o r to incision a f u r t h e r film was r e q u e s t e d - - a n d this s h o w e d b a r i u m in a v e r y large a n d o b v i o u s l y h y d r o n e p h r o t i e k i d n e y (Fig. 40), giving u n d i s p u t e d p r o o f of a renocolie fistula_ A s p i r a t i o n of t h e left k i d n e y yielded gas, pus, b a r i u m , a n d f o u l - s m e l l i n g urine. A m p l e d r a i n a g e w a s instituted. P o s t - o p e l ~ t i v e l y h e r p r o g r e s s was slow b u t satisfactory, so t h a t b y S e p t e m b e r n e p h r e e t o m y c o u l d b e p e r f o r m e d , at w h i c h t i m e t h i c k adhesions to t h e eolon w e r e f o u n d . A small p o r t i o n of t h e pelvis of t h e left k i d n e y r e m a i n e d a d h e r e n t to t h e colon at t h e p r e s u m e d site of t h e fistula. T h e fistula, was n o t p a t e n t . T h e r e m o v e d k i d n e y s h o w e d gross h y d r o n e p h r o t i c changes_ T h e p a t i e n t m a d e a n u n i n t e r r u p t e d recovery.
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C o r n m e n t . - - T h i s p a t i e n t ' s clinical features indicated a l o n g - s t a n d i n g 1eft u r i n a r y infection, and surgical o p i n i o n f a v o u r e d a p e r i n e p h r i c abscess or a p y o n e p h r o t i c kidney. The demonstration of the loculus of gas in the left renal region, in an area w h e r e p r e v i o u s l y an indefinite m a s s had been present, raised t h e differential diagnosis of a renoc01ic fistula a n d a p y o p n e u m o nephrosis. T h e h i s t o r y of the passage of u r i n e p e r r e c t u m , followed b y the a p p e a r a n c e of the loculus of gas, clearly indicated drainage of the left kidney into t h e gut, either t h r o u g h a p r e existing fistula or by the f o r m a t i o n of a fistula. It was therefore particularly d i s a p p o i n t i n g to iail to d e m o n s t r a t e this fistula at first b y the b a r i u m enema. E v e n m o r e s u r p r i s i n g was the virtual lack of distortion of the colon at the site of the fistula, a p o i n t particularly s t r e s s e d b y F e l d m a n (1937). N o d o u b t the explanation in this p a t i e n t was t h a t t h e fistula was very small in size a n d t h a t it closed after drainage of the kidney, since t h e p a t i e n t at no t i m e following the first operation p a s s e d any intestinal c o n t e n t s f r o m the drainage site. Case 2 . ~ e o v e s i c a l fistula. Mr. W. H., aged 23, was admitted to Hackney Hospital in June, 195 i, with a 3-year history of recurrent attacks of intestinal colic, watery diarrhoea, anorexia, general malaise, drenching night sweats, and fever. A perineal abscess was opened two years previously, and a pelvic abscess d r a i n e d s p o n t a n e o u s l y p e r r e c t u m . A barium Fig. 4 o. C'ase ~. Five days later, Barium in ~rossly hydromeal and enema at another hospital proved ineonncphrotic kidney. elusive. Pneumaturia was first noted three months before admission, associated with the passage of intestinal contents per urethram, frequency, and dysuria. On examination the only significant findings were a tender fixed mass felt per rectum in the rectovesical pouch, and pneumaturia at the end of micturition, the urine containing flecks of solid material. A provisional clinical diagnosis of a vesico-intestinal fistula probably due to Crohn's disease was made. Laboratory investigations showed protein in the urine, pus cells, and Bact. col~ communis on culture. The h~emoglobin was 12'5 g., the total white cells amounted to e i , 2 o e , of which 92 per cent were polynlorphs. A straight film of the abdomen showed some gas of uniform translucency in a normal-sized bladderRenal function and outlines were seen to be normal on both sides on intravenous pyelography. An erect view of the bladder demonstrated a gas and fluid level (F~'. 4I), but no opaque medium was seen outside the renal tract. A barium meal, followed through, showed a normal stomach and duodenum. The csecum was reached at 4 hours, with the patient fasting. There was gross distortion of the terminal ileum, which was ragged in appearance, and there was a suggestion of several sinuses. The c~ecum was pulled medially, but the ileocsecal valve could not be identified. The bladder filled wlth barium before the descending colon had filled, but the actual fistula was not shown (Fig. 42). With a barium enema no overflow into the small intestine occurred. The deformity of the c~ecum was confirmed. No vesiooealie fistula was demonstrated. On these radiological findings Crohn's disease with an ileovesical fistula was thought to be the most likely diagnosis. The patient developed an anterior perineal abscess requiring drainage. On culture of the pus B a s t . cell communis and B. proteus were obtained. This abscess prevented eystoscopy, but sigmoidoscopy revealed no significant feature. Laparotomy to close the fistula showed a somewhat dilated and kinked loop of terminal small gut, which was densely adherent to the bladder. The csecum was pulled medially, and the appendix could not be idannfied. There was no macroscopical evidence of Crohn's disease. The fistula between the bladder and terminal jleum was identified and closed. A further lice-ileal fistula was closed. An ileotransverse colostomy was performed because of the distortion of the ileum following the closure of the fistula. Biopsy from the edge of the fistula showed purulent granulation tissue.
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Fig. 4I.--Case z.
E r e c t v i e w of b l a d d c r at end of I . V . P . examination, d e m u n ~ t r a t i n g gas a n d fluid level.
Fig. 43.--Case 3. a o - m i n u t e I.V.P. film. H o m o g e n e o u s o u t l i n i n g of b l a d d e r a n d left u r e t e r w i t h gas. Bilateral h y d r o n e p h r o t i c change~.
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[qg. 42.---Cose ~. B a r i u m m e a l ftnllcw-through, 4 hr. p.o. D i s t o r t e d ileocmcal region, m e d i a l dq~pl,neement of ea~'cum, a n d p r o b a b l e sinuses. A c t u a l fistula w i t h b l a d d e r n o t s h o w n , b u t b m ' i u m present ila t h e bladder.
Ielg. 44.--Case 4-
S t r a i g h t filnl Gf abdolzmn. M o d e r a t e l y large bladder, completely filIed w i t h gas.
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Post-operatively an intraperitoneal abscess developed in the ileocmeal region, and a faecal fistula developed following drainage. This fistula closed spontaneously. Soon after laparotomy the urine cleared, the patient gained weight, and felt fit. On follow-up examination he stated that he still occasionally suffered from slight pneumaturia but no other symptom. His urine, however, remained clear, and he passed no further intestinal contents per urethram. Comment.--This m a n w a s t h e o n l y p a t i e n t in t h i s series w h o c o m p l a i n e d of p n e u m a t u r i a . Clinical e v i d e n c e gave a s t r o n g lead to t h e c a u s e of t h e p n c u m a t u r i a , a n d t h e pl-cscnce of a n i n t e s t i n o vesical fistula w a s a s s u m e d , in v i e w of t h e p a s s a g e of i n t e s t i n a l c o n t e n t s p e r u r e t h r a m . Radiological i n v e s t i g a t i o n s were p r i m a r i l y called for to d e t e r m i n e t h e site a n d c a u s e o f t h e fistula. F o l l o w i n g these, C r o h n ' s d i s e a s e w a s t h o u g h t to be t h e m o s t r e a s o n a b l e p r e - o p e r a t i v e d i a g n o s i s for t h e f o r m a tion of t h e p r o v e n fistula, b u t o p e r a t i o n s h o w e d n o t h i n g to s u p p o r t this. A s j u d g e d f r o m t h e b i o p s y s p e c i m e n a b e n i g n i n f l a m m a t o r y lesion m u s t be a c c e p t e d as t h e final diagnosis, and, s i n c e t h e a p p e n d i x could n o t be identified, it w o u l d a p p e a r likely t h a t a c h r o n i c s u p p u r a t i v e a p p e n d i c i t i s was t h e u n d e r lying cause. T h e difficulty in d e m o n s t r a t i n g the actual fistula is well s h o w n in this case, a l m o s t certainly o w i n g to t h e g r o s s d i s t o r t i o n o f t h e a n a t o m y of t h e t e r m i n a l s m a l l i n t e s t i n e . C a s e 3 , - - U r e t e r o - r e e t o - v a g i n a l fistula. Mrs. L_ P., aged 59, was first seen at Hackney Hospital in September, ~95 o, and presented the typical symptoms and signs of a carcinoma of the cervix. An intravenous pyelogxam at this time was normal. In October of that year a Wertheim's operation was performed. During the operation the left ureter appeared infiltrated. Part of the anterior wall of the rectum was removed accidentally during removal of the pelvic viscera. This rent was repaired with continuous cbronnc catgut sutures. T h e biopsy report was " solid, trabecular, squamous-celled carcinoma of the cervix, grade z ". T h e general condition post-operatively was satisfactory, but nine days after operation she passed stools per vaginam. This gradually cleared, and she was discharged on Jan. I9, I95I. She was re-admitted five days later becauae " urine just ran away from h e r " and at times it was " thick ". There was no faecal incontinence. At this time her blood-urea was 69 my. per cent. An intravenous pyelogram was performed on Feb. IS. Throughout the series the bladder was outlined by gas. In addition the left ureter wasMso clearly outlined by gas up to the renal pelvis (Big_ 43)- Excretion on both sides was poor early on, but at 20 minutes both kidneys showed hydronephrotlc changes. T h e radiologlcal conclusion was that a fistula existed between the left ureter and either the gut or the vagina, allowing air to enter the urinary tract. T h e site of the fistula was not established. Cystoscopy was performed twice after the radiological investigation. On the first occasion the bladder was dirty and exhibited cystitis. Both ureteric catheters were held up in the bladder wall. T h e vagina contained urine and faeces. On the second examination a large hole was seen between the upper vagina and rectum, and urine was present m the rectum, Again the catheters would not pass up into the ureters. Surgical opinion favoured a fistula between the lower left ureter and the rectum, the urine entering the vagina via the rectovaginal fistula. On April 5, the rectovaginai fistula was excised and closed, the vagina being readily separated from the rectmn. Apart from a urinary infection the patient did well post-operatively. T h e repaired fistula remained elosed. It was considered that the left ureter drained into the r e c t u m - - a m o u n t i n g to a ureterie transplantation. eemrnent.--Several p o i n t s r e m a i n s p e c u l a t i v e in this patient. I t is n o w c o n s i d e r e d t h a t a barium enema probably would have demonstrated the fistulous communication between the upper r e c t u m a n d t h e left u r e t e r . T h i s fistula w a s a s s u m e d in v i e w of t h e p r e s e n c e of u r i n e in t h e r e c t u m ; its cause is u n c e r t a i n , b u t it w a s p r o b a b l y n e o p l a s t i c in origin r a t h e r t h a n t r a u m a t i c , s i n c e t h e r e was n e o p l a s t i c infiltration of t h e l o w e r e n d o f t h e left u r e t e r at t h e t i m e o f t h e o p e r a t i o n . T h i s fistula w o u l d e x p l a i n t h e p r e s e n c e o f gas i n t h e left u r e t e r as s h o w n o n t h e I,V.P. T h e r e c t o v a g i n a l fistula w a s m o s t likely t r a u m a t i c in origin, in v i e w o f t h e a c c i d e n t a l d a m a g e to t h e a n t e r i o r rectal wall. T h i s s e e m s c o r r o b o r a t e d b y t h e a b s e n c e of o b v i o u s infiltration of t h e r e c t a wall as well as b y t h e fact t h a t t h e r c c t o v a g i n a l fistula d i d n o t r e c u r f o l l o w i n g repair. T h e p r e s e n c e of gas in t h e b l a d d e r r e m a i n s u n e x p l a i n e d . C y s t o s c o p i c e v i d e n c e r u l e s o u t a fistula b e t w e e n t h e b l a d d e r a n d a g a s - c o n t a i n i n g v i s c u s . S o m e air m i g h t h a v e e n t e r e d t h e b l a d d e r f r o m t h e left u r e t e r , w h i c h was filled b y colonic gas, b u t t h i s is n o t w h o l l y c o n v i n c i n g , s i n c e n e i t h e r of t h e u r e t e r i c orifices c o u l d be p a s s e d b y c a t h e t e r s . N e v e r t h e l e s s t h i s p o s s i b i l i t y c a n n o t be r u l e d out. It is m o r e likely t h a t air w a s i n t r o d u c e d into t h e b l a d d e r f r o m w i t h o u t b y u r e t h r a l c a t h e t e r i z a t i o n at a n u n k n o w n
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time. I n the absence of autopsy control this case, therefore, is tentatively classified as one of a uretero-recto-vaginal fistula. Case 4---"Primary p n e u m a t u r l a " of t h e Madder. Mr. L K., aged 40, was first seen at the Medical Arts Clinic in Regina in April, I95o, when he complaincd of dizzy spells, nocturia, cloudy urinc, and loss of energy. He thought he had passed a stone the previous winter_ Following trauma to the right kidney in I939 a right nephrectomy had been performed. Examination of the urine revealed glycosuria, as well as pyuria. An intravenous pyelogram demonstrated poor function and moderate dilatation of the calicine system in the remaining left kidney. No opaque urinary calculi were seen. Throughout the examination the bladder was seen to be filled by gas (Fig. 44). Cystoscopy two weeks later demonstrated a double stricture of the urethra, and a foul, greyish slough covering the major portion of the floor and part of the lateral walls of the bladder. It was impossible to identify the ureteral orifices. The subsequent course of the patient is not pertinent to this paper, except that films of the abdomen obtained in July 195o, and again in the summer of 1953, did not reveal recurrence of gas in the bladder~ the patient requiring extensive treatment fur repeated urinsry infections. On tw0 occasions coliform bacilli were cultured from the urine. C o m m e n t . - - T h e patient was not fully investigated radiologically, b u t he seems to fit rather well with the criteria laid down by A r t h u r and Johnson (i948). A diabetic, the subject of two urethral strictures, he did not volunteer whether he passed gas during micturition. T h e I.V.P. showed gas in the bladder in such amounts that the condition could hardly be overlooked, or mistaken for gas in the intestine. A fistula with one of the n e i g h b o u r m g gas-containing viscera was not excluded radiologically, b u t no history of a fistula was elicited, and cystoscopy failed to reveal one. Although a fistula could not be ruled out completely, in view of the extensive greyish slough covering a large part of the bladder wall, tile subsequent course, repeated negative cystoscopies, and the absence of gas in the bladder on later films of the a b d o m e n would exclude it. At the original examination urine culture was not done, b u t it seems reasonable to assume that Bact. coE communis, found on cultures at a later stage, was the organism responsible for the fermentation of the urine in this diabetic patient.
THE L I T E R A T U R E Although a very extensive medical and surgical literature exists on the subject of pneumaturia, little is said about the radiological manifestations of gas in the urinary tract in text-books of radiology. Only Wesson (I946), in his book, mentions spontaneous gas formation in the kidney and gives an excellent example of a pneumonephrosis (the case of Math~ and E. de la Pena (i933)). I n s t r u m e n t a l pneumopyelography, however, is occasionally mentioned. Some recent papers have described the radiological demonstration of gas in part of the urinary tract. Spontaneous gas formation in the kidneys was first diagnosed by radiography by Randall (i927) , and was confirmed by operation. A most interesting case was presentcd by Surraco (I932). I n his patient s u p p u r a t i o n within the cavity of a huge renal hydatid cyst resulted in gas formation, shown radiographically as a gas and fluid level. Math& and E. de la Pena (T933) reported a case of p n e u m o n e p h r o s i s - - a pyonephrosis in the u p p e r pole of a duplex left kidney, with gas formation. Alexander (i94 z) described a further case of pneumonephrosis in a diabetic woman. Excellent radiographs became available during the course of the illness, showing first a spontaneous gas pyelogram on t h e left side three days after the passage of a calculus, later that the gas extended and eventually surrounded the whole kidney. Bassalleck (i95~) described a case of bilateral renal caleuh, those o n t h e left being surrounded by gas. He considered this to be due to fermentation in the infected kidney. T h e spontaneous presence of gas in the bladder was first diagnosed by X rays by Mulsow and Gillies (~934) in a non-diabetic patient. Riley and Bragdon (I937) mention the demonstration 0~ gas in the bladder in a diabetic suffering From cystitis. A r t h u r and Johnson (i948), Danzer (~95o), and lately Fineman, Ferher, and Roginsky (1952), have described the presence of gas in the bladder
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shown radiographically under the temls ' spontaneous pneumaturia ', ' pneumaturia ', and ' primary pneumaturia '. In view of the large literature on urinary-intestinal fistula:, it is surprising how rarely gas has been noted or commented upon in these cases. Parks (1951) described intermittent spontaneous pneumo-urography following end-to-end ureterosigmoid transplantation in 6 out of 33 cases. Faingold, Hansen, and Rigler (1953) recently reviewed the literature on cystitis emphyscmatosa and reported 4 cases diagnosed radiologically. Pathology.--Fistul~e, irrespective of their underlying cause, can connect almost any part of the u r i n a l - tract with adjacent viscera, if these contain air or gas it may escape into the urinary tract and so become radiologically demonstrable. By far the commonest of the urogenital tract fistulae are the vesico-intestinal fistula. Higgins (r936) analysed the aetiological factors in 328 cases described in the literature_ The majority were of an inflammatory basis (5 ° per cent), and of these two thirds were due to diverticulitis of the colon. Next came neoplasms, followed by traumatic and congenital fistulae. Renovisceral and ureteroviscerai fistula~ are uncommon. They are not a disease sui generis, but a complication of an already existing disease. Wesson (1938) considered that rer~ocolic fistul~e, the commonest renovisceral type of fistula, were more frequent than generally believed, but Ratcliff and Barnes (1939) only found 38 cases at the tinle of their report. In addition to these authors Hirsch and Bass (i937) , Weiser (1929) , Higgins and Hicken (1933) , Deuticke (i935) , and Feldman (1937) stress the fact that it is generally primary renal pathology in the form of pyelonephritis, pyonephrosis, tuberculosis, or nephrolithiasis with infection which causes the formation of a renocolic fistula, once a perinephric extension of the infection has been established. Fistul~ between the kidney and the stomach have been described by Wildbolz (1937-8), the duodenum by Pulvertaft (1935) , the small intestine by Friedrich (~935), and the lungs and bronchi by Friedrich (i935) , Deuticke (1935) and Crenshaw (~932)_ Conversely, viscerurenal fistulae may occur, as described by Weiser (i9z 9 a, b) in his exhaustive survey oF the literalure. It is interesting to nole that most of the uncommon fistulae were described in the older literature, Their rarity nowadays is nu doubt due to the fact that modern diagnostic facilities and treatment methods have improved to such extent that few patients now reach that stage in the course of their disease, where fistuI~e with the kidneys or ureters can furm. Spontaneous gas formation may occur in either the kidneys or the bladder. In the kidney this is a distinct rarity, exemplified by the cases of Randall (19z7), Mathd and E. de la Pena (1933), Alexander (I941), and Bassalleck (1951), all cases of longstanding renal pathology. Where mentioned, it was generally Bact. coll communis, an aerobic gas-forming organism, which was cultured. In the bladder spontaneous gas formation is usually found in diabetic patients, but it can also occur in the absence of glycosuria, in non-diabetic patients. Urinary rctentmn with severe cystitis has usually been associated in these patients suffering from pneumaturia. Both aerobic gas-forming organisms may cause the fermentation of the urine, as well as, very rarely, anaerobic gas-forming organisms, as described by Wetser (i9~ 9 a, b) in a patient with a fulminating gas-gangrene infection. C l i n i c a l Aspects. Clinical aspects of the various conditions under discussion vary considerably, and can only be touched upon briefly. I n renovisceral fistulae the symptomatology is divided into those symptoms and signs due to the usually longstanding primary underlying condition, and those due to the establishment and persistence of the abnormal communication. The former are clearly outside the scope of this paper. Formation of the fistula with the intestinal tract may result in the passage of stones, pus, and urine per rectum or even in vomit, mad similarly renal contents may be coughed up if the fistula communicates with the bronchi. Bizarre clinical features may occur. A reduction in the size of a mass in the upper abdomen has not infrequently been noted, after the fistula has formed. The
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symptoms of vesico-intestinal fistulae are again separated into those of the underlying condition, and those of fistula formation. The latter classically include the passage of faeces, food, and gas per urethram ; urine may also be passed per rectum. The cases of pneumonephrosis quoted gave essentially a history of longstanding renal pathology. Two of these patients also complained of pncumaturia. In so-called ' primary pneumaturia ' the symptoms are those of cystitis, frequently recurrent in nature, with or without those of lower urinary tract obstruction. The symptomatology of diabetes mellitus in its various forms was present in approximately 5o per cent of these cases. Fineman and others (i952) state " the gas is passed at thc end of voiding, but the patient is not always aware of the passage of gas from the bladder ". Arthur and Johnson (i948) felt that the pneumaturia is of little consequence to the patient, but that its presence should suggest the possibility of glycosuria as the underlying cause. Radiological D i a g n o s i s . - - W h e n gas is noticed in the urinary tract on a film of the abdomen, it should arouse immediate interest as to its cause. If on inquiry it is found that recent urological instrumentation has not been performed on the patient, further radiological investigation is called for, always allowing for clinical considerations. Similarly, the complaint of pneumaturia should lead to radiological investigation in a high proportion of cases, in order to ascertain the cause of this symptom. It may be very difficult to be certain about small amounts of gas in either kidney, ureter, or bladder, in view of overlying intestinal gas shadows, larger amounts of gas within the confines of a renal pelvis, or calices in a hydronephrotie kidney, or surrounding calculi can be diagnosed, as well as large amounts of gas in the bladder. In addition there may be evidence of a renal mass, or of nephrolithiasis. Very rarely gas can also extend into the perirenal tissues, so that the kidney appears surrounded by gas, as in Alexander's case_ It must be understood that in probably most cases of urinary fistulae no gas may be visible in the urinary tract, even in the presence of a clinically symptomatic pneumaturia. When gas has been found in the urinary tract the fundamental diagnostic problem rests on a decision whether it is due to fermentation, i.e., spontaneous gas formation, or secondary to a fistula, instrumental introduction having been excluded. Knowledge and consideration of the clinical findings is essential. Intravenous pyelography, and in chosen cases retrograde pyelography, will have to be considered as a further diagnostic aid. Intestinal contrast studies, both as a barium meal followed through and as a barium enema, may be essential ; and in the case of the bladder cystography may have to be performed. Each patient must be considered on his own merits. It must not be assumed that a fistula is easily demonstrable. The literature abounds in references to the difficulty in demonstrating the actual fistula. This is partly due to the fact that an abnormal communication is often traversed by a contrast medium only in one direction, and not in the other, and partly because of the associated distortion of the normal anatomy. From a practical point of view the demonstration of the fistula itself is of secondary importance in many cases, the diagnosis of the cause of the fistula being of paramount importance to the patient as regards treatment. Interpretation of all the findings has to be cautious, and a diagnosis of spontaneous gas-formation in any part of the urinary tract should be offered only if the diagnosis of fistula formation can be reasonably excluded. Primary pneumaturia of the bladder has to be differentiated from the interesting condition variously called 'cystitis emphysematosa ', ' pneumatosis of the bladder', 'cystitis pneumatosis cystica ', etc., in which all the layers of the bladder wall may be studded with minute gas-containing vesicles i - 6 mm. in size. This condition has been described by Ravich and Katzen (i932), Redewill (~934), Burrell (I936), Herbut (1952), and others, and lately from the radiological point of view by Faingold and others (~953)- Certain similarities exist in this condition in comparison with
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primary pneumaturia, but the pathognomonic feature is the demonstration of the gas-filled vesicles on cystoscopy. Faingold and his colleagues demonstrated a most valuable radiological sign in the e o n d i t i o n - - a concentric ring of translucence outlining the bladder wall on a straight film of the abdomen. T h i s was seen in 3 of their 4 patients, and was quite unlike the gas-filled l u m e n of the bladder in primary pneurn~turia. According to Faingold sufficient gas may escape from the vesicles to fill and outline the l u m e n of the bladder, this being present in the film of their first patient. This latter appearance is t h e n indistinguishable from a case of primary pneumaturia, or indeed any case where the bladder is filled with gas. Faingold and his colleagues do not discuss the differentiation of cystitis emphysernatosa from primary pneumaturia. T h e relationship, or otherwise, between the two conditions is at present undecided, and depends on future work.
SUMMARY i. P n e u m a t u r i a as a radiologieal sign is rarely noted in the urinary tract on straight radiographs of the abdomen. I t is a most valuable sign requiring further radiological investigation in chosen cases. 2. Four case reports are given to illustrate the radiological aspects and problems arising from this finding. 3. T h e pertinent literature, pathology, and clinical aspects have been reviewed. 4. A radiologieal diagnostic approach to patients found to have p n e u m a t u r i a has been outlined.
Acknowledgements.--I am indebted to m y former colleagues at Hackney Hospital, M r . J. R. St.G. Stead and Miss Gladys Dodds, for permission to use their case records of the first 3 cases ; to Dr. Lloyd Bower for his records of'Case 4 ; and to Dr. T . H. C. Barclay and Dr. A. J. L o n g m o r e for valuable advice. REFERENCES ALEXANDER,J. C. (I94I), J_ Urol., 45, 57a. ARTHUR, L- M. and JorrNsoN, H. W. (I948), Ibid., 6o, 659. BASSALLeCK, H. (1951), Z. ~¥ol., 44, z3. BIJRRELL, N. L. (1'936), J. Urol., 36, 690. CnENSHAW, J. L. (I932), Ibid., 28, 427 . DANZER, J. T. (I95O), Radiology, 5~, 244. DEUTICKE, P. (1935) , Arch. klin. Chit., 182, 69. FAtNGOLD, J. E., HANSEN, C. O., and RIGLER, L. G. (I953) , Radiology, 61, 346. FISLDMAN, M. (I937), Arner.~. dig. Dis., 4, iio. FINEMAN, S., FERBER, W. L., and RO~INSKY, D. N. (195a), ,Radiology, 59, 63. FP,EIDI~IClt, I-I. (1935) ' Chlrurg., 7, 745H~I~EUT, P. A. (I952), Urological Pathology. London: Kimpton. HIGGII~S, C. C. (I936), J. Urd., 36, 694. - - - - and HIeKEN, N. F. (I933), Arch. Surg_, Chicago, 27, 817. Hmscn, E. W., and BAss, H. (i937) , J. Urol., 38, 371. MATHI~, C. P., and DE LA PENA, E. (I933), Vrol. cutan. Rev., 37, 732. MULSOW, F. W., and GILLIES, C. L. (I934),J. b~rol., 32, ~6I. PARKS, R. E. (I95I), Amer. J: Roentgenol., 66, 222. PI:LVERTA~T, R. J. V. (1935), Lancet, I, 24. RANDALL, A. (I927), Trans. Amer. Ass. gen_-urin. Surg., 2o, 261. RATCLIEV', R. K., and BARNES, A. C. (I939),J. [J¥ol., 42, 311RAVlCH, A., and KATZEN, P. (I932), 7. Amer. reed. Ass., 98, 1256. REDEWILL, F. H. (I934), Urol. cutan. Rev., 38, 537RILEY, F. G., and BRAGDOI,¢,F. H. (i937),J. Amer. rned. Ass., IO8, 1596. SENATOR, H. (1891), quoted by ALEXANI)I~R,J. C. (1941). SumtAco, L. A. (i93a), Anal. d. l. Fac. d. 1Fled., Montevideo, 17, 668. WEISER, A. (I929 a), Z. urol. Chit., 28, 113. (1929 b), Ihid_, 28, Izo. WESSON, M. B. (I938), J. Urol., 39, 589 . - - - - (i946), Urologic Roentgcnology. London : Kimpton. WILDBOLZ, H. (i937-8), "7. M t Sinai Hosp., 4, 579-
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