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Bone Formation in the Ureter: A Case Report
J:'nE JoLTH?\Al., OF' UROLOGY
Yol. 75, No. 5, :Ma.y 1956 Printed in lJ.S.A"
BONE FOIUHATION 1:-J THE URETER: A CASE REPORT M. K KLINGER Frnm the Departrnent 1\f [jrology, Beth El Hosp-ila(. Brooklyn, N. Y.
Extraskeletal or bone formation is known to occur in the urinary tract primarily 1md.er two sets of circumstances. The first i:;; the deposition of the bone in the bladder wall and in abdominal 1vall scars following operations on the bladder and prostate glancP-a The other is the experimental induction of bone formation when a section of epithelium of the urinary tract is transplanted to suitable portions of the parietal connective tissue or fascia. The laUer was dcm-on8trated both N euhof\ and Huggins. 5 -s The spontaneous appearance of bone in the human ureter is rare. The present report deals with such a ease. There are no similar instances recorded in the literatun". CASJ
A 26-year-old rnarried woman was first seen in U),'5:3. Iler chief complaint was recurrent left ffank of approximately three years' duration. The attadrn of pain were sometimes by burning on urination. J'\o previous urological studies had been done. Examination at the time of the fin,t, visit disclosed 0111:y some, tenderness over the left kidney. A c:atheteri½ed specimen of urine showed innumerable pus cells. plain film of the urinary tract showed a shadow with a calcified rim in the course of the left ureter suggestive of a ureteral calculus at the level of the fourth lumbar \Tertehra. An excretory urograrn showed prompt excretion and normal outlines on the right side. On the left excretion was dnlayed. The calyces and pelvis were dilated. The left ureter was not outlined. A cystoscopic examination showed a normal bladder. The urcternl orifices were normal. A,, ureteral catheter ·was passed through the left orifice for a distam:e of 12 cm. easily. Ho,vever, it could be advanced no farther. An x-ray indicated that the tip of the catheter ,vas at the lower edge of the shadow observed in the plain urogram. The was advised to enter the hospital for further A second urinalysis again showed pus cells and E. coli grew on culture. Studies for acid-fast organi,mis were negative, Hematological and blood chemistry studies were normal. " ,.•:_".,",,_",;;•0,\' and catheterization of the left ureter were repeated, 7
Accepted for publication January 11, 1956. 'Lewis, D,: Myositis ossificans in suprnpubic wound. J. A. J\f. A., 80: l2l, 1923. 2 Goldstein, H. H.: Myositis ossifieans foJJowing suprapubic prostatectom_y. ,J, Urol 24: 2Jl, 1930. '1 Collings, C. W. and Walebir, F.: .J. UroL, 46: 494, Hl41. 'Neuhof, H.: Fascia trnnsplants into visceral defects. Surg., Gynec. & Obst., 24: 38'.-I, 1917. 5 Huggins, C. B.: Influence of urinar,, tract mucosa on experimental formation of bone, Proc. Soc. Exp. Biol. & Med., 27: 349, 1929-30. 'Huggins, C. B.: Implantation of ureteral segments to abdominal waJL Proc. Soc. Exp. Biol. & Med., 28: 125, Hl30<1l. 7 Huggins, C. B.: Formation of bone under influence of epithelium of urinary tract. Arcb. Surg., 'J.7: 203, 1933. 8 Abbott, A. C. and Goodwin, A . :\I.: Observations on bone formation in abdominal wall following 1,ransplantation of mucous membrane of urinary bladder. Cana.cl. Med. Assoe . .J., 26: 3lX'>, 1932. 793
794
M. E. KLINGER
with the same result as previously. Contrast medium injected through the ureteral catheter passed around the tip of the catheter and down the ureter. No dye was visible beyond the obstructing object. On the supposition that we were dealing with some type of obstructing ureteral calculus, albeit an odd looking one, operation was advised and performed. The ureter was approached through the customary flank incision, identified, and isolated without difficulty. For a short distance surrounding the obstruction there was a pronounced ureteral and periureteral reaction consisting of thickening and induration of the ureteral wall and the periureteral areolar tissue. These findings were thought to be consistent with the localized ureteral and periureteral reaction commonly seen at the site of a calculus. The ureteral wall over the "calculus" was incised and the customary grating sensation noted as the knife's edge came in contact with the stone. However, considerable difficulty was encountered in freeing the object from the wall of the ureter. Its liberation was finally accomplished and it was delivered. It had a cylindrical form and a dense cartilaginous texture. Its true nature was unsuspected at this time. A catheter was passed easily to the renal pelvis then to the bladder. The ureteral incision was then closed with several sutures approximating the edges loosely. A Penrose drain was inserted at the site of the ureterotomy and the wound closed in the customary manner. Pathologist's report (Dr. David Spain): The specimen consisted of a bony hard piece of tissue that measured 0.5 cm. in diameter at its widest point. Micro-
FIG. 1
BONE FORMATIOS IN URETER
79Fi
scopically it consisted of an irregular arrangement of fully formed bony trabeculae with fat tissue and marrmv filling in the spaces. Figure 1 represents a photomicrograph of the sectioned specimen, and in it one can clearly see bone and bone marrmv elements. The convalescence -was uncomplicated except that urine continued to drain from the wound beyond the time when it normally would have been to cease. Accordingly, another cystoscopy was done and an attempt made to catheterize the left ureter. The catheter met an obstruct.ion at the same point as preoperatively, indicating, apparently, that healing by scar had taken place with resultant occlusion of tho ureteral lumen. Within several days after this procedure, hmvever, drainage suddenly stopped. 'vVith the cessation of drainage there was no pain, no fever, no palpable enlargement of the kidney and no mass in the operated region. It was presurned that the left kidney had ceased hmctioning. Confirmation was obtained by means of an excretory urograrn which showed no dye in the left kidney even after long delayed exposures. Furthermore tJ1e renal shadow was not enlarged. The patient -was informed that re-operation was advisable and that nephrectomy was a possibility. However, since she was completely asymptomatic at. the time and continued to remain so to the time of writing, she steadfastly refused. Subsequent urographic studies showed no change in the kidney's status and repeated attempts to catheterize the left ureter were fruitless. This case is principally because it is a medical curiosity. Had the true nature of the lesion been recognized at the time of operation, the management would have been different. A more satisfactory end result doubtlessly -would have been obtained had the entire segment of the involved ureter been excised and an end-to-end ureteral anastomosis done. It is interesting to speculate as to how this bone deposition occurred. That ureteral mucosa can induce the formation of bone uuder experirnental conditions is known. Normally it does of course, cause its adjacent connective tiss11e to undergo such rnet.aplasia . However, it is conceivable, in the present case, that, under the abnormal influences of infection and irritation, conditions akin to the experimental ones were present, and under such circumstances bone formation in the subepithelial tissues was possible, and did, in fact, occur. SUMMARY
A case of bone formation in the ureter of a patient mistakenly thought have a ureteral calculus is presented. The explanation for this unusual finding probably lies in subepithelial rnetaplasia under the influence of the ureteral mucosa in an abnormal enviromnent of infection. Surgical removal of the object was followed by ureteral occlusion and apparently a.utonephrectorny. 3.45 Schermerhorn
17,l'IT., Y.
Yol. 75, No. 5, :Ma.y 1956 Printed in lJ.S.A"
BONE FOIUHATION 1:-J THE URETER: A CASE REPORT M. K KLINGER Frnm the Departrnent 1\f [jrology, Beth El Hosp-ila(. Brooklyn, N. Y.
Extraskeletal or bone formation is known to occur in the urinary tract primarily 1md.er two sets of circumstances. The first i:;; the deposition of the bone in the bladder wall and in abdominal 1vall scars following operations on the bladder and prostate glancP-a The other is the experimental induction of bone formation when a section of epithelium of the urinary tract is transplanted to suitable portions of the parietal connective tissue or fascia. The laUer was dcm-on8trated both N euhof\ and Huggins. 5 -s The spontaneous appearance of bone in the human ureter is rare. The present report deals with such a ease. There are no similar instances recorded in the literatun". CASJ
A 26-year-old rnarried woman was first seen in U),'5:3. Iler chief complaint was recurrent left ffank of approximately three years' duration. The attadrn of pain were sometimes by burning on urination. J'\o previous urological studies had been done. Examination at the time of the fin,t, visit disclosed 0111:y some, tenderness over the left kidney. A c:atheteri½ed specimen of urine showed innumerable pus cells. plain film of the urinary tract showed a shadow with a calcified rim in the course of the left ureter suggestive of a ureteral calculus at the level of the fourth lumbar \Tertehra. An excretory urograrn showed prompt excretion and normal outlines on the right side. On the left excretion was dnlayed. The calyces and pelvis were dilated. The left ureter was not outlined. A cystoscopic examination showed a normal bladder. The urcternl orifices were normal. A,, ureteral catheter ·was passed through the left orifice for a distam:e of 12 cm. easily. Ho,vever, it could be advanced no farther. An x-ray indicated that the tip of the catheter ,vas at the lower edge of the shadow observed in the plain urogram. The was advised to enter the hospital for further A second urinalysis again showed pus cells and E. coli grew on culture. Studies for acid-fast organi,mis were negative, Hematological and blood chemistry studies were normal. " ,.•:_".,",,_",;;•0,\' and catheterization of the left ureter were repeated, 7
Accepted for publication January 11, 1956. 'Lewis, D,: Myositis ossificans in suprnpubic wound. J. A. J\f. A., 80: l2l, 1923. 2 Goldstein, H. H.: Myositis ossifieans foJJowing suprapubic prostatectom_y. ,J, Urol 24: 2Jl, 1930. '1 Collings, C. W. and Walebir, F.: .J. UroL, 46: 494, Hl41. 'Neuhof, H.: Fascia trnnsplants into visceral defects. Surg., Gynec. & Obst., 24: 38'.-I, 1917. 5 Huggins, C. B.: Influence of urinar,, tract mucosa on experimental formation of bone, Proc. Soc. Exp. Biol. & Med., 27: 349, 1929-30. 'Huggins, C. B.: Implantation of ureteral segments to abdominal waJL Proc. Soc. Exp. Biol. & Med., 28: 125, Hl30<1l. 7 Huggins, C. B.: Formation of bone under influence of epithelium of urinary tract. Arcb. Surg., 'J.7: 203, 1933. 8 Abbott, A. C. and Goodwin, A . :\I.: Observations on bone formation in abdominal wall following 1,ransplantation of mucous membrane of urinary bladder. Cana.cl. Med. Assoe . .J., 26: 3lX'>, 1932. 793
794
M. E. KLINGER
with the same result as previously. Contrast medium injected through the ureteral catheter passed around the tip of the catheter and down the ureter. No dye was visible beyond the obstructing object. On the supposition that we were dealing with some type of obstructing ureteral calculus, albeit an odd looking one, operation was advised and performed. The ureter was approached through the customary flank incision, identified, and isolated without difficulty. For a short distance surrounding the obstruction there was a pronounced ureteral and periureteral reaction consisting of thickening and induration of the ureteral wall and the periureteral areolar tissue. These findings were thought to be consistent with the localized ureteral and periureteral reaction commonly seen at the site of a calculus. The ureteral wall over the "calculus" was incised and the customary grating sensation noted as the knife's edge came in contact with the stone. However, considerable difficulty was encountered in freeing the object from the wall of the ureter. Its liberation was finally accomplished and it was delivered. It had a cylindrical form and a dense cartilaginous texture. Its true nature was unsuspected at this time. A catheter was passed easily to the renal pelvis then to the bladder. The ureteral incision was then closed with several sutures approximating the edges loosely. A Penrose drain was inserted at the site of the ureterotomy and the wound closed in the customary manner. Pathologist's report (Dr. David Spain): The specimen consisted of a bony hard piece of tissue that measured 0.5 cm. in diameter at its widest point. Micro-
FIG. 1
BONE FORMATIOS IN URETER
79Fi
scopically it consisted of an irregular arrangement of fully formed bony trabeculae with fat tissue and marrmv filling in the spaces. Figure 1 represents a photomicrograph of the sectioned specimen, and in it one can clearly see bone and bone marrmv elements. The convalescence -was uncomplicated except that urine continued to drain from the wound beyond the time when it normally would have been to cease. Accordingly, another cystoscopy was done and an attempt made to catheterize the left ureter. The catheter met an obstruct.ion at the same point as preoperatively, indicating, apparently, that healing by scar had taken place with resultant occlusion of tho ureteral lumen. Within several days after this procedure, hmvever, drainage suddenly stopped. 'vVith the cessation of drainage there was no pain, no fever, no palpable enlargement of the kidney and no mass in the operated region. It was presurned that the left kidney had ceased hmctioning. Confirmation was obtained by means of an excretory urograrn which showed no dye in the left kidney even after long delayed exposures. Furthermore tJ1e renal shadow was not enlarged. The patient -was informed that re-operation was advisable and that nephrectomy was a possibility. However, since she was completely asymptomatic at. the time and continued to remain so to the time of writing, she steadfastly refused. Subsequent urographic studies showed no change in the kidney's status and repeated attempts to catheterize the left ureter were fruitless. This case is principally because it is a medical curiosity. Had the true nature of the lesion been recognized at the time of operation, the management would have been different. A more satisfactory end result doubtlessly -would have been obtained had the entire segment of the involved ureter been excised and an end-to-end ureteral anastomosis done. It is interesting to speculate as to how this bone deposition occurred. That ureteral mucosa can induce the formation of bone uuder experirnental conditions is known. Normally it does of course, cause its adjacent connective tiss11e to undergo such rnet.aplasia . However, it is conceivable, in the present case, that, under the abnormal influences of infection and irritation, conditions akin to the experimental ones were present, and under such circumstances bone formation in the subepithelial tissues was possible, and did, in fact, occur. SUMMARY
A case of bone formation in the ureter of a patient mistakenly thought have a ureteral calculus is presented. The explanation for this unusual finding probably lies in subepithelial rnetaplasia under the influence of the ureteral mucosa in an abnormal enviromnent of infection. Surgical removal of the object was followed by ureteral occlusion and apparently a.utonephrectorny. 3.45 Schermerhorn
17,l'IT., Y.