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Malignant Change in Heterotopic Bone Formation Following Suprapubic Cystolithotomy
0022-534 7/84/1323-0558$02.00/0 Vol. 132, September Printed in U.S.A.
THE JOURNAL OF UROLOGY
Copyright© 1984 by The Williams & Wilkins Co.
MALIGNANT CHANGE IN HETEROTOPIC BONE FORMATION FOLLOWING SUPRAPUBIC CYSTOLITHOTOMY BEDRI KANDEMIR, YUCEL ARITAS
AND
MURAT SADE
From the Departments of Pathology, Surgery and Urology, Erciyes University School of Medicine, Kayseri, Turkey
ABSTRACT
We report the first case of heterotopic bone formation with malignant changes. The development of this lesion shortly after excision of a benign heterotopic bone lesion at the same site is another distinctive feature of this case. The development of heterotopic bone tissue under the influence of transitional epithelium that is inoculated into the abdominal wall after fascial transplantation for repair of ureteral and vesical defects, and suprapubic prostatectomy has been known since the beginning of this century. 1- 5 A review of the literature revealed that epithelial and osseous components of this phenomenon are of a benign nature. The first tumor of this kind with malignant degeneration in the epithelial component is reported.
CASE REPORT
F. 0., a 52-year-old farmer, was seen initially in our hospital in February 1981 for a left lower abdominal mass 2 years in duration. History was significant for a bladder stone operation 9 years previously. Physical examination revealed a hard 6 X 6 cm. mass in the left lower quadrant. Routine blood tests and urinalyses were normal. Excretory urography (IVP) revealed a pelvic mass less dense than the bones, with indistinct borders suggestive of new bone formation (fig. 1). The urinary tract was normal. On February 16 the mass was excised extraperitoneally. The mass was adherent to surrounding soft tissues but not to the bladder, which was not entered during the operation. The removed tumor was 6 cm. in diameter, with irregular lobulations and a distinct, smooth fibrous capsule. The cut surface revealed a grayish-white tumor with small irregular compartments that were separated by thin bone septae. The inner surfaces of these compartments were lined with mucous membrane resembling urinary mucosa (fig. 2). On microscopic section the tumor was composed of sharply circumscribed foci of osteoid and atypical, less calcified bone formation within vascular osteoblastic connective tissue around the highly proliferated transitional cell islands (fig. 3, A). No malignant changes were encountered in serial sections. On November 12 the patient was rehospitalized with a 4 x 4 cm. hard mass at the same location 2 months in duration, as well as dysuria and frequency. Routine blood tests, urinalysis and a chest x-ray were normal. IVP findings were similar to the previous study, suggesting new bone formation. The urinary tract was normal. Reoperation was performed via a lower median incision with the patient under general anesthesia. The mass was located extraperitoneally and was not attached to the bladder. The lesion was removed completely and convalescence was uneventful. Grossly, the tumor was similar to the first lesion except that the surface was irregular and did not have a complete surrounding capsule. The histologic appearance was the same as that of the first specimen except for malignant changes in the epithelial component. The malignant foci, which had infiltrated the surrounding soft tissues, were composed of nests and cords of
FIG. 1. IVP (cystogram phase) shows pelvic mass on left side (arrows).
Accepted for publication April 19, 1984. 558
FIG. 2. Macroscopic appearance of cut surface of first specimen
atypical transitional cells of varying size and shape. There was no osseous tissue formation around the malignant foci (fig. 3, B).
After the microscopic diagnosis of malignancy, slides of the first specimen were re-examined and new serial sections were made but no evidence of malignancy could be detected. In numerous new serial sections prepared from the margins of the first specimen transitional cell islands were located near to but not on the margins. Postoperatively, the patient received cobalt therapy for 30 sessions. He is doing well 2 years postoperatively, with no complaints and no evidence of metastases or recurrence.
559 an ,CV'~.•c.;rn., bone masses around the ~..,,,,,-,,,m,,,. ,, To date the ,rn~ff•-•C•-' cases of this phenomenon are of benign nature. 1-" In our case transitional epithelial cells most probably were inoculated into the soft tissues of the abdominal wall during the vesicai operation and within 7 years an osseous mass developed, which is in accordance with the cases reported in the literature. The originality of our case is the development of transitional cell carcinoma in the second mass and lack of bone formations around the malignant epithelial islands. At this point the recurrence of the mass with malignant changes needs some emphasis. In our opinion the presence of malignancy in the recurrent lesion can be explained by malignant degeneration of the retained transitional cells after the first operation. However, it is difficult to state how these cells remained or were inoculated into the operative site because the original mass had been excised completely. Moreover, repeated numerous sections of margins of the original mass did not show transitional cells. However, the presence of transitional cells near the margin may shed a dim light to the case. Inoculation of transitional cells from the unnoticed minute crack in the margin might be a possibilityo This case illustrates that heterotopic bone formation after urinary epithelial inoculations could give rise to benign as well as malignant osseous tumorso REFERENCES
FIG. 3. A, histologic appearance of specimen 1 shows bony tissue formation around hyperplastic transitional epithelium. H & E, reduced from X250. B, histologic appearance of specimen 2 shows transitional cell carcinoma invasion into surrounding soft tissue. H & E, reduced from X400. DISCUSSION
Experimental studies have shown that urinary and gastrointestinal epithelium could proliferate in soft tissues and exert
L Abeshouse, B. S.: Heterotopic bone formation following suprapubic prostatectomy. J. Urol., 59: 50, 1948. 2. Neuhof, ff: Fascia transplantation into visceral defects: an experimental and clinical studyo Surg., Gyneco & Obsto, 24: 383, 19170 3. Sewell, W. S., Siceluff, J. G. and Horton, J. D.: Extraskeletal bone formation following suprapubic prostatectomy: case reporto Jo Urol., 62: 842, 19490 4. Bell, J. W. and Knudtson, K. Po: Soft tissue osteogenesis with gall bladder mucosa! autografts. Lab. Investo, 10: 397, 1961. 5. Huggins, Co R: The formation of bone under the influence of epithelium of the urinary tract. Arch. Surgo, 22: 377, 19310 60 Apostolidis, No S., Legakis, N. C., Gregoriadis, G. C., Androulakakis, P. k and Romanos, A. N.: Heterotopic bone formation in abdominal operation scars. Report of six cases with review of the literature. Amer. J. Surgo, 142: 555, 1981.
THE JOURNAL OF UROLOGY
Copyright© 1984 by The Williams & Wilkins Co.
MALIGNANT CHANGE IN HETEROTOPIC BONE FORMATION FOLLOWING SUPRAPUBIC CYSTOLITHOTOMY BEDRI KANDEMIR, YUCEL ARITAS
AND
MURAT SADE
From the Departments of Pathology, Surgery and Urology, Erciyes University School of Medicine, Kayseri, Turkey
ABSTRACT
We report the first case of heterotopic bone formation with malignant changes. The development of this lesion shortly after excision of a benign heterotopic bone lesion at the same site is another distinctive feature of this case. The development of heterotopic bone tissue under the influence of transitional epithelium that is inoculated into the abdominal wall after fascial transplantation for repair of ureteral and vesical defects, and suprapubic prostatectomy has been known since the beginning of this century. 1- 5 A review of the literature revealed that epithelial and osseous components of this phenomenon are of a benign nature. The first tumor of this kind with malignant degeneration in the epithelial component is reported.
CASE REPORT
F. 0., a 52-year-old farmer, was seen initially in our hospital in February 1981 for a left lower abdominal mass 2 years in duration. History was significant for a bladder stone operation 9 years previously. Physical examination revealed a hard 6 X 6 cm. mass in the left lower quadrant. Routine blood tests and urinalyses were normal. Excretory urography (IVP) revealed a pelvic mass less dense than the bones, with indistinct borders suggestive of new bone formation (fig. 1). The urinary tract was normal. On February 16 the mass was excised extraperitoneally. The mass was adherent to surrounding soft tissues but not to the bladder, which was not entered during the operation. The removed tumor was 6 cm. in diameter, with irregular lobulations and a distinct, smooth fibrous capsule. The cut surface revealed a grayish-white tumor with small irregular compartments that were separated by thin bone septae. The inner surfaces of these compartments were lined with mucous membrane resembling urinary mucosa (fig. 2). On microscopic section the tumor was composed of sharply circumscribed foci of osteoid and atypical, less calcified bone formation within vascular osteoblastic connective tissue around the highly proliferated transitional cell islands (fig. 3, A). No malignant changes were encountered in serial sections. On November 12 the patient was rehospitalized with a 4 x 4 cm. hard mass at the same location 2 months in duration, as well as dysuria and frequency. Routine blood tests, urinalysis and a chest x-ray were normal. IVP findings were similar to the previous study, suggesting new bone formation. The urinary tract was normal. Reoperation was performed via a lower median incision with the patient under general anesthesia. The mass was located extraperitoneally and was not attached to the bladder. The lesion was removed completely and convalescence was uneventful. Grossly, the tumor was similar to the first lesion except that the surface was irregular and did not have a complete surrounding capsule. The histologic appearance was the same as that of the first specimen except for malignant changes in the epithelial component. The malignant foci, which had infiltrated the surrounding soft tissues, were composed of nests and cords of
FIG. 1. IVP (cystogram phase) shows pelvic mass on left side (arrows).
Accepted for publication April 19, 1984. 558
FIG. 2. Macroscopic appearance of cut surface of first specimen
atypical transitional cells of varying size and shape. There was no osseous tissue formation around the malignant foci (fig. 3, B).
After the microscopic diagnosis of malignancy, slides of the first specimen were re-examined and new serial sections were made but no evidence of malignancy could be detected. In numerous new serial sections prepared from the margins of the first specimen transitional cell islands were located near to but not on the margins. Postoperatively, the patient received cobalt therapy for 30 sessions. He is doing well 2 years postoperatively, with no complaints and no evidence of metastases or recurrence.
559 an ,CV'~.•c.;rn., bone masses around the ~..,,,,,-,,,m,,,. ,, To date the ,rn~ff•-•C•-' cases of this phenomenon are of benign nature. 1-" In our case transitional epithelial cells most probably were inoculated into the soft tissues of the abdominal wall during the vesicai operation and within 7 years an osseous mass developed, which is in accordance with the cases reported in the literature. The originality of our case is the development of transitional cell carcinoma in the second mass and lack of bone formations around the malignant epithelial islands. At this point the recurrence of the mass with malignant changes needs some emphasis. In our opinion the presence of malignancy in the recurrent lesion can be explained by malignant degeneration of the retained transitional cells after the first operation. However, it is difficult to state how these cells remained or were inoculated into the operative site because the original mass had been excised completely. Moreover, repeated numerous sections of margins of the original mass did not show transitional cells. However, the presence of transitional cells near the margin may shed a dim light to the case. Inoculation of transitional cells from the unnoticed minute crack in the margin might be a possibilityo This case illustrates that heterotopic bone formation after urinary epithelial inoculations could give rise to benign as well as malignant osseous tumorso REFERENCES
FIG. 3. A, histologic appearance of specimen 1 shows bony tissue formation around hyperplastic transitional epithelium. H & E, reduced from X250. B, histologic appearance of specimen 2 shows transitional cell carcinoma invasion into surrounding soft tissue. H & E, reduced from X400. DISCUSSION
Experimental studies have shown that urinary and gastrointestinal epithelium could proliferate in soft tissues and exert
L Abeshouse, B. S.: Heterotopic bone formation following suprapubic prostatectomy. J. Urol., 59: 50, 1948. 2. Neuhof, ff: Fascia transplantation into visceral defects: an experimental and clinical studyo Surg., Gyneco & Obsto, 24: 383, 19170 3. Sewell, W. S., Siceluff, J. G. and Horton, J. D.: Extraskeletal bone formation following suprapubic prostatectomy: case reporto Jo Urol., 62: 842, 19490 4. Bell, J. W. and Knudtson, K. Po: Soft tissue osteogenesis with gall bladder mucosa! autografts. Lab. Investo, 10: 397, 1961. 5. Huggins, Co R: The formation of bone under the influence of epithelium of the urinary tract. Arch. Surgo, 22: 377, 19310 60 Apostolidis, No S., Legakis, N. C., Gregoriadis, G. C., Androulakakis, P. k and Romanos, A. N.: Heterotopic bone formation in abdominal operation scars. Report of six cases with review of the literature. Amer. J. Surgo, 142: 555, 1981.