Beitr. Path. Bd. 155,208-211 (1975)
Department of Pathology (Provisional Managing Director: Prof. Dr. F. Niedobitek) and Department of Urology (Director: Prof. Dr. R. Nagel), Free University of Berlin, Westend Clinic
Nephrogenic Adenoma A Rare Epithelial Tumor of the Urinary Bladder in a Child Nephrogenes Adenom Ein se1tener epithelialer Harnblasentumor bei einem Kind K. DONHUIJSEN and W. LEISTENSCHNEIDER With
2
Figures ·Received September 6, 1974' Accepted November 14, 1974
Key words: Nephrogenic adenoma - Urinary bladder - Hamartoma Dysontogenic tumors
In children and adolescents bladder tumors are rare (Javadpour and Mostofi, 1969). Mesenchymal tumors of the bladder appear more frequently in children than epithelial tumors (Siegel and Pincus, 1966). This report describes a case of an infrequent benign tumor of the bladder of which according to our knowledge - only 22 cases have been recorded in the literature until now. The patient was an 81h-year-old female with double kidneys on both sides. A ureterocele of walnut size had been resected on the right side during the first year of life and a pyelo-uretero-anastomosis had been made on the right at the age of 2 years because of megaloureter. At the age of 8, multiple papillary tumors of pinhead to pea size were removed by transurethral resection primarily from the rear wall, both side walls and the roof of the bladder. Trigonum, ostia and the neck of the bladder were free from tumor. 4 months later recurrent tumor was removed from the side walls, the transition to the roof and from the base of the bladder. There is no evidence of local recurrence 4 months after the last treatment.
Histological examination of the surgical material showed small pieces of tissue exhibiting definitely papillary structures covered by a single layer
Nephrogenic Adenoma·
209
Fig. I. Nephrogenic adenoma of the urinary bladder with papillary and adenomatoid elements. H & E; X 50.
L' Fig. 2. E; X
Typicale single layer of low columnar epithelium of nephrogenic adenoma. H & 200.
210 . K. Donhuijsen and W. Leistensdmeider of cuboidal to low columnar epithelium with uniform basophilic nuclei. Numerous invaginations are lined with similar epithelium which is also found in often closely packed tubules of varying diameter. The edematous stroma frequently contains large capillaries. Typical stratified urothelium is only occasionally found. Bladder tumors in children and juveniles are predominantly of mesenchymal origin; in decreasing frequency sarcomas, myxomas, hemangiomas, neurofibromas and fibromas were found (Siegel and Pincus, 1966). The rarer epithelial tumors in the first and second decade of life are nearly exclusively papillomas, only occasionally transitional cell carcinomas were described (Javadpour and Mostofi, 1969). The above bladder tumor was called nephrogenic adenoma by Friedman and Kuhlenbeck (1950). From the 22 cases reported up to now 3 have been diagnosed before the third decade, 8 during the third decade and 9 during the seventh decade of life. 17 of the 22 cases concerned males. The clinical course and histomorphological picture were always benign, with the exception of a mesonephrogenic adenocarcinoma in a 43-year-old man (Dow and Young, 1968). Recurrence of the tumor was found over several months in 4 patients including the case reported above. The histogenesis of the so-called nephrogenic adenoma is unexplained. Several authors (Mostofi, 1954; Goldman, 1972; Sussman, Brice and Gray, 1974) consider it a metaplasia, but others feel that it is a hamartoma (Christoffersen and Moeller, 1972; Friedman and Kuhlenbeck, 1950; Hasen, 1962). Combination with a double kidney was found in 3 cases, with hydronephrosis in 3 cases and with a previously ruptured urinary bladder m 2 cases. In the differential diagnosis, adenomatoid changes which occur more frequently in the bladder must be excluded. Besides the various forms of adenocarcinoma of the bladder, it has to be thought of the following benign changes: hyperplastic paraurethral and paraprostatic glands, cystitis glandularis and cystitis cystica, urachal and cloacal rests, adenofibromas as well as endometriosis of the bladder. All authors agree that surgical removal of the tumor is the adequate treatment followed by regular control examinations just as with the bladder papilloma.
Zusammenfassung Berichtet wird iiber einen seltenen Harnblasentumor bei einem achtjahrigen Madchen,
dessen Histologie dem im Schrifttum in 22 Fallen beschriebenen sogenannten nephrogenen Adenom entspricht. In der Altersverteilung dominiert das 3. und 7. Lebensjahrzehnt. Das
Nephrogenic Adenoma· 2II mannliche Geschlecht ist bevorzugt. Auf eine auffallend haufige Kombination mit MiBbildungen der ableitenden Harnwege wird hingewiesen. Tumorresektion und regelmaBige Kontrolluntersuchungen erscheinen notwendig, da iiber Rezidive in 4 Fallen und maligne Entartung in einem Fall berichtet wird.
References 1.
2.
3. 4. 5. 6. 7· 8.
9.
Christoffersen, J., and Moeler, J. E.: Adenomatoid tumors of the unnary bladder. Scand. J. Uro!' Nephro!. 6, 295-298 (1972). Dow, ]. A., and Young, J. D.: Mesonephric adenocarcinoma of the bladder. J. Urol. 100,466-469 (1968). Friedman, N. B., and Kuhlenbeck, H.: Adenomatoid tumors of the bladder reproducing renal structures (Nephrogenic adenomas). J. Urol. 64, 657-670 (1950). Goldman, R. 1.: Nephrogenic metaplasia (Nephrogenic adenoma, adenomatoid tumor) of the bladder. J. Urol. 108, 565-567 (1972). Hasen, H. B.: Nephrogenic adenoma of the bladder. J. Urol. 88, 629-630 (1962). ]avadpour, N., and Mostofi, F. K.: Primary epithelial tumors of the bladder in the first two decades of life. J. Urol. 101, 706-710 (1969). Mostofi, F. K.: Potentialities of bladder epethelium. J. Urol. 71, 705-714 (1954). Siegel, W. H., and Pincus, M. B.: Epithelial bladder tumors in children. J. Urol. 101, 55-56 (19 66 ). Sussman, E. B., Brice, M., and G. F. Gray: Nephrogenic metaplasia of the bladder. J. Urol. I I 1,34-35 (1974).
Konrad Donhuijsen, Institut fiir Pathologie, Klinikum Westend, D-l Berlin 19, Spandauer Damm 130, West Germany
Department of Pathology (Provisional Managing Director: Prof. Dr. F. Niedobitek) and Department of Urology (Director: Prof. Dr. R. Nagel), Free University of Berlin, Westend Clinic
Nephrogenic Adenoma A Rare Epithelial Tumor of the Urinary Bladder in a Child Nephrogenes Adenom Ein se1tener epithelialer Harnblasentumor bei einem Kind K. DONHUIJSEN and W. LEISTENSCHNEIDER With
2
Figures ·Received September 6, 1974' Accepted November 14, 1974
Key words: Nephrogenic adenoma - Urinary bladder - Hamartoma Dysontogenic tumors
In children and adolescents bladder tumors are rare (Javadpour and Mostofi, 1969). Mesenchymal tumors of the bladder appear more frequently in children than epithelial tumors (Siegel and Pincus, 1966). This report describes a case of an infrequent benign tumor of the bladder of which according to our knowledge - only 22 cases have been recorded in the literature until now. The patient was an 81h-year-old female with double kidneys on both sides. A ureterocele of walnut size had been resected on the right side during the first year of life and a pyelo-uretero-anastomosis had been made on the right at the age of 2 years because of megaloureter. At the age of 8, multiple papillary tumors of pinhead to pea size were removed by transurethral resection primarily from the rear wall, both side walls and the roof of the bladder. Trigonum, ostia and the neck of the bladder were free from tumor. 4 months later recurrent tumor was removed from the side walls, the transition to the roof and from the base of the bladder. There is no evidence of local recurrence 4 months after the last treatment.
Histological examination of the surgical material showed small pieces of tissue exhibiting definitely papillary structures covered by a single layer
Nephrogenic Adenoma·
209
Fig. I. Nephrogenic adenoma of the urinary bladder with papillary and adenomatoid elements. H & E; X 50.
L' Fig. 2. E; X
Typicale single layer of low columnar epithelium of nephrogenic adenoma. H & 200.
210 . K. Donhuijsen and W. Leistensdmeider of cuboidal to low columnar epithelium with uniform basophilic nuclei. Numerous invaginations are lined with similar epithelium which is also found in often closely packed tubules of varying diameter. The edematous stroma frequently contains large capillaries. Typical stratified urothelium is only occasionally found. Bladder tumors in children and juveniles are predominantly of mesenchymal origin; in decreasing frequency sarcomas, myxomas, hemangiomas, neurofibromas and fibromas were found (Siegel and Pincus, 1966). The rarer epithelial tumors in the first and second decade of life are nearly exclusively papillomas, only occasionally transitional cell carcinomas were described (Javadpour and Mostofi, 1969). The above bladder tumor was called nephrogenic adenoma by Friedman and Kuhlenbeck (1950). From the 22 cases reported up to now 3 have been diagnosed before the third decade, 8 during the third decade and 9 during the seventh decade of life. 17 of the 22 cases concerned males. The clinical course and histomorphological picture were always benign, with the exception of a mesonephrogenic adenocarcinoma in a 43-year-old man (Dow and Young, 1968). Recurrence of the tumor was found over several months in 4 patients including the case reported above. The histogenesis of the so-called nephrogenic adenoma is unexplained. Several authors (Mostofi, 1954; Goldman, 1972; Sussman, Brice and Gray, 1974) consider it a metaplasia, but others feel that it is a hamartoma (Christoffersen and Moeller, 1972; Friedman and Kuhlenbeck, 1950; Hasen, 1962). Combination with a double kidney was found in 3 cases, with hydronephrosis in 3 cases and with a previously ruptured urinary bladder m 2 cases. In the differential diagnosis, adenomatoid changes which occur more frequently in the bladder must be excluded. Besides the various forms of adenocarcinoma of the bladder, it has to be thought of the following benign changes: hyperplastic paraurethral and paraprostatic glands, cystitis glandularis and cystitis cystica, urachal and cloacal rests, adenofibromas as well as endometriosis of the bladder. All authors agree that surgical removal of the tumor is the adequate treatment followed by regular control examinations just as with the bladder papilloma.
Zusammenfassung Berichtet wird iiber einen seltenen Harnblasentumor bei einem achtjahrigen Madchen,
dessen Histologie dem im Schrifttum in 22 Fallen beschriebenen sogenannten nephrogenen Adenom entspricht. In der Altersverteilung dominiert das 3. und 7. Lebensjahrzehnt. Das
Nephrogenic Adenoma· 2II mannliche Geschlecht ist bevorzugt. Auf eine auffallend haufige Kombination mit MiBbildungen der ableitenden Harnwege wird hingewiesen. Tumorresektion und regelmaBige Kontrolluntersuchungen erscheinen notwendig, da iiber Rezidive in 4 Fallen und maligne Entartung in einem Fall berichtet wird.
References 1.
2.
3. 4. 5. 6. 7· 8.
9.
Christoffersen, J., and Moeler, J. E.: Adenomatoid tumors of the unnary bladder. Scand. J. Uro!' Nephro!. 6, 295-298 (1972). Dow, ]. A., and Young, J. D.: Mesonephric adenocarcinoma of the bladder. J. Urol. 100,466-469 (1968). Friedman, N. B., and Kuhlenbeck, H.: Adenomatoid tumors of the bladder reproducing renal structures (Nephrogenic adenomas). J. Urol. 64, 657-670 (1950). Goldman, R. 1.: Nephrogenic metaplasia (Nephrogenic adenoma, adenomatoid tumor) of the bladder. J. Urol. 108, 565-567 (1972). Hasen, H. B.: Nephrogenic adenoma of the bladder. J. Urol. 88, 629-630 (1962). ]avadpour, N., and Mostofi, F. K.: Primary epithelial tumors of the bladder in the first two decades of life. J. Urol. 101, 706-710 (1969). Mostofi, F. K.: Potentialities of bladder epethelium. J. Urol. 71, 705-714 (1954). Siegel, W. H., and Pincus, M. B.: Epithelial bladder tumors in children. J. Urol. 101, 55-56 (19 66 ). Sussman, E. B., Brice, M., and G. F. Gray: Nephrogenic metaplasia of the bladder. J. Urol. I I 1,34-35 (1974).
Konrad Donhuijsen, Institut fiir Pathologie, Klinikum Westend, D-l Berlin 19, Spandauer Damm 130, West Germany
