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Bilateral brenner tumors: A case report and review of the literature
Medical hztelligence
BILATERAL BRENNER TUMORS: A CASE REPORT AND REVIEW OF THE LITERATURE
j. DANIELSLAMPING,M.D.,* A.XDJaslEs G. Bt.vwHE, M.D., F.A.C.O.G., F.A.C.S.~"
Abstract Although the first Brenner tumor was reported in 1898 by MacNaughtoa-Jones, it was not until 1907 that Fritz Brenner deselqbed the tumor that bears his name. Since then more than 500 cases have been reported with bilateragty ranging fiom 3.7 to 8.0 per cent. However, this percentage is probably high. The tumor was originally thought to arise from follicular epithelium. Several other etiolog4es of the Bremter tumor have been suggested, including celomic smface epithelium, Walthard rests, teratoma, urothel~am, and fete ovarii. The majority of Brem~er tumors are inert, but there have been reports suggesting endocrine activity. The question of malignant transformation of the Brem~er tumor has gained attention since Von Numers desc~qbed the first malignant Brenner tumor in 1945. The determination of malignancy of these tumors has been hampered by vague histolog& criteria for malignancy. The eo~tsion surrounding the histogenesis, hormonal capabilities, and malignant potential of the Brenner tumor has persisted. A case report of bilateral Bremwr tumors of the ovaries is presented as well as a review of the literature with emphasis on the bilaterality of Brem~er tlonofs.
An interesting case o f bilateral B r e n n e r tumors associated with proliferative endometrium anti multiple uterine leiomyomas encouraged a review o f the literature relating to B r e n n e r tumors. This review revealed that confilsion s u r r o u n d s the histogenesis, hormonal capabilities, and malignant potential o f Brenner tumors. We present a case o f bilateral B r e n n e r tumors o f the ovaries with a survey o f *Resident, Department of Obstetrics and Gynecology, St. John's Mercy Medical Center, Creve Coeur, Missouri. "~Chairman, Department of Obstetrics and Gynecology, and Chief, Section of Gynecologic Oncology, St. John's Mercy Medical Center, Creve Coeur, Missouri.
the literature concerning B r e n n e r emphasizing their bilaterality.
tumors
CASE R E P O R T A 39 )'ear old gravida It, para II white female complained o f increasingly severe d y s m e n o r r h e a for two )'ears in spite o f ovarian suppression. A 6 cm. firm n o d u l a r right adnexal mass was palpable d u r i n g physical examination. On admission, the uterus was anterior, irregular, and enlarged. T h e left ovary was enlarged and the right was twice normal size, hard, a n d tender. Laparoscopy confirmed the e n l a r g e m e n t "of the ovaries and d e m o n s t r a t e d an enlarged irregular uterus. A total abdominal hysterectomy and bilateral salpingo-oophorectomy and incidental a p p e n d e c t o m y were p e r t o r m e d . Frozen section examination o f the right ovary was p e r f o r m e d . T h e diagnosis was a benign B r e n n e r tumor. T h e patient was discharged on the eighth postoperative day after an uneventfid recovery. T h e right ovary measured 6 by 4 by 3 cm. and when sectioned revealed a 3.5 by 3 by 2 cm., well delineated, yellowish gray, firm mass, which in some areas completely replaced the ovary. T h e left ovary measured 3.5 by 2.5 by 1.5 cm., showed n u m e r o u s subcapsular cysts filled with clear serous fluid, and contained a 1 by 1 by 0.7 cm. yellowish gray nodule. T h e tubes were u n r e m a r k a b l e and the utertts was grossly distorted by leiomyomas. T h e right a n d left ovaries contained B r e n n e r tumors and n u m e r o u s follicular cysts. T h e e n d o m e t r i u m was proliferative. DISCUSSION MacNaughton-Jones TM r e p o r t e d the first B r e n n e r t u m o r in 1898. However, in 1907 Fritz B r e n n e r 2 described the t u m o r that bears his name. Since 1907 more than 500 B r e n n e r tumors have been r e p o r t e d , including 37 cases o f bilaterality. 1'~ According to Hertig and Gore, 8 the B r e n n e r t u m o r accounts for 1.7 p e r cent o f all solid ovarian tumors. T h e bilaterality o f - B r e n n e r tumors is variously r e p o r t e d to range from 3.7 to 8.0 p e r c e n t ? ' ~"6. ~_.~.._,9 However, the incidence o f bilaterality is probably lfigh, since not all cases o f unilateral B r e n n e r tumors are reported. Novak and
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H U M A N I ' A T H O L O G Y - - V O L U M E 8, N U M B E R 5 W o o d r u f f "z~ in a study o f 90 B r e l m e r tumors r e p o r t e d no cases o f bilaterality. Authorities agree that the B r e n n e r t u m o r usually occurs between the fourth and sixth decades. It is usually an asymptomatic incistental operative finding. Grossly it is a solid although occasionally cystic t u m o r a n d is rarely bilateral. Histologically it is composed o f nests o f solid or cystic epithelial cells surr o u n d e d by a dense fibrous stroma? ~ Brenner's original histogenetic concept p r o p o s e d a derivation from follicular epithelium. His concept is not s u p p o r t e d today, because a B r e n n e r t n m o r seldom coexists with a t u m o r o f similar histogenetic origin, such as a granulosa cell tumor. Also B r e n n e r tmnors are rarely malignant, whereas approximately 34 per cent o f granulosa cell tumors are malignant3 ~ O t h e r suggested etiologies o f B r e n n e r tumors are celomic surface epithelium, Walt h a r d rests, teratoma, nrothelium, and rete ovarii. W a l t h a r d rests were suggested by Meyer, ~7 but this is anatomically tmlikely since B r e n n e r tumors often occur in the hihtm but not in the oviduct? ~ A teratomatous origin was proposed because o f the frequent association of B r e n n e r tumors and mucinous cystadenomas; the latter were thought to be variants o f teratomas. According to present theories, however, serous and mucinous cystadenomas are derived from celomic surface epithelium. Consequently the teratomatous theory is untenable. T h e r e m a i n i n g p r o p o s e d origins (urothelium, rete ovarii, and celomic epithelium) are difficult to separate. Sternberg a~ has combined these theories by posttflating an origin from urothelial metaplasia o f ovarian surface epitheliunL Miles et al? s assumed that the external ovarian surface has the potential to form epithelium similar to b l a d d e r epithelium. Proponents o f the rete ovarii theory have interpreted the rete a n d thus the B r e n n e r t u m o r as being a m e s o n e p h r i c structure. T h e r e is also evidence to suggest that the rete nmy be derived from celomic epithelium, zr Tire controversy persists concerning the hormonal activity o f B r e n n e r tumors. T h e majority o f these tumors are inert; however, there are recent reports suggesting endocrine activity3 z Schiffmann ~a in 1932, and later T e o h zs and MacKinlay, 15 suggested a possible hormonal relationship be[ween B r e n n e r tumors and the e n d o m e t r i u m resuhing in abnormal uterine bleeding. However, most attthorities considered this association coincidental. In 1962 Ming and Goldman r9 reported that 75 per" cent o f the benign B r e n n e r tumors ira their series o f postmenopausal women were associated with either hyperplastic, carcinomatous, o r polypoid endo-
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nletrium. Shay and JanovskF~ r e p o r t e d a case o f a malignant B r e n n e r t u m o r associated with endometrial adenocarcinoma. Hamwi 7 and others have investigated testosterone synthesis by a B r e n n e r tumor. T h e application o f nhrastrnctural techn i q u e s a n d cytologic staining methods has allowed f u r t h e r investigation concerning the hormonal potential o f B r e n n e r tumors. Silverberg ~5 in his clinicopathologic review concluded that the evidence for specific endocrine activity of B r e n n e r tumors was weak. Using electron microscopy, Silverberg26 indicated that there might be a subclinical level o f hormonal activity associated with B r e n n e r tumors. Morris and Scull) a~ p r o p o s e d that any ovarian t u m o r can have endocrine activity. This activity presumably restilts from stimulation o f the ovarian stroma to differentiate into functionally active cells. H u g h e s d o n 9 believes that tiffs stimtflation may be tire result o f stretching of the ovarian stroma by an exp a n d i n g tumor. T h e possibility o f malignant transformation o f the B r e n n e r t u m o r has gained increased p r o m i n e n c e since Von Numers a~ described the first conclusively malignant B r e n n e r t u m o r in 1945. Idelson's review H in 1963 emplmsized the lack o f definite histologic criteria for malignancy. Hull and CampbelP ~ reviewed the literature relating to malignant B r e n n e r tumors a n d p r o p o s e d newly defined diagnostic criteria. T h e questionable validity o f man); o f the r e p o r t e d malignant B r e n n e r tumors has obscured the exact incidence o f tiffs malignancy, which is variously r e p o r t e d to be between 5 a n d 9 p e r cent. 6 Miles and N o r r i s ) s reviewing all the cases o f malignant a n d suspected malignant B r e n n e r tumors on file at the A r m e d Forces Institute o f Pathology, emplmsized a B r e n n e r neoplasm that is intermediate between the benign and fnlly malignant B r e n n e r tumor. T h e B r e n n e r t u m o r has occasionally been associated with other ovarian disease, frequently a mucinous cystadenoma. Epple and Bossert 4 r e p o r t e d a B r e n n e r t u m o r associated with a mucinous cystadenocarcinoma o f the contralateral ovary and a squamous cell carcinoma o f the cervix. Both a B r e n n e r t u m o r in tire wall o f a benign cystic teratonm and a malignant teratonm were r e p o r t e d from the Mayo Clinic. B r e n n e r tumors have also been associated with chronic oophoritis, simple and follicular cysts, germinal inclusion cysts, hilus cell hyperplasia, ovarian adenocarcinoma, adenofibroma, and endometriosis. Gifford and Birch in 1969 r e p o r t e d two cases o f bilateral B r e n n e r tnmors and a d d e d them to their review o f previously r e p o r t e d cases.a,6, ~2,29 T h e y stated that "In contrast to
MEDICAL t h e u n i l a t e r a l t u m o r s , it a p p e a r s t h a t b i l a t e r a l t u m o r s o c c u r at a s l i g h t l y e a r l i e r a g e , a r e associated with lower parity and higher abortion incidence and have a lower rate of malignancy. On the other band, nnilateral and bilateral Brenner tumors are similar in terms of symptomatology, laterality of tumor size and postmenopausally associated endometrial activity. ''6 The treatment of Brenner tumors is unilateral oophorectomy. In postmenopausal w o m e n t h e p r o c e d u r e o f c h o i c e is a h y s t e r ectomy with bilateral salpingo-oophorectomy) 3 I f m a l i g n a n t c h a n g e is e n c o u n t e r e d in t h e surgical specimen, a total abdominal hysterectomy with bilateral salpingo-ooplmrectomy is r e c o m m e n d e d as t h e i n i t i a l t r e a t m e n t in menopausal and postmenopausal women. If a m a l i g n a n t B r e n n e r t u m o r is e n c o u n t e r e d in a y o u n g e r w o m a n , t h e r a p y is m o r e likely to b e i n d i v i d u a l i z e d a n d c o n s e r v a t i v e , s u c h as a unilateral oophorectomy. The value of radiat i o n t h e r a p y as a t h e r a p e u t i c m o d a l i t y f o r r e s i d u a l o r r e c u r r e n t t u m o r is p r e s e n t l y u n determined.
References 1~ Badway, R. E., Jorgenson, O. it., and Cromer, J. K.: Bilateral Brenner tumors--review of literature and report of case. Med. Ann. D. C.,33:106, 1964. 2. Brenner, F.: Das Oophoroma Folliculare. Frankfurt. Z. Path., 1:150, 1907. 3. Christian, C. D., andJanovski, N. A.: Bilateral Brenner tumors. Ant. J. Obstet. Gynecol., 83:105, 1962. -4. Epple, tl. tl., and Bossert, L. J.: Three simultaneous neoplasms of the female genitalia: a Brenner tumor, a pseuclomucinous cystadenocarcinoma of the contralateral ovary, and a squamous cell carcinoma of the cervix. Obstet. Gynecol., 11:661, 1958. 5. Farrar, tt. K.,Jr., and Greene, R. R.: Bilateral Brenner tumors of tile ovary. Am. J. Obstet. Gynecol., 80: 1089, 1960. 6. Gifford, A. B.," and Birch, tt. W.: Bilateral Brenner tumors of the ovary. J. Med. Assoc. Georgia, 58:145, 1969. 7. tlamwi, G.J., et al.: Testosterone synthesis by a Brenner tumor, Part 1. Clinical evidence of masculinization during pregnanc). Part Ii. In ~itro biosynthetic steroid conversion of a Brenner tumor. Am. J. Obstet. Gynecol., 86:!015, 1963. 8. tlertig, A. T., and Gore, I1.: Tumors of the ovary and fallopiaxt tube. In Atlas of T u m o r Pathology, Sec. I X, Fasc. 33. Washington, D. C., Armed Forces Institute of Pathology, 1961, p. 124. 9. llughesdon, P. E.: Thecal and allied reactions in epithelial ovarian turnouts. ]. Obstet. Gynecol. Brit. Emp., 65:702, 1958. 10. tlnll, M. G. R., and Campbell, G. R.: The ntalignant Brenner tumor. Obstet. Gynecol., 42:527,, 1973. 11. Idelson, M. G.: Malignancy in Brenner tumors of the ovary, with comments on histogenesis and possible estrogen production. Obstet. Gynecol. Survey, 18: 246, 1963. 12. Kendall, B., and Bowers, P. A.: Bilateral Brenner tumor of the ovaries. Am. J. Ohstet. Gynecol., 80:439, 1960. 13. Kismer, R. W.: Gynecolo~" Principles attd Practice. Ed. 2. Chicago, Year Book Medical Publishers, Inc., 1972, p. 409. 14. Kraus, F. T.: Gynecologic Pathology. St. Louis, The C. V. Mosby Company, 1967, pp. 324,313.
INTELLIGENCE
15. MacKinlay, C. J.: Brenner tumours of the ovary. J. Obstet. Gynecol. Brit. Emp., 65:58, 1056. 16. MacNaughtou-Jones, t1.: Uterine fibroid with an anomalous ovarian tnmour. Trails. Lond. Obstet. Soc., 40:154,213, 1898. 17. Meyer, R.: Uber verschiedene Erschlintmgsformen der als typus Brenner bekannten Eirstockageschwulst, ihre Zuordnung unter Andere Overialgerschwillste. Arch. G)naekol., 148:5tl, 1932. 18. Miles, 1'. A., Joj, M. C., and Norris, tt. J.: l'roliferative 9 and malignant Brenner tumors of the ovary. Cancer, 30:174, 1972. 19. Ming, S. C., and Goldman, tt4 tlormonal activity of Brenner tumors in postmenopausal women. Am. J. Obstet. Gynecol., 83:666, 1962. 20. Morris,J. M., and Scully, R. E.: Endocrine Pathology of the Ovary. St. Louis, Tile C. V. Mosby Co., 1958. 21. Novak, E. R., and Woodruff, J. D.: Novak's Gynecologic and Obstetric Pathotogy. Ed. 5. lqdladelphia. W. B. Saunders Company, 1962. 22. Novak, E. R., Woodruff, J. D., and Linthicum, J. M.: Evaluation of the unclassified tumors of the. Ovarian T u m o r Registry (1942-1952). Am. J. Ohstet. Gynecol., 87:999. 1963. 23. Schiflmann, J.: Postklimakterische Blutung und Brennerscher ovarial-tumor. Arch GynS.k.. 150:159, 1932. 24. Shay, M. D., and Janovs~, N. A.: Mangnant Brenner tumor associated with endometrial adeuocardinoma. Obstet. Gynecol., 22:246, 1963. 25. Silverberg, S. G.: Brenuer tumor of the ovary. Cancer, 28:588, 1971. 26. Silverberg. S. G., and Willson, M. A.: Uhrastructure of the Brenner tumor. Amer. J. Obstet. Gynecol., 112: 91, 1972. 27. Sternberg, W. I1.: Nonfunctioning Ovarian Neoplasms in the Ovary. Baltimore, Tile Williams & Wilkins Co., 1963, p. 209. 28. Teoh, T. B.: The histogenesis of Brenner tumors of the ovary, J. Path. Bact., 66:441, 1953. 29. Varden, L. C.: Bilateral Brenner tumors of the ovaries. *led. Ann. D. C., 33:70, 196t. 30. Von Numers, C.: A contribution to the case knowledge and histology of the Brenner tumor. Acta Obstet. Gynecol. Scand., 25(Suppl. 2):114, 1945.
MUCOCELE OF THE APPENDIX SECONDARY TO OBSTRUCTION BY ENDOMETRIOSIS MARC R. HAI'KE, M . D . , * AND BRADLEY BIGELOW, M.D.'~
Abstract
A mucocele of the appendix secondaO' to obstruction by endometriosis is reported and the relevant literature reviewed. The theories of the pathogenesis of appeudiceal mucocele are reviewed *Medical Fellow, I ) e l m r t m c n t o1 Lal)or;.ttory Medicine atttl l'athology (Surgical P,tthology), University o f M i n n e s o t a School o f Medicine, Minneapolis, Minnesota. tAssociate l'rofessor o f Pathology, New York University School o f Medicine, New York, New York.
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BILATERAL BRENNER TUMORS: A CASE REPORT AND REVIEW OF THE LITERATURE
j. DANIELSLAMPING,M.D.,* A.XDJaslEs G. Bt.vwHE, M.D., F.A.C.O.G., F.A.C.S.~"
Abstract Although the first Brenner tumor was reported in 1898 by MacNaughtoa-Jones, it was not until 1907 that Fritz Brenner deselqbed the tumor that bears his name. Since then more than 500 cases have been reported with bilateragty ranging fiom 3.7 to 8.0 per cent. However, this percentage is probably high. The tumor was originally thought to arise from follicular epithelium. Several other etiolog4es of the Bremter tumor have been suggested, including celomic smface epithelium, Walthard rests, teratoma, urothel~am, and fete ovarii. The majority of Brem~er tumors are inert, but there have been reports suggesting endocrine activity. The question of malignant transformation of the Brem~er tumor has gained attention since Von Numers desc~qbed the first malignant Brenner tumor in 1945. The determination of malignancy of these tumors has been hampered by vague histolog& criteria for malignancy. The eo~tsion surrounding the histogenesis, hormonal capabilities, and malignant potential of the Brenner tumor has persisted. A case report of bilateral Bremwr tumors of the ovaries is presented as well as a review of the literature with emphasis on the bilaterality of Brem~er tlonofs.
An interesting case o f bilateral B r e n n e r tumors associated with proliferative endometrium anti multiple uterine leiomyomas encouraged a review o f the literature relating to B r e n n e r tumors. This review revealed that confilsion s u r r o u n d s the histogenesis, hormonal capabilities, and malignant potential o f Brenner tumors. We present a case o f bilateral B r e n n e r tumors o f the ovaries with a survey o f *Resident, Department of Obstetrics and Gynecology, St. John's Mercy Medical Center, Creve Coeur, Missouri. "~Chairman, Department of Obstetrics and Gynecology, and Chief, Section of Gynecologic Oncology, St. John's Mercy Medical Center, Creve Coeur, Missouri.
the literature concerning B r e n n e r emphasizing their bilaterality.
tumors
CASE R E P O R T A 39 )'ear old gravida It, para II white female complained o f increasingly severe d y s m e n o r r h e a for two )'ears in spite o f ovarian suppression. A 6 cm. firm n o d u l a r right adnexal mass was palpable d u r i n g physical examination. On admission, the uterus was anterior, irregular, and enlarged. T h e left ovary was enlarged and the right was twice normal size, hard, a n d tender. Laparoscopy confirmed the e n l a r g e m e n t "of the ovaries and d e m o n s t r a t e d an enlarged irregular uterus. A total abdominal hysterectomy and bilateral salpingo-oophorectomy and incidental a p p e n d e c t o m y were p e r t o r m e d . Frozen section examination o f the right ovary was p e r f o r m e d . T h e diagnosis was a benign B r e n n e r tumor. T h e patient was discharged on the eighth postoperative day after an uneventfid recovery. T h e right ovary measured 6 by 4 by 3 cm. and when sectioned revealed a 3.5 by 3 by 2 cm., well delineated, yellowish gray, firm mass, which in some areas completely replaced the ovary. T h e left ovary measured 3.5 by 2.5 by 1.5 cm., showed n u m e r o u s subcapsular cysts filled with clear serous fluid, and contained a 1 by 1 by 0.7 cm. yellowish gray nodule. T h e tubes were u n r e m a r k a b l e and the utertts was grossly distorted by leiomyomas. T h e right a n d left ovaries contained B r e n n e r tumors and n u m e r o u s follicular cysts. T h e e n d o m e t r i u m was proliferative. DISCUSSION MacNaughton-Jones TM r e p o r t e d the first B r e n n e r t u m o r in 1898. However, in 1907 Fritz B r e n n e r 2 described the t u m o r that bears his name. Since 1907 more than 500 B r e n n e r tumors have been r e p o r t e d , including 37 cases o f bilaterality. 1'~ According to Hertig and Gore, 8 the B r e n n e r t u m o r accounts for 1.7 p e r cent o f all solid ovarian tumors. T h e bilaterality o f - B r e n n e r tumors is variously r e p o r t e d to range from 3.7 to 8.0 p e r c e n t ? ' ~"6. ~_.~.._,9 However, the incidence o f bilaterality is probably lfigh, since not all cases o f unilateral B r e n n e r tumors are reported. Novak and
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H U M A N I ' A T H O L O G Y - - V O L U M E 8, N U M B E R 5 W o o d r u f f "z~ in a study o f 90 B r e l m e r tumors r e p o r t e d no cases o f bilaterality. Authorities agree that the B r e n n e r t u m o r usually occurs between the fourth and sixth decades. It is usually an asymptomatic incistental operative finding. Grossly it is a solid although occasionally cystic t u m o r a n d is rarely bilateral. Histologically it is composed o f nests o f solid or cystic epithelial cells surr o u n d e d by a dense fibrous stroma? ~ Brenner's original histogenetic concept p r o p o s e d a derivation from follicular epithelium. His concept is not s u p p o r t e d today, because a B r e n n e r t n m o r seldom coexists with a t u m o r o f similar histogenetic origin, such as a granulosa cell tumor. Also B r e n n e r tmnors are rarely malignant, whereas approximately 34 per cent o f granulosa cell tumors are malignant3 ~ O t h e r suggested etiologies o f B r e n n e r tumors are celomic surface epithelium, Walt h a r d rests, teratoma, nrothelium, and rete ovarii. W a l t h a r d rests were suggested by Meyer, ~7 but this is anatomically tmlikely since B r e n n e r tumors often occur in the hihtm but not in the oviduct? ~ A teratomatous origin was proposed because o f the frequent association of B r e n n e r tumors and mucinous cystadenomas; the latter were thought to be variants o f teratomas. According to present theories, however, serous and mucinous cystadenomas are derived from celomic surface epithelium. Consequently the teratomatous theory is untenable. T h e r e m a i n i n g p r o p o s e d origins (urothelium, rete ovarii, and celomic epithelium) are difficult to separate. Sternberg a~ has combined these theories by posttflating an origin from urothelial metaplasia o f ovarian surface epitheliunL Miles et al? s assumed that the external ovarian surface has the potential to form epithelium similar to b l a d d e r epithelium. Proponents o f the rete ovarii theory have interpreted the rete a n d thus the B r e n n e r t u m o r as being a m e s o n e p h r i c structure. T h e r e is also evidence to suggest that the rete nmy be derived from celomic epithelium, zr Tire controversy persists concerning the hormonal activity o f B r e n n e r tumors. T h e majority o f these tumors are inert; however, there are recent reports suggesting endocrine activity3 z Schiffmann ~a in 1932, and later T e o h zs and MacKinlay, 15 suggested a possible hormonal relationship be[ween B r e n n e r tumors and the e n d o m e t r i u m resuhing in abnormal uterine bleeding. However, most attthorities considered this association coincidental. In 1962 Ming and Goldman r9 reported that 75 per" cent o f the benign B r e n n e r tumors ira their series o f postmenopausal women were associated with either hyperplastic, carcinomatous, o r polypoid endo-
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nletrium. Shay and JanovskF~ r e p o r t e d a case o f a malignant B r e n n e r t u m o r associated with endometrial adenocarcinoma. Hamwi 7 and others have investigated testosterone synthesis by a B r e n n e r tumor. T h e application o f nhrastrnctural techn i q u e s a n d cytologic staining methods has allowed f u r t h e r investigation concerning the hormonal potential o f B r e n n e r tumors. Silverberg ~5 in his clinicopathologic review concluded that the evidence for specific endocrine activity of B r e n n e r tumors was weak. Using electron microscopy, Silverberg26 indicated that there might be a subclinical level o f hormonal activity associated with B r e n n e r tumors. Morris and Scull) a~ p r o p o s e d that any ovarian t u m o r can have endocrine activity. This activity presumably restilts from stimulation o f the ovarian stroma to differentiate into functionally active cells. H u g h e s d o n 9 believes that tiffs stimtflation may be tire result o f stretching of the ovarian stroma by an exp a n d i n g tumor. T h e possibility o f malignant transformation o f the B r e n n e r t u m o r has gained increased p r o m i n e n c e since Von Numers a~ described the first conclusively malignant B r e n n e r t u m o r in 1945. Idelson's review H in 1963 emplmsized the lack o f definite histologic criteria for malignancy. Hull and CampbelP ~ reviewed the literature relating to malignant B r e n n e r tumors a n d p r o p o s e d newly defined diagnostic criteria. T h e questionable validity o f man); o f the r e p o r t e d malignant B r e n n e r tumors has obscured the exact incidence o f tiffs malignancy, which is variously r e p o r t e d to be between 5 a n d 9 p e r cent. 6 Miles and N o r r i s ) s reviewing all the cases o f malignant a n d suspected malignant B r e n n e r tumors on file at the A r m e d Forces Institute o f Pathology, emplmsized a B r e n n e r neoplasm that is intermediate between the benign and fnlly malignant B r e n n e r tumor. T h e B r e n n e r t u m o r has occasionally been associated with other ovarian disease, frequently a mucinous cystadenoma. Epple and Bossert 4 r e p o r t e d a B r e n n e r t u m o r associated with a mucinous cystadenocarcinoma o f the contralateral ovary and a squamous cell carcinoma o f the cervix. Both a B r e n n e r t u m o r in tire wall o f a benign cystic teratonm and a malignant teratonm were r e p o r t e d from the Mayo Clinic. B r e n n e r tumors have also been associated with chronic oophoritis, simple and follicular cysts, germinal inclusion cysts, hilus cell hyperplasia, ovarian adenocarcinoma, adenofibroma, and endometriosis. Gifford and Birch in 1969 r e p o r t e d two cases o f bilateral B r e n n e r tnmors and a d d e d them to their review o f previously r e p o r t e d cases.a,6, ~2,29 T h e y stated that "In contrast to
MEDICAL t h e u n i l a t e r a l t u m o r s , it a p p e a r s t h a t b i l a t e r a l t u m o r s o c c u r at a s l i g h t l y e a r l i e r a g e , a r e associated with lower parity and higher abortion incidence and have a lower rate of malignancy. On the other band, nnilateral and bilateral Brenner tumors are similar in terms of symptomatology, laterality of tumor size and postmenopausally associated endometrial activity. ''6 The treatment of Brenner tumors is unilateral oophorectomy. In postmenopausal w o m e n t h e p r o c e d u r e o f c h o i c e is a h y s t e r ectomy with bilateral salpingo-oophorectomy) 3 I f m a l i g n a n t c h a n g e is e n c o u n t e r e d in t h e surgical specimen, a total abdominal hysterectomy with bilateral salpingo-ooplmrectomy is r e c o m m e n d e d as t h e i n i t i a l t r e a t m e n t in menopausal and postmenopausal women. If a m a l i g n a n t B r e n n e r t u m o r is e n c o u n t e r e d in a y o u n g e r w o m a n , t h e r a p y is m o r e likely to b e i n d i v i d u a l i z e d a n d c o n s e r v a t i v e , s u c h as a unilateral oophorectomy. The value of radiat i o n t h e r a p y as a t h e r a p e u t i c m o d a l i t y f o r r e s i d u a l o r r e c u r r e n t t u m o r is p r e s e n t l y u n determined.
References 1~ Badway, R. E., Jorgenson, O. it., and Cromer, J. K.: Bilateral Brenner tumors--review of literature and report of case. Med. Ann. D. C.,33:106, 1964. 2. Brenner, F.: Das Oophoroma Folliculare. Frankfurt. Z. Path., 1:150, 1907. 3. Christian, C. D., andJanovski, N. A.: Bilateral Brenner tumors. Ant. J. Obstet. Gynecol., 83:105, 1962. -4. Epple, tl. tl., and Bossert, L. J.: Three simultaneous neoplasms of the female genitalia: a Brenner tumor, a pseuclomucinous cystadenocarcinoma of the contralateral ovary, and a squamous cell carcinoma of the cervix. Obstet. Gynecol., 11:661, 1958. 5. Farrar, tt. K.,Jr., and Greene, R. R.: Bilateral Brenner tumors of tile ovary. Am. J. Obstet. Gynecol., 80: 1089, 1960. 6. Gifford, A. B.," and Birch, tt. W.: Bilateral Brenner tumors of the ovary. J. Med. Assoc. Georgia, 58:145, 1969. 7. tlamwi, G.J., et al.: Testosterone synthesis by a Brenner tumor, Part 1. Clinical evidence of masculinization during pregnanc). Part Ii. In ~itro biosynthetic steroid conversion of a Brenner tumor. Am. J. Obstet. Gynecol., 86:!015, 1963. 8. tlertig, A. T., and Gore, I1.: Tumors of the ovary and fallopiaxt tube. In Atlas of T u m o r Pathology, Sec. I X, Fasc. 33. Washington, D. C., Armed Forces Institute of Pathology, 1961, p. 124. 9. llughesdon, P. E.: Thecal and allied reactions in epithelial ovarian turnouts. ]. Obstet. Gynecol. Brit. Emp., 65:702, 1958. 10. tlnll, M. G. R., and Campbell, G. R.: The ntalignant Brenner tumor. Obstet. Gynecol., 42:527,, 1973. 11. Idelson, M. G.: Malignancy in Brenner tumors of the ovary, with comments on histogenesis and possible estrogen production. Obstet. Gynecol. Survey, 18: 246, 1963. 12. Kendall, B., and Bowers, P. A.: Bilateral Brenner tumor of the ovaries. Am. J. Ohstet. Gynecol., 80:439, 1960. 13. Kismer, R. W.: Gynecolo~" Principles attd Practice. Ed. 2. Chicago, Year Book Medical Publishers, Inc., 1972, p. 409. 14. Kraus, F. T.: Gynecologic Pathology. St. Louis, The C. V. Mosby Company, 1967, pp. 324,313.
INTELLIGENCE
15. MacKinlay, C. J.: Brenner tumours of the ovary. J. Obstet. Gynecol. Brit. Emp., 65:58, 1056. 16. MacNaughtou-Jones, t1.: Uterine fibroid with an anomalous ovarian tnmour. Trails. Lond. Obstet. Soc., 40:154,213, 1898. 17. Meyer, R.: Uber verschiedene Erschlintmgsformen der als typus Brenner bekannten Eirstockageschwulst, ihre Zuordnung unter Andere Overialgerschwillste. Arch. G)naekol., 148:5tl, 1932. 18. Miles, 1'. A., Joj, M. C., and Norris, tt. J.: l'roliferative 9 and malignant Brenner tumors of the ovary. Cancer, 30:174, 1972. 19. Ming, S. C., and Goldman, tt4 tlormonal activity of Brenner tumors in postmenopausal women. Am. J. Obstet. Gynecol., 83:666, 1962. 20. Morris,J. M., and Scully, R. E.: Endocrine Pathology of the Ovary. St. Louis, Tile C. V. Mosby Co., 1958. 21. Novak, E. R., and Woodruff, J. D.: Novak's Gynecologic and Obstetric Pathotogy. Ed. 5. lqdladelphia. W. B. Saunders Company, 1962. 22. Novak, E. R., Woodruff, J. D., and Linthicum, J. M.: Evaluation of the unclassified tumors of the. Ovarian T u m o r Registry (1942-1952). Am. J. Ohstet. Gynecol., 87:999. 1963. 23. Schiflmann, J.: Postklimakterische Blutung und Brennerscher ovarial-tumor. Arch GynS.k.. 150:159, 1932. 24. Shay, M. D., and Janovs~, N. A.: Mangnant Brenner tumor associated with endometrial adeuocardinoma. Obstet. Gynecol., 22:246, 1963. 25. Silverberg, S. G.: Brenuer tumor of the ovary. Cancer, 28:588, 1971. 26. Silverberg. S. G., and Willson, M. A.: Uhrastructure of the Brenner tumor. Amer. J. Obstet. Gynecol., 112: 91, 1972. 27. Sternberg, W. I1.: Nonfunctioning Ovarian Neoplasms in the Ovary. Baltimore, Tile Williams & Wilkins Co., 1963, p. 209. 28. Teoh, T. B.: The histogenesis of Brenner tumors of the ovary, J. Path. Bact., 66:441, 1953. 29. Varden, L. C.: Bilateral Brenner tumors of the ovaries. *led. Ann. D. C., 33:70, 196t. 30. Von Numers, C.: A contribution to the case knowledge and histology of the Brenner tumor. Acta Obstet. Gynecol. Scand., 25(Suppl. 2):114, 1945.
MUCOCELE OF THE APPENDIX SECONDARY TO OBSTRUCTION BY ENDOMETRIOSIS MARC R. HAI'KE, M . D . , * AND BRADLEY BIGELOW, M.D.'~
Abstract
A mucocele of the appendix secondaO' to obstruction by endometriosis is reported and the relevant literature reviewed. The theories of the pathogenesis of appeudiceal mucocele are reviewed *Medical Fellow, I ) e l m r t m c n t o1 Lal)or;.ttory Medicine atttl l'athology (Surgical P,tthology), University o f M i n n e s o t a School o f Medicine, Minneapolis, Minnesota. tAssociate l'rofessor o f Pathology, New York University School o f Medicine, New York, New York.
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