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Uterus didelphys unicollis with adenocarcinoma in one horn and atypical endometrial hyperplasia in the other: Case report
222
Communications
in brief
multiple malformations are not found, thus maximizing cost effectiveness. Chromosomal abnormalities are generally and appropriately considered in any infant with multisystem anomalies. In this case in which, initially, only a single malformation was thought to exist, tissues for chromosomal studies might not have been obtained if there had not been a standard protocol for the evaluation of stillbirths. Since the overall incidence of chromosomal anomalies in stillborn infants is .5%. to 75?.’ the yield of this protocol is sufficiently high to warrant routine evaluation. It allows for both the diagnosis of chromosomal abnormalities and the possibility of preventing recurrence by subsequent prenatal chromosomal testing.
major illnesses. except for hypertension thar had hwrl I I twe(I with diuretics. Examination disclosed that the paticnl \\;t~ obese and hypertensive but in no acute distress. ~1 he IIICIIII was small and irregular. The cel\ix \\a> clean. I’II~I <’ \logic findings showed a ver-y good estrogenic cflccr. She wva\ taughr liegel’s exercises. and a Reisw pws;w~ was itlw ted lot \UIIport. Papanicolaou smeat-s ~\erc repo~.ted trl be nrgaI~\c. ‘I’hc pessary gave excellent qupport ;uld \\aa tlrar~rrtl ,~nti i c’positioned at intervals.
REFERENCES
1.
Milunsky. A.: The Prenatal
Diagnosis of Hereditary Disorders. Springfield. Illinois. 1973, Charles C Thomas, Publisher. 2. Machin, G. A., and Crolla. J. A.: Chromosome constitution of 500 infants dying during the perinatal period, Humangenetik 23: 183, 1974.
Uterus didelphys unicollis with adenocarcinoma in one horn and atypical endometrial hyperplasia in the other: Case report EDUARD
EICHNER,
KA-I‘HLEEN
A.
M.D. SIMAK.
M.D
DiGsion of Obstetnrs and Qnrcology. and DizG~n Pathology, Mount Sinai Hospital, Clezleland, Ohio
oj
THE H I STORY of the patient who is the subject of this report exemplifies the difficulties involved in making the correct diagnosis. Braun’ reviewed the English literature and reported eight patients in whom the dela) in diagnosis ranged from 3 months to 3 years, since in no patient were both cavities curetted at the original procedure. Other reports* also emphasize this problem.
M. Y., an unmarried, nulligravid, white woman, 57 years old, was first seen in May, 1978, complaining of urinary frequency with incontinence. Menarche had occurred at age 11. The menstrual cycles had always been regular but were associated with severe cramps and prolonged bleeding. There had been no bleeding since an abrupt and spontaneous menopause 8 years earlier. Prior Papanicolaou smears had all been negative. The patient denied having had any operations OI Reprint requests: Eduard Eichner, M.D.. Severance cal Arts Bldg.. 5 Severance Circle, Cleveland Heights, 44118. *Full bibliography available on request. 0002.9378/H1/020222+04$OO.4O/O
0
1981
The
C. V. Mosby
MediOhio
CO.
The patient was hospitali/ 1 week later for IXI~IIF *;[?I v(‘tom). Pwoperative e\aluatiotl disclosed that the I)a’irili wah obche and hvpertensivr and 11,1tl J srvcwl> h\pr)pl,lhll( IvI’I kidnev. Ttle r-emainder of rhc lludings KC’IC 1, ithln now~l limits, rxcept for an abnormal clcctr-c,c;irtliograrll. 1 IIC I)rttiwIt withstood the operative procedul-c \ er\ well a11(1 con\ .ilesc ed \bithout difficult\. The correct diagnosis was made oni\ atic]the parhologis~ (K. A. S.I r\;lminetl the cpecinlrn: I-he findings were utci-us didelptiw urli(~)llis witI1 ill\.r>iw, undometrial tarcinoma of’ the left IloIn (Fig. L’, :{ I ,ttld ,tt\plc‘il ddenom.ttou\ hvpcrplasia of the entlonietl-ium 1x1 rhc, right horn (Fig. 2. B). The two funrli t eprc.sentrd I\ h,tc h,~d IXXII called tibroids. Tubes. waries. and vaginal cuff \\C‘IV fl w rlt’ malignant infiltration. Becawr of Ihc cxtenI ot ~\OIIITII-I.I~ invasion, the patient received IHO applications art wlpc)stat radiation bv Nolan applicator-s, totaling 6.906 ratls IO the \‘LKillal apex. (:onvalescence has tonr~nucd wtlafac IOI~I~V. :md there are no ur-inar, problem\. 1Uicholscm
anti
associates”
\ww
tlw
fli-st
10 cl;tssit!
and subdivide the malignant malformed tltc’ri into anatomic divisions. Becatlse of ciuplication and incornpleteness in some reports, thrre is difficult! itr giving [he exact number of‘ cases reported, bllt exceeds 3.5. In reviewing the a\-ailable
it probal~l~
rrport~;.:~ KC were unable to identify any patient with Amultaneous primary tumors in both horns. aithough thih ha\ been reported in cervical malignancy. Tsibris and aboc iates,’ studied estrogen and progesterone rcc.
Communications
Fig. 1. Prehysterectomy specimens. >I, Endometrial and fragments of endometrial epithelium without ing moderately well-differentiated adenocarcinoma
biopsy specimen (79-S-5893) showing red cells atypia. B, Curettage specimen (79-S-7585) showof the endometrium, in part papillary.
in brief
223
224
Communications
in brief
Fig. 2. Hysterectomy specimens. .4, Right horn (79-S-7737). Adenocarcinoma in papillary and cribriform patrertl. invading the myometrium. R, Left hot-n (79-S-7737). Endometrittm with irregular prolif’eration of the glands, papillary infolding, and a suggestion of back-to-back patter-n. Nuclei at-r \a)-iabl) placed. and some are large and hyperchromatic. A rare mitosis is seen. These changes arc Li~calla clisrribnted in an otherwise menopausal endometrium,
Communications
reports* recorded adenocarcinoma ranging from well dit’f’erentiated to completely anaplastic. Other malignancieh recorded were chorixarcinoma and carcinosal‘coma. We \Iich to thank Ms. L. Landau and the hospital librarians for their exhaustive search of the literature, and Mr. R. Newhouse and his staff for the microphotographs. “Full
bibliography
available
on request
in brief
225
REFERENCES
I. Braun, R. D.: Uterus didelphys and endometrial carcinoma. A case report, Obstet. Gynecol. 35:93, 11170. 2. Nicholson, R., Inza. R., and DiPaola, G.: Asociacion de malformation y carcinoma uterino-Report. Prcnsa Med. Argent. 48:2234. 1961. (In Spanish.) 3. Tsibris, J. C. 14.. Fort, F. I... Cantor. B.. Gelman, S. R.. Rigall. F. C:.. and Spellacy, W. N.: F.ndometrial estrogen and progesterone receptors in a uterus didelphts. ,J. Reprod. Med. 24:182. 1980.
Communications
in brief
multiple malformations are not found, thus maximizing cost effectiveness. Chromosomal abnormalities are generally and appropriately considered in any infant with multisystem anomalies. In this case in which, initially, only a single malformation was thought to exist, tissues for chromosomal studies might not have been obtained if there had not been a standard protocol for the evaluation of stillbirths. Since the overall incidence of chromosomal anomalies in stillborn infants is .5%. to 75?.’ the yield of this protocol is sufficiently high to warrant routine evaluation. It allows for both the diagnosis of chromosomal abnormalities and the possibility of preventing recurrence by subsequent prenatal chromosomal testing.
major illnesses. except for hypertension thar had hwrl I I twe(I with diuretics. Examination disclosed that the paticnl \\;t~ obese and hypertensive but in no acute distress. ~1 he IIICIIII was small and irregular. The cel\ix \\a> clean. I’II~I <’ \
REFERENCES
1.
Milunsky. A.: The Prenatal
Diagnosis of Hereditary Disorders. Springfield. Illinois. 1973, Charles C Thomas, Publisher. 2. Machin, G. A., and Crolla. J. A.: Chromosome constitution of 500 infants dying during the perinatal period, Humangenetik 23: 183, 1974.
Uterus didelphys unicollis with adenocarcinoma in one horn and atypical endometrial hyperplasia in the other: Case report EDUARD
EICHNER,
KA-I‘HLEEN
A.
M.D. SIMAK.
M.D
DiGsion of Obstetnrs and Qnrcology. and DizG~n Pathology, Mount Sinai Hospital, Clezleland, Ohio
oj
THE H I STORY of the patient who is the subject of this report exemplifies the difficulties involved in making the correct diagnosis. Braun’ reviewed the English literature and reported eight patients in whom the dela) in diagnosis ranged from 3 months to 3 years, since in no patient were both cavities curetted at the original procedure. Other reports* also emphasize this problem.
M. Y., an unmarried, nulligravid, white woman, 57 years old, was first seen in May, 1978, complaining of urinary frequency with incontinence. Menarche had occurred at age 11. The menstrual cycles had always been regular but were associated with severe cramps and prolonged bleeding. There had been no bleeding since an abrupt and spontaneous menopause 8 years earlier. Prior Papanicolaou smears had all been negative. The patient denied having had any operations OI Reprint requests: Eduard Eichner, M.D.. Severance cal Arts Bldg.. 5 Severance Circle, Cleveland Heights, 44118. *Full bibliography available on request. 0002.9378/H1/020222+04$OO.4O/O
0
1981
The
C. V. Mosby
MediOhio
CO.
The patient was hospitali/ 1 week later for IXI~IIF *;[?I v(‘tom). Pwoperative e\aluatiotl disclosed that the I)a’irili wah obche and hvpertensivr and 11,1tl J srvcwl> h\pr)pl,lhll( IvI’I kidnev. Ttle r-emainder of rhc lludings KC’IC 1, ithln now~l limits, rxcept for an abnormal clcctr-c,c;irtliograrll. 1 IIC I)rttiwIt withstood the operative procedul-c \ er\ well a11(1 con\ .ilesc ed \bithout difficult\. The correct diagnosis was made oni\ atic]the parhologis~ (K. A. S.I r\;lminetl the cpecinlrn: I-he findings were utci-us didelptiw urli(~)llis witI1 ill\.r>iw, undometrial tarcinoma of’ the left IloIn (Fig. L’, :{ I ,ttld ,tt\plc‘il ddenom.ttou\ hvpcrplasia of the entlonietl-ium 1x1 rhc, right horn (Fig. 2. B). The two funrli t eprc.sentrd I\ h,tc h,~d IXXII called tibroids. Tubes. waries. and vaginal cuff \\C‘IV fl w rlt’ malignant infiltration. Becawr of Ihc cxtenI ot ~\OIIITII-I.I~ invasion, the patient received IHO applications art wlpc)stat radiation bv Nolan applicator-s, totaling 6.906 ratls IO the \‘LKillal apex. (:onvalescence has tonr~nucd wtlafac IOI~I~V. :md there are no ur-inar, problem\. 1Uicholscm
anti
associates”
\ww
tlw
fli-st
10 cl;tssit!
and subdivide the malignant malformed tltc’ri into anatomic divisions. Becatlse of ciuplication and incornpleteness in some reports, thrre is difficult! itr giving [he exact number of‘ cases reported, bllt exceeds 3.5. In reviewing the a\-ailable
it probal~l~
rrport~;.:~ KC were unable to identify any patient with Amultaneous primary tumors in both horns. aithough thih ha\ been reported in cervical malignancy. Tsibris and aboc iates,’ studied estrogen and progesterone rcc.
Communications
Fig. 1. Prehysterectomy specimens. >I, Endometrial and fragments of endometrial epithelium without ing moderately well-differentiated adenocarcinoma
biopsy specimen (79-S-5893) showing red cells atypia. B, Curettage specimen (79-S-7585) showof the endometrium, in part papillary.
in brief
223
224
Communications
in brief
Fig. 2. Hysterectomy specimens. .4, Right horn (79-S-7737). Adenocarcinoma in papillary and cribriform patrertl. invading the myometrium. R, Left hot-n (79-S-7737). Endometrittm with irregular prolif’eration of the glands, papillary infolding, and a suggestion of back-to-back patter-n. Nuclei at-r \a)-iabl) placed. and some are large and hyperchromatic. A rare mitosis is seen. These changes arc Li~calla clisrribnted in an otherwise menopausal endometrium,
Communications
reports* recorded adenocarcinoma ranging from well dit’f’erentiated to completely anaplastic. Other malignancieh recorded were chorixarcinoma and carcinosal‘coma. We \Iich to thank Ms. L. Landau and the hospital librarians for their exhaustive search of the literature, and Mr. R. Newhouse and his staff for the microphotographs. “Full
bibliography
available
on request
in brief
225
REFERENCES
I. Braun, R. D.: Uterus didelphys and endometrial carcinoma. A case report, Obstet. Gynecol. 35:93, 11170. 2. Nicholson, R., Inza. R., and DiPaola, G.: Asociacion de malformation y carcinoma uterino-Report. Prcnsa Med. Argent. 48:2234. 1961. (In Spanish.) 3. Tsibris, J. C. 14.. Fort, F. I... Cantor. B.. Gelman, S. R.. Rigall. F. C:.. and Spellacy, W. N.: F.ndometrial estrogen and progesterone receptors in a uterus didelphts. ,J. Reprod. Med. 24:182. 1980.