Primary malignant mixed M?llerian tumor arising from the mesorectum with a synchronous ovarian cancer: a case report and review of the literature.

0.43 MB·English

by  Huang, Chuang-Chi

Huangetal.JournalofMedicalCaseReports2011,5:15
JOURNAL OF MEDICAL
http://www.jmedicalcasereports.com/content/5/1/15
CASE REPORTS
CASE REPORT Open Access
Primary malignant mixed Müllerian tumor arising
from the mesorectum with a synchronous
ovarian cancer: a case report and review of the
literature
Chuang-Chi Huang1, Cheng-Jen Ma1, Wan-Ting Huang2, Te-Fu Chan3,4,5, Jaw-Yuan Wang1,4,6,7,8*
Abstract
Introduction: Extragenital malignant mixed Müllerian tumor is an extremely rare presentation of malignant mixed
Müllerian tumor, especially when combined with a synchronous ovarian cancer.
Case presentation: We report the clinical course and pathologic findings of a case of mesorectal malignant mixed
Müllerian tumor with synchronous ovarian cancer, in a 50-year-old, gravida 0, para 0, Han Chinese woman with
regular menstruation. This is the sixteenth case in the English literature of extragenital malignant mixed Müllerian
tumor combined with synchronous or metachronous malignancy reported.
Conclusion: Although extragenital malignant mixed Müllerian tumor is very rare and has a poor prognososis, a
longer survival time might be achieved with treatment by cytoreductive surgery, radiotherapy and chemotherapy.
Introduction menstruation. Six months ago, she visited another medi-
Malignant mixed Müllerian tumor (MMMT) is an cal center in Southern Taiwan for abdominal bloating,
uncommon tumor in females and the occurrence of this where bilateral ovarian tumors were diagnosed. At
disease outside the genital tract is extremely rare. In a laparotomy, a left ovarian cystic tumor (35 × 20 × 10
review of the English literature since 1955, only 48 cases cm) and a right ovarian tumor (12 × 8.5 × 6 cm) with
of extragenital MMMT have been reported other than normal uterus and cervix were noted. An additional
the presented case. Sixteen out of these 49 (32.7%) tumor of about 12 × 9 × 8 cm in size was also found in
extragenital MMMTs [1], including this case, were asso- the mesorectum of the rectosigmoid colon. Resection of
ciated with synchronous or metachronous colonic can- the mesorectum and bilateral oophorectomy was per-
cer or gynecologic malignancy and serous carcinoma of formed at the first operation at another medical center.
the peritoneum (Table 1). The MMMT often presents The histopathology report revealed bilateral ovarian can-
in elderly menopausal women and is a highly aggressive cer (endometrioid adenocarcinoma) and malignant
tumor. We report the clinical course and pathologic mixed Müllerian tumor from the mesorectum with
findings of an extragenital MMMT arising from the biphasic differentiation (adenocarcinomatous and spin-
mesorectum in a perimenopausal woman and a review dle cell sarcomatous elements). No heterologous ele-
of the English literature. ment was identified. No further treatment was
performed after the first time of operation. However,
Case presentation she felt progressive abdominal bloating and dysuria
The patient case was a 50-year-old, gravid 0, para 0 recently. She, therefore, visited the department of sur-
(G0P0), unmarried Han Chinese woman with regular gery of our hospital. On physical examination a lower
abdominal mass was palpated. An abdominal computed
*Correspondence:[email protected] tomography scan revealed a large low density mass in
1DepartmentofSurgery,KaohsiungMedicalUniversityHospital,Kaohsiung the pelvic cavity (Figure 1). The maximum size of this
MedicalUniversity,Kaohsiung,Taiwan
lesion was about 15 cm in its long-axis diameter. This
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Table 1Previous reportsofmalignantmixed Müllerian tumor (MMMT) withsynchronous ormetachronous neoplasm
Case Year Author Age Primarysite Tissuetype Associatedtumor Treatment Prognosis
1 1983 Hermannand 72 Abdominal Heterologous Ovarianserouspapillary Operation,CT(Adriamycin Deathatsix
[15] Tessler retroperitoneum carcinoma,metachronous (doxorubicin),cytoxan,DTIC, months
vincristine)
2 1988 Chenand 58 Pelvic Homologous Ovarianserouspapillary Operation,RT Deathat11
[16] Wolk peritoneum carcinoma,metachronous months
3 1989 El-Jabbouret 76 Ascending Heterologous Colonicadenocarcinoma, Operation Deathat14days
[17] al. colon synchronous
peritoneum
4 1991 Gardeand 65 Diaphragmatic Heterologous Ovarianendometrioid Operation,CT(Adriamycin Deathatsix
[18] Jonesetal. peritoneum adenocarcinoma, (doxorubicin),cisplatin, months
metachronous ifosfamide)
5 1991 Solisetal. 54 Pelvic Heterologous Serouscarcinomaof Operation,CT(Adriamycin Unknown
[19] peritoneum peritoneum,synchronous (doxorubicin),cisplatin,
cytoxan)
6[9] 1994 Garamvoelgyi 59 Pelvic Heterologous Endometrial Operation,CT(ifosfamide) Deathat24
etal. peritoneum adenocarcinoma, months
metachronous
7[9] 1994 Garamvoelgyi 64 Pelvic Homologous Fallopiantubecacinoma Operation Deathateight
etal. peritoneum insitu,synchronous months
8[9] 1994 Garamvoelgyi 84 Retrouterine Heterologous Colonicadenocarcinoma, Operation Deathattwo
etal. peritoneum synchronous monthsfromheart
disease
9 1995 Miraetal. 62 Pelvic Heterologous Ovarianendometrioid Operation Survivalfor28
[20] peritoneum adenocarcinoma, months
metachronous
10 1997 Roseetal. 71 Peritoneum Homologous Uterinecervical Operation,CT(cisplatin, Deathatsix
[21] adenocarcinoma, ifosfamide) months
synchronous
11 2001 Shenetal. 33 Pelvic Heterologous Endometrial Operation Deathat12
[22] peritoneum adenocarcinoma, months
metachronous
12 2001 Shenetal. 40 Pelvic Heterologous Fallopiantubecarcinoma, Operation Unknown
[22] metachronous
13 2005 Mikamietal. 53 Mesentery Heterologous Fallopiantubecarcinoma, Operation,CT Survivalforsix
[23] metachronous months
14 2005 Shaco-Levy 85 Omentum Heterologous Colonicadenocarcinoma, Operation Survivalforthree
[24] metachronous months
15 2006 Maetal. 62 Mesentery Homologous Ovarian Operation,CT(ifosfamide, Deathat30
[1] adenocarcinofibroma, carboplatin,etoposide) months
synchronous
16 2008 Currentcase 50 Mesentery Homologous Ovarianadenocarcinoma, Operation Deathat10
synchronous months
CT,computedtomography;DTIC,Dacarbazine;RT,radiotherapy
mass affected the bladder and the rectosigmoid colon. resection of the rectosigmoid colon with an end-to-end
Laboratory tests showed that the serum lactate dehydro- anastomosis was performed. Unfortunately, 10 days
genase level was 271 IU/L. The serum CA 125 level was later, the patient had an anastomotic leakage caused by
elevated up to 154.3 U/mL, while the serum CA19-9 the penetration of the drain tube which was noted when
level was within the normal range. a colonoscopy was performed. Consequently, an ileost-
On suspicion of the recurrence of a tumor, another omy was constructed for fecal diversion as the healing
laparotomy was performed. The pelvic cavity was fully of the leakage site had failed. The pathologic findings
occupied by a huge cystic mass with adjacent organ showed neoplastic cells with areas of local glandular and
involvement. A tumor measuring 12 × 10 × 8 cm arising squamoid differentiation. In addition, bizarre giant
from the mesorectum was identified - the terminal tumor cells in the carcinoma component were also
ileum was also involved. The tumor infiltrated into the noted (Figure 2A and 2B). Patternless oval to spindled
pelvic floor and the retroperitoneum and a palliative neoplastic cells were noted in the sarcoma component
Huangetal.JournalofMedicalCaseReports2011,5:15 Page3of5
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chemotherapy, hospice care was suggested and she was
referred to the previous medical center.
Discussion
MMMT arising from the female genital tract is a rare
disease, comprising less than 1% of all gynecological
malignancies, and MMMT of extragenital origin is even
rarer. MMMT arises from the Müllerian system which
develops to form the fallopian tubes, uterus and the
upper portion of the vagina and often occurs in meno-
pausal women. Since histological evaluation shows both
carcinoma (epithelial) and sarcoma (mesenchymal) com-
ponents, this disorder is also named carcinosarcoma.
MMMT is classified into homologous or heterologous
according to the sarcomatous component. Extragenital
MMMT can occur at any site of peritoneum and is one
type of primary peritoneal carcinomas (PPC) which was
Figure 1 A large low-density mass lesion was noted in the first described by Swerdlow in 1959 [2]. It has the char-
pelviccavityandasignificantmasseffectatrectosigmoidand acteristics of involvement in the peritoneum by carci-
bladderwasalsonoted(arrow).
noma without an obvious primary site [3].
The majority of PPCs present in pathology as serous
papillarycarcinomas,aswellas peritonealmixed epithe-
(Figure 2C). Immunohistochemical studies showed that
lialcarcinomas,whiletheextragenitalMMMTsarerarely
CK7 and CD10 staining were positive but that the CK20
reported.PPCisararecancercloselyrelatedtoepithelial
staining was negative. After one and a half months in
ovarian cancer and develops in cells from the lining of
our department, she recovered uneventfully and was
the pelvis and abdomen (peritoneum). These cells are
transferred to the division of medical oncology for che-
similar to the cells on the surface of the ovaries. Like
motherapy. Chemotherapy, with regimen of bleomycin,
ovariancancer,PPCtendstospreadalongthesurfaceof
etoposide and cisplatin, was arranged but pancytopenia
thepelvisandabdomen.SymptomsofpatientswithPPC
with nosocomial infection was noted after the che-
are similar to those with ovarian cancer, including
motherapy. Due to the poor response to systemic
abdominalpainorbloating,nausea,vomiting,indigestion
and change in bowel habits. Women with PPC are
usuallytreatedsimilarlytothosewithwidespreadovarian
cancer.Thetherapeuticmodalitiesincludecytoreductive
surgery as much as possible, followed by the same che-
motherapy regimen administrated for ovarian cancer.
Looketal.assertedthatoptimalcytoreductioncouldsig-
nificantlyimprovetheprognosisofpatients[4].However,
PPCisofmultifocalorigin, whichisin contrastto ovar-
ian cancer, and usually infiltrates the peritoneal lining
surface. Consequently, cytoreductive surgery is not
alwaysoptimalandthistherapeuticmodalityneedstobe
evaluated in order to determine whether it is an appro-
priatetreatmentforPPC.
Most PPCs are serous papillary adenocarcinomas with
a relatively good prognosis but the primary peritoneal
MMMT, a rare type of PPC, usually has an unfavorable
outcome according to the previous literature [5].
MMMT of extragenital origin was first reported by
Figure 2 (A) These cells with local glandular and squamoid
Ober and Black in 1955 [6] and, until now, only 48
differentiationwerenotedinthecarcinomatouscomponent
(100×).(B)Thebizarretumorgiantcellwasnotedinthe cases have been reported in the English literature. It has
carcinomatouscomponent(arrow;400x).(C)Thepatternlessovalto been reported to have arisen from the peritoneum,
spindledneoplasticcellswasnotedinthesarcomatouscomponent mesentery, omentum, spleen, diaphragm and retroperi-
(200x).
toneum. Among all the reported cases, the majority
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were menopausal women with a median age of 62.8 peritonealperfusionplusadjuvantchemotherapyseemsto
years (range 33-87 years). Sixteen of the 49 patients be an effective treatment for recurrent or metastatic
(32.7%) presented with synchronous or metachronous MMMT.
malignancies including colonic (three cases), ovarian (six A similar case of MMMT of mesenteric origin was
cases including the present case), fallopian tubal (three reported by Ma et al.[1]. The patient died of extensive
cases), endometrial (two), cervical (one) and one syn- metastasis 30 months after the diagnosis of MMMT.
chronous serous carcinoma of the peritoneum. Due to a She received six courses of chemotherapy, including
high incidence of synchronous or metachronous colonic ifosfamide, VP-16 and carboplatin, as well as eight
cancer or gynecologic malignancy originating from the courses of Phyxol (paclitaxel) and cisplatin.
Müllerian duct, clinicians should carefully check the Recently,ithasbeendemonstratedthatthepresenceof
genital tract in detail during the resection of primary BRCA mutations may predispose to primary peritoneal
MMMT. cancersand thisneoplasm couldbe a part ofthe heredi-
Little information about the management of extrageni- tarybreastand ovarycancersyndrome [11].Immunohis-
tal MMMT is available. All suggestions for the treat- tochemicalstudies,itissuggestedthatexpressionofCD10
ment extragenital MMMT are based on individual cases. shouldbeexamined-itmaybeoneofthecharacteristics
Treatments including cytoreductive surgery and che- of MMMT [12,13]. However, the significance of CD10
motherapy have been reported. Surgical management is expressionneedstobeelucidatedbyfurtherstudies.Our
usually mandatory due to the clinical presentation patient’stumoralsohadanexpressionofCD10.Regarding
caused by the mass effect. However, a radical surgical the histological component in MMMT, Ozguroglu et al.
treatment is often obtained with difficulty. It seems that investigated the role of carcinomatous and sarcomatous
chemotherapy is more important than surgical treat- componentsontheresponsetochemotherapyanddisease
ment and the treatment choice of MMMT is similar to outcome.Italsoobservedthatpatientswithapredominat-
that of genital MMMT. ing carcinomatous component had a higher therapeutic
There are several reports regarding platinum-based response rate (87.5%) than those with a predominating
chemotherapy activity against MMMT of the ovary. sarcomatouscomponent(66.6%)[14].
Simon et al. reported a patient with MMMT of the
ovary who had a suboptimal response to single-agent Conclusion
cisplatin chemotherapy but who demonstrated a com- Extragenital MMMT is extremely rare and has a poor
plete response with ifosfamide, mesna, Adriamycin (dox- prognosis due to its aggressive biological behavior. Syn-
orubicin) and dacarbazine [7]. Paclitaxel/carboplatin chronousormetachronousgynecologictumorsoftenexist
(PC) or platinum/ifosfamide (PI) has been used for the and a detailed examination of the genital tract must be
chemotherapy of ovarian MMMT [8]. The median sur- made before and during the operation. Moreover,
vival time of patients receiving PC was 19 months. One improved survival times would probably be obtained if
patient receiving PC as first-line treatment demonstrated accurate diagnoses and aggressive treatment, including
a complete response and was free of disease after 33 cytoreductivesurgeryandchemotherapy,areappliedearly
months. The median survival time of patients managed
with PI was 23 months. Three patients with suboptimal Consent
disease demonstrated complete response after receiving Written informed consent was obtained from the patient
PI. This study showed the potential activity of PC in for publication of this case report and any accompany-
MMMT of the ovaries should be further explored. ing images. A copy of the written consent is available
Theroleofradiotherapyremainscontroversial.Whena for review by the Editor-in-Chief of this journal.
patientpresentswithagrosslyresidualtumor,radiother-
apy may be considered. Garamvoelgyi et al. reported a
Abbreviations
patientwhoreceived postoperativeradiotherapyandsur-
MMMT:malignantmixedMülleriantumor;PC:paclitaxe/carboplatin;PI:
vivedforeightmonths[9].Conversely,otherauthorscon- platinum/ifosfamide;PPC:primaryperitonealcarcinomas.
siderthatextragenital MMMTisone kindofPPCandis
similartoovarianepithelialtumor.Mulleretal.reported Acknowledgements
ThisworkwassupportedbyagrantfromtheKaohsiungMedicalUniversity
sixcasesofmetastasizedMMMTsreceivingcytoreductive Hospital(KMUH98-8I04)andbyanExcellenceforCancerResearchCenter
surgery plus intraperitoneal hyperthermic perfusion and Grant(DOH100-TD-111-002)throughthefundingbyDepartmentofHealth,
ExecutiveYuan.
adjuvanttreatmentofCDDP(cis-diamminedichloroplati-
num), mitomycin and ifosfamide applied via intraaortic Authordetails
catheter [10]. Fourpatientswerefoundwith noevidence 1DepartmentofSurgery,KaohsiungMedicalUniversityHospital,Kaohsiung
MedicalUniversity,Kaohsiung,Taiwan.2DepartmentofPathology,Kaohsiung
ofdiseaseaftertwo,four,14,and19months,respectively.
MedicalUniversityHospital,KaohsiungMedicalUniversity,Kaohsiung,
Thus, complete cytoreduction plus hyperthermic Taiwan.3DepartmentsofObstetricsandGynecology,CollegeofMedicine,
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