Download PDF
Am J Perinatol 2009; 26(7): 507-518
DOI: 10.1055/s-0029-1215428
© Thieme Medical Publishers

Pregnancy and Genital Sarcoma: A Systematic Review of the Literature

Koji Matsuo1 , Michele L. Eno2 , Dwight D. Im1 , 3 , Neil B. Rosenshein1 , 3
  • 1Obstetrics, Gynecology, and Reproductive Sciences, Baltimore, Maryland
  • 2University of Maryland School of Medicine, Baltimore, Maryland
  • 3The Gynecologic Oncology Center, Mercy Medical Center, Baltimore, Maryland
Further Information

Publication History

Publication Date:
13 March 2009 (online)

    Permissions and Reprints

ABSTRACT

We conducted a literature review to determine the clinical characteristics of genital sarcoma during pregnancy. The systematic literature search was conducted using the search engines PubMed and MEDLINE with keywords “sarcoma” and “pregnancy” and was limited to female genital organs such as ovary, uterus, cervix, vagina, vulva, and retroperitoneal sarcoma. Kaposi's sarcoma, metastatic sarcoma, history of sarcoma, bone sarcoma located in pelvis, and fetal sarcoma were excluded in this study. There were 40 cases of genital sarcoma during pregnancy between 1955 and 2007. The majority of the cases were uterine sarcoma (37.5%), followed by retroperitoneal sarcoma (27.5%), vulvar sarcoma (22.5%), and vaginal sarcoma (12.5%). Mean age of the patient was 27.8 ± 7.0. The distribution in the onset of symptoms had two peaks: first trimester (27.5%) and third trimester (50.0%). Growing mass (42.5%), abdominal pain (30.0%), and vaginal bleeding (22.5%) were the three most common symptoms. Incidental diagnosis was made in 22.5% and included during cesarean section (12.5%) and routine pelvic exam (7.5%). The cases initially not suspicious for malignancy were 42.5%. Thirty-three (82.5%) cases had live-born infants with term delivery in 55.2%. Mean birth weight was 2843 ± 791 g, and male infants were more common (66.7%). Intrauterine growth retardation was seen in 12.5% of cases. Preterm labor was a common complication. Median survival period was 2.5 years (95% confidence, 1.9 to 3.1). The 2-, 3-, and 5-year cumulative survival rates were 60%, 38%, and 17%, respectively. Genital sarcomas in pregnancy are rare. There is a delay in diagnosis due to low index of suspicion. A majority had live births, and the 5-year survival is similar to that of advanced-stage sarcoma in nonpregnant women.

KEYWORDS

Uterine sarcoma - vulvar sarcoma - vaginal sarcoma - retroperitoneal sarcoma - pregnancy - review

REFERENCES

  • 1 Platz C E, Benda J A. Female genital tract cancer.  Cancer. 1995;  75 270-294
    CrossrefPubMedGoogle Scholar
  • 2 Gadducci A, Cosio S, Romanini A, Genazzani A R. The management of patients with uterine sarcoma: a debated clinical challenge.  Crit Rev Oncol Hematol. 2008;  65 129-142
    CrossrefPubMedGoogle Scholar
  • 3 Major F J, Blessing J A, Silverberg S G et al.. Prognostic factors in early-stage uterine sarcoma. A Gynecologic Oncology Group study.  Cancer. 1993;  71 1702-1709
    CrossrefPubMedGoogle Scholar
  • 4 Marchese M J, Liskow A S, Crum C P, McCaffrey R M, Frick II H C. Uterine sarcomas: a clinicopathologic study, 1965–1981.  Gynecol Oncol. 1984;  18 299-312
    CrossrefPubMedGoogle Scholar
  • 5 Giuntoli II R L, Metzinger D S, DiMarco C S et al.. Retrospective review of 208 patients with leiomyosarcoma of the uterus: prognostic indicators, surgical management, and adjuvant therapy.  Gynecol Oncol. 2003;  89 460-469
    CrossrefPubMedGoogle Scholar
  • 6 Parker W H, Fu Y S, Berek J S. Uterine sarcoma in patients operated on for presumed leiomyoma and rapidly growing leiomyoma.  Obstet Gynecol. 1994;  83 414-418
    PubMedGoogle Scholar
  • 7 Buttram Jr V C, Reiter R C. Uterine leiomyomata: etiology, symptomatology, and management.  Fertil Steril. 1981;  36 433-445
    PubMedGoogle Scholar
  • 8 Dover R W, Ferrier A J, Torode H W. Sarcomas and the conservative management of uterine fibroids: a cause for concern?.  Aust N Z J Obstet Gynaecol. 2000;  40 308-312
    CrossrefPubMedGoogle Scholar
  • 9 Antonelli N M, Dotters D J, Katz V L, Kuller J A. Cancer in pregnancy: a review of the literature. Part I.  Obstet Gynecol Surv. 1996;  51 125-134
    CrossrefPubMedGoogle Scholar
  • 10 Aharoni A, Reiter A, Golan D, Paltiely Y, Sharf M. Patterns of growth of uterine leiomyomas during pregnancy. A prospective longitudinal study.  Br J Obstet Gynaecol. 1988;  95 510-513
    CrossrefPubMedGoogle Scholar
  • 11 Leunen K, Amant F, Debiec-Rychter M et al.. Endometrial stromal sarcoma presenting as postpartum haemorrhage: report of a case with a sole t(10;17)(q22;p13) translocation.  Gynecol Oncol. 2003;  91 265-271
    CrossrefPubMedGoogle Scholar
  • 12 Izban K F, Gooneratne S. Images in clinical medicine. Mullerian adenosarcoma and early pregnancy.  N Engl J Med. 2000;  343 1167
    CrossrefPubMedGoogle Scholar
  • 13 Bekkers R L, Massuger L F, Berg P P, Haelst U G, Bulten J. Uterine malignant leiomyoblastoma (epithelioid leiomyosarcoma) during pregnancy.  Gynecol Oncol. 1999;  72 433-436
    CrossrefPubMedGoogle Scholar
  • 14 Katsanis W A, O'Connor D M, Gibb R K, Bendon R W. Endometrial stromal sarcoma involving the placenta.  Ann Diagn Pathol. 1998;  2 301-305
    PubMedGoogle Scholar
  • 15 Lau T K, Wong W S. Uterine leiomyosarcoma associated with pregnancy: report of two cases.  Gynecol Oncol. 1994;  53 245-247
    CrossrefPubMedGoogle Scholar
  • 16 Laffargue F, Giacalone P L, Charpin C, Lachard A. Uterine angiomyolipoma associated with pregnancy.  Gynecol Oncol. 1993;  50 357-360
    CrossrefPubMedGoogle Scholar
  • 17 Younis J S, Okon E, Anteby S O. Uterine leiomyosarcoma in pregnancy.  Arch Gynecol Obstet. 1990;  247 155-160
    CrossrefPubMedGoogle Scholar
  • 18 Kyodo Y, Inatomi K, Abe T. Sarcoma associated with pregnancy.  Am J Obstet Gynecol. 1989;  161 94-96
    PubMedGoogle Scholar
  • 19 Visser A A, Brummer W E, Bruin B D. Embryonal rhabdomyosarcoma. A case report.  S Afr Med J. 1978;  54 70-71
    PubMedGoogle Scholar
  • 20 Nye E B. Endometrial sarcoma causing obstructed labour.  J Obstet Gynaecol Br Commonw. 1973;  80 999-1001
    PubMedGoogle Scholar
  • 21 Kececioglu Y. Sarcoma botryoides and pregnancy.  Obstet Gynecol. 1972;  39 70-72
    PubMedGoogle Scholar
  • 22 Wolfe S A, Seckinger D. Sarcoma of the uterus associated with pregnancy.  Am J Obstet Gynecol. 1969;  104 1215-1216
    PubMedGoogle Scholar
  • 23 Herting A T, Gore H. Atlas of Tumor Pathology. Section 9, Fascile 33, Part 2. Washington, DC; Armed Forces Institute of Pathology 1960: 235
    Google Scholar
  • 24 Behzatoğlu K, Bahadir B, Inhan G, Altuğ M. Primary vaginal leiomyosarcoma in pregnancy.  Gynecol Oncol. 2003;  91 627-629
    CrossrefPubMedGoogle Scholar
  • 25 Singhal S, Sharma S, De S et al.. Adult embryonal rhabdomyosarcoma of the vagina complicating pregnancy: a case report and review of the literature.  Asia Oceania J Obstet Gynaecol. 1990;  16 301-306
    PubMedGoogle Scholar
  • 26 Mhaskar R, Takkar D, Bijlani L, Mala G. Rath. Embryonal rhabdomyosarcoma of the vagina associated with pregnancy; a case report.  Eur J Obstet Gynecol Reprod Biol. 1989;  30 95-98
    CrossrefPubMedGoogle Scholar
  • 27 Roddick Jr J W, Honig J. Sarcoma botryoides of the vagina coincident with pregnancy.  Am J Obstet Gynecol. 1965;  92 268-270
    PubMedGoogle Scholar
  • 28 Schwartz P, Klein E I, Fields C. Sarcoma botryoides in pregnancy.  Am J Obstet Gynecol. 1963;  85 123-124
    PubMedGoogle Scholar
  • 29 Di Gilio A R, Cormio G, Resta L et al.. Rapid growth of myxoid leiomyosarcoma of the vulva during pregnancy: a case report.  Int J Gynecol Cancer. 2004;  14 172-175
    CrossrefPubMedGoogle Scholar
  • 30 Moore R G, Steinhoff M M, Granai C O, DeMars L R. Vulvar epithelioid sarcoma in pregnancy.  Gynecol Oncol. 2002;  85 218-222
    CrossrefPubMedGoogle Scholar
  • 31 Fried-Oginski W, Lovecchio J L, Farahani G, Smilari T. Malignant myxoid sarcoma of the Bartholin gland in pregnancy.  Am J Obstet Gynecol. 1995;  173 1633-1635
    CrossrefPubMedGoogle Scholar
  • 32 Bakri Y N, Akhtar M, el-Senoussi M, Wierzbicki R. Vulvar sarcoma: a report of four cases.  Gynecol Oncol. 1992;  46 384-390
    CrossrefPubMedGoogle Scholar
  • 33 Kuller J A, Zucker P K, Peng T C. Vulvar leiomyosarcoma in pregnancy.  Am J Obstet Gynecol. 1990;  162 164-166
    PubMedGoogle Scholar
  • 34 Jafari K, Lash A F, Webster A. Pregnancy and sarcoma.  Acta Obstet Gynecol Scand. 1978;  57 265-271
    PubMedGoogle Scholar
  • 35 Priore R M, Patton G D, Conti E A. Pregnancy complicated by nonpigmented sarcoma of the vulva. Report of a case and review of the literature.  Obstet Gynecol. 1966;  27 420-427
    PubMedGoogle Scholar
  • 36 Lysyj A, Bergquist J R. Pregnancy complicated by sarcoma.  Obstet Gynecol. 1963;  21 506-509
    PubMedGoogle Scholar
  • 37 Ulker V, Gungorduk K, Numanoglu C et al.. Complete surgical resection of retroperitoneal leiomyosarcoma in pregnancy: a case report.  Arch Gynecol Obstet. 2008;  277 353-356
    CrossrefPubMedGoogle Scholar
  • 38 Rousková L, Melichar B, Nikolov D H et al.. Fulminant course of metastatic liposarcoma after delivery—case report.  Eur J Gynaecol Oncol. 2007;  28 67-68
    PubMedGoogle Scholar
  • 39 Tebes S, Cardosi R, Hoffman M. Liposarcoma complicating pregnancy.  Gynecol Oncol. 2001;  83 610-612
    CrossrefPubMedGoogle Scholar
  • 40 De Jaco P, Giorgio M, Zantedeschi B, Mazzoleni G, Marabini A. A case of retroperitoneal liposarcoma in pregnancy.  Acta Obstet Gynecol Scand. 1993;  72 122-124
    CrossrefPubMedGoogle Scholar
  • 41 Allan S M. Primary peritoneal sarcoma: three case reports and review of the literature.  Br J Clin Pract. 1990;  44 753
    PubMedGoogle Scholar
  • 42 Kerner H, Lichtig C, Beck D. Extrauterine Müllerian adenosarcoma of the peritoneal mesothelium: a clinicopathologic and electron microscopic study.  Obstet Gynecol. 1989;  73 510-513
    PubMedGoogle Scholar
  • 43 Shimm D S, McDonough J F. Leiomyosarcoma of the broad ligament—report of a case.  Gynecol Oncol. 1987;  26 123-126
    CrossrefPubMedGoogle Scholar
  • 44 Foruhan B. Retroperitoneal sarcomas obstructing delivery: two case reports.  Br J Obstet Gynaecol. 1979;  86 747-748
    PubMedGoogle Scholar
  • 45 Goodman M. Reticulum cell sarcoma in pregnancy.  J Obstet Gynaecol Br Emp. 1958;  65 641-643
    PubMedGoogle Scholar
  • 46 Palen G M. Retroperitoneal lymphosarcoma complicating pregnancy; report of a case.  J Int Coll Surg. 1950;  13 376-382
    PubMedGoogle Scholar
  • 47 Moran B J, Yano H, Al Zahir N, Farquharson M. Conflicting priorities in surgical intervention for cancer in pregnancy.  Lancet Oncol. 2007;  8 536-544
    CrossrefPubMedGoogle Scholar
  • 48 Krausz C, Quintana-Murci L, Forti G. Y chromosome polymorphisms in medicine.  Ann Med. 2004;  36 573-583
    CrossrefPubMedGoogle Scholar
  • 49 Kasami M, Yoshida M, Isogaki J et al.. Bilateral breast tumors, malignant phyllodes tumor and invasive lobular carcinoma in a 46,XX/46,XY mosaic female with family history of breast cancer.  Pathol Int. 1997;  47 147-154
    CrossrefPubMedGoogle Scholar
  • 50 Pyakurel P, Montag U, Castaños-Vélez E et al.. CGH of microdissected Kaposi's sarcoma lesions reveals recurrent loss of chromosome Y in early and additional chromosomal changes in late tumour stages.  AIDS. 2006;  20 1805-1812
    CrossrefPubMedGoogle Scholar
  • 51 Mbulaiteye S M, Sternberg L R, Nsubuga M M et al.. Absence of Y-chromosome sequences in tumors from African women with AIDS-related Kaposi sarcoma.  Cancer Lett. 2007;  248 229-233
    CrossrefPubMedGoogle Scholar
  • 52 Schuborg C, Mertens F, Rydholm A et al.. Cytogenetic analysis of four angiosarcomas from deep and superficial soft tissue.  Cancer Genet Cytogenet. 1998;  100 52-56
    CrossrefPubMedGoogle Scholar
  • 53 Gladstone B, Parikh P M, Balsara B et al.. Rhabdomyosarcoma. A cytogenetically interesting case report.  Cancer Genet Cytogenet. 1993;  66 43-46
    CrossrefPubMedGoogle Scholar
  • 54 Mandahl N, Jin Y S, Heim S et al.. Trisomy 5 and loss of the Y chromosome as the sole cytogenetic anomalies in a cavernous hemangioma/angiosarcoma.  Genes Chromosomes Cancer. 1990;  1 315-316
    CrossrefPubMedGoogle Scholar

Koji MatsuoM.D. 

Obstetrics, Gynecology, and Reproductive Sciences, University of Maryland School of Medicine, University of Maryland Medical Center

22S Greene Street, P.O. Box 290, Baltimore, MD 21201

Email: koji.matsuo@gmail.com

      >