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A giant superinfected uterine angioleiomyoma with distant septic metastases: an extremely rare presentation of a benign process and a systematic review of the literature

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Abstract

Purpose

Uterine angioleiomyoma is a rare type of leiomyoma variant and there are few cases reported in the literature. The definitive diagnosis is usually obtained only after the histopathologic examination because there are no specific imaging criteria for this disease. The objective of this article is to review published cases about this clinical condition.

Methods

We report a case of giant angioleiomyoma superinfected by S. agalactiae with the development of latero-cervical distant metastasis in a premenopausal woman. Firstly, the case herein reported was orientated as an endometrial stroma sarcoma in the peri-operative histologic examination by frozen sections. It was treated with laparotomic total hysterectomy, bilateral salpingo-oophorectomy, inframesocolic omentectomy and pelvic and paraaortic lymph node dissection. Postoperative definitive anatomopathological analyses using a proper immunohistochemical panel revealed a case of uterine angioleiomyoma. We also review other case reports published about this clinical condition.

Results

We present the first case reported in the literature, in our knowledge, of a giant angioleiomyoma superinfected by S. agalactiae with the development of distant septic metastases. Immunohistochemistry permitted the definitive diagnosis of angioleiomyoma. Treatments previously reported are hysterectomy or tumor resection and any patient recurred.

Conclusions

The definitive diagnosis is usually obtained after the definitive histopathologic examination since the use of immunohistochemical study has an important role in this regard. Complete surgical removal of the lesion is the treatment of choice, with no recurrent cases reported to date.

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References

  1. Hachisuga T, Hashimoto H, Angioleiomyoma Enjoji M (1984) A clinicopathologic reappraisal of 562 cases. Cancer 54(1):126–130

    Article  CAS  Google Scholar 

  2. Sharma C, Sharma M, Chander B, Soni A, Soni PK (2014) Angioleiomyoma uterus in an adolescent girl: a highly unusual presentation. J Pediatr Adolesc Gynecol 27(3):e69–71

    Article  Google Scholar 

  3. Sahu L, Tempe A, Agrawal A (2012) Angioleiomyoma of uterus. J Obstet Gynaecol 32(7):713–714

    Article  CAS  Google Scholar 

  4. Sikora-Szczęśniak DL (2016) Uterine angioleiomyoma—a rare variant of uterine leiomyoma: review of literature and case reports. Prz Menopauzalny 15(3):165–169

    PubMed  PubMed Central  Google Scholar 

  5. Garg G, Mohanty SK (2014) Uterine angioleiomyoma: a rare variant of uterine leiomyoma. Arch Pathol Lab Med 138(8):1115–1118

    Article  Google Scholar 

  6. Zizi-Sermpetzoglou A, Myoteri D, Arkoumani E, Koulia K, Tsavari A, Alamanou E et al (2015) Angioleiomyoma of the uterus: report of a distinctive benign leiomyoma variant. Eur J Gynaecol Oncol 36(2):210–212

    CAS  PubMed  Google Scholar 

  7. Jameson CF (1990) Angiomyoma of the uterus in a patient with tuberous sclerosis. Histopathology 16(2):202–203

    Article  CAS  Google Scholar 

  8. Konichezky M, Reif R, Bukovsky I (1980) Benign angiomyoma of the uterus with unusual macroscopic appearance. Int J Gynaecol Obstet 18(1):4–6

    Article  CAS  Google Scholar 

  9. Hennig Y, Caselitz J, Stern C, Bartnitzke S, Bullerdiek J (1999) Karyotype evolution in a case of uterine angioleiomyoma. Cancer Genet Cytogenet 108(1):79–80

    Article  CAS  Google Scholar 

  10. Agorastos T, Dinas K, Patsiaoura K (2001) Cystic degenerated angioleiomyoma mimicking ovarian pathology. Acta Obstet Gynecol Scand 80(9):863–865

    Article  CAS  Google Scholar 

  11. Hsieh C-H, Lui C-C, Huang S-C, Ou Y-C, ChangChien C-C, Lan K-C et al (2003) Multiple uterine angioleiomyomas in a woman presenting with severe menorrhagia. Gynecol Oncol 90(2):348–352

    Article  Google Scholar 

  12. Culhaci N, Ozkara E, Yüksel H, Ozsunar Y, Unal E (2006) Spontaneously ruptured uterine angioleiomyoma. Pathol Oncol Res 12(1):50–51

    Article  Google Scholar 

  13. Sakai Y (2007) Epithelioid vascular leiomyoma of the uterus mimicking glomangiomyoma. Arch Gynecol Obstet 275(1):59–61

    Article  Google Scholar 

  14. McCluggage WG, Boyde A (2007) Uterine angioleiomyomas: a report of 3 cases of a distinctive benign leiomyoma variant. Int J Surg Pathol 15(3):262–265

    Article  Google Scholar 

  15. Hakverdi S, Dolapçioğlu K, Güngören A, Yaldiz M, Hakverdi AU (2009) Multiple uterine angioleiomyomas mimicking an ovarian neoplasm: a case report. Eur J Gynaecol Oncol 30(5):592–594

    CAS  PubMed  Google Scholar 

  16. Thomas S, Radhakrishnan L, Abraham L, Matthai A (2012) Uterine Angioleiomyoma with atypia, raised CA-125 levels, and pseudo-meigs syndrome: an alarming presentation. Case Rep Pathol 2012:519473

    PubMed  PubMed Central  Google Scholar 

  17. Handler M, Rezai F, Fless KG, Litinski M, Yodice PC (2012) Uterine angioleiomyoma complicated by consumptive coagulopathy. Gynecol Oncol Case Rep 2(3):89–91

    Article  Google Scholar 

  18. Diwaker P, Pradhan D, Garg G, Bisaria D, Gogoi K, Mohanty SK (2015) Uterine angioleiomyoma: a rare variant of uterine leiomyoma–A case report and literature review. J Cancer Res Ther 11(3):649

    PubMed  Google Scholar 

  19. Gupta M, Suryawanshi M, Kumar R, Peedicayil A (2018) Angioleiomyoma of uterus: a clinicopathologic study of 6 cases. Int J Surg Pathol 26(1):18–23

    Article  Google Scholar 

  20. Koleskas D, Karagiannis G, Beukenholdt RW (2009) A case of a cervical angioleiomyoma presenting with menorrhagia and pelvic pain: a common presentation of a rare tumour. J Obstet Gynaecol 29(2):161–163

    Article  CAS  Google Scholar 

  21. Al-Sannaa GA, Al-Manea M (2011) Cervical angioleiomyoma. J Obstet Gynaecol 31(6):555

    Article  CAS  Google Scholar 

  22. Bouraoui S, El Hadj OEA, Rekik W, Goutallier-Ben Fadhel C, Kébir FZ, Lahmar A et al (2010) First case of angioleiomyoma originating from the ovary of an adult woman. Gynecol Obstet Invest. 70(1):8–10

    Article  Google Scholar 

  23. Lee S-J, Choi YS, Park K-K (2014) Ovarian angioleiomyoma: a case report. Int J Clin Exp Pathol 7(11):8235–8239

    PubMed  PubMed Central  Google Scholar 

  24. Hsu T-L, Changchien C-C, Huang C-C, Lin H (2008) Angioleiomyoma originating from the ovary of an eleven-year-old premenarchal girl. Gynecol Obstet Invest 65(4):262–265

    Article  Google Scholar 

  25. Cobellis L, Pecori E, Rigatti F, Scaffa C, Rotondi M, Messalli EM (2007) A rare case of female pelvic mass: angioleiomyoma of the broad ligament. Eur J Gynaecol Oncol 28(5):418–420

    CAS  PubMed  Google Scholar 

  26. Huang H-C, Chen Y-R, Tsai H-D, Cheng Y-M, Hsiao Y-H (2017) Angiomyofibroblastoma of the broad ligament: a case report. Int J Gynecol Pathol 36(5):471–475

    Article  CAS  Google Scholar 

  27. Chen X, Zhang X, Zhang S, Lü B (2010) Angioleiomyomas in the bilateral broad ligaments. Int J Gynecol Pathol 29(1):39–43

    Article  CAS  Google Scholar 

  28. Agarwal S, Gupta SK, Tejwani N (2009) Angioleiomyoma of broad ligament. J Gynecol Endosc Surg 1(2):116–117

    Article  CAS  Google Scholar 

  29. Güven D, Erdogan O, Koçak I, Ustün C (2009) Giant angiomyoma of the broad ligament. J Obstet Gynaecol 29(3):261–263

    Article  Google Scholar 

  30. Pierro A, Rotondi F, Cilla S, De Ninno M, Mattoni M, Berardi S et al (2018) Giant angioleiomyoma of uterus: a case report with focus on CT imaging. Radiol Case Rep 13(2):371–375

    Article  Google Scholar 

  31. Kim H, Lee J-J, Choi Y, Lee M, Hwang H-J, Chung Y-J et al (2018) Successfully removed uterine angioleiomyoma by robot-assisted laparoscopic myomectomy. Obstet Gynecol Sci 61(3):425–429

    Article  Google Scholar 

  32. Hong J-A, Heo G-E, Kwak JJ, Chung S-H (2017) A case report of angioleiomyoma of uterus. Obstet Gynecol Sci 60(5):494–497

    Article  Google Scholar 

  33. Singh S, Naik M, Bag ND, Patra S (2017) Angioleiomyoma of uterus masquerading as malignant ovarian tumor. J Midlife Health 8(3):145–147

    PubMed  PubMed Central  Google Scholar 

  34. Grigoriadis C, Androutsopoulos G, Zygouris D, Arnogiannaki N, Terzakis E (2014) Uterine angioleiomyoma causing severe abnormal uterine bleeding. Clin Exp Obstet Gynecol 41(1):102–104

    CAS  PubMed  Google Scholar 

  35. Lazarov N, Lazarov L, Lazarov S. [Angioleiomyoma utery in a female patient with damaged health condition. Diagnostic and terapeutic difficulties]. Akush Ginekol (Sofiia). 2011;50(4):54–8.

  36. Jaszcz W, Pieczonka L (1975) Case of uterine angiomyoma. Patol Pol 26(3):447–451

    CAS  PubMed  Google Scholar 

  37. Jin CH, Yi KW, Kim Y-S, Shin J-H, Kim T, Hur J-Y et al (2013) Uterine angioleiomyoma: unusual appearance at laparoscopy. J Minim Invasive Gynecol 20(2):149–150

    Article  Google Scholar 

  38. Walocha JA, Litwin JA, Miodoński AJ (2003) Vascular system of intramural leiomyomata revealed by corrosion casting and scanning electron microscopy. Hum Reprod 18(5):1088–1093

    Article  Google Scholar 

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Author information

Author notes

  1. José-Luis Sánchez-Iglesias and Sira Capote contributed equally.

Authors and Affiliations

  1. Unit of Gynecologic Oncology, Department of Obstetrics and Gynecology, Hospital Universitari Vall D’Hebron, Autonomous University of Barcelona, Barcelona, Spain

    José-Luis Sánchez-Iglesias, Melchor Carbonell-Socias, Silvia Cabrera, Lidia Illán-Hernández, Maria-Assumpció Pérez-Benavente, Sonia Monreal-Clua & Antonio Gil-Moreno

  2. Gynecology Unit, Department of Obstetrics and Gynecology, Fundació Hospital de L’Esperit Sant, Avinguda Josep Pons I Rabadà S/N, Santa Coloma de Gramenet, E-08923, Barcelona, Spain

    Sira Capote

  3. Benign Gynecology Unit, Department of Obstetrics and Gynecology, Hospital Universitari Vall D’Hebron, Autonomous University of Barcelona, Barcelona, Spain

    Montserrat Cubo-Abert

Authors
  1. José-Luis Sánchez-Iglesias
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  2. Sira Capote
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  3. Montserrat Cubo-Abert
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  4. Melchor Carbonell-Socias
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  6. Lidia Illán-Hernández
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  7. Maria-Assumpció Pérez-Benavente
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  8. Sonia Monreal-Clua
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  9. Antonio Gil-Moreno
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Contributions

JLS-I: Project development, Data Collection, Manuscript writing. SC: Project development, Data Collection, Manuscript writing, Manuscript editing. MC-A: Manuscript editing. MC-S: Data Collection. S Cabrera: Data Collection. LI-H: Data Collection, Manuscript writing. MAP-B: Manuscript editing. SM-C: Data collection. AG-M: Final Manuscript editing.

Corresponding author

Correspondence to Sira Capote.

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Sánchez-Iglesias, JL., Capote, S., Cubo-Abert, M. et al. A giant superinfected uterine angioleiomyoma with distant septic metastases: an extremely rare presentation of a benign process and a systematic review of the literature. Arch Gynecol Obstet 300, 841–847 (2019). https://doi.org/10.1007/s00404-019-05267-w

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