Uterine angioleiomyoma: clinicopathological evaluation of 5 cases

Year 2020, Volume: 17 Issue: 2, 360 - 363, 25.06.2020

Nilgün Söğütçü , Süleyman Cemil Oğlak

https://doi.org/10.38136/jgon.693599

Abstract

Aim: Uterine angioleiomyoma (ALM) is a very rare benign perivascular neoplasm. In this study, clinicopathologic and immunohistochemical features and treatment modalities of the cases were evaluated with a comprehensive literature review.
Materials and Methods: 5 cases of uterine ALM admitted to the pathology department of our hospital were found in the pathology database. Hematoxylin-Eosin (HE) stained preparations of the cases were removed from the archive and reviewed. Immunohistochemical markers, namely muscle-specific actin, h-caldesmon, desmin, CD10, melan-A, HMB-45 and WT-1, were done.
Results: The mean age of the patients was 40 years. Abnormal uterine bleeding and abdominal pain were the cause of admission in all patients. In one case (multiple ALM ), infertility was also present. A single tumour mass was observed in 4 cases, while multiple tumours focus (2 separate tumour focuses) were present in one case. The smallest tumour size was 5.2 cm, and the largest was 10 cm. The tumour was submucosal in one case and intramural in four cases. Macroscopically, all cases were sharp-edged, solid, white-grey, with a vortex nodular appearance. Two cases had solid and venous variants, one had venous and cavernous variants, one had venous variants, and one had all three variants. All cases were stained with desmin, h-caldesmon, smooth muscle actin; Staining with CD10, melan-A, HMB-45 and WT-1 was not observed.
Conclusion: Uterine ALM is a rare, benign tumour and preoperative diagnosis is very difficult since there are no specific imaging findings. Therefore, it is crucial for the clinician and pathologist to remember this rare tumour and to distinguish it from similar ones.

Keywords

Angioleiomyoma, myomectomy, immunohistochemistry, myopericytoma

Uterin Anjioleiomyom: 5 Olgunun Klinikopatolojik Değerlendirilmesi

Abstract

Amaç: Anjioleiomyom (ALM) sıklıkla alt ekstremite, baş ve gövde bölgesini içeren, iyi huylu bir perivasküler neoplazmadır. Uterin ALM oldukça nadir bir tümördür. Bu çalışmada olguların klinikopatolojik ve immünohistokimyasal özellikleri ve tedavi yöntemleri kapsamlı bir literatür taraması ile değerlendirildi.
Gereç ve Yöntemler: Hastanemiz patoloji servisine başvuran 5 uterin ALM vakası, patoloji veri tabanından bulundu. Vakaların Hemotoksilen-Eozin (HE) boyalı preparatları arşivden çıkarılarak yeniden gözden geçirildi. Vakalarda immünohistokimyasal olarak düz kas aktin (SMA), h-caldesmon, desmin, CD10, melan-A, HMB-45, ve WT-1 boyaları çalışıldı (Ventana benchmark XT).
Bulgular: Hastaların ortalama yaşı 40 idi. Hastaların hepsinde hastaneye başvuru sebebi anormal uterin kanama ve abdominal ağrı idi. 1 vakada (multipl ALM vakası) infertilite şikayeti de mevcuttu. 4 vakada tek bir tümör kitlesi izlenmekteyken, 1 vakada multipl tümör odağı (2 ayrı tümör odağı) mevcuttu. Tümör boyutu en küçük 5.2 cm, en büyük 10 cm olarak saptandı. 1 vakada tümör submukozal yerleşimli olup, diğer 4 vakada intramural yerleşimliydi. Makroskobik olarak tüm vakalar keskin sınırlı, solid, beyaz gri renkte, girdapsı nodüler görünümdeydi.2 vakada solid ve venöz varyant, 1 vakada venöz ve kavernöz varyant, 1 vakada venöz varyant, 1 vakada da her üç varyant izlendi.Vakaların hepsi desmin, h-kaldesmon, düz kas aktin ile boyanırken; CD10, melan-A, HMB-45 ve WT-1 ile boyanma izlenmedi.
Sonuç: Uterin ALM oldukça nadir, benign bir tümör olup, spesifik görüntüleme bulguları olmadığından preoperatif tanı son derece zordur. Tanı ancak patolojik çalışma ile mümkündür. Bu nedenle, klinisyen ve patoloğun bu nadir görülen tümörü hatırlaması ve histopatolojik inceleme ve gerektiğinde uygun immünohistokimyasal panel ile benzerlerinden ayırması önemlidir.

Keywords

Anjiyoleiomyom, myomektomi, immunohistokimya, myoperisitoma

References

• 1. Weiss SW, Goldblum JR. Benign tumors of smooth muscle. In: Weiss SW, Goldblum JR, eds. Enzinger and Weiss's Soft Tissue Tumors. 4th ed. St Louis, MO: The CV Mosby Co, 2001:699–700.
• 2. Hisaoka M, Quade B. Angioleiomyoma: pericytic (perivascular) tumours. In: Fletcher CDM, Bridge JA, Hogendoorn PCW, Mertens F, eds. World Health Organization Classification of Tumours of Soft Tissue and Bone. Lyon, France: IARC Press, 2013:118-21.
• 3. Wang CP, Chang YL, Sheen TS. Vascular leiomyoma of the head and neck. Laryngoscope 2004; 114(4): 661–5.
• 4. Ide F, Mishima K, Saito I. Angiomyoma in the submandibular gland: a rare location for ubiquitous tumour. J Laryngol Otol. 2003;117(12):1001–2.
• 5. Paal E, Miettinen M. Retroperitoneal leiomyomas: a clinicopathologic and immunohistochemical study of 56 cases with comparison to retroperitoneal leiomyosarcomas. Am J Surg Pathol. 2001;25(11):1355–63.
• 6. Sánchez-Iglesias JL, Capote S, Cubo-Abert M, Carbonell-Socias M, Cabrera S, Illán-Hernández L, 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. 2019;300(4):841-7.
• 7. Garg G, Mohanty SK. Uterine angioleiomyoma: a rare variant of uterine leiomyoma. Arch Pathol Lab Med. 2014;138(8):1115–8.
• 8. Zaloudek CJ, Hendrickson MR, Soslow RA. Mesenchymal tumors of uterus. In: Kurman RJ, Ellenson LH,Ronnett BM, eds. Blaustein’s Pathology of Female Genital Tract. New York, NY: Springer, 2011:453.
• 9. Singh S, Naik M, Bag ND, et al. Angioleiomyoma of uterus masquerading as malignant ovarian tumor. J Midlife Health. 2017;8(3):145–7.
• 10. Hachisuga T, Hashimoto H, Enjoji M. Angioleiomyoma. A clinicopathologic reappraisal of 562 cases. Cancer. 1984;54(1):126-30.
• 11. McCluggage WG, Boyde A. Uterine angioleiomyomas: a report of 3 cases of a distinctive benign leiomyoma variant. Int J Surg Pathol. 2007;15(3):262-5.
• 12. Gupta M, Suryawanshi M, Kumar R, Peedicayil A. Angioleiomyoma of Uterus: A Clinicopathologic Study of 6 Cases. Int J Surg Pathol 2017;26(1):18-23.
• 13. Hsieh CH, Lui CC, Huang SC, Ou YC, ChangChien CC, Lan KC, et al. Multiple uterine angioleiomyomas in a woman presenting with severe menorrhagia. Gynecol Oncol 2003;90(2):348-52.
• 14. Hakverdi S, Dolapçioğlu K, Güngören A, Yaldiz M, Hakverdi AU. Multiple uterine angioleiomyomas mimicking an ovarian neoplasm: a case report. Eur J Gynaecol Oncol. 2009;30(5):592-4.
• 15. Sharma C, Sharma M, Chander B, Soni A, Soni PK. Angioleiomyoma uterus in an adolescent girl: a highly unusual presentation. J Pediatr Adolesc Gynecol. 2014;27(3):69-71.
• 16. Culhaci N, Ozkara E, Yüksel H, Ozsunar Y, Unal E. Spontaneously ruptured uterine angioleiomyoma. Pathol Oncol Res. 2006;12(1):50-1.
• 17. Agorastos T, Dinas K, Patsiaoura K. Cystic degenerated angioleiomyoma mimicking ovarian pathology. Acta Obstet Gynecol Scand. 2001;80(9):863-5.
• 18. Rosai J. Tumours of smooth muscle: soft tissues. In: Rosai J, ed. Rosai and Ackerman’s Surgical Pathology. Vol. 2. St Louis, MO: Mosby Elsevier, 2011:2163.
• 19. Morimoto N. Angiomyoma (vascular leiomyoma): a clinicopathologic study. Med J Kagoshima Univ. 1973;24:663-83.
• 20. Nucci MR, Carcangiu ML, Neilsen GP, Oliva E, Quade B. Mesenchymal tumors and tumor-like lesions. In: Kurman RJ, Carcangiu ML, Herrington CS, Young RH, eds. WHO Classification of Tumors of Female Reproductive Organs. Lyon, France: IARC Press, 2014:198.
• 21. Jeong-A Hong, Gyeong-Eun Heo, Jeong Ja Kwak, Soo-Ho Chung. A case report of angioleiomyoma of uterus. Obstet Gynecol Sci. 2017;60(5):494-7.
• 22. Pierro A, Rotondi F, Cilla S, De Ninno M, Mattoni M, Berardi S, et al. Giant angioleiomyoma of uterus: A case report with focus on CT imaging. Radiol Case Rep. 2018;13(2):371-5.
• 23. Duhig JJ, Ayer JP. Vascular leiomyoma: a study of 61 cases. Arch Pathol. 1959;68:424-30.
• 24. Zizi-Sermpetzoglou A, Myoteri D, Arkoumani E, Koulia K, Tsavari A, Alamanou E, et al. Angioleiomyoma of the uterus: report of a distinctive benign leiomyoma variant. Eur J Gynaecol Oncol. 2015;36(2):210-2.
• 25. Oliva E, Carcangiu ML, Carinelli SG. Mesenchymal tumors. In: Kurman RJ, Carcangiu ML, Herrington CS, Young RH, eds. WHO Classification of Tumors of Female Reproductive Organs. Lyon, France: IARC Press, 2014:141-7.