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High-grade squamous intraepithelial lesion arising adjacent to vulvar lymphangioma circumscriptum: a tertiary institutional experience.
Oncotarget 2016; 7(30):48120-48129O

Abstract

Lymphangioma circumscriptum of the vulva occurs in patients who have undergone radical hysterectomy, lymph node dissection, or radiation therapy for management of advanced uterine cancer. Since vulvar lymphangioma circumscriptum typically presents as multiple, grossly verrucous vesicles of various sizes, it may be impossible to clinically distinguish vulvar lymphangioma circumscriptum from other vulvoperineal cutaneous diseases. In the present study, 16 (1.6%) out of the 1,024 vulvar biopsy or excision specimens were diagnosed as lymphangioma circumscriptum. In two (12.5%) out of the 16 cases, unusual histopathological findings were observed. Both patients had previously undergone radical hysterectomy with lymph node dissection and postoperative radiation therapy or concurrent chemoradiation therapy for advanced cervical cancer. Microscopic examination revealed high-grade squamous intraepithelial lesions, which were located immediately adjacent to the normal squamous epithelium covering the dilated subepithelial lymphatic vessels. Further, human papillomavirus genotyping confirmed that both patients were infected with high-risk human papillomavirus. High-grade squamous intraepithelial lesion cannot be grossly distinguished from vulvar lymphangioma circumscriptum because the multiple, verrucous vesicles that constitute the characteristic gross appearance of vulvar lymphangioma circumscriptum hinder its distinction. In this regard, our cases of high-grade squamous intraepithelial lesion, located adjacent to vulvar lymphangioma circumscriptum, support the notion that active surgical excision is necessary for the treatment of vulvar lymphangioma circumscriptum.

Authors+Show Affiliations

Department of Pathology, Graduate School, Kyung Hee University, Seoul, Republic of Korea.Department of Obstetrics and Gynecology, Pusan National University Yangsan Hospital, Pusan National University School of Medicine, Yangsan-si, Gyeongsangnam-do, Republic of Korea.Department of Obstetrics and Gynecology, Pusan National University Yangsan Hospital, Pusan National University School of Medicine, Yangsan-si, Gyeongsangnam-do, Republic of Korea.Department of Pathology, Severance Hospital, Yonsei University College of Medicine, Seoul, Republic of Korea.

Pub Type(s)

Journal Article

Language

eng

PubMed ID

27329721

Citation

Bae, Go Eun, et al. "High-grade Squamous Intraepithelial Lesion Arising Adjacent to Vulvar Lymphangioma Circumscriptum: a Tertiary Institutional Experience." Oncotarget, vol. 7, no. 30, 2016, pp. 48120-48129.
Bae GE, Yoon G, Song YJ, et al. High-grade squamous intraepithelial lesion arising adjacent to vulvar lymphangioma circumscriptum: a tertiary institutional experience. Oncotarget. 2016;7(30):48120-48129.
Bae, G. E., Yoon, G., Song, Y. J., & Kim, H. S. (2016). High-grade squamous intraepithelial lesion arising adjacent to vulvar lymphangioma circumscriptum: a tertiary institutional experience. Oncotarget, 7(30), pp. 48120-48129. doi:10.18632/oncotarget.10158.
Bae GE, et al. High-grade Squamous Intraepithelial Lesion Arising Adjacent to Vulvar Lymphangioma Circumscriptum: a Tertiary Institutional Experience. Oncotarget. 2016 Jul 26;7(30):48120-48129. PubMed PMID: 27329721.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - High-grade squamous intraepithelial lesion arising adjacent to vulvar lymphangioma circumscriptum: a tertiary institutional experience. AU - Bae,Go Eun, AU - Yoon,Gun, AU - Song,Yong Jung, AU - Kim,Hyun-Soo, PY - 2016/03/21/received PY - 2016/06/06/accepted PY - 2016/6/23/pubmed PY - 2018/3/3/medline PY - 2016/6/23/entrez KW - high-grade squamous intraepithelial lesion KW - human papillomavirus KW - lymphangioma circumscriptum KW - vulva SP - 48120 EP - 48129 JF - Oncotarget JO - Oncotarget VL - 7 IS - 30 N2 - Lymphangioma circumscriptum of the vulva occurs in patients who have undergone radical hysterectomy, lymph node dissection, or radiation therapy for management of advanced uterine cancer. Since vulvar lymphangioma circumscriptum typically presents as multiple, grossly verrucous vesicles of various sizes, it may be impossible to clinically distinguish vulvar lymphangioma circumscriptum from other vulvoperineal cutaneous diseases. In the present study, 16 (1.6%) out of the 1,024 vulvar biopsy or excision specimens were diagnosed as lymphangioma circumscriptum. In two (12.5%) out of the 16 cases, unusual histopathological findings were observed. Both patients had previously undergone radical hysterectomy with lymph node dissection and postoperative radiation therapy or concurrent chemoradiation therapy for advanced cervical cancer. Microscopic examination revealed high-grade squamous intraepithelial lesions, which were located immediately adjacent to the normal squamous epithelium covering the dilated subepithelial lymphatic vessels. Further, human papillomavirus genotyping confirmed that both patients were infected with high-risk human papillomavirus. High-grade squamous intraepithelial lesion cannot be grossly distinguished from vulvar lymphangioma circumscriptum because the multiple, verrucous vesicles that constitute the characteristic gross appearance of vulvar lymphangioma circumscriptum hinder its distinction. In this regard, our cases of high-grade squamous intraepithelial lesion, located adjacent to vulvar lymphangioma circumscriptum, support the notion that active surgical excision is necessary for the treatment of vulvar lymphangioma circumscriptum. SN - 1949-2553 UR - https://www.unboundmedicine.com/medline/citation/27329721/High_grade_squamous_intraepithelial_lesion_arising_adjacent_to_vulvar_lymphangioma_circumscriptum:_a_tertiary_institutional_experience_ L2 - http://www.impactjournals.com/oncotarget/misc/linkedout.php?pii=10158 DB - PRIME DP - Unbound Medicine ER -