Abstract
Purpose
To investigate any association between the presence of an adnexal cystic lymphangioma (ACL) and an enlarged leiomyomatous uterus.
Methods
A retrospective observational study was conducted by two expert radiologists using a 10-year MRI database (2008–2018); 85 patients (mean age: 45.5 years ± 10.9) were considered eligible due to the presence of a single (n = 31) or multiple (n = 54) leiomyomas causing distortion of the uterine contour and uterine enlargement. The association of specific leiomyoma features (longest diameter (Dmax), location, number) and uterine volume with the presence of ACL was statistically tested. Diagnosis of ACL was based on typical imaging features (n = 14) and intraoperative/histological findings (n = 3).
Results
ACL (unilateral = 9, bilateral = 8) was recorded in 17/85 (20%) of patients; it was more frequently observed when the largest leiomyoma was located in the uterine fundus (33.3%). Patients with ACL had significantly more leiomyomas (median: 5 vs. 2, p = 0.043), greater Dmax of largest leiomyoma (median: 13.3 vs. 7.2 cm, p < 0.001), and larger uterine volumes (median: 676.7 vs. 223.1 cm3, p < 0.001) compared to patients without ACL. ROC curve analysis for a number of leiomyomas showed that the optimal cut-off for the prediction of ACL was the presence of 5 leiomyomas with 53.8% sensitivity and 84% specificity (AUC = 0.65, 95% CI 0.51–0.83, p = 0.049), Dmax of largest leiomyoma 9.1 cm with 76.5% sensitivity and 77.9% specificity (AUC = 0.83, 95% CI 0.73–0.94, p < 0.001), and uterine volume 311 cm3 with 71% sensitivity and 75% specificity (AUC = 0.79, 95% CI 0.66–0.92, p < 0.001).
Conclusions
The presence of ACL is significantly associated with number of leiomyomas, Dmax of largest leiomyoma, and uterine volume; prospective evaluation of our results is needed to investigate its clinical significance.
Similar content being viewed by others
Abbreviations
- ACL:
-
Adnexal cystic lymphangioma
- AUC:
-
Area under the curve
- C-section:
-
Cesarean section
- Dmax:
-
Maximal diameter
- FIGO:
-
Federation international of gynecology and obstetrics
- MDCT:
-
Multidetector computed tomography
- MRI:
-
Magnetic resonance imaging
- ROC:
-
Receiver operating characteristic
- SI:
-
Signal intensity
References
Sparic R, Mirkovic L, Malvasi A, Tinelli A (2016) Epidemiology of uterine myomas: a review. Int J Fertil Steril 9:424-435
Roisman I, Manny J, Fields S, Shiloni E (1989) Intra-abdominal lymphangioma. Br J Surg 76:485-489
Thomas AM, Leung A, Lynn J (1985) Abdominal cystic lymphangiomatosis: report of a case and review of the literature. Br J Radiol 58:467-469
Iwasa T, Tani A, Miyatani Y et al (2009) Lymphangioma of the ovary accompanied by chylous ascites. J Obstet Gynaecol Res 35:812-815
Singer T, Filmar G, Jormark S, Seckin T, Divon M (2010) Rare case of ovarian cystic lymphangioma. J Minim Invasive Gynecol 17:97-99
Tripathi M, Parshad S, Karwasra RK, Gupta A, Srivastva S, Sarwal A (2015) Retroperitoneal lymphangioma in an adult: a case report of a rare clinical entity. Case Rep Surg. https://doi.org/10.1155/2015/732531
Wilson SR, Bohrer S, Losada R, Price AP (2006) Retroperitoneal lymphangioma: an unusual location and presentation. J Pediatr Surg 41:603-605
Rice M, Pearson B, Treadwell WB (1943) Malignant lymphangioma of the ovary. Am J Obstet Gynecol 45:884-888
Aristizabal SA, Galindo JH, Davis JR, Boone ML (1977) Lymphangiomas involving the ovary. Report of a case and review of the literature. Lymphology 10:219-223
Romeo V, Maurea S, Mainenti PP et al (2015) Correlative imaging of cystic lymphangiomas: ultrasound, CT and MRI comparison. Acta Radiol Open. https://doi.org/10.1177/2047981614564911
Bhavsar T, Saeed-Vafa D, Harbison S, Inniss S (2010) Retroperitoneal cystic lymphangioma in an adult: a case report and review of the literature. World J Gastrointest Pathophysiol 1:171-176
Hauser H, Mischinger HJ, Beham A et al. (1997) Cystic retroperitoneal lymphangiomas in adults. Eur J Surg Oncol 23:322-326
Gonen KA, Abali R, Oznur M, Erdogan C (2013) Lymphangioma: surrounding the ovarian vein and ovary. BMJ Case Rep. https://doi.org/10.1136/bcr-2013-200020
Akyildiz EU, Peker D, Ilvan S, Calay Z, Cetinaslan I, Oruc N (2006) Lymphangioma of the ovary: a case report and review of the literature. J BUON 11:91-93
Kido A, Togashi K, Koyama T, Yamaoka T, Fujiwara T, Fujii S (2003) Diffusely enlarged uterus: evaluation with MR imaging. Radiographics 23:1423-1439
Kubik-Huch RA, Weston M, Nougaret S et al (2018) European Society of Urogenital Radiology (ESUR) Guidelines: MR Imaging of Leiomyomas. Eur Radiol 28:3125-3137
Kearney CE, Hall GH, Purdie DW, Turnbull LW (2001) Ovarian lymphangioma: MRI appearances. Clin Radiol 56:685-687
Moyle PL, Kataoka MY, Nakai A, Takahata A, Reinhold C, Sala E (2010) Nonovarian cystic lesions of the pelvis. Radiographics 30:921-938
Monro MG, Critchley HOD, Fraser IS et al (2018) The two FIGO systems for normal and abnormal uterine bleeding symptoms and classification of causes of abnormal uterine bleeding in the reproductive years: 2018 revisions. Int J Gynaecol Obstet 143:393-408
Radhouane A, Mayada S, Khaled N (2016) Lymphangioma of the ovary: etiology and management. Eur J Obstet Gynecol Reprod Biol 203:342-343
Joo HJ, Lee TJ, Lee SH, Lee EJ (2016) Lymphangioma arising from the ovary. Lymphology 49:21-26
Kleppe M, Kraima AC, Kruitwagen RF et al (2015) Understanding lymphatic drainage pathways of the ovaries to predict sites for sentinel nodes in ovarian cancer. Int J Gynecol Cancer 25:1405-1414
Pillai S, O’Brien D, Stewart CJ (2013) Bilateral ovarian lymphangioma (lymphangioleiomyoma). Int J Gynecol Pathol 32:171-175
Kelsey TW, Ginbey E, Chowdhury MM, Bath LE, Anderson RA, Wallace WH (2016) A Validated Normative Model for Human Uterine Volume from Birth to Age 40 Years. PLoS One. https://doi.org/10.1371/journal.pone.0157375.eCollection2016.
Naik SA (2011) Rare case of ovarian cystic lymphangioma managed at laparoscopy. J Gynecol Endosc Surg 2:97-100
Yildiz C, Karadayi K, Sarkis C, Cetin A (2011) Huge cystic lymphangioma mimicking ovarian malignancy: a case report. Turk J Gastroenterol 22:344-346
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of interest
The authors declare that they have no conflict of interest.
Ethical approval
All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional research committee and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards.
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
About this article
Cite this article
Bourgioti, C., Chatoupis, K., Tzavara, C. et al. Αdnexal cystic lymphangiomas in patients with massive leiomyomatous uterus: a not so uncommon finding on pelvic MRI. Abdom Radiol 45, 537–546 (2020). https://doi.org/10.1007/s00261-019-02106-z
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00261-019-02106-z