Association of retroperitoneal lymphangioleiomyomatosis with endosalpingiosis: a case report

Lorente-Herce, José Manuel; Ruiz-Luque, Virgilio; Aguilar-Luque, José; Martínez-García, Pablo; Díaz-Gómez, Daniel

doi:10.4076/1757-1626-2-6331

Cited by 4 publications

(2 citation statements)

References 10 publications

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“…Most (81.4%, n = 171) included a histological picture, 8.1% (n = 17) an ultrasound image, 11.0% (n = 23) a CT-scan, and 11.4% (n = 24) an MRI image. More than half (57.1%, n = 120) of the studies included only information on the anatomical distribution and no depiction of the phenotype [ 1 , 8 , 104 , 105 , 106 , 107 , 108 , 109 , 110 , 111 , 112 , 113 , 114 , 115 , 116 , 117 , 118 , 119 , 120 , 121 , 122 , 123 , 124 , 125 , 126 , 127 , 128 , 129 , 130 , 131 , 132 , 133 , 134 , 135 , 136 , 137 , 138 , 139 , 140 , 141 , 142 , 143 , 144 , 145 , 146 , 147 , 148 , 149 , 150 , 151 , 152 , 153 , 154 , ...…”

Section: Resultsmentioning

confidence: 99%

Intraoperative Appearance of Endosalpingiosis: A Single-Center Experience of Laparoscopic Findings and Systematic Review of Literature

Burla

Kalaitzopoulos

Mrozek

et al. 2022

JCM

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Background: Endosalpingiosis is assumed to be the second most common benign peritoneal pathology after endometriosis in women. Although recent studies indicate a significant association with gynecologic malignancies, many underlying principles remain unclear. This work aimed to systematically describe the intraoperative appearance of endosalpingiosis. Methods: Data and intraoperative videos of patients with histologically verified endosalpingiosis were retrospectively reviewed. The main outcome measures were macroscopic phenotype and anatomical distribution. Additionally, a systematic review searching PubMed (Medline) and Embase was conducted. Results: In the study population (n = 77, mean age 40.2 years (SD 16.4)), the mean size of lesions was 3.6 mm and the main visual pattern was vesicular (62%). The most frequent localization was the sacrouterine ligaments (24.7%). In the systematic review population (n = 1174 (210 included studies overall), mean age 45.7 years (SD 14.4)), there were 99 patients in 90 different studies with adequate data to assess the appearance of the lesions. The mean size of the lesions was 48.5 mm, mainly with a cystic visual pattern (49.5%). The majority of the lesions affected the ovaries (23.2%), fallopian tubes (20.4%), or lymph nodes (18.5%). Comparing this study to the literature population, the main differences concerned the size (p < 0.001) and main visual patterns (p < 0.001) of lesions. Conclusions: The usual intraoperative findings of endosalpingiosis appeared less impressive than described in the literature. In our study population, lesions of a few millimeters in size with a vesicular appearance were mostly seen, most frequently in the sacrouterine ligament area. Intraoperative recognition by the gynecologic surgeon and histologic diagnosis should play an important role in further understanding this entity, scientifically and clinically.

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Section: Resultsmentioning

confidence: 99%

Intraoperative Appearance of Endosalpingiosis: A Single-Center Experience of Laparoscopic Findings and Systematic Review of Literature

Burla

Kalaitzopoulos

Mrozek

et al. 2022

JCM

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“…To our knowledge, a total of 6 cases of lymphangiomyomatosis associated with ectopic tubal-like epithelium have been described, 16,[26][27][28][29] and none have undergone molecular analysis. All of the cases were large cysts greater than 4 cm in dimension located in the retroperitoneum/pelvis of female patients; morphologically half of the cases had evidence of a pre-existing lymph node replaced by the neoplastic lymphangiomyomatosis component.…”

Section: Discussionmentioning

confidence: 99%

Retroperitoneal Cystic Adenopecoma with Genetic Analysis: A Rare Neoplasm

et al. 2022

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Lymphangiomyomatosis is a member of the PEComa family, and usually involves the pulmonary parenchyma of middle-aged females. Infrequently, it may involve abdominal and retroperitoneal lymph nodes, and rarely it has been described to be associated with fallopian tube-type ciliated epithelium co-existing in one neoplasm. To increase our understanding of this unusual tumor, we describe the morphology and genetics of one case and review the literature. We present the case of a 50-year-old female found to have 12.5 and 7.7 cm cystic retroperitoneal masses, describe its unique pathological features and review the literature on the previously reported cases. Based on its unique morphological, immunohistochemical, and molecular features we propose the term adenoPEComa to represent this entity. This case represents a rare example of adenoPEComa with lymphangiomyomatosis of the lymph nodes. It is the first example that has undergone next-generation sequencing revealing a mutation in TSC2 making it a confirmed member of the PEComa family of tumors.

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Evidence for lymphatic pathogenesis of endosalpingiosis: the more things change, the more they stay the same

Russell

Anderson

2016

Pathology

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Association of retroperitoneal lymphangioleiomyomatosis with endosalpingiosis: a case report

Cited by 4 publications

References 10 publications

Intraoperative Appearance of Endosalpingiosis: A Single-Center Experience of Laparoscopic Findings and Systematic Review of Literature

Intraoperative Appearance of Endosalpingiosis: A Single-Center Experience of Laparoscopic Findings and Systematic Review of Literature

Retroperitoneal Cystic Adenopecoma with Genetic Analysis: A Rare Neoplasm

Evidence for lymphatic pathogenesis of endosalpingiosis: the more things change, the more they stay the same

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