Sonographic Reporting Completeness and Sonographic Features of Xanthogranulomatous endometritis: A Systematic Review and Quality Scoring Analysis of Case Reports.

Xiang Ji designed the study, developed the scoring system, performed the literature search and data extraction, analyzed the data, wrote the original draft, and revised the manuscript. The author confirms that he has read and approved the final version of the manuscript and takes full responsibility for the integrity and accuracy of the data.

Among the 18 included patients, 16 reported exact age. The mean age of these patients was 68.7 ± 10.9 years (mean ± standard deviation), with an age range of 45 to 87 years. The remaining two patients were described as being in their “70 s” and “late 70 s”, respectively. The novel standardized scoring scheme was applied to the 18 included studies. The total scores of the included reports yielded a mean of 3.2 out of a maximum of 8 points, with a range from 1 to 6. The detailed scores for each paper are presented in Table  2 . Completeness of sonographic reporting in included case reports. Analysis of individual score items revealed the following: Basic lesion description: At least one descriptor of lesion nature was included in 18 reports (100%). Lesion Measurement: A specific measurement was provided in 13 reports (72.2%). Endometrium‐myometrium relationship: This was described in 12 reports (66.7%). Color doppler assessment: Its use was mentioned and applied in only 3 reports (16.7%). Spectral doppler parameters: RI or PI values were provided in 2 reports (11.1%). Provision of ultrasound images: Images were provided in 5 reports (27.8%). The frequency of described sonographic features across the 18 reports is summarized as follows: Uterus and Endometrium: Uterine enlargement was reported in 38.9% and endometrial thickening or irregularity in 61.1% of cases. Intracavitary Lesions: Intrauterine fluid collection or a mass was reported in the majority of cases (88.9%). Among reports providing specific descriptions, cystic texture was most frequent (68.8%). Vascular Features: Of the three reports that documented the use of Doppler techniques, two described rich intralesional vascularity with a low resistive index (RI < 0.7). The frequency of described sonographic features across the 18 reports is summarized in Table  3 . The frequency of described sonographic features across the reports. Endometrium endometrial thickening or irregularity Intracavitary lesion – presence fluid collection or mass Intracavitary lesion ‐ characteristics (when specified) Echogenicity level ( n  = 4) Hyperechoic Hypoechoic Lesion texture ( n  = 16) Solid Cystic Mixed Uniformity of echogenicity ( n  = 9) Uniform Heterogeneous 2 (11.1%) 2 (11.1%) 4 (22.2%) 11 (68.8%) 1 (5.6%) 3 (16.7%) 6 (33.3%) (Denominator = reports with specific description) (Denominator = 4) 2/4 (50.0%) 2/4 (50.0%) (Denominator = 16) 4/16 (25.0%) 11/16 (68.8%) 1/16 (6.3%) (Denominator = 9) 3/9 (33.3%) 6/9 (66.7%) Intracavitary lesion ‐ morphology (when specified) Polypoidal Lesion measurement Any measurement provided Any color doppler assessment provided Blood flow detected No blood flow detected 3 (16.7%) 2 (11.1%) 1 (5.6%) (Denominator = 3) 2/3 (66.7%) 1/3 (33.3%) Resistive index provided Low resistive index (RI < 0.7) 2 (11.1%) 2 (11.1%) (Denominator = 2) 2/2 (100%)

This study, utilizing a novel scoring system alongside systematic review methodology, conducted the first comprehensive evaluation of the completeness of sonographic reporting in case reports on XGE and summarized its characteristic sonographic findings. To our knowledge, this is also the first attempt to systematically assess the completeness of sonographic reporting in case reports of rare diseases, using the scoring table as an initial tool for such analysis. The kidneys and the gallbladder are the most common sites affected by xanthogranulomatous inflammation [ 26 ]. XGE is extremely rare [ 22 ], and it typically occurs in postmenopausal women, with a median age of 72 years and an age range of 45–88 years. Common clinical presentations include lower abdominal pain, irregular vaginal bleeding, and increased discharge, often secondary to conditions such as cervical stenosis, pyometra, endometrial hyperplasia, or malignancy. Infection is considered the most likely etiology, with other contributing factors including intrauterine hemorrhage and endometriosis. It may also occur after radiotherapy‐induced endometrial or cervical tumor necrosis [ 3 ]. The pathogenesis of XGE is still subject to debate. The pathogenesis may involve chronic inflammation associated with pyometra caused by cervical stenosis or cervical cancer, where obstruction, inflammation, lipid sources, free radical generation, and lipid peroxidation could collectively contribute to the development of XGE [ 20 ]. The patients in the included reports were all postmenopausal, with a median age of 70.5 years, consistent with the commonly reported high‐risk age group. Frequently observed sonographic features of XGE included intrauterine fluid collection or pyometra and endometrial thickening or irregularity. These findings are likely attributable to secretory retention from cervical stenosis or obstruction, chronic suppurative inflammation, and the subsequent histiocytic infiltration—including characteristic foam cells—mucosal disruption, and microabscess formation. Regarding the uniformity of echogenicity, while detailed description was lacking in many reports, heterogeneity was the most common finding (6 out of 9 reports that described the uniformity of echogenicity, 66.7%), which may reflect the underlying process of inflammatory attack and tissue repair. Vascular assessment with Doppler ultrasound was infrequently reported (16.7%). Among the three studies that performed Color Doppler, two reported detectable blood flow; both studies that provided spectral Doppler indices reported low‐resistance values (RI < 0.7). Given the very small number of cases, these observations should be considered hypothesis‐generating and require validation in larger studies. Histopathology still remains the gold standard for diagnosing XGE. The primary value of ultrasound lies in identifying abnormalities, suggesting diagnostic directions, and guiding biopsy. Since XGE may present with intrauterine fluid, endometrial thickening or irregularity, or even hypervascular masses, its imaging features overlap with those of endometrial carcinoma, making differential diagnosis based solely on ultrasound particularly challenging. However, this overlap underscores the importance of maintaining diagnostic awareness. When ultrasound reveals unexplained pyometra or other intrauterine fluid collection, especially when accompanied by a thickened or irregular endometrium in postmenopausal women, XGE should be considered in the differential diagnosis. The value of ultrasound in this context lies in highlighting the imaging similarities between this benign condition and malignant lesions. This could remind clinicians to actively seek evidence supporting XGE during pathological sampling, thereby avoiding misdiagnosis. An analysis of the sonographic descriptions in the included case reports revealed that they primarily focused on basic morphological changes. For instance, hemodynamic information was missing in the vast majority of reports. Incorporating such information could complement existing descriptions and contribute to a more comprehensive understanding of the sonographic presentation of XGE. This study systematically reviewed the sonographic features of XGE and, as a novel initiative, introduced a scoring system to evaluate the completeness of sonographic reporting in published case reports. The application of this tool revealed a considerable heterogeneity in reporting practices, with the majority of cases providing only fundamental morphological details while hemodynamic information from Doppler ultrasound was missing in most reports. By highlighting these variations, our scoring approach offers a practical tool to facilitate more objective and quantifiable assessments of sonographic reporting in future studies on XGE. The scoring system introduced in this study was developed based on two sources: the IETA consensus for standardized terminology in endometrial assessment, and routine clinical practice in gynecologic ultrasound. Rather than adopting all items from the consensus, we selected core elements essential for case reports of XGE, recognizing that sonographic descriptions in the literature on XGE are often brief. Applying the full set of IETA consensus criteria would result in uniformly low scores that fail to distinguish between more and less complete reporting in case reports. The current items represent a foundational set that can be used and refined as more cases accumulate. We view this tool as a practical starting point for improving reporting completeness in this rare condition. The tool assesses two dimensions: descriptive completeness (whether key sonographic features such as morphology, echogenicity, and vascularity are reported) and image completeness (whether original images are provided). This dual structure reflects our view that complete reporting should include both what the observer saw and what can be independently verified. We also encourage, as good practice, the inclusion of operator experience in the methods section, and—in line with routine clinical practice and established guidelines—the provision of complete image sets. For XGE, this includes grayscale, color Doppler, and spectral Doppler where applicable. These elements are not scored but are recommended to enhance transparency and to facilitate future image review by expert panels. The tool is designed specifically for rare diseases, where each case report contributes meaningfully to the limited evidence base. It may be adapted to other rare conditions by modifying the descriptive items according to organ‐specific reporting guidelines. The ultimate goal is not to establish diagnostic criteria, but to improve the completeness of reported data—including thorough sonographic descriptions and verifiable images—thereby enabling a more comprehensive understanding of the sonographic features of rare diseases. The main limitation of this work stems from its reliance on published case reports, which are inherently subject to publication and reporting bias. However, the scoring system introduced in this study may help mitigate this bias by encouraging more complete sonographic reporting in future case reports of rare diseases.

This study summarizes the sonographic features most frequently described in published case reports of XGE, which center on intrauterine abnormalities and endometrial changes. Frequently observed sonographic features of XGE included intrauterine fluid collection or pyometra and endometrial thickening or irregularity. The application of our novel scoring table revealed that the ultrasound descriptions in existing case reports are often incomplete, with doppler assessment being a notable gap. Future reports would benefit from more detailed Doppler and morphological descriptions, along with complete image sets (e.g., grayscale, color Doppler, spectral Doppler where applicable) in line with clinical practice and guidelines. These practices would contribute to a more comprehensive and verifiable understanding of the sonographic features of this rare condition. Transparent reporting of operator experience is also encouraged to further enhance the interpretability of future case reports. We hope that the scoring approach introduced here may also serve as a starting point for evaluating the completeness of sonographic reporting in future systematic reviews of case reports of rare diseases.

Xanthogranulomatous inflammation is a rare chronic inflammation and a mixture of foam lipid loaded tissue cells and inflammatory cells [ 1 , 2 ], which may affect the kidney and gallbladder, and rarely involve endometrium. Xanthogranulomatous endometritis (XGE), an extraordinary disease, may be associated with endometrial carcinoma [ 3 ]. If not properly treated, this disease can have fatal consequences [ 4 ]. Therefore, it is important to make a correct diagnosis. Ultrasound plays an important role in the examination of gynecological diseases because of non‐invasive nature, real‐time capability, safety, and accuracy [ 5 , 6 , 7 ]. The first case of XGE was reported by Barua et al. in 1978 [ 8 ], however, there is currently few English literature describing the ultrasonographic features of XGE till date [ 9 , 10 ]. This study has two primary objectives: (1) to systematically review and summarize the sonographic characteristics of XGE, and (2) to introduce and apply a novel scoring system—demonstrated on XGE—for evaluating the completeness of sonographic reporting in case reports of rare diseases. This scoring system—the first of its kind—was developed to enable a structured assessment of the available evidence, hoping to inform future studies and improve reporting quality.

The lead author Xiang Ji affirms that this manuscript is an honest, accurate, and transparent account of the study being reported; that no important aspects of the study have been omitted; and that any discrepancies from the study as planned (and, if relevant, registered) have been explained.

A systematic literature review was performed on PubMed, Web of Science and EMBASE according to the PRISMA guidelines by two independent researchers (Ji X and He WT). The search terms were: (“Xanthogranulomatous Endometritis” OR “XGE”). The search was conducted for published papers up to October 2025. Case reports written in English describing female patients with a pathological confirmation of XGE who underwent ultrasound examination were included. Studies were excluded if they were irrelevant, non‐English, not case reports, involved non‐human subjects, or lacked any description of the ultrasound findings. Two reviewers (Ji X and He WT) independently screened the titles, abstracts, and full texts against the inclusion and exclusion criteria. A standardized data extraction form was used in Microsoft Excel 2010 to collect the following information from each included study: first author, year of publication, patient age, and ultrasonographic findings. The process of study screening and selection is detailed in the PRISMA flow diagram (Figure  1 ). PRISMA flow diagram for the study. To systematically evaluate the completeness of sonographic reporting in the included case reports, we developed an 8‐point scoring system based on the essential components of routine sonographic assessment and IETA consensus [ 11 ], the system assesses six key elements. The assessment items and scoring criteria are detailed in Table  1 . The assessment items and scoring criteria. Uterine size abnormality: (e.g., “enlarged uterus” or “bulky uterus”). Lesion nature (IETA criteria): Mention of any of the following: Uterine size abnormality: (e.g., “enlarged uterus” or “bulky uterus”). Lesion nature (IETA criteria): Mention of any of the following: – Basic presence (e.g., “intrauterine fluid collection/effusion” or “intrauterine mass/occupying lesion”). – Echogenicity uniformity (e.g., “uniform” or “non‐uniform/heterogeneous”). – Lesion texture (e.g., “cystic”, “solid”, or “mixed”). Basic presence (e.g., “intrauterine fluid collection/effusion” or “intrauterine mass/occupying lesion”). Echogenicity uniformity (e.g., “uniform” or “non‐uniform/heterogeneous”). Lesion texture (e.g., “cystic”, “solid”, or “mixed”). – Echogenicity level (e.g., “hyperechoic/strong echo”, “isoechoic”, or “hypoechoic/weak echo”. C. Precise morphology (IETA criteria): Mention of specific morphological terms (e.g., “nodular”, “mass‐like”, “polypoidal”, or “cauliflower like”). Echogenicity level (e.g., “hyperechoic/strong echo”, “isoechoic”, or “hypoechoic/weak echo”. C. Precise morphology (IETA criteria): Mention of specific morphological terms (e.g., “nodular”, “mass‐like”, “polypoidal”, or “cauliflower like”). C. Precise morphology (IETA criteria): Mention of specific morphological terms (e.g., “nodular”, “mass‐like”, “polypoidal”, or “cauliflower like”). 1 point: Contains any 1 of categories A, B, or C. 2 points: Contains any 2 of categories A, B, or C. 3 points: Contains all 3 categories (A, B, and C). Provides specific dimensions of the lesion or uterus (e.g., length, width, thickness, endometrial thickness). 0 points: No specific measurement values provided. 1 point: At least one specific measurement value provided. Describes the state of the endometrium or the relationship of the lesion to the myometrium/endometrium. 0 points: Not mentioned. 1 point: Explicitly describes relevant features (e.g., “endometrial thickening/irregularity” or “lesion with clear/unclear demarcation from the myometrium”, “lesion involving the myometrium”). Mentions the use of Color Doppler and describes blood flow. 0 points: Use of Doppler not mentioned. 1 point: Explicitly mentions the use of CDFI and describes blood flow signals (e.g., “rich vascularity” or “no internal flow”). Provides hemodynamic parameters. 0 points: No parameters provided. 1 point: Provides RI (Resistive Index) or PI (Pulsatility Index) values. Whether ultrasound images are provided in the paper for reference. 0 points: No ultrasound images provided. 1 point: At least one ultrasound image provided. Data analysis was performed using SPSS 28. Continuous variables are expressed as mean ± standard deviation, frequency data is given as number and percent.