Purpose: To assess the effectiveness of treatments for Morbihan disease. Methods: Following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines, a systematic review of the literature was performed on April 1, 2018, using PubMed, Google Scholar, and Excerpta Medica dataBASE with terms used to describe Morbihan disease, including “Morbihan Disease,” “Morbihan Syndrome,” “lymphedema rosacea,” and “lymphedematous rosacea”. Case reports or case series were included if they fulfilled the following criteria: published in English, peer-reviewed, and reported Morbihan disease. Results: A total of 89 patients—87 patients from 49 articles and 2 cases from the authors’ institution—were included in the final analysis. The median age of patients was 51 years (range: 14–79), and 66 of 89 (74%) patients were men. Male gender correlated with lack of complete response to treatment (odds ratio: 0.25; 95% confidence interval: 0.06–0.97; p = 0.02), while presence of papules or pustules correlated with complete response to treatment (odds ratio: 4.07; 95% confidence interval: 1.04–17.68; p = 0.03). Longer antibiotic duration correlated with response to treatment (p = 0.03), favoring complete over partial response (p = 0.02). Mean antibiotic duration in patients who responded was 4.43 months (standard deviation: 3.49), with complete responders requiring 6.50 months (standard deviation: 4.57). Oral corticosteroids, isotretinoins, and combination therapies did not correlate with treatment response. Conclusions: The presence of papules and pustules correlates with a complete response to treatment, while male gender correlates with a partial response. Patients may benefit from 4- to 6-month duration of tetracycline-based antibiotics. Prospective studies are needed to assess the impact of antibiotic and isotretinoin dose and duration on treatment response.

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Purpose: To measure orbital cavernous hemangioma size using 3 segmentation methods requiring different degrees of subjective judgment, and to evaluate interobserver agreement using these methods. Methods: Fourteen patients with orbital cavernous hemangiomas were included in the study. Pretreatment T2-weighted MRIs were analyzed by 2 observers using 3 methods, including 1 user-dependent image segmentation method that required high degrees of subjective judgment (ellipsoid) and 2 parameter-dependent methods that required low degree of subjective judgment (GrowCut and k-means clustering segmentation). Interobserver agreement was assessed using Lin’s concordance correlation coefficients. Results: Using the ellipsoid method, the average tumor sizes calculated by the 2 observers were 1.68 ml (standard deviation [SD] 1.45 ml) and 1.48 ml (SD 1.19 ml). Using the GrowCut method, the average tumor sizes calculated by the 2 observers were 3.00 ml (SD 2.46 ml) and 6.34 ml (SD 3.78 ml). Using k-means clustering segmentation, the average tumor sizes calculated by the 2 observers were 2.31 ml (SD 1.83 ml) and 2.12 ml (SD 1.87 ml). The concordance correlation coefficient for the ellipsoid, GrowCut, and k-means clustering methods were 0.92 (95% CI, 0.83–0.99), 0.12 (95% CI, –0.21 to 0.44), and 0.95 (95% CI, 0.90–0.99), respectively. Conclusions: k-means clustering, a parameter-dependent method with low degree of subjective judgment, showed better interobserver agreement compared with the ellipsoid and GrowCut methods. k-means clustering clearly delineated tumor boundaries and outlined components of the tumor with different signal intensities.

BackgroundInverted papillomas (IPs) are benign, locally aggressive neoplasms with a high recurrence rate that most commonly arise from the lateral nasal wall. Rarely, IP can originate from the lacrimal sac and/or nasolacrimal duct (NLD) system. A 58-year-old man presented with chronic epiphora and an enlarging mass inferior to his left medial canthal tendon (MCT) for 2 years.ObjectiveThe patients’ clinical presentation and surgical management are described. A literature review on IP of the lacrimal sac and NLD system was conducted.MethodsCase report.ResultsWe present a case of an IP that arose primarily from the lacrimal sac and grew below the MCT rather than extending above it. This is one of few published case reports detailing the use of a combined endoscopic and external approach for the resection of an IP of the lacrimal sac.ConclusionCombined endoscopic and external resection is a viable approach for complete removal of extensive IP of the lacrimal sac and NLD system. IP arising from the lacrimal sac may grow below the MCT or extend above it as previously reported.