International multicentre retrospective cohort study of ocular adnexal marginal zone B-cell lymphoma

Hindsø, Tine Gadegaard; Esmaeli, Bita; Holm, Frederik; Mikkelsen, Lauge Hjorth; Rasmussen, P; Coupland, Sarah E.; Finger, Paul T.; Graue, Gerardo F.; Grossniklaus, Hans E.; Honavar, Santosh G; Khong, Jwu Jin; McKelvie, Penelope; Mulay, Kaustubh; Sjö, Lene Dissing; Vemuganti, Geeta K.; Thuro, Bradley A.; Heegaard, Steffen

doi:10.1136/bjophthalmol-2019-314008

Cited by 31 publications

(32 citation statements)

References 26 publications

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“…In most cases, the disease was in a T2N0M0 stage and no relapse was noted. This finding is consistent with the current literature that supports the use of EBRT for most cases of primary OEL, especially for low-grade lymphomas such as EMZL, which represented the majority of cases in our series [38][39][40][41][42]. A newly published review by the American Academy of Ophthalmology on treatment of OEL has documented that EBRT has a very good effect on local control, disease-free survival and overall survival in patients with EMZL [43].…”

Section: Discussionsupporting

confidence: 92%

“…No association was detected in our study between the T category and recurrence. The AJCC proposed additional factors recommended just for clinical care: the International Prognostic Index (IPI), tumor cell growth fraction (Ki-67, MIB-1) and lactate dehydrogenase level [ 34 , 35 , 36 , 37 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 , 49 , 50 , 51 , 52 , 53 , 54 , 55 ]. Nonetheless, these factors were not considered in this series as complete data were available only for a small percentage of studied patients.…”

Section: Discussionmentioning

confidence: 99%

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Orbital and Eyelid B-Cell Lymphoma: A Multicenter Retrospective Study

Savino

Midena

Blasi

et al. 2020

Cancers

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Background: The aim of this study was to analyze patients diagnosed, staged and treated for orbital and eyelid B-cell lymphoma (OEL). Methods: One hundred and forty-one cases of OEL were included in this study. Primary endpoints were to analyze the histopathologic findings, the main risk factors and the type of treatment and to correlate them with recurrence of OEL. The secondary endpoint was to determine the progression-free survival (PFS) time. Results: Extranodal marginal zone B-cell lymphoma was the most frequent subtype (66%), followed by small lymphocytic lymphoma (12.7%), diffuse large B-cell lymphoma (DLBCL) (9.2%), follicular lymphoma (6.6%), mantle cell lymphoma (4.3%) and Burkitt lymphoma (1.2%). The probability of relapse was influenced by the histopathologic subtype DLBCL (OR = 7.7, 95% CI 1.8–32.3) and treatment with chemotherapy (OR = 14.9, 95% CI 2.6–83.7). Multivariate analysis showed that the histopathologic subtype DLBCL and chemotherapy treatment retained statistical significance for a poorer PFS, with hazard ratios of 8.581 (p = 0.0112) and 9.239 (p = 0.0094), respectively. Conclusions: Five lymphoma subtypes were found in patients with OEL. The histopathologic subtype and the type of treatment were found to be the main factors influencing treatment outcome.

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Section: Discussionsupporting

confidence: 92%

Section: Discussionmentioning

confidence: 99%

Orbital and Eyelid B-Cell Lymphoma: A Multicenter Retrospective Study

Savino

Midena

Blasi

et al. 2020

Cancers

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“…36 47 Orbital involvement and advanced stage seem to also contribute to prognosis; however, this may be explained by the fact that these highgrade lymphomas were frequently located in the orbit and were commonly advanced at diagnosis. Similar to the 90%-96% disease-specific survival of ocular adnexal marginal zone lymphoma, 37 no deaths were witnessed among patients with EMZL in our cohort. Similarly, no deaths were also documented among the patients with FL.…”

Section: Survival Analysissupporting

confidence: 84%

Paediatric ocular adnexal lymphoma: a population-based analysis

et al. 2020

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ObjectiveTo investigate the incidence, clinicopathological characteristics and survival of ocular adnexal lymphoma (OAL) in the paediatric population.Methods and analysisIn this retrospective case series, the Surveillance, Epidemiology and End Results database was accessed to identify individuals with OAL ≤18 years of age, diagnosed between 1973 and 2015. OAL located in the eyelid, conjunctiva, lacrimal apparatus and orbit were included. Main outcome measures were the age-adjusted incidence rates (IRs) per 1 000 000 population at risk (calculated for the period 2000–2015) and descriptive statistics of demographic and clinicopathological features.ResultsThe IR of paediatric OAL was 0.12 (95% CI 0.08 to 0.16) per 1 000 000. Males (0.15; 95% CI 0.10 to 0.22) and blacks (0.24; 95% CI 0.13 to 0.42) had a higher tendency for OAL development. A total of 55 tumours in 54 children were identified. The majority were localised (78.4%), conjunctival (49.1%) lymphomas. Extranodal marginal zone lymphoma (EMZL, 45.5%, n=25) was the most frequent subtype, followed by diffuse large B-cell lymphoma (DLBCL, 9.1%, n=5), B lymphoblastic lymphoma (7.3%, n=4), follicular lymphoma (5.5%, n=3), Burkitt lymphoma (5.5%, n=3), anaplastic large cell lymphoma (ALCL, 3.6%, n=2), small lymphocytic lymphoma (1.8%, n=1), diffuse large B-cell lymphoma, immunoblastic (1.8%, n=1) and panniculitis-like T-cell lymphoma (1.8%, n=1). Localised, low-grade, conjunctival lymphomas were frequently treated with complete excision with or without radiation, while high-grade and distant tumours usually received chemotherapy. Only 29.1% of paediatric OAL cases were treated with radiation. Three out of five (60%) patients with DLBCL died of lymphoma at a median follow-up of 21 (range 10–86) months, and 1 out of 2 (50%) patients with ALCL died of lymphoma at 23 months from diagnosis.ConclusionOAL in the paediatric population is rare. The majority of OAL are EMZL and are characterised by excellent prognosis. The histological subtype was found to be the main predictor of outcome with cancer-specific deaths observed in patients with DLBCL and ALCL.

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“…11,21,22 Patients with EMZL and FL were found to have a good prognosis, with a 5-year DSS of 93.4% and 86.5%, respectively, which is similar to previous studies of the OA. 23,24 The current recommendation for treating localized stage I-IIE EMZL and FL of the OA is EBRT, applying 24-36 Gy in conventional daily fractions. 25 In line with this, the present study found no significant difference in DSS between low-stage EMZL and FL patients treated with EBRT as monotherapy and patients treated with combination regimens of chemotherapy and EBRT.…”

Section: Discussionmentioning

confidence: 99%

Lymphoma of the Lacrimal Gland — An International Multicenter Retrospective Study

Vest

Mikkelsen

Holm

et al. 2020

American Journal of Ophthalmology

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To characterize the clinical features of subtype-specific lacrimal gland lymphoma and their effect on patient survival.DESIGN: Multicenter retrospective interventional case series.METHODS: Patient data were collected from 6 international eye cancer centers from January 1, 1980, through December 31, 2017. All patients with histologically verified primary or secondary lymphoma of the lacrimal gland were included. Primary endpoints were overall survival (OS) and disease-specific survival (DSS).RESULTS: A total of 260 patients with lacrimal gland lymphoma were identified. The median age was 58 years and 52% of patients were men. Non-Hodgkin B-cell lymphomas constituted 99% (n [ 258) and T-cell lymphomas constituted 1% (n [ 2). The most frequent lymphoma subtypes were extranodal marginal zone Bcell lymphoma (EMZL) (n [ 177, 68%), follicular lymphoma (FL) (n [ 26, 10%), diffuse large B-cell lymphoma (DLBCL) (n [ 25, 10%), and mantle cell lymphoma (MCL) (n [ 17, 7%). Low-grade lymphomas (EMZL and FL) were most commonly treated with external beam radiotherapy (EBRT), whereas highgrade lymphomas (DLBCL and MCL) were treated with chemotherapy in combination with rituximab and/or EBRT. The prognosis was relatively good with a 5-year OS and DSS of 73.8% and 87.5%, respectively. Lymphoma subtype was a statistically significant predictor for DSS, with EMZL (5-year DSS: 93.4%) having the best prognosis and DLBCL (5-year DSS: 52.6%) having the poorest.CONCLUSIONS: This is the largest reported collection of data of subtype-specific lacrimal gland lymphoma. The subtype distribution of lacrimal gland lymphoma resembles that of the ocular adnexa. Prognosis is good and the histologic subtype is a significant predictor for disease-specific survival.

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International multicentre retrospective cohort study of ocular adnexal marginal zone B-cell lymphoma

Cited by 31 publications

References 26 publications

Orbital and Eyelid B-Cell Lymphoma: A Multicenter Retrospective Study

Orbital and Eyelid B-Cell Lymphoma: A Multicenter Retrospective Study

Paediatric ocular adnexal lymphoma: a population-based analysis

Lymphoma of the Lacrimal Gland — An International Multicenter Retrospective Study

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