Abstract
BackgroundAlthough studies have investigated the risk of second primary malignancies (SPMs) associated with lymphoma of various sites, limited studies have investigated this risk in patients with lymphoma originating within the ocular adnexa. We conducted a retrospective study to assess incidence of secondary malignancies in patients with a prior diagnosis of ocular adnexal lymphoma (OAL) and to determine latency periods and age-groups at increased risk for SPM occurrence.MethodsRetrospective analysis was performed on data obtained from Surveillance, Epidemiology, and End Results (SEER) 9 database. Patients with an initial primary malignancy diagnosis of OAL between 1973 and 2015 were included in the study. Standardized incidence ratios (SIR) and excess absolute risks (EAR) compared to a SEER reference population with similar sex, race, age, and calendar year were computed for SPMs. Excess absolute risk is per 10,000 individuals; alpha of 0.05 was used.ResultsOf 1834 patients with primary ocular adnexal lymphoma, 279 developed a secondary malignancy during average follow-up of 110.03 months (+/− 88.46), denoting higher incidence than expected (SIR 1.20; 95% CI, 1.07 to 1.35; EAR 30.56). Amongst the primary lymphoma cohort, 98.7% (1810/1834) of patients had non-Hodgkin’s lymphoma and amongst those that developed secondary malignancies, 99.6% (278/279) had non-Hodgkin’s lymphoma. Patients exhibited increased incidence of lymphohematopoietic and non-lymphohematopoietic second malignancies and no secondary malignancies of the eye or orbit. Patients had increased incidence of secondary malignancies in the first year (SIR 2.07; 95% CI, 1.49 to 2.79; EAR 150.37) and 1–5 years following lymphoma diagnosis (SIR 1.24; 95% CI, 1.01 to 1.51; EAR 34.89). Patients with various OAL subtypes demonstrated differing patterns of site-specific and overall SPM risk.ConclusionsPatients with prior diagnosis of ocular adnexal lymphoma possess increased risk of hematologic and non-hematologic secondary malignancies. Risk of secondary malignancy could vary by lymphoma subtype. Patients with ocular adnexal lymphoma may benefit from regular surveillance to promote early detection of second primary malignancies.
Highlights
Studies have investigated the risk of second primary malignancies (SPMs) associated with lymphoma of various sites, limited studies have investigated this risk in patients with lymphoma originating within the ocular adnexa
These neoplasms are broadly classified as Hodgkin’s lymphoma (HL) and non-Hodgkin’s lymphoma (NHL), NHL accounts for the vast majority of ocular adnexal lymphoma (OAL)
This can be attributed to increased occurrences of both hematologic and non-hematologic malignancies; SPMs of lymphohematopoietic origin included Hodgkin’s lymphoma (SIR 5.64; 95% Confidence interval (CI), 1.16 to 16.47; excess absolute risks (EAR) 1.6), non-Hodgkin’s lymphoma (SIR 5.29; 95% confidence intervals (95% CI), 3.95 to 6.93; EAR 27.27), and leukemia (SIR 2.20; 95% CI, 1.23 to 3.62; EAR 5.28) whereas SPMs of nonlymphohematopoietic origin included non-epithelial skin sites (SIR 4.91; 95% CI, 1.59 to 11.46; EAR 2.58), the kidney (SIR 2.08; 95% CI, 1.08 to 3.64; EAR 4.03), and the nervous system excluding brain (SIR 19.90; 95% CI, 2.41 to 71.89; EAR 1.23) (Table 2)
Summary
Studies have investigated the risk of second primary malignancies (SPMs) associated with lymphoma of various sites, limited studies have investigated this risk in patients with lymphoma originating within the ocular adnexa. We conducted a retrospective study to assess incidence of secondary malignancies in patients with a prior diagnosis of ocular adnexal lymphoma (OAL) and to determine latency periods and age-groups at increased risk for SPM occurrence. OAL can originate from the orbit and surrounding structures, including the conjunctiva, orbital soft tissues, eyelid, lacrimal glands/drainage system or other adnexa [3]. These neoplasms are broadly classified as Hodgkin’s lymphoma (HL) and non-Hodgkin’s lymphoma (NHL), NHL accounts for the vast majority of OAL. The most common major lymphoma subtypes responsible for OAL are extranodal marginal-zone B-cell lymphoma (EMZL) (55–66%), follicular lymphoma (FL) (10–29%), diffuse large B-cell lymphoma (DLBCL) (9–13%), mantle cell lymphoma (MCL) (6–11%), and small lymphocytic lymphoma (CLL/SLL) (2–5%) [6, 7]
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