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La carúncula lagrimal forma parte de los anexos oculares y es asiento ocasional de neoplasias, las cuales en su mayoría son benignas. Dentro de estos tumores se encuentran los oncocitomas, los cuales están constituidos por células oncocíticas (oxifílicas) y poseen una baja incidencia con menos del 3 % de los tumores y bajos reportes de casos en la literatura. A pesar de que existen varios estudios en Cuba de tumores de anexos oculares no hay evidencia actualizada de casos con oncocitoma. Por ello se considera necesaria la presentación de este caso. Se trata de paciente blanca, femenina de 83 años de edad con antecedentes de hipertensión arterial, compensada bajo tratamiento y de carcinoma basocelular, operada hace siete años. Acudió por presentar aumento de volumen indoloro en el ángulo interno del ojo izquierdo. Al examen físico oftalmológico se observó lesión de 1 cm, gris-rosácea, bien delimitada. Se procedió a su exéresis con sospecha clínica de carcinoma basocelular y se envió espécimen para estudio anatomopatológico, el cual concluyó como oncocitoma quístico papilar (cistoadenoma papilar eosinofílico) de carúncula.
The lacrimal caruncle is part of the ocular adnexa and is the occasional seat of neoplasms, most of which are benign. Among these tumors are oncocytomas, which are constituted by oncocytic (oxyphilic) cells and have a low incidence with less than 3% of tumors and low case reports in the literature. Although there are several studies in Cuba of ocular adnexal tumors, there is no updated evidence of cases with oncocytoma. Therefore, it is considered necessary to present this case. The patient is a white, 83-year-old female with a history of arterial hypertension, compensated under treatment and basal cell carcinoma, operated seven years ago. She presented with painless enlargement of the inner corner of the left eye. Ophthalmologic physical examination revealed a 1 cm lesion, grayish-pinkish, well demarcated. The lesion was excised with clinical suspicion of basal cell carcinoma and the specimen was sent for anatomopathologic study, which concluded as papillary cystic oncocytoma (eosinophilic papillary cystoadenoma) of the caruncle.
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Objective:To evaluate the influence of the clinical staging and different risk factors for the prognosis of ocular adnexal lymphoma.Methods:An ambispective cohort study was conducted.Seventy-four patients diagnosed with primary ocular adnexal lymphoma by pathology at Tianjin Medical University Eye Hospital from November 2010 to December 2018 were enrolled.TNM staging was performed according to local tumor extent, lymph node or systemic involvement.Ann Arbor staging was carried out according to lymph node involvement and extranodal extension.The pathological subtype was classified according to World Health Organization classification of lymphoma.The outcome of disease progression or death was analyzed.Kaplan-Meier method was used for univariate survival analysis.Cox proportional hazard model was employed for multivariate survival analysis to predict the risk factors affecting prognosis, hazard ratio ( HR) and 95% confidence interval ( CI) were estimated.This study adhered to the Declaration of Helsinki.The study protocol was approved by an Ethics Committee of Tianjin Medical University Eye Hospital (No.2021KY[L]-32). Written informed consent was obtained from all patients before entering the cohort. Results:For TNM staging, there were 68 cases in stage <T4, accounting for 91.9%, 6 cases in T4, accounting for 8.1%, 71 cases in N0, accounting for 95.9%, 3 cases in ≥N1, accounting for 4.1%, and no case was in stage M. For Ann Arbor staging, there were 72 cases in stage ⅠE, accounting for 97.3%, and 2 cases in stage ⅡE, accounting for 2.7%.As for pathological classification, 64 cases had mucosa-associated lymphoid tissue (MALT) lymphoma, accounting for 86.5% and 10 cases had non-MALT lymphoma, accounting for 13.5%.The follow-up of the patients was 3 to 117 months, with a median follow-up of 53 months.There were 6 cases dying of disease and 19 cases progressed.The 3-year and 5-year overall survival rates were 96.6% and 86.6%, respectively.The 3-year and 5-year progression-free survival rates were 75.6% and 65.9%, respectively.According to single-factor analysis, T4 stage, non-MALT type and Ki67 positive rate ≥10% were related to declined overall survival rate ( P<0.05). T4 stage, ≥N1 stage, ≥Ann Arbor Ⅱ stage, non-MALT type and Ki67 positive rate ≥10% were related to declined progression-free survival rate ( P<0.05). According to multiple-factor analysis, pathological type ( HR=33.193, 95% CI: 3.388-325.156, P=0.003) was the independent risk factor for overall survival rate.N stage ( HR=11.683, 95% CI: 2.720-50.173, P=0.001) and pathological type ( HR=11.337, 95% CI: 3.841-33.464, P<0.001) were independent risk factors for progression-free survival rate. Conclusions:TNM staging and pathological type are important clinical prognostic indicators for ocular adnexal lymphoma.Patients with high TNM stage or non-MALT lymphoma should be monitored closely.
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RESUMO O oncocitoma é um tipo de neoplasia rara na prática clínica e descrito na literatura, principalmente quando localizado no olho. Quando localizado nos anexos oculares, é mais frequentemente na carúncula. Analisou-se o caso de uma paciente de 74 anos, caucasiana, que relatou desconforto visual no olho esquerdo, e cujo exame físico mostrou lesão tumoral na carúncula esquerda, com volume moderado, presença de neovascularização e secreção excessiva. A excisão cirúrgica da lesão foi realizada sob sedação, e a peça foi enviada para avaliação anatomopatológica. A lesão foi diagnosticada histologicamente como oncocitoma, sem malignidade, e a paciente não apresentou recidiva após o procedimento. Embora raro, esse tumor deve ser reconhecido pelos oftalmologistas, devido ao risco já relatado de desenvolvimento de adenocarcinoma.
ABSTRACT Oncocytoma is a neoplasm rarely observed in clinical practice and reported in the literature, especially when located in the eye. When described in the ocular adnexa, it is most often located in the caruncle. The case of a 74-year-old Caucasian female patient is reported. She complained of visual discomfort in the left eye, and physical examination showed a tumoral lesion in the left caruncle, of moderate volume, presence of neovascularization, and excessive secretion. Surgical excision of the lesion was performed under sedation, and the specimen was sent for pathological examination. The lesion was histologically diagnosed as oncocytoma with no malignancy, and the patient presented no recurrence after the procedure. Although rare, this tumor must be recognized by ophthalmologists due to the risk of developing adenocarcinoma, as already reported.
Subject(s)
Humans , Female , Aged , Adenoma, Oxyphilic/pathology , Eye Neoplasms/pathology , Lacrimal Apparatus Diseases/pathology , Adenoma, Oxyphilic/surgery , Eye Neoplasms/surgery , Lacrimal Apparatus Diseases/surgeryABSTRACT
Primary ocular adnexal mucosa-associated marginal zone lymphoma is the most common ocular adnexal lymphoma. In recent years, its incidence has been increasing, and some techniques of immunology and molecular biology can make us better identify lymphoma with other lymphadenopathies, and some imaging tools can better judge the stage. In addition to radiotherapy and chemotherapy, antibiotic therapy and immune therapy have made progresses in treatment. This paper reviews the research progress of the diagnosis, staging and treatment of the disease.
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Objective To evaluate the clinical features,therapies and prognosis in patients with mucosa-assoeiated lymphoid tissue (MALT) lymphoma in ocular adnexal marginal zone (OAML).Methods A retrospective analysis was made upon clinical data from 21 patients with OAML admitted into Beijing Tongren Hospital from June,2008 to December,2011.Results There were 12 (57.1 ％) men and 9(42.9％) women,with a median age of 57 (23-79) years old.Majority of patients had localized pathological changes.Among them,16 patients (76.2％) were in stage Ⅰ E,and 5 (23.8％) in stage Ⅳ E.Surgical resection as the sole treatment was performed in 13 patients (61.9％),and positron emission tomography CT(PET-CT) imaging demonstrated normal fluorine 18-fluorodeoxyglucose (FDG) uptake after surgical resection,who were managed with no further therapy.All the 13 patients were followed up for median 14 (5-38) months,and all in complete remission.Combination chemotherapy was given to 8(38.1％) patients.Three patients in stage Ⅰ E treated with COP (cyclophosphamide,vineristine and prednisone) or CHOP (cyclophosphamide,adriamycin,vincristine and prednisone) were all in partial remission.Five patients in stage ⅣE were treated with COP/CHOP in combination with rituximab,and all in complete remission.The 3-year overall survival rate and disease-free survival rate in the total patients were 100.0％ and 74.9％ respectively.Conclusions The patients with OAML generally have localized disease,show indolent clinical course,and present low lymphoma-related mortality.Surgical resection is a very important treatment in the patients with local disease.Systemic chemotherapy should be considered in patients at advanced stages.Rituximab in combination with chemotherapy can improve the remission rate.
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Infectious factors contribute to human cancers.In past few years,chlamydia,hepatitis C virus (HCV)and helicobacter pylori (HP)had been found in the mucosa-associated lymphoid tissue (MALT)lymphomas of ocular adnexa.The infectious factors had been considered to be associated with the oncogenesis,management and treatment of tumor.Antibiotic therapy against infectious factors may herald a future with a curtailed role for traditional therapies of surgery,radiation,and chemotherapy.Unlike MALT lymphoma of gastric related to a single infectious factor,multiple organisms may play a role in the etiology in MALT lymphoma of ocular adnexa.MALT lymphoma of ocular adnexa is seldom in clinic.The characteristics of MALT lymphoma of ocular adnexa,the relationship of MALT lymphoma of ocular adnexa and causal agents are reviewed.
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PURPOSE: To report 3 cases of hyper-IgG4 syndrome involving ocular adnexa. CASE SUMMARY: A 66-year-old woman presented with swelling of the bilateral upper eyelids with ocular pain that began 1 year before. Nodular episcleral injection of the left eye and other generalized symptoms, such as cough, decreased hearing ability, multiple nodular lesions of the bilateral lungs and right kidney, together suggested rheumatic disease. Orbital computed tomographic images revealed diffuse swelling of the bilateral lacrimal glands. After immunostaining a surgically-biopsied specimen from the lacrimal gland for IgG4 expression, 15% of infiltrated lymphoplasmacytic cells were IgG4-positive. Similar findings were shown with biopsied specimens from the lung and kidney; therefore, the patient was diagnosed with Hyper-IgG4 syndrome. A 49-year-old woman complained of a mass in the left upper eyelid that began 4 years earlier. Orbital computed tomographic images showed a 5-mm-sized mass in the left upper eyelid. Ocular adnexal Hyper-IgG4 syndrome was confirmed by the immunostained biopsy from the left upper eyelid, showing infiltration of IgG4-positive lymphoplasmacytic cells. A 51-year-old woman presented with swelling of the bilateral lacrimal glands. Enlargement of the bilateral lacrimal glands were apparent in orbital computed tomographic images. After anti-IgG4 antibody staining of a biopsied specimen from the right lacrimal gland, dense infiltration of IgG4-positive lymphoplasmacytic cells was observed. The patient was also diagnosed with Hyper-IgG4 syndrome.
Subject(s)
Aged , Female , Humans , Middle Aged , Biopsy , Cough , Eye , Eyelids , Hearing , Immunoglobulin G , Kidney , Lacrimal Apparatus , Lung , Orbit , Rheumatic DiseasesABSTRACT
The medical literature contains few reports of ectopic lacrimal gland. Ectopic lacrimal gland has been observed in the eye and in various sites in the ocular adnexa. The authors had experienced one case of ectopic lacrimal gland in the super olateral side of orbit. The authors report this case with the review of literatures.