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The purpose of this article was to report a case of intraconal lacrimal gland tissue and to review the literature on lacrimal gland choristoma. The magnetic resonance imaging findings of a biopsy-proven orbital case are also presented. A PubMed database search was performed using the key terms heterotopic, ectopic, aberrant, choristoma, and lacrimal gland to identify all the previously documented studies on lacrimal gland choristoma, in English, Spanish, and French. We classified the lacrimal gland choristoma cases classified according to the location of the lesions, clinical appearance, management, and outcome. The search targeting the period between 1887 and 2019 returned 79 articles, which were reviewed. We found a total of 113 cases of choristomas with normal lacrimal gland tissue. Only two of them were not associated with the eye or its adnexa while the remaining 111 lesions were found either on the ocular surface (n=46) or in the orbit (n=34). Intraocular choristomas were found in 18 patients, and the rest of the lesions were noted either on the eyelids (n=10) or in the lacrimal drainage system (n=3). Orbital and intraocular choristomas are the most harmful lesions as orbital choristomas are frequently associated with permanent diplopia while intraocular lacrimal gland choristomas have a poor visual prognosis and are a common cause of enucleation of the eye. In one of the reported cases, a corneal lacrimal gland choristoma had been experimentally induced by activating the FGF10 signaling pathway. Lacrimal gland choristomas are not uncommon. This peculiar type of lesion has been experimentally induced and may appear in a variety of locations associated with the globe and its adnexa. RESUMO Descrever um caso de tecido de glândula lacrimal orbitário intracônico e revisar a literatura sobre choristomas lacrimais. Os termos heterotópico, ectópico, aberrante, choristoma e glândula lacrimal foram usados para procurar manuscritos em inglês, espanhol e francês. Todos os artigos encontrados foram revisados. Os choristomas lacrimais relatados foram classificados segundo a localização das lesões, aparência clínica, manejo e desfecho. Imagens de ressonância magnética de um novo caso orbitário comprovado por biópsia também são apresentadas. Foram revisados setenta e nove artigos, de 1887 a 2019, descrevendo 113 casos de choristomas constituídos por tecido de glândula lacrimal normal. Apenas 2 casos (2%) não estavam associados com o olho ou seus anexos. A maioria das 111 lesões restantes foram encontradas na superfície dos olhos (n=46) e nas órbitas (n=34). Os choristomas intraoculares foram encontrados em 18 pacientes. As demais lesões foram diagnosticadas nas pálpebras (n=10) e no sistema lacrimal (n=3). Os choristomas orbitais e intraoculares foram as lesões mais deletérias. Na órbita eles são frequentemente associados com diplopia permanente. Os choristomas lacrimais intraoculares imitam neoplasias e são uma causa comum de enucleação. Os choristomas lacrimais não são incomuns. Este tipo peculiar de lesão já foi induzido experimentalmente uma única vez e pode aparecer em uma variedade de locais associados ao globo e sua anexos. Os choristomas intraoculares têm um prognóstico visual ruim.

Background : : : : The partial loss of the lacrimal gland by obstruction, injury, removal of tumor tissue or other pathological changes may lead to loss of tear flow, bringing harmful consequences to the body. The aim was to evaluate the production of tears after removal of approximately 50% of the main lacrimal gland. Materials, Methods & Results : For this, lacrimal glands from the left eyes of seven dogs, male, adult, with weight ranging from 6 to 13kg were partially removed. In each animal, an incision of approximately 3.0 cm of skin, orbicularis and cutaneous muscle of the eye between the eyeball and the zygomatic process of frontal bone of the lateral left side was performed. After incision of the conjunctiva, close to the zygomatic process, the orbital ligament was located and partially sectioned to expose the main lacrimal gland. With a stainless steel wire graph, the gland was measured and retired approximately 50% of the parenchyma. The fragment was placed in a bottle containing 10% formalin for histological evaluation. The tears production was measured with lacrimal Schirmer test at 28 oC and 70% relative humidity 24 hours before surgery and on the 15th, 30th and 60th days after surgery. At the 60th day, samples of gland lacrimal of the same animals were colleted for histopathologic examination of number, mean diameter of the acini and their nuclei, nucleus: cytoplasm ratio and cell area in a sample of 30 acini per animal and the density of collagen fibers. In the evaluations of the tears production with the Schirmer Test, between the pre-operative (PE), 15, 30 and 60 days post-operatively (PO) were noticed significant differences (p=0.0033) at times between PE and 15 days of PO and between 15 and 60 days of PO. By comparing histological evaluation of lacrimal gland samples in the PE and after 60 days, there were significant differences in the lacrimal gland of nuclear area (p > 0), nuclear cytoplasm proportion (p=0.0006) and cell area (p=0.0083). The collagen fibers formation did not differ significantly between PE and 60 days after partial removal of the gland (p=0.7104). All the ethical principles of ethical use of animals in experiments recommended by the Brazilian College of Animal Experimentation and Ethics Committee of the use of animals at the Federal University of Uberlandia. Discussion : The partial removal allowed the lacrimal gland remaining to offset the production of aqueous tear film: the values found in tear production were above normal throughout the trial period. Induction of cells secreting tears to hyperactivity occurred to offset the demand of production after removing part of the gland. Thus, it was stimulated increase nuclear - giant and multiple nuclei in constant transcription-protein synthesis – and small cytoplasm with little storage tear. This fact explains the increase in cell at 60 days, the decrease in tear production after 24 hours and its return to normal parameters at 60 days postoperatively. In this experiment, the density of collagen fibers found throughout the parenchyma tear between the PE and after 60 days postoperatively was not significant, possibly due to the deposition of fibers are directed to the incision site and the inflammatory reaction, along with the deposition collagen fibers was terminated with 60 days postoperatively. Therefore, the partial removal of the main tear gland in dogs does not interfere significantly in the fall of tear production on day 60 of evaluation in which the tears production came back within the normal patterns.

Purpose: The goal of the present study was to establish a protocol for primary culture of lacrimal gland acinar cells (LGACs) and to assess the effect of adding insulin to the culture media. Methods: LGACs were isolated and cultured from lacrimal glands of Wistar male rats. The study outcomes included cell number, viability, and peroxidase release over time and in response to three concentrations of insulin (0.5, 5.0, and 50.0 μg/mL). Results: In LGAC primary culture, cells started to form clusters by day 3. There was a time-response pattern of peroxidase release, which rose by day 6, in response to carbachol. Culture viability lasted for 12 days. An insulin concentration of 5.0 μg/mL in the culture medium resulted in higher viability and secretory capacity. Conclusions: The present method simplifies the isolation and culture of LGACs. The data confirmed the relevance of adding insulin to maintain LGACs in culture. RESUMO Objetivo: O objetivo do estudo foi estabelecer um protocolo de cultura primária para o isolamento de células acinares da glândula lacrimal (CAGL) e avaliar a relevância de insulina no meio de cultura. Métodos: CAGL foram isoladas e cultivadas a partir das glândulas lacrimais de ratos Wistar machos. Os parâmetros analisados foram: o número de células, viabilidade e secreção da peroxidase ao longo do tempo e em resposta a três concentrações de insulina (0,5; 5,0 e 50,0 μg/ml). Resultados: Na cultura primária de CAGL as células passaram a se agrupar por volta do dia 3. A secreção de peroxidase em resposta ao carbacol aumentou no dia 6. O período de cultura viável foi limitado à 12 dias. Insulina à 5,0 μg/ml no meio de cultura resultou em viabilidade e capacidade secretora maior. Conclusão: o estudo descreveu um método para simplificar o isolamento e cultivo de CAGL. Os dados apresentados confirmam a importância da insulina na manutenção da cultura de CAGL.

PURPOSE: The primary ocular targets of chronic graft-versus-host disease (GVHD) are the lacrimal gland (LG) and the conjunctiva, and their involvement results in keratoconjunctivitis sicca (KCS). The purpose of the present study was to evaluate the frequency of signs and symptoms of KCS present in patients submitted to BMT, to identify the cellular phenotype of the conjunctival and lacrimal gland biopsies of these patients by immunohistochemistry and to correlate the findings with the presence of dry eye due to GVHD. METHODS: Forty-seven patients were clinically classified into two groups: Group I, with ocular GVHD, submitted to allogeneic BMT and Group II, without ocular GVHD, submitted to autologous and allogeneic BMT. Thorough eye examination, including clinical lacrimal function tests and biopsies of the conjunctiva and of the LG were performed in the pre- and posttransplantation period. The biopsies were submitted to immunohistochemical analysis using a panel of monoclonal antibodies. RESULTS: Of the 28 (82.4%) patients with chronic GVHD, 13 (46.4%) presented ocular GVHD. Of the six patients without GVHD, one (16.7%) presented ocular GVHD. None of those submitted to autologous BMT presented ocular GVHD and 14 (41.2%) of 34 patients with allogeneic BMT presented ocular GVHD. All patients with ocular GVHD (Group I) had symptoms and the most frequent were burning, foreign body sensation, blurred vision and dryness. The rose bengal test was one of the most sensitive in this study and slit lamp examination was very useful in the evaluation of corneal and conjunctival alterations, since these alterations were always present in patients with dry eye in our study. Neither symptoms and signs of dry eye nor significant immunologic reaction was observed in the conjunctiva and in the LG of patients without ocular GVHD (Group II). There was an increase in the T lymphocyte population, of T helper lymphocyte (Th/i) and T suppressor-cytotoxic lymphocyte (Ts/c) subpopulation in the conjunctiva and in the LG of patients with ocular GVHD after the transplantation. CONCLUSIONS: Patients submitted to allogeneic BMT may develop ocular GHVD characterized by KCS. The immunohistochemical study of the conjunctiva and lacrimal gland biopsies of these patients suggested that these tissues are the target of the T cell mediated immunological reaction.OBJETIVOS: A glândula lacrimal e a conjuntiva são os tecidos oculares primariamente afetados pela doença enxerto-versus-hospedeiro (DEVH) crônica, e seu envolvimento clínico é caracterizado por ceratoconjuntivite seca (CCS). Os objetivos deste estudo foram os de avaliar a freqüência dos sintomas e sinais de CCS em pacientes submetidos ao transplante de medula óssea (TMO) e identificar a população celular por análise imuno-histoquímica de biópsias da conjuntiva e glândula lacrimal desses pacientes, correlacionando esses achados com o quadro clínico ocular. MÉTODOS: Quarenta e sete pacientes foram classificados em dois grupos: Grupo I, pacientes com DEVH ocular, submetidos ao TMO alogênico e Grupo II, pacientes sem DEVH ocular, submetidos ao TMO alogênico e autólogo. Exame ocular completo, incluindo testes clínicos da função lacrimal, e biópsias de conjuntiva e glândula lacrimal foram realizadas nos períodos pré e pós-transplante. Estudou-se a população celular, por meio de análise imuno-histoquímica das biópsias, utilizando um painel de anticorpos monoclonais. RESULTADOS: Dos 28 (82,4%) pacientes com DEVH crônica, 13 (46,4%) apresentaram DEVH ocular. Dos seis pacientes sem DEVH, um (16,7%) apresentou DEVH ocular. Nenhum paciente submetido a TMO autólogo apresentou DEVH ocular e 14 (41,2%) dos 34 pacientes com TMO alogênico desenvolveram DEVH ocular. Todos os pacientes com DEVH ocular (Grupo I) apresentaram sintomas e os mais freqüentes foram ardor, sensação de corpo estranho, visão borrada e secura ocular. O teste de rosa bengala foi um dos mais sensíveis e o exame biomicroscópico foi considerado muito útil na avaliação das alterações conjuntivais e corneanas, as quais estavam sempre presentes nos pacientes com DEVH ocular deste estudo. Na conjuntiva e na glândula lacrimal dos pacientes sem DEVH ocular não houve reação imunológica significante, concordando com os testes de função lacrimal. Houve aumento da população de linfócitos T, dos linfócitos T auxiliadores (Th/i) e linfócitos T supressores-citotóxicos (Ts/c) após o transplante na conjuntiva e glândula lacrimal de pacientes com DEVH ocular. CONCLUSÕES: Pacientes submetidos ao TMO alogênico podem desenvolver DEVH ocular, caracterizado por ceratoconjuntivite seca. O estudo imuno-histoquímico de biópsias da conjuntiva e glândula lacrimal desses pacientes sugere que esses tecidos são alvo de reação imunológica mediada pelos linfócitos T.

Tumors of the lacrimal gland are rare in clinical practice. Among all of them, the most common epithelial tumor is the lacrimal gland pleomorphic adenoma, which is a benign indolent tumor that usually affects adults in the third and fourth decades of life. We present an unusual case of lacrimal gland pleomorphic adenoma. Its management, radiological findings and outcomes are also described, along with a brief review of the literature. Os tumores da glândula lacrimal são raros na prática clínica. Dentre eles, o tumor epitelial mais comum é o adenoma pleomórfico da glândula lacrimal, que consiste em um tumor benigno, indolente, que geralmente acomete adultos na terceira e quarta décadas de vida. Apresentaremos um caso raro de adenoma pleomórfico de glândula lacrimal, bem como a condução do caso, achados radiológicos e o seu desfecho, além de um breve resumo da literatura.

A 78-year-old male patient presented with double vision, painless palpable mass under the right superolateral orbital rim, downward displacement and restricted adduction of the right eye. His visual acuity was 20/50 OD and 20/20 OS. Hertel exophthalmometry was 21 mm OD and 17 mm OS. Computed tomographic scans showed an infiltrative orbital mass with ill-defined, irregular margins, involving the lacrimal gland and the lateral rectus muscle. The patient underwent an anterior transcutaneous transseptal orbitotomy with incisional biopsy and surgical debulking. Histopathologic evaluation revealed primary ductal adenocarcinoma of the lacrimal gland. Following the metastatic work up, he underwent an eyelid-sparing orbital exenteration. Microscopically, the tumor elements were characterized by large polygonal cells with vesicular nuclei, prominent nucleoli and amphophilic cytoplasm. The tumor components comprised duct-type structures with papillary and cribriform patterns, surrounded by prominent basement membrane. The tumor cells were positive for cytokeratin-7, matrix metalloproteinase (MMP)-2, MMP-9, MMP-13 and proto-oncogene Her-2/neu, but negative for cytokeratin-5, cytokeratin-20, p63, prostate-specific antigen, S-100 protein and thyroid transcription factor. These histopathologic findings were compatible with poorly differentiated ductal adenocarcinoma of the lacrimal gland, T3N0M0. Twenty-four months after orbital exenteration, the patient was diagnosed with ipsilateral parotid gland and cervical lymph node metastases and died of disease. Paciente do sexo masculino e com 78 anos de idade apresentou diplopia, massa palpável abaixo da margem orbitária direita, deslocamento inferior do bulbo ocular direito e limitação da adução do olho direito. A acuidade visual foi 20/50 OD e 20/20 OE. A exoftalmometria de Hertel foi 21 mm OD e 17 mm OE. Tomografia computadorizada mostrou uma massa orbitária, infiltrativa e com margens irregulares, envolvendo a glândula lacrimal e o músculo reto lateral. O paciente foi submetido a uma orbitotomia anterior com biópsia incisional. O exame histopatológico revelou adenocarcinoma ductal primário da glândula lacrimal. Em seguida, o paciente foi submetido a uma exenteração orbitária com preservação das pálpebras. Microscopicamente, os elementos tumorais foram caracterizados por células poligonais grandes com citoplasma anfofílico, núcleo vesicular e nucléolo proeminente. Os componentes tumorais incluíram estruturas ductais com padrões cribriforme e papilífero e cercadas por membrana basal proeminente. As células tumorais foram positivas para citoqueratina 7, metaloproteinase 2 da matriz, metaloproteinase 9 da matriz, metaloproteinase 13 da matriz e Her-2/neu, mas negativas para citoqueratina 5, citoqueratina 20, p63, antígeno prostático específico, proteína S-100 e fator de transcrição da tiroide. Estes achados histopatológicos foram compatíveis com o diagnóstico de adenocarcinoma ductal pouco diferenciado da glândula lacrimal, T3N0M0. Vinte e quatro meses após a exenteração orbitária, o paciente foi diagnosticado com metástases nos linfonodos cervicais ipsilaterais e na glândula parótida ipsilateral e faleceu.