Oncocitoma em carúncula: relato de caso / Oncocytoma of the caruncle: a case report

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.

Rhinosporidiosis of the lacrimal sac masquerading as chronic dacryocystitis: a rare presentation

Rhinosporidiosis is a chronic infection of the mucous membrane caused by the Rhinosporiduim seeberi, which infects through transepithelial penetration. Although described worldwide, this entity is mostly found in the western hemisphere, afflicting young people, predominantly males, associated in many cases with recreational or professional contact with bath in ponds, rivers, or stagnant waters. The clinical features are varied depending on the affected membrane, in some cases mimicking other diseases postponing the correct diagnosis. Although nasal obstruction and epistaxis are the common clinical presentations in sinonasal rhinosporidiosis, patients with epiphora without a nasal mass often challenge the diagnosis. In the present case, we have documented a case of isolated lacrimal sac rhinosporidiosis masquerading as chronic dacryocystitis, which was successfully managed by endoscopic excision, accompanied by a literature review.

Pilomatrix carcinoma of the lacrimal caruncle: a case report / Carcinoma pilomatricial da carúncula lacrimal: relato de caso

ABSTRACT A 45-year-old man presented with a 3-month history of a mass located in the caruncle of his right eye. An incisional biopsy had been performed one month prior by another specialist, and the histopathology report showed basal cell carcinoma. The mass was completely excised with a 2 mm safety margin, and the large conjunctival defect was reconstructed with one sheet of amniotic membrane allograft. A histological diagnosis of pilomatrix carcinoma was established. To prevent recurrence after surgery, we added bevacizumab (25 mg/mL, 1.25 mg/mL per drop) eye drops four times per day for three months. At the one-year follow-up, the patient showed no evidence of local recurrence or distant metastasis after initial excision and remains under close follow-up. Pilomatrix carcinoma should be considered in the differential diagnosis of a caruncular mass.

RESUMO Um homem de 45 anos apresentou história de massa na carúncula no olho direito durante 3 meses. Uma biópsia incisional foi realizada 1 mês antes por outro especialista e o laudo histopatológico mostrava carcinoma basocelular. A massa foi completamente excisada, com uma margem de segurança de 2 mm, e a grande lesão conjuntival foi reconstruída com uma folha de aloenxerto de membrana amniótica. Foi estabelecido um diagnóstico histológico de carcinoma pilomatricial. Para evitar a recorrência após a cirurgia, adicionamos colírio de bevacizumabe (25 mg/mL, 1,25 mg/mL por gota) quatro vezes ao dia durante três meses. No seguimento de 1 ano, o paciente não apresentou evidência de recidiva local ou metástase distante após a excisão inicial e continua sob acompanhamento próximo. O carcinoma pilomatricial deve ser considerado no diagnóstico diferencial de uma massa caruncular.

Bilateral simultaneous endoscopic dacryocystorhinostomy: outcome and impact on the quality of life of the patients

Introduction: Bilateral simultaneous endoscopic dacryocystorhinostomy (endo-DCR) has received little attention in the literature, thus many surgeons continue to address bilateral nasolacrimal duct obstruction at two stages, rather than in the same setting. Objective: To evaluate the feasibility and the outcome of simultaneous bilateral Endo- DCR and its impact on the quality of life of the patients. Methods: We have conducted a retrospective analysis of patients who underwent bilateral simultaneous endo-DCR between March 2013 and February 2017 at our tertiary care institution. The reviewed data included clinical presentation; operative details; success rate; pre and postoperative evaluation of the symptoms of the patients, using the Nasolacrimal Duct Obstruction Symptom Score Questionnaire; satisfaction of the patients, and improvement in the quality of life, assessed by the Glasgow Benefit Inventory (GBI) questionnaire. Results: Out of 128 cases in which endo-DCRs were performed, 13 were bilateral (26 sides). Postoperative success was documented in 24 of the 26 sides (92.3%), with a mean follow-up duration of 16.2 months. The two failed sides were reported in the same case. The preoperative symptom score ranged between 12 and 80 (mean ± standard deviation [SD]: 38.23 ± 15.7). The postoperative symptom score was significantly lower (mean ± SD: 5.4 ± 12.9). The success rates in unilateral and bilateral cases were comparable, with no statistically significant difference. A notable improvement in the quality of life of the patients was also reported, with a mean GBI score of 81.38 ± 12.37. Conclusion: Our results support that a simultaneous bilateral endo-DCR is a safe procedure that offers a high success rate, spares the patient from the stress of a second surgery, provides the patient with a bilateral resolution of the symptoms, and confers an immediate improvement in the quality of life of the patients (AU)

Oncocytoma of the lacrimal gland: a case report / Oncocitoma de glândula lacrimal: relato de caso

ABSTRACT Here we describe a rare case of a benign tumor in the lacrimal gland of a healthy 4-year-old girl. Mild proptosis was the only abnormality observed on clinical examination. Magnetic resonance imaging of the right orbit revealed an oval, solid, well-circumscribed, homogeneous mass extending from the lacrimal gland and measuring 2.5 × 2.3 × 1.7 cm without any evidence of invasion into adjacent bones. The lesion was surgically excised and histological analyses defined the diagnosis of oncocytoma of the lacrimal gland. Although rare, oncocytoma should be included in the differential diagnosis of lacrimal gland tumors.

Resumo Nós descrevemos um raro caso de tumor benigno na glândula lacrimal em uma criança sadia de 4 anos de idade. Clinicamente, a paciente apresentava apenas uma discreta proptose. A ressonância nuclear magnética (RNM) de órbita direita revelou a presença de uma massa oval, sólida, bem-circunscrita, homogênea, se extendendo a partir da glândula lacrimal, medindo 2,5 cm x 2,3 cm x 1,7 cm, sem nenhum sinal evidente de invasão a estrutura óssea adjacente. A lesão foi cirurgicamente removida e analizada histopatologicamente, sendo estabelecido o diagnóstico de oncocitoma de glândula lacrimal. Apesar de raro, o oncocitoma deve ser incluído no diagnóstico diferencial de qualquer tumor originado da glândula lacrimal.

Adenoid cystic carcinoma of the lacrimal sac: case report / Carcinoma adenóide cístico do saco lacrimal: relato de caso

ABSTRACT Lacrimal sac tumors are rare with a clinical presentation that typically includes obstruction of the lacrimal drainage system and epiphora as the most frequent symptom. Cribriform adenoid cystic carcinoma (ACC) is the most common malignant epithelial tumor of the lacrimal gland and minor salivary glands; however, its occurrence in the lacrimal drainage apparatus is extremely rare. Given the rarity of ACC, definitive diagnosis is almost invariably late conferring a poor prognosis. Herein we report the case of a 41-year-old woman with primary ACC of the lacrimal sac and describe the ophthalmological examination, diagnosis, and multidisciplinary treatment of this rare type of tumor.

RESUMO Tumores do saco lacrimal são raros. A apresentação clínica muitas vezes mostra uma obstrução no sistema de drenagem lacrimal sendo a epífora o sinal mais frequente. Carcinoma adenóide cístico cribriforme (ACC) é o tumor epitelial maligno mais comum da glândula lacrimal e glândulas salivares menores, mas a sua ocorrência no aparelho de drenagem lacrimal é extremamente rara. Infelizmente, devido a raridade destes tumores, o diagnóstico preciso é quase sempre atrasado, o que por sua vez leva a um pior prognóstico. Nós relatamos o caso de uma mulher de 41 anos de idade, com ACC primário do saco lacrimal e analisamos o exame oftalmológico, diagnóstico e tratamento multidisciplinar deste tipo de tumor.

Pleomorphic Adenoma Lacrimal Gland.

Pleomorphic adenoma is the most common epithelial tumour of the lacrimal gland1. Histological confirmation is necessary for diagnosis. I report a case of pleomorphic adenoma in an otherwise well 39 year old female patient and review the clinical and histological features of this tumour. These lesions are usually cured by simple surgical excision3, Inadequate removal leads to recurrence6. The incidence of pleomorphic adenoma is 3.4 to 4.8 % of the orbital lesions5.

Primary ductal adenocarcinoma of the lacrimal gland: case report / Adenocarcinoma ductal primário da glândula lacrimal: relato de caso

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.

Ten-year follow-up of a case series of primary epithelial neoplasms of the lacrimal gland: clinical features, surgical treatment and histopathological findings / Dez anos de acompanhamento de uma série de casos de neoplasias epiteliais primárias da glândula lacrimal: características clínicas, tratamento cirúrgico e achados histopatológicos

PURPOSE: To describe and analyze the features of a cases series of patients with primary epithelial neoplasms of the lacrimal gland, its surgical treatment, and histopathological findings. METHODS: Retrospective evaluation of files from patients with primary epithelial neoplasms of the lacrimal gland in the period from 1997 to 2007. All patients with primary epithelial tumors of the lacrimal gland were included in this study. Data on gender, age, clinical features, surgical treatment, histopathological findings and follow-up were collected. The slides with histological sections of the tumors were reviewed by the same pathologist. RESULTS: During the study period, there were 12 patients, 5 (41.7 percent) with benign tumors, all pleomorphic adenomas (benign mixed tumor) and 7 (58.3 percent) with malignant neoplasms, thus distributed: four cases of adenoid cystic carcinoma, two of mucoepidermoid carcinoma and one carcinoma expleomorphic adenoma. Globally, patients mean age was 54.1 years-old (ranging from 14 to 70 years-old), with mean age of 52.4 years-old (ranging from 14 to 65 years-old) for benign neoplasms, and 55.3years-old for malignant neoplasms (ranging from 26 to 70 years-old). Clinical follow-up information, ranging from 2 to 10 years-old, was available for all patients. Three patients developed distant metastasis and died of disease. CONCLUSIONS: The most frequent primary epithelial neoplasms of the lacrimal gland were pleomorphic adenoma and adenoid cystic carcinoma during the study period. Malignant tumors were more frequent than benign tumors. The histopathological diagnosis and the disease initial stage can play a significant role in patient's survival.

OBJETIVO: Descrever e analisar as características de uma série de casos de portadores de neoplasias epiteliais primárias da glândula lacrimal, o tratamento cirúrgico, assim como os achados histopatológicos. MÉTODOS: Avaliação retrospectiva dos arquivos de pacientes com neoplasias epiteliais primárias da glândula lacrimal, no período de 1997 até 2007. Todos os pacientes com tumores epiteliais primários da glândula lacrimal foram incluídos neste estudo. Foram analisados os dados sobre sexo, idade, características clínicas, tratamento cirúrgico, achados histopatológicos e seguimento dos pacientes. As lâminas com secções histológicas dos tumores foram revisadas pelo mesmo patologista. RESULTADOS: No período do estudo, foram encontrados 12 pacientes, sendo 5 (41,7 por cento) portadores de tumores benignos, todos adenomas pleomórficos (tumor benigno misto), e 7 (58,3 por cento) com neoplasias malignas, assim distribuídos: quatro casos de carcinoma adenóide cístico, dois de carcinoma mucoepidermóide e um de carcinoma ex-adenoma pleomórfico. Analisando-se de modo global, a idade média dos portadores foi de 54,1 anos (variando de 14 a 70 anos); com média de idade de 52,4 anos (variando de 14 a 65 anos) para neoplasias benignas, e 55,3 para neoplasias malignas (variando de 26 a 70 anos). Informações do seguimento, variando de 2 a 10 anos, estavam disponíveis para todos os pacientes. Três pacientes desenvolveram metástases distantes e morreram devido à doença. CONCLUSÕES: A maioria das neoplasias epiteliais primárias da glândula lacrimal foi o adenoma pleomórfico e o carcinoma adenóide cístico no período de estudo. Os tumores malignos foram mais frequentes que os benignos. O diagnóstico histopatológico e o estadiamento inicial da doença podem desempenhar uma papel significante na sobrevida do paciente.

Management of Dermoid Tumor in the Medial Canthal Area

Dermoid tumors in the medial canthal area are rare, but when present they commonly adhere to the lacrimal canaliculi. Three patients presented with a mass in the medial canthal area. The authors performed excisional biopsies, and the masses were diagnosed as dermoid tumors. In two patients, canalicular lacerations were found after mass excision, which suggested that the masses had been firmly adherent to the lacrimal canaliculi. The lacerated canaliculi were repaired after bicanalicular silicone intubation. In the remaining patient, lacrimal silicone intubation was performed at the beginning of surgery, and the mass was successfully dissected from the canaliculi, leaving them intact. Excision of dermoid tumors in the medial canthal area requires careful dissection to avoid canalicular laceration. Bicanalicular silicone intubation at the beginning of surgery is helpful for the identification of the canaliculi and for the prevention of canalicular laceration during dermoid tumor excision.

The Clinical Usefulness of 18F-FDG PET/CT for the Evaluation of Lymph Node Metastasis in Periorbital Malignancies

OBJECTIVE: The aim of this study was to assess the clinical role of 18F-FDG PET/CT for the evaluation of lymph node metastasis in periorbital malignancies, compared with CT alone. MATERIALS AND METHODS: We analyzed eighteen PET/CT and CT scans in 15 patients with biopsy-proven periorbital malignancies. We compared the diagnostic capabilities of PET/CT and CT with regard to nodal metastasis by level-by-level analysis and by N staging prediction. The reference standards were surgical pathology (n = 7) from dissected lymph node specimens and the results from radiological follow-up (n = 11, mean 20.5 months; range 10-52 months). Moreover, any changes in patient care as prompted by PET/CT were recorded and compared with treatment planning for CT alone. RESULTS: PET/CT had a sensitivity of 100%, while CT had a sensitivity of 57% (p = 0.03) for nodal metastasis by level-by-level analysis. PET/CT had a specificity of 97%, positive predictive value of 93%, negative predictive value of 100%, and diagnostic accuracy of 98%, while the CT values for these same parameters were 97%, 89%, 82%, and 84%, respectively. PET/CT correctly predicted N staging with an accuracy of 100%, while CT was only 83% accurate (p = 0.01). Regarding the impact on patient care, the extent of surgery for regional lymph nodes and the treatment decision were modified by PET/CT in 39% of patients. CONCLUSION: PET/CT could provide useful information in the management of regional lymph node metastases in patients with periorbital malignancies.

Osteoma do etmóide com invasão orbitária: relato de três casos e revisão da literatura / Ethmoid sinus osteoma with orbital invasion: report of three cases and literature

São apresentados três raros casos de osteoma do etmóide, com extensão para o quadrante medial da órbita e que apresentavam algumas particularidades que os tornavam ainda mais inusitados, como terem acontecido em mulheres, em faixa etária não usual e com queixa de epífora. Os achados radiográficos foram típicos da afecção e os casos foram operados, com resolução do problema.

We report three rare cases of ethmoid osteoma extending to the medial quadrant of the orbit that had singular particularities, such as occurring in women, at an unusual age group, and complaint of epiphora. The radiographic images were typical of this condition. Patients were submitted to surgery with complete resolution of the disease.

Disappearance of primitive basaloid cells after external irradiation in adenoid cystic carcinoma of the lacrimal gland.

OBJECTIVE: To study the histopathological findings of basaloid adenoid cystic carcinoma after high dose orbital irradiation. MATERIAL AND METHOD: The histopathology study was done in a 38-year-old Thai female patient who had basaloid adenoid cystic carcinoma of the right lacrimal gland confirmed by incisional biopsy. After a 60-Gy external irradiation, the mass decreased in size and finally could not be palpated, but subsequent orbital imaging still showed infiltrative soft tissue mass at the right lacrimal gland with bony defects at posterosuperior and lateral walls without extraorbital extension. Orbital exenteration and lateral orbitectomy was performed. The specimen was submitted for histopathological examination. RESULTS: Microscopic examination revealed periglandular infiltration with chronic inflammation of the palpebral lobe of lacrimal gland. The orbital lobe was extensively replaced with fibrous tissue. There was only a small area of cribiform pattern of the adenoid cystic carcinoma left; no basaloid pattern was seen in the tissue. CONCLUSION: The primitive cells (basaloid pattern) disappear after orbital irradiation for adenoid cystic carcinoma of the lacrimal gland. Replacement with fibrous tissue is seen.

Oncocytoma of the caruncle.

A rare case of oncocytoma of the caruncle in a 75-year-old female is reported. Clinical differential diagnosis and histopathologic features are discussed.

Lesiones de la glándula lagrimal / Lagrimal gland lesions

Se analizaron retrospectivamente, 104 lesiones de la glándula lagrimal en un período de 40 años comprendido entre 1957 y 1996. Del total, 21 casos correspondieron a neoplasias malignas, todas ellas de origen epitelial; 38 casos a quistes (dacriops), 16 casos a tumores benignos y 28 casos a glándula lagrimal normal. Sólo se encontró un caso de proceso inflamatorio o dicrioadenitis crónica

Fine needle aspiration cytology of ophthalmic lesions.

Forty cases of ophthalmic lesions were subjected to fine needle aspiration cytology for pre-operative diagnosis which included retinoblastomas (5 cases), inflammatory and neoplastic lesions of the eyelid and lacrimal glands (29 cases) and extra-ocular orbital lesions (6 cases) inclusive of 3 cases of rhabdomyosarcomas. The lesions were distributed in both the eyes almost in equal proportions. Histological confirmation was obtained in 85% of cases; false positive in only one case with malignant neoplasm of the eyelid and false negative also in only one case with inflammatory pseudotumour which were, histologically, proved to be adenoma of tarsal gland and lymphocytic lymphoma respectively.

Cyst of accessory lacrimal gland

When a patient is presented with a subconjunctival cyst, it is not only hard to reveal its true nature clinically but also easy to rupture during excision. We experienced cases with cysts of the accessory lacrimal gland in two patients with subconjunctival cysts. They had lid swelling at initial presentation and underwent surgical excision of subconjunctival cysts located in superior portion of the upper tarsal plate. The lining of these cysts composed of ductal epithelia. Biochemical analyses for serum and cystic fluid were performed in one case, in which was found high Ig A titer in the cystic fluid. These cysts seemed to originate from the duct of Wolfring's accessory lacrimal gland, considering their anatomic locations and pathologic findings. Complete removal of the cyst is important, because recurrences have been reported in cases of incomplete removal or simple aspiration.

Unusual presentation of lacrimal gland tumours.

The diagnosis of orbital tumour is a challenge to the ophthalmologist. The aim of this paper is to highlight the unusual presentation of lacrimal gland tumours.