RESUMEN
PURPOSE: Botulinum toxin (BoNTA) lacrimal gland injections are one treatment option for managing epiphora. Outcomes for epiphora in existing studies are subjective and lack grading. This study utilized validated grading instruments to evaluate the efficacy of BoNTA lacrimal gland injections. METHODS: Prospective study evaluating the TEARS score and quality of life in patients receiving incobotulinum toxin (Xeomin) lacrimal gland injections for epiphora. Epiphora was graded using the TEARS score. Quality of life was evaluated using the watery eye quality of life score. Etiology, benefit duration, complications and additional injections were recorded. RESULTS: Of the 28 patients, (median age 67.5 years), 19 (68%) had facial palsy gustatory epiphora (GE). Median effect duration was 13 weeks. Improvements in tearing frequency (T), clinical effects (E), and activity limitation (A) were seen in 57%, 57%, and 71% of patients, respectively. Median watery eye quality of life score improved from 42.4/100 ± 10.1 to 10.6 ± 15.5 (p < 0.001). Alongside crocodile tears, reflex tearing R-scores improved in 68% of patients with GE vs. 11% with non-GE causes (p = 0.004618). In total 89% of patients with GE and 56% with non-GE requested repeat injections. Self-reported percentage improvement ranged from 0% to 100% (median 60%, interquartile range 10%-76.25%). CONCLUSIONS: This is the first study reporting the outcomes of BoNTA lacrimal gland injections using validated TEARS and watery eye quality of life scores. Clinical and quality of life improvements are seen in most patients. Patients with GE achieve an additional improvement in dry eye-related reflex tearing alongside crocodile tears which both improve with BoNTA. TEARS data supports using BoNTA in these patients and is a useful validated grading instrument for epiphora.
Asunto(s)
Toxinas Botulínicas Tipo A , Enfermedades del Aparato Lagrimal , Aparato Lagrimal , Fármacos Neuromusculares , Humanos , Anciano , Estudios Prospectivos , Calidad de Vida , Fármacos Neuromusculares/uso terapéutico , Toxinas Botulínicas Tipo A/uso terapéutico , Lágrimas , Enfermedades del Aparato Lagrimal/diagnóstico , Enfermedades del Aparato Lagrimal/tratamiento farmacológico , Enfermedades del Aparato Lagrimal/etiologíaRESUMEN
BACKGROUND: Lacrimal gland adenoid cystic carcinoma (LGACC) has historically been associated with a poor prognosis even with localized disease, with a survival of 56% at 5 years. In 1988, we treated the first patient with neoadjuvant intra-arterial cytoreductive chemotherapy (IACC). Since then, we have used this protocol as the standard approach. We aim to analyze the outcomes of patients with LGACC treated with the protocol and compare them to a population-based cohort to assess if IACC can improve survival. METHODS: We prospectively assessed all non-metastatic patients with LGACC treated with IACC at a single institution between 1988 and 2021. For a comparison group, we identified all non-metastatic patients with LGACC treated with excision from the Surveillance, Epidemiology, and End Results (SEER) registry. We calculated disease-specific survival using the Kaplan-Meier and Cox proportional-hazards modeling methods. RESULTS: Thirty-five non-metastatic patients with LGACC treated with IACC were identified at a single institution, and 64 patients with non-metastatic LGACC treated with excision were identified in the SEER database. The 5- and 10-year disease-specific survival rates for patients treated with IACC were 84% (95%CI 71-97) and 76% (95%CI 60-92), respectively. While the 5- and 10-year disease-specific survival rates for the population-based cohort were 72% (95%CI 62-82) and 46% (95%CI 32-60). The survival analysis favored IACC, with a 60% lower risk of death (HR: 0.4; 95%CI 0.2-0.9). CONCLUSION: IACC improves disease-specific survival in comparison to a population-based cohort treated with excision. Additional patients treated with IACC at multiple institutions are required to provide further external validity.
Asunto(s)
Carcinoma Adenoide Quístico , Neoplasias del Ojo , Neoplasias de Cabeza y Cuello , Enfermedades del Aparato Lagrimal , Aparato Lagrimal , Humanos , Carcinoma Adenoide Quístico/tratamiento farmacológico , Aparato Lagrimal/patología , Terapia Neoadyuvante , Procedimientos Quirúrgicos de Citorreducción , Estudios Retrospectivos , Enfermedades del Aparato Lagrimal/tratamiento farmacológico , Enfermedades del Aparato Lagrimal/patología , Neoplasias del Ojo/tratamiento farmacológico , Neoplasias del Ojo/patologíaRESUMEN
PURPOSE: To investigate the effect of botulinum toxin (BTX) and partial orbital lobe dacryoadenectomy (POLD) on the main lacrimal gland functions using the direct assessment of its secretory activity. METHODS: A pilot study involving 14 subjects (mean age, 42.16 years) with proximal bicanalicular block (n = 9) or complete punctal effacement (n = 5) received either intraglandular 2.5 units of BTX (n = 7) or underwent the POLD procedure (n = 7). Dynamic assessment of tear secretion from the palpebral lobes was performed as per standard protocols. The parameters studied included Schirmer I, changes in the glandular outflow, and the neurosensory secretory lag. RESULTS: Schirmer I values showed a mean reduction of 5.25 mm (range, 2-8 mm) following BTX injection, and mean tear flow decreased from 1.46 µl/min to 1.11 µl/min (P = 0.15). The POLD group showed a significant mean reduction of 0.45 µl/min in mean tear flow at 1 month (P = 0.03) and 0.32 µl/min at 3 months of follow-up (P = 0.20). However, there was no statistically significant change of the Schirmer values in the POLD group at 3 months. CONCLUSION: Direct assessment of lacrimal gland function can be used to monitor changes following the gland therapies. Although POLD shows a significant reduction in short term, significant surgical improvisation would be needed for it to be effective in long term. A larger study is ongoing to further our understanding.
Asunto(s)
Toxinas Botulínicas Tipo A , Enfermedades del Aparato Lagrimal , Aparato Lagrimal , Humanos , Adulto , Aparato Lagrimal/cirugía , Proyectos Piloto , Toxinas Botulínicas Tipo A/uso terapéutico , Enfermedades del Aparato Lagrimal/diagnóstico por imagen , Enfermedades del Aparato Lagrimal/tratamiento farmacológico , Enfermedades del Aparato Lagrimal/cirugía , LágrimasRESUMEN
Background: Hemolacria or the presence of blood in tears is a rare condition, and there are only a few cases reported in the literature. Hemolacria is associated with multiple underlying diseases, including vicarious menstruation due to extragenital endometriosis. Case report: We present a 26-year-old woman with hemolacria and abdominal pain related to her menstrual cycle. The patient was diagnosed with bilateral ovarian endometriomas. After ruling out other possible causes of hemolacria, a progestin-only treatment was applied, with improvement of the abdominal pain and complete remission of hemolacria. Conclusions: When faced with hemolacria, a thorough anamnesis and physical examination must be performed, sometimes involving more than one specialist to reach a diagnosis. Considering hemolacria is a sign of a subjacent pathology, its treatment should be specific one for the disease in each case.
Antecedentes: La hemolacria o presencia de sangre en las lágrimas es una afección poco frecuente y sólo hay unos pocos casos descritos en la literatura. La hemolacria se asocia a múltiples enfermedades subyacentes, incluida la menstruación vicaria debida a endometriosis extragenital. Caso clínico: Presentamos a una mujer de 26 años con hemolacria y dolor abdominal relacionado con su ciclo menstrual. La paciente fue diagnosticada de endometriomas ováricos bilaterales. Tras descartar otras posibles causas de hemolacria, se aplicó un tratamiento sólo con progestágenos, con mejoría del dolor abdominal y remisión completa de la hemolacria. Conclusiones: Ante una hemolacria se debe realizar una anamnesis y exploración física minuciosa, en la que a veces interviene más de un especialista para llegar al diagnóstico. Teniendo en cuenta que la hemolacria es signo de una patología subyacente, su tratamiento debe ser el específico para la enfermedad en cada caso.
Asunto(s)
Humanos , Femenino , Adulto , Endometriosis/diagnóstico , Enfermedades del Aparato Lagrimal/etiología , Progestinas/uso terapéutico , Lágrimas , Endometriosis/tratamiento farmacológico , Hemorragia , Enfermedades del Aparato Lagrimal/tratamiento farmacológicoRESUMEN
Objectives: To evaluate the demographic characteristics, clinical presentation, microbiologic profile, and treatment results of patients with primary canaliculitis. Materials and Methods: Patients diagnosed and treated for primary canaliculitis between May 2014 and May 2021 were analyzed retrospectively. Results: There were 26 patients with primary canaliculitis, including 17 females (65.4%) and 9 males (34.6%) with a mean age of 50.6±16.4 years (range: 9-80 years). Canaliculitis affected the right eye in 11 patients, the left eye in 13 patients, and bilateral involvement was seen in 2 patients. Inferior canaliculus involvement was more frequent (73%). The most common complaint was epiphora (46.1%). Five patients (19.2%) were wrongly diagnosed as chronic conjunctivitis. The time interval between the beginning of symptoms and canaliculitis diagnosis was 18.2±14.3 months (range: 1-60 months). Canaliculotomy and curettage of canalicular content with dacryolith removal were performed in 23 patients. After surgery, antibiotic irrigation of the canaliculus was added to the treatment regimen in 12 of these 23 patients. Intracanalicular antibiotic therapy was administered to the remaining 3 patients. The most cultured organism was Actinomyces (6 patients). Gemella (1 patient), Porphyromonas (1 patient), Candida parapsilosis (1 patient), Citrobacter koseri (1 patient) were also grown in culture. The follow-up time of patients was 26.2±23.7 months (range: 6-83 months). All symptoms and findings resolved in all patients in one month. In two patients, recurrence occurred at 4 and 16 months after surgical treatment. With appropriate treatment, no further recurrence was seen in either patient over 24-month follow-up. One patient presented with iatrogenic canaliculus blockage during follow-up. Conclusion: Primary canaliculitis is often overlooked and can be misdiagnosed. The most common symptom was epiphora. All patients with epiphora and chronic conjunctivitis should be examined carefully for canaliculitis.
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Canaliculitis , Conjuntivitis , Dacriocistitis , Enfermedades del Aparato Lagrimal , Masculino , Femenino , Humanos , Adulto , Persona de Mediana Edad , Anciano , Canaliculitis/diagnóstico , Canaliculitis/tratamiento farmacológico , Canaliculitis/cirugía , Dacriocistitis/diagnóstico , Dacriocistitis/cirugía , Dacriocistitis/microbiología , Estudios de Seguimiento , Estudios Retrospectivos , Antibacterianos/uso terapéutico , Enfermedades del Aparato Lagrimal/tratamiento farmacológicoAsunto(s)
Toxinas Botulínicas Tipo A , Toxinas Botulínicas , Enfermedades del Aparato Lagrimal , Aparato Lagrimal , Obstrucción del Conducto Lagrimal , Humanos , Enfermedades del Aparato Lagrimal/tratamiento farmacológico , Toxinas Botulínicas/uso terapéutico , Toxinas Botulínicas Tipo A/uso terapéuticoRESUMEN
Crocodile tear syndrome (CTS) is a late complication of facial nerve palsy characterized by unilateral lacrimation in response to gustatory stimulation. We present 2 cases of patients diagnosed with CTS after recovering from unilateral idiopathic facial nerve palsy. Both patients underwent transconjunctival lacrimal gland incobotulinumtoxinA injection, with doses of 5-16 units. The patients were seen in clinic for post-treatment follow-up at 2 weeks, 3 months, and 6 months. Outcomes were measured by treatment efficacy and adverse drug effects. Following treatment, both patients reported resolution of gustatory lacrimation. The patient treated with 16 U experienced transient ptosis and diplopia following injection, whereas the patient treated with 5-7.5 U experienced no adverse effects.
Asunto(s)
Enfermedades del Sistema Nervioso Autónomo , Parálisis de Bell , Blefaroptosis , Toxinas Botulínicas Tipo A , Trastornos Congénitos de Denervación Craneal , Parálisis Facial , Anomalías Maxilomandibulares , Enfermedades del Aparato Lagrimal , Aparato Lagrimal , Fármacos Neuromusculares , Reflejo Anormal , Toxinas Biológicas , Humanos , Aparato Lagrimal/inervación , Toxinas Botulínicas Tipo A/farmacología , Toxinas Botulínicas Tipo A/uso terapéutico , Fármacos Neuromusculares/farmacología , Fármacos Neuromusculares/uso terapéutico , Enfermedades del Aparato Lagrimal/diagnóstico , Enfermedades del Aparato Lagrimal/tratamiento farmacológico , Parálisis Facial/complicaciones , Parálisis Facial/tratamiento farmacológico , Parálisis de Bell/complicaciones , Parálisis de Bell/tratamiento farmacológico , Enfermedades del Sistema Nervioso Autónomo/complicaciones , Enfermedades del Sistema Nervioso Autónomo/tratamiento farmacológico , Toxinas Biológicas/farmacología , Toxinas Biológicas/uso terapéuticoRESUMEN
Lacrimal gland injection of botulinum toxin type A inhibits the secretion of tears. As a new method to treat or alleviate the symptom of tears or epiphora, it has the characteristics of simple operation, definite curative effect, repeatable treatment and no irreversible complications. It provides an optional treatment scheme for many patients with refractory tears or epiphora. This article reviews the pharmacological characteristics of botulinum toxin type A, the mechanism of inhibiting tear secretion, the method and dose of lacrimal gland injection, indications and contraindications, clinical efficacy evaluation, complications, existing problems to be solved and prospects for reference.
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Toxinas Botulínicas Tipo A , Enfermedades del Aparato Lagrimal , Aparato Lagrimal , Toxinas Botulínicas Tipo A/farmacología , Toxinas Botulínicas Tipo A/uso terapéutico , Humanos , Enfermedades del Aparato Lagrimal/tratamiento farmacológico , Lágrimas , Resultado del TratamientoRESUMEN
PURPOSE: To report the therapeutic efficacy of integrating neoadjuvant chemotherapy with conventional bimodal therapies for lacrimal gland adenoid cystic carcinoma by providing an additional 8 years of follow-up data on the same cohort of patients whose cumulative 10-year disease-free survival outcomes were reported in 2013. DESIGN: Non-randomized, retrospective, interventional case series. METHODS: Nineteen consecutive patients treated with neoadjuvant intra-arterial cytoreductive chemotherapy (IACC), orbital exenteration, chemoradiotherapy, and adjuvant intravenous chemotherapy at a single institution were included. Analyses were undertaken of locoregional recurrences and distant metastases, disease-free survival time, TNM tumor stage at presentation, response to IACC, and prognostic impact of positive resection margins. The main outcome measures were overall survival, disease-free survival, disease relapse, positive tumor resection margins, and tumor stage at presentation. RESULTS: Eight patients with an intact lacrimal artery (group 1), 7 with AJCC stage T4a-c, had significantly better overall survival (87.5% versus 14.3% at 15 years), disease-specific mortality, and recurrences (all < .001, log-rank test) than prior conventionally treated patients from the Bascom Palmer Eye Institute. Group 1 was superior to group 2, patients lacking an intact lacrimal artery, concerning overall survival (P = .042) and recurrence (P = .017), but with no significant difference in disease-specific mortality (P = .23). Group 2 was associated with a significantly lower cause-specific mortality than the institutional comparator group (P = .039). Prior tumor resection with lateral wall osteotomy and failure to adhere to all protocol elements were adverse prognostic factors for suboptimal outcomes. Positive tumor margins increased the risk of all-cause mortality 4.1 times (P = .036, stratified Cox proportional hazards regression) and disease-specific mortality 8.0 times (P = .043, stratified Cox proportional hazards regression) than a patient with negative margins. CONCLUSIONS: Extended follow-up supplemented with AJCC staging data supports neoadjuvant IACC as an integral component of a trimodal treatment strategy in patients with an intact lacrimal artery. Protocol elements implemented as designed appear to have improved overall survival and decreased disease relapse in this cohort. This extended long-term IACC dataset suggests that a critical bar of at least 15 years of follow-up is appropriate for assessing the efficacy of current conventional and future globe-sparing bimodal therapies.
Asunto(s)
Carcinoma Adenoide Quístico , Neoplasias del Ojo , Neoplasias de Cabeza y Cuello , Enfermedades del Aparato Lagrimal , Aparato Lagrimal , Carcinoma Adenoide Quístico/tratamiento farmacológico , Procedimientos Quirúrgicos de Citorreducción , Neoplasias del Ojo/tratamiento farmacológico , Neoplasias del Ojo/patología , Estudios de Seguimiento , Humanos , Aparato Lagrimal/patología , Enfermedades del Aparato Lagrimal/tratamiento farmacológico , Enfermedades del Aparato Lagrimal/patología , Márgenes de Escisión , Terapia Neoadyuvante , Recurrencia Local de Neoplasia , Estudios RetrospectivosRESUMEN
The purpose of this study is to identify the location of the orbital part of the lacrimal gland using external landmarks to facilitate effective botulinum toxin A (BTX-A) injections for epiphora treatment. Dissections were performed on 45 hemifaces from 27 cadavers. The length, anterior protrusion, and thickness of the orbital part of the lacrimal gland were measured directly. The midpoint of the line running horizontal through the medial and lateral canthus was used as the horizontal reference point. Vertical lines perpendicular to the midpoint of the horizontal reference line were the vertical reference lines. The angles from the horizontal reference line to the lacrimal gland center and to the frontal tubercle were also measured. The length and thickness of the lacrimal gland were 12.8 and 2.4 mm, respectively. The lacrimal gland had an anterior protrusion of 4.1 mm from the superolateral orbital margin. The superior and inferior margins of the lacrimal gland were located 35.7° and 15.8° from the two reference lines, respectively. The angle from the horizontal reference line to the lacrimal gland center was 35.1°, which was similar to that to the frontal tubercle (34.4°). The orbital part of the lacrimal gland was more inferior in elderly subjects and the angle between the horizontal reference line and its center was 35.1°, which was similar to the location of the frontal tubercle. The location of this tubercle can help clinicians to perform BTX-A injections into the lacrimal gland, thereby enhancing the effectiveness of epiphora treatment.
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Toxinas Botulínicas Tipo A , Enfermedades del Aparato Lagrimal , Aparato Lagrimal , Anciano , Humanos , Inyecciones , Enfermedades del Aparato Lagrimal/tratamiento farmacológico , ÓrbitaRESUMEN
Purpose: To determine the efficacy and safety of botulinum toxin injection into the lacrimal gland as a symptomatic treatment of crocodile tear syndrome (CTS). Methods: Our study included six patients of unilateral gustatory hyper lacrimation following either an episode of facial paralysis or post trauma or any related surgery that posed a risk of damaging the facial nerve. Detailed history regarding previous trauma, duration of facial paralysis, previous significant surgery, and duration of steroid use following facial paralysis was noted. Schirmer's test was done at baseline and 3 months follow-up. Patients' consent was taken prior to treatment with botulinum toxin injection in the lacrimal gland. Repeat injection of 5 U botulinum toxin (type A) was administered into the lacrimal gland of all patients transconjunctivally within an interval of 1 week. All patients were followed up with Schirmer's test at 6 weeks and 3 months. Any complications during treatment were recorded. Results: All six patients showed complete or partial disappearance of reflex lacrimation while chewing following botulinum injection measured by a significant reduction in Schirmer's value. When comparing Schirmer test values before (27.8 ± 3.58 mm) and after (11.6 ± 2.28 mm) BTX-A injection, the differences observed (P = 0.002) were statistically significant (P < 0.05). Only two patients developed mild transitory ptosis. No other complications were noted. Conclusion: Transconjunctival botulinum toxin injection into the lacrimal gland is an effective and safe method to decrease reflex lacrimation during eating or chewing in CTS or gustatory hyper-lacrimation syndrome.
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Toxinas Botulínicas Tipo A , Parálisis Facial , Enfermedades del Aparato Lagrimal , Aparato Lagrimal , Blefaroptosis , Parálisis Facial/complicaciones , Cardiopatías Congénitas , Humanos , Anomalías Maxilomandibulares , Enfermedades del Aparato Lagrimal/complicaciones , Enfermedades del Aparato Lagrimal/diagnóstico , Enfermedades del Aparato Lagrimal/tratamiento farmacológico , Enfermedades del Sistema Nervioso , Reflejo Anormal , LágrimasRESUMEN
Adenoid cystic carcinoma of the lacrimal gland is an aggressive, malignant epithelial neoplasm. We report the case of a 30-year-old male with lacrimal gland adenoid cystic carcinoma treated with neoadjuvant intra-arterial chemotherapy through the internal carotid artery, followed by orbital exenteration and chemoradiation. Treatment response was evaluated using a novel combination of pre- and posttreatment genome sequencing coupled with immunohistochemical evaluation, which showed diffuse tumor apoptosis. A posttreatment decrease in variant allele frequency of the NOTCH1 mutation, and robust tumor cytoreduction on imaging, supports exploration of NOTCH1 analysis as a potential marker of cisplatin sensitivity. The use of genome sequencing and immunohistochemical evaluation could provide a more targeted therapeutic assessment of neoadjuvant intra-arterial chemotherapy in the management of lacrimal gland adenoid cystic carcinoma.
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Carcinoma Adenoide Quístico , Neoplasias del Ojo , Enfermedades del Aparato Lagrimal , Aparato Lagrimal , Adulto , Carcinoma Adenoide Quístico/diagnóstico , Carcinoma Adenoide Quístico/tratamiento farmacológico , Carcinoma Adenoide Quístico/genética , Procedimientos Quirúrgicos de Citorreducción , Neoplasias del Ojo/diagnóstico , Neoplasias del Ojo/tratamiento farmacológico , Neoplasias del Ojo/genética , Humanos , Aparato Lagrimal/patología , Enfermedades del Aparato Lagrimal/diagnóstico , Enfermedades del Aparato Lagrimal/tratamiento farmacológico , Enfermedades del Aparato Lagrimal/patología , MasculinoRESUMEN
Neoadjuvant intra-arterial cytoreductive chemotherapy is used for the treatment of lacrimal gland adenoid cystic carcinomas (ACC) to improve outcomes in this condition with an otherwise dismal prognosis. We share our experience in the management of an advanced case of ACC using a novel, highly targeted intra-arterial cytoreductive chemotherapy delivery technique involving both the internal and external carotid circulation, with an attempt to correlate the effect histologically. Refinement of the chemotherapy delivery using the tumor's vascular anatomy and appropriate blood vessel selection may lead to future globe sparing procedures without compromising survival.
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Carcinoma Adenoide Quístico , Neoplasias del Ojo , Enfermedades del Aparato Lagrimal , Aparato Lagrimal , Biomarcadores de Tumor , Carcinoma Adenoide Quístico/tratamiento farmacológico , Neoplasias del Ojo/tratamiento farmacológico , Humanos , Enfermedades del Aparato Lagrimal/diagnóstico , Enfermedades del Aparato Lagrimal/tratamiento farmacológico , Terapia NeoadyuvanteRESUMEN
PURPOSE: To identify the frequency of intraepithelial (Pagetoid) spread beyond the ocular surface-namely beyond conjunctiva and cornea-in patients undergoing orbital exenteration for advanced periocular Sebaceous carcinoma (SC). DESIGN: A retrospective, noncomparative observational case series. SUBJECTS: Patients undergoing orbital exenteration for biopsy-proven SC, at Moorfields Eye Hospital between 1997 and 2013. METHODS: Review of clinical records and histological specimens, with particularly reference to involvement of conjunctiva and the extent of Pagetoid infiltration beyond the examinable ocular surface-here termed "hidden" disease. MAIN OUTCOME MEASURES: Histological evidence of intraepithelial SC within the lacrimal sac or lacrimal gland. RESULTS: Twenty-nine patients had clinical data and histological specimens adequate for review. Seventeen (59%) did not have a discrete mass (clinically or histologically) and, on clinical examination, were thought to only have extensive intraepithelial carcinoma; foci of microscopic invasion were, however, detected histologically in 11/17 (65%) of these specimens. Moreover, the in situ carcinoma was found to have invaded far in lacrimal gland ductules in 1/17 patients, in the lacrimal sac (in 2 patients; 12%) or in both the gland and sac (in 2 patients); these 5/17 (29%) cases all showed extensive poorly differentiated intraepithelial SC. Of the 12 other patients who had both Pagetoid spread and a clinically evident nodule, 3 had histological evidence of "hidden" disease. CONCLUSION: Although-due to their being operated in the era prior to the accepted usage of topical therapy for this condition-some of these exenterations might have had particularly advanced in situ SC, over a quarter of patients with periocular SC warranting orbital exenteration show "hidden" intraepithelial tumor within the lacrimal gland and sac. This important finding might significantly reduce the efficacy (particularly in the lacrimal gland) of the various topical therapies used for in situ SC of the ocular surface, and it also emphasizes the importance of excising both the lacrimal gland and sac in all orbital exenterations for this particular tumor.
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Carcinoma in Situ , Carcinoma , Neoplasias del Ojo , Enfermedades del Aparato Lagrimal , Aparato Lagrimal , Neoplasias de las Glándulas Sebáceas , Carcinoma in Situ/tratamiento farmacológico , Neoplasias del Ojo/tratamiento farmacológico , Humanos , Enfermedades del Aparato Lagrimal/tratamiento farmacológico , Estudios Retrospectivos , Neoplasias de las Glándulas Sebáceas/tratamiento farmacológicoRESUMEN
PURPOSE: Ocular manifestations of immunoglobulin G4 (IgG4)-related disease are common in children although remain ill-defined because of the disease's rarity. We describe a pediatric case of IgG4-related orbital disease (IgG4-ROD) who presented with persistent conjunctival infiltration before developing lacrimal gland enlargement 3 years later. METHODS: This was a case report. RESULTS: An 8-year-old girl developed forniceal salmon-patch-like conjunctival lesions in her left eye that were refractory to topical corticosteroids. Investigations, including an orbital MRI and 2 conjunctival biopsies, were negative for lymphoma. She was treated with topical corticosteroids and then nonsteroidal antiinflammatory drops. The lesions decreased mildly, and no new lesion emerged. After 3 years, the patient developed a ptosis, new salmon-patch conjunctival lesions, and papillae. Vision deteriorated to 20/80 because of severe punctate epithelial erosions in the left eye, and the Schirmer test was significantly reduced. A repeat MRI revealed an enlarged left lacrimal gland. A biopsy was performed and was compatible with IgG4-ROD. An elevated IgG4 level of 4.61 g/L was also found. The patient was successfully treated with oral prednisone but flared on tapering the dosage. Rituximab was therefore initiated with excellent clinical response, and prednisone was discontinued. Vision returned to 20/20 after aggressive lubrification, punctal plugs, and autologous serum eye drops. Tear function came back to normal, and local treatments were stopped. CONCLUSIONS: This case describes a pediatric case of IgG4-ROD presenting initially with conjunctival follicular reaction, later developing lacrimal gland involvement. Therefore, it is important to consider IgG4-ROD in chronic atypical follicular conjunctival lesions in children, even in the absence of orbital disease.
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Enfermedades de la Conjuntiva/diagnóstico , Enfermedad Relacionada con Inmunoglobulina G4/diagnóstico , Enfermedades del Aparato Lagrimal/diagnóstico , Enfermedades Orbitales/diagnóstico , Inhibidores de la Angiogénesis/uso terapéutico , Niño , Enfermedades de la Conjuntiva/tratamiento farmacológico , Femenino , Glucocorticoides/uso terapéutico , Humanos , Enfermedad Relacionada con Inmunoglobulina G4/tratamiento farmacológico , Enfermedades del Aparato Lagrimal/tratamiento farmacológico , Enfermedades Orbitales/tratamiento farmacológico , Prednisolona/uso terapéutico , Rituximab/uso terapéutico , Factor A de Crecimiento Endotelial Vascular/antagonistas & inhibidoresRESUMEN
Primary melanoma of the lacrimal sac is a rare entity, with high mortality and a propensity for recurrence. This report details a patient with widely metastatic melanoma discovered after biopsy of abnormal lacrimal sac tissue during routine dacryocystorhinostomy. The patient subsequently underwent local excision and treatment with pembrolizumab. At the time of this writing, it has been 24 months since the original diagnosis with resolution of his lacrimal and orbital lesions and improvement in all metastatic lesions. This case highlights the growing use of cancer genomics and immunotherapeutic agents in orbital aspects of oncology and reinforces the role of a multidisciplinary approach in the treatment of such diseases.
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Dacriocistorrinostomía , Enfermedades del Aparato Lagrimal , Melanoma , Conducto Nasolagrimal , Anticuerpos Monoclonales Humanizados , Humanos , Enfermedades del Aparato Lagrimal/diagnóstico , Enfermedades del Aparato Lagrimal/tratamiento farmacológico , Enfermedades del Aparato Lagrimal/cirugía , Melanoma/tratamiento farmacológico , Melanoma/cirugía , Recurrencia Local de NeoplasiaRESUMEN
PURPOSE: We have evaluated the potential radioprotective, antioxidant and anti-apoptotic effects of resveratrol (RSV) against high-dose radioactive iodine (RAI) therapy associated damage of the lacrimal glands by biochemical, histopathological and immunohistochemical methods. MATERIALS AND METHODS: Thirty Wistar-albino rats were randomly divided into three groups; the control group received no treatment or medication, the RAI group received RAI but no medication and the RSV group received oral RAI and intraperitoneal RSV. RSV was started at day one, before RAI administration, and continued for 8 days. Bilateral intraorbital (IG), extraorbital (EG), and Harderian (HG) lacrimal glands were evaluated in all rats for histopathological, immunohistochemical, tissue cytokine and oxidant and antioxidant level assessment. RESULTS: RSV group restored inflammation, fibrosis, vacuolization, change in nucleus characteristics, lipofuscin-like accumulation and cellular morphologic patterns were statistically significant in all lacrimal gland types, compared to the RAI group (p < .05 for all variables). Similarly, elevated Caspase-3 and TUNEL levels in the RAI group were significantly alleviated in the RSV group in all lacrimal gland types (p < .05 for all variables). RAI administration significantly elevated TNF-α, IL-6, NF-кb levels, and decreased IL-10 levels (p < .05 for all parameters) whereas TOS levels significantly increased and TAS levels were significantly decreased. However, RSV significantly diminished TNF-α, IL-6, IL-4, and NF-кb levels. Furthermore, RSV significantly decreased TOS and increased TAS levels (p < .05 for all variables). CONCLUSIONS: We conclude that with its anti-cancer effect as well as its antioxidant effect RSV has protected the histopathological pattern of the lacrimal glands from the damage, decreased inflammation in histopathologic assessments, and decreased tissue cytokine levels, apoptosis and DNA fragmentation on the lacrimal glands after RAI.
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Radioisótopos de Yodo/efectos adversos , Enfermedades del Aparato Lagrimal/tratamiento farmacológico , Aparato Lagrimal/patología , Traumatismos Experimentales por Radiación/tratamiento farmacológico , Resveratrol/farmacología , Animales , Antioxidantes/farmacología , Modelos Animales de Enfermedad , Femenino , Radioisótopos de Yodo/uso terapéutico , Aparato Lagrimal/metabolismo , Aparato Lagrimal/efectos de la radiación , Enfermedades del Aparato Lagrimal/diagnóstico , Enfermedades del Aparato Lagrimal/etiología , Estrés Oxidativo , Traumatismos Experimentales por Radiación/complicaciones , Traumatismos Experimentales por Radiación/diagnóstico , Ratas , Ratas WistarRESUMEN
PURPOSE: To assess whether exenteration specimens obtained after neoadjuvant intra-arterial cytoreductive chemotherapy (IACC) for adenoid cystic carcinoma of the lacrimal gland demonstrate significant ocular histopathologic alterations that might preclude future pursuit of globe-preserving therapy. METHODS: Retrospective histopathologic analysis of globes in IACC-treated exenteration specimens among the same cohort of patients whose survival outcomes have been reported. RESULTS: Twenty patients had specimens available. Nineteen globes revealed no abnormalities of the iris, ciliary body, lens, retinal pigment epithelium, choroid, or chorioretinal vasculature. Eighteen globes showed no optic nerve abnormalities. One globe from a patient who refused exenteration until adenoid cystic carcinoma recurrence supervened demonstrated optic nerve edema with a peripapillary hemorrhage and cotton wool spot, as well as hemorrhage and necrosis within an extraocular muscle. Eighteen globes showed no retinal abnormalities attributable to intra-arterial chemotherapy. Three globes showed incidental retinal findings: 2 globes contained 1 to 2 small peripheral retinal hemorrhages and 1 had a pigmented retinal hole. Seven demonstrated mild, chronic extraocular muscle inflammation, and 13 had unremarkable musculature. The single patient who received IACC via the internal carotid rather than the external carotid artery developed ophthalmic artery occlusion with orbital apex syndrome prior to exenteration, and diffuse necrosis and hemorrhage were evident histopathologically. CONCLUSIONS: Neoadjuvant IACC does not cause significant histopathologic damage to key ocular structures or compromise visual function in patients receiving intra-arterial chemotherapy through the external carotid artery. However, delivering chemotherapy through the internal carotid artery may result in visually significant thrombotic vascular events. The generally benign histopathological findings in these exenteration specimens support the concept of IACC delivery through the external carotid system as the cornerstone of a future globe-preserving strategy for lacrimal gland adenoid cystic carcinoma.
Asunto(s)
Carcinoma Adenoide Quístico , Neoplasias del Ojo , Enfermedades del Aparato Lagrimal , Aparato Lagrimal , Carcinoma Adenoide Quístico/tratamiento farmacológico , Procedimientos Quirúrgicos de Citorreducción , Neoplasias del Ojo/tratamiento farmacológico , Humanos , Enfermedades del Aparato Lagrimal/tratamiento farmacológico , Terapia Neoadyuvante , Recurrencia Local de Neoplasia , Órbita , Estudios RetrospectivosRESUMEN
Background: Cyclopentolate is not commonly used as mydriatic drug in veterinary medicine because of limited data on the local and systemic effects in animals. Aim: To determine the effects of topical 1% cyclopentolate hydrochloride on intraocular pressure (IOP), horizontal pupil diameter (HPD) and tear production in the cat and rabbit's eye during the first hour and up to 36 hours after treatment. Methods: One drop of 1% cyclopentolate hydrochloride was used in the left eye in 10 clinically and ophthalmologically healthy domestic cats and 10 rabbits. IOP and HPD were recorded every 5 minutes during the first hour, then every 2 hours during the following 12-hour period, and at 24 and 36 hours after application. Schirmer tear test (STT) was measured at 30 and 60 minute after treatment, then in same time points as IOP and HPD. Rebound tonometer (TonoVet®) was used to assess IOP, Jameson calliper to measure HPD and STT to determine the tear production. Results: 1% cyclopentolate increased IOP in cats, reaching a maximum (28.1 ± 5.4 mmHg) at T50 and in rabbits at T25 (16.7 ± 1.3 mmHg). Maximal mydriasis in cats was observed at T40 and lasted 24-36 hours, but in rabbits at T25, and returned to pre-treatment values at T10h-T12h. In cats, STT decreased in both eyes 30 minutes after treatment and remained lower throughout the 36-hour period. In rabbits, STT decreased in the treated eye 30 minutes after treatment, but all following STT measurements returned to normal pre-treatment levels. Conclusion: Study showed novel data about the effects of 1% cyclopentolate to IOP, HPD, STT in cats and rabbits. Cyclopentolate in cats caused mydriasis 20-40 minutes after the treatment by increasing IOP, at the same time, pupil diameter reached pre-treatment values 24-36 hours after treatment. In rabbit's mydriasis occurred faster, 10-25 minutes after treatment without significant IOP increase and mydriasis lasted 10-12 hours. Significant STT decrease was recorded in cats, but more likely were connected to stress factors. This drug could be considered as a therapeutical alternative in rabbit more than in cats.