Infección de cavidad anoftálmica por Scedosporium sp. en paciente inmunocompetente: Reporte de caso / Anophthalmic socket infection caused by Scedosporium sp. in immunocompetent patient: Case Report

Resumen Scedosporium sp. es un género de hongos filamentosos que de forma infrecuente se relaciona a infecciones localizadas o diseminadas en pacientes inmunocom prometidos, o inmunocompetentes con trauma penetrante contaminado. Reportamos el caso de un paciente masculino de 31 años con antecedente de trauma penetrante por arma de fuego requiriendo enucleación de su ojo izquierdo, quien 12 años posterior al trauma inicial presenta necrosis de la cavidad anoftálmica con extrusión del implante y lisis del párpado inferior, finalmente identificando Scedosporium sp. como germen causal, y un mucocele en el seno frontal como reservorio. El paciente recibió tratamiento médico con voriconazol sistémico, desbridamiento quirúrgico amplio, y resección endoscópica del quiste de retención mucoso, con una evolución satisfactoria. Las infecciones micóticas de la cavidad anoftálmica son infrecuentes, requiriendo una alta sospecha diagnóstica y trata miento agresivo para evitar consecuencias potencialmente devastadoras.

Abstract Scedosporium sp. is a genus of filamentous fungi that is infrequently related to localized or disseminated infections in either immunocompromised patients, or immunocompetent patients with penetrating contaminated trauma. We report the case of a 31-year-old patient with a history of penetrating gunshot wound that required enucleation of his left eye, who 12 years after initial trauma presents necrosis of the anophthalmic socket with implant extrusion and lysis of inferior eyelid, finally identifying the causal agent to be Scedosporium sp. The patient received medical treatment with systemic voriconazole, and wide surgical debridement, with a satisfactory evolution. Fungal infections of anophthalmic sockets are infrequent, requiring a high diagnostic suspicion and aggressive treatment to avoid potentially devastating consequences.

Low-cost three-dimensional printed orbital template-assisted patient-specific implants for the correction of spherical orbital implant migration

Purpose: To describe the outcomes of a patient-specific implant (PSI), fabricated using a three-dimensional (3D) printed orbital template and placed in the basin of the inferior orbital fissure to correct inferotemporally migrated spherical orbital implant. Methods: This is a single-center, prospective, consecutive, interventional, case series of six patients, with non-porous, spherical, orbital implant migration that underwent implant recentration surgically with a novel technique. Migration was subclassified either as decentration that did not affect the prosthetic retention or as displacement that affected the prosthetic retention in the eye socket. Only implant displacements were treated. The primary outcome measure was centration of the implant clinically and radiologically, with ability to retain the prosthesis. Results: At a mean follow-up of 21 months, all six orbital spherical implants remained centered. There were no cases of extrusion, exposure, or migration of either implants. There were no cases of PSI displacement. Additional procedures to optimize the aesthetic outcome of the customized ocular prosthesis (COP) required were simultaneous fornix formation suture in three patients, subsequent fornix formation with mucus membrane graft in two patients, and levator resection and sulcus hyaluronic acid gel injection in one patient each. The mean PSI implant weight was 2.66 ± 0.53 g. The mean COP weight was 2.2 ± 0.88 g postoperatively. The median patient satisfaction with the procedure was 9 on 10. Conclusion: A 3D printing-assisted PSI placed in the basin of the inferior orbital fissure allows recentration of the migrated implant over a follow-up of 21 months without complications.

Frequência de ocorrência de cavidade anoftálmica na região centro-oeste paulista e características dos portadores / Frequency of occurrence of anophthalmic socket in the Middle West region of the state of São Paulo and the carriers characteristics

OBJETIVO: Observar a frequência de ocorrência de casos de cavidade anoftálmica na região centro-oeste paulista e descrever o perfil demográfico dos portadores em estudo populacional. MÉTODOS: Estudo transversal, de caráter observacional e de amostragem aleatória, realizado em 12 cidades da região centro-oeste do estado de São Paulo, para as quais o centro de referência é a cidade de Botucatu. Os participantes foram determinados por sorteio que levou em conta o local de moradia, tendo sido estabelecida uma amostra de 11.453 indivíduos. Todos os exames foram feitos utilizando uma Unidade Oftalmológica Móvel. O protocolo da pesquisa consistiu de dados demográficos e exame oftalmológico completo (anamnese, antecedentes oculares e sistêmicos, antecedentes familiares, avaliação da acuidade visual com e sem correção, tonometria, biomicroscopia, fundoscopia e exame refracional). Todos os dados obtidos foram transferidos para tabela Excel e submetidos à análise descritiva e apresentados como frequência de ocorrência. RESULTADOS: A frequência de ocorrência de cavidade anoftálmica na região centro-oeste paulista foi de 0,96‰. Dentre os sujeitos examinados, foram encontrados 11 casos de cavidade anoftálmica, com acometimento de 0,7‰, no sexo feminino e 1,3‰, do sexo masculino. Dentre as causas de cavidade anoftálmica foram encontradas glaucoma (olho cego doloroso), microftalmia, trauma e endoftalmite. Os indivíduos eram, em sua maioria, de idade superior ou igual a 40 anos, tendo sido encontrado apenas um caso com idade abaixo de 19 anos. CONCLUSÃO: A cavidade anoftálmica ocorreu em 0,96‰ dos habitantes da região centro-oeste paulista, acometendo mais frequentemente os homens e com grande variação de idade de acometimento.

PURPOSE: To observe the frequency of the occurrence of the anophthalmic socket in the Middle West region of the state of São Paulo and to describe the demographic profile of the carriers in a population-based data. METHODS: A cross-sectional study involve a random sampling carried out in twelve cities of the Middle West region of the state of São Paulo, for which the reference center is the city of Botucatu was done. The participators were chosen by assortment which considered the houses of these people. It was established a sampling with 11,453 people. All the exams were realized using a Mobile Ophthalmologic Unit. The research protocol included the demographic data and the complete ophthalmologic exam (anamnesis, ocular and systemic antecedents, familiar antecedents, visual acuity with and without correction, tonometry, biomicroscopy, fundoscopy, and refraction exam). All the data were transferred to an Excel spreadsheet and submitted to a descriptive analysis and were presented by the frequency of the occurrence. RESULTS: Ophthalmic socket frequency in the Middle West region of the state of São Paulo was 0.96‰. We found 11 cases of anophthalmic socket, with involvement of 0.7 ‰ in females and 1.3 ‰, male. The most common causes of anophthalmic socket were glaucoma (blind painful eye), microphthalmia, trauma, and endophthalmitis. The majority of the people were 40 years old or more, and we found just one person younger than 19 years old. CONCLUSION:The anophthalmic socket occurred in 0.96‰ of the habitants of the Middle West region of São Paulo State, occurring mainly in male and with large variety of the affected ages.

Tear and ocular surface profile in adult anophthalmic sockets

Objective@#To determine the tear and ocular surface profile of the anophthalmic socket in relation to the contralateral normal eye.@*Methods@#Twenty-five adult patients with unilateral anophthalmic sockets were included into the study. They were at least 2 months post-enucleation or post-evisceration and without any topical medications on the anophthalmic socket and control eye for at least 2 weeks. Assessment was performed using the following parameters: (1) meibomian gland evaluation, (2) ocular surface staining, (3) degree of conjunctival inflammation, (4) Schirmer I and II, and (5) conjunctival impression cytology. @*Results@#Mucoid discharge (52%) was the most common complaint in anophthalmic sockets, followed by itchiness (40%), tearing (36%), and dryness (4%). Compared to control eyes, the anophthalmic sockets had more pronounced and statistically significant lid wiper epitheliopathy, conjunctival staining, and bulbar inflammation. Meibomian gland dysfunction, Schirmer I and II, and conjunctival impression cytology showed no difference between the 2 groups. There was a correlation between the symptoms complained and the ocular staining patterns of the anophthalmic sockets.@*Conclusion@#Anophthalmia predisposes to various ocular surface problems, such as a change in the composition of tears, specifically an increase in the mucin component and a decrease in the aqueous and lipid components, resulting to increased tear viscosity.

Reconstruction of Contracted Anophthalmic Socket with Acellular Dermal Allograft

PURPOSE: To study the use of acellular dermal allografts for the reconstruction of contracted anophthalmic sockets. METHODS: A retrospective analysis was performed on 12 patients with contracted sockets who underwent reconstructive surgery using acellular dermal allografts from 2002 to 2006. RESULTS: The mean age of the patients was 44.4 years. The mean duration of conjunctival sac contracture after removal of the eyeball was 21.4 years. In 4 patients, acellular dermal allografts were used as spacer grafts to elevate the eyelids, and in 8 patients the allografts were used as a conjunctival grafting material. All patients were able to wear ocular prostheses comfortably 2.2 months after surgery with acceptable cosmesis. There were no serious complications, such as infections or graft rejections during follow-up period. CONCLUSIONS: When used as conjunctival grafting materials and tarsal substitutes to elevate patients' eyelids, acellular dermal allografts gave cosmetically and functionally acceptable results in the surgical rehabilitation of contracted sockets.

A Case of Nonspecific Orbital Inflammation in Anophthalmia

PURPOSE: We report a case of orbital pseudotumor developed in an anophthalmic socket, presenting no typical symptoms or signs. METHODS: A 67-year-old woman was referred for treatment of necrotizing scleritis of her left eye and painful orbital pseudotumor of her right eye. The right eye had been removed 10 years previously. There was an irregular mass in superomedial portion of anterior orbit. Orbital MRI showed poorly defined T1 iso and low T2 signal intensity of a lesion in the medial anterior portion of the right orbit. Because systemic steroid administration was limited, 2 mg of betamethasone was injected locally, after which the size of the lesion was decreased and the orbital pain improved. CONCLUSIONS: The diagnosis of an orbital pseudotumor developed in an anophthalmic socket may be difficult because of the lack of typical eyeball-related signs or symptoms such as proptosis, red eye, or decreased vision. But, the easy access to the lesion can provide early and effective treatment through direct injection of glucocorticoid.

Anophthalmic Conjunctival Sac Reconstruction using Acellular Dermal Allograft

PURPOSE: To investigate the clinical results and safety of the acellular dermal allograft (SureDerm(R), Hans Biomed co., Korea) as a new spacer graft in anophthalmic socket contracture. METHODS: We grafted SureDerm(R) as the replacing spacer in 8 patients for the treatment of anophthalmic socket contracture. Under local anesthesia, SureDerm(R) was sutured at the levator aponeurosis and upper margin of the tarsus using 6-0 Vicryl in the upper lid. In the lower lid, SureDerm(R) was sutured at the tarsal conjunctiva and lower margin of the incised conjunctiva. The patients were followed-up individually for 6 to 15 months. RESULTS Surgical results in all 8 eyelids (6 lower lids, 2 upper lids) were satisfactory. The grafted SureDerm(R) survived in all patients with little contracture. There were no other complications such as infection, rejection, or exposure. CONCLUSIONS: We consider SureDerm(R) a good replacing spacer material for the treatment of anophthalmic socket contracture.

Clinical Results of Porous Polyethylene Orbital Implants in Postenucleation Retinoblastoma Patients

PURPOSE: To report the clinical results of implantation of porous polyethylene orbital implant (Medpor(R))after enucleation of eyes in patients with retinoblastoma. METHODS: The medical records of 33 children who had undergone Medpor(R) implantation after enucleation of eyes in patients with retinoblastoma were reviewed retrospectively. RESULTS: Surgery was performed in 33 eyes of 33 patients. The mean age at the time of operation was 24 months (ranged from 2 to 85 months). The mean follow-up period was 20 months (ranged from 12 to 31months). The diameters of the orbital implant used were 18 mm in 20 patients, and 20 mm in 13 patients. All parents were satisfied with the motility and cosmesis of the prosthesis. Exposure of the porous polyethylene orbital implant was found in 11 eyes (33.3%) at the mean postoperative time of 15 months (ranged from 7 to 29 months). The implant exposures in 10 eyes were not responsive to supportive therapy and underwent subconjunctival scleral patch grafts and direct conjunctival closure with success. The small exposure of the remaining eye was spontaneously healed under a careful observation with supportive therapy. CONCLUSIONS: Medpor(R) implantation in children with retinoblastoma showed good surgical results. However, careful follow-up examination is needed due to potential conjunctival pressure erosion and implant exposure. If exposure of the implant does occur, active surgical management is suggested instead of supportive management.

Surgical Effect of the Secondary Implantation of Hydroxyapatite

Intraorbital hydroxyapatite implant has advantages such as nondisplacement, prolapse and good prosthetic motility. The surgical effect of the secondary implantation of hydroxyapatite sphere was evaluated for 18 anophthairnic sockets of 18 patients. After follow-up period of over 6 months, extraocular muscle motility, prosthesis motility, improvement of the superior sulcus deformity, and enophthalmos were evaluated. Postoperative good EOM motility was noted in 7 cases (43.8%). Of the 15 cases without drilling, good prosthetic motility was noted in I case (6.7%), fair in 3 cases (20.0%), and poor in 11 cases (73.3%). One of the two eyes with drilling had good motility and the other one eye showed fair prosthetic motility. Superior sulcus deformity and enophthalinos improved in 12 cases-(75%) and 14 cases (82.4%). In postoperative complication, displacement or prolapse of the implant was not noted in any patient. and one patient. with postradiation showed m.inimal wound dehescence with spontaneous healing.