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1.
Acta Ophthalmol ; 2024 Mar 27.
Artículo en Inglés | MEDLINE | ID: mdl-38538530

RESUMEN

PURPOSE: To evaluate axial length (AL), orbital width (OW) and height (OH) development in congenital microphthalmia and anophthalmia (MICA) using serial ultrasonography measurements. METHODS: A longitudinal prospective cohort (n = 74) of unilaterally and bilaterally affected MICA patients was followed from 2013 to 2022 at the university hospital in Amsterdam, the Netherlands. Clinical entity, age, severity category based on axial length, conformer treatment and intra-orbital cysts were registered. The main outcome measures were the absolute and relative growth of AL, OW and OH. Surgical and intra-orbital cyst cases were described separately. RESULTS: Absolute microphthalmic eye size increased in 27/49 (55%) unilateral MICA eyes, but growth arrest/decrease in the remaining could shift the case to a more severe category over time. A final affected/unaffected orbital symmetry ≥80% was seen in the large majority of unilateral cases (45/46 for OW, 43/46 for OH). Cases with AL < 10.5 mm had orbital symmetry <80% more often. Most orbital symmetry changes were seen in moderate and severe unilateral cases treated with 3D-printed conformer therapy starting at age <1 year, with 6/10 (60%) symmetry increase, 30% unchanged symmetry and 10% symmetry decrease. All cases older than 6.5 years (n = 6) did not show any change anymore, regardless of treatment. For bilateral and unilateral mild cases, orbital dimensions kept the same proportions during follow-up, with or without conformer treatment. CONCLUSIONS: Using severity categories in MICA based on relative AL may aid the decision to start conformer treatment, as most orbital symmetry changes were seen in moderate and severe unilateral cases receiving 3D-printed conformer therapy that started under age 1.

2.
J Neurooncol ; 166(1): 79-87, 2024 Jan.
Artículo en Inglés | MEDLINE | ID: mdl-38150061

RESUMEN

PURPOSE: Progressive pediatric optic pathway gliomas (OPGs) are treated by diverse systemic antitumor modalities. Refined insights on the course of intra-tumoral components are limited. METHODS: We performed an exploratory study on the longitudinal volumetric course of different (intra-)tumor components by manual segmentation of MRI at the start and after 3, 6 and 12 months of bevacizumab (BVZ) treatment. RESULTS: Thirty-one patients were treated with BVZ (median 12 months, range: 2-39 months). During treatment the total tumor volume decreased with median 19.9% (range: - 62.3 to + 29.7%; n = 30) within the first 3 months, decreased 19.0% (range: - 68.8 to + 96.1%; n = 28) between start and 6 months and 27.2% (range: -73.4 to + 36.0%; n = 21) between start and 12 months. Intra-tumoral cysts were present in 12 OPGs, all showed a decrease of volume during treatment. The relative contrast enhanced volume of NF1 associated OPG (n = 11) showed an significant reduction compared to OPG with a KIAA1549-BRAF fusion (p < 0.01). Three OPGs progressed during treatment, but were not preceded by an increase of relative contrast enhancement. CONCLUSION: Treatment with BVZ of progressive pediatric OPGs leads to a decrease of both total tumor volume and cystic volume for the majority of OPGs with emphasis on the first three months. NF1 and KIAA1549-BRAF fusion related OPGs showed a different (early) treatment effect regarding the tumor enhancing component on MRI, which did not correlate with tumor volume changes. Future research is necessary to further evaluate these findings and its relevance to clinical outcome parameters.


Asunto(s)
Quistes , Neurofibromatosis 1 , Glioma del Nervio Óptico , Niño , Humanos , Bevacizumab/farmacología , Bevacizumab/uso terapéutico , Proteínas Proto-Oncogénicas B-raf , Glioma del Nervio Óptico/diagnóstico por imagen , Glioma del Nervio Óptico/tratamiento farmacológico , Glioma del Nervio Óptico/patología , Imagen por Resonancia Magnética
3.
Ophthalmic Plast Reconstr Surg ; 39(6S): S65-S80, 2023 12 01.
Artículo en Inglés | MEDLINE | ID: mdl-38054987

RESUMEN

PURPOSE: Dysthyroid optic neuropathy (DON) is a sight-threatening complication of thyroid eye disease (TED). This review provides an overview of the epidemiology, pathogenesis, diagnosis, and current therapeutic options for DON. METHODS: A literature review. RESULTS: DON occurs in about 5% to 8% of TED patients. Compression of the optic nerve at the apex is the most widely accepted pathogenic mechanism. Excessive stretching of the nerve might play a role in a minority of cases. Increasing age, male gender, smoking, and diabetes mellitus have been identified as risk factors. Diagnosis of DON is based on a combination of ≥2 clinical findings, including decreased visual acuity, decreased color vision, relative afferent pupillary defect, visual field defects, or optic disc edema. Orbital imaging supports the diagnosis by confirming apical crowding or optic nerve stretching. DON should be promptly treated with high-dose intravenous glucocorticoids. Decompression surgery should be performed, but the response is incomplete. Radiotherapy might play a role in the prevention of DON development and may delay or avoid the need for surgery. The advent of new biologic-targeted agents provides an exciting new array of therapeutic options, though more research is needed to clarify the role of these medications in the management of DON. CONCLUSIONS: Even with appropriate management, DON can result in irreversible loss of visual function. Prompt diagnosis and management are pivotal and require a multidisciplinary approach. Methylprednisolone infusions still represent first-line therapy, and surgical decompression is performed in cases of treatment failure. Biologics may play a role in the future.


Asunto(s)
Oftalmopatía de Graves , Papiledema , Humanos , Masculino , Glucocorticoides/uso terapéutico , Oftalmopatía de Graves/complicaciones , Oftalmopatía de Graves/diagnóstico , Oftalmopatía de Graves/terapia , Metilprednisolona , Nervio Óptico
4.
Br J Ophthalmol ; 2023 Nov 27.
Artículo en Inglés | MEDLINE | ID: mdl-38041682

RESUMEN

BACKGROUND: Orbital decompression is commonly performed to correct proptosis in patients with Graves' orbitopathy (GO). In literature, the incidence of new-onset constant diplopia after orbital decompression varies. Few studies have evaluated changes in duction and cyclodeviation after orbital decompression. We evaluated the changes in duction, cyclodeviation, eye position and degree of diplopia after orbital decompression. METHODS: We retrospectively analysed data from patients who underwent orbital decompression at our hospital between January 2016 and July 2020. Data regarding the type of decompression, eye position, duction, cyclodeviation and level of diplopia according to the Gorman score were recorded. RESULTS: Data from 281 eyes/orbits of 156 patients were analysed. Proptosis decreased from 23.8±2.5 to 20.9±2.5 mm. Horizontal and vertical duction range decreased (horizontal d=0.40 and vertical 0.30; p=0.000) after surgery; however, the change was not clinically significant (≤5°). Horizontal deviation changed towards esodeviation (d=-0.45; p=0.000), whereas vertical deviation remained stable (d=0.15; p=0.161). Preoperative cyclotorsion in the primary and downgaze positions changed towards incyclodeviation (primary: d=0.30, p=0.021; downgaze d=0.30, p=0.039). Diplopia improved in 22% (18 patients), whereas new-onset constant diplopia developed in 11% (12 patients). Elevation has an excellent predictive value of causing new-onset constant diplopia when measured preoperatively as <19°, sensitivity 88.0%, specificity 41.7% (area under the curve 0.812 (95% CI 0.660 to 0.963); p=0.000). CONCLUSION: After orbital decompression, incidence of new-onset constant diplopia was 11% and 22% of the patients had partial or complete improvement of their diplopia. The changes in duction, cyclodeviation and horizontal deviation showed a statistical but not clinical difference. Patients with GO and a preoperatively restricted elevation of <19° are at risk of developing new-onset constant diplopia. In addition, preoperative incyclodeviation may worsen after decompression surgery.

5.
Ned Tijdschr Geneeskd ; 1672023 10 25.
Artículo en Holandés | MEDLINE | ID: mdl-37930160

RESUMEN

Tear complaints can arise from either an increased tear production or from a disturbed tear drainage. Increased tear production from the lacrimal gland is a neuroregulated response to a dried out or irritated ocular surface. Dryness often results from a reduced quality of the tear film, but can also be caused by eyelid malposition with increased globe exposure. Impaired tear drainage usually occurs when the lacrimal drainage system is blocked at the level of the ductus nasolacrimalis, the canaliculi or the lacrimal punctae. Anatomical knowledge of the lacrimal system is necessary to distinguish between the different causes. Using cases and illustrations, we provide insight into the diagnostic considerations for a patient with a watery eye.


Asunto(s)
Enfermedades de los Párpados , Enfermedades del Aparato Lagrimal , Aparato Lagrimal , Humanos , Enfermedades del Aparato Lagrimal/diagnóstico , Enfermedades del Aparato Lagrimal/terapia , Lágrimas/fisiología
6.
Blood ; 142(18): 1570-1575, 2023 11 02.
Artículo en Inglés | MEDLINE | ID: mdl-37595284

RESUMEN

In this retrospective study, BRAF mutation status did not correlate with disease extent or (event-free) survival in 156 adults with Langerhans cell histiocytosis. BRAFV600E was associated with an increased incidence of second malignancies, often comprising hematological cancers, which may be clonally related.


Asunto(s)
Histiocitosis de Células de Langerhans , Neoplasias Primarias Secundarias , Humanos , Adulto , Neoplasias Primarias Secundarias/epidemiología , Neoplasias Primarias Secundarias/genética , Proteínas Proto-Oncogénicas B-raf/genética , Estudios Retrospectivos , Incidencia , Histiocitosis de Células de Langerhans/epidemiología , Histiocitosis de Células de Langerhans/genética , Histiocitosis de Células de Langerhans/patología , Mutación
7.
Interv Neuroradiol ; : 15910199231164833, 2023 Mar 22.
Artículo en Inglés | MEDLINE | ID: mdl-36945855

RESUMEN

BACKGROUND: Cavernous sinus dural arteriovenous fistula (CSDAVF) is a rare condition that radiologists would encounter in their careers. We aim to describe the clinical and radiological characteristics of this condition, and to provide a management workflow. METHODS: In our retrospective study, we studied 27 patients with CSDAVF from January 2007 to August 2020. Patients with direct cavernous sinus AVFs and patients with incomplete date were excluded. Clinical and radiological data were collected and analyzed. RESULTS: Fourteen patients were conservatively treated with spontaneous resolution while 13 patients had endovascular intervention performed. In the intervention group, seven patients had intra-cranial reflux seen on radiological imaging and six patients had clinical deterioration, hence requiring intervention. Clinically, among our patients, 21 had proptosis, 20 had conjunctiva hyperaemia, 18 had extraocular movement limitation, 13 had raised intraocular pressure, 11 had chemosis, ten had ocular pain, nine had ocular bruit, eight had headache and six had worsening visual acuity. Radiologically, a concurrence was seen between superior ophthalmic vein thrombosis and spontaneous resolution of the CSDAVF, as compared to those who underwent intervention. A paradoxical increase of ocular symptoms was seen despite a decrease of flow or stagnation of contrast in radiological imaging of CSDAVF. CONCLUSIONS: In our study, 52% of CSDAVF closed spontaneously. As deterioration of ocular symptoms in patients with CSDAVF might also reflect spontaneous progressive occlusion, it warrants dynamic vascular imaging to check the status of venous outflow. Patients with CSDAVF with corticovenous reflux or deterioration of visual acuity need more urgent (endovascular) treatment.

8.
Eye (Lond) ; 37(1): 48-53, 2023 01.
Artículo en Inglés | MEDLINE | ID: mdl-34999720

RESUMEN

OBJECTIVES: To describe the features, management approaches, and outcomes of orbito-cranial schwannomas. METHODS: Retrospective review of ten patients with orbito-cranial schwannomas managed in six orbital services over 22 years. Data collected included demographics, presenting features, neuroimaging characteristics, histology, management approach, complications, and outcomes. RESULTS: Mean age of the patients was 41.4 ± 19.9 years, and 6 (60%) were females. The majority presented with proptosis (90%), limited extraocular motility (80%), eyelid swelling (60%), and optic neuropathy (60%). Most lesions (80%) involved the entire anterior-posterior span of the orbit, with both intra- and extraconal involvement. All tumours involved the orbital apex, the superior orbital fissure, and extended at least to the cavernous sinus. Surgical resection was performed for all. Seven (70%) of the tumours were completely or subtotally resected combining an intracapsular approach by an orbital-neurosurgical collaboration, with no recurrence on postoperative follow-up (6-186 months). Three underwent tumour debulking. Of these, two remained stable on follow-up (6-34 months) and one showed progression of the residual tumour over 9 years (cellular schwannoma on histology) necessitating stereotactic radiotherapy (SRT) for local control. Adjuncts to the orbito-cranial resection included perioperative frozen section (n = 5), endoscopic transorbital approach (n = 2), and image-guided navigation (n = 1). Post-surgical adjuvant SRT was used in three subjects. CONCLUSIONS: These results highlight the possibility of successful surgical control in complex orbito-cranial schwannomas. A combined neurosurgical/orbital approach with consideration of an intracapsular resection is recommended. Recurrence may not occur with subtotal excision and observation may be reasonable. Adjunctive SRT for progression or residual tumour can be considered.


Asunto(s)
Neurilemoma , Enfermedades del Nervio Óptico , Femenino , Humanos , Adulto Joven , Adulto , Persona de Mediana Edad , Masculino , Neoplasia Residual , Endoscopía/métodos , Órbita , Estudios Retrospectivos , Neurilemoma/diagnóstico , Neurilemoma/cirugía , Resultado del Tratamiento
9.
Br J Ophthalmol ; 107(9): 1239-1245, 2023 09.
Artículo en Inglés | MEDLINE | ID: mdl-35477668

RESUMEN

BACKGROUND/AIMS: To evaluate treatment with custom, three-dimensional (3D) printed conformers for socket expansion in congenital microphthalmia and anophthalmia (MICA). METHODS: Retrospective analysis of prospective cohort from 2016 to 2020. All children received custom-made 3D-printed conformers increasing in size. We measured height, width, thickness, surface and volume of first and consecutive conformers, as well as horizontal palpebral fissure length (HPF) at start and follow-up visits. We analysed these parameters for severely (<45%) and moderately (>45%-75%) affected children, based on affected axial length on ultrasonography. RESULTS: We included 18 cases (9 severe, 9 moderate) with a total of 174 conformers (88 severe, 86 moderate) and a mean follow-up of 2.8 years (range 1.3-4.8). The mean relative HPF increased from 77% to 93% with 16/17 cases reaching >80%, and 12/17 cases >90% symmetry. Horizontal and vertical conformer dimensions increased up to 10 months of treatment, with a steeper slope for the severe group (10.5% vs 5.5% for height and 9.0% vs 6.1% for width gain per treatment month, for severe and moderate MICA, respectively). After 10 months of treatment conformer height and width increased only slightly. No serious complications were observed. CONCLUSION: 3D-design and printing of solid conformers results in highly acceptable horizontal eyelid symmetry in the treatment of congenital MICA. The mean increase in conformer height and width in the first 10 months should be about 170% for moderate and about 200% for severe MICA. The presented conformer size formulas can aid ophthalmologists and ocularists to plan conformer treatment.


Asunto(s)
Anoftalmos , Microftalmía , Niño , Humanos , Estudios Retrospectivos , Flujo de Trabajo , Estudios Prospectivos , Diseño Asistido por Computadora , Impresión Tridimensional
10.
Acta Ophthalmol ; 101(1): e106-e112, 2023 Feb.
Artículo en Inglés | MEDLINE | ID: mdl-35934887

RESUMEN

INTRODUCTION AND PURPOSE: Patients with Graves' orbitopathy (GO) may develop restricted elevation; this can lead to hypotropia, sometimes in combination with an abnormal head posture. Recession of one or both inferior rectus muscles is the first line surgery to restore eye motility in these patients. However, this may result in A pattern strabismus. This study was performed to determine the rate of occurrence of this type of incomitant strabismus and potential predictive factors. METHODS: All patients undergoing surgery on one or two inferior rectus muscles over a 10-year period were screened retrospectively for the A pattern, defined as a ≥5° difference in squint angle between the primary gaze and downgaze. The extraocular muscle thickness in patients with acquired A pattern was determined by computed tomography (CT) and compared with a control group consisting of patients randomly selected from the total cohort. RESULTS: In a total of 590 patients, surgery was performed on the inferior rectus muscle(s) during the study period; the A pattern was identified in 59 patients. Simultaneous surgery was performed on one or both medial rectus muscles in 32% of the patients. This group had significant incyclotorsion (p = 0.000) and less depression (p = 0.000) postoperatively. The mean amount of recession was 4.38 ± 1.53 mm in the A pattern group and 3.91 ± 1.37 mm in the control group (p = 0.032). The amount of depression was 50.2° ± 7.4° in the A pattern group and 57.3° ± 4.4° in the control group (p = 0.045). The inferior rectus muscle was significantly thicker in the A pattern than in the control group (p = 0.027), while there was no significant difference in the thickness of the superior oblique muscle between the two groups (p = 0.870). Of all patients with the A pattern, 47% required further surgery to achieve adequate binocular single vision. CONCLUSION: Increased preoperative inferior rectus muscle thickness and relatively limited depression could be predictors of postoperative A pattern inferior rectus recession in patients with GO. Step-by-step procedures are preferable in this surgically challenging group of patients.


Asunto(s)
Oftalmopatía de Graves , Estrabismo , Humanos , Oftalmopatía de Graves/complicaciones , Oftalmopatía de Graves/diagnóstico , Oftalmopatía de Graves/epidemiología , Músculos Oculomotores/diagnóstico por imagen , Músculos Oculomotores/cirugía , Estudios Retrospectivos , Incidencia , Resultado del Tratamiento , Procedimientos Quirúrgicos Oftalmológicos/métodos , Estrabismo/epidemiología , Estrabismo/etiología , Estrabismo/cirugía
11.
Cancers (Basel) ; 14(24)2022 Dec 10.
Artículo en Inglés | MEDLINE | ID: mdl-36551572

RESUMEN

BACKGROUNDS: Bevacizumab (BVZ) is used as a subsequent line of treatment for pediatric optic pathway glioma (OPG) in the case of progression. Data on the treatment effect concerning tumor progression and visual function are scarce and nationwide studies are lacking. METHODS: We performed a retrospective, nationwide, multicentre cohort study including all pediatric patients with OPG treated with BVZ in the Netherlands (2009-2021). Progression-free survival, change in visual acuity and visual field, MRI-based radiologic response, and toxicity were evaluated. RESULTS: In total, 33 pediatric patients with OPG were treated with BVZ (median 12 months). Visual acuity improved in 20.5%, remained stable in 74.4%, and decreased in 5.1% of 39 of all analysed eyes. The monocular visual field improved in 73.1%, remained stable in 15.4%, and decreased in 7.7% of 25 analysed eyes. Radiologic response at the end of therapy showed a partial response in 7 patients (21.9%), minor response in 7 (21.9%), stable disease in 15 (46.9%), and progressive disease in 3 (9.3%). Progression-free survival at 18 and 36 months after the start of BVZ reduced from 70.9% to 38.0%. Toxicity (≥grade 3 CTCAE) during treatment was observed in five patients (15.2%). CONCLUSION: Treatment of BVZ in pediatric patients with OPG revealed stabilisation in the majority of patients, but was followed by progression at a later time point in more than 60% of patients. This profile seems relatively acceptable given the benefits of visual field improvement in more than 70% of analysed eyes and visual acuity improvement in more than 20% of eyes at the cessation of BVZ.

12.
Int J Ophthalmol ; 15(11): 1852-1856, 2022.
Artículo en Inglés | MEDLINE | ID: mdl-36404960

RESUMEN

AIM: To examine the publication trend of oculoplastic articles throughout the last decade in general ophthalmology journals. METHODS: A review of all abstracts published between January 2010 to December 2019 in general, clinical ophthalmic journals was conducted. Articles that were categorized as original articles in general and clinical journals were included in the study. RESULTS: Totally 10 281 abstracts were included. Of them 465 (4.5%) were oculoplastic publications. The mean number of annual-publications was 46.5 and the mean annual-rate of oculoplastic publications was 4.51%. A significant decreasing trend in the number of oculoplastic publication in the last decade was found (P<0.01, R 2=0.770). However, there was no significant change in the annual-rate of oculoplastic publications during the last decade (P=0.191, R 2=0.203). From the 465 oculoplastic articles: 179 (38.5%) were articles about eyelid diseases, 160 (34.40%) were about orbit diseases, 92 (19.80%) were about lacrimal diseases and 34 (7.30%) were about thyroid eye disease (TED). A significant decreasing trends in the number of orbital and eyelids publications were found (P<0.01, P<0.01). However, there were no significant changes in the annual-rate of orbital, eyelids, TED and lacrimal-diseases publications throughout the last decade. CONCLUSION: Oculoplastic subspecialty deals with a wide range of pathologies in different ages. However, less than 5% of the articles in general, clinical, high impact factor ophthalmology journals are about oculoplastic diseases. One of the best way for ophthalmologists from different subspecilties, nowadays, to be updated, is to read high-impact-factor, general ophthalmology journals. Therefore, it is important that those journals will include articles about breakthroughs in oculoplastic.

13.
Eur J Ophthalmol ; 32(3): 1406-1410, 2022 May.
Artículo en Inglés | MEDLINE | ID: mdl-34096347

RESUMEN

PURPOSE: To examine the absolute number and the proportions of articles published in general high-ranked ophthalmology journals for each ophthalmic subspecialty during the last decade, and to examine the publishing trends over the study period. METHODS: All original articles published between January 2010 and December 2019 in the selected general clinical ophthalmic journals were included in the study. All abstracts of original articles were reviewed and deemed to be related to 1 of the 11 ophthalmic subspecialties. RESULTS: Six journals and 10,232 abstracts were reviewed. Articles focused on medical retina were the most common in the last decade (35.22%) while articles focused on strabismus were the least common (2.11%). The total number of articles published per year decreased during the last decade (p < 0.01). There was a significant reduction in the number of publications per year focused on anterior-chamber (p = 0.012), cataract and refractive-surgeries (p = 0.014), oculoplastic (p < 0.01), and strabismus (p = 0.011). In each year during the last decade, the highest proportion of publications was focused on medical retina while the lowest proportion of publications in most of the years was focused on strabismus. There was a significant decrease during the years in the proportion of articles focused on oculoplastic (p < 0.01). CONCLUSIONS: During the last decade, there have been differences in the proportion of publications of different ophthalmology subspecialties in high impact factor journals. This probably derives from demographic changes and advances in diagnosis and treatment. The proportion of articles focused on medical retina was the highest during all years while the proportion of articles focused on strabismus was consistently the lowest.


Asunto(s)
Oftalmología , Publicaciones Periódicas como Asunto , Estrabismo , Bibliometría , Humanos , Edición
14.
Eur J Ophthalmol ; 32(1): 717-724, 2022 Jan.
Artículo en Inglés | MEDLINE | ID: mdl-33706571

RESUMEN

PURPOSE: Recurrent contracted sockets are complex situations where previous surgeries have failed, disabling the wear of an ocular prosthesis. A combined method of surgery and long-term fixation using custom-made, three-dimensional (3D) printed conformers is evaluated. METHODS: Retrospective case series of nine patients with recurrent excessive socket contraction and inability to wear a prosthesis, caused by chemical burns (n = 3), fireworks (n = 3), trauma (n = 2) and enucleation and radiotherapy at childhood due to optic nerve glioma (n = 1) with three average previous socket surgeries (range 2-6). Treatment consisted of a buccal mucosal graft and personalized 3D-printed conformer designed to be fixated to the periosteum and tarsal plates for minimal 2 months. Primary outcome was the retention of an ocular prosthesis. Secondary outcome was the need for additional surgeries. RESULTS: Outcomes were measured at final follow-up between 7 and 36 months postoperatively (mean 20 months). Eight cases were able to wear an ocular prosthesis after 2 months. Three cases initially treated for only the upper or only the lower fornix needed subsequent surgery for the opposite fornix for functional reasons. Two cases had later surgery for cosmetic improvement of upper eyelid position. Despite pre-existing lid abnormalities (scar, entropion, lash deficiency), cosmetic outcome was judged highly acceptable in six cases because of symmetric contour and volume, and reasonably acceptable in the remaining two. CONCLUSIONS: Buccal mucosal transplant fixated with a personalized 3D-designed conformer enables retention of a well-fitted ocular prosthesis in previously failed socket surgeries. Initial treatment of both upper and lower fornices is recommended to avoid subsequent surgeries for functional reasons.


Asunto(s)
Ojo Artificial , Implantes Orbitales , Niño , Humanos , Recurrencia Local de Neoplasia , Órbita , Impresión Tridimensional , Estudios Retrospectivos
15.
Acta Ophthalmol ; 100(6): e1340-e1348, 2022 Sep.
Artículo en Inglés | MEDLINE | ID: mdl-34951116

RESUMEN

PURPOSE: Graves' orbitopathy (GO) is a rare condition in children often considered to be a less severe condition than at an older age. The aim of our study was to analyse if there are any factors that distinguish paediatric from adult GO in order to provide guidelines for assessing and managing paediatric GO. METHODS: Study design is a multicentre retrospective observational case series; 115 paediatric patients diagnosed with GO who visited our university medical centres in the Netherlands and Iran between 2003 and 2019 were submitted for complete ophthalmological examinations, serological testing and/or orbital imaging. Main outcome measures focussed on the natural course and clinical picture as well as medical and surgical treatment in paediatric GO. RESULTS: Clinical findings included proptosis (n = 97; 84.3%), eyelid retraction (n = 77; 67%) and diplopia (n = 13; 11.3%). Ninety-two patients (80%) presented with mild disease, 21 (18.3%) with moderate-severe disease and two (1.7%) with severe GO. Five patients (4.3%) underwent intravenous glucocorticoids and 25 patients underwent orbital decompression surgery. Strabismus surgery due to primary involvement of extraocular muscles was performed in two patients (1.7%). Overall, rehabilitative surgical treatment was planned in 31 patients (26.9%) with inactive disease. Two patients experienced reactivation of the disease. CONCLUSION: Despite the fact that paediatric and adult GO are considered two separate entities, they might be the same disease with two different clinical phenotypes. Paediatric GO population presents with a comparable clinical picture regarding both soft tissue involvement and proptosis, which may require surgical intervention. Proptosis was present in the majority of paediatric GO patients. Orbital decompression was performed in 21.7% of patients.


Asunto(s)
Exoftalmia , Oftalmopatía de Graves , Descompresión Quirúrgica/efectos adversos , Exoftalmia/diagnóstico , Exoftalmia/etiología , Exoftalmia/cirugía , Oftalmopatía de Graves/diagnóstico , Oftalmopatía de Graves/cirugía , Humanos , Órbita/cirugía , Estudios Retrospectivos
16.
PLoS One ; 16(10): e0258548, 2021.
Artículo en Inglés | MEDLINE | ID: mdl-34673789

RESUMEN

Pediatric optic pathway glioma (OPG) can seriously decrease visual function in the case of progression. Systemic anticancer therapy (SAT) is considered the treatment of first choice for unresectable OPG. New SAT modalities for the treatment of progressive OPG have been introduced in the last decade, including VEGF and MAPK pathway inhibition. This systematic review evaluated the effect of SAT on change in visual acuity and visual field in OPG. A systematic review was performed on SAT for OPG (January 1990 to August 2020). MEDLINE and EMBASE (Ovid) were searched for studies reporting on change in visual acuity and visual field after treatment with SAT for OPG. Overall, 11 series, including 358 patients, fulfilled the eligibility criteria. After follow-up of median 3.7 years (range: cessation of SAT- 8.2 years), improvement in binocular VA was found in 0-45% of studies, stability in 18-77% and a decrease in 0-82%. Two studies reported on change in visual field (improvement in 19% and 71% of patients), although either the change was not defined or the testing strategy was lacking. Considerable heterogeneity was present among the included studies, such as variety in the combinations of SAT administered, status of neurofibromatosis type 1, definition regarding change in visual acuity, 1- or 2-eye analysis, diversity in anatomic location, and extent of follow-up, all of which made meta-analysis inappropriate. This systematic review suggests that the impact of SAT in OPG on visual function is still unclear. The wide ranges reported on the efficacy of SAT and the observed heterogeneity highlight the need for prospective studies with uniform definitions of outcome parameters.


Asunto(s)
Glioma del Nervio Óptico , Adolescente , Niño , Preescolar , Humanos , Visión Ocular
17.
Acta Ophthalmol ; 99(8): 850-860, 2021 Dec.
Artículo en Inglés | MEDLINE | ID: mdl-33576183

RESUMEN

PURPOSE: Patients with vertical strabismus and consecutive diplopia due to Graves' orbitopathy (GO) might need recession of the rectus superior muscle. The aim of this retrospective analysis is to provide dose-effect values and motility analysis for these patients. METHODS: Dose-effect relations (deviation reduction in primary position per amount of recession in mm), monocular excursions and size of the field of binocular single vision (BSV) were evaluated in three different groups of patients. Group 1 (n = 33) patients with primary fibrosis of superior rectus muscle undergoing recession of the ipsilateral rectus superior, Group 2 (n = 14) superior rectus muscle recession after recession of inferior rectus on the contralateral eye and Group 3 (n = 15) simultaneous ipsilateral superior rectus recession and contralateral inferior rectus muscle recession. Surgery was performed using the dose-effect for inferior rectus recession of 4 prism dioptre (PD,Δ ) reduction/per mm recession and the intraoperative traction test. RESULTS: Restoration of BSV in the central 20° of gaze could be reached in 88%, 79% and 67% of patients in the three groups, respectively. Restoration of BSV in downgaze (up to 30°) was a little less successful, 79%, 57% and 53%, respectively. The median dose-effect was 3.2Δ reduction/mm recession in Group 1 (Spearman correlation r = 0.75, p < 0.0001) and 3.3Δ /mm in Group 2 (n.s.) and 4Δ /mm in Group 3 (r = 0.67, p = 0.016). The basis for the improvement was the symmetrization of ductions. CONCLUSION: If superior rectus recession is necessary in GO, higher dosing around 3Δ reduction/per mm recession should be applied. In case of large deviations, combined vertical surgery (inferior rectus and superior rectus contralateral) is required and reasonable (low cyclotorsion). Single-step or two-step procedures lead to similar results.


Asunto(s)
Movimientos Oculares/fisiología , Oftalmopatía de Graves/cirugía , Músculos Oculomotores/cirugía , Procedimientos de Cirugía Plástica/métodos , Estrabismo/etiología , Visión Binocular/fisiología , Agudeza Visual , Adulto , Anciano , Anciano de 80 o más Años , Femenino , Estudios de Seguimiento , Oftalmopatía de Graves/complicaciones , Oftalmopatía de Graves/fisiopatología , Humanos , Masculino , Persona de Mediana Edad , Músculos Oculomotores/fisiopatología , Procedimientos Quirúrgicos Oftalmológicos/métodos , Estudios Retrospectivos , Estrabismo/fisiopatología , Estrabismo/cirugía , Resultado del Tratamiento
18.
J Cosmet Dermatol ; 19(11): 2845-2858, 2020 Nov.
Artículo en Inglés | MEDLINE | ID: mdl-32185876

RESUMEN

BACKGROUND: Inadvertent intra-arterial injection of dermal fillers including calcium hydroxylapatite (CaHA) can result in serious adverse events including soft tissue necrosis, permanent scarring, visual impairment, and blindness. When intra-arterial injection occurs, immediate action is required for optimal outcomes, but the infrequency of this event means that many physicians may never have experienced this scenario. The aim of this document is to provide evidence-based and expert opinion recommendations for the recognition and management of vascular compromise following inadvertent injection of CaHA. METHODS: An international group of experts with experience in injection of CaHA and management of vascular complications was convened to develop a consensus on the optimal management of vascular compromise following intra-arterial CaHA injection. The consensus members were asked to provide preventative advice for the avoidance of intravascular injection and to produce a treatment protocol for acute and delayed presentation. To ensure all relevant treatment options were included, the recommendations were supplemented with a PubMed search of the literature. RESULTS: For prevention of intra-arterial CaHA injection, consensus members outlined the importance of a thorough knowledge of facial vascular anatomy and patient history, as well as highlighting potential risk zones and optimal injection techniques. Individual sections document how to recognize the symptoms of vascular occlusion leading to vision loss and tissue necrosis as well as detailed treatment protocols for the management of these events. For impending tissue necrosis, recommendations are provided for early and delayed presentations with treatment protocols for acute and follow-up treatment. A separate section details the treatment options for open and closed wounds. CONCLUSIONS: All physicians should be prepared for the eventuality of intra-arterial injection of a dermal filler, despite its rarity. These consensus recommendations combine advice from aesthetic experts with the latest reports from the published literature to provide an up-to-date office-based protocol for the prevention and treatment of complications arising from intra-arterial CaHA injection.


Asunto(s)
Técnicas Cosméticas , Rellenos Dérmicos , Calcio , Consenso , Técnicas Cosméticas/efectos adversos , Rellenos Dérmicos/efectos adversos , Durapatita/efectos adversos , Humanos
19.
Acta Ophthalmol ; 98(5): 514-520, 2020 Aug.
Artículo en Inglés | MEDLINE | ID: mdl-32100474

RESUMEN

PURPOSE: Current clinical classifications do not distinguish between the severity of the MICrophthalmia/Anophthalmia (MICA) spectrum with regard to treatment urgency. We aim to provide parameters for distinguishing mild, moderate and severe MICA using clinical and biometrical characteristics. METHODS: We performed a single-centre, cross-sectional analysis of prospective cohort of 58 MICA children from September 2013 to February 2018 seen at the Amsterdam University Medical Center, The Netherlands. All patients with a visible underdeveloped globe were included. We performed full ophthalmic evaluation including horizontal palpebral fissure length, axial length by ultrasound and/or MRI measurements, paediatric and genetic evaluation. Cases were subdivided based on clinical characteristics. Biometrical data were used to calculate the relative axial length (rAL) and the relative horizontal palpebral fissure length (rHPF) compared with the healthy contralateral eye for unilateral cases. RESULTS: In previously untreated patients, a strong correlation exists between rAL and rHPF, distinguishing between severe, moderate and mild subjects using rAL of 0-45%, 45-75% and 75%-100%, respectively. Clinical subgroups were randomly dispersed throughout the scatterplot. CONCLUSION: Current classifications lack clinical implications for MICA patients. We suggest measuring eyelid length and axial length to classify the severity and determine treatment strategy. The 'severe' group has obvious asymmetry and abnormal socket configuration for which therapy should quickly be initiated; the 'moderately' affected group has normal socket anatomy with a microphthalmic eye with disturbing asymmetry for which treatment should be initiated within months of development; the 'mild' group has a slightly smaller axial length or less obvious eyelid asymmetry for which reconstructive correction is possible, but expansive conformer treatment is unnecessary.

20.
Ophthalmic Plast Reconstr Surg ; 36(4): 385-389, 2020.
Artículo en Inglés | MEDLINE | ID: mdl-31917766

RESUMEN

PURPOSE: Intravenous antibiotic prophylaxis is used for many clean-contaminated surgeries or clean surgeries with an implant, but its value for clean orbital surgery has not been determined. This study investigated infection risks and adverse effects related to antibiotics in patients undergoing orbital surgery. METHODS: A prospective, nonrandomized comparative case series of all patients undergoing orbital surgery with participating surgeons between October 1, 2013, and March 1, 2015. Types of surgery, antibiotic regimens, corticosteroid use, antibiotic side effects, and surgical site infections (SSIs) were entered into an electronic database and subsequently analyzed. Cases in which patients received postoperative oral antibiotics were analyzed separately. RESULTS: Of 1,250 consecutive orbital surgeries, 1,225 met inclusion criteria. A total of 1208 patients were included in the primary analysis: 603 received no antibiotic prophylaxis (group A), and 605 received a single dose of intravenous antibiotic (group B). Five patients (0.42%) developed an SSI, 3 in group A and 2 in group B. The difference in SSI rates was not statistically significant between the 2 groups (p = 0.66). Antibiotic prophylaxis, alloplastic implants, paranasal sinus entry, and corticosteroid use were not associated with differences in SSI rates. All SSIs resolved on a single course of oral antibiotics; an implant was removed in 1 case. There were no complications associated with a single dose of intravenous prophylaxis. However, 12% of 17 patients (group C) who received 1 week of oral postoperative prophylactic antibiotics developed antibiotic-related complications (diarrhea, renal injury), yielding a number needed to harm of 8.5. CONCLUSIONS: In this large series, antibiotic prophylaxis does not appear to have reduced the already low incidence of SSI following orbital surgery. Given the detriments of systemic antibiotics, the rarity of infections related to orbital surgery, and the efficacy of treating such infections should they occur, patients undergoing orbital surgery should be educated to the early symptoms of postoperative infection and followed closely, but do not routinely require perioperative antibiotics.


Asunto(s)
Profilaxis Antibiótica , Infección de la Herida Quirúrgica , Antibacterianos/uso terapéutico , Humanos , Estudios Prospectivos , Estudios Retrospectivos , Infección de la Herida Quirúrgica/prevención & control
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