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Journal of AAPOS : the Official... Jun 2024Thyroid eye disease (TED) can result in proptosis and ocular misalignment, leading to eye pain, diplopia, and vision loss. Teprotumumab, a humanized antibody against...
BACKGROUND
Thyroid eye disease (TED) can result in proptosis and ocular misalignment, leading to eye pain, diplopia, and vision loss. Teprotumumab, a humanized antibody against insulin-like growth factor 1 receptor, was approved in 2020 for the treatment of TED. The purpose of this study was to describe the effect of a full course of teprotumumab on ocular misalignment.
METHODS
The medical records of patients who underwent treatment with teprotumumab for active moderate-to-severe TED at a single institution from April 2020 to September 2023 were reviewed retroactively. Sensorimotor examination was performed at each visit using simultaneous prism-cover testing. Demographic information and previous history of radioactive iodine, steroids, strabismus surgery, and smoking were extracted from the record for analysis.
RESULTS
A total of 19 patients were treated during the study period, of whom 11 had strabismus and diplopia. The initial absolute horizontal misalignment in these 11 was 6.0 ± 1.5, vertical misalignment was 7.7 ± 2.4, and total misalignment was 11.5 ± 2.0. On completion of treatment, these measurements decreased by 2.0 ± 1.5, 2.2 ± 1.0, and 3.2 ± 1.6, respectively (P = 0.10, 0.02, and 0.04, resp.). Eight patients (73%) had a decrease in their strabismus, and 5 (46%) reported complete resolution of their diplopia at the final visit. No factors were predictive of which patients would have resolution of their misalignment. Of the remaining 3 patients who had no improvement in ocular alignment, 2 (66%) underwent strabismus surgery. Of the 8 patients with improvement of strabismus, only a single patient (13%) underwent strabismus surgery for persistent diplopia.
CONCLUSIONS
In our study cohort, a full course of teprotumumab coincided with complete resolution of diplopia in 46% of patients and a decrease in strabismus in 73% of patients.
PubMed: 38944235
DOI: 10.1016/j.jaapos.2024.103959 -
International Journal of Surgery Case... May 2024Primary intracranial neuroendocrine tumors (NETs) are exceedingly rare, often posing diagnostic challenges, particularly in non-secreting variants. These tumors may...
INTRODUCTION AND IMPORTANCE
Primary intracranial neuroendocrine tumors (NETs) are exceedingly rare, often posing diagnostic challenges, particularly in non-secreting variants. These tumors may initially present with nonspecific symptoms, leading to delayed diagnosis and potential neurological complications.
CASE REPORT
We present the case of a 33-year-old male admitted with a one-year history of progressively worsening headache accompanied by acute left ptosis and diplopia. Initial examination revealed left eye ptosis and hypotropia, indicative of third and fourth cranial nerve paralysis. Cavoscopy revealed a mild anterior wall bulge of the sphenoid with normal mucosa. MRI imaging unveiled an expansive clival process extending towards the sellar region and left cavernous sinus, completely occupying the sphenoid sinus and exerting mass effect on the pituitary stalk. Hormonal assays were within reference ranges, ruling out a hormonally-active tumor. Endoscopic endonasal surgery for biopsy revealed a low-grade neuroendocrine tumor positive for cytokeratin AE1/AE2, chromogranin A, synaptophysin, and beta-catenin, with a Ki-67-labeling index <2 %. Somatostatin receptor scintigraphy confirmed intense hyper fixation of the tracer in the sphenoidal tumor, supporting its neuroendocrine origin. The patient declined surgical intervention after informed consent, opting for C1 somatostatin analogs prior to radiotherapy. One-year follow-up demonstrated symptom stability with no tumor progression.
CLINICAL DISCUSSION
To this day, no consensus among reports concerning the optimal management of these cases. Imaging assessment is crucial to validate the primary nature of the tumor and to exclude any distant localization. Various therapeutic modalities, such as surgery, radiotherapy, and somatostatin analogs, should be considered based on the specific characteristics and extent of the tumor.
CONCLUSION
Our case is a clear reminder that neuroendocrine tumors should be considered as a differential diagnosis for skull base neoplasms.
PubMed: 38943936
DOI: 10.1016/j.ijscr.2024.109750 -
Case Reports in Neurological Medicine 2024The etiology of chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) remains elusive and is believed to involve multiple contributing factors. There have...
The etiology of chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) remains elusive and is believed to involve multiple contributing factors. There have been cases linking CIDP to the coronavirus disease 2019 (COVID-19) mRNA vaccine. However, there are no documented instances following alternative vaccines. We report a case of a 48-year-old woman, previously vaccinated with Pfizer-BioNTech's COVID-19 vaccine (BNT162b2), who subsequently received the Moderna mRNA-1273 vaccine. Within 2 days postvaccination, she developed diplopia and numbness in the lower limbs' distal extremities. Cerebrospinal fluid analysis exhibited protein-cell dissociation, while F-wave studies showed demyelinating activity in the bilateral tibial nerves. Given the disease's progressive nature, the patient was presumed to have CIDP and commenced steroid pulse therapy and intravenous immunoglobulin therapy. The onset of CIDP may be associated with variations in mRNA sequences and vaccine constituents.
PubMed: 38939233
DOI: 10.1155/2024/8584482 -
Journal of Surgical Case Reports Jun 2024Orbital metastasis originating from breast carcinoma, particularly ductal carcinoma, represents a rare clinical entity, with lobular carcinoma usually being more common....
Orbital metastasis originating from breast carcinoma, particularly ductal carcinoma, represents a rare clinical entity, with lobular carcinoma usually being more common. Long-term surveillance in breast cancer patients is crucial for early detection of metastasis. Herein, we present a case of a 70-year-old woman with a history of left ductal breast carcinoma, diagnosed and treated 12 years ago. She then developed left eye vision loss, diplopia, enophthalmos, and chemosis in October 2024. Imaging revealed orbital metastasis involving the left superior and lateral rectus extraocular muscles. Biopsy confirmed the diagnosis of orbital metastases arising from ductal breast carcinoma. This case underscores the significance of long-term surveillance in breast cancer patients, as metastasis can manifest years after the initial diagnosis. Despite its rarity, orbital metastasis warrants consideration in the differential diagnosis of ocular symptoms in patients with a history of breast carcinoma. Treatment primarily aims at palliation and preserving visual function, with prognosis typically poor.
PubMed: 38938683
DOI: 10.1093/jscr/rjae428 -
International Ophthalmology Jun 2024Various materials have been proposed for reconstructing orbital fractures. The materials used must meet certain criteria to ensure their suitability for restoring the...
PURPOSE
Various materials have been proposed for reconstructing orbital fractures. The materials used must meet certain criteria to ensure their suitability for restoring the structure and function of the organ. These criteria include biocompatibility, ease of application, non-toxicity, hypo-allergenicity, and non-carcinogenicity. In this study, we systematically reviewed the studies regarding the biomaterials in orbital implants and their clinical application.
METHODS
A comprehensive search across various databases, including PubMed, Scopus, EMBASE, Cochrane Library, and Web of Science, was conducted until April 10th, 2023. After retrieving the search results and eliminating duplicates, final studies were included after screening through defined criteria. Human and animal studies assessing the clinical application of biomaterials in orbital implants were included. The quality of the case series and controlled intervention studies were evaluated using the NIH tool, and for animal studies, the risk of bias was assessed using SYRCLE's tool.
RESULTS
Seventeen studies were included according to defined criteria. These studies aimed to explore the clinical application of biomaterials and examine the associated complications in orbital implants.
CONCLUSION
We found that using biomaterials did not result in elevated intraocular pressure (IOP). However, we did observe certain complications, with infection, residual diplopia, and enophthalmos being the most frequently reported issues.
Topics: Animals; Humans; Biocompatible Materials; Orbital Fractures; Orbital Implants; Plastic Surgery Procedures
PubMed: 38937319
DOI: 10.1007/s10792-024-03183-w -
In Vivo (Athens, Greece) 2024To investigate the treatment outcomes and determinants of prognosis in patients experiencing visual acuity (VA) deterioration due to inflammatory isolated sphenoid sinus...
BACKGROUND/AIM
To investigate the treatment outcomes and determinants of prognosis in patients experiencing visual acuity (VA) deterioration due to inflammatory isolated sphenoid sinus disease (ISSD) who underwent endonasal endoscopic surgery (EES).
PATIENTS AND METHODS
Thirteen patients with 14 lesions treated with EES between March 2010 and April 2022 were included. Evaluation included improvements in VA using the logarithm of the minimum angle of resolution (LogMAR) scale, resolution rates of associated symptoms, and identification of factors predicting VA recovery. A literature review was conducted to assess the outcomes for ISSD-related VA impairments.
RESULTS
The most common etiology is mycetoma (n=5), followed by an equal representation of mucocele and sphenoiditis (n=4). The mean interval from symptom onset to intervention was 4.7 months, with an average follow-up duration of 14.4 months. Seven eyes exhibited preoperative VA of 2.1 LogMAR or worse, with diplopia/ptosis (n=8) and headache (n=5) being the predominant co-occurring symptoms. After surgery, all ancillary symptoms improved, with an overall VA recovery rate of 87.5% (improvement more than 0.2 logMAR units). Mucocele exhibited the best improvements, whereas sphenoiditis showed the least progress (p=0.021). Poor baseline VA (p=0.026) and combined diplopia/ptosis (p=0.029) were identified as negative prognostic factors for VA recovery.
CONCLUSION
Our findings suggest a favorable prognosis for VA recovery following EES in patients with inflammatory ISSDs, with response variations based on disease entity. However, further research is needed to personalize therapeutic strategies for enhanced outcomes.
Topics: Humans; Female; Male; Middle Aged; Adult; Visual Acuity; Aged; Treatment Outcome; Sphenoid Sinus; Sphenoid Sinusitis; Endoscopy; Prognosis; Young Adult; Inflammation; Vision Disorders
PubMed: 38936949
DOI: 10.21873/invivo.13651 -
Neuro-ophthalmology (Aeolus Press) 2024Isolated paresis of the inferior rectus muscle (IRM) represents a rare occurrence. This case report involves a young adult male who, abruptly during a Valsalva maneuver,...
Isolated paresis of the inferior rectus muscle (IRM) represents a rare occurrence. This case report involves a young adult male who, abruptly during a Valsalva maneuver, encountered acute vertical diplopia due to right IRM paresis, resolving spontaneously within a few hours. The patient presented without identifiable risk factors, and magnetic resonance imaging revealed a minor left thalamic ischemic lesion. A cardiac study identified the presence of a patent foramen ovale, potentially associated with a paradoxical embolism, indicating its likely role in this transient ischemic event.
PubMed: 38933753
DOI: 10.1080/01658107.2024.2308545 -
Neuro-ophthalmology (Aeolus Press) 2024Millard-Gubler syndrome is a pontine syndrome caused by a lesion in the lower pons region. It is characterised by ipsilateral facial paralysis and VI paresis and...
Millard-Gubler syndrome is a pontine syndrome caused by a lesion in the lower pons region. It is characterised by ipsilateral facial paralysis and VI paresis and contralateral brachiocrural palsy. We present the case of a female patient, G4P2A1, at 21 weeks of gestation, with preeclampsia, complaints of blurred vision, diplopia, and right hemiparesis, in whom a clinical diagnosis of Millard-Gubler syndrome was made. Neuroimaging showed an intraparenchymal haemorrhage towards the central portion of the bulbopontine junction. An extensive aetiological study was carried out to determine the cause of the hypertensive disorder syndrome during pregnancy. The patient improved satisfactorily from the neurological deficit after delivery of an early stillbirth.
PubMed: 38933747
DOI: 10.1080/01658107.2024.2303708 -
Journal of Clinical Medicine Jun 2024Blowout fractures are common midfacial fractures in which one or several of the bones of orbital vault break. This is usually caused by a direct trauma to the eye with a...
Blowout fractures are common midfacial fractures in which one or several of the bones of orbital vault break. This is usually caused by a direct trauma to the eye with a blunt object such as a fist. Fracturing of the fragile orbital bones can lead to changes in the orbital volume, which may cause enophthalmos, diplopia, and impaired facial aesthetics. : The aim of this study is to investigate whether there is an association between volume change of the bony orbit and age, gender, or trauma mechanism. : A retrospective study of patients with unilateral blowout or blow-in fractures treated and examined in Päijät-Häme Central Hospital, Lahti, Finland was conducted. Altogether, 127 patients met the inclusion criteria. Their computed tomographs (CT) were measured with an orbit-specific automated segmentation-based volume measurement tool, and the relative orbital volume change between fractured and intact orbital vault was calculated. Thereafter, a statistical analysis was performed. A -value less than 0.05 was considered significant. : We found that relative increase in orbital volume and age have a statistically significant association ( = 0.022). Trauma mechanism and gender showed no significant role. : Patient's age is associated with increased volume change in fractures of the bony orbit.
PubMed: 38930147
DOI: 10.3390/jcm13123618 -
Diagnostics (Basel, Switzerland) Jun 2024Orbital floor fractures (OFFs) represent an interesting chapter in maxillofacial surgery, and one of the main challenges in orbit reconstruction is shaping and cutting...
Classical Orbital Floor Post-Traumatic Reconstruction vs. Customized Reconstruction with the Support of "In-House" 3D-Printed Models: A Retrospective Study with an Analysis of Volumetric Measurement.
BACKGROUND
Orbital floor fractures (OFFs) represent an interesting chapter in maxillofacial surgery, and one of the main challenges in orbit reconstruction is shaping and cutting the precise contour of the implants due to its complex anatomy.
OBJECTIVE
The aim of the retrospective study was to demonstrate, through pre- and postoperative volumetric measurements of the orbit, how the use of a preformed titanium mesh based on the stereolithographic model produced with 3D printers ("In-House" reconstruction) provides a better reconstruction volumetric compared to the intraoperatively shaped titanium mesh.
MATERIALS AND METHODS
The patients with OFF enrolled in this study were divided into two groups according to the inclusion criteria. In Group 1 (G1), patients surgically treated for OFF were divided into two subgroups: G1a, patients undergoing orbital floor reconstruction with an intraoperatively shaped mesh, and G1b, patients undergoing orbital floor reconstruction with a preoperative mesh shaped on a 3D-printed stereolithographic model. Group 2 (G2) consisted of patients treated for other traumatic pathologies (mandible fractures and middle face fractures not involving orbit). Pre- and postoperative orbital volumetric measurements were performed on both G1 and G2. The patients of both groups were subjected to the measurement of orbital volume using Osirix software (Pixmeo SARL, CH-1233 Bernex, Switzerland) on the new CT examination. Both descriptive (using central tendency indices such as mean and range) and regressive (using the Bravais-Pearson index, calculated using the GraphPad program) statistical analyses were performed on the recorded data.
RESULTS
From 1 January 2017 to 31 December 2021, of the 176 patients treated for OFF at the "Magna Graecia" University Hospital of Catanzaro 10 fulfilled the study's inclusion criteria: 5 were assigned to G1a and 5 to G1b, with a total of 30 volumetric measurements. In G2, we included 10 patients, with a total of 20 volumetric measurements. From the volumetric measurements and statistical analysis carried out, it emerged that the average of the volumetric differences of the healthy orbits was ±0.6351 cm, the standard deviation of the volumetric differences was ±0.3383, and the relationship between the treated orbit and the healthy orbit was linear; therefore, the treated orbital volumes tend to approach the healthy ones after surgical treatment.
CONCLUSION
This study demonstrates that if the volume is restored within the range of the standardized mean, the diplopia is completely recovered already after surgery or after one month. For orbital volumes that do not fall within this range, functional recovery could occur within 6 months or be lacking. The restoration of the orbital volume using pre-modeled networks on the patient's anatomical model, printed internally in 3D, allows for more accurate reconstructions of the orbital floor in less time, with clinical advantages also in terms of surgical timing.
PubMed: 38928663
DOI: 10.3390/diagnostics14121248