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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 -
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 -
Plastic and Reconstructive Surgery.... Jun 2024Mucoceles can often present as a complication after prior sinus surgery or maxillofacial trauma when mucosal drainage is obstructed. Their presence in the orbit...
Mucoceles can often present as a complication after prior sinus surgery or maxillofacial trauma when mucosal drainage is obstructed. Their presence in the orbit following orbital wall fracture and subsequent repair represent an exceedingly rare complication whose occurrence is limited to a few reported cases in the literature. In these patients, continuous cyst expansion and subsequent mass effect may lead to ophthalmic symptoms, including orbital pain, proptosis, diplopia, and globe dystopia. We report the discovery of an orbital mucocele after orbital floor fracture repair and its possible association with the nonporous reconstruction plate utilized for fixation. When a patient with history of orbital wall reconstruction presents with new-onset ocular symptoms, an orbital mucocele should be considered as a potential diagnosis.
PubMed: 38911571
DOI: 10.1097/GOX.0000000000005917 -
Journal of Radiology Case Reports 2024Multiple myeloma is a plasma cell neoplasm, which may present as a solitary plasmacytoma and, uncommonly, as an extramedullary plasmacytoma. Intracranial plasmacytomas...
Multiple myeloma is a plasma cell neoplasm, which may present as a solitary plasmacytoma and, uncommonly, as an extramedullary plasmacytoma. Intracranial plasmacytomas may manifest in central nervous system involvement as cranial nerve palsies. Cranial nerve six palsy is the most common in cases of malignancy. However, isolated abducens palsy presenting as multiple myeloma recurrence is very uncommon. Here, we detail two cases in which intracranial plasmacytoma lesions were present within the region of the Dorello canal, resulting in acute isolated unilateral diplopia from disease recurrence in the absence of systemic marrow involvement.
Topics: Humans; Abducens Nerve Diseases; Multiple Myeloma; Neoplasm Recurrence, Local; Male; Magnetic Resonance Imaging; Middle Aged; Aged; Diagnosis, Differential; Plasmacytoma; Female; Diplopia; Brain Neoplasms
PubMed: 38910589
DOI: 10.3941/jrcr.v18i1.5240 -
Journal of Vision Jun 2024Binocular double vision in strabismus is marked by diplopia (seeing the same object in two different directions) and visual confusion (seeing two different objects in... (Review)
Review
Binocular double vision in strabismus is marked by diplopia (seeing the same object in two different directions) and visual confusion (seeing two different objects in the same direction). In strabismus with full visual field, the diplopia coexists with visual confusion across most of the binocular field. With visual field loss, or with use of partial prism segments for field expansion, the two phenomena may be separable. This separability is the focus of this review and offers new insights into binocular function. We show that confusion is necessary but is not sufficient for field expansion. Diplopia plays no role in field expansion but is necessary for clinical testing of strabismus, making such testing difficult in field loss conditions with confusion without diplopia. The roles of the three-dimensional structure of the real world and the dynamic of eye movements within that structure are considered as well. Suppression of one eye's partial view under binocular vision that develops in early-onset (childhood) strabismus is assumed to be a sensory adaption to diplopia. This assumption can be tested using the separation of diplopia and confusion.
Topics: Humans; Vision, Binocular; Visual Fields; Diplopia; Strabismus; Eye Movements
PubMed: 38899959
DOI: 10.1167/jov.24.6.13 -
Internal Medicine (Tokyo, Japan) Jun 2024An 86-year-old woman was admitted to our hospital with cryptogenic progressive dyspnea and dysphagia following a tracheostomy procedure 4 months prior to presentation....
An 86-year-old woman was admitted to our hospital with cryptogenic progressive dyspnea and dysphagia following a tracheostomy procedure 4 months prior to presentation. She exhibited fluctuating diplopia, bilateral vocal fold paralysis, normal nerve test results, negative findings for serum anti-acetylcholine receptor and anti-muscle-specific kinase antibodies, and positive findings for anti-LDL-receptor related protein 4 (LRP4). A videofluoroscopic swallowing study (VFSS) with edrophonium revealed an improvement in bulbar paralysis. Consequently, the patient was diagnosed with double-seronegative myasthenia gravis (DSN-MG) and began immunomodulatory therapy. This case emphasizes the diagnostic challenges of bulbar-type DSN-MG and underscores the value of a VFSS with edrophonium for diagnosing this condition.
PubMed: 38897960
DOI: 10.2169/internalmedicine.3348-23 -
BMJ Open Ophthalmology Jun 2024Graves' ophthalmopathy is a complex autoimmune disorder that can significantly affect quality of life (QoL), vision and physical appearance. Recently, a deeper...
BACKGROUND
Graves' ophthalmopathy is a complex autoimmune disorder that can significantly affect quality of life (QoL), vision and physical appearance. Recently, a deeper understanding of the underlying pathogenesis has led to the development of novel treatment options.
AIMS
The purpose of this review is to explore the current literature on conventional and novel treatment modalities and to evaluate which interventions provide the most favourable psychological and clinical outcomes in patients with moderate to severe, active Grave's ophthalmopathy. For example, QoL is an important psychosocial outcome of disease management. However, available literature demonstrates that not all clinically effective treatment options improve patients' QoL.
METHODS
A systematic literature review was conducted to assess the clinical and psychosocial outcomes of different therapies for Graves' ophthalmopathy. An extensive database search of Ovid Medline, Ovid Embase and Cochrane Central Register of Controlled Trials was conducted. Studies generated were reviewed and the relevant selected data were retrieved and analysed.
RESULTS
Results showed intravenous steroids, rituximab (RTX), tocilizumab and teprotumumab were all significantly effective in improving Clinical Activity Scores. Orbital radiotherapy showed a slight improvement in proptosis and diplopia. All interventions were safe with few serious adverse events being reported across all studies. All treatment modalities demonstrated beneficial improvements in both components of the Graves' Ophthalmopathy-QoL (QoL) questionnaire, apart from orbital radiotherapy which only demonstrated improvements in the visual functioning subscale. Teprotumumab was identified to be the most effective intervention for improving both clinical and psychosocial outcomes. However, further research needs to be conducted to evaluate its side effect profile and cost-effectiveness. Nonetheless, with time it has the potential to be a first-line treatment option in the management of active moderate to severe Graves' ophthalmopathy.
Topics: Humans; Graves Ophthalmopathy; Quality of Life; Antibodies, Monoclonal, Humanized; Rituximab; Immunologic Factors; Glucocorticoids
PubMed: 38886120
DOI: 10.1136/bmjophth-2023-001515