Trigeminal Distribution Research Articles

Ceftazidime-Cefazolin Empiric Therapy for Pediatric Gradenigo Syndrome.

Gradenigo Syndrome (GS), a rare complication of petrous apicitis secondary to acute otitis media, is characterized by (an often incomplete) triad of otorrhea, abducens nerve palsy, and facial pain along the trigeminal nerve distribution. There are several causative pathogens of petrous apicitis, including Streptococcus and Staphylococcus species, while Pseudomonas aeruginosa is the most common. However, the case report literature often describes antibiotic management of GS with antibiotics that do not cover Pseudomonas, potentially predisposing to further intracranial complications or mortality. The purpose of this work was to describe a case of pediatric Gradenigo Syndrome, successfully treated with sufficiently broad-spectrum antibiotics. This is case report. A previously healthy 5-year-old boy with a history of swimming presented with esotropia and acute otitis media. Initial symptoms included otorrhea, otalgia, and pruritis, which were refractory to ciprofloxacin-dexamethasone drops. He subsequently developed a right sixth nerve palsy, suggestive of Gradenigo Syndrome, and neuroimaging showed evidence of petrous apicitis, clival osteomyelitis, and internal carotid artery stenosis. The causative organism was not elucidated to laboratory error. Given this uncertainty, he was successfully treated with empiric intravenous ceftazidime and cefazolin. After 16 weeks, he recovered fully without the need for surgery. In the setting of delayed or absent culture results with suspicion of skull-base infection, our case supports the use of empiric antibiotic therapy with sufficient coverage of all common pathogens including Streptococcus/Staphylococcus and Pseudomonas aeruginosa species, the latter of which is often not adequately covered by antibiotic regimens described in the literature.

Comparison of Different 3D Surface Registration-Based Methods to Assess Facial Asymmetry.

Facial asymmetry is gaining an increasing diagnostic interest in many clinical contexts. Several three-dimensional surface-based methods have been proposed for its assessment; however, they might provide non-equivalent data. Since there is a lack of comparative studies in these terms, this study aims to compare three methods for assessing the asymmetry of the face and facial thirds, thus addressing whether the potential differences can be considered clinically acceptable or not. Two 'maxillofacial' methods based on the trigeminal nerve distribution and one 'orthodontic' method based on reference horizontal planes were used to identify the facial thirds on 3D facial models of 80 Italian healthy adults to calculate the asymmetry of the face, and the upper, middle, and lower thirds of the face differently selected by each method. As a measure of asymmetry, the Root Mean Square value was calculated through a mirroring surface-based registration. Intra- and inter-operator reliability was verified for each method. Differences and interchangeability between the methods were tested, respectively, by two-way repeated measures ANOVA (Analysis of Variance) and Bland-Altman and Similarity Percentage model analysis. Additionally, the time required to perform each method was assessed. All methods demonstrated excellent intra- and inter-operator reliability. While the ANOVA analysis found significant differences (p < 0.001) for the majority of facial Regions of Interest between each method, the Bland-Altman analysis revealed that the differences were clinically acceptable (<0.50 mm) for all facial regions between the trigeminal methods, and for the face and the upper third of the face between the orthodontic method, which was revealed to be faster, and the trigeminal ones. The additional similarity percentage model provided visual support for the complete interchangeability of the two trigeminal methods, as evidenced by the lower Coefficient of Variation value. There is no best method for assessing facial asymmetry that applies to all types of clinical settings, as we have shown that different methods may not be completely interchangeable. However, we suggest that the methods based on the trigeminal subdivision can be used interchangeably in contexts where the morpho-functional analysis of maxillofacial regions with different embryological origins is considered. Thus, the clinical setting imposes the choice of one method over another and, as we have pointed out, the consequent comparison of data with those obtained with methods whose interchangeability has been demonstrated.

A retrospective clinico epidemiological study of pattern of dermatosis involving trigeminal distribution in a tertiary care center

Trigeminal nerve is the principal sensory nerve of face. Many dermatological conditions known occur in relation to trigeminal nerve distribution clinically presenting as unilateral facial lesions with strict midline demarcation. To study the clinical pattern and epidemiological determinants of dermatoses along trigeminal nerve among the patients of both the sexes, involving all the age groups. Thirty two patients presenting with skin lesions along trigeminal nerve presenting to Dermatology department of a tertiary care center, Bangalore, Karnataka, are selected for the study. Study design is retrospective hospital record based study conducted from January 2023 to January 2024. In this study, out of 32 patients most common dermatoses along trigeminal nerve were infection of which herpes zoster was seen thirteen (40.62%) patients, followed by nevi (n=6, 18.75%), pigmentary disorders (n=5, 15.62%), vascular disorder (n=4, 12.5%) and auto inflammatory disorder (n=4, 12.5%) was seen. Most common branch affected was ophthalmic followed maxillary branch .Females were more common and mean age was 24 years. Small sample size and rare clinical cases are included Studies focusing on the pattern of dermatoses involving the trigeminal nerve contribute to the advancement of medical knowledge and facilitate the development of novel diagnostic techniques and therapeutic modalities. By elucidating the underlying pathophysiological mechanisms and identifying potential risk factors, research in this field can lead to the development of more effective treatment strategies tailored to the specific needs of patients.

Neurological and Systemic Pitfalls in the Diagnosis of Cluster Headaches: A Case-Based Review.

To describe different pitfalls in the diagnosis of primary cluster headaches (CHs) with the guidance of seven case vignettes. The question of whether primary CHs and migraines are totally different entities has been long debated. Autonomic features can be detected in as many as 60% of migraine patients. Although some genetic similarities have been found, CACNA1A mutations have not been detected among CH patients with hemimotor aura in contrast to hemiplegic migraine. Recently, functional MRI studies have shown that the left thalamic network was the most discriminative MRI feature in distinguishing migraine from CH patients. Compared to migraine, CH patients showed decreased functional interaction between the left thalamus and cortical areas mediating interception and sensory integration. However, clinically the most significant feature had been the restlessness and agitation seen during headache attacks patients with CHs. This feature is also important in distinguishing cluster patients from other patients having other trigeminal autonomic cephalalgias except for a subset of patients with hemicrania continua. CH is an important member of the group of headache disorders characterized by their association with one or more autonomic features in the trigeminal nerve distribution and termed Trigeminal Autonomic Cephalalgias (TACs). Although CH is a relatively rare condition, judged by the distress it generally causes to the affected individual, early diagnosis and institution of appropriate therapy seem mandatory. Correct diagnosis of CHs needs avoidance of pitfalls. Such pitfalls generally include differentiation from migraine, differentiation from other side locked headache disorders, from other trigeminal autonomic cephalalgias (TACs), and lastly, recognition of rare presentations of cluster-like manifestations with hemiplegic aura and simulating trigeminal and glossopharyngeal neuralgias. Differentiation between primary and symptomatic CHs related to sellar pathologies and systemic medical conditions is of equal importance. In the present review such issues are discussed with the assistance of seven case vignettes.

Intracerebral hemorrhage in Sturge Weber Syndrome: A case report

Background. Encephalotrigeminal angiomatosis also called Sturge Weber Syndrome (SWS) is neurocutaneous abnormality with angioma affecting the leptomeninges and the face skin, commonly in the eyes and maxillary distribution of trigeminal nerve. The characteristic of SWS is dilated face skin veins, and referred to nevus flammeus and port wine stain. Case reports. We report a case of Sturge Weber Syndrome in a 41 years old male with uncontrolled hypertension and smoking, left-sided weakness, and left-sided facial port wine stain. Conclusions. Neurological examination was suggestive of SWS and further radiological examination confirmed the diagnosis.

Role of Magnetic Resonance Imaging in the Evaluation of Trigeminal Neuralgia Using Steady-State Imaging.

Trigeminal neuralgia (TN) poses diagnostic challenges due to its complex origins, often associated with neurovascular compression. Advanced imaging techniques, particularly magnetic resonance imaging (MRI) with thefast imaging employing steady-state acquisition (FIESTA) sequence, offer crucial insights into TN pathophysiology. This prospective cross-sectional observational study aimed to elucidate MRI's utility in diagnosing TN and correlating imaging findings with clinical manifestations and treatment outcomes. A cohort of 41 patients clinically suspected of TN underwent MRI evaluation at Acharya Vinoba Bhave Rural Hospital, Sawangi (Meghe), Wardha, utilizing various sequences including FIESTA. Analysis revealed a higher incidence among females, predominant unilateral presentation, and a higher prevalence of abnormal MRI findings, with neurovascular compression as the leading etiology. Correlation analysis demonstrated significant associations between facial pain localized to the trigeminal nerve distribution, triggering factors, and abnormal MRI findings. Gender distribution did not significantly influence MRI findings. Treatment outcomes favored microvascular surgery over conservative management in cases of neurovascular compression. This study underscores MRI's pivotal role, particularly FIESTA, in TN evaluation, guiding personalized treatment strategies and emphasizing the importance of integrated clinical and imaging approaches. Further research is warranted to validate these findings and explore additional imaging modalities for a deeper understanding of TN pathogenesis.

Thalamic segmentation based on diffusion tensor imaging in patients with trigeminal neuralgia

Classical trigeminal neuralgia (CTN) refers to episodic pain that is strictly confined to the trigeminal distribution area, and the thalamus is an important component of the trigeminal sensory pathway. Probabilistic tracking imaging algorithm was used to identify specific connections between the thalamus and the cortex, in order to identify structural changes in the thalamus of patients with CTN and perform thalamic segmentation. A total of 32 patients with CTN and 32 healthy controls underwent DTI-MRI scanning (3.0 T). Differences in fractional anisotropy (FA), axial diffusivity (AD), radial diffusivity (RD) and mean diffusivity (MD) between the groups were studied. Correlation analysis was performed with clinical course and pain level. Compared to the healthy controls, patients in the CTN group had significantly reduced FA, increased AD, RD and MD in somatosensory subregion of the bilateral thalamus, increased RD in frontal subregion, increased RD and MD in motor subregion. Correlation analysis showed that patient history was positively correlated with pain grading, and that medical history was positively correlated with significantly reduced FA in somatosensory subregion, negatively correlated with increased RD and MD in motor subregion. We used DTI-based probabilistic fiber tracking to discover altered structural connectivity between the thalamus and cerebral cortex in patients with CTN and to obtain a thalamic segmentation atlas, which will help to further understand the pathophysiology of CTN and serve as a future reference for thalamic deep brain stimulation electrode implantation for the treatment of intractable pain.

Facial Onset Sensory and Motor Neuronopathy with Neurotrophic Keratitis: A Case Report

Facial-onset sensory and motor neuronopathy (FOSMN) is a rare, progressive neurodegenerative disorder characterized by sensory loss and motor deficits, primarily affecting the face and upper extremities. The condition begins with sensory impairments in the trigeminal nerve distribution, extending to involve the scalp, neck, upper limbs, and trunk. Symptoms include facial weakness, bulbar disturbances (dysphonia, dysarthria, dysphagia, and sialorrhea), and lower motor neuron signs (weakness, atrophy, fasciculation, and cramps) in the limbs. A hallmark of FOSMN is the diminished or absent corneal reflex, indicating trigeminal nerve involvement and leading to potential complications like neurotrophic keratitis (NK), which can cause corneal ulceration and blindness. The etiology of FOSMN remains uncertain, with theories proposing autoimmune or neurodegenerative origins. Diagnosis is challenging due to the overlap with other ocular surface disorders, necessitating careful examination and differential diagnosis. Management focuses on symptomatic relief, including eye protection to prevent corneal complications. Awareness and early detection of ocular symptoms in FOSMN are crucial for preventing severe outcomes like NK and blindness.

Novel incision to debulk eyelids in a case of Orbital Periorbital Plexiform Neurofibroma (OPPN): A case report

: Plexiform neurofibroma with neurofibromatosis 1 is a autosomal dominant condition and a relatively rare condition. It is also termed as Von Recklinghausen disease. PN involving the structures like eyelid, orbit, periorbital and facial structures are labelled as orbital-periorbital PN(OPPN). These are slowly progressing lesions and are present since birth but increase in size during childhood and are locally infiltrating in nature.: To describe a lid incision to simultaneously debulk both upper and lower eyelid, preserving the vascularity in a case of Orbital Periorbital Plexiform Neurofibroma (OPPN).: We report a case of 22 year male who presented with a disfiguring swelling of right side of face since childhood. After thorough clinical and radiological evaluation it was diagnosed to be a OPPN. Staged debulking was planned and after stage 1 debulking, tissue was sent for HPE which revealed overgrowth of peripheral nerve components and connective tissue dermis showing infiltrating tumour composed of oval to spindle cells with pleomorphic nuclei and moderate cytoplasm. Staged debulking with lid reduction surgery was performed.: Neurofibroma is a rare entity with plexiform neurofibroma occurring in 5-15% of patients. OPPN infiltrates locally without respecting the anatomical planes but follows the trigeminal nerve distribution usually. Indications for debulking procedure are usually the disfigurement and optimal timing is also not certain because the extent and rate of growth cannot be predicted.: The facial appearance and outcome in patients with OPPN can be significantly improved through lid reduction surgery.

MRI evaluation of trigeminal neuralgia patients in a rural hospital of central India

Background Trigeminal neuralgia is characterized by repetitive paroxysmal electric shock-like pain in the trigeminal nerve distribution. The most common cause implicated in this condition is neurovascular conflict, primarily by the superior cerebellar artery in the prepontine cistern. Other common causes include cerebellopontine angle tumours and aneurysms at the brainstem level. Magnetic resonance imaging (MRI) is the imaging modality of choice in evaluating trigeminal neuralgia, with 3D-FIESTA (Fast Imaging employing steady-state acquisition) being the mainstay for assessing neurovascular conflict. This study’s objective is to assess the role of MRI in evaluating trigeminal neuralgia and to study the spectrum of MRI findings associated with various causes. Methods The study type is a cross-sectional observational prospective study conducted at Acharya Vinoba Bhave Rural Hospital, Sawangi, on 41 patients coming to the Radiology department with clinical suspicion of trigeminal neuralgia. Descriptive statistics will summarize participants’ imaging characteristics. At the same time, comparison tests will be employed to assess the association between imaging features and diagnosis of MRI with the distribution of patient symptoms, laterality, aetiology, artery, and branch of trigeminal nerve involved. Considering ethical concerns, participants’ privacy will be protected, and all data will be handled with the utmost confidentiality.

Facial Onset Sensory and Motor Neuronopathy: A Case Series and Literature Review

Introduction: Facial Onset Sensory and Motor Neuronopathy (FOSMN) typically presents with paresthesias in the trigeminal nerve distribution and weakness that progresses rostro-caudally.&#x0D; Objective: To present two new cases of FOSMN, summarize the current literature, and address areas for future study.&#x0D; Methods: Observational data was collected from two patients with FOSMN from our institution. A literature review of FOSMN was completed using PubMed.&#x0D; Results: We identified 100 cases of FOSMN, including our two new cases. 93% presented with facial paresthesias. 97% had bulbar symptoms. Five had family history of ALS. Abnormal Blink reflex was most common on EMG/NCS. CSF was typically normal, but a rare severe case showed elevated protein. Mutations included: TARDBP, OPMD, D90A-SOD1, CHCHD10, VCP, and SQSTM1. Neuropathological studies showed neurodegenerative changes without inflammation. Some cases have reported transient stabilization or improvement to immunomodulatory therapy.&#x0D; Case Reports: A 72-year-old man presented with right-sided trigeminal paresthesias that progressed in a rostro-caudal fashion, dysphagia, and hand weakness. He died 4-5 years after symptom onset. A 69-year-old man presented with left-sided jaw paresthesias, dysphagia and dysarthria. He was trialed on IVIG for 1.5 years without improvement and died 2.6 years after symptom onset.&#x0D; Conclusion: FOSMN is a rare disorder with a unique clinical and electrophysiological phenotype. The pathophysiology has been associated with neurodegeneration and multiple gene mutations have correlated to FOSMN. Some reports suggest transient response to immunomodulatory therapy, though prospective studies are lacking. CSF protein elevation may be seen in severe disease. Future studies will help further elucidate the approach to diagnosis, treatment, and prognostic counseling (biomarkers).&#x0D;

Numb Chin Syndrome: Atypical presentation of metastatic breast cancer

Introduction/Background: Numb chin syndrome (NCS) is an uncommon but known complication of cancer. Prompt recognition is imperative to directed treatment. In some instances, numb chin may be complicated by other findings.&#x0D; &#x0D; Case Report: A 53-year-old female with past medical history of estrogen receptor positive invasive ductal breast carcinoma underwent partial mastectomy, chemotherapy, and radiation. After 6 years remission, new lung and liver metastases occurred, and chemotherapy resumed. Imaging was negative. Eight weeks later, she developed left chin numbness followed by progressive difficulty retaining food and drink. Her left lower lip weakened.&#x0D; &#x0D; Neurological examination revealed left lower lip weakness (showing a “droop” with attempted smile). The area of decreased sensation to sharp touch had expanded from the left chin to the nasolabial fold. The remainder of her neurological examination was unremarkable. Contrast-enhanced MRI was repeated which revealed a 1.9 cm enhancing left parotid mass.&#x0D; &#x0D; Summary/Conclusion: NCS is defined as an ipsilateral loss of chin sensation. If there is no history of trauma or dental injury and especially if the presentation is atypical, then cancer is the most likely etiology. This case began as an uncomplicated NCS that rapidly expanded to involve a greater portion of the trigeminal nerve distribution, in addition to a branch of the facial nerve. Thus, each focal deficit was not the result of separate distal nerve lesions, but rather partial proximal cranial neuropathies at their parotid gland propinquity.

Role of Physical Therapy in Trigeminal Neuralgia: A Case With 4-Year Follow-up

BACKGROUND: Trigeminal neuralgia presents with acute stabbing unilateral facial pain along the nerve distribution precipitated by innocuous sensory stimuli. Trigeminal neuralgia can be unrelenting and can lead to severe physical and emotional consequences. Medical and surgical intervention often has adverse effects, and reports of physical therapy treatment are uncommon. CASE PRESENTATION: The patient in this case presented for physical therapy with a history of head/neck surgery, migraines and headaches, joint pain, and depression, with severe sharp shock-like pain aggravated by innocuous oral activity in the trigeminal nerve distribution. Over 10 sessions, she received manual therapy for joint, myofascial, and nerve mobilization, and upper limb stretching and strengthening. OUTCOME AND FOLLOW-UP: Her pain level decreased substantially and interfered with only 1 oral activity at 4-year follow-up. DISCUSSION: Her presentation may inform the differential diagnosis of craniofacial pain. Her management integrating manual therapy with exercise and the positive clinical outcome suggest a new avenue for treatment of trigeminal neuralgia. JOSPT Cases 2023;3(2):119–126. Epub: 13 April 2023. doi:10.2519/josptcases.2023.11446

Postherpetic pruritus successfully treated with Onabotulinum toxin: A case report (P12-10.003)

<h3>Objective:</h3> Postherpetic pruritus is an underreported but significant sequelae of Herpes zoster infection. It can present isolated or in conjunction with postherpetic neuralgia. <h3>Background:</h3> A 78-year-old woman who presented with Shingles in V1 dermatome distribution of the left trigeminal nerve developed postherpetic neuralgia and pruritus. Standard treatment with antivirals resolved the pain but the postherpetic itching persisted. Trials of antidepressant and nerve pain medications such as Duloxetine and Doxepin couldn’t alleviate the symptoms or were not tolerated. Topical corticosteroid cream provided the only relief at the time. However, it induced profound skin thinning. <h3>Design/Methods:</h3> Due to concomitant blepharospasm, the patient was injected with a pattern Onabotulinum toxin targeting the blepharospasm and additional units were added in the areas of pruritus on the scalp and forehead. <h3>Results:</h3> The result was complete resolution of blepharospasm and dramatic improvement of the itching. The symptoms reoccurred a few months later and subsequent treatments with Onabotulinum toxin were performed, and consistently had notable improvement. The only side effect was post injection bruising after the initial injections which subsided rapidly in the days following. <h3>Conclusions:</h3> In conclusion, Botulinum toxin administration might constitute a valuable option for symptomatic relief of postherpetic itching, especially after the failure of first-choice treatment. <b>Disclosure:</b> Ms. Stoilova has nothing to disclose. Mrs. Torres has received personal compensation in the range of $0-$499 for serving as a Panelist with IQVIA. The institution of Dr. Martinez has received personal compensation in the range of $5,000-$9,999 for serving as a Consultant for Merz. The institution of Dr. Martinez has received personal compensation in the range of $500-$4,999 for serving as a Consultant for Biohaven. The institution of Dr. Martinez has received personal compensation in the range of $500-$4,999 for serving as a Consultant for Amgen/Novartis. The institution of Dr. Martinez has received personal compensation in the range of $500-$4,999 for serving as a Consultant for Abbott. The institution of Dr. Martinez has received personal compensation in the range of $10,000-$49,999 for serving as a Consultant for Abbvie/Allergan. The institution of Dr. Martinez has received personal compensation in the range of $500-$4,999 for serving on a Speakers Bureau for Abbvie/Allergan. The institution of Dr. Martinez has received personal compensation in the range of $5,000-$9,999 for serving on a Speakers Bureau for Merz. The institution of Dr. Martinez has received research support from Medtronic. The institution of Dr. Martinez has received research support from Boston Scientific. The institution of Dr. Martinez has received research support from Abbvie/Allergan.

Trigeminal Somatosensory Evoked Potentials During Neurosurgical Procedures Using Electrical Stimulation of the Lips Under General Anesthesia.

Background Intraoperative neurophysiology monitoring is rapidly evolving with the advent of newer modalities. Long latency sensory evoked potentials from the trigeminal nerve distribution have rarely been demonstrated during neurosurgical procedures. Trigeminal sensory evoked potential (TSEP) can be used to prevent nerve injury during surgical procedures, such as those for trigeminal neuralgia and tumors involving the trigeminal nerve and pathway. Methodology We attempted to record TSEP from 12 subjects who underwent various neurosurgical procedures with low doses of inhalational anesthetic agents. We stimulated the upper and lower lip and recorded from C6 and Fz locations. We used 14-17 mA current stimuli with a pulse width of 50-150 microseconds and a stimulation rate of2.1 Hz. Results We could obtain a clear, reproducible TSEP response in two out of 12 subjects. We observed a TSEP waveform with negative peaks at 13 and 27 milliseconds and a positive wave at around 19 milliseconds. Conclusions The TSEP produced by the electrical stimulation of the upper and lower lip can be detected from the scalp C5, C6, and Fz area even during neurosurgical procedures, even if inhalational anesthesia was used at induction, but only in a small proportion of cases. It appeared to reflect the activity of trigeminal cortical response. Avoiding the notch filter and turning off the inhalational agents are essential for a good response.

Facial Asymmetry of Italian Children: A Cross-Sectional Analysis of Three-Dimensional Stereophotogrammetric Reference Values

Reference data on the asymmetry of facial thirds of children is still scarce, although it can offer meaningful comparative information for clinical studies. This study aims to provide reference data on the facial asymmetry of Italian children using a 3D analysis of facial thirds divided according to the trigeminal nerve distribution (upper, middle, and lower). A 3D surface-based approach was conducted on the digital models of the faces of 135 children (74 M, 61 F), acquired by stereophotogrammetry. In addition to sex, two different age classes were analyzed (4–8 years and 9–12 years). For each facial third, the asymmetry was expressed as root-mean-square distance (RMS) by calculating the point-to-point distances between the original and the reflected 3D models. A 3-way ANOVA test verified significant differences between the two sexes, the two age classes, and the three facial thirds (p-value &lt; 0.05), and also their interaction. Significant differences were found between the sexes (females were more symmetric, p = 0.005) and the two age groups (younger were more symmetric p &lt; 0.001). According to Tukey’s HSD post-hoc test, among the thirds, the middle one proved to be significantly more symmetrical (p &lt; 0.001). No significant interaction impacting the asymmetry was found when the main factors were considered in any combination. Reference data on the “normal” facial asymmetry of Italian children was provided for further clinical purposes. Normal children have low average RMS values (0.30–0.51 mm) and younger, in particular females, proved more symmetrical than older children, while among the facial surfaces, the middle proved most symmetrical in both sexes, although with little clinical relevance. Since there is no consensus on the amount of symmetry deemed clinically acceptable, further studies on larger randomized samples are auspicial.

A case report on type I sturge weber syndrome with bilateral port-wine stain

Sturge-Weber syndrome (SWS) is a type of neuro-cutaneous diseases which is seen in 1 in 50,000 populations and affects both the sexes equally. It usually manifests with a facial Port Wine Stain (PWS). Facial port-wine stains are usually isolated findings, however, when associated with cerebral and ocular vascular malformations they form part of the classical triad of Sturge-weber syndrome (SWS). The prevalence of PWS is estimated at three to five children per 1000 live births; there are ~26 million people worldwide with PWS birthmarks. The majority of facial PWS (~90%) are unilateral in a trigeminal dermatomal distribution. Here, we report a case of 39 years old female with SWS who presented with classical triad of SWS i.e., bilateral port-wine stain, epilepsy and severe glaucoma.

Carotid-Cavernous Fistula: Hemodynamic Dysfunction after Shingles? A Case Report and Review Literature

The patient, a 67-year-old female, presented with esotropia, diplopia, and eye redness, as seen in Figure 1. The diplopia started two months prior to the initial visit while the eye started to crossed in. The patient also complained of severe daily headaches along with ringing in her ears. She believes the symptoms started while she was recovering from shingles on her right side of the forehead. Prior MRI imaging was reported to be unremarkable. The patient was referred to neuro-ophthalmology service with possible diagnosis of thyroid eye disease given the red eye and esotropia. Patient had no history of other medical conditions except shingles in trigeminal distribution that occurred about 4-6 weeks before the onset of her symptoms. Central VA was recorded to be unaffected at 20/20 in both eyes with correction. On sensorimotor exam, there was severe horizontal diplopia at the primary, increasing in left, and right gazes due to bilateral VI nerve palsy. Intraocular pressure testing 22mmHg right eye and 19mmHg left eye, indicating ocular hypertension in the right eye. She had corkscrew blood vessels in both eyes. Optical coherence tomography (OCT) revealed normal retinal nerve fiber layer (RNFL) thickness in both eyes.

NCMP-20. NUMB CHIN SYNDROME: ATYPICAL PRESENTATION OF METASTATIC BREAST CANCER

Abstract OBJECTIVES 1. To report a variation of Numb Chin Syndrome (NCS) 2. Raise awareness and early localization of NCS to more efficiently develop a targeted treatment plan to prevent tumor spread and potentially improve clinical outcomes and survival. INTRODUCTION/BACKGROUND NCS is an uncommon but known complication of cancer. Prompt recognition is imperative to directed treatment. In some instances, numb chin may be complicated by other findings. CASE REPORT A 53-year-old female with past medical history of estrogen receptor positive invasive ductal breast carcinoma underwent partial mastectomy, chemotherapy, and radiation. After 6 years remission, new lung and liver metastases occurred, and chemotherapy resumed. Imaging was negative. Eight weeks later, she developed left chin numbness followed by progressive difficulty retaining food and drink. Her left lower lip weakened. Neurological examination revealed left lower lip weakness (showing a “droop” with attempted smile). The area of decreased sensation to sharp touch had expanded from the left chin to the nasolabial fold. The remainder of her neurological examination was unremarkable. Contrast-enhanced MRI was repeated which revealed a 1.9 cm enhancing left parotid mass. SUMMARY/ CONCLUSION NCS is defined as an ipsilateral loss of chin sensation. If there is no history of trauma or dental injury and especially if the presentation is atypical, then cancer is the most likely etiology. This case began as an uncomplicated NCS that rapidly expanded to involve a greater portion of the trigeminal nerve distribution, in addition to a branch of the facial nerve. Thus, each focal deficit was not the result of separate distal nerve lesions, but rather partial proximal cranial neuropathies at their parotid gland propinquity.

ODP318 Idiopathic Granulomatous Hypophysitis : A Rare Cause of Hypopituitarism

Abstract Introduction Pituitary hypophysitis is an inflammatory disorder classified as primary or secondary. The five histologic subtypes of primary hypophysitis are lymphocytic, granulomatous, xanthomatous, IgG4-related, and necrotizing. Idiopathic granulomatous hypophysitis (IGH) has an incidence of 1 in 10 million and accounts for &amp;lt; 1% of cases involving panhypopituitarism, headache, and visual disturbances. We present the case of IGH in a patient initially suspected to have pituitary adenoma. Case Presentation A 54-year-old female with a history of hypertension presented to the emergency department with nausea, vomiting, and acute worsening of a chronic headache of 1-week duration. Her past medical history was significant for a suspected pituitary macroadenoma of 1.7 cm, which was incidentally detected 7 months before the current presentation. She reported a history of 9 lb weight gain in one month, hot flashes, and diplopia, but denied hirsutism, polyuria, polydipsia, and galactorrhea. Prior evaluation revealed normal prolactin, TSH, and visual field testing. Her medication and familial history were unremarkable. Physical exam during the current presentation revealed stable vital signs, left eye exophthalmos, left anisocoria, decreased sensation in the left V1 and V2 trigeminal nerve distribution, and right abducens nerve palsy. Her initial laboratory workup was consistent with panhypopituitarism. Brain Magnetic Resonance Imaging (MRI) revealed an extensive, locally invasive, exuberantly enhancing infiltrative process measuring at least 5.6 cm in maximal transverse diameter involving the pituitary gland with bilateral cavernous sinus and Meckel's caves invasion and extension into bilateral superior orbital fissures. The patient underwent supraorbital craniotomy with subtotal resection of the tumor. Tuberculosis, sarcoidosis, and neoplasm were ruled out by appropriate tests, and the patient was diagnosed to have IGH on histology. Her symptoms improved postoperatively and the patient was discharged on levothyroxine and hydrocortisone. Conclusion Clinically and radiologically, IGH is a rare pathology that can closely mimic a pituitary adenoma. Through this case, we highlight the importance of performing a comprehensive evaluation with a full hormonal workup and imaging, with the exclusion of secondary causes while assessing patients with a sellar mass. Presentation: No date and time listed