Lethal mycotic pseudoaneurysm presenting as isolated sixth nerve palsy.

Pseudoaneurysm of the internal carotid artery caused by skull base osteomyelitis (SBO) is a lethal condition seen in immunocompromised patients, predominantly those with diabetes mellitus. Cranial nerve involvement is a common complication and generally indicates a poor prognosis. We report the case of a 62-year-old diabetic patient who presented with isolated sixth cranial nerve palsy. She had uncontrolled blood sugar levels and high erythrocyte sedimentation rate, and she suffered from pyelonephritis. Neuroimaging detected SBO with multiple secondary mycotic pseudoaneurysms prominent at the petrocavernous junction. Ischemia is the most common etiology for an isolated abducens nerve palsy, but in certain cases neuroimaging is warranted to prevent life-threatening complications. This case highlights the importance and urgency of identifying and managing such conditions.

Fusobacterium necrophorum an Underrecognized Cause of Petrous Apicitis Presenting with Gradenigo Syndrome: A Case Report.

BACKGROUND With the advent of antibiotics, petrous apicitis (PA), inflammation of the petrous temporal bone, has become a rare complication of otitis media. Even more uncommon is Gradenigo syndrome (GS), a result of PA, characterized by a triad of otitis media or purulent otorrhea, pain within the regions innervated by the first and second division of the trigeminal nerve, and ipsilateral abducens nerve palsy. Recent literature has demonstrated increasing reports of Fusobacterium necrophorum isolated in cases of GS. CASE REPORT A 21-year-old man presented with otalgia, reduced hearing, and severe headache. Examination revealed right-sided purulent otorrhea, anesthesia within the trigeminal nerve distribution, and an ipsilateral abducens nerve palsy. F. necrophorum was isolated from an ear swab and a blood culture. Computed tomography and magnetic resonance imaging (MRI) demonstrated otomastoiditis, PA, cavernous sinus thrombosis, and severe stenosis of the petrous internal carotid artery. He was treated with intravenous benzylpenicillin, underwent a mastoidectomy and insertion of a ventilation tube, and was started on a 3-month course of dabigatran. Interval MRI showed improved internal carotid artery caliber, persistent petrous apex inflammation, and normal appearance of both cavernous sinuses. Follow-up clinical review noted persistent abducens and trigeminal nerve dysfunction. CONCLUSIONS We identified 190 cases of PA; of these, 80 presented with the classic Gradenigo triad. Fusobacterium sp. were cultured in 10% of GS cases, making them the most frequent isolates. Due to the fastidious nature of F. necrophorum, it may be underrepresented in the historical literature, and we recommend that empiric antibiotics cover anaerobic organisms.

Spinal procedures, pneumocephalus, and cranial nerve palsies: A review of the literature.

Purpose: Minimally invasive and surgical spine procedures are commonplace with various risks and complications. Cranial nerve palsies, however, are infrequently encountered, particularly after procedures such as lumbar punctures, epidural anesthesia, or intrathecal injections, and are understandably worrisome for clinicians and patients as they may be interpreted as secondary to a sinister etiology. However, a less commonly considered source is a pneumocephalus which may, in rare cases, abut cranial nerves and cause a palsy as a benign and often self-resolving complication. Here, we present the case of a patient who underwent an intrathecal methotrexate infusion for newly diagnosed non-Hodgkin's T-cell lymphoma and subsequently developed an abducens nerve palsy due to pneumocephalus. We highlight the utility of various imaging modalities, treatment options, and review current literature on spinal procedures resulting in cranial nerve palsies attributable to pneumocephalus presenting as malignant etiologies.

Gradenigo Syndrome in a 6-Year-Old Boy with Acute Otitis Media: A Case Report.

Gradenigo Syndrome (GS) is not a common intracranial complication of acute otitis media infections that needs proper diagnosis and management. This case report presents a 6-year-old child diagnosed with unilateral headache, right eye diplopia, and right abducens nerve palsy. MRI and cranial nerve examination facilitate the confirmation of GS. The patient was treated with proper antibiotics and showed good outcomes. This case report highlights the importance of early detection and understanding of the clinical picture associated with GS, which have a crucial role in achieving desired outcomes and avoiding serious complications.

COVID-19 and sudden-onset ocular neurogenic palsy in prior healthy patients: a systematic review.

BACKGROUND: The aim of this systematic review is to identify cases of neurogenic ocular palsy in the presence of COVID-19 and to document patient characteristics, type of palsy and possible aetiologies. METHODS: A systematic search of PubMed, Medline and CINAHL databases was conducted on the 6th of January 2023 to identify cases of neurogenic ocular palsy in patients with current or previous COVID-19 infection. Data were pooled to summarise the neurogenic palsy, patient clinical characteristics and proposed palsy mechanisms. RESULTS: The combined database search yielded 1197 articles. Of these, 23 publications consisting of 25 patients met the inclusion criteria. Most patients were male (68%) and ranged in age from 2 to 71 years (median=32.7, SD=21.4). Seven patients (28%) were children aged 2 to 10 years old. Abducens palsies were most common (68%) and the most common ocular presentation was diplopia (76%) with an average time of onset 15 days from testing positive to COVID-19 or having symptoms of the virus. Proposed mechanism of development of a neurogenic palsy secondary to COVID-19 infection was classified into one of three categories: vascular/thrombotic, a viral neuro-invasive or inflammatory virus-mediated immune response. DISCUSSION: This study suggests that COVID-19 infection may be linked to oculomotor, trochlear and abducens nerve palsies and the underlying mechanisms may vary but are difficult to definitively establish. Further studies investigating the onset of neurogenic palsy secondary to COVID-19 infection is required.

Sixth cranial nerve palsy and dense cerebral venous sinus thrombosis in a child with nephrotic syndrome.

Nephrotic syndrome (NS) is a common glomerular disease characterised by massive proteinuria, hypoalbuminaemia, oedema and hyperlipidaemia. Cerebral venous sinus thrombosis (CVST) is a rare complication in children with NS. Here, we report a case of relapsing NS on steroid therapy, in a male in early childhood who presented with symptoms of headache, vomiting and double vision. On prism cover test there was 25 PD esotropia with abduction restriction in the left eye. Fundus examination showed bilateral papilloedema. He was diagnosed as sixth cranial nerve palsy of left eye. Neuroimaging reported dense CVST. He was managed with subcutaneous low molecular weight heparin and steroids. After 2 months of treatment, there was a complete resolution of esotropia and optic disc oedema. This case highlights the importance of early diagnosis of acute onset esotropia and sagittal sinus thrombosis in a case of NS.

Acute abducens nerve palsy with acute disseminated encephalomyelitis-like presentation following COVID-19 vaccination.

We report two adult cases of abducens nerve palsy presenting immediately (within weeks) after they received the first dose of Covishield vaccination. Magnetic resonance imaging (MRI) of the brain obtained after the onset of diplopia demonstrated demyelinating changes. The patients had associated systemic symptoms. Post-vaccination demyelination typically known as acute disseminated encephalomyelitis (ADEM) associated with several vaccines is more common in children. Although the mechanism of the nerve palsy remains unclear, it is suspected to be related to the post-vaccine neuroinflammatory syndrome. Cranial nerve palsies and ADEM-like presentations may represent part of the neurologic spectrum following COVID-vaccination in adults, and ophthalmologists should be aware of these sequelae. Although cases of sixth nerve palsy following COVID vaccination are already reported, associated MRI changes have not been reported from India.

Bilateral sixth nerve palsy with subdural hematoma: a unique presentation of B12 deficiency.

Vitamin B12 is inextricably associated with the development and maintenance of neuronal functions. It is classically associated with subacute combined degeneration and peripheral neuropathy; however, cranial neuropathy is uncommon. We observed the rarest neurological manifestation of B12 deficiency. A 12 months infant had history of lethargy, irritability, anorexia, paleness, vomiting, and neurodevelopmental delay for 2 months. He also developed inattention and altered sleep pattern. His mother noticed bilateral inward rotation of both eyes. On examination, the infant had bilateral lateral rectus palsy. The infant was found to have anemia (7.7g/dL) and severe B12 deficiency (74pg/mL). On MRI, there was cerebral atrophy, subdural hematoma (SDH) and wide cisternal spaces and sulci. On supplementation with cobalamin, he improved clinically though mild restriction of lateral gaze on the left side persists. Follow up MRI showed significant improvement in cerebral atrophy with resolution of SDH. To date, such clinical presentation of B12 deficiency has never been reported. The authors suggest B12 supplementation for at risk population esp at antenatal stage and lactating mothers in national programs. The treatment of this condition should be initiated early to prevent long term sequelae.

Diagnosis of idiopathic intracranial hypertension: A proposal for evidence-based diagnostic criteria.

BACKGROUND: Based on expert opinion, abducens nerve palsy and a neuroimaging criterion (≥3 neuroimaging signs suggestive of elevated intracranial pressure) were added to the diagnostic criteria for idiopathic intracranial hypertension. Our objective was to validate this. METHODS: This prospective study included patients with new-onset idiopathic intracranial hypertension for a standardized work-up: interview, neuro-ophthalmological exam, lumbar puncture, neuroimaging. Neuroimaging was evaluated by a blinded neuroradiologist. RESULTS: We included 157 patients classified as idiopathic intracranial hypertension (56.7%), probable idiopathic intracranial hypertension (1.9%), idiopathic intracranial hypertension without papilledema (idiopathic intracranial hypertension-without papill edema; 0%), suggested idiopathic intracranial hypertension-without papill edema (4.5%), or non-idiopathic intracranial hypertension (36.9%). Moderate suprasellar herniation was more common in idiopathic intracranial hypertension than non-idiopathic intracranial hypertension (71.4% versus 47.4%, p < 0.01), as was perioptic nerve sheath distension (69.8% versus 29.3%, p < 0.001), flattening of the globe (67.1% versus 11.1%, p < 0.001) and transverse sinus stenosis (60.2% versus 18.9%, p < 0.001). Abducens nerve palsy was of no diagnostic significance. Sensitivity of ≥3 neuroimaging signs was 59.5% and specificity was 93.5%. CONCLUSION: Moderate suprasellar herniation, distension of the perioptic nerve sheath, flattening of the globe and transverse sinus stenosis were associated with idiopathic intracranial hypertension. We propose that idiopathic intracranial hypertension can be defined by two out of three objective findings (papilledema, opening pressure ≥25 cm cerebrospinal fluid and ≥3 neuroimaging signs).

Pearls & Oy-sters: Trigeminal Cystic Schwannoma Presenting With Foster Kennedy Syndrome, Sixth Nerve Palsy, and Focal Seizures.

Foster Kennedy syndrome refers to a finding of optic atrophy in one eye from direct compression of the optic nerve by a mass lesion and contralateral papilledema in the nonatrophic optic nerve caused by an increased intracranial pressure. When the fundoscopy finding is not due to a direct compressive mass, the term pseudo-Foster Kennedy syndrome is used; this can be caused by any process or processes that result in optic atrophy in one eye and optic disc edema in the other. Identifying Foster Kennedy syndrome in a patient calls for expedient neuroimaging looking for an intracranial mass lesion. In this article, we present the case of a patient presenting with vision loss and Foster Kennedy syndrome who was found to have a large trigeminal cystic schwannoma. While several other accompanying symptoms were not evident from the patient complaint, a careful history and physical examination revealed additional localizing clues: unilateral sensory changes in the face and pterygoid and masseter atrophy, unilateral cranial nerve VI palsy, and episodes of intense déjà vu sensation, which were presumed to represent temporal lobe-onset focal aware seizures. Trigeminal schwannomas are a rare entity, and they are even more rarely cystic. This case highlights an unusual scenario where a slow expansion of the tumor ultimately resulted in vision loss and presentation of the patient to medical attention.

A comparative multicentric long-term study of un-augmented modified Nishida procedure vs augmentation in unilateral sixth nerve palsy.

PURPOSE: To compare the effectiveness of three procedures: modified Nishida procedure alone vs modified Nishida procedure combined with medial rectus recession (MRc) vs modified Nishida procedure combined with MRc and botulinum toxin (BT) for severe unilateral sixth nerve palsy. DESIGN: Consecutive, interventional case series. METHODS: The medical records of a consecutive series of patients with severe unilateral sixth nerve palsy who underwent modified Nishida procedure in multiple centres were reviewed. Surgical technique was decided preoperatively at the surgeon's discretion. The preoperative and postoperative findings were compared. RESULTS: Of the 43 patients with abducens palsy that received the procedure, 32 were included (mean age 38.6 ± 19.8 years). Mean preoperative deviation was 63.0 ± 27.3 prism dioptres (PD) and mean limitation of abduction -4.5 ± 1.2. Five patients underwent a modified Nishida procedure alone, 24 patients had an additional MRc and 3 patients were also injected with BT. Overall, the average correction of modified Nishida technique by itself was 29.4 ± 6.6 PD (range 20-36) and adding a MRc corrected 62.6 ± 23.8 PD (range 24-120). Modified Nishida procedure, MRc and BT altogether corrected 95.0 ± 18.0 PD (range 75-110). No postoperative complications were observed in any of the patients. CONCLUSIONS: Excellent outcomes with fewer complications are obtained with modified Nishida procedure alone. The need for additional procedures such as MRc and BT which increase the effect in primary position can be determined depending on passive duction and preoperative horizontal deviation.

Diplopia in a Child: Gradenigo Syndrome Is an Unforgettable Disease.

Gradenigo syndrome is a clinical triad of abducens nerve palsy, retro-orbital pain (trigeminal ganglionitis), and chronic otorrhea (otitis media). The etiology of Gradenigo syndrome results from apical petrositis secondary to suppurative otitis media. Although apical petrositis has gradually become uncommon in modern society due to the widespread use of antibiotics, Gradenigo syndrome should be considered in the differential diagnosis of a child's diplopia.

Síndrome de Moebius y ortotropía en posición primaria: ¿es infrecuente esta asociación? (Colombia) / Moebius syndrome and orthotropia in primary position: is this association uncommon? (Colombia) / Síndrome de Moebius e ortotropia em posição primária: é uma associação incomum? (Colômbia)

El síndrome de Moebius es una enfermedad congénita poco común que se caracteriza por el compromiso unilateral o bilateral del VI y VII par craneal, lo que compromete los músculos que controlan la oculomotricidad, produciendo una parálisis en la abducción del globo ocular y los músculos involucrados en la expresión facial. Su presentación clínica y grados de severidad son variables, puede presentar compromiso simétrico o asimétrico. Adicionalmente, gran parte de los casos se relacionan con trastornos del lenguaje, anomalías musculoesqueléticas y orofaciales. En el presente artículo se presenta el caso de una paciente femenina de 3 años producto de un embarazo trigemelar con diagnóstico clínico de síndrome de Moebius al nacer, confirmado por neuroimagen en la que se evidencia la ausencia bilateral del nervio facial en ángulos pontocerebelosos, adicionalmente con un defecto completo en los movimientos oculares de abducción y aducción lo que impide el estrabismo convergente común en estos pacientes.

Moebius syndrome is a rare congenital disease characterized by unilateral or bilateral involvement of the VI and VII cranial nerves, which compromises the muscles that control ocular motricity with paralysis in the abduction of the eyeball and the muscles involved in the facial expression. Its clinical presentation and degrees of severity are variable, and it can be symmetric or asymmetric. Additionally, most of the cases are related to language disorders, musculoskeletal and orofacial anomalies. This paper presents the case of a 3-year-old female patient, product of a trigemellar pregnancy with a clinical diagnosis of Moebius syndrome at birth, confirmed by neuroimaging, which shows the bilateral absence of the facial nerve in point-lateral angles. Additionally she has a complete defect in abduction and adduction eye movements, which prevents the common convergent strabismus in these patients.

A síndrome de Moebius é uma doença congênita rara caracterizada pelo envolvimento unilateral ou bilateral dos nervos cranianos VI e VII, que compromete os músculos que controlam a oculomotricidade com paralisia na abdução do globo ocular e dos músculos envolvidos na expressão facial. Sua apresentação clínica e graus de gravidade são variáveis, podendo ser um comprometimento simétrico ou assimétrico. Além disso, a maioria dos casos está relacionada a distúrbios de linguagem, anomalias musculoesqueléticas e orofaciais. Este paper apresenta o caso de uma paciente de 3 anos de idade, fruto de uma gravidez trigêmea com diagnóstico clínico de Síndrome de Moebius ao nascimento, confirmado por neuroimagem em que é evidente a ausência bilateral do nervo facial nos ângulos ponto-cerebelares. Além disso, ela tem um defeito completo nos movimentos oculares de abdução e adução, o que impede o estrabismo convergente comum nesses pacientes.

[CASE SERIES OF EARLY NEUROIMAGING IN PATIENTS WITH VASCULAR RISK FACTORS AND ISOLATED SIX CRANIAL NERVE PALSY, DEMONSTRATING THE DILEMMA OF EARLY IMAGING].

INTRODUCTION: There are various etiologies for isolated third, fourth and sixth cranial nerve palsies. The most common etiology in adults aged 50 years and older with vascular risk factors such as diabetes, hyperlipidemia, and hyperlipidemia, is microvascular ischemia. The role of early neuroimaging in older patients with vascular risk factors presenting with acute isolated ocular motor nerve palsy is controversial. AIMS: We present six cases of patients aged 50 years and older, with vascular risk factors, who suffered from acute isolated paralysis of the sixth nerve. All cases were diagnosed with a non-microvascular ischemia etiology. RESULTS: The identified etiologies included petroclival meningioma with involvement of the cavernous sinus, metastatic hepatoma, plasmacytoma, venous thrombosis, arterial-venous malformation and aneurysm. CONCLUSIONS: Despite the high cost and presumed low yield, early neuroimaging can change the clinical management in certain cases. DISCUSSION: Previous studies have found the yield of early neuroimaging in older vasculopathic patients with acute 4th or 6th cranial nerve palsies to be 1-15%. In spite of these reports, our cases demonstrate the need for early onset imaging in these cases.

[Clinical observation of superior rectus transposition with/without augmented suture and vertical rectus transposition for the treatment of strabismus caused by complete abducens nerve palsy].

Objective: To evaluate the efficacy and safety of superior rectus transposition (SRT) with/without augmented suture and vertical rectus transposition (VRT) for the treatment of strabismus caused by complete abducens nerve palsy. Methods: This was a retrospective cohort study. Forty-two patients (42 eyes) with complete abducens nerve palsy underwent strabismic surgeries from January 2015 to November 2020 in Tianjin Eye Hospital. According to the different procedures, the patients were divided into three groups: SRT group (16 cases, SRT with medial rectus recession), superior rectus transposition with augmented suture (SRTA) group (13 cases, SRT with Buckley suture and medial rectus recession) and VRT group (13 cases). The preoperative and postoperative (1, 6 and 12 months) data including deviations, ocular motility, binocular vision and surgical complications among three groups were analyzed and compared. χ2 test was used for comparison of count data among three groups. The measurement data were compared among three groups by the repeated measures ANOVA. LSD-t test was used for within-group comparison and between-group comparison. Results: There was no difference in sex ratio, age and course of disease among the groups (all P>0.05). The horizontal deviations of the three groups at 1, 6 and 12 months after surgeries was lower than that before surgeries, and the difference was statistically significant (all P<0.001). The horizontal deviations of the SRT group, SRTA group and VRT group at 12 months after surgeries were (+0.8±5.8), (+0.8±4.5), (+1.2±2.5) prism diopters (PD), respectively, lower than that of the preoperative (+82.8±17.2), (+77.7±26.1), (+71.5±18.6) PD. However, there was no significant difference among different postoperative follow-up timepoints (all P>0.05). There was no difference in horizontal deviations before surgeries and at 1, 6 and 12 months after surgeries among three groups (P>0.05). There were significant differences in the scales of abduction motility among preoperative, postoperative 1, 6 and 12 months measurements for three group (all P<0.001). The scales of abduction before surgeries in the SRT group, SRTA group, and VRT group were (-4.4±0.5), (-4.4±0.5), (-4.5±0.5) scale and at 12 months after surgeries were (-2.3±0.7), (-2.2±0.5), (-2.1±0.6) scale respectively. But there was no change among different postoperative follow-up timepoints (all P>0.05). Preoperative and postoperative 1-, 6-and 12-month abduction motility was similar among three groups (P>0.05). There were significant differences in the scales of adduction limitation among preoperative, postoperative 1-, 6-and 12-months measurements for three group (all P<0.05). But there was no change among different postoperative follow-up timepoints (all P>0.05). There were significant differences between the SRT group [(-0.9±0.6), (-0.8±0.6) scale] and the SRTA groups [(-1.5±0.5), (-1.4±0.5) scale] (t=-2.62, -2.52) and between the SRTA group and the VRT group [(-0.8±0.8), (-0.6±0.7) scale] (t=2.62, 3.01) at 6 and 12 months after surgeries (all P<0.05). The outcomes of binocular vision at postoperative 12 months were similar among three groups (P>0.05). No patient had torsional diplopia and anterior segment ischemia. Only 2 patients from the SRTA group had hypotropia of 4 to 5 PD in the primary position associated with supraduction limitation. Conclusions: SRT with/without augmented suture and VRT are effective and safe procedures for the treatment of strabismus caused by complete abducens nerve palsy. They could correct deviations, improve abduction motility and restore binocular vision, with stable outcomes and a small risk of vertical and torsional diplopia.

Abducens and Trochlear Nerve Palsies After COVID-19 Vaccination: Report of Two Cases.

The authors report two cases of an abducens palsy and a trochlear nerve palsy, respectively, in two patients who received a coronavirus disease 2019 (COVID-19) vaccine 2 weeks previously. Given the lack of other symptoms, normal test results, and spontaneous resolution of the diplopia, a likely association with the COVID-19 vaccine was suggested. [J Pediatr Ophthalmol Strabismus. 2022;59(5):e50-e53.].

Abducens Nerve Palsy as a Presenting Symptom of Multiple Sclerosis.

Multiple sclerosis (MS) is a chronic disorder characterized by demyelination of the central nervous system. It often presents in women aged 18-35 with neurological symptoms such as visual loss, paresthesia, focal weakness, and ataxia. Demyelination in the brainstem can result in internuclear ophthalmoplegia causing binocular horizontal diplopia. Our report details a patient with horizontal diplopia from an isolated abducens (sixth) nerve palsy as the initial symptom of MS. While rare, this demonstrates the importance of including MS in the differential diagnosis for an isolated abducens nerve palsy, especially in younger patients with no known vascular risk factors.

A rare cause of acquired esotropia: Leprosy.

Leprosy is an infective chronic granulomatous disease involving the skin and peripheral nerves caused by Mycobacterium leprae. Complications such as lagophthalmos, corneal opacity and uveitis are common, but cranial nerve involvement is rarely seen. The fifth and seventh cranial nerves are most commonly involved. We report a case of acquired esotropia due to sixth nerve palsy following a rare cranial nerve involvement by leprosy.

Petrous Apicitis: A Systematic Review and Case Presentation.

INTRODUCTION: Petrous apicitis (PA) is a rare but dangerous complication of acute otitis media. The objective of this study is to present a case of PA and systematically review the existing literature on PA to characterize clinical presentation, diagnosis, management, and outcomes in the antibiotic era. METHODS: A comprehensive search from 1983 to June 1, 2020, of PubMed, MEDLINE, Cochrane Library, and EmBase databases was conducted. Studies with clinical data regarding patients with PA were included. Non-English literature or studies with insufficient individual patient data were excluded. Sixty-seven studies were included with a total of 134 patients. RESULTS: A total of 67 articles were found to meet criteria for inclusion. The mean age of presentation was 33 years. Recent acute otitis media was reported in 78 patients (58.2%). Only 3 patients (2.2%) were immunocompromised, and 8 patients (6.0%) had a history of diabetes. Gradenigo's triad of abducens palsy, otorrhea, and retro-orbital or facial pain was reported in 28 patients (20.9%); however, these presenting symptoms were common individually (51.5%, 48.5%, and 64.2%, respectively). Hearing loss (35.8%), facial weakness (17.9%), and vertigo (7.5%) were also reported.The most frequently cultured pathogen was Pseudomonas (34.2%), followed by Streptococcus and Staphylococcus. All 134 patients underwent imaging, with computed tomography being the most frequently used modality (56.0%). Nearly all patients received antibiotic therapy (95.6%), with 91 (67.9%) undergoing surgery ranging from myringotomy (26.9%) to petrosectomy (25.4%). Five patients (5.7%) died because of complications related to PA. Mean follow-up was 11.0 months. CONCLUSIONS: Petrous apicitis has a variable presentation with potential for severe morbidity. Mortality rates are low, and presentation with Gradenigo's triad is uncommon. Appropriate medical management with surgical drainage can avoid long-term sequelae.

Scrub Typhus Presenting as Unilateral Abducens Nerve Palsy: A Case Report.

Abducens nerve palsies associated with infectious diseases are rare. Scrub typhus is an acute, febrile, infectious illness caused by Orientia tsutsugamushi carried out by vector mite zoonosis and is highly endemic in the so-called "tsutsugamushi triangle". The organism has been reported to be capable of entering the nervous system, causing meningitis and focal neurologic abnormalities. We report a 23 years old previously healthy girl who presented with fever, pain abdomen, vomiting and classical pathognomic black eschar mark on the right proximal medial calf region. After exclusion of other common infectious causes, scrub typhus serology immunoglobulin M was positive and was diagnosed with scrub typhus associated with unilateral abducens nerve palsy which responded to doxycycline therapy. On the background of strong clinical suspicion, we underline its significance in the interpretation of the serologic testing and its role in guiding the further treatment respectively. Keywords: abducens nerve palsy; case report; Orientia tsutsugamushi; scrub typhus; zoonoses.