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PURPOSE: To characterize the size of extraocular muscles (EOMs) in a pediatric population with thyroid dysfunction using orbital echography. METHODS: Patients under age 18 with thyroid dysfunction who presented to an academic ophthalmology department from 2009 to 2020 and received orbital echography were included in this IRB-approved retrospective study. Data collected included age, clinical activity score (CAS), thyroid stimulating immunoglobulin (TSI), and extraocular recti muscle thickness on echography. Patients were organized into three age cohorts, after which statistical analysis compared recti measurements to previously reported normal ranges. RESULTS: Twenty patients with thyroid dysfunction were included. When comparing average recti muscle thicknesses of study patients to those of previously published normal children in similar age ranges, the levator-superior rectus complex was significantly increased in all age groups of children with thyroid dysfunction (p-values = <.004), and the levator-superior rectus complex was most frequently enlarged compared to published normal values (78% of eyes). CAS was not correlated with EOM size in the youngest group (5-10 years old, p-values >.315) but was significantly correlated in older groups (11-17 years old, p-values <.027). TSI was not correlated with EOM size in any group (p-values >.206). CONCLUSIONS: Echographic reference ranges for EOMs in children with thyroid dysfunction were established. There are increased rates of levator-superior rectus complex enlargement in children with TED compared to adults with TED, and EOM size is correlated with CAS in children older than 10 years. Though limited, these findings may serve as an additional tool for ophthalmologists to ascertain disease activity in pediatric patients with thyroid dysfunction.
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Músculos Oculomotores , Glándula Tiroides , Adulto , Humanos , Niño , Anciano , Adolescente , Preescolar , Músculos Oculomotores/diagnóstico por imagen , Estudios Retrospectivos , Ojo , UltrasonografíaRESUMEN
A 56-year-old female with a history of chronic systemic steroid use for asthma control displayed orbital congestion, exophthalmos, a mild abduction deficit, and optic neuropathy. Laboratory workup was unrevealing. Neuroimaging showed increased orbital fat compartments, though the orbital fat was unremarkable on biopsy. The patient was diagnosed with iatrogenic Cushing's syndrome of the orbit and underwent orbital decompression. Early published literature declared this orbitopathy benign. However, newer cases describe more pathologic changes, suggesting the disease is diagnosed later and/or treatment is delayed.
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A neonate presented with a large full-thickness upper eyelid coloboma with near-complete exposure of the cornea. After failing lubrication and a moisture chamber, he was fit with a customized scleral contact lens that protected the ocular surface. It was tolerated well, and ocular surface health was maintained for 13 months to permit the growth of eyelid tissue for future oculoplastic surgery. Delaying reconstruction by using scleral contact lenses as management for large eyelid colobomas has not been previously described. A temporizing measure such as this could be considered for large eyelid colobomas in neonates.
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Coloboma , Lentes de Contacto , Procedimientos de Cirugía Plástica , Coloboma/diagnóstico , Coloboma/cirugía , Córnea , Párpados/cirugía , Humanos , Recién Nacido , Masculino , EscleróticaRESUMEN
PURPOSE: To evaluate extraocular muscle response to teprotumumab using orbital echography in thyroid eye disease. METHODS: This retrospective study included adult thyroid eye disease patients with pre- and post-teprotumumab orbital echography. Data collected included: age, Hertel measurements, clinical activity score, Gorman diplopia scores, ocular motility, and recti muscle diameters measured by echography. The patient's more proptotic eye before treatment initiation was designated as the study orbit. Ocular motility was assessed by totaling the ductions in all 4 cardinal directions. Orbital echography was obtained pre- and post-treatment to assess response of extraocular muscle diameters. RESULTS: Six patients with a mean age of 67 years were included. There was a mean improvement in proptosis of 4.3 mm in the study eye with 11/12 orbits showing improvement in globe position ( p < 0.05). All patients had a decrease in clinical activity score with a mean reduction of 2.5. Four patients had an improvement in Gorman diplopia score. Ocular motility in the study orbits improved by a total mean of 26.9° ( p < 0.05). Mean total extraocular muscle diameter was reduced from 27.4 to 23.4 mm ( p < 0.001). On average, superior recti were largest pre- and post-treatment, followed by inferior, medial, then lateral recti. However, inferior recti showed the greatest reduction of 23% ( p < 0.02). CONCLUSIONS: Orbital echography demonstrated extraocular muscle reduction in all patients after teprotumumab, correlating with improved clinical activity score, ocular motility, and proptosis. Orbital echography is a safe and cost-effective imaging alternative to monitor therapeutic response to teprotumumab.
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Exoftalmia , Oftalmopatía de Graves , Adulto , Anciano , Anticuerpos Monoclonales Humanizados , Diplopía , Oftalmopatía de Graves/diagnóstico , Oftalmopatía de Graves/tratamiento farmacológico , Humanos , Músculos Oculomotores/diagnóstico por imagen , Órbita/diagnóstico por imagen , Estudios Retrospectivos , UltrasonografíaRESUMEN
PURPOSE: To determine whether the age-adjusted Charlson comorbidity index (age-CCI) in sino-orbital fungal disease patients correlates with disease-specific mortality. METHODS: Hospital billing systems at 2 academic institutions were queried for patients with ICD-9, ICD-10, and CPT codes used in fungal disease who also had orbital disease and significant visual loss. Thirty-two patients at Institution A and 18 patients at Institution B met the inclusion criteria of microbiologic or pathologic confirmation of fungal infection and completion of inpatient ophthalmology evaluation. Patients without radiographic abnormality in the sinus or orbit were excluded. Demographic, diagnostic, treatment, and outcome variables were recorded. Our primary outcome was death due to fungal disease. RESULTS: Of the 50 medical records examined, 44 patients met the criteria for fungal-related death outcome on multivariate analysis. The regression coefficient for age-CCI and fungal-related mortality was 0.242 (95% CI, 0.012-0.779) with a p value of 0.038. CONCLUSIONS: Age-CCI is significantly associated with fungal-related mortality. This relationship remains significant when controlling for 5 covariates of fungal organism phylum, presence or absence of CNS disease, exenteration, local treatment use, and presence or absence of an immunosuppressive diagnosis. Age-CCI shows promise as a clinical and research tool in the evaluation of invasive fungal disease involving the orbit.
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Infecciones Fúngicas Invasoras , Enfermedades Orbitales , Comorbilidad , Hongos , Humanos , Enfermedades Orbitales/diagnóstico , Enfermedades Orbitales/epidemiología , Estudios RetrospectivosRESUMEN
PURPOSE: To describe a novel transcutaneous infraorbital nerve biopsy technique which can be performed to aid in the diagnosis of perineural invasion (PNI) of facial cutaneous squamous cell carcinoma (SCC). METHODS: A single-center retrospective chart review was performed. Patients diagnosed with SCC with PNI via an infraorbital nerve biopsy between February 2019 and February 2020 were included. Data collected consisted of patient demographics, medical history, clinical presentation and exam, histologic and radiographic findings, treatment, and outcomes. RESULTS: Four patients (3 male, 1 female) met inclusion criteria. The mean age at diagnosis was 79.5 years (range 66-85 years). Three of the four patients had a history of facial skin lesions, including actinic keratosis and SCC, involving the nose, cheek, or ear. One patient had no history of cutaneous malignancy. All patients presented with cranial neuropathies, including total V2 hypoesthesia. The most common presenting symptom was facial pain, followed by diplopia, unilateral facial weakness, and hypoesthesia in the V1 and/or V2 distribution. Transcutaneous infraorbital nerve biopsy in all patients revealed squamous cell carcinoma with no biopsy complications. CONCLUSION: Definitive diagnosis of PNI can be challenging but is important to minimize tumor-related morbidity. Infraorbital nerve biopsy can establish this diagnosis, especially in the context of negative or indeterminate imaging findings. This work comprises the first description of a transcutaneous approach to infraorbital nerve biopsy, which is a minimally invasive technique that can be performed in an outpatient procedure suite with limited to no sedation.
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Carcinoma de Células Escamosas , Neoplasias Cutáneas , Anciano , Anciano de 80 o más Años , Biopsia , Femenino , Humanos , Masculino , Invasividad Neoplásica , Estudios RetrospectivosRESUMEN
Orbital actinomyces is a rare diagnosis with only a few cases reported in the literature. It can be difficult to diagnose due to its slow, indolent course, and nonspecific findings on imaging and clinical examination, and frequently it can masquerade as other pathologies such as neoplasm and inflammatory disease. The authors present a case of actinomyces masquerading as meningioma with findings of hyperostosis and a superior orbital roof interosseous tract on imaging.
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Hiperostosis , Neoplasias Meníngeas , Meningioma , Actinomyces , Humanos , Neoplasias Meníngeas/diagnóstico , Meningioma/diagnóstico , ÓrbitaRESUMEN
OBJECTIVE: To evaluate the accuracy of the initial diagnosis in the case of fungal infections of the orbit and identify factors that may influence patient outcomes. METHODS: An institutional review board-approved retrospective chart review was conducted across 2 large academic centres to identify cases of fungal infections involving the orbit from January 1, 1998, to November 15, 2019. Data collected included patient demographics, past medical history, examination findings, diagnosis, treatment, imaging, and outcomes. RESULTS: Fifty cases of fungal infection involving the orbit were identified. Of these, 33 (66.0%) were initially misdiagnosed as nonfungal diagnoses. Sixteen patients (32.0%) received multiple initial diagnoses. The most common diagnoses on presentation were bacterial cellulitis (nâ¯=â¯12 of 50; 24.0%) and bacterial sinusitis (nâ¯=â¯12 of 50; 24.0%). These were followed by vascular and orbital inflammatory conditions (nâ¯=â¯9 of 50; 18.0%): 5 patients (10.0%) were clinically diagnosed with giant cell arteritis, 3 (6.0%) with nonspecific orbital inflammation, and 1 (2.0%) with optic neuritis. In this subset of patients, 77.8% (nâ¯=â¯7 of 9) were treated initially with systemic steroids. Additional initial diagnoses included neoplastic mass lesions, mucocele, dacryocystitis, cavernous sinus thrombosis, hemorrhage, tick-borne illness, allergic rhinitis, and allergic conjunctivitis. Misdiagnosis was significantly correlated with involvement of the masticator space on imaging (pâ¯=â¯0.04). CONCLUSION: Fungal infections of the orbit are misdiagnosed in 2 of 3 cases. Nearly 15% of patients who are later diagnosed with fungal disease of the orbit were initially treated with systemic steroids. Misdiagnosis is more frequent when the masticator space is involved.
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Micosis , Enfermedades Orbitales , Humanos , Órbita/patología , Estudios Retrospectivos , Micosis/diagnóstico , Micosis/patología , Enfermedades Orbitales/microbiología , Errores DiagnósticosRESUMEN
Teprotumumab has been shown to improve proptosis and clinical activity scores (CAS) in patients with thyroid eye disease, but little has been published regarding its effects on eyelid retraction. The purpose of this work was to evaluate changes in eyelid position in thyroid eye disease patients after teprotumumab. Eight patients completed eight cycles of teprotumumab. Data collected included exophthalmometry; clinical activity scores; margin reflex distance (MRD) 1; MRD2; and pre-, during, and posttreatment photographs. ImageJ analysis was also used to evaluate eyelid position in photographs. Proptosis significantly improved in 15 of 16 orbits [mean 4.75 ± 2.07 mm reduction (P = 0.0001) in study orbits and mean 3.00 ± 2.14 mm reduction (P = 0.0048) in nonstudy orbits]. CAS was significantly reduced (pretreatment mean 4.88 mm and posttreatment mean 1.88 mm, P = 0.006). MRD1 decreased in 11 of 16 orbits and increased in five orbits (P = 0.18 in study orbits and P = 0.22 in nonstudy orbits). MRD2 decreased in six of 16 orbits and increased in eight orbits (P = 0.49 in study orbits and P = 0.43 in nonstudy orbits). Patients exhibited variable changes in eyelid position with teprotumumab. There was a statistically insignificant decrease in MRD1 after teprotumumab. Proptosis reduction led to unpredictable changes in MRD1 and MRD2. Severity of eyelid retraction did not correlate with clinical activity score response to teprotumumab. There are inherent difficulties in evaluating eyelid position in thyroid eye disease, which may necessitate a paradigm shift in how patients are examined, measured, and photographed.
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PURPOSE: To report a case series of subjective and objective hearing function changes associated with teprotumumab treatment for thyroid eye disease. OBSERVATIONS: A 74-year-old female with a history of Graves' disease with thyroid eye disease was treated with teprotumumab. She had a history of bilateral tinnitus and noticed a subjective improvement in her tinnitus after the second infusion. Audiology testing obtained before, during, and after completion of infusions showed symmetric and rapidly progressive worsening of the patient's sensorineural hearing loss. In contrast, a 42-year-old male with a history of Grave's disease endorsed worsening intermittent tinnitus and low-pitched hearing loss after initiation of teprotumumab. Audiology testing before, during, and after completion of infusions showed stable and normal hearing function bilaterally. CONCLUSION AND IMPORTANCE: This case series highlights the importance of objective testing in patients prior to and after teprotumumab initiation as subjective hearing changes may not accurately reflect objective hearing function. In addition, this report suggests that teprotumumab may play a role in potentiating sensorineural hearing loss.