Tocilizumab as a Useful Tool for Thyroid Eye Disease in Pediatric Population: A Case Report.

Thyroid eye disease (TED) characterizes by inflammation and remodeling of orbital tissues. Although the majority of pediatric TED is mild, some children present progressive and severe disease. The approach to severe disease in this group of patients, especially when noncortico-responsive, is challenging. The purpose of this work was to describe the effective use of tocilizumab as second-line therapy in steroid-resistant pediatric TED. A 13-year-old female with a history of Graves' disease presented with right eye proptosis for at least 8 months associated with mild pain on eye movements and ocular surface complaints. The ophthalmologic evaluation revealed Hertel exophthalmometry readings of 22 mm on the right eye (OD) and 19 mm on the left (OS). The remaining ophthalmic examination was unremarkable. Intravenous methylprednisolone pulses of 500 mg were initiated without any improvement after 4 treatments. Following multidisciplinary team discussion, therapy was switched to monthly tocilizumab injections at 4 mg/kg. Significant reduction of proptosis and resolution of pain and ocular surface complaints were noted immediately after the treatment switch. Exophthalmometry readings after the end of treatment that included 4 tocilizumab injections were 20 mm on OD and 19 mm on OS. No side effects were reported during the entire follow-up. Six months after treatment cessation, the patient remains stable, without any signs of orbitopathy relapse and no ophthalmologic complaints. This is the second case report showing the effectiveness of tocilizumab in pediatric TED and the first one showing its efficacy when steroids fail. Our results support the potential safety and efficacy of this immunosuppressor in children with TED.

Rhino-Orbital Mucormycosis After COVID-19 Recovery: A Case Report.

The pandemic caused by SARS-CoV-2 remains a health care concern, despite vaccination programs. Mucormycosis, especially rhino-orbital-mucormycosis, has been described as a severe complication of COVID-19. Although it has been described mostly in India and other developing countries, few cases in the western world have also been described. We present a case of rhino-orbito-mucormycosis after recovery from severe COVID-19 in Portugal. A 75-year-old diabetic and obese man presented with right proptosis associated with right eye pain and low vision one month after recovery from severe COVID-19. Considering the most probable etiology for this clinical picture, anti-fungal therapy with liposomal amphotericin B was promptly initiated, followed by endoscopic sinus debridement. However, due to persistent and progressive infection, and after a multidisciplinary revision of the case, orbital exenteration was performed. One year after surgery, the patient is stable, without clinical or imagological signs of relapse of the disease. Although the evolution of the pandemic, along with vaccination programs, led to a lower incidence of severe COVID-19 disease, there are still patients presenting with severe COVID-19, requiring intensive care and at risk for serious complications. This case illustrates the importance of being aware of the development of post-COVID-19 mucormycosis and the need for close surveillance of patients recovering from severe COVID-19. COVID-19 prompt diagnosis and multidisciplinary approach are essential for a timely intervention achieving better survival while minimizing morbidity.

Bilateral Episcleral Corkscrew Vessels: Expedition into the Unknown - Case Report.

We aim to report a case of a middle-aged woman with bilateral idiopathic elevated episcleral venous pressure (IEEVP) and its difficulties in the diagnostic approach. Particularly in this case, the atypical feature of ocular hypertension without glaucomatous optic nerve damage may be misleading. We present a 66-year-old woman with longstanding bilateral "red eyes." Clinical findings included bilateral episcleral vessel engorgement and tortuosity and raised intraocular pressure with open iridocorneal angles. Despite ocular hypertension, glaucomatous neuropathy was absent and confirmed by normal structural (optical coherence tomography) and functional (standard automated perimetry) tests. The systemic workup was unremarkable. Magnetic resonance angiography showed bilateral dilated superior ophthalmic veins. Cerebral digital subtraction angiography was requested, and no carotid-cavernous fistula (or other significant vascular findings) was identified. The diagnosis of IEEVP was assumed. In conclusion, our case highlights the systematic investigation necessary in cases of bilateral episcleral vessel engorgement and tortuosity and the possible differential diagnosis to be considered to rule out life-threatening causes of elevated episcleral venous pressure. It is important for clinicians to be aware of IEEVP even in patients with atypical features that despite significative ocular hypertension had no glaucomatous damage.

Management of post-septal complications of acute rhinosinusitis in children: A 14-year experience in a tertiary hospital.

INTRODUCTION: Acute rhinosinusitis accounts for most of the cases of orbital infections and is the most common cause of periorbital oedema in children. Up to 10% of patients with orbital complications of acute rhinosinusitis may experience vision loss and other complications such as meningitis, intracranial abscess or even death. Therefore, these patients require prompt diagnosis and proper treatment. OBJECTIVES: This study aims to report the clinical presentation and management of post-septal orbital complications of acute rhinosinusitis in the paediatric population. MATERIALS AND METHODS: A retrospective medical chart review of all children aged under 18 years old who were diagnosed with post-septal orbital complications of acute rhinosinusitis at a tertiary academic hospital, between 01/2007 and 12/2020. Patients were grouped according to the Chandler Classification (groups 2-5). RESULTS: Fifty-five children (mean age of 6.91 ± 4.61 years) fulfilled the entry criteria for post-septal orbital complications of acute rhinosinusitis, based on clinical evaluation by an otorhinolaryngologist and CT-scan findings. Forty (72.72%) patients were also evaluated by an ophthalmologist. Most patients were male (76.36%). Twenty-four patients had post-septal cellulitis (43.63%), 21 patients had a subperiosteal abscess (38.18%) and 10 patients had an orbital abscess (18.18%). Eyelid swelling was the most frequent sign, followed by fever. Microbiology varied considerably and gram-positive agents were clearly predominant. Eighteen (32.73%) patients had been treated with oral antibiotics prior to hospital admission, exhibiting a significantly higher risk of recurrence of orbital infection (p = 0.020). Ethmoid and maxillary sinuses were the most involved paranasal sinuses (90.91%). Thirty-three patients (60%) were successfully treated medically, and 22 patients (40%) required surgical drainage. Seven patients (12.73%) developed further complications and six recovered without sequelae. The mean length of hospital stay was 8.0 ± 5.0 days and recurrence of orbital infection occurred in six patients (10.91%). The absolute neutrophil blood count was significantly different amongst Chandler groups (p = 0.021), with higher counts in patients with subperiosteal abscess. The duration of hospitalization was significantly higher in patients submitted to surgery (p < 0.001). CONCLUSION: Post-septal orbital complications of acute rhinosinusitis are infrequent but dangerous events in the paediatric population. Close collaboration with Ophtalmology is paramount, as the child's vision is at risk. Eyelid swelling and proptosis are early signs. CT-scan imaging plays an invaluable role in the diagnosis and decision-making. Predictive indicators for surgery were not found. However, emergency endoscopic nasal surgery with abscess drainage should be considered whenever vision is at risk, if there is no improvement after aggressive medical treatment, and in cases of intracranial complications.

Orbital Apex Syndrome due to Orbital Mucormycosis after Teeth Infection: A Successful Case Report.

Rhino-orbital-cerebral mucormycosis is a severe and rapidly progressive fungal infection associated with low survival rates. Early diagnosis and proper management are mandatory. We report the case of a 20-year-old female with diabetic ketoacidosis and teeth infection, who presented with sudden orbital apex syndrome. Several surgical procedures were necessary to provide a definite histopathological diagnosis of the fungal infection. Ultimately, given the progressive aggravation of the infection and the ineffectiveness of antifungal and antibiotic agents, an orbital exenteration was performed. A mycobacteriological examination revealed a polymicrobial culture with Mucorales. One year after the initial presentation, the patient is alive and with a good general health condition.

Graded müllerectomy for correction of graves upper eyelid retraction: effect on eyelid movements.

PURPOSE: To measure the effect of müllerectomy from posterior approach on the amplitudes of spontaneous blinking and downward eyelid saccades. METHODS: Spontaneous blinks and downward upper eyelid saccadic movements of 16 patients (23 eyelids) with Graves orbitopathy were measured before and after müllerectomy from posterior approach. A new video system was used to continuously register the blinking activity while subjects viewed a commercial movie for 5 minutes. Downward eyelid saccades (30° of downgaze) were also measured with the video system. RESULTS: Müllerectomy had no effect on the amplitude of the blink. However, as the eyelid margins were significantly lowered by the surgery, the amplitude of the blink movements relative to the pupil center increased substantially. The number of movements occluding the pupil center increased from 0% to 13%. Due to the increased efficiency of blinking, the blink rate decreased. Surgery induced a mean increase of 1.1 mm of downward saccades. CONCLUSIONS: The effects of müllerectomy on the blinks are indirect and related to correction of eyelid retraction. The relative amplitude of blink movements increases and blink rate decreases. Müllerectomy does affect the downward eyelid saccades increasing the ability of the upper eyelid to relax on downgaze.

[Orbital vascular pathology: clinical and imagiological differential diagnosis]. / Patologia vascular da órbita: diagnóstico diferencial clínico e imagiológico.

Orbital vascular lesions represent an important group of orbital pathology, because of their high prevalence, particularly in the pediatric population. They are also the most controversial group of lesions, due to the polemics in regard to their nature. Based on cases of their experience, the authors analyze the orbital vascular pathology, especially its clinical and imagiological characteristics, important to the diagnosis.