{"title":"Tocilizumab - a disease-modulating treatment for thyroid associated ophthalmopathy?","authors":"K Farde, F Träisk","doi":"10.1080/01676830.2025.2452181","DOIUrl":"https://doi.org/10.1080/01676830.2025.2452181","url":null,"abstract":"<p><strong>Purpose: </strong>To analyze Tocilizumab (TCZ, an interleukin 6 inhibitor) as a second-line treatment for thyroid-associated ophthalmopathy (TAO).</p><p><strong>Methods: </strong>In this retrospective observational study, the charts of patients with moderately severe to severe TAO who received intravenous Tocilizumab as a second-line treatment 2020-2023 were reviewed.</p><p><strong>Results: </strong>Twenty-three patients were enrolled in the study. At the follow-up visit 24 weeks after the initiation of treatment, 14 out of 22 patients (63.6%, one missing data) had a reduction of proptosis of 2 mm or more. The mean proptosis decreased from 21.07 (±3.23) to 19.18 (±2.52) mm in the right eye (RE) and from 20.61 (±3.12) to 19.95 (±2.81) mm in the left eye (LE). Twenty patients (87%) also showed a reduction of the clinical activity score (CAS) of at least two points with a mean decrease from 5.22 (±1.53) pre-treatment to 2.09 (±1.38) post-treatment points. The median patient total follow-up time after treatment was ten (range 6-26) months. The comparisons reached strong significance between pre-treatment and 24 weeks, as well as pre-treatment and final visit assessments for both proptosis and clinical activity scores. Serum TRAb levels also dropped significantly, and only two patients needed re-treatment with TCZ. Improvement of diplopia did not reach statistical significance.</p><p><strong>Conclusions: </strong>Intravenous Tocilizumab as a second-line treatment for TAO led to a significant improvement of proptosis and CAS at 24 weeks follow-up, with a sustained effect over time.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"1-5"},"PeriodicalIF":0.9,"publicationDate":"2025-01-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143013976","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Svein Harald Mørkve, Gaute Kjellevold Wathle, Nicola Logallo
{"title":"Endovascular treatment of carotid-cavernous fistulas in a medium-sized Scandinavian neurovascular center.","authors":"Svein Harald Mørkve, Gaute Kjellevold Wathle, Nicola Logallo","doi":"10.1080/01676830.2024.2448802","DOIUrl":"https://doi.org/10.1080/01676830.2024.2448802","url":null,"abstract":"<p><strong>Purpose: </strong>Carotid-cavernous fistulas (CCFs) are treated almost exclusively by endovascular techniques, but the frequency of treatments is limited in smaller centers. We analyzed all CCFs treated in our hospital to determine if high-quality treatment of CCFs can be provided in a medium-volume neurovascular center.</p><p><strong>Methods: </strong>Retrospective quality-control cohort study.</p><p><strong>Results: </strong>From 2005 to 2021, 28 CCF-treatments were performed in our hospital, all endovascular. Fourteen were direct and fourteen were indirect CCFs. All direct CCFs were treated using transarterial coil embolization, and in seven of these the ICA was preserved. All indirect CCFs were treated using transvenous coil embolization. Complete cure was achieved after one procedure in 24 (85.7%) patients, and in all patients (100%) after retreatments. All CCF-patients experienced significant improvement of symptoms after treatment and 79.2% were asymptomatic after 6-12 months. Two patients with direct CCFs were blind on their ipsilateral eye before treatment and remained so after treatment. Three patients had reduced vision or visual field defects ipsilateral to their indirect CCFs that normalized after treatment. One patient suffered a minor asymptomatic thromboembolic event during the treatment (3.6%), otherwise there were no complications to treatments.</p><p><strong>Conclusions: </strong>High-quality treatment of CCFs can be provided in a medium-volume neurovascular center.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"1-8"},"PeriodicalIF":0.9,"publicationDate":"2025-01-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143013972","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Bicanalicular canaliculitis.","authors":"Mohammad Javed Ali","doi":"10.1080/01676830.2025.2454357","DOIUrl":"https://doi.org/10.1080/01676830.2025.2454357","url":null,"abstract":"","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"1"},"PeriodicalIF":0.9,"publicationDate":"2025-01-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143013945","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Paulina Truong, Saif Aldeen Alryalat, Osama Al Deyabat, Andrew G Lee, Patricia Chévez-Barrios, Amina Malik
{"title":"Necrosis of pleomorphic adenoma of the lacrimal gland with acute and chronic arteriolar occlusion.","authors":"Paulina Truong, Saif Aldeen Alryalat, Osama Al Deyabat, Andrew G Lee, Patricia Chévez-Barrios, Amina Malik","doi":"10.1080/01676830.2025.2449988","DOIUrl":"https://doi.org/10.1080/01676830.2025.2449988","url":null,"abstract":"<p><p>Pleomorphic adenoma of the lacrimal gland (PALG) is a benign neoplasm typically presenting with gradual, painless globe displacement and/or lid swelling. We report an atypical case of PALG in a 53-year old male presenting acutely, mimicking orbital cellulitis. Imaging demonstrated an extraconal rim-enhancing soft-tissue lesion medial to the left lacrimal gland, involving superior rectus and levator palpebrae superioris. Surgical excision and histopathology revealed PALG with central infarction, necrosis, and chronic and acute pseudocapsular arteriolar occlusion. These findings correlated with the fluctuating symptoms and suggested a multifactorial mechanism involving both underlying vasculopathy, thrombosis, and tumor growth. Infarction in PALG may occur iatrogenically or from spontaneous tumor infarction, and can cause an acute inflammatory presentation. We discuss unique histopathologic features of necrotic PALG and mechanisms of infarction. Recognition of these features and the varied presentations of PALG is crucial for proper diagnosis. Complete resection is necessary to prevent recurrence or malignant transformation.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"1-6"},"PeriodicalIF":0.9,"publicationDate":"2025-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142962461","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Marissa K Shoji, Benjamin I Meyer, Nina Diklich, Sugi Panneerselvam, Matthew Camacho, Kevin D Clauss, Thomas E Johnson, David T Tse, Brian C Tse
{"title":"Adult and pediatric orbital rhabdomyosarcoma: comparison of characteristics and outcomes.","authors":"Marissa K Shoji, Benjamin I Meyer, Nina Diklich, Sugi Panneerselvam, Matthew Camacho, Kevin D Clauss, Thomas E Johnson, David T Tse, Brian C Tse","doi":"10.1080/01676830.2024.2445702","DOIUrl":"https://doi.org/10.1080/01676830.2024.2445702","url":null,"abstract":"<p><strong>Introduction: </strong>Rhabdomyosarcoma (RMS) is a common pediatric orbital malignancy but is extremely rare in adults. This study assesses clinical and radiographic features, management, and outcomes in adult orbital RMS patients with comparison to pediatric patients.</p><p><strong>Methods: </strong>A retrospective chart review from 2000-2023 at Bascom Palmer Eye Institute was conducted evaluating patients aged 0 to 100-years-old with biopsy-confirmed orbital RMS. Medical records were reviewed for demographics, clinical features, imaging, histopathology, management, and outcomes. Statistical analysis was conducted with Mann-U Whitney and chi-squared testing.</p><p><strong>Results: </strong>Twenty-four patients were included, 15 children (mean age 6.4 ± 4.4 years) and 9 adults (35.7 ± 12.4 years). Patients in both groups presented with eyelid edema followed by proptosis with similar symptom duration (<i>p</i> = 0.31). Lesions were frequently located medially and inferiorly in both groups. At presentation, adults had significantly more metastatic disease in addition to bone, extraocular muscle, intracranial, and parameningeal involvement. The most common pediatric RMS subtype was embryonal (80%), whereas most adults were alveolar (77.8%, <i>p</i> = 0.001). Treatment in both groups frequently included chemoradiation. Both groups demonstrated similar local recurrence (<i>p</i> = 0.72), overall survival (<i>p</i> = 0.86), and ophthalmologic sequelae (<i>p</i> = 0.45), although pediatric follow-up duration was notably longer.</p><p><strong>Conclusions: </strong>To our knowledge, this study is the largest report of adult orbital RMS from a single institution, highlights key comparisons in features and outcomes between adult and pediatric orbital RMS, and provides an updated literature review. While pediatric and adult orbital RMS clinical presentations are similar, adult disease more often demonstrates aggressive features, including alveolar subtype, local structure involvement, and lower disease-free survival.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"1-12"},"PeriodicalIF":0.9,"publicationDate":"2025-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142962347","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Mostafa Mohamed Diab, Ashish Ranjan, Mohammad Javed Ali, Nandini Bothra
{"title":"Atypical lacrimal sac mucoceles.","authors":"Mostafa Mohamed Diab, Ashish Ranjan, Mohammad Javed Ali, Nandini Bothra","doi":"10.1080/01676830.2024.2443627","DOIUrl":"https://doi.org/10.1080/01676830.2024.2443627","url":null,"abstract":"<p><strong>Purpose: </strong>To report the clinical features and operative findings of a series of patients with lacrimal sac mucoceles extending above the anatomic level of the medial canthal tendon (MCT).</p><p><strong>Methods: </strong>A retrospective chart review of all patients presenting with lacrimal sac mucocele extending above the anatomic level of the MCT over a period of five years from 2019 and 2023. All patients underwent surgical management. Diagnosis of lacrimal sac mucocele was based on clinical examination, imaging where indicated and intraoperative confirmation. Data obtained include demographics, clinical presentations, duration, imaging features, type of surgical intervention, intraoperative findings, and outcomes.</p><p><strong>Results: </strong>A total of 62 patients were analyzed, of whom 67.7% were female. The mean age at presentation was 49.11 years. The mean duration of the lacrimal sac swelling was 18.15 months (SD = 21.94). Epiphora, discharge, and dacryocystitis were frequent associations. Regurgitation on Pressure over the lacrimal sac area (ROPLAS) decompressed the swelling in 35 (56.5%) patients.In 61.3% of patients, the medial canthus remained in its normal position, while the medial canthal tendon (MCT) exerted pressure on the overextending mucocele to varying degrees, resulting in a dumbbell-shaped appearance in certain cases. In 38.7% of patients, the mucocele caused superior displacement of the medial canthus compared to the contralateral side. The surgical interventions were successful in resolving the mucocele.</p><p><strong>Conclusions: </strong>Lacrimal sac mucoceles can extend above the level of the MCT. While a lacrimal sac swelling above the MCT should raise suspicion for a mass lesion of neoplastic etiology, it is not a tell-tale sign.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"1-6"},"PeriodicalIF":0.9,"publicationDate":"2025-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142956631","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Tatiana Boza, Doris Quiroz, Mariana Nadais Aidar, Denny M Garcia, Antônio Augusto Velasco E Cruz
{"title":"Upper and lower eyelid contour and positional changes after deep skin grafts in ablepharon macrostomia syndrome.","authors":"Tatiana Boza, Doris Quiroz, Mariana Nadais Aidar, Denny M Garcia, Antônio Augusto Velasco E Cruz","doi":"10.1080/01676830.2024.2430339","DOIUrl":"https://doi.org/10.1080/01676830.2024.2430339","url":null,"abstract":"<p><p>Ablepharon macrostomia syndrome is a rare congenital disorder caused by autosomal-dominant <i>TWIST2</i> mutations. This condition is characterized by redundant skin, low-set ears, macrostomia, ambiguous genitalia, and underdevelopment of the both upper and lower eyelids. The shortening of the anterior lamella, septum and levator aponeurosis lead to a severe corneal exposure within the first hours of life. Since McCarthy and West's first report in 1977, 21 AMS cases have been documented. We report a new AMS case with a quantitative analysis of palpebral fissure changes following skin grafts over the upper and lower smooth tarsal muscles and lateral tarsorrhaphy.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"1-5"},"PeriodicalIF":0.9,"publicationDate":"2025-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142956637","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Technical notes for a method of eyebrow reconstruction by retroauricular scalp graft: problems and countermeasures.","authors":"Hidetaka Miyazaki, Jonnah Kristina Teope, Yasuhiro Takahashi, Hirohiko Kakizaki","doi":"10.1080/01676830.2024.2444515","DOIUrl":"https://doi.org/10.1080/01676830.2024.2444515","url":null,"abstract":"<p><strong>Purpose: </strong>To describe a technique using retroauricular scalp graft for eyebrow reconstruction, along with problems encountered and countermeasures in treatment.</p><p><strong>Methods: </strong>We present a patient with eyebrow loss following resection of a malignant schwannoma. We initially covered the defect from the upper eyelid to the eyebrow area with artificial dermis for hemostasis and to increase the granulation of the graft bed. Considering hair texture, aesthetic unit and color match, the eyebrow area was grafted with a scalp harvested from the retroauricular region. The skin from the subclavian area was used for the upper eyelid defect. Tie-over fixation was performed.</p><p><strong>Results: </strong>After a slightly longer immobilization period of 8.5 days, the tie-over fixation was removed. All grafted scalp and skin were fully engrafted. The reconstructed eyebrows had natural hair flow and softness. The texture of retroauricular hair closely resembled that of natural eyebrow hairs. The color and texture the upper eyelid grafts were also matched well. Surgical scar at the retroauricular hairline was effectively concealed by surrounding hair.</p><p><strong>Conclusion: </strong>Eyebrow reconstruction could be successfully achieved using from the retroauricular scalp with some ingenuity in order to be successful. These include improving the hemodynamic status of the grafted bed, considering aesthetic units, and extending the fixation period.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"1-5"},"PeriodicalIF":0.9,"publicationDate":"2025-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142962463","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Tracy J Lu, John Nguyen, Andrea L Kossler, Aaron Fay, Natalie A Homer
{"title":"Positional lagophthalmos variability in facial nerve palsy with and without upper eyelid loading.","authors":"Tracy J Lu, John Nguyen, Andrea L Kossler, Aaron Fay, Natalie A Homer","doi":"10.1080/01676830.2024.2444497","DOIUrl":"https://doi.org/10.1080/01676830.2024.2444497","url":null,"abstract":"<p><strong>Purpose: </strong>Lagophthalmos from facial nerve palsy is traditionally measured with patients in an upright position and may fail to identify positional variability. This study aims to assess the effects of body position, surgical technique, implant material, and patient demographics on lagophthalmos.</p><p><strong>Methods: </strong>A multicenter prospective study was performed to evaluate positional changes in paralytic lagophthalmos and the effects of various patient and surgical factors.</p><p><strong>Results: </strong>Sixty-four eyelids were included. Thirty-two (50%) of eyelids had upper eyelid weight implantation with a median post-operative follow-up of 67 weeks. The mean change in lagophthalmos from sitting to supine was a 1.4 mm increase (<i>p</i> < 0.001). There was a larger lagophthalmos variability in patients with eyelid weights compared to those without (<i>p</i> = 0.047), and in patients with gold compared to platinum weights (<i>p</i> = 0.026). Positional increase in lagophthalmos did not correlate with weight position (i.e. pretarsal vs. supratarsal) (<i>p</i> = 0.26), weight load (<i>p</i> = 0.70), House-Brackmann severity (<i>p</i> = 0.16), age (<i>p</i> = 0.14), or gender (<i>p</i> = 0.13).</p><p><strong>Conclusions: </strong>Surgeons should routinely measure lagophthalmos in the supine position, as nocturnal corneal exposure may warrant ocular lubrication, occlusion, or additional surgery. Platinum weights may be preferred over gold due to less positional lagophthalmos variability.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"1-5"},"PeriodicalIF":0.9,"publicationDate":"2025-01-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142956635","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Emma M Hartness, Arnulfo Garza Reyes, Rupin N Parikh, Nicholas J Lepa, Leili Rahimi, Erin M Shriver
{"title":"A clinical challenge: delayed diagnosis of autoimmune polyglandular syndrome type II in a patient with thyroid eye disease.","authors":"Emma M Hartness, Arnulfo Garza Reyes, Rupin N Parikh, Nicholas J Lepa, Leili Rahimi, Erin M Shriver","doi":"10.1080/01676830.2024.2433120","DOIUrl":"https://doi.org/10.1080/01676830.2024.2433120","url":null,"abstract":"<p><p>A 35-year-old woman with thyroid eye disease (TED) was found to have autoimmune polyglandular syndrome type II (APS-II) 1 year after developing symptoms of this rare syndrome, during which she underwent 8 emergency department visits before receiving a targeted endocrinology workup. Thyroid disease is the second most common autoimmune syndrome associated with APS-II after primary adrenal insufficiency. Identification of this syndrome is critical as it can be life-threatening if left untreated. Additionally, if associated hypothyroidism is treated with levothyroxine, adrenal crisis can occur. The patient described symptoms, some of which overlapped with those of TED, and a significant delay in diagnosis occurred. Discussion of this patient's course highlights the importance of ophthalmologists keeping APS-II in mind when seeing patients with thyroid eye disease.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"1-4"},"PeriodicalIF":0.9,"publicationDate":"2025-01-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142956626","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}