Pub Date : 2026-02-01Epub Date: 2025-12-09DOI: 10.1080/01676830.2025.2596739
Tanya Gupta, Caroline G Ploeger, Mohsen B Kashkouli, Eyas M Hattab, Jeremy D Clark
Purpose: To present three new cases of primary intraosseous orbital hemangioma (PIOH), including rare lacrimal bone involvement, and to provide an updated systematic review of previously reported cases.
Methods: We retrospectively reviewed three patients with orbital PIOH from our institution. Clinical, radiographic, histopathologic, and surgical findings were analyzed. A comprehensive systematic review of 84 previously published PIOH cases was performed to assess demographics, anatomic location, imaging, and treatment outcomes.
Results: All three patients had firm, progressively enlarging orbital masses. Imaging showed well-circumscribed, expansile lesions confirmed as PIOH histologically. Case 1 involved the zygomatic and sphenoid bones; Case 2, the maxilla and lacrimal bone; and Case 3, the frontal bone. All underwent orbitotomy with subtotal or complete excision and reconstruction. No recurrence was observed at 1-34 month follow-up. In the systematic review, the zygoma (50%) and frontal bone (44%) were most frequently involved. Common symptoms included swelling, proptosis, pain, and globe displacement. CT was the most used imaging modality. Surgical excision, often with preoperative embolization, remains the preferred treatment. Only one recurrence (1.8%) was reported out of the 56 cases with follow-up data.
Conclusions: Orbital PIOH is a rare vascular tumor best managed by tailored surgical excision. Our findings and updated systematic review emphasize individualized planning, particularly in rare sites like the lacrimal bone.
{"title":"Orbital intraosseous hemangioma; report of three cases and systematic review.","authors":"Tanya Gupta, Caroline G Ploeger, Mohsen B Kashkouli, Eyas M Hattab, Jeremy D Clark","doi":"10.1080/01676830.2025.2596739","DOIUrl":"10.1080/01676830.2025.2596739","url":null,"abstract":"<p><strong>Purpose: </strong>To present three new cases of primary intraosseous orbital hemangioma (PIOH), including rare lacrimal bone involvement, and to provide an updated systematic review of previously reported cases.</p><p><strong>Methods: </strong>We retrospectively reviewed three patients with orbital PIOH from our institution. Clinical, radiographic, histopathologic, and surgical findings were analyzed. A comprehensive systematic review of 84 previously published PIOH cases was performed to assess demographics, anatomic location, imaging, and treatment outcomes.</p><p><strong>Results: </strong>All three patients had firm, progressively enlarging orbital masses. Imaging showed well-circumscribed, expansile lesions confirmed as PIOH histologically. Case 1 involved the zygomatic and sphenoid bones; Case 2, the maxilla and lacrimal bone; and Case 3, the frontal bone. All underwent orbitotomy with subtotal or complete excision and reconstruction. No recurrence was observed at 1-34 month follow-up. In the systematic review, the zygoma (50%) and frontal bone (44%) were most frequently involved. Common symptoms included swelling, proptosis, pain, and globe displacement. CT was the most used imaging modality. Surgical excision, often with preoperative embolization, remains the preferred treatment. Only one recurrence (1.8%) was reported out of the 56 cases with follow-up data.</p><p><strong>Conclusions: </strong>Orbital PIOH is a rare vascular tumor best managed by tailored surgical excision. Our findings and updated systematic review emphasize individualized planning, particularly in rare sites like the lacrimal bone.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"236-245"},"PeriodicalIF":0.8,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145716232","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}
Pub Date : 2026-02-01Epub Date: 2025-07-09DOI: 10.1080/01676830.2025.2528226
Haris Afridi, Kainat Afzal, Fnu Shahzaib
{"title":"Comment on: \"selumetinib use as targeted therapy for plexiform neurofibroma: a comprehensive review of the literature\" - mirdametinib as the emerging standard in NF1-PN.","authors":"Haris Afridi, Kainat Afzal, Fnu Shahzaib","doi":"10.1080/01676830.2025.2528226","DOIUrl":"10.1080/01676830.2025.2528226","url":null,"abstract":"","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"227"},"PeriodicalIF":0.8,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144592658","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}
Pub Date : 2026-02-01Epub Date: 2025-08-12DOI: 10.1080/01676830.2025.2543388
Josefina Herrera, Sophia Moshegov, Deepaysh Charanjeet, Jessica Tong, Thomas Hardy, Dinesh Selva, Jean-Louis Desousa, Simon F Taylor, Krishna Tumuluri
Purpose: To provide Australian data and a literature review on the clinical, radiological, and microbiological features and outcomes in patients with orbital abscess secondary to dacryocystitis.
Methods: Multicentre retrospective review of orbital abscess secondary to acute dacryocystitis from 2019 to 2024 in Australia.
Results: Eight cases were identified. The median age was 56 years (range, 26-80 years) with no gender predilection. Common presenting symptoms and signs were motility restriction (n = 6, 75%), proptosis (n = 5, 62.5%), decreased visual acuity (n = 4, 50%), chemosis (n = 3, 37.5%) and RAPD (n = 2, 25%). Radiologically inferomedial (n = 3, 37.5%) and medial (n = 2, 25%) orbital involvement was most commonly followed by intraconal (n = 2, 25%) and inferior (n = 1, 12.5%) orbit. Microbiological cultures revealed gram-negative bacteria (n = 3), gram-positive bacteria (n = 1), anaerobic growth (n = 1), polymicrobial growth (n = 1), MRSA (n = 1), and no growth (n = 1). Treatment included intravenous antibiotics in all cases, 4 patients underwent orbital abscess drainage combined with DCR, 1 had orbital drainage alone, 1 had endoscopic DCR alone, 1 had canthotomy with cantholysis, and 1 patient did not receive surgical treatment. One patient lost vision (light perception) with remainder having stable or improved visual acuity at follow up.
Conclusion: Acute surgical management needs to be considered in majority of cases with dacryocystitis and orbital abscess. Signs of orbital involvement in acute dacryocystitis should be identified promptly to prevent irreversible visual loss.
{"title":"Orbital abscess secondary to acute dacryocystitis.","authors":"Josefina Herrera, Sophia Moshegov, Deepaysh Charanjeet, Jessica Tong, Thomas Hardy, Dinesh Selva, Jean-Louis Desousa, Simon F Taylor, Krishna Tumuluri","doi":"10.1080/01676830.2025.2543388","DOIUrl":"10.1080/01676830.2025.2543388","url":null,"abstract":"<p><strong>Purpose: </strong>To provide Australian data and a literature review on the clinical, radiological, and microbiological features and outcomes in patients with orbital abscess secondary to dacryocystitis.</p><p><strong>Methods: </strong>Multicentre retrospective review of orbital abscess secondary to acute dacryocystitis from 2019 to 2024 in Australia.</p><p><strong>Results: </strong>Eight cases were identified. The median age was 56 years (range, 26-80 years) with no gender predilection. Common presenting symptoms and signs were motility restriction (<i>n</i> = 6, 75%), proptosis (<i>n</i> = 5, 62.5%), decreased visual acuity (<i>n</i> = 4, 50%), chemosis (<i>n</i> = 3, 37.5%) and RAPD (<i>n</i> = 2, 25%). Radiologically inferomedial (<i>n</i> = 3, 37.5%) and medial (<i>n</i> = 2, 25%) orbital involvement was most commonly followed by intraconal (<i>n</i> = 2, 25%) and inferior (<i>n</i> = 1, 12.5%) orbit. Microbiological cultures revealed gram-negative bacteria (<i>n</i> = 3), gram-positive bacteria (<i>n</i> = 1), anaerobic growth (<i>n</i> = 1), polymicrobial growth (<i>n</i> = 1), MRSA (<i>n</i> = 1), and no growth (<i>n</i> = 1). Treatment included intravenous antibiotics in all cases, 4 patients underwent orbital abscess drainage combined with DCR, 1 had orbital drainage alone, 1 had endoscopic DCR alone, 1 had canthotomy with cantholysis, and 1 patient did not receive surgical treatment. One patient lost vision (light perception) with remainder having stable or improved visual acuity at follow up.</p><p><strong>Conclusion: </strong>Acute surgical management needs to be considered in majority of cases with dacryocystitis and orbital abscess. Signs of orbital involvement in acute dacryocystitis should be identified promptly to prevent irreversible visual loss.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"40-47"},"PeriodicalIF":0.8,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144822836","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}
Pub Date : 2026-02-01Epub Date: 2025-10-01DOI: 10.1080/01676830.2025.2566458
Judy Gaffar, Victoria Leung, André Chollet, Elie Boghossian, Vijayabalan Balasingam, Pascal Lavergne, Evan Kalin-Hajdu
Eyelid and conjunctiva sparing exenteration (ECSE) allows for the extirpation of pathologies that lie deep within the orbit without disrupting the anatomy of the eyelids. Despite the cosmetic benefits of maintaining the eyelids, few cases of ECSE have been reported, and reconstruction following the procedure has only been detailed in the form of primary closure or a temporalis muscle transfer. The authors present two patients with deep orbital pathologies that extended into the intracranial space. Each patient underwent an extended ECSE followed by reconstruction with a microvascular free flap. The combination of ECSE plus microvascular free flap reconstruction offers an alternative approach to previously published techniques and may improve esthetic outcomes when exenteration is required for deep orbital pathologies.
{"title":"Eyelid and conjunctiva sparing exenteration followed by microvascular free flap reconstruction - report of two cases.","authors":"Judy Gaffar, Victoria Leung, André Chollet, Elie Boghossian, Vijayabalan Balasingam, Pascal Lavergne, Evan Kalin-Hajdu","doi":"10.1080/01676830.2025.2566458","DOIUrl":"10.1080/01676830.2025.2566458","url":null,"abstract":"<p><p>Eyelid and conjunctiva sparing exenteration (ECSE) allows for the extirpation of pathologies that lie deep within the orbit without disrupting the anatomy of the eyelids. Despite the cosmetic benefits of maintaining the eyelids, few cases of ECSE have been reported, and reconstruction following the procedure has only been detailed in the form of primary closure or a temporalis muscle transfer. The authors present two patients with deep orbital pathologies that extended into the intracranial space. Each patient underwent an extended ECSE followed by reconstruction with a microvascular free flap. The combination of ECSE plus microvascular free flap reconstruction offers an alternative approach to previously published techniques and may improve esthetic outcomes when exenteration is required for deep orbital pathologies.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"171-175"},"PeriodicalIF":0.8,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145207817","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}
Purpose: To compare the clinical presentation, imaging features and outcomes of patients with orbital myositis (OM) and thyroid associated orbitopathy (TAO).
Methods: A retrospective and comparative study of 60 patients diagnosed with OM in a tertiary Eye Clinic between 2012 and 2023. Cases were matched with 60 TAO controls by sex and age.
Results: Clinical presentation of OM differs from TAO with a unilateral involvement and an acute onset of symptoms in 93% and 65% of cases, respectively. OM usually affect a single extraocular muscle (63%), typically the medial rectus, whereas TAO affect multiple and several EOM, with higher incidence of increased orbital fat volume. Tendon sparing on MRI was not discriminant between both conditions. Patients with OM had a better remission rate at 12 months (48% vs 14%, p < 0.001). Optic neuritis occurred in only one patient with OM vs. 11 patients with TAO (p < 0.001) and 12 patients had a relapse of OM with a contralateral involvement in 3/12 patients (25%).
Conclusions: This comparative study highlights key clinical and imaging features that distinguish OM from TAO. OM is associated with a better clinical outcome and prognosis with a reduced risk of optic neuritis and relapse compared with TAO patients.
目的:比较眼眶肌炎(OM)与甲状腺相关性眼病(TAO)的临床表现、影像学特征及转归。方法:对2012年至2023年在某三级眼科门诊诊断为OM的60例患者进行回顾性比较研究。病例按性别和年龄与60例TAO对照组相匹配。结果:OM的临床表现与TAO不同,分别有93%和65%的病例单侧受累和急性发作症状。OM通常影响单个眼外肌(63%),典型的是内侧直肌,而TAO影响多个和多个眼外肌,眼眶脂肪体积增加的发生率较高。在两种情况下,MRI上的肌腱保留没有区别。OM患者在12个月时有更好的缓解率(48% vs 14%, p)。结论:这项比较研究突出了OM与TAO的关键临床和影像学特征。与TAO患者相比,OM具有更好的临床结果和预后,视神经炎和复发的风险降低。
{"title":"A comparative study of the characteristics and prognosis of orbital myositis versus thyroid-associated orbitopathy.","authors":"Augustin Lecler, Alistair Baber, Marie Callet, Gaelle Clavel, Julien Fournel, Thibaud Chazal","doi":"10.1080/01676830.2025.2565786","DOIUrl":"10.1080/01676830.2025.2565786","url":null,"abstract":"<p><strong>Purpose: </strong>To compare the clinical presentation, imaging features and outcomes of patients with orbital myositis (OM) and thyroid associated orbitopathy (TAO).</p><p><strong>Methods: </strong>A retrospective and comparative study of 60 patients diagnosed with OM in a tertiary Eye Clinic between 2012 and 2023. Cases were matched with 60 TAO controls by sex and age.</p><p><strong>Results: </strong>Clinical presentation of OM differs from TAO with a unilateral involvement and an acute onset of symptoms in 93% and 65% of cases, respectively. OM usually affect a single extraocular muscle (63%), typically the medial rectus, whereas TAO affect multiple and several EOM, with higher incidence of increased orbital fat volume. Tendon sparing on MRI was not discriminant between both conditions. Patients with OM had a better remission rate at 12 months (48% vs 14%, <i>p</i> < 0.001). Optic neuritis occurred in only one patient with OM vs. 11 patients with TAO (<i>p</i> < 0.001) and 12 patients had a relapse of OM with a contralateral involvement in 3/12 patients (25%).</p><p><strong>Conclusions: </strong>This comparative study highlights key clinical and imaging features that distinguish OM from TAO. OM is associated with a better clinical outcome and prognosis with a reduced risk of optic neuritis and relapse compared with TAO patients.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"112-120"},"PeriodicalIF":0.8,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145276223","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}
Pub Date : 2026-02-01Epub Date: 2025-10-15DOI: 10.1080/01676830.2025.2571039
Flavia Quaranta Leoni, Antonella Leonardi, Francesco M Quaranta Leoni
Inverse Bell's phenomenon (IBP) is a rare but clinically significant complication of eyelid surgery, characterized by paradoxical downward deviation of the globe during attempted eyelid closure. This study presents a retrospective case series of four patients who developed IBP within three months after upper eyelid surgery at a tertiary oculoplastic center (2018-2024) together with a review of the literature. Presentations ranged from transient IBP that resolved spontaneously to persistent cases associated with lagophthalmos or ocular surface disease. Two patients regained a normal Bell's reflex after conservative or surgical management, whereas two developed chronic IBP despite intervention. Literature review indicates that most cases are transient, although persistent IBP may result from scarring, fibrosis, or neuromuscular disruption. Careful preoperative assessment of Bell's reflex, close postoperative monitoring, and early individualized management are essential. While most cases resolve without sequelae, persistent IBP may necessitate surgical intervention and carries a risk of long-term ocular surface morbidity.
{"title":"Inverse Bell's phenomenon following eyelid surgery: a case series and comprehensive review.","authors":"Flavia Quaranta Leoni, Antonella Leonardi, Francesco M Quaranta Leoni","doi":"10.1080/01676830.2025.2571039","DOIUrl":"10.1080/01676830.2025.2571039","url":null,"abstract":"<p><p>Inverse Bell's phenomenon (IBP) is a rare but clinically significant complication of eyelid surgery, characterized by paradoxical downward deviation of the globe during attempted eyelid closure. This study presents a retrospective case series of four patients who developed IBP within three months after upper eyelid surgery at a tertiary oculoplastic center (2018-2024) together with a review of the literature. Presentations ranged from transient IBP that resolved spontaneously to persistent cases associated with lagophthalmos or ocular surface disease. Two patients regained a normal Bell's reflex after conservative or surgical management, whereas two developed chronic IBP despite intervention. Literature review indicates that most cases are transient, although persistent IBP may result from scarring, fibrosis, or neuromuscular disruption. Careful preoperative assessment of Bell's reflex, close postoperative monitoring, and early individualized management are essential. While most cases resolve without sequelae, persistent IBP may necessitate surgical intervention and carries a risk of long-term ocular surface morbidity.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"187-192"},"PeriodicalIF":0.8,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145304024","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}
Pub Date : 2026-02-01Epub Date: 2025-07-23DOI: 10.1080/01676830.2025.2528979
Satoshi Ueki, Takumi Mashio
Purpose: To analyze the correlation between a magnetic resonance imaging (MRI) score, we proposed in a previous report, and conventional quantitatively evaluated MRI findings, and to evaluate the clinical utility of the MRI score in patients with Graves' ophthalmopathy (GO).
Methods: Using ImageJ, we retrospectively re-analyzed magnetic resonance images of 25 patients with GO who were evaluated in the previous study. The signal intensity and area measured by ImageJ were normalized for each affected extraocular muscle (EOM) in each patient (normalized quantified inflammation and enlargement). Furthermore, the ratio of the value for the affected EOM to the normal values was calculated in each patient (quantified inflammation and enlargement compared to normal values). We analyzed the correlations between MRI scores and quantitative MRI finding before and after 6 g of intravenous methylprednisolone (IVMP).
Results: The correlation coefficient between the normalized MRI score and normalized quantified inflammation and enlargement was 0.59 before IVMP and 0.69 after IVMP. The correlation coefficient between the MRI score and quantified inflammation and enlargement compared to normal values was 0.58 before IVMP and 0.73 after IVMP. The correlation coefficients for all items were statistically significant (P-value < .05).
Conclusion: This study found a high correlation between the MRI score, which is our proposed semiquantitative method for assessing EOM swelling and inflammation, and the results of conventional quantitative evaluation methods. This suggests that the MRI score, which can be more readily used in clinical practice, may be as reliable as quantitative evaluation methods.
{"title":"Correlation between quantified magnetic resonance imaging (MRI) findings and \"MRI score\" in Graves' ophthalmopathy.","authors":"Satoshi Ueki, Takumi Mashio","doi":"10.1080/01676830.2025.2528979","DOIUrl":"10.1080/01676830.2025.2528979","url":null,"abstract":"<p><strong>Purpose: </strong>To analyze the correlation between a magnetic resonance imaging (MRI) score, we proposed in a previous report, and conventional quantitatively evaluated MRI findings, and to evaluate the clinical utility of the MRI score in patients with Graves' ophthalmopathy (GO).</p><p><strong>Methods: </strong>Using ImageJ, we retrospectively re-analyzed magnetic resonance images of 25 patients with GO who were evaluated in the previous study. The signal intensity and area measured by ImageJ were normalized for each affected extraocular muscle (EOM) in each patient (normalized quantified inflammation and enlargement). Furthermore, the ratio of the value for the affected EOM to the normal values was calculated in each patient (quantified inflammation and enlargement compared to normal values). We analyzed the correlations between MRI scores and quantitative MRI finding before and after 6 g of intravenous methylprednisolone (IVMP).</p><p><strong>Results: </strong>The correlation coefficient between the normalized MRI score and normalized quantified inflammation and enlargement was 0.59 before IVMP and 0.69 after IVMP. The correlation coefficient between the MRI score and quantified inflammation and enlargement compared to normal values was 0.58 before IVMP and 0.73 after IVMP. The correlation coefficients for all items were statistically significant (<i>P</i>-value < .05).</p><p><strong>Conclusion: </strong>This study found a high correlation between the MRI score, which is our proposed semiquantitative method for assessing EOM swelling and inflammation, and the results of conventional quantitative evaluation methods. This suggests that the MRI score, which can be more readily used in clinical practice, may be as reliable as quantitative evaluation methods.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"10-14"},"PeriodicalIF":0.8,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144691948","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}
Eyelid xanthogranuloma is a rare non-Langerhans cell histiocytic disorder that typically occurs in children as juvenile xanthogranuloma or in adults as part of systemic diseases such as necrobiotic xanthogranuloma or adult -onset asthma with periocular xanthogranuloma. Isolated eyelid involvement without systemic association is distinctly uncommon. We report a 26-year- old immunocompetent male presenting with a solitary, firm, nodular swelling over the lateral right lower eyelid for three months. Magnetic resonance imaging (MRI) orbit revealed a well-defined subcutaneous lesion (10x 8 mm) without orbital extension. Complete surgical excision was performed and histopathology confirmed non- Langerhans cell xanthogranuloma. Systemic evaluation was unremarkable. The patient received a single postoperative intralesional corticosteroid injection to reduce recurrence risk and remained asymptomatic with no recurrence or systemic disease over one year of follow-up. This case highlights the importance of considering xanthogranuloma in the differential diagnosis of isolated eyelid masses in young adults and demonstrates excellent outcome with surgical excision and adjunctive intralesional steroid therapy. A thorough systemic evaluation is mandatory to exclude underlying systemic disease and to guide appropriate long- term surveillance.
{"title":"Solitary eyelid xanthogranuloma in an immunocompetent young adult: diagnostic challenges and insights.","authors":"Sonali Vinay Kumar, Manoj Gopal Madakshira, Vinay Kumar, Natasha V Kumar, Alok Sati, Jaya Kaushik","doi":"10.1080/01676830.2025.2571038","DOIUrl":"10.1080/01676830.2025.2571038","url":null,"abstract":"<p><p>Eyelid xanthogranuloma is a rare non-Langerhans cell histiocytic disorder that typically occurs in children as juvenile xanthogranuloma or in adults as part of systemic diseases such as necrobiotic xanthogranuloma or adult -onset asthma with periocular xanthogranuloma. Isolated eyelid involvement without systemic association is distinctly uncommon. We report a 26-year- old immunocompetent male presenting with a solitary, firm, nodular swelling over the lateral right lower eyelid for three months. Magnetic resonance imaging (MRI) orbit revealed a well-defined subcutaneous lesion (10x 8 mm) without orbital extension. Complete surgical excision was performed and histopathology confirmed non- Langerhans cell xanthogranuloma. Systemic evaluation was unremarkable. The patient received a single postoperative intralesional corticosteroid injection to reduce recurrence risk and remained asymptomatic with no recurrence or systemic disease over one year of follow-up. This case highlights the importance of considering xanthogranuloma in the differential diagnosis of isolated eyelid masses in young adults and demonstrates excellent outcome with surgical excision and adjunctive intralesional steroid therapy. A thorough systemic evaluation is mandatory to exclude underlying systemic disease and to guide appropriate long- term surveillance.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"181-186"},"PeriodicalIF":0.8,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145303993","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}
Pub Date : 2026-02-01Epub Date: 2025-10-15DOI: 10.1080/01676830.2025.2571446
Mostafa M Diab, Eman A Alibrahim, Hind M Alkatan, Adel H Alsuhaibani
Giant cell reparative granuloma (GCRG) is a rare, benign fibro-osseous lesion, with orbital involvement being particularly uncommon in young children. We describe a 4-year-old boy with progressive right orbital inferior dystopia and proptosis, without history of trauma, sinus disease, or systemic illness. Imaging showed a well-circumscribed superonasal orbital mass with osseous erosion. Through an upper conjunctival fornix approach combined with superior lateral cantholysis, the mass was debulked to preserve vital orbital structures. The lesion appeared highly vascular and yellowish-brown intraoperatively, and histopathology confirmed GCRG. Postoperatively, a short course of low-dose oral corticosteroids was given. The patient achieved complete resolution with no recurrence at 22-month follow-up. This case represents one of the youngest reported patients with isolated orbital GCRG and demonstrates that conservative, function-preserving surgery with adjuvant corticosteroids can be effective. The favorable outcome supports a possible inflammatory component in its pathogenesis and suggests that radical excision may not be necessary in all pediatric cases.
{"title":"Isolated orbital giant cell reparative granuloma in a 4-year-old: case report and literature review.","authors":"Mostafa M Diab, Eman A Alibrahim, Hind M Alkatan, Adel H Alsuhaibani","doi":"10.1080/01676830.2025.2571446","DOIUrl":"10.1080/01676830.2025.2571446","url":null,"abstract":"<p><p>Giant cell reparative granuloma (GCRG) is a rare, benign fibro-osseous lesion, with orbital involvement being particularly uncommon in young children. We describe a 4-year-old boy with progressive right orbital inferior dystopia and proptosis, without history of trauma, sinus disease, or systemic illness. Imaging showed a well-circumscribed superonasal orbital mass with osseous erosion. Through an upper conjunctival fornix approach combined with superior lateral cantholysis, the mass was debulked to preserve vital orbital structures. The lesion appeared highly vascular and yellowish-brown intraoperatively, and histopathology confirmed GCRG. Postoperatively, a short course of low-dose oral corticosteroids was given. The patient achieved complete resolution with no recurrence at 22-month follow-up. This case represents one of the youngest reported patients with isolated orbital GCRG and demonstrates that conservative, function-preserving surgery with adjuvant corticosteroids can be effective. The favorable outcome supports a possible inflammatory component in its pathogenesis and suggests that radical excision may not be necessary in all pediatric cases.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"201-207"},"PeriodicalIF":0.8,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145303971","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}
Pub Date : 2026-02-01Epub Date: 2025-10-21DOI: 10.1080/01676830.2025.2571441
Camilla Pagnacco, Hardeep Singh Mudhar, Sachin M Salvi
A 51-year-old male presented with a rapidly growing right caruncular nodular lesion. Excisional biopsy performed elsewhere showed an apocrine adenocarcinoma of the caruncle with positive margins. Clinically, there was no obvious residual disease, mapping biopsies were negative, and cryotherapy of the surgical base was performed.The tumor recurred both anteriorly in the lower fornix and deeper in the anterior orbit a year later. These were surgically excised with adjuvant radiotherapy (60 Gy in 30 fractions). Unfortunately, the tumor continued to recur posteriorly to progressively first involve optic canal and cavernous sinus, then planus sphenoidale (with loss of right eye vision), and then intracranially with each recurrence treated with stereotactic radiosurgery. Six years from last treatment, the latest MRI revealed the involvement of Meckel's cave and optic chiasm, which is now being treated with hormonal treatment. This is the first case of invasive apocrine adenocarcinoma of the caruncle with intracranial extension treated multimodally with surgery, radiotherapy, repeated stereotactic radiosurgery, and hormone therapy.
{"title":"First report of caruncular invasive apocrine adenocarcinoma with intracranial extension treated with a multimodal approach: a case report and literature review.","authors":"Camilla Pagnacco, Hardeep Singh Mudhar, Sachin M Salvi","doi":"10.1080/01676830.2025.2571441","DOIUrl":"10.1080/01676830.2025.2571441","url":null,"abstract":"<p><p>A 51-year-old male presented with a rapidly growing right caruncular nodular lesion. Excisional biopsy performed elsewhere showed an apocrine adenocarcinoma of the caruncle with positive margins. Clinically, there was no obvious residual disease, mapping biopsies were negative, and cryotherapy of the surgical base was performed.The tumor recurred both anteriorly in the lower fornix and deeper in the anterior orbit a year later. These were surgically excised with adjuvant radiotherapy (60 Gy in 30 fractions). Unfortunately, the tumor continued to recur posteriorly to progressively first involve optic canal and cavernous sinus, then planus sphenoidale (with loss of right eye vision), and then intracranially with each recurrence treated with stereotactic radiosurgery. Six years from last treatment, the latest MRI revealed the involvement of Meckel's cave and optic chiasm, which is now being treated with hormonal treatment. This is the first case of invasive apocrine adenocarcinoma of the caruncle with intracranial extension treated multimodally with surgery, radiotherapy, repeated stereotactic radiosurgery, and hormone therapy.</p>","PeriodicalId":47421,"journal":{"name":"Orbit-The International Journal on Orbital Disorders-Oculoplastic and Lacrimal Surgery","volume":" ","pages":"193-200"},"PeriodicalIF":0.8,"publicationDate":"2026-02-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145349297","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}