Nontraumatic orbital hematoma secondary to labor and childbirth: a case report and review of the literature.

PURPOSE: Non-traumatic orbital hemorrhage without underlying vascular malformations or predisposing conditions is uncommon, and particularly rare in the context of maternal labor. This study combines a novel case report and retrospective review to analyze reported cases and propose insights. METHODS: This study is both a unique case report and literature review examining PubMed publications with articles traced back to original sources through citations for inclusion. Analysis included clinical presentation, visual examination, hematoma characteristics, neuroimaging, management strategies, and outcomes. RESULTS: We present a 37-year-old multigravida woman at 40 weeks gestation who developed acute right-sided proptosis, diplopia, retrobulbar pain, and periorbital edema during the second stage of labor. Computed tomography (CT) revealed a subperiosteal hemorrhage, with subsequent magnetic resonance imaging (MRI) excluding vascular anomalies. Symptoms resolved within two months. Only 14 cases of maternal orbital hematoma associated with labor have been reported. The average age was 28 with 42% (6/14) being primigravid. Including our case, forty percent (6/15) developed symptoms during the second stage of labor, 40% (6/15) immediately postpartum, and 20% (3/15) over 24 hours postpartum. Overall, 33% (5/15) had potentially contributing conditions including coagulopathies, delivery complications, or vascular malformations. Unilateral orbital hemorrhage occurred in 87% (13/15). Surgical intervention was necessary in 13% (2/15). Most (87%, 13/15) underwent observation or medical management with full recovery of symptoms. CONCLUSIONS: Non-traumatic orbital hematomas associated with maternal labor are rare and likely related to increased valsalva during delivery and heightened blood volume in pregnancy. Neuro-imaging and systemic workup are recommended to assess for vascular anomalies or underlying coagulopathies. The overall prognosis is favorable with most having full recovery.

Infection of a Nylon Foil Orbital Implant Due to Fusarium brachygibbosum and Lomentospora prolificans After Intranasal Methamphetamine Use.

The authors describe a case of nylon foil implant infection caused by Fusarium brachygibbosum , and Lomentospora prolificans following medial orbital wall fracture repair in the setting of postoperative nasal methamphetamine use. A 61-year-old male presented with OS pain and swelling after a physical assault on his face. A CT of maxillofacial bones without contrast showed a moderately comminuted fracture of the medial wall of the left orbit with depression of fracture fragments into the left ethmoid air cells. Six days after repair of the medial wall fracture, the patient returned with a new onset headache, OS pain, and swelling to the left medial canthal area. He reported snorting methamphetamine approximately 48 hours before his current presentation. CT imaging showed fat stranding and soft tissue density in the extraconal space adjacent to the left medial rectus muscle and chronic fracture deformity of lamina papyracea with approximately 4 mm of medial displacement of the fracture fragments. The patient showed little clinical improvement after 48 hours of intravenous antibiotics, which led to the removal of the nylon foil implant by a left orbitotomy. Intraoperative tissue cultures grew coagulase-negative Staphylococcus , F. brachygibbosum , and Lomentospora (Scedosporium) prolificans . The patient was subsequently transitioned to oral clindamycin 600 mg three times daily and voriconazole 200 mg two times daily. To the authors' knowledge, this is the first case report to document an association between snorted methamphetamine and a fungal infection of an orbital implant.

Reconstruction of Large Full-Thickness Lower Eyelid Defects With Remnant Eyelid Rotation, Semicircular Skin and Muscle Flap, and Lateral Tarsoconjunctival Flap.

PURPOSE: To describe and assess the results of reconstruction of large, full-thickness defects of the central or medial lower eyelid using a semicircular skin flap, rotation of the remnant lateral eyelid, and a lateral tarsoconjunctival flap. METHODS: The surgical approach is described, and the authors performed a retrospective chart review of consecutive patients between 2017 and 2023 reconstructed with this technique. Outcomes were assessed for size of eyelid defect, vision, subjective symptoms, facial and palpebral aperture symmetry, eyelid position and closure, corneal examination, surgical complications, and need for subsequent surgical intervention. Postoperative appearance was graded on malposition, distortion, asymmetry, contour deformity, and scarring (MDACS). RESULTS: Charts of 45 patients were identified. The average size of the lower eyelid defect was 18 mm (ranging from 12 to 26 mm). Facial and palpebral aperture symmetry were acceptable and all patients had preserved visual acuity and eyelid position and closure. The MDACS cosmetic score was perfect (0) in 15.6% (7/45) of eyelids, good (1-4) in 80.0% (36/45) of eyelids, and mediocre (5-14) in 4.4% (2/45) of eyelids. Second stage reconstruction was not necessary in 32 (71.1%) cases. There were no serious surgical complications, but minor complications included redness of the eyelid margin and pyogenic granulomas. CONCLUSIONS: Medial rotation of the remnant lower eyelid with a lateral semicircular skin and muscle flap over a lateral tarsoconjunctival flap was very effective in this series. Benefits include scarring within the facial skin tension lines, maintained vision throughout the recovery period, no eyelid retraction, and often single stage reconstruction.

Metastatic Epithelioid Sarcoma of the Eyelid in a 47-Year-Old Man: A Case Report.

Epithelioid sarcoma is a rare soft tissue neoplasm of uncertain differentiation that typically affects the distal extremities. Primary orbital epithelioid sarcoma is rare, and no reports exist characterizing metastases of this tumor to the orbit and ocular adnexa. In this article, the authors describe a rare case of eyelid metastasis in a 47-year-old man with epithelioid sarcoma of the right fibula diagnosed 16 months earlier who was otherwise doing well on the adjuvant tazemetostat therapy. In addition, the authors perform a retrospective review of cases of primary orbital epithelioid sarcoma reported in the literature, of which 4 patients responded favorably to surgical excision whereas 2 others eventually died as a direct result of their disease.

Kimura Disease of the Periorbita and Earlobe in a 15-Year-Old Male: A Case Report and Review of the Literature.

PURPOSE: The authors describe a case of Kimura disease (KD) affecting the lacrimal gland, the ipsilateral periorbita, and the contralateral earlobe. The authors also review the literature for characteristics and outcomes of KD affecting the periorbita or earlobe. METHODS: The authors review retrospectively a single chart and abstract data from the English language literature. RESULTS: A 15-year-old Samoan male presented with a left upper eyelid mass and later the contralateral earlobe and ipsilateral periorbita. Surgical resection was partially effective and normal vision and eye motility were maintained. Histopathology was consistent with KD. Thirty-seven cases of periorbital KD were identified in the literature. The average age of patients with periorbital KD was 31.9 years (standard deviation: 17.8 years), with 81.1% (30/37) of patients being male and 69.4% (25/36) Asian. Over half of periorbital KD patients had lacrimal gland involvement (51.3%; 19/37). Fifteen patients were initially treated with surgery; 3 (20%) had recurrence of the disease. Another 15 patients were initially treated with corticosteroids; 12 (80%) had recurrence of the disease. Ophthalmologic data, when reported, demonstrated that, most patients had no visual (77.8%; 14/18 patients) or motility disturbances (82.4%; 14/17 patients) but most had proptosis 71.4% (10/14). Only 4 cases of earlobe involvement in KD were identified, all in women. CONCLUSIONS: In the literature, orbital KD was rare but often affected the lacrimal gland and caused proptosis, commonly in young adult Asian males. Vision and extraocular motility were usually normal. Earlobe involvement was very rare. This unique case of KD that affected the orbit and contralateral earlobe corroborates the prior literature that KD is prone to recurrence but may not adversely affect vision or extraocular motility.

Pediatric Bartonella henselae neuroretinitis masking co-infections.

PURPOSE: Neuroretinitis (NR) is an inflammatory disorder that presents with painless vision loss due to optic disc edema, peripapillary detachment, and macular lipid exudation. We report the first two documented cases of co-infections of pediatric NR due to Bartonella henselae with HSV and Toxocara cati, respectively. OBSERVATIONS: A 10-year-old female with acute right-sided facial droop, right eye pain, and acute visual loss of the right eye is diagnosed with co-infection of Bartonella and HSV retinitis and is successfully treated with acyclovir, rifampin, and doxycycline. A 13-year-old female with progressive visual loss of the left eye is diagnosed with co-infection of Bartonella and ocular toxocariasis and is successfully treated with doxycycline, rifampin, prednisolone, and albendazole. CONCLUSIONS AND IMPORTANCE: Early recognition and multi-modal treatment is necessary to prevent delayed diagnosis and treat the underlying NR causes for optimal visual recovery.

Optic nerve biopsy in leukemic infiltrative optic neuropathy: a case report and review of the literature.

Optic nerve infiltration is a rare but known complication of the central nervous system (CNS)-involving lymphoma and leukemic disorders. The diagnosis is often presumed and patients are empirically treated with systemic therapy and/or local radiation. Optic nerve biopsy is usually avoided due to the risk of permanent vision loss secondary to the procedure. We present a case of biopsy-proven leukemic optic neuropathy without optic nerve sheath or cerebrospinal fluid (CSF) involvement in a patient previously in remission from T-cell prolymphocytic leukemia (T-PLL). To our knowledge, this is the first documented case of T-PLL with biopsy-proven optic nerve invasion without CSF involvement and suggests possible perineural invasion or a sanctuary site from chemotherapy. We suggest that for patients with poor vision and suspected leukemic infiltration without other evidence of CNS involvement, both optic nerve and optic sheath biopsy should be performed for diagnosis and treatment.

Bilateral orbital inflammation in a 6-month old with SARS-CoV-2 infection.

A 6-month-old female presented with bilateral periorbital edema for 7 days. Laboratory testing was significant for active SARS-CoV-2 infection. Neuroimaging demonstrated soft tissue changes within the bilateral orbits and enlargement of the bilateral lacrimal glands. Although the patient initially improved with corticosteroid treatment, she later returned with recurrent left periorbital and eyelid edema. Orbital biopsy was performed and demonstrated findings in the lacrimal gland and the adjacent fibroconnective tissues that are similar to those of prior lung specimens seen in SARS-CoV-2 patients. Final diagnosis was bilateral orbital inflammation with features presumed secondary to SARS-CoV-2 infection. To the best of our knowledge, this is one of the first reports to document bilateral orbital inflammation as a sign of SARS-CoV-2 infection in the pediatric population with the associated pathological findings.

Bilateral acquired nasolacrimal duct obstruction secondary to amyloidosis in a 15-year-old.

A 15-year-old boy who presented with a 1-week history of increasing erythema, edema, and tenderness of the right upper and lower eyelids was found to have acquired nasolacrimal duct obstruction (NLDO) secondary to primary amyloidosis. To our knowledge, this is the youngest case of bilateral NLDO secondary to primary amyloidosis (biopsy proven for right NLDO and presumed for left NLDO) reported in the literature. This case highlights the importance of lacrimal sac biopsy in patients with acquired NLDO of unclear etiology. Given the prevalence of the primary amyloidosis subtype in cases of ocular or adnexal amyloidosis, patients should undergo immediate workup for systemic disease.

Ophthalmic changes in patients with hemifacial atrophy (Parry-Romberg syndrome).

PURPOSE: Parry-Romberg syndrome (PRS) is a rare condition characterized by progressive, unilateral facial atrophy. We hypothesize that patients with this condition may have involvement of the ocular structures. Here, we report our ophthalmic, clinical and anatomical findings in an observational study of six patients with long-standing PRS. METHODS: Patients diagnosed with PRS were invited to participate in a clinic visit during which the following tests were administered and data recorded: best-corrected vision, refractive error, Ishihara color plates, Hertel exophthalmometry, gonioscopy, complete slit-lamp and dilated fundus examination, Intra Ocular Lens Master measurements and keratometry. Two-sample T tests were used to compare data between affected and unaffected eyes, as well as affected eyes and a normative population. RESULTS: Six patients underwent complete eye examinations. The mean spherical equivalent of the affected eye was + 3.83 D, while that of the unaffected eye was + 0.13 D. The atrophic hemiface averaged 2.8 mm of enophthalmos on Hertel exophthalmometry. The axial length of the atrophic eye was 0.91 mm shorter than the unaffected eye. Compared to normative data, in patients with PRS, the difference between eyes was statistically different for each of the following variables: visual acuity, spherical equivalent, corneal diameter, axial length and flat and steep keratometry. CONCLUSIONS: This is one of the first quantitative, exploratory studies with ophthalmic measurements in patients with PRS. Our results suggest the globe may demonstrate atrophic changes similar to other soft tissues in the face known to be affected by this condition.

Topical Corticosteroid Use Associated With Increased Degree of Ptosis and Rate of Ptosis Repair Failure.

PURPOSE: To compare the degree of ptosis and the risk of ptosis repair failure among patients with and without a history of topical corticosteroid use. METHODS: Retrospective, case-controlled study examining topical corticosteroid use among adults with ptosis who underwent external levator advancement/resection (ELR) or Müller muscle conjunctival resection with at least 3 months postoperative follow-up. Comparative statistical analyses of surgical outcomes were performed amongst patients with and without history of topical corticosteroid use. RESULTS: A total of 240 patients (406 eyelids) met study criteria, of which 36 patients (44 eyelids) had history of topical corticosteroid use. Mean preoperative margin reflex distance was 0.20 mm and 0.58 mm for topical corticosteroid and non-corticosteroids users (p = 0.01). Mean preoperative levator function was 9.78 mm and 10.38 mm for topical corticosteroid and non-corticosteroid users (p = 0.02). The rate of ptosis repair failure was 30% and 16% in patients with and without a history of topical corticosteroid use (odds ratio 2.25, 95% confidence interval 1.10-4.55; p = 0.03). The rate of recurrence per surgical type in eyelids with and without history of topical corticosteroid use was: external levator advancement/resection 11/27 (41%) and 48/266 (18%) (odds ratio = 3.12, confidence interval 1.36-7.15 0; p = 0.01); Müller muscle conjunctival resection 2/17 (12%) and 9/96 (9%) (odds ratio 1.29, confidence interval 0.25-6.56; p = 0.76). CONCLUSIONS: Topical corticosteroid use is associated with more severe presenting ptosis and increased rates of ptosis repair failure. Compared to Müller muscle conjunctival resection, there is a significantly higher rate of ptosis repair failure in patients undergoing external levator advancement/resection.

Polytetrafluoroethylene frontalis suspension in blepharospasm with eyelid apraxia: an effective and well-tolerated adjunct to botulinum toxin therapy.

Purpose: To report the efficacy of polytetrafluoroethylene (PTFE) frontalis suspension for blepharospasm with eyelid apraxia and postoperative botulinum toxin requirements. Methods: Retrospective chart review of patients with blepharospasm and eyelid apraxia who underwent frontalis suspension. The primary outcome was a surgical success, defined by surgeon- and patient-reported success in postoperative eyelid opening. Comparative statistical analyses of botulinum toxin dosage and treatment intervals were performed amongst patients before and after frontalis suspension ptosis repair. Results: Five patients (10 eyelids) met the study criteria, of which 40% were female. Mean age was 63.2 years. All patients had successful surgical outcomes based on physician-reported and patient reported satisfaction with the postoperative eyelid opening. Average follow-up was 14 months. Preoperative botulinum toxin treatments averaged 80.4 units (range 32-110, SD 33.2) to the periocular region over an average of 9.6-week intervals. Postoperative botulinum toxin treatments averaged 61.4 units (range 24-110, SD 34.7) to the periocular region over an overage of 9.8-week intervals. No patients experienced postoperative exposure keratopathy, extrusion of the sling, or postoperative infection. Conclusions: Frontalis suspension using PTFE suture in the setting of blepharospasm with eyelid apraxia was found to be a safe and effective procedure. Frontalis suspension in this population is not a substitute for botulinum toxin treatment but may allow for reduced treatment dosage. Frontalis suspension appears to increase patient functionality with improved eyelid opening in patients with blepharospasm with eyelid apraxia.

A shotgun wedding.

A 69-year-old woman developed a carotid-cavernous fistula (CCF) after firing a shotgun. Initially, the patient had mild visual symptoms, but later on developed prominent features of CCF including chemosis, proptosis, ophthalmoparesis and conjunctival injection . The fistula was embolized via an intravascular coiling procedure. We are unaware of another patient who developed a CCF due to blunt force from shotgun use.

Visual preservation in congenital orbital fibrosis.

OBJECTIVE: Congenital orbital fibrosis (COF) is a nonprogressive, unilateral, congenital process in which variable fibrosis is demonstrated in the orbit, resulting in restrictive strabismus, upper eyelid malposition, and axial displacement of the globe. We present 4 new pediatric cases of COF and discuss factors that impact visual development. We also describe a patient with local compressive optic neuropathy/edema who underwent optic nerve sheath fenestration (ONSF) for visual preservation. DESIGN: Literature review and retrospective case series. RESULTS: Four male COF patients (mean age of 11 months) were examined. Two patients presented with decreased ocular motility of the affected eye. Two patients presented with exophthalmos, and one presented with enophthalmos. Two patients presented with ptosis, and one presented with eyelid retraction. Two patients presented with optic nerve atrophy, and one presented with optic nerve edema. Magnetic resonance imaging demonstrated involvement of the superior, medial, and inferior rectus and superior oblique muscles in 3 patients and the lateral rectus and inferior oblique muscles in 2 patients. Three patients underwent orbitotomy. Histology was consistent with fibrosis. Three patients demonstrated amblyopia, and 2 responded to treatment. The patient with optic nerve edema underwent ONSF. At 4 months' follow-up, the edema had resolved. CONCLUSIONS: COF can present with either anterior or posterior globe displacement. Patients must undergo a complete ophthalmic evaluation to identify modifiable factors. Strabismus and ptosis should be addressed for optimal visual development. Amblyopia therapy should be instituted quickly. Patients who present with active optic nerve edema may benefit from ONSF for local compressive optic neuropathy.