Mariel Angelou Parulan, Gangadhara Sundar, Yew Kwang Ong, Tseng Tsai Yeo, Victor Lee & Miriam Santiago Kimpo
We report a case of a 2-year-old female who presented with bilateral progressive proptosis, visual loss, nasal obstruction, and breathing difficulty. Magnetic resonance imaging revealed a large sino-orbital mass that was extending to the orbital apex and skull base. An initial diagnosis of rhabdomyosarcoma was made elsewhere on the basis of the presence of round and spindle cell tumor. Subsequent biopsy with immunohistochemical staining was positive for nuclear staining with β-catenin, shifting the diagnosis to a myofibroblastic tumor, favoring desmoid-type fibromatosis. With image guidance, near complete excision of tumor was performed by a multidisciplinary team, while respecting danger zones such as the skull base and the optic nerve. Following a recurrence over 2 months, additional excision was performed with a 6-month treatment of methotrexate and vinblastine. Desmoid tumor is a rare form of soft tissue tumor uncommonly seen in the orbital area. Although benign, it is known to be recurrent and infiltrative. Few data are known and further information will aid in the management of these tumors.
Clara J. Men, Frances Wu, Bradford W. Lee, Jonathan H. Lin, Bobby S. Korn & Don O. Kikkawa
A 5-year-old otherwise healthy girl presented to the oculoplastic service with a painless superotemporal subconjunctival mass in the left eye. Visual acuity was within normal limits, and there was no evidence of proptosis or orbital enlargement. Excision was performed to remove the anterior portion of the mass for alleviation of symptoms. On histopathological analysis, the mass was comprised of fibroadipose tissue consistent with dermolipoma and contained a hard nodule found to be a calcified tooth. In the periocular region, odontogenic choristoma (tooth) is a rare lesion, and has been reported to occur within teratomas, dermoid cysts, and displaced oral embryonic epithelium. We describe an unusual case of a tooth occurring within a sporadic dermolipoma. The clinical presentation, examination, management, and histopathology are reviewed.
Levi N. Kanu, Catherine Y. Liu, Daniel J. Oh, Peter W. MacIntosh & Pete Setabutr
We describe six patients with 12 separate episodes of self-inflicted periocular foreign body injuries, which presented to our institution recently. All patients were male, relatively young (mean 28.5 years old), incarcerated, and had significant underlying psychiatric conditions. The subjects had inserted staples (6), paperclips (2), or other small metallic wire segments (4) into the periocular region. Most cases (9/12) involved concurrent self-inflicted injury to other body parts. Ten cases involved foreign bodies inserted through the palpebral conjunctiva into the upper eyelid, while two cases involved insertion into the orbit. Identification and surgical retrieval of foreign bodies was successful in most cases (9/11) but was not attempted in one case. Self-inflicted periocular injuries, while rare, are challenging cases for which the ophthalmologist should be prepared. A multidisciplinary approach, including psychiatric assessment and treatment, is important for optimal care.
S. Kumar & T. Diamond
Paraneoplastic syndrome is a rare but reversible cause of non-thyroid-related extraocular muscle enlargement. We present a 71-year-old lady with diplopia, restricted eye movements, suppressed thyroid-stimulating hormone and enlargement of all extraocular muscles while on thyroxine replacement for hypothyroidism. She had distant history of metastatic breast cancer treated with chemotherapy, surgical resection and tamoxifen. She had negative anti-thyroid autoantibodies and thyroid ultrasound was not consistent with autoimmune thyroid disease. Carcinoembryonic antigen and cancer antigens 15-3, 125 and 72-4 were elevated, and whole-body positron emission tomography-computed tomography showed avid liver, left adrenal and skeletal lesions, with liver biopsy confirming breast cancer recurrence. She received prednisone and chemotherapy (letrozole, palbociclib) and achieved normalisation of eye movements and reduction in her EOME at 9-month follow-up. Our case highlights the importance of exploring paraneoplastic syndrome as a treatable cause of EOME in a patient lacking features of thyroid orbitopathy and autoimmune thyroid disease.
David S. Curragh, Srikandan Kamalarajah, Brendan Lacey, Stephen T. White, Alan A. McNab, George Kalantzis, Peter J. Dolman, Dinesh Selva & Saul N. Rajak
Purpose: We present a series of primary orbital implant replacement for cases of implant exposure to describe our experience of this one-staged surgical approach.
Methods: This study reports on a one-stage technique which involved the removal of the exposed implant or dermis fat graft (DFG) and insertion of a secondary (replacement) in the same procedure, with a variety of materials, including autologous tissue. Re-exposure in a socket where a DFG was placed was defined as a new defect in the newly epithelialized conjunctiva or dehiscence of the dermis-conjunctiva junction. All cases of primary replacement for the management of exposed orbital implant, porous and non-porous, were included, even when there were clinical signs suggestive of infection. The primary outcome was the rate of re-exposure, requiring additional surgical procedures. Infection following primary replacement was a secondary outcome.
Results: Seventy-eight patients had primary replacement for the management of an exposed orbital implant. 6.4% had re-exposure at a mean follow-up of 49.7 months (9.1% for ball implants and 4.5% for DFG). The rate of exposure was higher in those with prior signs of infection than those without (8% vs. 3.6%). Re-exposure occurred in 4.5% of cases with DFG implantation, 4.3% of cases with non-porous implants and in 20% of cases with porous implants.
Conclusion: Primary replacement for management of exposed orbital implant, porous and non-porous, has a high rate of successful outcome even in cases with presumed or confirmed infection.
Mansooreh Jamshidian-Tehrani, Amin Nabavi, Abolfazl Kasaee, Narges Hasanpoor, Eahsan Elhami, Shervin Sharif-Kashani, Ahamd Masoumi, M. Hossein Nowroozzadeh & Ali Sadeghi-Tari
Purpose: To evaluate alterations in orbital color Doppler imaging (CDI) parameters and their correlation to disease activity and severity in patients with thyroid eye disease (TED).
Methods: Seventy-six orbits of 45 TED patients and 40 orbits of 40 normal controls were enrolled in this cross-sectional study. According to clinical activity score (CAS), patients were categorized to active (CAS ≥ 3) or inactive disease (CAS < 3). Patients were also classified as having mild, moderate or severe disease. Peak systolic velocity (PSV), end diastolic velocity (EDV), and resistance index (RI) in ophthalmic artery and central retinal artery, and maximum and minimum velocity in superior ophthalmic vein and central retinal vein were determined in all subjects.
Results: There was a significant difference in maximum velocity of superior ophthalmic vein and EDV and RI of ophthalmic artery between patients with TED and normal subjects. Superior ophthalmic vein maximum and minimum velocity and ophthalmic artery RI were significantly higher in patients with active disease than inactive cases. Disease severity did not affect the blood flow parameters independently. A cutoff point of 3.99 cm/s in superior ophthalmic vein maximum velocity yielded a sensitivity of 91.2% and specificity of 81.2% in detecting active disease.
Conclusion: Retrobulbar blood flow is altered in TED and is related to disease activity. Superior ophthalmic vein maximum velocity could be helpful in differentiation of active and inactive cases.
Kerstin Stähr, Anja Eckstein, Laura Holtmann, Anke Schlüter, Meaghan Dendy, Stephan Lang & Stefan Mattheis
Introduction: Different minimally invasive surgical approaches to the orbit allow individualized bone resection to reduce proptosis and decompress the optic nerve in patients with Graves’ orbitopathy (GO). This study aims to compare piezosurgery to an oscillating saw used to resect bone from the lateral orbital wall.
Methods: In a retrospective study, we analyzed balanced orbital decompressions performed on 174 patients (318 cases) with GO. An oscillating saw was used in 165 cases (saw group) and piezosurgery in 153 cases (piezo group). Peri- and postoperative complications, reduction of proptosis, new onset of diplopia and improvement of visual acuity in cases of pre-operative optic nerve compression were analyzed.
Results: We observed no significant differences in the surgical outcome between the two groups. Proptosis reduction was 4.6 mm in the saw group (p < 0.01) and 5.3 mm in the piezo group (p < 0.01). Intraoperative handling of the piezosurgery device was judged superior to the oscillating saw, due to soft tissue conservation and favourable cutting properties. Duration of the surgery did not differ between the groups. No serious adverse events were recorded in both groups.
Conclusion: The application of piezosurgery in orbital decompression is more suitable than an oscillation saw due to superior cutting properties such as less damage to surrounding soft tissue or a thinner cutting grove.
Pimkwan Jaru-Ampornpan, Shannon S. Joseph, Ana B. Diniz Grisolia & Cesar A. Briceno
The authors describe the first report in the literature of delayed orbital hemorrhage that may be partly caused by supratherapeutic anticoagulation. A 52-year-old man with supratherapeutic international normalized ratio (INR) presented with acute proptosis, orbital pain and diplopia 9 months after the floor and medial orbital wall fracture repair using nylon foil implant. He was found to have hemorrhaged into the capsule surrounding the orbital implant. Three weeks later, the patient underwent implant removal after warfarin was discontinued for 5 days and INR was normalized. His symptoms resolved postoperatively. This case describes a unique risk factor of delayed orbital hemorrhage in patients with previous orbital fracture repair, and highlights that coagulopathy should be investigated in patients presenting with acute proptosis with a history of orbital fracture repair. The authors also provide a comprehensive and up-to-date literature review on previously reported cases with delayed hemorrhagic complications from alloplastic orbital implants.
LETTER TO THE EDITOR
Alan A. McNab
No abstract available
Pimkwan Jaru-Ampornpan, Shannon S. Joseph, Ana B. Diniz Grisolia & Cesar A. Briceno
No abstract available
Wilson, Caroline W.; Fisher, Mark T.; Pagedar, Nitin A.; Kass, Gretchen; Terry, William W.; Shriver, Erin M.
The aim of exenteration reconstruction is to stabilize the postsurgical wound bed to promote expeditious healing particularly in patients who are undergoing adjuvant radiation and/or chemotherapy. Porcine urinary bladder matrix has previously been used successfully as a wound-healing scaffold in treatment of burns and in acute, chronic, and surgical wounds, but the use of these products has not previously been reported in the exenterated orbit. The authors present a case of the novel use of porcine urinary bladder matrix in a pediatric patient who underwent exenteration for recurrent embryonal rhabdomyosarcoma, subsequent split-thickness skin grafting, and adjuvant radiation.
Hodson, Trevor; Cho, Raymond I.
Chronic inflammatory demyelinating polyneuropathy is known to cause enlargement of the cranial nerves, particularly the trigeminal nerve. Several cases of orbital neuromas associated with this condition have been published, but surgical treatment has not been reported. The authors present a case of bilateral supraorbital neuromas associated with chronic inflammatory demyelinating polyneuropathy that underwent surgical excision and histopathologic examination.