Customized Nasal Prosthesis for a Jones Lacrimal Bypass Tube Using 3-Dimensional Planning and Printing Technology.

A 35-year-old woman was treated for extensive squamous cell carcinoma of the nasal septal mucosa with total rhinectomy, including removal of the nasal septum, and chemoradiotherapy. A magnet-retained nasal prosthesis was fitted. She had developed right-sided epiphora from total proximal lacrimal canalicular obstruction, for which an angled Jones lacrimal bypass tube was inserted. The tube, however, intermittently rotated in the nasal cavity, causing recurrent epiphora and irritation at the caruncular site. With the aid of 3-dimensional technology, we designed a septum for the prosthesis that stabilized the tube within the nasal cavity. At the follow-up 2 years later, the patient was satisfied with the nasal prosthesis and lacrimal stent. To our knowledge, this report is the first to describe a patient-specific nasal prosthesis adapted for a Jones tube after total rhinectomy.

Motility of the Ocular Prosthesis in Anophthalmic Patients: Objective and Patient-perceived Findings.

AIM: To study the impact of the prosthesis motility on the Quality of Life (QoL) in anophthalmic patients. METHODS: Cross-sectional, observational study of 100 anophthalmic patients, of whom 64% had an acryl implant, 6% an Allen implant, 1% a hydroxyapatite implant, 4% a dermis fat graft, 16% no implant, and 9% an unknown implant. We quantitatively assessed the motility of the prosthesis with Kestenbaum glasses and the QoL with a validated questionnaire covering five domains: General functional abilities and care, wearing comfort, physical appearance, psychological and social functioning. Associations between measured prosthetic eye motility, patient-perceived motility, and satisfaction were made. RESULTS: Motility of the prosthesis was impaired with an average loss of 76%, and correlated with Cosmetic satisfaction (adduction P = .02, abduction P = .008, elevation P = .04) and Social satisfaction (adduction P = .03, abduction P = .003). The patient-perceived motility of the prosthesis correlated with General functioning abilities (horizontal P = .0004, vertical P = .0004), Comfort (horizontal P = .001, vertical P = .003), Cosmetic satisfaction (horizontal P = .0002, vertical P = .0002), Psychological satisfaction (horizontal P = .001, vertical P = .001), and Social satisfaction (horizontal P = .002, vertical P = .003). CONCLUSIONS: Ocular prosthetic motility has a significant impact on patient-perceived satisfaction and physical appearance, and predicts coping with the prosthetic condition on the psychosocial level. This highlights the need of introducing patient-reported outcome measures in the prosthetic rehabilitation of the anophthalmic patient.

Idiopathic Orbital Myositis Revisited.

PURPOSE OF REVIEW: To review current knowledge regarding idiopathic orbital myositis. RECENT FINDINGS: Recent publications have focused on possible causes of orbital myositis and the process to reach a diagnosis of idiopathic orbital myositis. With inflamed and enlarged extraocular muscles, features to distinguish between competing diagnostic possibilities are based on imaging in the context of history and clinical signs. Idiopathic orbital myositis is characterized by the clinical triad of acute onset of orbital pain exacerbated on eye movement, double vision, and redness or swelling of the eyelids or conjunctiva, along with the radiological finding of homogeneous, fusiform enlargement of one or more extraocular muscles. In atypical or inconclusive clinico-radiological findings for a diagnosis of idiopathic orbital myositis, or where the clinical behavior changes or fails to respond to corticosteroid treatment, a systemic and oncologic work-up and muscle biopsy are warranted to exclude specific local or systemic disease as cause of the inflamed and enlarged muscle. As our understanding of idiopathic orbital myositis evolves, the diagnostic focus is shifting toward earlier identification of underlying local or systemic disease through systemic work-up and muscle biopsy.

Delayed orbital fibrosing inflammation from a retained crayon.

A 39-year-old woman presented with sudden onset of double vision, right upper eyelid swelling and ptosis, and orbital pain. Imaging revealed an irregular mass of the upper right orbit with central non-enhancing areas. Upon inquiry, the patient recalled an intraorbital trauma with a crayon in her childhood, 35 years ago. Via translid anterior orbitotomy, remnants of a blue crayon embedded in an orbital fat mass were removed. Histopathology showed scavenger reaction of macrophages and sclerosis. Energy-dispersive X-ray spectroscopy revealed the presence of silicate particles. Repeated courses of corticosteroids were given. The patient deteriorated with reduced vision and frozen globe owing to severe orbital fibrosis of the entire orbit.

Measuring quality of care and life in patients with an ocular prosthesis.

PURPOSE: In patients with an anophthalmic condition, the primary determinants of success of ocular prosthetic rehabilitation are satisfaction with care and quality of life (QoL). The aim of this study is to develop a condition-specific questionnaire as a patient-reported outcome measure for patients with an ocular prosthesis. METHODS: Observational cross-sectional prospective study. We included 100 patients (52 female, 48 male, > 18 years old) with an anophthalmic and ocular prosthetic condition existing for 2 years or more. The patients completed a pre-tested 72-item questionnaire regarding their experience on living with an ocular prosthesis in four domains of QoL: single vision and care, wearing comfort, physical appearance and motility, and psychosocial functioning. Associations with demographic factors and condition- and prosthesis-related variables were investigated with multivariate analysis. The questionnaire was reduced with principal component analysis to obtain the Global Ocular Prosthesis Score (GOPS). RESULTS: Satisfaction scores for each QoL domain were high with a mean visual analogue score between 7.2 and 7.6. Patients were generally satisfied with the physical appearance of the artificial eye and reported adequate psychosocial functioning. Patients described the reduced peripheral visual field and socket discharge as chief complaints. The test was reduced to a 20-item questionnaire. The mean GOPS was 70.87 (median 75.00). CONCLUSIONS: Patients with longstanding ocular prosthetic wear are satisfied with their physical appearance and report adequate psychosocial functioning. A concise 20-item questionnaire for the anophthalmic condition is a valuable tool to quantitatively measure patient-reported outcome of ocular prosthetic rehabilitation. TRIAL REGISTRATION NUMBER: NCT04321382, 03/2020, retrospectively registered.

Aspiration Pneumonia After External Dacryocystorhinostomy Under Local Anesthesia Without Sedation: A Case Report.

Amid the global coronavirus disease 2019 (COVID-19) outbreak, an 89-year-old male with chronic kidney disease presented with acute dacryocystitis and a persistent dry cough. After a course of antibiotics, external dacryocystorhinostomy was performed under local anesthesia without sedation. During planned hemodialysis in the early hours after the procedure, the patient developed nausea and hematemesis followed by severe dyspnea and hypoxemia. The patient was diagnosed with aspiration pneumonia, a previously unreported complication in lacrimal surgery.

Diplopia Following the Insertion of a Canalicular Bypass Tube: Etiology, Risk Factors, Management, and Outcomes.

PURPOSE: To report the etiology, management, and possible risk factors for diplopia after canalicular bypass surgery. METHODS: A multicenter retrospective, noncomparative case series of patients who developed diplopia following canalicular bypass surgery were assessed. RESULTS: Twenty-four cases of diplopia were identified across 12 institutions. Tubes were inserted as a primary procedure with external dacryocystorhinostomy (DCR) (1; 4%) or without DCR (10; 42%) or as a secondary procedure after external (8; 33%) or endonasal (5; 21%) DCR. Factors predisposing to local damage were noted in 17 (71%): these factors included preexisting autoimmune/inflammatory condition (7 cases), medial canthal tumor resection (5 cases), preoperative radiotherapy (2 cases), 2 drug treatments (topical and systemic), and 1 local surgery. Horizontal diplopia was due to restriction of abduction and first noted at a median of 3.5 months (mean: 17.8 months, range: 1 day to 112 months) and persisted in 23 (96%) cases with a mean restriction of -2, affecting primary gaze in 4 patients and activities of daily living in 13 (42%). Seventeen patients received various treatments: 10 were operated on resulting in cure in 1 and improvement in 9. A stable degree of diplopia persisted in all but one patient. CONCLUSIONS: Restriction of abduction causing horizontal diplopia is a rare complication with canalicular bypass surgery and a notably high proportion occurred after tube placement without DCR; carunculectomy was not ubiquitous. Although in some the diplopia may be improved with intervention, the chance of cure is low. This complication should probably be included during informed consent for canalicular bypass tubes.

Three-dimensional characterisation of the globe position in the orbit.

PURPOSE: Current methods to analyse the globe position, including Hertel exophthalmometry and computed tomography (CT), are limited to the axial plane and require the lateral orbital rim and cornea as landmarks. This pilot study aimed to design a method to measure the position of the globe in the axial, coronal and sagittal plane and independent from orbital bony and corneal references. METHODS: With the aid of three-dimensional CT reconstruction technology, we determined the globe position in the orbit based on the centre of the globe. Method validation was performed using data of consecutive orbital CT scans from the control group and from the patients with Graves' orbitopathy who underwent orbital decompression surgery with removal of the lateral orbital margin. RESULTS: The inter- and intra-observer reliability was excellent with a high intraclass correlation coefficient (> 0.99, 95% CI [0.97; 1.00]). In the decompressed orbits, there was a statistically significant globe position shift along the anterior-posterior axis (P = 0.0005, 95% CI [0.63; 3.66]), but not along the medial-lateral and superior-inferior axis. CONCLUSION: The 3D CT method can accurately and reliably characterise the globe position shift in the three dimensions without using orbital and corneal anatomical landmarks. The method can be useful to determine the globe shift in proptosis, enophthalmos, hypoglobus and hyperglobus, even in the presence of strabismus and orbital bone defects.

Perioperative Prophylactic Antibiotics in 1,250 Orbital Surgeries.

PURPOSE: Intravenous antibiotic prophylaxis is used for many clean-contaminated surgeries or clean surgeries with an implant, but its value for clean orbital surgery has not been determined. This study investigated infection risks and adverse effects related to antibiotics in patients undergoing orbital surgery. METHODS: A prospective, nonrandomized comparative case series of all patients undergoing orbital surgery with participating surgeons between October 1, 2013, and March 1, 2015. Types of surgery, antibiotic regimens, corticosteroid use, antibiotic side effects, and surgical site infections (SSIs) were entered into an electronic database and subsequently analyzed. Cases in which patients received postoperative oral antibiotics were analyzed separately. RESULTS: Of 1,250 consecutive orbital surgeries, 1,225 met inclusion criteria. A total of 1208 patients were included in the primary analysis: 603 received no antibiotic prophylaxis (group A), and 605 received a single dose of intravenous antibiotic (group B). Five patients (0.42%) developed an SSI, 3 in group A and 2 in group B. The difference in SSI rates was not statistically significant between the 2 groups (p = 0.66). Antibiotic prophylaxis, alloplastic implants, paranasal sinus entry, and corticosteroid use were not associated with differences in SSI rates. All SSIs resolved on a single course of oral antibiotics; an implant was removed in 1 case. There were no complications associated with a single dose of intravenous prophylaxis. However, 12% of 17 patients (group C) who received 1 week of oral postoperative prophylactic antibiotics developed antibiotic-related complications (diarrhea, renal injury), yielding a number needed to harm of 8.5. CONCLUSIONS: In this large series, antibiotic prophylaxis does not appear to have reduced the already low incidence of SSI following orbital surgery. Given the detriments of systemic antibiotics, the rarity of infections related to orbital surgery, and the efficacy of treating such infections should they occur, patients undergoing orbital surgery should be educated to the early symptoms of postoperative infection and followed closely, but do not routinely require perioperative antibiotics.

Applications of three-dimensional printing in orbital diseases and disorders.

PURPOSE OF REVIEW: To comprehensively review the applications of advanced three-dimensional printing technology in the management of orbital abnormalities. RECENT FINDINGS: Three-dimensional printing has added value in the preoperative planning and manufacturing of patient-specific implants and surgical guides in the reconstruction of orbital trauma, congenital defects and tumor resection. In view of the costs and time, it is reserved as strategy for large and complex craniofacial cases, in particular those including the bony contour. There is anecdotal evidence of a benefit of three-dimensional printing in the manufacturing of prostheses for the exenterated and anophthalmic socket, and in the fabrication of patient-specific boluses, applicators and shielding devices for orbital radiation therapy. In addition, three-dimensional printed healthy and diseased orbits as phantom tangible models may augment the teaching and learning process of orbital surgery. SUMMARY: Three-dimensional printing allows precision treatment tailored to the unique orbital anatomy of the patient. Advancement in technology and further research are required to support its wider use in orbital clinical practice.

Diagnosis of enlarged extraocular muscles: when and how to biopsy.

PURPOSE OF REVIEW: To review current knowledge regarding diagnosis of nonthyroid orbital disorders with extraocular muscle enlargement. RECENT FINDINGS: Recent publications have focused on immunoglobulin G4-related disease as a possible cause of enlarged extraocular muscles, on patterns of strabismus that raise a clinical suspicion of intramuscular lymphoma, and on surgical techniques to access the muscles for tissue biopsy. SUMMARY: With enlarged extraocular muscles, features to distinguish between competing diagnostic possibilities are based on imaging in the context of history and clinical signs. Infraorbital nerve enlargement in the presence of muscle enlargement strongly favours a diagnosis of immunoglobulin G4-related disease and reactive lymphoid hyperplasia. As our understanding of minimally invasive orbital surgery evolves, the diagnostic focus is shifting toward earlier identification through muscle biopsy.

The many facets of dacryoadenitis.

PURPOSE OF REVIEW: To review the spectrum of lacrimal gland inflammation, with particular reference to the old condition, idiopathic dacryoadenitis, and the new, immunoglobulin G4 (IgG4)-related dacryoadenitis. RECENT FINDINGS: Idiopathic dacryoadenitis remains the most common inflammatory lesion of the lacrimal gland, for which surgical treatment is successful. There is mounting evidence that the presence of IgG4-positive plasma cells in a lacrimal gland specimen is nonspecific, for being found in other inflammatory lacrimal gland lesions, even lymphoproliferative ones. To identify IgG4-related dacryoadenitis, particularly when there is no documented disease elsewhere, the criteria have been strengthened toward abundant IgG4-positive plasma cell counting. SUMMARY: The gold standard of diagnosis of noninfectious lacrimal gland inflammation is tissue biopsying, which is commonly therapeutic in the case of idiopathic dacryoadenitis. Although it was initially suggested that IgG4-related dacryoadenitis is a possible cause of idiopathic dacryoadenitis, it becomes obvious that it has more resemblance to the lymphoproliferative tumors.

Surgical debulking for idiopathic dacryoadenitis: a diagnosis and a cure.

PURPOSE: Idiopathic inflammatory tumor of the lacrimal gland, also called idiopathic dacryoadenitis, generally is treated with high-dose, long-term systemic corticosteroids, despite their limited success, high recurrence rate, and incidence of drug-induced side effects. This study describes the outcome of patients with idiopathic dacryoadenitis who were managed with surgical debulking. DESIGN: Retrospective case series from 2 tertiary referral centers. PARTICIPANTS: Forty-six patients (46 lacrimal glands). METHODS: Review of the clinical records, radiologic scans, and histopathologic specimens, with additional immunoglobulin G4 immunostaining. MAIN OUTCOME MEASURES: Clinical signs and symptoms at 2 months after the surgery and off medications. RESULTS: Before referral, 41% (19 of 46) of the patients had received systemic high-dose corticosteroids, after which they all showed recurrence, of whom 26% (5 of 19) became dependent on corticosteroids. At referral, all patients underwent debulking surgery of the inflammatory lacrimal gland mass for diagnostic and therapeutic reasons. Additionally, intralesional or systemic low-dose corticosteroids were given during the operation or the first postoperative days in 54% (25 of 46) of the patients. At 2 months after the debulking surgery, a full clinical recovery was seen in 80% (37 of 46) of the patients. A recurrence occurred in 8% (3 of 37) of the patients 4 months and 2.2 and 4.6 years later. Surgical failure (20%; 9 of 46) was correlated with prior corticosteroid treatment (P = 0.002, Fisher exact test), but not with sclerosing inflammation present in 28% (13 of 46). The median follow-up time was 7.2 years (range, 0.7-18 years). CONCLUSIONS: Debulking biopsy procedures for idiopathic dacryoadenitis, in addition to being diagnostic, may be therapeutic.

Series of extranodal natural killer/T-cell lymphoma, nasal type, with periorbital involvement.

PURPOSE: To describe the clinical presentation, diagnosis and treatment of periorbital extranodal natural killer/T-cell lymphoma, nasal type. METHODS: Case series of three patients with periorbital involvement of extranodal natural killer/T-cell lymphoma, nasal type, of whom clinical data, orbital imaging and immunohistochemical analysis were collected. For the purpose of this study, all histopathological and immunohistochemical slides were re-examined. RESULTS: All patients presented with painless eyelid swelling and a history of sinonasal disease, of whom one with bilateral panuveitis, not responding to systemic antibiotics. Extraocular muscle involvement was present in 2 cases upon presentation and in 1 case later on. Initial paranasal and orbital biopsies were negative in 2 patients, with only the second orbital biopsy leading to the diagnosis. Natural killer/T-cell and cytotoxic markers were present in all cases, as well as Epstein-Barr virus encoded RNA in situ hybridization. The patients died respectively 5, 9 and 35 months from diagnosis despite treatment with chemotherapy and radiotherapy. CONCLUSION: Extranodal natural killer/T-cell lymphoma, nasal type, should be suspected in a painless periorbital cellulitis with chronic sinusitis, not responding to conventional therapy. A high index of suspicion is necessary in biopsies showing angiodestruction and necrosis. Epstein-Barr virus encoded RNA in situ hybridization and expert hematopathologist consultation is necessary to decrease the delay in diagnosis.

The transconjunctival orbitotomy: A versatile approach to the orbit and beyond.

In the management of orbital disorders and defects, minimally invasive surgical approaches have become increasingly efficient for their reduction of operative trauma and access without compromise of therapeutic benefit or diagnostic yield. Various approaches have focused on bone- and canthal-sparing techniques and concealed and small skin incisions. We review the current state of knowledge of procedures to enter the orbit via the conjunctiva. Any quadrant of the orbit can be accessed via the conjunctiva. Surgical incisions involve the orbital palpebral, forniceal, and bulbar conjunctiva. According to the location, nature, and size of the lesion, the transconjunctival orbitotomy can be used as a single procedure, in combination with a caruncular approach or as an adjunct in a multidisciplinary procedure for lesions extending deep into or outside the orbit. The working space and field of operating view can be expanded by releasing the horizontal tension of the eyelid with a lateral cantholysis, lateral paracanthal blepharotomy, or medial lid split procedure. Complications related to the conjunctival incision are reduced to dry eye disease.

The Immunoarchitecture of Human Extraocular Muscles.

Purpose: The purpose of this study was to describe the immunoarchitecture of normal extraocular muscles (EOMs) in terms of presence, distribution, and organization of various immune cells. Methods: We performed unilateral orbital exenterations in six fresh human cadavers from elderly patients, followed by dissection of the medial, lateral, superior and inferior rectus, superior and inferior oblique, and superior palpebral levator muscle in their entirety. We further cross sectioned each EOM in an anterior, central, and posterior third. After immunohistochemical staining for CD3, CD8, CD20, CD138, CD68, and podoplanin, quantitative analysis was performed. Results: We found all EOMs (rectus, oblique, and levator muscles) to harbor both T- and B-lymphocytes, with a B-lymphocyte dominance and an absence of plasma cells. The highest prevalence of immune cells was seen in the muscle bellies, with, on average, 488 ± 63 CD3+ T-lymphocytes and 44 ± 110 CD20+ B-lymphocytes per mm2, and significant differences from the anterior (T-lymphocytes) and posterior (T- and B-lymphocytes) thirds. T- and B-lymphocytes were primarily organized in hotspots in the vicinity of blood vessels. In addition, a small resident population of macrophages scattered throughout the specimens was detected. No lymphatic vessels were found in any of the EOMs. Conclusions: These findings can serve as a reference dataset in the assessment of EOM biopsies in the diagnostic process of inflammatory orbital and systemic disorders. Moreover, from a regenerative perspective, our results highlight the importance of taking into account the presence of a resident immune cell population when studying the host immune response on transplanted tissues or engineered constructs.

Management of thyroid eye disease: a Consensus Statement by the American Thyroid Association and the European Thyroid Association.

Thyroid eye disease (TED) remains challenging for clinicians to evaluate and manage. Novel therapies have recently emerged, and their specific roles are still being determined. Most patients with TED develop eye manifestations while being treated for hyperthyroidism and under the care of endocrinologists. Endocrinologists, therefore, have a key role in diagnosis, initial management, and selection of patients who require referral to specialist care. Given that the need for guidance to endocrinologists charged with meeting the needs of patients with TED transcends national borders, and to maximize an international exchange of knowledge and practices, the American Thyroid Association and European Thyroid Association joined forces to produce this Consensus Statement.

Management of Thyroid Eye Disease: A Consensus Statement by the American Thyroid Association and the European Thyroid Association.

Thyroid eye disease (TED) remains challenging for clinicians to evaluate and manage. Novel therapies have recently emerged, and their specific roles are still being determined. Most patients with TED develop eye manifestations while being treated for hyperthyroidism and under the care of endocrinologists. Endocrinologists, therefore, have a key role in diagnosis, initial management, and selection of patients who require referral to specialist care. Given that the need for guidance to endocrinologists charged with meeting the needs of patients with TED transcends national borders, and to maximize an international exchange of knowledge and practices, the American Thyroid Association and European Thyroid Association joined forces to produce this consensus statement.