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.

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.

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.

Three-dimensional design of a geometric model for an ocular prosthesis in ex vivo anophthalmic socket models.

PURPOSE: Fitting a customized ocular prosthesis for anophthalmic patients entails an artisanal labour-exhausting process and is standardly based on impression moulding of the socket, which may be anatomically inaccurate. The objective of the study was to design an impression-free socket mould with three-dimensional (3D) technology. METHODS: The ex vivo anophthalmic socket models included one silicone, one fresh pig cadaver head and three fresh-frozen human cadaver heads. After intra-socket application with iodine substance, five observers obtained eighteen low-dose cone beam computed tomography (CBCT) scans and one observer one high-dose CBCT scan of each model. The observers designed non-impression 3D moulds of the socket with 3D software. For the human cadaver sockets 3D geometric models of the ocular prosthesis were rendered from the 3D mould of the socket and the mirrored cornea of the contralateral eye. RESULTS: The posterior surface of the 3D mould was highly accurate, with a mean absolute deviation of 0.28 mm, 0.53 mm, 0.37 mm and mean upper deviation of 0.53 mm, 0.86 mm, 1.17 mm, respectively, for the phantom, pig and human model. The intra- and interobserver repeatability and reproducibility of the 3D moulds and designs was good (<0.35 mm). The largest variation in the 3D geometric model was found at the junction of the 3D mould and mirrored cornea. CONCLUSION: 3D design of an impression-free geometric model for an ocular prosthesis with low-dose CBCT is highly accurate in ex vivo anophthalmic socket models. This novel method is a critical step towards the manufacturing of 3D printed ocular prostheses and requires validation in anophthalmic patients.

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.

The prevalence of anophthalmic socket syndrome and its relation to patient quality of life.

BACKGROUND: Following removal of the eye, soft tissue changes of the eyelids and orbit may develop into an anophthalmic socket (AS) syndrome, which is often attributed to orbital volume deficiency. While adequate primary orbital volume replacement is nowadays standard of care, patients may still present with facial asymmetry. The aim of this study is to provide insights into these changes and their impact on patient quality of life (QoL). METHODS: Cross-sectional study of 59 patients with longstanding ocular prosthetic wear after enucleation or evisceration surgery. The alignment, function, and laxity of the eyelids of the anophthalmic side were compared to those of the fellow side. The QoL was assessed with a 4-item questionnaire specific for the prosthetic condition. The different aspects of AS syndrome were analysed in relation to disease-specific and prosthetic data and to the patient QoL scores. RESULTS: Clinical AS syndrome was prevalent in 53% of patients with acquired anophthalmia. The anophthalmic side was statistically significantly different from the fellow side for the known AS syndrome features such as superior sulcus depression, margin reflex distance 1, and enophthalmia, but also for new features such as levator muscle function and lagophthalmia (P < 0.05). The difference was correlated with duration of prosthetic wear, prior orbital radiotherapy, and size of the prosthesis (P < 0.05). QoL scores were not correlated to the separate features of AS syndrome, except for a positive correlation between wearing comfort of the prosthesis and upper eyelid ptosis (P < 0.05). CONCLUSION: Patients with an ocular prosthesis show a relatively high prevalence of one or more distinct clinical features of AS syndrome, which do not negatively affect patient QoL. These findings underscore the importance to tailor prosthetic and surgical treatment to the patient's perceived QoL rather than to the objective clinical findings.

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.

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 orbital mass lesions: clinical, radiological, and pathological recommendations.

The orbit can harbor mass lesions of various cellular origins. The symptoms vary considerably according to the nature, location, and extent of the disease and include common signs of proptosis, globe displacement, eyelid swelling, and restricted eye motility. Although radiological imaging tools are improving, with each imaging pattern having its own differential diagnosis, orbital mass lesions often pose a diagnostic challenge. To provide an accurate, specific, and sufficiently comprehensive diagnosis, to optimize clinical management and estimate prognosis, pathological examination of a tissue biopsy is essential. Diagnostic orbital tissue biopsy is obtained through a minimally invasive orbitotomy procedure or, in selected cases, fine needle aspiration. The outcome of successful biopsy, however, is centered on its representativeness, processing, and interpretation. Owing to the often small volume of the orbital biopsies, artifacts in the specimens should be limited by careful peroperative tissue handling, fixation, processing, and storage. Some orbital lesions can be characterized on the basis of cytomorphology alone, whereas others need ancillary molecular testing to render the most reliable diagnosis of therapeutic, prognostic, and predictive value. Herein, we review the diagnostic algorithm for orbital mass lesions, using clinical, radiological, and pathological recommendations, and discuss the methods and potential pitfalls in orbital tissue biopsy acquisition and analysis.

Jones lacrimal bypass tubes in children and adults.

BACKGROUND/AIMS: Although a Jones tube is considered the mainstay for epiphora in patients with total blockage of the canalicular system, it has been discouraged in children for reasons of inadequate self-care and maintenance. The purpose of this study is to compare the long-term outcome of Jones tube surgery in paediatric versus adult patients. METHODS: Retrospective, interventional case series of a single academic institution. The medical records of all children (≤16 years old) and adults (>16 years old) who underwent conjunctivorhinostomy with placement of a 130° angled extended Jones tube were reviewed. The outcome measures were patency and anatomical position of the tube, type and frequency of complications and subjective relief of epiphora. RESULTS: The study included 10 children (11 eyes) (range, 5.1-16.0 years old) and 102 adults (127 eyes) (range, 19.7-82.4 years old). The success and complication rate did not differ between the two age groups. Tube dislodgement and obstruction occurred in 4 (36.4%) of the paediatric tubes with an incidence rate of 6.1%/year and in 47 (37.0%) of the adult tubes with an incidence rate of 9.3%/year (p=0.3867). Two adults required routine self-irrigation of the tube. The median follow-up was 6.7 years for the children and 8.7 years for the adults (p=0.3430). CONCLUSION: With a similar outcome profile and minimal self-care, young age is not a prognostic nor limiting factor for surgery with angled Jones tubes. Exchange with tubes of a longer length is not required during growth.

Orbital tumor associated with a microphthalmic eye and colobomatous cleft: Pilocytic astrocytoma (glioma) or massive retinal gliosis.

A 11-year-old boy with congenital microphthalmos of the right eye presented with gradual protrusion of his ocular prosthesis. MRI showed an orbital mass adjacent to the microphthalmic eye. After removal of the eye and the orbital soft tissue mass a gliotic mass, resembling a pilocytic astrocytoma WHO grade 1 (glioma) was diagnosed. Through a colobomatous cleft in the eye the tumour spread in the orbit. There were no clinical signs of neurofibromatosis 1. This case showed a very rare association between a microphthalmic and colobomatous eye and pilocytic astrocytoma, grade 1. However a far advanced and infiltrative massive retinal gliosis cannot be definitively excluded as differential diagnosis.

Use of the Masquerade Flap in Ablepharon-Macrostomia Syndrome: A Case Report.

PURPOSE: To report a case of ablepharon-macrostomia syndrome and surgical treatment options. METHODS: Case report and literature review. RESULTS: A prematurely born male baby presented with severe ablepharon, hypertelorism, macrostomia, low-set dysplastic ears, broad nasal bridge, coarse and redundant body skin, absent scalp and body hair, lax abdominal wall, absent nipples, camptodactyly, and ambiguous genitalia. Despite intensive ocular lubrication, severe exposure keratopathy developed within the first days after birth. The eyes were closed using masquerade flaps for 6 weeks. In a secondary procedure at the adjusted age of 3 weeks, the flaps were partially divided, and visual input and development were successfully achieved, while maintaining corneal protection. CONCLUSIONS: We present a rare case of a prematurely born infant with a severe phenotype of ablepharon-macrostomia syndrome, surgically treated with masquerade flaps to preserve corneal health and allow bilateral visual input.

Consensus on Diagnostic Criteria of Idiopathic Orbital Inflammation Using a Modified Delphi Approach.

Importance: Current practice to diagnose idiopathic orbital inflammation (IOI) is inconsistent, leading to frequent misdiagnosis of other orbital entities, including cancer. By specifying criteria, diagnosis of orbital inflammation will be improved. Objective: To define a set of criteria specific for the diagnosis of IOI. Design, Setting, and Participants: A 3-round modified Delphi process with an expert panel was conducted from June 8, 2015, to January 25, 2016. Fifty-three orbital scientist experts, identified through membership in the Orbital Society, were invited to participate in on online survey and they scored, using 5-point Likert scales, items that are eligible as diagnostic criteria from the literature and from personal experience. The items were clustered around the anatomic subtypes of IOI: idiopathic dacryoadenitis and idiopathic orbital fat inflammation (2 nonmyositic IOIs), and idiopathic orbital myositis (myositic IOI). Items with dissensus were rescored in the second round, and all items with consensus (median, ≥4; interquartile range, ≤1) were ranked by importance in the third round. Main Outcomes and Measures: Consensus on items to be included in the criteria. Results: Of the 53 experts invited to participate, a multinational panel of 35 (66%) individuals with a mean (SD) years of experience of 31 (11) years were included. Consensus was achieved on 7 of 14 clinical and radiologic items and 5 of 7 pathologic items related to diagnosis of nonmyositic IOI, and 11 of 14 clinical and radiologic items and 1 of 5 pathologic items for myositic IOI. There was agreement among panelists to focus on surgical tissue biopsy results in the diagnosis of nonmyositic IOI and on a trial with systemic corticosteroids in myositic IOI. Panelists agreed that a maximum number of 30 IgG4-positive plasma cells per high-power field in the orbital tissue is compatible with the diagnosis of IOI. Conclusions and Relevance: An international panel of experts endorsed consensus diagnostic criteria of IOI. These criteria define a level of exclusion suggested for diagnosis and include tissue biopsy for lesions not confined to the extraocular muscles. This consensus is a step toward developing guidelines for the management of IOI, which needs to be followed by validation studies of the criteria.

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.

Objective and subjective outcomes of strabismus surgery in Graves' orbitopathy: a prospective multicentre study.

PURPOSE: To assess the change and interrelationship of the field of binocular single vision (BSV) and the quality of life (QoL), tested with two different tools, after one or two strabismus surgeries in patients with Graves' orbitopathy (GO). METHODS: Prospectively, consecutive patients with GO who were scheduled for their first strabismus surgery were recruited from five centres specialized in the treatment of GO. One week preoperatively and 3 months after the last operation, a full ophthalmic and orthoptic examination was performed. Change in field of BSV, GO-QoL and thyroid eye disease-QoL (TED-QoL) was recorded. RESULTS: A total of 59 met all the eligibility criteria of whom 15 underwent two strabismus operations. The median (interquartile range) preoperative score of the field of BSV was 0 (0-0), which improved to 73 (53-85) after the correction(s) (p < 0.001). After the first surgery, a significant higher score of the field of BSV was found in the patients who underwent one operation (76; 60-86) compared with those who underwent two 0 (0-63) operations (p < 0.001). After the second surgery, this score increased to 62 (40-76; p = 0.05). A moderate correlation was found between the score of the field of BSV and the GO-QoL visual functioning (VF) questionnaires (r = 0.485; p < 0.001). Both the GO-QoL and TED-QoL for VF and appearance (AP) showed significantly higher scores after the treatment (p < 0.001). CONCLUSION: After strabismus surgery in GO patients, both the field of BSV and quality of life questionnaires increase. In approximately 30% of the patients, an additional strabismus surgery is performed. This second surgery significantly expands the field of BSV and the quality of life.

Orbital Inflammation in Pregnant Women.

OBJECTIVE: To analyze overlaps between pregnancy and orbital inflammation (OI). DESIGN: Retrospective observational case series. METHODS: Eight new cases from 1997 to 2015 and 2 previously published cases were identified for inclusion in this investigation to provide the fullest clinical picture. Medical records, imaging studies, and the results of biopsies were reviewed. RESULTS: Three categories of association were discovered: (1) OI arising for the first time during pregnancy (5 cases); (2) OI arising within 3 months of delivery (2 cases); and (3) previously diagnosed OI reactivated or exacerbated by pregnancy (3 cases). One patient had a preexistent systemic autoimmune disease and another's was later diagnosed. One patient had attacks during sequential pregnancies. Findings included eyelid swelling and erythema, conjunctival chemosis, pain on eye movement, minimal diplopia, the usual absence of proptosis, and general preservation of visual acuity. Imaging studies disclosed extraocular muscle swelling (8 cases), most frequently of a single lateral rectus muscle. There were 2 cases of dacryoadenitis; 1 of these and an additional case displayed inflammation of the retrobulbar fat. Corticosteroids effected resolution of most symptoms. Singleton births were normal with the exceptions of an intrauterine fetal demise owing to acrania and a molar pregnancy. CONCLUSION: OI usually affects a single rectus muscle (typically the lateral) and, less often, the lacrimal gland and is often mild when it arises during or after pregnancy. Independent systemic autoimmune disease is an uncommon feature. Corticosteroids were efficacious except in 1 case with severe orbital scarring. No definitive causal relationships between pregnancy and OI could be established based on the clinical data.