Delayed onset enophthalmos and hypoglobus mimicking silent sinus syndrome following midface trauma.

Craniomaxillofacial trauma is primarily diagnosed and managed by oral and maxillofacial surgeons. Among the cases encountered, midface fractures involving orbital walls are highly prevalent. In these fractures, involvement of the orbital walls, particularly floor of the orbit, can lead to considerable aesthetic and functional limitations. From a maxillofacial perspective, indications for surgical repair of orbital floor encompass marked decrease in ocular motility, fracture affecting more than 50% of surface area, an increase in orbital volume exceeding 18% and enophthalmos greater than 2 mm. In the absence of these discernible signs, surgical intervention is not generally indicated. However, in this case, an early adolescent with a history of midface trauma and minimal orbital floor fracture 8 months earlier presented with progressively delayed onset enophthalmos and hypoglobus closely resembling features of silent sinus syndrome. The enophthalmos and hypoglobus were corrected by placing custom-made non-resorbable high-density polyethylene implant in the orbital floor. Postoperative follow-up demonstrated aesthetically and functionally satisfactory outcomes.

Ten-Year Follow-Up of Orbital Volume Augmentation With Calcium Hydroxyapatite Filler in Postenucleation Socket Syndrome.

PURPOSE: To describe the efficacy and persistence of injectable calcium hydroxyapatite (CAHY) to correct orbital volume deficit in postenucleation socket syndrome. METHODS: An observational study was conducted as a clinical review of all patients in the authors' practice who received injectable CAHY placed in the extraconal and intraconal space to increase orbital volume with a 10-year follow up. The amount of CAHY to be injected was defined according to the degree of orbital volume deficit. Patients previously treated with radiotherapy or with a conjunctival fornix insufficient to accommodate the external prosthesis were excluded. All the patients with at least 10 years of follow up were included in the study. RESULTS: Thirty-one postenucleation socket syndrome patients received injectable CAHY for orbital volume augmentation, with a 10-year follow up. The mean amount of preoperative relative enophthalmos measured by Hertel's exophthalmometry was 14.16 ± 2.15. An increase in the mean orbital volume of 3.35 ± 0.91 at 6 months and 2.97 ± 1.35 at 10 years was obtained. The mean follow-up was 219 ± 18 months (range, 184-240). Patients demonstrated clinical and cosmetic improvement that was observed to continue for 10 years. The complications were peribulbar ecchymosis, 2 extrusions of the internal prosthesis, and 2 ptosis. CONCLUSIONS: Injectable CAHY provides safe, simple, repeatable, and cost-effective technique to treat volume deficiency in the enophthalmic orbit in the long term. The volume augmentation obtained with this semipermanent filler demonstrated a lasting effect in the orbit with negligible loss of volume at 10 years.

Clinical manifestations, management, and outcomes of primary silent sinus syndrome: a systematic review.

INTRODUCTION: Silent sinus syndrome (SSS) is a rare disorder of the maxillary sinus, which may present with orbital symptoms. Most reports of silent sinus syndrome are limited to small series or case reports. This systematic review comprehensively characterizes the various clinical presentations, management, treatment, and outcomes in patients with SSS. METHODS: A systematic literature search of the PubMed, Cochrane, Web of Science, and Scopus databases. Inclusion criteria were studies describing the presentation, management, or treatment of SSS or chronic maxillary atelectasis. RESULTS: One hundred fifty-three articles were included in the final review (n=558 patients). Mean age at diagnosis was 38.8 +- 14.1 years, with a relatively even distribution among sexes. Enophthalmos and/or hypoglobus were the most frequent symptoms, along with diplopia, headache, or facial pressure/pain. Most patients (87%) underwent functional endoscopic sinus surgery (FESS), and 23.5% received orbital floor reconstruction. Post-treatment, patients had significant reductions in enophthalmos (2.67 +- 1.39 vs. 0.33 +- 0.75 mm) and hypoglobus (2.22 +- 1.43 vs. 0.23 +- 0.62 mm). Most patients (83.2%) achieved partial or total resolution of clinical symptoms. CONCLUSIONS: SSS has a variable clinical presentation, with enophthalmos and hypoglobus being most common. FESS with or without orbital reconstruction are effective treatments to address the underlying pathology and structural deficits.

Orbital Vascular Malformations: Relationship Between Enophthalmos and Clinically Apparent Distensibility with Valsalva.

PURPOSE: Determining the hemodynamic characteristics of an orbital vascular malformation is a critical step in management. The purpose of this study is to assess the relationship between enophthalmos and clinically apparent distensibility of orbital vascular malformations, to optimize imaging and treatment. METHODS: In this cross-sectional cohort study consecutive patients at a single institution were screened for study entry. Data extracted included age, sex, Hertel measurements, presence or absence of distensibility during the Valsalva maneuver, whether lesions were primarily venous or lymphatic based on imaging, and location of the lesion relative to the globe. Enophthalmos was defined as ≥ 2 mm difference from the opposite side. Parametric and nonparametric statistics were used, and linear regression was performed to examine factors predictive of Hertel measurement. RESULTS: Twenty-nine patients met the inclusion criteria. Relative enophthalmos ≥2 mm was significantly associated with distensibility ( p = 0.03; odds ratio = 5.33). Distensibility and venous dominant morphology were the 2 most important factors associated with enophthalmos on regression analysis. The relative position of the lesion anterior or posterior to the globe did not have a significant bearing on baseline enophthalmos. CONCLUSIONS: The presence of enophthalmos increases the likelihood that an orbital vascular malformation is distensible. This group of patients was also more likely to be characterized by venous dominant malformations. Baseline clinical enophthalmos may serve as a useful surrogate marker for distensibility and venous dominance, which may be useful in guiding the selection of appropriate imaging.

Assessing effectiveness of endoscope-assisted medial orbital wall fracture repair vs. no surgery using benefit-risk metrics and literature review.

PURPOSES: To evaluate clinical usefulness of endoscope-assisted medial orbital wall fracture repair via the retrocaruncular approach (rc-EAMOWFR) vs. no surgery (NS), and to perform a narrative review of relevant literature. METHODS: This was a retrospective cohort study enrolling isolated medial orbital wall fracture (IMOWF) eyes presented to two German level 1 trauma centers during a 7-year interval. The predictor variable was treatment type (rc-EAMOWFR vs. NS), and the main outcomes were late enophthalmos (LE) and retrobulbar hemorrhage (RH) assessed at 9-15 posttraumatic months. Descriptive and bivariate statistics were computed at α = 95%. Binary adjustments enabled calculation of number needed to treat (NNT), to harm (NNH), and likelihood to be helped or harmed (LHH) for demonstrating benefit-risk tradeoffs. Moreover, a narrative review was also performed. RESULTS: The sample comprised 502 patients (28.3% females; mean age, 46.5±19.2 years) with 541 IMOWF eyes (5.9% NS; 7.2% LE; 1.3% RH). Operated eyes had significantly lower LE events than NS eyes (symptomatic IMOWF: P < .0001; 95% confidence interval [CI], .03 to .16; NNT = 2 [95% CI, 1.1 to 6.1]; asymptomatic IMOWF: P < .0001; 95% CI, .01 to .07; NNT = 2 [95% CI, 1.1 to 1.8]). There were 7 (1.5%) RH events following rc-EAMOWFR (P = .99; 95% CI, .06 to 17.4; NNH = 68 [95% CI, 38.3 to 254.2]). LHH calculations posited that rc-EAMOWFR was 34 times more likely to prevent LE than to cause RH, regardless of fracture symptoms. Our results conformed to those of other 15 studies. CONCLUSIONS: The results of this study suggest that all IMOWFs be treated. rc-EAMOWFR performed in every 68 IMOWFs would be at risk of one RH event, but prevent 34 eyes from LE due to untreated fractures. Nearly 72% of untreated IMOWFs develop LE after 9 months.

Silent sinus syndrome: potentially misleading features that should be recognized.

OBJECTIVE: Silent sinus syndrome (SSS) usually manifests clinically as hypoglobus and enophthalmos. Patients may experience different symptoms and may present to an assortment of specialties and delay diagnosis and management. The objective of this article was to describe the different and sometimes misleading signs and symptoms of SSS to improve the level of suspicion and reduce time to diagnosis. METHODS: A retrospective consecutive audit of the records of all patients diagnosed with SSS between 2015 and 2019 in the Oxford Eye Hospital, Oxford University Hospitals NHS Foundation Trust. Demographic and clinical data including presentation, diagnosis, and symptoms were obtained from the patients' medical files. RESULTS: Ten patients were included; mean age was 42.5 ± 11.5 years (range, 16-56 years). Four patients were initially referred to an ophthalmologist with globe asymmetry, diplopia, eyelid asymmetry, or retraction. Three patients were initially referred to an ear, nose, and throat specialist with facial asymmetry or infraorbital paraesthesia. Two patients were referred from the maxillofacial department with an incidental finding, and the last patient was seen initially by the neurology team with headaches. CONCLUSION: SSS has a variable presentation. Patients may have common or misleading signs. Patients may attend different clinics and subspecialties, and physicians should be aware of the broad range of presenting signs in this condition to prevent delay in diagnosis and further morbidity.

[Ocular involvement in Parry-Romberg syndrome]. / Porazhenie organa zreniya pri sindrome Parri­Romberga.

Parry-Romberg syndrome (PRS) is a rare degenerative disease of unknown etiology, characterized by slow progressive hemifacial atrophy of the soft tissues (subcutaneous fat, muscles), cartilage and bones. Beside hemifacial atrophy, various ophthalmic and periocular manifestations of PRS has been described. Progressive enophthalmos and eyelid changes are only some of many other symptoms, as the disease can affect all layers of the eyeball. This article reviews literature on ophthalmic manifestations of PRS and reports our own case and treatment approach for a patient with this pathology.

Orbital Index: A Novel Comprehensive Quantitative Tool for Prediction of Delayed Enophthalmos in Orbital Floor Fracture Management.

SUMMARY: Early identification of surgical indication is critical to optimizing outcomes in orbital floor fracture management. However, identifying those at risk for delayed enophthalmos and requiring subsequent repair has remained a challenge. This study aimed to validate the Orbital Index, a prediction tool using computed tomography to stratify risk for delayed enophthalmos and establish a threshold for repair. The Orbital Index stratifies fractures by size, location, and inferior rectus rounding (a surrogate for fascioligamentous sling disruption) on a scale 0 to 6. A 22-year (1998 to 2020) multi-institution analysis of unilateral isolated orbital floor fractures was performed. Index scores were assigned to each scan, unoperated patients invited for blinded Hertel exophthalmometry assessment, and enophthalmos measurements correlated with Index scores. Interobserver scoring reproducibility was assessed with weighted Cohen kappa. Preintervention and postintervention Likert scale surveys were administered to determine whether this tool improved understanding and communication. The Orbital Index demonstrated high fidelity and interobserver reproducibility and identified a score of four or greater as a surgical threshold. Of 1769 computed tomography scans, 395 met criteria and were included for analysis. Eighty of 395 were managed operatively (operative rate, 20.3 percent). Of 315 patients managed nonoperatively, 41 (13.0 percent) agreed to follow-up evaluation and 28 (68.3 percent) were found to have enophthalmos. Unoperated patients with an Orbital Index score of 4 or higher were more likely to have enophthalmos than those with a score of 3 or less ( p = 0.001). The mean weighted Cohen kappa was 0.73, corroborating reproducibility. Communication ( p = 0.0003) and ability to correctly identify surgical need ( p = 0.01) were improved with use of this tool. The Orbital Index is a reproducible tool to stratify risk for enophthalmos in orbital floor fracture management.

Symptomatic enophthalmos due to sphenoid wing dysplasia appearing over 12 years in a patient with neurofibromatosis type 1: a case report and literature review.

Sphenoid wing dysplasia (SWD) is a common orbital complication of neurofibromatosis type 1 (NF1). However, enophthalmos associated with SWD is extremely rare, and details of its natural history are unclear. We present the case of a 14-year-old boy with an early childhood diagnosis of NF1 presenting with left blepharophimosis and enophthalmos for several months. Imaging demonstrated enlargement of the left lateral SWD, progression of the posteromedial deviation of the orbital contents, and sphenoid/ethmoid sinus deformation due to left temporal lobe compression over 12 years. Two characteristic changes were revealed on imaging: enlargement of the middle cranial fossa and deformation of the sphenoid/ethmoid sinuses. The orbital contents were compressed by the intracranial pressure of the temporal lobe and were displaced posteromedially into the space created by the deformed sphenoid/ethmoid sinuses. Because orbital symptoms can gradually become apparent over years with the progression of SWD and skeletal growth, long-term follow-up of orbital symptoms is recommended in patients with NF1.

Silent Sinus Syndrome: A Case Report.

To describe a case of silent sinus syndrome in the perspective of imaging studies, on 2 January 2020 we present this case of a 26-year-old Bangladeshi man with unilateral right facial asymmetry and no sino-nasal symptoms. He was referred to the Ophthalmology Department with complain of right palpebral ptosis and facial asymmetry for 7 months. On physical examination, painless enophthalmos and hypo globus of the right eye was seen. The computed tomography scan of the paranasal air sinuses showed opacification of the right maxillary sinus along with retraction of the walls of the sinus. Regardless of the clinical doubt, the conclusion can only be given by imaging studies, radiologist play a key role here.

Bilateral Enophthalmos as an Unusual Presenting Feature of Non-Hodgkin's Orbital Lymphoma.

Lymphoma is the commonest orbital malignancy. The typical presentation is proptosis or swelling, which warrants imaging and confirmation by tissue biopsy. Enophthalmos is a much rarer clinical sign and if bilateral and symmetrical can often present late. We describe a patient who presented with bilateral enophthalmos and symptomatic, secondary entropion due to bilateral non-Hodgkin's lymphoma in which orbital fat was replaced by a monoclonal proliferation of small B cells. Low-dose orbital radiotherapy and entropion surgery relieved the patient's symptoms.

Etiology, Early Diagnosis and Proper Treatment of Silent Sinus Syndrome Based on Review of the Literature and Own Experience.

PURPOSE: The authors' aim was to review the literature in terms of the etiology of the syndrome, the frequency of Silent Sinus Syndrome (SSS) and surgical procedure, as well as to present their own experience. METHODS: The authors used PubMed, Medline, and Science Direct websites to find and review the most significant papers related to SSS. The case reports of SSS published between 2010 and 2020 were reviewed. A retrospective case review of 8 patients with SSS treated at the authors' departments was done. RESULTS: The silent sinus syndrome has been reported in both children and adults. It is relatively rare and should be differentiated from congenital sinus hypoplasia or atelectasis. It most often affects the maxillary sinus. SSS is usually diagnosed when facial asymmetry or vision problems occur. Late diagnosis requires endoscopic sinus surgery, involving orbital wall reconstruction. The etiology of the syndrome, including the role of bacterial flora found in the sinuses, is unclear. CONCLUSION: Early diagnosis of SSS enables avoiding orbital complications and limits surgical intervention to endoscopic surgery. Further research into bacteriology may help to understand the pathophysiology of the silent sinus syndrome.

Silent Sinus Syndrome Secondary to Lymphoma: An Unusual Case With Radiological Evidence of Rapid Progression.

OBJECTIVE: To describe a case of silent sinus syndrome secondary to malignancy and discuss the pertinent clinical findings. Silent Sinus Syndrome (SSS) refers to a rare, asymptomatic condition whereby occlusion of the maxillary sinus ostium results in gradual resorption of air, creation of negative pressure and collapse of the maxillary walls. METHODS: Review of medical records and literature review using NCBI/PubMed. RESULTS: We describe a case of a 54-year-old gentleman presenting solely with enophthalmos. He had been diagnosed with stage IVa small lymphocytic lymphoma (SLL) 1.5 years prior to this, which was being managed with active surveillance. CT demonstrated severe bowing of the anterior and posterolateral wall, inferior displacement of the floor of the orbit and right enophthalmos, thus supporting a diagnosis of silent sinus syndrome. Compared to previous staging CT at the time of the lymphoma diagnosis these findings were entirely new, and soft tissue in the pterygomaxillary fissure was found to be enlarged. The patient underwent endoscopic sinus surgery and a right maxillary mega-antrostomy was performed to ventilate the maxillary sinus and prevent progression of eye symptoms. A biopsy was taken from the pterygopalatine fossa, which was confirmed to be chronic lymphocytic leukemia (CLL). CONCLUSION: This case is unique both in being secondary to malignancy, as well as being rapidly progressive given the presence of radiologically normal appearances 1.5 years prior to presentation. Although a rare condition, prompt recognition of SSS is vital to prevent ophthalmological complications. This report highlights malignancy as a potential cause in cases with focal bony remodeling.

Refining Indications for Orbital Floor Fracture Reconstruction: A Risk-Stratification Tool Predicting Symptom Development and Need for Surgery.

BACKGROUND: Challenges in orbital floor fracture management include delayed symptom onset and controversial surgical indications based on radiographic findings. This study assessed which imaging characteristics most reliably predict symptomatology to generate a tool quantifying individual need for surgery on initial presentation. METHODS: The clinical course for all patients with isolated orbital fractures at a single institution from 2015 to 2017 were reviewed. Trauma mechanism, computed tomographic scan findings, and symptoms necessitating surgery (diplopia, enophthalmos) were noted. Univariable and multivariable regression modeling was used to generate a predictive risk model for operative fractures. RESULTS: One hundred twenty-one patients with isolated orbital fractures were identified. Mechanism of injury included falls (41 percent), assault (37 percent), and vehicular trauma (17 percent). Patient follow-up averaged 4.4 ± 4.8 months. Average orbital floor fracture area was 2.4 cm2 (range, 0.36 to 6.18 cm2), and orbital volume herniation averaged 0.70 cm3 (range, 0.01 to 4.23 cm3). Twenty-one patients (17.3 percent) required surgical intervention for symptomatic fractures. The strongest predictors of symptoms were orbital volume increase greater than 1.3 cm3 (OR, 10.5; p = 0.001) and inferior rectus displacement within/below the fracture line (OR, 3.7; p = 0.049). Mechanical fall was risk-reducing (OR, 0.08; p = 0.005). Symptom risk was stratified from low (3.6 percent) to high risk (71 percent) (C-statistic = 0.90). The volume of herniated orbital contents was significantly more predictive of symptoms than fracture area (C-statistic = 0.81 versus C-statistic = 0.66; p = 0.02). CONCLUSIONS: The proposed risk tool allows highly accurate, early prediction of symptomatic orbital floor fractures. Findings suggest that orbital volume change, not fracture area, more reliably informs operative indications, along with inferior rectus muscle caudal malposition. A simplified stepwise decision algorithm demonstrates the potential utility of this risk-assessment tool. CLINICAL QUESTION/LEVEL OF EVIDENCE: Risk, III.

Self-centering second-generation patient-specific functionalized implants for deep orbital reconstruction.

Deep and complete reconstruction of the orbital cavity has been shown to be essential for preventing enophthalmos and hypoglobus in patients with orbital defects or deformities. Additively manufactured patient-specific titanium implants provide unlimited options in design. However, implant malpositioning can still occur, even when intraoperative imaging and navigation are used. In this study, we investigated novel orbital implants containing features facilitating self-centering. Accuracy of implant placement and reconstruction of the orbital dimensions were compared retrospectively between self-centering second-generation patient-specific functionalized orbital implants (study group) and CAD-based individualized implants (control group). Design features of implants in the study group included functionalization with navigation tracks, a preventive design, and flanges - so called stabilizers - towards opposite orbital walls. Implant position was evaluated by fusion of preoperative virtual plans and the post-therapeutic imaging. Aberrances were quantified by 3D heatmap analysis. 31 patients were assigned to the study group and 50 to the control group, respectively. In the study group, most implants were designed with either one (n = 18, 58.06%) or two (n = 10, 32.26%) stabilizers. Twice (6.45%), one stabilizer had to be shortened intraoperatively. Implant fit analysis revealed a significantly more precise (p < 0.001) positioning in the study group (n = 22/31) than in the control group (n = 42/50). Self-centering second-generation patient-specific functionalized orbital implants showed significantly more accurate implant positioning, facilitating the transformation of virtual plans into patient's anatomy. The presented design provides an additional instrument for intraoperative quality control besides intraoperative imaging and navigation.