Orbital Cutaneous Fistula as a Delayed Complication after Fracture Repair Surgery: A Case-based Literature Review

Orbital Cutaneous Fistula as a Delayed Complication after Fracture Repair Surgery: A Case-based Literature Review | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Case Report Orbital Cutaneous Fistula as a Delayed Complication after Fracture Repair Surgery: A Case-based Literature Review Weijie Zhang, Yuan Deng This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-7370260/v1 This work is licensed under a CC BY 4.0 License Status: Published Journal Publication published 26 Nov, 2025 Read the published version in BMC Ophthalmology → Version 1 posted 10 You are reading this latest preprint version Abstract Purpose: To describe the epidemiological changes in orbital cutaneous fistula formation and emphasize the importance of selecting implant materials for orbital fracture repair surgeries. Patient presentation: Eightpatients were included in this study. All patients had a history of orbital fracture surgery, with Medpor implanted in 3 patients, Medpor titanium in 2 patients, titanium mesh in 2 patients, and bioresorbable material in 1 patient. The average duration between orbital cutaneous fistula formation and the first operation was 4.58 ± 2.93 years. Orbital cutaneous fistulas were found around the orbital region (6 cases in the infraorbital region, 1 case in the lateral orbital region, and 1 case in the supraorbital region). The Valsalva maneuverinduced purulent secretion from the external fistula opening. Other implications include enophthalmos, limited eye mobility, diplopia, ectropion, acquired deformitiesof the epicanthal, nasolacrimal occlusion, and chronic dacryocystitis. CT imaging revealsinflammatory effusion in the surrounding paranasal sinuses (e.g., the maxillary, ethmoid and frontal sinus) before the second operation. The implanted porous materials were found intraoperatively to be poorly vascularized, with surrounding scar formation. Postoperative pathology revealed focal granulomatous inflammation. After the removal of the implants and the fistula resection operation, the functional defects were essentially repaired, with permanent impairment of appearance. Conclusion: Orbital cutaneous fistula is a late complication of orbital reconstruction surgery that requires more attention during long-term follow-up. Figures Figure 1 Figure 2 Figure 3 Figure 4 Figure 5 Introduction Orbital cutaneous fistulas are cutaneous fistulas located around the orbit, mostly arising from chronic granulomatous inflammation induced by foreign bodies [1] , and can be accompanied by purulent secretion. A previous study [2] reported that orbital cutaneous fistulas were caused mostly by penetrating trauma with foreign body residues (especially active plant-based foreign bodies). It usually occurs weeks to months after injury, leading to functional and aesthetic damage. On the basis of this consensus, as well as the development of radiography technologies such as CT/MRI, one-stage removal of orbital active foreign bodies is generally carried out[ 3 ], which significantly reduces the incidence of orbital cutaneous fistula caused by foreign bodies. However, with the widespread development of orbital fracture reconstruction operations and the extension of follow-up time, orbital cutaneous fistulas related to implants have attracted our attention. This complication usually occurs several years after the first reconstruction surgery. In this study, we retrospectively reviewed 8 patients with orbital cutaneous fistulas after primary orbital reconstruction surgery and analyzed their clinical manifestations and pathological etiology to provide a basis for selecting implant materials for orbital fracture repair surgeries. Case Presentation Patient 1, Patient 2 and Patient 7. These 3 patients had Medpor implantation histories. The intervals between primary orbital reconstruction surgery and the formation of orbital cutaneous fistulas were 9 years, 6 years and 5 years (Table 1 ). Patient 1 (Fig. 1 A) reported redness and swelling of the left facial region when spicy or irritating food was eaten after the first surgery, which could be controlled with oral antibiotic therapy. Orbital cutaneous fistula at the infraorbital region with purulent discharge when performing the Valsalva maneuver 2 months before the second surgery, accompanied by 3 mm of measurable enophthalmos. Patient 2 (Fig. 1 B) experienced recurring redness and swelling of the lateral canthus of the left eye 3 months before the second surgery, accompanied by purulent discharge when the Valsalva maneuver was performed, diplopia and epiphora. The ophthalmic examination revealed hypophasis, ectropion of the lower eyelid, restricted eye movement, and 3 mm of measurable enophthalmos. Patient 7 (Fig. 1 G) presented with a soft tissue mass, local redness and swelling, and intermittent purulent discharge 5 years after the first orbital reconstruction surgery. Moreover, these 3 patients all presented with purulent infiltration in the maxillary sinus and chronic inflammation of periorbital soft tissue (Fig. 2 A-D). The Medpor implants with a rarely vascularized appearance (Fig. 3 A) were removed during the second reconstruction surgery. After the second reconstruction surgery, the infraorbital region depression and lower eyelid ectropion of patients 1 and 2 remained at the 3rd month of follow-up. (Fig. 5 A, 5 B). Table 1 Demographic data Case Primary implants Interval Indication for secondary surgery Fistula location Secondary implants 1 Medpor 9 years Recurring redness and swelling, fistula with purulent discharge when performing valsalva maneuver, enophthalmos Infraorbital region Rapidsorb 2 Medpor 6 years Recurring redness and swelling, fistula with purulent discharge when performing valsalva maneuver, diplopia, and epiphora Lateral orbital region Rapidsorb 3 Medpor Titanium 5 years Fistula with purulent discharge when performing valsalva maneuver Infraorbital region Rapidsorb 4 Medpor Titanium 6 years soft tissue mass, ectropion, restricted eye movement, diplopia, and epiphora Infraorbital region Rapidsorb 5 Rapidsorb 2 months Soft tissue mass, local redness and swelling Infraorbital region Rapidsorb 6 Titanium 5 years Soft tissue mass, local redness and swelling Supraorbital/ subfrontal region Titanium 7 Medpor 5 years Soft tissue mass, local redness and swelling, fistula with purulent discharge when performing valsalva maneuver Infraorbital region Titanium 8 Titanium 6 months Deformity of inner canthus, enophthalmos, soft tissue mass, local redness and swelling, nasolacrimal occlusion, chronic dacryocystitis, purulent secretion fistula with purulent discharge when performing valsalva maneuver Infraorbital region Titanium Interval: Interval between the primary orbital reconstruction surgery and orbital fistula formation Patients 3 and 4. These 2 patients had histories of Medpor Titanium implantation. Patient 3 reported fistula formation with purulent discharge via the Valsalva maneuver at the suborbital region 5 years after the first surgery (Fig. 1 C). Patient 4 (Fig. 1 D) presented with symptoms such as a round soft tissue mass or nodule with a local depression in the suborbital region, ectropion of the lower eyelid, restricted eye movement, diplopia, and epiphora 6 years after the first surgery. Moreover, the maxillary sinus of both patients was filled with pus, as shown by CT scanning (Fig. 3 E-H), which we inferred to have resulted mostly from the excellent structural wrapping performance of the Medpor titanium implants. We also noted erosion of the implants (Fig. 2 B) in Patient 3, which contradicts the consensus that Medpor Titanium is a permanent material. Patient 5. This patient was implanted with bioresorbable material (Rapidsorb) during primary orbital repair surgery, and 2 months later, a soft tissue mass accompanied by local redness and swelling was found in the inferior orbital region, without surface ulceration (Fig. 1 E). The treatment for this patient included removal of the bioresorbable implants combined with titanium implantation. Patients 6 and 8. Patient 6 (Fig. 1 F) underwent titanium implantation during the first orbital construction surgery 5 years prior. An orbital cutaneous soft tissue mass without surface ulceration formed in the supraorbital region, with local redness and swelling 3 months before the second surgery. CT revealed frontal sinus empyema (Fig. 3 I-L). Patient 8 (Fig. 1 H) was implanted with titanium material during the first orbital reconstruction 3 months prior. This patient had severe deformity of the inner canthus and inferior orbital region, 4 mm measurable enophthalmos, and limited eye movement. In addition to purulent discharge from the orbital cutaneous fistula, patient 8 also presented symptoms of nasolacrimal occlusion and secondary dacryocystitis. For treatment, an incision at the fistula site was made deep into the orbital periosteum. The entire fistula and the surrounding granulation tissues were then thoroughly excised with an incision at the fistula site through the skin. Meanwhile, the primary implants were removed, and the adjacent paranasal sinus was thoroughly irrigated with antibiotic lavage fluid. Moreover, bioresorbable implants (in 5 patients) or titanium implants (in 3 patients) were used for secondary orbital reconstruction (Table 1 ). After treatment, the fistulae of 7 patients were closed, and there was no swelling or redness in the periorbital region. Additionally, fluid in the sinus cavity disappeared. However, the orbital cutaneous fistula of Patient 6 did not heal after removal of the titanium implant. We speculated that frontal sinusitis resulted from nasal frontal fracture, frontal sinus opening surgery was conducted under nasal endoscopy, and the orbital cutaneous fistula healed 1 week after surgery. Plastic surgery was performed in the same way to correct ectropion in Patient 4 (Fig. 4 A). In Patient 8, lacrimal sac excision was performed considering the extensive cicatricial adhesion around the lacrimal drainage and lacrimal sac abscess. Other complications, including ectropion in Patient 1 (Fig. 4 B) and cicatricial blepharon deformities in Patient 3 (Fig. 4 C), required further elective surgeries to obtain a satisfactory appearance and function. Postoperative pathology revealed chronic granulomatous inflammation around the implants (Fig. 5 ), and the inner wall of the fistula was covered by squamous epithelium. Discussion Previous studies have reported that the long-term complications of implants used in orbital reconstruction include infection, hemorrhage, implant migration, exposure, extrusion and sino-orbital cutaneous fistula[ 4 – 11 ]. In addition, orbital cutaneous fistula, as a rather rare complication, occurred after silicon[ 12 ] and Medpor[ 13 ] implant reconstruction was described in 2 case reports. The leading etiological factor of orbital cutaneous fistula has been reported to be trauma-induced foreign body retention[ 2 ]. In this study, we reported a case series of orbital cutaneous fistulas related to implants in orbital reconstruction, which seriously affected patient appearance, severely undermined the quality of life of patients, and usually required plastic surgery. The results revealed that orbital cutaneous fistulas often occur years after reconstructive surgery as late complications. Typical clinical manifestations include cutaneous fistula or soft tissue masses located in the periorbital region, adjacent orbital or facial swelling when spicy or irritating foods are eaten, and intermittent purulence discharge when the Valsalva maneuver is performed. Moreover, the accompanying necrosis of periorbital soft tissue often results in local depressed deformation and eyelid ectropion. CT scanning revealed an irregular high-density soft tissue mass, mucosal tissue with sinus wall thickening, and effusion in the sinus cavity. The causes of orbital cutaneous fistula formation after orbital reconstruction surgery are as follows: local chronic inflammatory reactions and the migration of epithelial cells[ 2 ]. Chronic inflammatory reactions are the initial factors resulting in orbital cutaneous fistulas. The residual foreign bodies, which refer to various reconstructed implants, stimulate chronic inflammatory reactions, leading to granulation proliferation, abscess formation, and, eventually, orbital cutaneous fistula formation. Moreover, adjacent nasal sinusitis is also a source of inflammation after orbital reconstruction surgery. In this study, 2 patients (patient 1 and patient 2) experienced local redness or swelling before definite cutaneous fistula formation or incorporation of purulent discharge, which proved that the fistula might have been mild or vague for a relatively long period of time. In addition, radiological examination can reveal only paranasal sinus effusion or mucosal thickening around the implants, which is not specific for diagnosis. Another necessary factor for fistula formation is the layered epithelium lining the inner wall of the orbital cutaneous fistula. The epithelial cells usually come from two sources: squamous epithelial cells migrating from the wounded skin and columnar epithelial cells from the paranasal sinus mucosa[ 14 ]. The latter source could also explain why orbital cutaneous fistula, a postoperative complication of orbital exenteration surgery, occurs more often in patients with ethmoid sinus involvement and medical orbital fracture[ 14 , 15 ]. In addition, as the conjunctival approach is now widely applied as an advanced minor incision in orbital fracture repair operations, conjunctival epithelial cells can also be a latent epithelial source of the fistula. In our study, the histological examinations of these 8 patients revealed squamous epithelium, which cannot easily exclude the participation of the paranasal sinus mucosae, as a mild chronic inflammatory microenvironment could stimulate squamous metaplasia. Notably, the time to fistula formation seemed longer for the Medpor and Medpor titanium implants (5–9 years) than for the bioresorbable implants (2 months) in our study. Among the different porous orbital implants used after evisceration or enucleation, Medpor results in slower and insufficient vascularization than hydrocyapatite (HA) and bioceramic implants do, resulting in significantly higher late exposure rates (more than 2 years)[ 16 ]. The early exposure rate (less than 2 years) of Medpor implants was comparable among these implant types[ 17 – 19 ]. These studies are consistent with our finding that Medpor implants tend to induce late complications. Therefore, our study indicated that long-term or even life-long follow-up is needed after orbital reconstruction. In our experience, we recommend early intervention when focal soft tissue masses or fistulas form. Management involves thorough excision of the fistula as well as the encompassing inflammatory tissues, removal of the initial implanted materials, and thorough cleaning of the maxillary sinus cavity with an antibiotic rinse. Bioresorbable or titanium materials could also be implanted at the same time to correct enophthalmos and diplopia. Systemic antibiotics were used after surgery to prevent infection. When accompanied by other complications, including local depression, ectropion, and scarring, multiple oculoplastic surgeries might be needed. In conclusion, orbital cutaneous fistula is an insidious and intractable late complication after orbital fracture reconstruction surgery. The clinical manifestations and radiographic findings of orbital cutaneous fistula at the early stage lack specificity and can easily be ignored clinically unless a fistula at the skin appears. More awareness and attention need to be given to orbital cutaneous fistulas during the long-term follow-up of orbital fracture surgeries. Other possible risk factors include implant migration, communication with paranasal canals, chronic nasosinusitis, compound fractures and transconjunctival surgical incisions, etc. Moreover, granulomatous reactions around the implants shown by radiographic imaging need more attention and early intervention. Conclusion In this study, we report a case series of orbital cutaneous fistulas as rare complications of orbital reconstruction surgery and provide a detailed spectrum of clinical manifestations, radiological investigations, management strategies and outcomes. On the basis of these findings, we believe that long-term follow-up and early intervention are needed to prevent orbital cutaneous fistula formation. Declarations Data availability The datasets generated and analyzed during the current study are available from the corresponding author upon reasonable request Acknowledgment No acknowledgments exist. Authors’ contributions Weijie Zhang: Writing-Original draft, data analysis, project administration. Yuan Deng: Conceptualization, Resources, Writing-Review and Editing, Supervision. Funding This research was funded by the National Natural Science Foundation of China (82301165) and the Fundamental Research Funds for the Central Universities (YG2023QNA24). Ethics approval and consent to participate All participating patients provided informed consent. The study adhered to the tenets of the Declaration of Helsinki. Ethical approval was obtained from the Ethics Committee of Shanghai Ninth People’s Hospital (SH9H-2023-T375-1). Consent for publication Consent for publication and signed informed consent was obtained from all patients. Competing interests The authors declare that they have no competing interests. References Nasr AM, Haik BG, Fleming JC, Al-Hussain HM, Karcioglu ZA: Penetrating orbital injury with organic foreign bodies. Ophthalmology 1999, 106(3):523-532. Wang WJ, Li CX, Sebag J, Ni C: Orbital fistula. Causes and treatment of 20 cases. Archives of ophthalmology (Chicago, Ill : 1960) 1983, 101(11):1721-1723. Zhao Y, Li Y, Li Z, Deng Y: Removal of Orbital Metallic Foreign Bodies With Image-Guided Surgical Navigation. Ophthalmic plastic and reconstructive surgery 2020, 36(3):305-310. Choudhry OJ, Christiano LD, Arnaout O, Adel JG, Liu JK: Reconstruction of pterional defects after frontotemporal and orbitozygomatic craniotomy using Medpor Titan implant: cosmetic results in 98 patients. Clin Neurol Neurosurg 2013, 115(9):1716-1720. Dubois L, Steenen SA, Gooris PJJ, Bos RRM, Becking AG: Controversies in orbital reconstruction-III. Biomaterials for orbital reconstruction: a review with clinical recommendations. Int J Oral Maxillofac Surg 2016, 45(1):41-50. Meyer DR: Alloplastic materials for orbital surgery. Current opinion in ophthalmology 1995, 6(5):43-52. Brown AE, Banks P: Late extrusion of alloplastic orbital floor implants. Br J Oral Maxillofac Surg 1993, 31(3):154-157. Massaro-Giordano M, Kirschner RA, Wulc AE: Orbital floor implant migration across the ethmoidal sinuses and nasal septum. American journal of ophthalmology 1998, 126(6):848-850. Gilhotra JS, McNab AA, McKelvie P, O'Donnell BA: Late orbital hemorrhage around alloplastic orbital floor implants: a case series and review. Clinical & experimental ophthalmology 2002, 30(5):352-355. McNab AA: Sino-orbital fistula: two case reports. Clinical & experimental ophthalmology 2000, 28(4):324-325. Levine MR, Perla B: Maxillary sinus orbital fistula secondary to repair of an orbital floor fracture. Ophthalmic plastic and reconstructive surgery 1995, 11(1):66-69. Huelse R, Freuschle A, Hörmann K, Stuck BA: [Orbitocutaneous fistula after orbital reconstruction]. HNO 2013, 61(4):344-346. Cho W-K, Ko AC, Korn BS, Kikkawa DO: Orbitocutaneous Fistula Secondary to Buried Polyethylene Mesh Implant 12 Years After Injury. Ophthalmic plastic and reconstructive surgery 2017, 33(5):e107-e108. Tassone P, Gill KS, Hsu D, Nyquist G, Krein H, Bilyk JR, Murchison AP, Evans JJ, Heffelfinger RN, Curry JM: Naso- or Orbitocutaneous Fistulas after Free Flap Reconstruction of Orbital Exenteration Defects: Retrospective Study, Systematic Review, and Meta-Analysis. J Neurol Surg B Skull Base 2017, 78(4):337-345. Limawararut V, Leibovitch I, Davis G, Rees G, Goldberg RA, Selva D: Sino-orbital fistula: a complication of exenteration. Ophthalmology 2007, 114(2):355-361. Lin CW, Liao SL: Long-term complications of different porous orbital implants: a 21-year review. Br J Ophthalmol 2017, 101(5):681-685. Jordan DR, Gilberg S, Bawazeer A: Coralline hydroxyapatite orbital implant (bioeye): experience with 158 patients. Ophthalmic plastic and reconstructive surgery 2004, 20(1):69-74. Alwitry A, West S, King J, Foss AJ, Abercrombie LC: Long-term follow-up of porous polyethylene spherical implants after enucleation and evisceration. Ophthalmic plastic and reconstructive surgery 2007, 23(1):11-15. Liao SL, Kao SC, Tseng JH, Lin LL: Surgical coverage of exposed hydroxyapatite implant with retroauricular myoperiosteal graft. Br J Ophthalmol 2005, 89(1):92-95. Additional Declarations No competing interests reported. Cite Share Download PDF Status: Published Journal Publication published 26 Nov, 2025 Read the published version in BMC Ophthalmology → Version 1 posted Editorial decision: Revision requested 01 Oct, 2025 Reviews received at journal 24 Sep, 2025 Reviews received at journal 23 Sep, 2025 Reviewers agreed at journal 25 Aug, 2025 Reviewers agreed at journal 24 Aug, 2025 Reviewers invited by journal 24 Aug, 2025 Editor assigned by journal 24 Aug, 2025 Editor invited by journal 21 Aug, 2025 Submission checks completed at journal 21 Aug, 2025 First submitted to journal 21 Aug, 2025 You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. Our growing team is made up of researchers and industry professionals working together to solve the most critical problems facing scientific publishing. Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-7370260","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":505039443,"identity":"ba5fe952-5428-4d37-8c35-14b3e2e0f21f","order_by":0,"name":"Weijie Zhang","email":"","orcid":"","institution":"Ninth People’s Hospital, Shanghai Jiao Tong University","correspondingAuthor":false,"prefix":"","firstName":"Weijie","middleName":"","lastName":"Zhang","suffix":""},{"id":505039444,"identity":"14260a55-7c69-48f5-b706-eb1c0bc0a569","order_by":1,"name":"Yuan Deng","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAAqUlEQVRIiWNgGAWjYFACHgYJBgYbHn7+BtK0pMlIzjhAmpbDNgYNCURq4JfuPXjj447zPAYMBxg/fMwhQovknHPJljPP3OYxZ25glpy5jQgtBjdyzKR5227zWDYcYGPmJVrL37ZzPAYHEkjRwth2gAQtknPOGFv2tiXzSM442EycX/ilewxv/Gyzs+fnbz744SMxWkCRAgWMDcSoR9EyCkbBKBgFowAHAACpsjQeTk+UfwAAAABJRU5ErkJggg==","orcid":"","institution":"Ninth People’s Hospital, Shanghai Jiao Tong University","correspondingAuthor":true,"prefix":"","firstName":"Yuan","middleName":"","lastName":"Deng","suffix":""}],"badges":[],"createdAt":"2025-08-14 06:08:39","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-7370260/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-7370260/v1","draftVersion":[],"editorialEvents":[{"content":"https://doi.org/10.1186/s12886-025-04526-6","type":"published","date":"2025-11-26T15:57:16+00:00"}],"editorialNote":"","failedWorkflow":false,"files":[{"id":90438186,"identity":"34d5c877-5fc5-4b7d-9579-90db568e05d8","added_by":"auto","created_at":"2025-09-02 17:32:15","extension":"png","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":12918690,"visible":true,"origin":"","legend":"\u003cp\u003eClinical presentations of the 8 patients after primary orbital reconstruction surgery.\u003cstrong\u003e \u003c/strong\u003eA-H show the orbital cutaneous fistulae of patients1--8.\u003c/p\u003e","description":"","filename":"Figure1.png","url":"https://assets-eu.researchsquare.com/files/rs-7370260/v1/8e3fbaae113c6586dc0110ac.png"},{"id":90437830,"identity":"3cccb88c-28f9-4cdd-bc4b-c99e60f0ea12","added_by":"auto","created_at":"2025-09-02 17:24:15","extension":"png","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":4347522,"visible":true,"origin":"","legend":"\u003cp\u003eCT scansof three patients with fistula formation before secondary orbital reconstruction surgery.\u003cstrong\u003e \u003c/strong\u003eLeft (A-D): Patient1. Center (E-F): Case 3. Right (I-L): Case 6. CT and 3-D scanning showing implants used for orbital floor fracture (asterisks) and maxillary or frontal sinus effusion (green arrow).\u003c/p\u003e","description":"","filename":"Figure2.png","url":"https://assets-eu.researchsquare.com/files/rs-7370260/v1/921fcbd2db2303a85f57702e.png"},{"id":90437831,"identity":"9138affa-306c-4362-ae97-b274b0276471","added_by":"auto","created_at":"2025-09-02 17:24:15","extension":"png","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":9559219,"visible":true,"origin":"","legend":"\u003cp\u003eImplants of different materials removed during secondary orbital reconstruction surgeries. A. Mediative implant of Patient 1 with an appearance of insufficient vascularization. B. Medially worn titanium implant in Patient 3 with local erosion and insufficient vascularization. C. Remnants of the RapidSorb implant from Patient 5 after degradation and absorption \u003cem\u003ein vivo\u003c/em\u003e for 2 months.\u003c/p\u003e","description":"","filename":"Figure3.png","url":"https://assets-eu.researchsquare.com/files/rs-7370260/v1/f326428c9cf1a9d89a8891bc.png"},{"id":90437832,"identity":"10f048f3-a2a3-4659-bc2a-0f1c42f0d7a3","added_by":"auto","created_at":"2025-09-02 17:24:15","extension":"png","order_by":4,"title":"Figure 4","display":"","copyAsset":false,"role":"figure","size":9587347,"visible":true,"origin":"","legend":"\u003cp\u003ePostoperative appearance of three patients after secondary orbit reconstruction and the fistula operation. A. The ectropion of the lower eyelid in Patient4 was repaired via a one-stage procedure with secondary orbit reconstruction surgery. B. Patient in Patient 1 experiencedpostoperative complications of cicatricial ectropion. C. Patient in Patient 3 presented with a depressed scar in the lower eyelid region.\u003c/p\u003e","description":"","filename":"Figure4.png","url":"https://assets-eu.researchsquare.com/files/rs-7370260/v1/533bcaf62d56116070797e2b.png"},{"id":90438876,"identity":"6bff93b6-ebc9-4698-92b5-ca2355b86179","added_by":"auto","created_at":"2025-09-02 17:40:15","extension":"png","order_by":5,"title":"Figure 5","display":"","copyAsset":false,"role":"figure","size":10359419,"visible":true,"origin":"","legend":"\u003cp\u003ePostoperative pathology. Histological examination showing the squamous epithelium lining the inner wall of the fistula (asterisk), fibrosis, and local accumulation of inflammatory cells (arrows).\u003c/p\u003e","description":"","filename":"Figure5.png","url":"https://assets-eu.researchsquare.com/files/rs-7370260/v1/0ec3d03ab3d094e967f36f9c.png"},{"id":97178303,"identity":"b194cfde-438f-4577-ad91-84a136007bd2","added_by":"auto","created_at":"2025-12-01 16:07:35","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":44954987,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-7370260/v1/44230000-fb80-453b-898c-b2fabf027d37.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"Orbital Cutaneous Fistula as a Delayed Complication after Fracture Repair Surgery: A Case-based Literature Review","fulltext":[{"header":"Introduction","content":"\u003cp\u003eOrbital cutaneous fistulas are cutaneous fistulas located around the orbit, mostly arising from chronic granulomatous inflammation induced by foreign bodies\u003csup\u003e[1]\u003c/sup\u003e, and can be accompanied by purulent secretion. A previous study\u003csup\u003e[2]\u003c/sup\u003e reported that orbital cutaneous fistulas were caused mostly by penetrating trauma with foreign body residues (especially active plant-based foreign bodies). It usually occurs weeks to months after injury, leading to functional and aesthetic damage. On the basis of this consensus, as well as the development of radiography technologies such as CT/MRI, one-stage removal of orbital active foreign bodies is generally carried out[\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e], which significantly reduces the incidence of orbital cutaneous fistula caused by foreign bodies. However, with the widespread development of orbital fracture reconstruction operations and the extension of follow-up time, orbital cutaneous fistulas related to implants have attracted our attention. This complication usually occurs several years after the first reconstruction surgery.\u003c/p\u003e\u003cp\u003eIn this study, we retrospectively reviewed 8 patients with orbital cutaneous fistulas after primary orbital reconstruction surgery and analyzed their clinical manifestations and pathological etiology to provide a basis for selecting implant materials for orbital fracture repair surgeries.\u003c/p\u003e"},{"header":"Case Presentation","content":"\u003cp\u003e\u003cb\u003ePatient 1, Patient 2 and Patient 7.\u003c/b\u003e These 3 patients had Medpor implantation histories. The intervals between primary orbital reconstruction surgery and the formation of orbital cutaneous fistulas were 9 years, 6 years and 5 years (Table\u0026nbsp;\u003cspan refid=\"Tab1\" class=\"InternalRef\"\u003e1\u003c/span\u003e). Patient 1 (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003eA) reported redness and swelling of the left facial region when spicy or irritating food was eaten after the first surgery, which could be controlled with oral antibiotic therapy. Orbital cutaneous fistula at the infraorbital region with purulent discharge when performing the Valsalva maneuver 2 months before the second surgery, accompanied by 3 mm of measurable enophthalmos. Patient 2 (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003eB) experienced recurring redness and swelling of the lateral canthus of the left eye 3 months before the second surgery, accompanied by purulent discharge when the Valsalva maneuver was performed, diplopia and epiphora. The ophthalmic examination revealed hypophasis, ectropion of the lower eyelid, restricted eye movement, and 3 mm of measurable enophthalmos. Patient 7 (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003eG) presented with a soft tissue mass, local redness and swelling, and intermittent purulent discharge 5 years after the first orbital reconstruction surgery. Moreover, these 3 patients all presented with purulent infiltration in the maxillary sinus and chronic inflammation of periorbital soft tissue (Fig.\u0026nbsp;\u003cspan refid=\"Fig2\" class=\"InternalRef\"\u003e2\u003c/span\u003eA-D). The Medpor implants with a rarely vascularized appearance (Fig.\u0026nbsp;\u003cspan refid=\"Fig3\" class=\"InternalRef\"\u003e3\u003c/span\u003eA) were removed during the second reconstruction surgery. After the second reconstruction surgery, the infraorbital region depression and lower eyelid ectropion of patients 1 and 2 remained at the 3rd month of follow-up. (Fig.\u0026nbsp;\u003cspan refid=\"Fig4\" class=\"InternalRef\"\u003e5\u003c/span\u003eA, \u003cspan refid=\"Fig4\" class=\"InternalRef\"\u003e5\u003c/span\u003eB).\u003c/p\u003e\u003cp\u003e\u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab1\" border=\"1\"\u003e\u003ccaption language=\"En\"\u003e\u003cdiv class=\"CaptionNumber\"\u003eTable 1\u003c/div\u003e\u003cdiv class=\"CaptionContent\"\u003e\u003cp\u003eDemographic data\u003c/p\u003e\u003c/div\u003e\u003c/caption\u003e\u003ccolgroup cols=\"6\"\u003e\u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e\u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e\u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e\u003cdiv align=\"left\" class=\"colspec\" colname=\"c4\" colnum=\"4\"\u003e\u003c/div\u003e\u003cdiv align=\"left\" class=\"colspec\" colname=\"c5\" colnum=\"5\"\u003e\u003c/div\u003e\u003cdiv align=\"left\" class=\"colspec\" colname=\"c6\" colnum=\"6\"\u003e\u003c/div\u003e\u003cthead\u003e\u003ctr\u003e\u003cth align=\"left\" colname=\"c1\"\u003e\u003cp\u003eCase\u003c/p\u003e\u003c/th\u003e\u003cth align=\"left\" colname=\"c2\"\u003e\u003cp\u003ePrimary\u003c/p\u003e\u003cp\u003eimplants\u003c/p\u003e\u003c/th\u003e\u003cth align=\"left\" colname=\"c3\"\u003e\u003cp\u003eInterval\u003c/p\u003e\u003c/th\u003e\u003cth align=\"left\" colname=\"c4\"\u003e\u003cp\u003eIndication for secondary surgery\u003c/p\u003e\u003c/th\u003e\u003cth align=\"left\" colname=\"c5\"\u003e\u003cp\u003eFistula location\u003c/p\u003e\u003c/th\u003e\u003cth align=\"left\" colname=\"c6\"\u003e\u003cp\u003eSecondary\u003c/p\u003e\u003cp\u003eimplants\u003c/p\u003e\u003c/th\u003e\u003c/tr\u003e\u003c/thead\u003e\u003ctbody\u003e\u003ctr\u003e\u003ctd align=\"left\" colname=\"c1\"\u003e\u003cp\u003e1\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c2\"\u003e\u003cp\u003eMedpor\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c3\"\u003e\u003cp\u003e9 years\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c4\"\u003e\u003cp\u003eRecurring redness and swelling,\u003c/p\u003e\u003cp\u003efistula with purulent discharge when performing valsalva maneuver, enophthalmos\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c5\"\u003e\u003cp\u003eInfraorbital region\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c6\"\u003e\u003cp\u003eRapidsorb\u003c/p\u003e\u003c/td\u003e\u003c/tr\u003e\u003ctr\u003e\u003ctd align=\"left\" colname=\"c1\"\u003e\u003cp\u003e2\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c2\"\u003e\u003cp\u003eMedpor\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c3\"\u003e\u003cp\u003e6 years\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c4\"\u003e\u003cp\u003eRecurring redness and swelling,\u003c/p\u003e\u003cp\u003efistula with purulent discharge when performing valsalva maneuver, diplopia, and epiphora\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c5\"\u003e\u003cp\u003eLateral orbital region\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c6\"\u003e\u003cp\u003eRapidsorb\u003c/p\u003e\u003c/td\u003e\u003c/tr\u003e\u003ctr\u003e\u003ctd align=\"left\" colname=\"c1\"\u003e\u003cp\u003e3\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c2\"\u003e\u003cp\u003eMedpor Titanium\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c3\"\u003e\u003cp\u003e5 years\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c4\"\u003e\u003cp\u003eFistula with purulent discharge when performing valsalva maneuver\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c5\"\u003e\u003cp\u003eInfraorbital region\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c6\"\u003e\u003cp\u003eRapidsorb\u003c/p\u003e\u003c/td\u003e\u003c/tr\u003e\u003ctr\u003e\u003ctd align=\"left\" colname=\"c1\"\u003e\u003cp\u003e4\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c2\"\u003e\u003cp\u003eMedpor Titanium\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c3\"\u003e\u003cp\u003e6 years\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c4\"\u003e\u003cp\u003esoft tissue mass, ectropion, restricted eye\u003c/p\u003e\u003cp\u003emovement, diplopia, and epiphora\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c5\"\u003e\u003cp\u003eInfraorbital region\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c6\"\u003e\u003cp\u003eRapidsorb\u003c/p\u003e\u003c/td\u003e\u003c/tr\u003e\u003ctr\u003e\u003ctd align=\"left\" colname=\"c1\"\u003e\u003cp\u003e5\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c2\"\u003e\u003cp\u003eRapidsorb\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c3\"\u003e\u003cp\u003e2 months\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c4\"\u003e\u003cp\u003eSoft tissue mass, local redness and swelling\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c5\"\u003e\u003cp\u003eInfraorbital region\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c6\"\u003e\u003cp\u003eRapidsorb\u003c/p\u003e\u003c/td\u003e\u003c/tr\u003e\u003ctr\u003e\u003ctd align=\"left\" colname=\"c1\"\u003e\u003cp\u003e6\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c2\"\u003e\u003cp\u003eTitanium\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c3\"\u003e\u003cp\u003e5 years\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c4\"\u003e\u003cp\u003eSoft tissue mass, local redness and swelling\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c5\"\u003e\u003cp\u003eSupraorbital/ subfrontal region\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c6\"\u003e\u003cp\u003eTitanium\u003c/p\u003e\u003c/td\u003e\u003c/tr\u003e\u003ctr\u003e\u003ctd align=\"left\" colname=\"c1\"\u003e\u003cp\u003e7\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c2\"\u003e\u003cp\u003eMedpor\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c3\"\u003e\u003cp\u003e5 years\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c4\"\u003e\u003cp\u003eSoft tissue mass, local redness and swelling, fistula with purulent discharge when performing valsalva maneuver\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c5\"\u003e\u003cp\u003eInfraorbital region\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c6\"\u003e\u003cp\u003eTitanium\u003c/p\u003e\u003c/td\u003e\u003c/tr\u003e\u003ctr\u003e\u003ctd align=\"left\" colname=\"c1\"\u003e\u003cp\u003e8\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c2\"\u003e\u003cp\u003eTitanium\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c3\"\u003e\u003cp\u003e6 months\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c4\"\u003e\u003cp\u003eDeformity of inner canthus, enophthalmos, soft tissue mass, local redness and swelling, nasolacrimal occlusion, chronic dacryocystitis, purulent secretion fistula with purulent discharge when performing valsalva maneuver\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c5\"\u003e\u003cp\u003eInfraorbital region\u003c/p\u003e\u003c/td\u003e\u003ctd align=\"left\" colname=\"c6\"\u003e\u003cp\u003eTitanium\u003c/p\u003e\u003c/td\u003e\u003c/tr\u003e\u003c/tbody\u003e\u003c/colgroup\u003e\u003ctfoot\u003e\u003ctr\u003e\u003ctd colspan=\"6\"\u003eInterval: Interval between the primary orbital reconstruction surgery and orbital fistula formation\u003c/td\u003e\u003c/tr\u003e\u003c/tfoot\u003e\u003c/table\u003e\u003c/div\u003e\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003cp\u003e\u003c/p\u003e\u003cp\u003e\u003cb\u003ePatients 3 and 4.\u003c/b\u003e These 2 patients had histories of Medpor Titanium implantation. Patient 3 reported fistula formation with purulent discharge via the Valsalva maneuver at the suborbital region 5 years after the first surgery (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003eC). Patient 4 (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003eD) presented with symptoms such as a round soft tissue mass or nodule with a local depression in the suborbital region, ectropion of the lower eyelid, restricted eye movement, diplopia, and epiphora 6 years after the first surgery. Moreover, the maxillary sinus of both patients was filled with pus, as shown by CT scanning (Fig.\u0026nbsp;\u003cspan refid=\"Fig3\" class=\"InternalRef\"\u003e3\u003c/span\u003eE-H), which we inferred to have resulted mostly from the excellent structural wrapping performance of the Medpor titanium implants. We also noted erosion of the implants (Fig.\u0026nbsp;\u003cspan refid=\"Fig2\" class=\"InternalRef\"\u003e2\u003c/span\u003eB) in Patient 3, which contradicts the consensus that Medpor Titanium is a permanent material.\u003c/p\u003e\u003cp\u003e\u003cb\u003ePatient 5.\u003c/b\u003e This patient was implanted with bioresorbable material (Rapidsorb) during primary orbital repair surgery, and 2 months later, a soft tissue mass accompanied by local redness and swelling was found in the inferior orbital region, without surface ulceration (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003eE). The treatment for this patient included removal of the bioresorbable implants combined with titanium implantation.\u003c/p\u003e\u003cp\u003e\u003cb\u003ePatients 6 and 8.\u003c/b\u003e Patient 6 (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003eF) underwent titanium implantation during the first orbital construction surgery 5 years prior. An orbital cutaneous soft tissue mass without surface ulceration formed in the supraorbital region, with local redness and swelling 3 months before the second surgery. CT revealed frontal sinus empyema (Fig.\u0026nbsp;\u003cspan refid=\"Fig3\" class=\"InternalRef\"\u003e3\u003c/span\u003eI-L). Patient 8 (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003eH) was implanted with titanium material during the first orbital reconstruction 3 months prior. This patient had severe deformity of the inner canthus and inferior orbital region, 4 mm measurable enophthalmos, and limited eye movement. In addition to purulent discharge from the orbital cutaneous fistula, patient 8 also presented symptoms of nasolacrimal occlusion and secondary dacryocystitis.\u003c/p\u003e\u003cp\u003eFor treatment, an incision at the fistula site was made deep into the orbital periosteum. The entire fistula and the surrounding granulation tissues were then thoroughly excised with an incision at the fistula site through the skin. Meanwhile, the primary implants were removed, and the adjacent paranasal sinus was thoroughly irrigated with antibiotic lavage fluid. Moreover, bioresorbable implants (in 5 patients) or titanium implants (in 3 patients) were used for secondary orbital reconstruction (Table\u0026nbsp;\u003cspan refid=\"Tab1\" class=\"InternalRef\"\u003e1\u003c/span\u003e).\u003c/p\u003e\u003cp\u003eAfter treatment, the fistulae of 7 patients were closed, and there was no swelling or redness in the periorbital region. Additionally, fluid in the sinus cavity disappeared. However, the orbital cutaneous fistula of Patient 6 did not heal after removal of the titanium implant. We speculated that frontal sinusitis resulted from nasal frontal fracture, frontal sinus opening surgery was conducted under nasal endoscopy, and the orbital cutaneous fistula healed 1 week after surgery. Plastic surgery was performed in the same way to correct ectropion in Patient 4 (Fig.\u0026nbsp;\u003cspan refid=\"Fig5\" class=\"InternalRef\"\u003e4\u003c/span\u003eA). In Patient 8, lacrimal sac excision was performed considering the extensive cicatricial adhesion around the lacrimal drainage and lacrimal sac abscess. Other complications, including ectropion in Patient 1 (Fig.\u0026nbsp;\u003cspan refid=\"Fig5\" class=\"InternalRef\"\u003e4\u003c/span\u003eB) and cicatricial blepharon deformities in Patient 3 (Fig.\u0026nbsp;\u003cspan refid=\"Fig5\" class=\"InternalRef\"\u003e4\u003c/span\u003eC), required further elective surgeries to obtain a satisfactory appearance and function. Postoperative pathology revealed chronic granulomatous inflammation around the implants (Fig.\u0026nbsp;\u003cspan refid=\"Fig4\" class=\"InternalRef\"\u003e5\u003c/span\u003e), and the inner wall of the fistula was covered by squamous epithelium.\u003c/p\u003e\u003cp\u003e\u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003ePrevious studies have reported that the long-term complications of implants used in orbital reconstruction include infection, hemorrhage, implant migration, exposure, extrusion and sino-orbital cutaneous fistula[\u003cspan additionalcitationids=\"CR5 CR6 CR7 CR8 CR9 CR10\" citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e]. In addition, orbital cutaneous fistula, as a rather rare complication, occurred after silicon[\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e] and Medpor[\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e] implant reconstruction was described in 2 case reports. The leading etiological factor of orbital cutaneous fistula has been reported to be trauma-induced foreign body retention[\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e]. In this study, we reported a case series of orbital cutaneous fistulas related to implants in orbital reconstruction, which seriously affected patient appearance, severely undermined the quality of life of patients, and usually required plastic surgery.\u003c/p\u003e\u003cp\u003eThe results revealed that orbital cutaneous fistulas often occur years after reconstructive surgery as late complications. Typical clinical manifestations include cutaneous fistula or soft tissue masses located in the periorbital region, adjacent orbital or facial swelling when spicy or irritating foods are eaten, and intermittent purulence discharge when the Valsalva maneuver is performed. Moreover, the accompanying necrosis of periorbital soft tissue often results in local depressed deformation and eyelid ectropion. CT scanning revealed an irregular high-density soft tissue mass, mucosal tissue with sinus wall thickening, and effusion in the sinus cavity.\u003c/p\u003e\u003cp\u003eThe causes of orbital cutaneous fistula formation after orbital reconstruction surgery are as follows: local chronic inflammatory reactions and the migration of epithelial cells[\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e]. Chronic inflammatory reactions are the initial factors resulting in orbital cutaneous fistulas. The residual foreign bodies, which refer to various reconstructed implants, stimulate chronic inflammatory reactions, leading to granulation proliferation, abscess formation, and, eventually, orbital cutaneous fistula formation. Moreover, adjacent nasal sinusitis is also a source of inflammation after orbital reconstruction surgery. In this study, 2 patients (patient 1 and patient 2) experienced local redness or swelling before definite cutaneous fistula formation or incorporation of purulent discharge, which proved that the fistula might have been mild or vague for a relatively long period of time. In addition, radiological examination can reveal only paranasal sinus effusion or mucosal thickening around the implants, which is not specific for diagnosis. Another necessary factor for fistula formation is the layered epithelium lining the inner wall of the orbital cutaneous fistula. The epithelial cells usually come from two sources: squamous epithelial cells migrating from the wounded skin and columnar epithelial cells from the paranasal sinus mucosa[\u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e]. The latter source could also explain why orbital cutaneous fistula, a postoperative complication of orbital exenteration surgery, occurs more often in patients with ethmoid sinus involvement and medical orbital fracture[\u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e, \u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e]. In addition, as the conjunctival approach is now widely applied as an advanced minor incision in orbital fracture repair operations, conjunctival epithelial cells can also be a latent epithelial source of the fistula. In our study, the histological examinations of these 8 patients revealed squamous epithelium, which cannot easily exclude the participation of the paranasal sinus mucosae, as a mild chronic inflammatory microenvironment could stimulate squamous metaplasia.\u003c/p\u003e\u003cp\u003eNotably, the time to fistula formation seemed longer for the Medpor and Medpor titanium implants (5\u0026ndash;9 years) than for the bioresorbable implants (2 months) in our study. Among the different porous orbital implants used after evisceration or enucleation, Medpor results in slower and insufficient vascularization than hydrocyapatite (HA) and bioceramic implants do, resulting in significantly higher late exposure rates (more than 2 years)[\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e]. The early exposure rate (less than 2 years) of Medpor implants was comparable among these implant types[\u003cspan additionalcitationids=\"CR18\" citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR19\" class=\"CitationRef\"\u003e19\u003c/span\u003e]. These studies are consistent with our finding that Medpor implants tend to induce late complications. Therefore, our study indicated that long-term or even life-long follow-up is needed after orbital reconstruction.\u003c/p\u003e\u003cp\u003eIn our experience, we recommend early intervention when focal soft tissue masses or fistulas form. Management involves thorough excision of the fistula as well as the encompassing inflammatory tissues, removal of the initial implanted materials, and thorough cleaning of the maxillary sinus cavity with an antibiotic rinse. Bioresorbable or titanium materials could also be implanted at the same time to correct enophthalmos and diplopia. Systemic antibiotics were used after surgery to prevent infection. When accompanied by other complications, including local depression, ectropion, and scarring, multiple oculoplastic surgeries might be needed.\u003c/p\u003e\u003cp\u003eIn conclusion, orbital cutaneous fistula is an insidious and intractable late complication after orbital fracture reconstruction surgery. The clinical manifestations and radiographic findings of orbital cutaneous fistula at the early stage lack specificity and can easily be ignored clinically unless a fistula at the skin appears. More awareness and attention need to be given to orbital cutaneous fistulas during the long-term follow-up of orbital fracture surgeries. Other possible risk factors include implant migration, communication with paranasal canals, chronic nasosinusitis, compound fractures and transconjunctival surgical incisions, etc. Moreover, granulomatous reactions around the implants shown by radiographic imaging need more attention and early intervention.\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eIn this study, we report a case series of orbital cutaneous fistulas as rare complications of orbital reconstruction surgery and provide a detailed spectrum of clinical manifestations, radiological investigations, management strategies and outcomes. On the basis of these findings, we believe that long-term follow-up and early intervention are needed to prevent orbital cutaneous fistula formation.\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eData availability\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe datasets generated and analyzed during the current study are available from the corresponding author upon reasonable request\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAcknowledgment\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNo acknowledgments exist.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAuthors\u0026rsquo; contributions\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eWeijie Zhang:\u003c/strong\u003e Writing-Original draft, data analysis, project administration. \u003cstrong\u003eYuan Deng:\u003c/strong\u003e Conceptualization, Resources, Writing-Review and Editing, Supervision.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eFunding\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThis research was funded by the National Natural Science Foundation of China (82301165) and the Fundamental Research Funds for the Central Universities (YG2023QNA24).\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eEthics approval and consent to participate\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eAll participating patients provided informed consent. The study adhered to the tenets of the Declaration of Helsinki. Ethical approval was obtained from the Ethics Committee of Shanghai Ninth People\u0026rsquo;s Hospital (SH9H-2023-T375-1).\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConsent for publication\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eConsent for publication and signed informed consent was obtained from all patients.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCompeting interests\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe authors declare that they have no competing interests.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003eNasr AM, Haik BG, Fleming JC, Al-Hussain HM, Karcioglu ZA: Penetrating orbital injury with organic foreign bodies. \u003cem\u003eOphthalmology \u003c/em\u003e1999, 106(3):523-532.\u003c/li\u003e\n\u003cli\u003eWang WJ, Li CX, Sebag J, Ni C: Orbital fistula. Causes and treatment of 20 cases. \u003cem\u003eArchives of ophthalmology (Chicago, Ill : 1960) \u003c/em\u003e1983, 101(11):1721-1723.\u003c/li\u003e\n\u003cli\u003eZhao Y, Li Y, Li Z, Deng Y: Removal of Orbital Metallic Foreign Bodies With Image-Guided Surgical Navigation. \u003cem\u003eOphthalmic plastic and reconstructive surgery \u003c/em\u003e2020, 36(3):305-310.\u003c/li\u003e\n\u003cli\u003eChoudhry OJ, Christiano LD, Arnaout O, Adel JG, Liu JK: Reconstruction of pterional defects after frontotemporal and orbitozygomatic craniotomy using Medpor Titan implant: cosmetic results in 98 patients. \u003cem\u003eClin Neurol Neurosurg \u003c/em\u003e2013, 115(9):1716-1720.\u003c/li\u003e\n\u003cli\u003eDubois L, Steenen SA, Gooris PJJ, Bos RRM, Becking AG: Controversies in orbital reconstruction-III. Biomaterials for orbital reconstruction: a review with clinical recommendations. \u003cem\u003eInt J Oral Maxillofac Surg \u003c/em\u003e2016, 45(1):41-50.\u003c/li\u003e\n\u003cli\u003eMeyer DR: Alloplastic materials for orbital surgery. \u003cem\u003eCurrent opinion in ophthalmology \u003c/em\u003e1995, 6(5):43-52.\u003c/li\u003e\n\u003cli\u003eBrown AE, Banks P: Late extrusion of alloplastic orbital floor implants. \u003cem\u003eBr J Oral Maxillofac Surg \u003c/em\u003e1993, 31(3):154-157.\u003c/li\u003e\n\u003cli\u003eMassaro-Giordano M, Kirschner RA, Wulc AE: Orbital floor implant migration across the ethmoidal sinuses and nasal septum. \u003cem\u003eAmerican journal of ophthalmology \u003c/em\u003e1998, 126(6):848-850.\u003c/li\u003e\n\u003cli\u003eGilhotra JS, McNab AA, McKelvie P, O\u0026apos;Donnell BA: Late orbital hemorrhage around alloplastic orbital floor implants: a case series and review. \u003cem\u003eClinical \u0026amp; experimental ophthalmology \u003c/em\u003e2002, 30(5):352-355.\u003c/li\u003e\n\u003cli\u003eMcNab AA: Sino-orbital fistula: two case reports. \u003cem\u003eClinical \u0026amp; experimental ophthalmology \u003c/em\u003e2000, 28(4):324-325.\u003c/li\u003e\n\u003cli\u003eLevine MR, Perla B: Maxillary sinus orbital fistula secondary to repair of an orbital floor fracture. \u003cem\u003eOphthalmic plastic and reconstructive surgery \u003c/em\u003e1995, 11(1):66-69.\u003c/li\u003e\n\u003cli\u003eHuelse R, Freuschle A, H\u0026ouml;rmann K, Stuck BA: [Orbitocutaneous fistula after orbital reconstruction]. \u003cem\u003eHNO \u003c/em\u003e2013, 61(4):344-346.\u003c/li\u003e\n\u003cli\u003eCho W-K, Ko AC, Korn BS, Kikkawa DO: Orbitocutaneous Fistula Secondary to Buried Polyethylene Mesh Implant 12 Years After Injury. \u003cem\u003eOphthalmic plastic and reconstructive surgery \u003c/em\u003e2017, 33(5):e107-e108.\u003c/li\u003e\n\u003cli\u003eTassone P, Gill KS, Hsu D, Nyquist G, Krein H, Bilyk JR, Murchison AP, Evans JJ, Heffelfinger RN, Curry JM: Naso- or Orbitocutaneous Fistulas after Free Flap Reconstruction of Orbital Exenteration Defects: Retrospective Study, Systematic Review, and Meta-Analysis. \u003cem\u003eJ Neurol Surg B Skull Base \u003c/em\u003e2017, 78(4):337-345.\u003c/li\u003e\n\u003cli\u003eLimawararut V, Leibovitch I, Davis G, Rees G, Goldberg RA, Selva D: Sino-orbital fistula: a complication of exenteration. \u003cem\u003eOphthalmology \u003c/em\u003e2007, 114(2):355-361.\u003c/li\u003e\n\u003cli\u003eLin CW, Liao SL: Long-term complications of different porous orbital implants: a 21-year review. \u003cem\u003eBr J Ophthalmol \u003c/em\u003e2017, 101(5):681-685.\u003c/li\u003e\n\u003cli\u003eJordan DR, Gilberg S, Bawazeer A: Coralline hydroxyapatite orbital implant (bioeye): experience with 158 patients. \u003cem\u003eOphthalmic plastic and reconstructive surgery \u003c/em\u003e2004, 20(1):69-74.\u003c/li\u003e\n\u003cli\u003eAlwitry A, West S, King J, Foss AJ, Abercrombie LC: Long-term follow-up of porous polyethylene spherical implants after enucleation and evisceration. \u003cem\u003eOphthalmic plastic and reconstructive surgery \u003c/em\u003e2007, 23(1):11-15.\u003c/li\u003e\n\u003cli\u003eLiao SL, Kao SC, Tseng JH, Lin LL: Surgical coverage of exposed hydroxyapatite implant with retroauricular myoperiosteal graft. \u003cem\u003eBr J Ophthalmol \u003c/em\u003e2005, 89(1):92-95.\u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":true,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"bmc-ophthalmology","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"boph","sideBox":"Learn more about [BMC Ophthalmology](http://bmcophthalmol.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/boph","title":"BMC Ophthalmology","twitterHandle":"BMC_series","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true},"keywords":"","lastPublishedDoi":"10.21203/rs.3.rs-7370260/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-7370260/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003e\u003cem\u003e\u003cstrong\u003ePurpose:\u003c/strong\u003e\u003c/em\u003e\u003cstrong\u003e \u003c/strong\u003eTo describe the epidemiological changes in orbital cutaneous fistula formation and emphasize the importance of selecting implant materials for orbital fracture repair surgeries.\u003c/p\u003e\n\u003cp\u003e\u003cem\u003e\u003cstrong\u003ePatient presentation: \u003c/strong\u003e\u003c/em\u003eEightpatients were included in this study. All patients had a history of orbital fracture surgery, with Medpor implanted in 3 patients, Medpor titanium in 2 patients, titanium mesh in 2 patients, and bioresorbable material in 1 patient. The average duration between orbital cutaneous fistula formation and the first operation was 4.58 ± 2.93 years. Orbital cutaneous fistulas were found around the orbital region (6 cases in the infraorbital region, 1 case in the lateral orbital region, and 1 case in the supraorbital region). The Valsalva maneuverinduced purulent secretion from the external fistula opening. Other implications include enophthalmos, limited eye mobility, diplopia, ectropion, acquired deformitiesof the epicanthal, nasolacrimal occlusion, and chronic dacryocystitis. CT imaging revealsinflammatory effusion in the surrounding paranasal sinuses (e.g., the maxillary, ethmoid and frontal sinus) before the second operation. The implanted porous materials were found intraoperatively to be poorly vascularized, with surrounding scar formation. Postoperative pathology revealed focal granulomatous inflammation. After the removal of the implants and the fistula resection operation, the functional defects were essentially repaired, with permanent impairment of appearance.\u003c/p\u003e\n\u003cp\u003e\u003cem\u003e\u003cstrong\u003eConclusion: \u003c/strong\u003e\u003c/em\u003eOrbital cutaneous fistula is a late complication of orbital reconstruction surgery that requires more attention during long-term follow-up.\u003c/p\u003e","manuscriptTitle":"Orbital Cutaneous Fistula as a Delayed Complication after Fracture Repair Surgery: A Case-based Literature Review","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2025-09-02 17:24:10","doi":"10.21203/rs.3.rs-7370260/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Revision requested","date":"2025-10-01T07:08:09+00:00","index":"","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-09-24T11:17:26+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2025-09-23T07:51:57+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"327174132570286500991740529677079540560","date":"2025-08-25T05:44:19+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"154457244985105932396566306556511462344","date":"2025-08-24T20:29:05+00:00","index":"hide","fulltext":""},{"type":"reviewersInvited","content":"","date":"2025-08-24T20:22:48+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2025-08-24T20:21:01+00:00","index":"","fulltext":""},{"type":"editorInvited","content":"","date":"2025-08-21T15:36:33+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2025-08-21T15:07:01+00:00","index":"","fulltext":""},{"type":"submitted","content":"BMC Ophthalmology","date":"2025-08-21T14:53:33+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"bmc-ophthalmology","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"boph","sideBox":"Learn more about [BMC Ophthalmology](http://bmcophthalmol.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/boph","title":"BMC Ophthalmology","twitterHandle":"BMC_series","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"d3140df5-b1be-47f1-93d8-a540b850e274","owner":[],"postedDate":"September 2nd, 2025","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"published-in-journal","subjectAreas":[],"tags":[],"updatedAt":"2025-12-01T16:00:26+00:00","versionOfRecord":{"articleIdentity":"rs-7370260","link":"https://doi.org/10.1186/s12886-025-04526-6","journal":{"identity":"bmc-ophthalmology","isVorOnly":false,"title":"BMC Ophthalmology"},"publishedOn":"2025-11-26 15:57:16","publishedOnDateReadable":"November 26th, 2025"},"versionCreatedAt":"2025-09-02 17:24:10","video":"","vorDoi":"10.1186/s12886-025-04526-6","vorDoiUrl":"https://doi.org/10.1186/s12886-025-04526-6","workflowStages":[]},"version":"v1","identity":"rs-7370260","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-7370260","identity":"rs-7370260","version":["v1"]},"buildId":"8U1c8b4HqxoKbykW_rLl7","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}