Ophthal Plast Reconstr Surg, Vol. 32, No. 4, 2016
patients.1 Risk factors include total neutrophil count of less than 1,000/mm3, AIDS, defects in phagocytosis, hematologic malignancy, steroids, immunosuppressives, diabetes mellitus, prosthetic devices, trauma, environmental exposure (proximity to building demolition or restoration, yard work, compost heaps), and residence in endemic areas such as the Middle East especially Sudan, the Indian subcontinent and the southern United States.1 The 2 patients presented here were immunocompromised and received chemotherapy for hematologic malignancies. While lateral or retro-orbital pain is a common initial complaint of sino-orbital Aspergillus,3 acute dacryocystitis has only rarely been reported as the initial complaint. Most reported cases of lacrimal involvement have been caused by allergic fungal sinusitis and are present in immune-competent adults. Kim et al.4 recently reported a case of allergic fungal sinusitis presenting with epiphora. Their patient required multiple surgical procedures to control the infection and improve the nasolacrimal duct obstruction. Also, Petkar et al.5 reported a patient with epiphora as the only presenting sign of allergic fungal sinusitis. The patient had resolution of epiphora following sinus surgery and medical management. No lacrimal surgery was required. Finally, Facer et al.6 reported a case of bilateral nasolacrimal involvement from eosinophilic fungal sinusitis. The patient presented with a fluctuant mass just under the right medial canthus. Medical management, sinus surgery, and dacryocystorhinostomy were curative. Imaging techniques such as CT and MRI aid in the diagnosis. Focal hypodense lesions and areas of enhancement or calcification may be appreciated in the orbit, paranasal sinuses or in our cases, the nasolacrimal duct of those affected. Focal bony destruction may occur.1,7 Beyond inspection, biopsies may be guided by imaging studies, and it has been recommended that hypodense lesions be particularly targeted as they often represent abscess.7 Repeat biopsies or multiple biopsies are sometimes required. As demonstrated in our second case, nasal endoscopy may give further evidence of an invasive fungal process. While this procedure can be a helpful adjunct, not all children can tolerate it at the bedside, even with sedation. The use of bedside nasal endoscopy is often at the discretion of the otolaryngologist. Treatment involves surgery and antifungal therapy. Antifungal options include amphotericin B, intraconazole, voriconazole, and lipid complex nystatin and echinocandins.3 Surgical treatment involves debridement either radical or conservative with the goal of clear surgical margins when possible. Aeration and drainage of the sinus with resection of involved tissue is commonly required.1 Radical versus conservative surgical excision remains a difficult decision as high mortality has been reported with central nervous system extension.1,3,7,8 Dhiwakar et al.7 advocate a conservative resection for anterior orbital processes while exenteration remains the treatment of choice for posterior disease. Our patients with anterior invasive sino-orbital fungal infections were treated with conservative debridement and antifungals. When the nasolacrimal system is affected, dacryocystorhinostomy may be necessary to repair secondary obstruction. In conclusion, these cases demonstrate that dacryocystitis may be the initial presentation of invasive fungal sinusitis in children. While not a common presenting sign, the clinician should maintain a heightened clinical suspicion of fungal sinusitis when immune-compromised pediatric patients present with dacryocystitis.
REFERENCES 1. Levin LA, Avery R, Shore JW, et al. The spectrum of orbital aspergillosis: a clinicopathological review. Surv Ophthalmol 1996;41:142–54.
Case Reports
2. Mauriello JA Jr, Yepez N, Mostafavi R, et al. Invasive rhinosinoorbital aspergillosis with precipitous visual loss. Can J Ophthalmol 1995;30:124–30. 3. Sivak-Callcott JA, Livesley N, Nugent RA, et al. Localised invasive sino-orbital aspergillosis: characteristic features. Br J Ophthalmol 2004;88:681–7. 4. Kim C, Kacker A, Chee RI, et al. Allergic fungal sinusitis causing nasolacrimal duct obstruction. Orbit 2013;32:143–5. 5. Petkar A, Rao L, Elizondo DR, et al. Allergic fungal sinusitis with massive intracranial extension presenting with tearing. Ophthal Plast Reconstr Surg 2011;27:e98–100. 6. Facer ML, Ponikau JU, Sherris DA. Eosinophilic fungal rhinosinusitis of the lacrimal sac. Laryngoscope 2003;113:210–4. 7. Dhiwakar M, Thakar A, Bahadur S. Invasive sino-orbital aspergillosis: surgical decisions and dilemmas. J Laryngol Otol 2003;117:280–5. 8. deShazo RD. Fungal sinusitis. Am J Med Sci 1998;316:39–45.
Granulicatella elegans Causing Periorbital Infection During Orthodontic Treatment Yong Joon Kim, M.D.*, Bo Mi Choi, M.D.†, and Kyung Seek Choi, M.D.* Abstract: Granulicatella elegans is a normal component of the oral flora and is an unusual causative agent of infective endocarditis. A case of G. elegans periorbital infection of the eyelid after dental treatment is reported. A healthy 35-yearold man presented with painful swelling of the left upper eyelid. He was empirically treated with oral amoxicillin for 1 week. He presented 3 months later with the same clinical features. G. elegans and Staphylococcus epidermidis were identified in bacterial cultures from wound aspirates. Probable relapse of periorbital infection was successfully treated with a 6-week course of oral amoxicillin. This is the first reported case of a non-bloodstream infection caused by G. elegans. Clinicians should be aware of G. elegans as an unusual causative agent of periorbital infection. Within the limitations of this case report, prolonged antibiotic therapy is recommended for a G. elegans periorbital infection to minimize the risk of relapse.
G
ranulicatella species are normal components of oral flora, and catalase-negative and oxidase-negative, facultatively anaerobic, Gram-positive cocci.1 Granulicatella is known as nutritionally variant streptococci (NVS) because of their requirement for pyridoxal or cysteine to be successfully isolated in the laboratory.2 G. adiacens and G. elegans have been isolated from various human samples. G. adiacens is a causative agent in infective endocarditis, brain abscess, septic arthritis, breast implant–associated infections, and ocular infections.3 In contrast, G. elegans has been isolated only in bloodstream infections (BSIs) including infective endocarditis. Herein, a rare case *Department of Ophthalmology, Soonchunhyang University College of Medicine, Soonchunhyang University Seoul Hospital, Seoul, Republic of Korea; and †Department of Internal Medicine, Bucheon St. Mary’s Hospital, The Catholic University of Korea, College of Medicine, Bucheon, Republic of Korea Accepted for publication June 4, 2014. Supported by the Soonchunhyang University Research Fund. The authors have no financial or conflicts of interest to disclose. Address correspondence and reprint requests to Kyung Seek Choi, M.D., Department of Ophthalmology, Soonchunhyang University College of Medicine, Soonchunhyang University Seoul Hospital, 59 Daesagwan-ro, Yongsan-gu, Seoul, Republic of Korea. E-mail: [email protected] DOI: 10.1097/IOP.0000000000000253
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
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Ophthal Plast Reconstr Surg, Vol. 32, No. 4, 2016
Case Reports
of a G. elegans periorbital infection after orthodontic treatment is presented. This is the first reported case of a non-BSI caused by G. elegans.
endocarditis.6 Dental procedures were thought to have contributed
CASE REPORT A healthy 35-year-old man presented with painful swelling of the left upper eyelid. He had undergone orthodontic treatment for several months. Initial visual acuity and intraocular pressure were within normal ranges. An inspection revealed a firm, tender, and erythematous lesion (Fig. A). Ocular motility was not limited in any direction, and no afferent pupillary defect was observed. Prominent conjunctival chemosis and injection were observed on biomicroscopy, but there was no inflammatory reaction in the anterior chamber or vitreous cavity. Because the patient refused further evaluation, oral amoxicillin/clavulanate (500 mg/8 h) was initiated empirically. The periorbital swelling and conjunctival chemosis improved prominently 1 week after treatment. Thereafter, the patient did not return for follow up. He presented 3 months later with the same clinical features (Fig. B). There was no evidence of systemic inflammation. Laboratory data showed a normal white blood count (8.3 × 109/L, neutrophils 65.5%) and high C-reactive protein (5.48 mg/L). CT revealed contrast-enhanced, swollen soft tissue in the left upper eyelid without intraorbital extension (Fig. C). Paranasal sinusitis was not detected by the CT. A conjunctival swab of the superior fornix for a bacterial culture was performed. Surgical drainage of the abscess and 2 times of wound culture were also performed with a skin incision. Because Gram staining showed a few Gram-positive cocci, empirical treatment was initiated with oral amoxicillin/clavulanate (500 mg/8 h). G. elegans and rare Staphylococcus epidermidis were identified in 3 bacterial cultures from the conjunctival swab and intraoperative aspirates using an automated microbiology system (VITEK 2, BioMerieux, Inc., Hazelwood, MO, U.S.A.) using growth-based technology. Both isolates were sensitive to penicillin, amoxicillin, ceftriaxone, and vancomycin. The swollen eyelid and conjunctival chemosis improved after 1 week of treatment, and the oral amoxicillin/clavulanate therapy was continued for another 5 weeks. No signs of infection were observed over the next 12 months.
DISCUSSION Granulicatella species are uncommon causative agents of infection. G. adiacens and G. elegans have been reported to cause BSI. Infective endocarditis caused by G. adiacens is more common than that caused by G. elegans.4 Strains of G. adiacens from infective endocarditis display fibronectin binding, unlike G. elegans.2,5 This difference between the 2 species may account for the different prevalence in infective endocarditis and other infections. Interestingly, only G. adiacens has been isolated from a non-BSI. No study has described a G. elegans–related infection other than BSI. In this case, G. elegans and S. epidermidis were isolated from both conjunctiva and an abscess during the second episode of infection. This coinfection distinguished our case from other reported Granulicatella infections. Satellitism around S. epidermidis is an important phenotypic characteristic of G. elegans.4 Therefore, the authors speculate that the coinfecting S. epidermidis triggered the vigorous growth of G. elegans in the eyelid. It is difficult to clarify the mode of infection in this case. He had no history of paranasal sinusitis or trauma. One possible explanation is that the isolated G. elegans and S. epidermidis originated from the oral cavity. Colonization of G. elegans and S. epidermidis has been observed in the human oral cavity, and transmission of these species has been suggested as a cause of infective
e82
Clinical features of the patient. Erythematous swelling of the left upper eyelid and conjunctiva were observed during the first (A) and second episodes (B). CT revealed diffuse thickening and enhancement of soft tissue in the left upper eyelid and conjunctiva (C).
to transmit the bacteria, which normally colonize the oral cavity. Because the authors did not perform a bacterial culture during the first episode, they cannot be certain that the first episode of infection was caused by the same agent. Nevertheless, the second episode was regarded as recurrent periorbital infection due to inappropriate antibiotic therapy, based on the similarity of clinical features and characteristics of G. elegans. The long interval between the 2 episodes could be explained by low virulence of species. It has been postulated that the difficulties treating infections of Granulicatella may be caused by their slow growth rate.7 Therefore, a longer course of antibiotic therapy might be required for an NVS infection to prevent relapse. Cargill et al.2 reported successful treatment of Granulicatella endocarditis with 6 weeks of antibiotics. Teo et al.3 reported a post-traumatic orbital abscess associated with G. adiacens. The infection was diagnosed 5 months after a penetrating injury and was treated with a 6-week course of oral levofloxacin. del Pozo et al.7 reported a probable relapse of a G. adiacens breast implant– associated infection after 5 days of levofloxacin treatment. The relapse was identified 2 months after treatment and completely resolved with 4 months of antibiotic therapy. In this case, the patient was treated with oral amoxicillin for only 7 days during the first episode. Although the isolated G. elegans was susceptible to amoxicillin, it was speculated that the duration of therapy may have been insufficient. A probable relapse of periorbital infection was successfully treated with a 6-week course of oral amoxicillin. In conclusion, clinicians should be aware of G. elegans as an unusual causative agent of periorbital infection. Within the limitations of this case report, prolonged duration antibiotic therapy was recommended for G. elegans periorbital infection to minimize the risk of relapse.
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
Ophthal Plast Reconstr Surg, Vol. 32, No. 4, 2016
REFERENCES 1. Collins MD, Lawson PA. The genus Abiotrophia (Kawamura et al.) is not monophyletic: proposal of Granulicatella gen. nov., Granulicatella adiacens comb. nov., Granulicatella elegans comb. nov. and Granulicatella balaenopterae comb. nov. Int J Syst Evol Microbiol 2000;50(pt 1):365–9. 2. Cargill JS, Scott KS, Gascoyne-Binzi D, et al. Granulicatella infection: diagnosis and management. J Med Microbiol 2012;61(pt 6):755–61. 3. Teo L, Looi A, Seah LL. An unusual causative agent for an orbital abscess: Granulicatella adiacens. Orbit 2011;30:162–4. 4. Christensen JJ, Facklam RR. Granulicatella and Abiotrophia species from human clinical specimens. J Clin Microbiol 2001;39:3520–3. 5. Okada Y, Kitada K, Takagaki M, et al. Endocardiac infectivity and binding to extracellular matrix proteins of oral Abiotrophia species. FEMS Immunol Med Microbiol 2000;27:257–61. 6. Ohara-Nemoto Y, Kishi K, Satho M, et al. Infective endocarditis caused by Granulicatella elegans originating in the oral cavity. J Clin Microbiol 2005;43:1405–7. 7. del Pozo JL, Garcia-Quetglas E, Hernaez S, et al. Granulicatella adiacens breast implant-associated infection. Diagn Microbiol Infect Dis 2008;61:58–60.
Localization and Retrieval of an Eyelid Metallic Foreign Body With an Oscillating Magnet and High-Resolution Ultrasonography Sylvia H. Yoo, M.D., Dan B. Rootman, M.D., Alice Goh, M.B.B.S., Aaron Savar, M.D., and Robert A. Goldberg, M.D. Abstract: A patient was found to have a metallic foreign body in the left anterior orbit on CT imaging, but the foreign body was not evident on clinical examination. On high-resolution ultrasonography, an object was identified in the left upper eyelid; however, the typical shadow with metallic foreign bodies was not seen. A high-power oscillating magnet was then applied to the eyelid, which revealed a subcutaneous metallic foreign body in the left upper eyelid. When used in conjunction, the high-resolution ultrasound and oscillating magnet successfully localized and facilitated retrieval of the metallic foreign body from the left upper eyelid.
Case Reports
time of injury. However, he continued to complain of periodic sharp pain in the medial left orbit. During a subsequent evaluation of long-term back pain, a CT scan was performed to investigate residual metallic objects used in preparation of MRI examination. The CT revealed a metallic foreign body in the superficial soft tissues of the medial left orbit (Fig. A). It was localized to the canalicular system, conjunctiva or eyelid. Despite careful examination by multiple ophthalmologists and oculoplastic surgeons, including lacrimal probing, the foreign body was not visualized or palpated. Further studies with A-scan ultrasound were unable to identify the foreign body, and B-scan ultrasonography by a retina specialist confirmed that the object was not localized to the sclera or intraocular space. The remainder of his ophthalmologic examination was normal, with the exception of a corneal scar in the OD from prior metallic foreign body removal. To investigate the periocular soft tissues further, static and dynamic high-resolution ultrasound (Logiq P6, GE Healthcare, Waukesha, WA, U.S.A.) was performed with a 15-MHz probe. A hyperechoic mass in the medial aspect of the left upper eyelid was identified (Fig. B). The mass moved dynamically with eyelid excursion (see Video 1, Supplemental Digital Content 1, available at: http://links.lww.com/IOP/ A97). An oscillating electromagnet (Scientronics Series 10K Eye Magnet; IMC Magnetics Corp, Jericho, NY) was then brought into the field of ultrasound imaging (Fig. C). The periodic application of an electromagnetic field caused the metallic body to oscillate in a regular pattern, confirming its identity (see Video 2, Supplemental Digital Content 2, available at: http://links.lww.com/IOP/A98). The position of the pulsating skin below the magnet was then marked (see Video 3, Supplemental Digital Content 3, available at: http:// links.lww.com/IOP/A99) and, after application of 2% lidocaine with 1:100,000 epinephrine, the skin was opened. By repeatedly applying the magnetic field, followed by sharp and blunt dissection, the metallic foreign body was identified and excised. (Fig. D; see Video 4, Supplemental Digital Content 4, http://links.lww.com/IOP/A100). The foreign body measured approximately 3 mm × 4 mm. The pathology report confirmed a firm black object, consistent with foreign body, with soft tissue showing foreign body reaction and brown pigment suggestive of hemosiderin. The patient reported improved pain symptoms and no complications since the procedure. The patient was subsequently able to undergo MRI that revealed pathology in his lumbosacral spine.
CASE REPORT This report conformed to the requirements of the United States Health Insurance Portability and Accountability Act. A 50-year-old man presented with periodic pain in the medial aspect of the left upper eyelid following trauma to OU in a work-related explosion 7 years before. The event had resulted in the embedding of multiple periocular metallic foreign bodies. The majority of these materials were removed manually at the
Jules Stein Eye Institute, UCLA, Los Angeles, California, U.S.A. Accepted for publication June 11, 2014. Presented at the European Society of Oculoplastic and Reconstructive Surgery on September 19–21, 2013 in Barcelona, Spain. The authors have no financial or conflicts of interest to disclose. Supplemental digital content is available for this article. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal’s website (www.op-rs.com.). Address correspondence and reprint requests to Sylvia H. Yoo, M.D., 100 Stein Plaza, Los Angeles, CA 90095. E-mail: [email protected] DOI: 10.1097/IOP.0000000000000257
DISCUSSION CT imaging of the orbit in this case demonstrated a superficial left periocular metallic foreign body many years after an explosive work-related trauma. Although it was clearly identifiable on imaging, multiple examinations of the eyelid, orbit, and globe were unable to localize the object. Such cases can be surgically frustrating, as wide dissection and surgical exploration can increase collateral tissue damage and lead to unwanted structural and/or functional consequences. For this reason, small metallic orbital foreign bodies are often not removed unless there is a compelling reason to do so, such as infection or functional deficit. In a chart review by Ho et al.,1 retained intraorbital metallic foreign bodies were overall found to be well tolerated, leading the authors to recommend conservative management. They did, however, discuss indications for removal including problematic location or particular metallic composition. Copper foreign bodies may produce a significant inflammatory response, requiring removal, while copper alloys may be better
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
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patients.1 Risk factors include total neutrophil count of less than 1,000/mm3, AIDS, defects in phagocytosis, hematologic malignancy, steroids, immunosuppressives, diabetes mellitus, prosthetic devices, trauma, environmental exposure (proximity to building demolition or restoration, yard work, compost heaps), and residence in endemic areas such as the Middle East especially Sudan, the Indian subcontinent and the southern United States.1 The 2 patients presented here were immunocompromised and received chemotherapy for hematologic malignancies. While lateral or retro-orbital pain is a common initial complaint of sino-orbital Aspergillus,3 acute dacryocystitis has only rarely been reported as the initial complaint. Most reported cases of lacrimal involvement have been caused by allergic fungal sinusitis and are present in immune-competent adults. Kim et al.4 recently reported a case of allergic fungal sinusitis presenting with epiphora. Their patient required multiple surgical procedures to control the infection and improve the nasolacrimal duct obstruction. Also, Petkar et al.5 reported a patient with epiphora as the only presenting sign of allergic fungal sinusitis. The patient had resolution of epiphora following sinus surgery and medical management. No lacrimal surgery was required. Finally, Facer et al.6 reported a case of bilateral nasolacrimal involvement from eosinophilic fungal sinusitis. The patient presented with a fluctuant mass just under the right medial canthus. Medical management, sinus surgery, and dacryocystorhinostomy were curative. Imaging techniques such as CT and MRI aid in the diagnosis. Focal hypodense lesions and areas of enhancement or calcification may be appreciated in the orbit, paranasal sinuses or in our cases, the nasolacrimal duct of those affected. Focal bony destruction may occur.1,7 Beyond inspection, biopsies may be guided by imaging studies, and it has been recommended that hypodense lesions be particularly targeted as they often represent abscess.7 Repeat biopsies or multiple biopsies are sometimes required. As demonstrated in our second case, nasal endoscopy may give further evidence of an invasive fungal process. While this procedure can be a helpful adjunct, not all children can tolerate it at the bedside, even with sedation. The use of bedside nasal endoscopy is often at the discretion of the otolaryngologist. Treatment involves surgery and antifungal therapy. Antifungal options include amphotericin B, intraconazole, voriconazole, and lipid complex nystatin and echinocandins.3 Surgical treatment involves debridement either radical or conservative with the goal of clear surgical margins when possible. Aeration and drainage of the sinus with resection of involved tissue is commonly required.1 Radical versus conservative surgical excision remains a difficult decision as high mortality has been reported with central nervous system extension.1,3,7,8 Dhiwakar et al.7 advocate a conservative resection for anterior orbital processes while exenteration remains the treatment of choice for posterior disease. Our patients with anterior invasive sino-orbital fungal infections were treated with conservative debridement and antifungals. When the nasolacrimal system is affected, dacryocystorhinostomy may be necessary to repair secondary obstruction. In conclusion, these cases demonstrate that dacryocystitis may be the initial presentation of invasive fungal sinusitis in children. While not a common presenting sign, the clinician should maintain a heightened clinical suspicion of fungal sinusitis when immune-compromised pediatric patients present with dacryocystitis.
REFERENCES 1. Levin LA, Avery R, Shore JW, et al. The spectrum of orbital aspergillosis: a clinicopathological review. Surv Ophthalmol 1996;41:142–54.
Case Reports
2. Mauriello JA Jr, Yepez N, Mostafavi R, et al. Invasive rhinosinoorbital aspergillosis with precipitous visual loss. Can J Ophthalmol 1995;30:124–30. 3. Sivak-Callcott JA, Livesley N, Nugent RA, et al. Localised invasive sino-orbital aspergillosis: characteristic features. Br J Ophthalmol 2004;88:681–7. 4. Kim C, Kacker A, Chee RI, et al. Allergic fungal sinusitis causing nasolacrimal duct obstruction. Orbit 2013;32:143–5. 5. Petkar A, Rao L, Elizondo DR, et al. Allergic fungal sinusitis with massive intracranial extension presenting with tearing. Ophthal Plast Reconstr Surg 2011;27:e98–100. 6. Facer ML, Ponikau JU, Sherris DA. Eosinophilic fungal rhinosinusitis of the lacrimal sac. Laryngoscope 2003;113:210–4. 7. Dhiwakar M, Thakar A, Bahadur S. Invasive sino-orbital aspergillosis: surgical decisions and dilemmas. J Laryngol Otol 2003;117:280–5. 8. deShazo RD. Fungal sinusitis. Am J Med Sci 1998;316:39–45.
Granulicatella elegans Causing Periorbital Infection During Orthodontic Treatment Yong Joon Kim, M.D.*, Bo Mi Choi, M.D.†, and Kyung Seek Choi, M.D.* Abstract: Granulicatella elegans is a normal component of the oral flora and is an unusual causative agent of infective endocarditis. A case of G. elegans periorbital infection of the eyelid after dental treatment is reported. A healthy 35-yearold man presented with painful swelling of the left upper eyelid. He was empirically treated with oral amoxicillin for 1 week. He presented 3 months later with the same clinical features. G. elegans and Staphylococcus epidermidis were identified in bacterial cultures from wound aspirates. Probable relapse of periorbital infection was successfully treated with a 6-week course of oral amoxicillin. This is the first reported case of a non-bloodstream infection caused by G. elegans. Clinicians should be aware of G. elegans as an unusual causative agent of periorbital infection. Within the limitations of this case report, prolonged antibiotic therapy is recommended for a G. elegans periorbital infection to minimize the risk of relapse.
G
ranulicatella species are normal components of oral flora, and catalase-negative and oxidase-negative, facultatively anaerobic, Gram-positive cocci.1 Granulicatella is known as nutritionally variant streptococci (NVS) because of their requirement for pyridoxal or cysteine to be successfully isolated in the laboratory.2 G. adiacens and G. elegans have been isolated from various human samples. G. adiacens is a causative agent in infective endocarditis, brain abscess, septic arthritis, breast implant–associated infections, and ocular infections.3 In contrast, G. elegans has been isolated only in bloodstream infections (BSIs) including infective endocarditis. Herein, a rare case *Department of Ophthalmology, Soonchunhyang University College of Medicine, Soonchunhyang University Seoul Hospital, Seoul, Republic of Korea; and †Department of Internal Medicine, Bucheon St. Mary’s Hospital, The Catholic University of Korea, College of Medicine, Bucheon, Republic of Korea Accepted for publication June 4, 2014. Supported by the Soonchunhyang University Research Fund. The authors have no financial or conflicts of interest to disclose. Address correspondence and reprint requests to Kyung Seek Choi, M.D., Department of Ophthalmology, Soonchunhyang University College of Medicine, Soonchunhyang University Seoul Hospital, 59 Daesagwan-ro, Yongsan-gu, Seoul, Republic of Korea. E-mail: [email protected] DOI: 10.1097/IOP.0000000000000253
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
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Ophthal Plast Reconstr Surg, Vol. 32, No. 4, 2016
Case Reports
of a G. elegans periorbital infection after orthodontic treatment is presented. This is the first reported case of a non-BSI caused by G. elegans.
endocarditis.6 Dental procedures were thought to have contributed
CASE REPORT A healthy 35-year-old man presented with painful swelling of the left upper eyelid. He had undergone orthodontic treatment for several months. Initial visual acuity and intraocular pressure were within normal ranges. An inspection revealed a firm, tender, and erythematous lesion (Fig. A). Ocular motility was not limited in any direction, and no afferent pupillary defect was observed. Prominent conjunctival chemosis and injection were observed on biomicroscopy, but there was no inflammatory reaction in the anterior chamber or vitreous cavity. Because the patient refused further evaluation, oral amoxicillin/clavulanate (500 mg/8 h) was initiated empirically. The periorbital swelling and conjunctival chemosis improved prominently 1 week after treatment. Thereafter, the patient did not return for follow up. He presented 3 months later with the same clinical features (Fig. B). There was no evidence of systemic inflammation. Laboratory data showed a normal white blood count (8.3 × 109/L, neutrophils 65.5%) and high C-reactive protein (5.48 mg/L). CT revealed contrast-enhanced, swollen soft tissue in the left upper eyelid without intraorbital extension (Fig. C). Paranasal sinusitis was not detected by the CT. A conjunctival swab of the superior fornix for a bacterial culture was performed. Surgical drainage of the abscess and 2 times of wound culture were also performed with a skin incision. Because Gram staining showed a few Gram-positive cocci, empirical treatment was initiated with oral amoxicillin/clavulanate (500 mg/8 h). G. elegans and rare Staphylococcus epidermidis were identified in 3 bacterial cultures from the conjunctival swab and intraoperative aspirates using an automated microbiology system (VITEK 2, BioMerieux, Inc., Hazelwood, MO, U.S.A.) using growth-based technology. Both isolates were sensitive to penicillin, amoxicillin, ceftriaxone, and vancomycin. The swollen eyelid and conjunctival chemosis improved after 1 week of treatment, and the oral amoxicillin/clavulanate therapy was continued for another 5 weeks. No signs of infection were observed over the next 12 months.
DISCUSSION Granulicatella species are uncommon causative agents of infection. G. adiacens and G. elegans have been reported to cause BSI. Infective endocarditis caused by G. adiacens is more common than that caused by G. elegans.4 Strains of G. adiacens from infective endocarditis display fibronectin binding, unlike G. elegans.2,5 This difference between the 2 species may account for the different prevalence in infective endocarditis and other infections. Interestingly, only G. adiacens has been isolated from a non-BSI. No study has described a G. elegans–related infection other than BSI. In this case, G. elegans and S. epidermidis were isolated from both conjunctiva and an abscess during the second episode of infection. This coinfection distinguished our case from other reported Granulicatella infections. Satellitism around S. epidermidis is an important phenotypic characteristic of G. elegans.4 Therefore, the authors speculate that the coinfecting S. epidermidis triggered the vigorous growth of G. elegans in the eyelid. It is difficult to clarify the mode of infection in this case. He had no history of paranasal sinusitis or trauma. One possible explanation is that the isolated G. elegans and S. epidermidis originated from the oral cavity. Colonization of G. elegans and S. epidermidis has been observed in the human oral cavity, and transmission of these species has been suggested as a cause of infective
e82
Clinical features of the patient. Erythematous swelling of the left upper eyelid and conjunctiva were observed during the first (A) and second episodes (B). CT revealed diffuse thickening and enhancement of soft tissue in the left upper eyelid and conjunctiva (C).
to transmit the bacteria, which normally colonize the oral cavity. Because the authors did not perform a bacterial culture during the first episode, they cannot be certain that the first episode of infection was caused by the same agent. Nevertheless, the second episode was regarded as recurrent periorbital infection due to inappropriate antibiotic therapy, based on the similarity of clinical features and characteristics of G. elegans. The long interval between the 2 episodes could be explained by low virulence of species. It has been postulated that the difficulties treating infections of Granulicatella may be caused by their slow growth rate.7 Therefore, a longer course of antibiotic therapy might be required for an NVS infection to prevent relapse. Cargill et al.2 reported successful treatment of Granulicatella endocarditis with 6 weeks of antibiotics. Teo et al.3 reported a post-traumatic orbital abscess associated with G. adiacens. The infection was diagnosed 5 months after a penetrating injury and was treated with a 6-week course of oral levofloxacin. del Pozo et al.7 reported a probable relapse of a G. adiacens breast implant– associated infection after 5 days of levofloxacin treatment. The relapse was identified 2 months after treatment and completely resolved with 4 months of antibiotic therapy. In this case, the patient was treated with oral amoxicillin for only 7 days during the first episode. Although the isolated G. elegans was susceptible to amoxicillin, it was speculated that the duration of therapy may have been insufficient. A probable relapse of periorbital infection was successfully treated with a 6-week course of oral amoxicillin. In conclusion, clinicians should be aware of G. elegans as an unusual causative agent of periorbital infection. Within the limitations of this case report, prolonged duration antibiotic therapy was recommended for G. elegans periorbital infection to minimize the risk of relapse.
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
Ophthal Plast Reconstr Surg, Vol. 32, No. 4, 2016
REFERENCES 1. Collins MD, Lawson PA. The genus Abiotrophia (Kawamura et al.) is not monophyletic: proposal of Granulicatella gen. nov., Granulicatella adiacens comb. nov., Granulicatella elegans comb. nov. and Granulicatella balaenopterae comb. nov. Int J Syst Evol Microbiol 2000;50(pt 1):365–9. 2. Cargill JS, Scott KS, Gascoyne-Binzi D, et al. Granulicatella infection: diagnosis and management. J Med Microbiol 2012;61(pt 6):755–61. 3. Teo L, Looi A, Seah LL. An unusual causative agent for an orbital abscess: Granulicatella adiacens. Orbit 2011;30:162–4. 4. Christensen JJ, Facklam RR. Granulicatella and Abiotrophia species from human clinical specimens. J Clin Microbiol 2001;39:3520–3. 5. Okada Y, Kitada K, Takagaki M, et al. Endocardiac infectivity and binding to extracellular matrix proteins of oral Abiotrophia species. FEMS Immunol Med Microbiol 2000;27:257–61. 6. Ohara-Nemoto Y, Kishi K, Satho M, et al. Infective endocarditis caused by Granulicatella elegans originating in the oral cavity. J Clin Microbiol 2005;43:1405–7. 7. del Pozo JL, Garcia-Quetglas E, Hernaez S, et al. Granulicatella adiacens breast implant-associated infection. Diagn Microbiol Infect Dis 2008;61:58–60.
Localization and Retrieval of an Eyelid Metallic Foreign Body With an Oscillating Magnet and High-Resolution Ultrasonography Sylvia H. Yoo, M.D., Dan B. Rootman, M.D., Alice Goh, M.B.B.S., Aaron Savar, M.D., and Robert A. Goldberg, M.D. Abstract: A patient was found to have a metallic foreign body in the left anterior orbit on CT imaging, but the foreign body was not evident on clinical examination. On high-resolution ultrasonography, an object was identified in the left upper eyelid; however, the typical shadow with metallic foreign bodies was not seen. A high-power oscillating magnet was then applied to the eyelid, which revealed a subcutaneous metallic foreign body in the left upper eyelid. When used in conjunction, the high-resolution ultrasound and oscillating magnet successfully localized and facilitated retrieval of the metallic foreign body from the left upper eyelid.
Case Reports
time of injury. However, he continued to complain of periodic sharp pain in the medial left orbit. During a subsequent evaluation of long-term back pain, a CT scan was performed to investigate residual metallic objects used in preparation of MRI examination. The CT revealed a metallic foreign body in the superficial soft tissues of the medial left orbit (Fig. A). It was localized to the canalicular system, conjunctiva or eyelid. Despite careful examination by multiple ophthalmologists and oculoplastic surgeons, including lacrimal probing, the foreign body was not visualized or palpated. Further studies with A-scan ultrasound were unable to identify the foreign body, and B-scan ultrasonography by a retina specialist confirmed that the object was not localized to the sclera or intraocular space. The remainder of his ophthalmologic examination was normal, with the exception of a corneal scar in the OD from prior metallic foreign body removal. To investigate the periocular soft tissues further, static and dynamic high-resolution ultrasound (Logiq P6, GE Healthcare, Waukesha, WA, U.S.A.) was performed with a 15-MHz probe. A hyperechoic mass in the medial aspect of the left upper eyelid was identified (Fig. B). The mass moved dynamically with eyelid excursion (see Video 1, Supplemental Digital Content 1, available at: http://links.lww.com/IOP/ A97). An oscillating electromagnet (Scientronics Series 10K Eye Magnet; IMC Magnetics Corp, Jericho, NY) was then brought into the field of ultrasound imaging (Fig. C). The periodic application of an electromagnetic field caused the metallic body to oscillate in a regular pattern, confirming its identity (see Video 2, Supplemental Digital Content 2, available at: http://links.lww.com/IOP/A98). The position of the pulsating skin below the magnet was then marked (see Video 3, Supplemental Digital Content 3, available at: http:// links.lww.com/IOP/A99) and, after application of 2% lidocaine with 1:100,000 epinephrine, the skin was opened. By repeatedly applying the magnetic field, followed by sharp and blunt dissection, the metallic foreign body was identified and excised. (Fig. D; see Video 4, Supplemental Digital Content 4, http://links.lww.com/IOP/A100). The foreign body measured approximately 3 mm × 4 mm. The pathology report confirmed a firm black object, consistent with foreign body, with soft tissue showing foreign body reaction and brown pigment suggestive of hemosiderin. The patient reported improved pain symptoms and no complications since the procedure. The patient was subsequently able to undergo MRI that revealed pathology in his lumbosacral spine.
CASE REPORT This report conformed to the requirements of the United States Health Insurance Portability and Accountability Act. A 50-year-old man presented with periodic pain in the medial aspect of the left upper eyelid following trauma to OU in a work-related explosion 7 years before. The event had resulted in the embedding of multiple periocular metallic foreign bodies. The majority of these materials were removed manually at the
Jules Stein Eye Institute, UCLA, Los Angeles, California, U.S.A. Accepted for publication June 11, 2014. Presented at the European Society of Oculoplastic and Reconstructive Surgery on September 19–21, 2013 in Barcelona, Spain. The authors have no financial or conflicts of interest to disclose. Supplemental digital content is available for this article. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal’s website (www.op-rs.com.). Address correspondence and reprint requests to Sylvia H. Yoo, M.D., 100 Stein Plaza, Los Angeles, CA 90095. E-mail: [email protected] DOI: 10.1097/IOP.0000000000000257
DISCUSSION CT imaging of the orbit in this case demonstrated a superficial left periocular metallic foreign body many years after an explosive work-related trauma. Although it was clearly identifiable on imaging, multiple examinations of the eyelid, orbit, and globe were unable to localize the object. Such cases can be surgically frustrating, as wide dissection and surgical exploration can increase collateral tissue damage and lead to unwanted structural and/or functional consequences. For this reason, small metallic orbital foreign bodies are often not removed unless there is a compelling reason to do so, such as infection or functional deficit. In a chart review by Ho et al.,1 retained intraorbital metallic foreign bodies were overall found to be well tolerated, leading the authors to recommend conservative management. They did, however, discuss indications for removal including problematic location or particular metallic composition. Copper foreign bodies may produce a significant inflammatory response, requiring removal, while copper alloys may be better
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