Ophthal Plast Reconstr Surg, Vol. 30, No. 2, 2014
(Fig. E and F) and she was pleased with the reduced exposure and epiphora.
COMMENT Generalized eruptive keratoacanthomas involve the skin and mucous membranes.1 They are characterized by multiple small umbilicated keratinous papules histologically resembling keratoacanthoma.1 Ferguson-Smith described the most common variant of multiple keratoacanthomas, characterized by early onset of tens to hundreds of medium to large-sized lesions that usually leave deep scars.2 FergusonSmith type may or may not have familial inheritance.2 One autosomal familial subtype named multiple self-healing squamous epitheliomata has been described in people with Scottish descent.2 Grzybowski variant is rare and characterized by nonfamilial, adult-onset type of disease, with smaller lesions involving the skin of the face, eyelids, palms, and soles.2 Typically these lesions are pruritic and painful, and heal forming deep scars.2 Rare case reports exist in which Ferguson-Smith type and Grzybowski-type keratoacanthomas have existed in the same patient, so-called Witten–Zak type keratoacanthomas.2 Further multiple keratoacanthomas may be associated with cancers of meibomian gland and colon in Muir-Torre syndrome or of the genitourinary tract in keratoacanthoma visceral malignancy syndrome.2 Therapies reported to have some benefit for multiple keratoacanthomas include systemic chemotherapy, intralesional agents, surgery, and radiation.2 Synthetic retinoids like isotretinoin and etretinate have been beneficial in Ferguson-Smith type but are usually ineffective in the more generalized Grzybowski type.2 Immune modulation by agents like methotrexate and cyclophosphamide have been reported to alleviate symptoms of pruritus, involute existing keratoacanthomas and prevent appearance of new lesions.1 Mittelveifhaus3 and Consigli et al.4 described challenges from progressive cicatricial ectropions in a case of disseminated eruptive keratoacanthomas. Mittelveifhaus3 emphasized that multiple reoperations using oversized skin grafts with traction sutures and pressure patching were needed to correct the recurrent eyelid ectropion. He also detected bacterial contamination of the tear lake and suggested this might contribute to recurrent corneal infections. Our patient likewise had significant cosmetic deformity and early corneal damage from her upper and lower eyelid cicatricial ectropions; her generalized distribution of lesions limited the donor site to the dorsal foot, which may be a good source but requires significant undermining to close. As yet, her symptoms are well controlled, but if recurrences occur, further grafting may be required and suitable donor sites may be limited.
Case Reports
Subcutaneous and Periorbital Emphysema Following Dental Procedure David Fleischman, M.D., M,S., Richard M. Davis, M.D., and Lyndon B. Lee, M.D. Abstract: Subcutaneous emphysema following a dental procedure is a relatively uncommon phenomenon that may produce a dramatic clinical presentation. The advent of highpowered dental equipment that uses directed and highly pressurized air has increased the risk of this complication, but few cases have been described in the ophthalmic literature. The authors present a case of subcutaneous periorbital emphysema in a young woman following a dental procedure. Aside from a temporary ptosis, there were no other ophthalmic complications. An attempt to decompress the air produced minimal relief. The condition resolved spontaneously within 1 week.
T
he introduction of air into the subcutaneous tissues in the periorbital region is a recognized phenomenon that can occur after trauma, infection with gas-forming organisms, following head, neck, and sinus surgery, or as a factitious disorder. Isolated periorbital emphysema has also been reported as an exceedingly rare complication of dental procedures, with only 4 cases described in the literature.1–4 As an isolated condition, periorbital subcutaneous emphysema is benign and selflimited. However, if air gains access to the orbit, significant and catastrophic visual complications including blindness from congestive orbitopathy may develop.5,6 Of the few cases of dental procedure–related periorbital emphysema, only 1 has been described in the ophthalmic literature.4 Given the dramatic ophthalmic presentation and the possibility of being confused with more serious cases of orbital emphysema, the authors herein describe a case of subcutaneous periorbital and orbital emphysema following a dental procedure, with the intention of raising awareness about this procedural complication. The case presented is in compliance with the United States Health Insurance Portability and Accountability Act.
CASE REPORT A healthy 15-year-old black girl with no medical problems presented to our ophthalmology clinic with acute periorbital swelling that occurred immediately following an otherwise-uncomplicated extraction of the upper left third molar under general anesthesia. She was initially evaluated in a local emergency department where CT imaging of the head with contrast revealed subcutaneous air tracking along facial planes from the left maxilla to the left orbit (Figs. 1 and 2). She was diagnosed with subcutaneous emphysema, discharged on
REFERENCES 1. Oakley A, Ng S. Grzybowski’s generalized eruptive keratoacanthoma: remission with cyclophosphamide. Australas J Dermatol 2005;46:118–23. 2. Feldman RJ, Maize JC. Multiple keratoacanthomas in a young woman: report of a case emphasizing medical management and a review of the spectrum of multiple keratoacanthomas. Int J Dermatol 2007;46:77–9. 3. Mittelveifhaus. Cicatricial ectropion in progressive skin diseases. Orbit 2001;20:91–9. 4. Consigli JE, González ME, Morsino R, et al. Generalized eruptive keratoacanthoma (Grzybowski variant). Br J Dermatol 2000;142:800–3.
Department of Ophthalmology, University of North Carolina Hospitals, Chapel Hill, North Carolina, U.S.A. Accepted for publication April 1, 2013. The authors have no financial or conflicts of interest to disclose. Supported by the Department of Ophthalmology, University of North Carolina Chapel Hill. University of North Carolina is a recipient of an unrestricted grant from the Research to Prevent Blindness, Inc., New York City, New York. Address correspondence and reprint requests to Lyndon B. Lee, m.d.,Department of Ophthalmology, University of North Carolina Hospitals, 5151 Bioinformatics Bldg No. 7040, Chapel Hill, North Carolina 27599. E-mail: [email protected] DOI: 10.1097/IOP.0b013e318295f982
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
e43
Ophthal Plast Reconstr Surg, Vol. 30, No. 2, 2014
Case Reports
unremarkable without conjunctival chemosis or edema. Dilated fundus examination in OU was normal. Given the appearance of significant confluent air from CT imaging, an attempt was made to decompress the eyelid by inserting a 30-gauge needle on a syringe into the subcutaneous space to reduce the blepharoptosis. A small amount of air was released, but the tissue remained swollen with persistent crepitus. The patient was discharged from the clinic with prophylactic oral amoxicillin and instructions to return in 1 week. Follow-up examination on day 7 showed nearly complete resolution of the ptosis and eyelid emphysema though trace amounts of subcutaneous crepitus remained (Fig. 4). The antibiotic therapy was continued to complete a 10-day course, and the patient recovered fully.
DISCUSSION
FIG. 1. Coronal CT image reveals left periorbital and superior orbital emphysema, as well as soft tissue emphysema along the left maxilla. CT, computed tomography.
oral antibiotics, and referred to the University of North Carolina (UNC) Ophthalmology Clinic. On presentation, the patient’s visual acuity was 20/25 in the (OD) and 20/20 in the (OS). Pupils were equal, round, and reactive to light with no afferent pupillary defect. Extraocular motility was full in OU. Testing of cranial nerves III, IV, V, VI, and VII was unremarkable. Intraocular pressure measured 20 mm·Hg OD and 15 mm·Hg OS. On examination of the periorbital adnexa, the left upper eyelid was ptotic and swollen without warmth or erythema (Fig. 3). Palpation of the periorbital tissue revealed crepitus in the left upper eyelid without tenderness. The swelling and crepitus was limited to the upper eyelid. Hertel measurements with a base of 103 mm were 20 mm OD and 24 mm OS. Anterior segment examination was
FIG 2. Axial CT image remarkable for significant left eyelid and superior orbital emphysema. CT, computed tomography.
e44
In this case, subcutaneous emphysema from a dental procedure was a benign occurrence. A previous report had described, in a circumstance leading to orbital and periorbital emphysema, vision loss from a compressive neuropathy.6 Other reports of subcutaneous emphysema from dental procedures have been published in dental, oral, and maxillofacial literature, and once in the ophthalmic literature.1–4 Subcutaneous emphysema as a complication of dental surgery was first reported in 1900 following a tooth extraction and has since been described in association with root canal treatment, crown placement, and periodontal surgery.7–12 It is postulated that the high-speed, air-driven instruments used in modern dental treatments combined with a disruption of the intraoral barrier introduces pressurized air into the subcutaneous tissues and facilitates the tracking of air along the facial planes. The roots of the first, second, and third molars communicate directly with the sublingual and submandibular spaces. Furthermore, the sublingual space is in direct communication with the pterygomandibular, parapharyngeal, and retropharyngeal spaces.2 Subcutaneous air following these pathways may result in local swelling in the neck, mandible, or cheek. However, isolated periorbital involvement remains a relatively rare occurrence. In the case of upper third molar extraction, air conceivably tracks along the pterygomaxillary region to enter the periorbital tissue plane. In the patient reported here, the emphysema was limited to the subcutaneous tissues of the upper eyelid. The orbit remained uninvolved as an intact orbital septum prevented subcutaneous air from entering the orbital space. CT imaging confirmed the extent of spread. Subcutaneous emphysema may present during the dental procedure or within hours of its completion. Other important conditions to consider include allergy, angioedema, hemorrhage, and infection. More serious complications can occur when air enters the parapharyngeal and retropharyngeal spaces resulting in airway compromise through pneumomediastinum or pneumothorax. There is no consensus on the appropriate management of periorbital subcutaneous emphysema in a patient who does not appear to have vision-threatening complications.5,13 However, close observation for signs of airway involvement is warranted. Even if there are no immediate signs of compromised vision or a threatened airway, the patient should be observed until the clinician is satisfied that the path of the air has been fully demarcated and is no longer advancing.14 Imaging, while recommended, may not be necessary in the absence of worrisome clinical findings. Once a stable, nonprogressive exam has been documented, the patient can be successfully managed on an outpatient basis with close follow up. In contrast, patients with orbital signs and visual compromise have had air enter the
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
Ophthal Plast Reconstr Surg, Vol. 30, No. 2, 2014
FIG 3. Photograph of the patient reveals a proptotic left orbit. Povidone-iodine solution is present on the eyelid prior to needle decompression.
FIG. 4. One-week follow up. Near resolution of orbital and periorbital subcutaneous emphysema.
orbit and may require acute management. Imaging is warranted in these cases. If significant changes (e.g., proptosis, loss of vision, increased intraocular or intraorbital pressure) have occurred, the orbit must be decompressed. Direct decompression of the air mass with a needle-coupled syringe and indirect treatment with lateral canthotomy and cantholysis have both been used successfully.15,16 Needle decompression is not as useful in periorbital emphysema, given the diffuse spread of air in the subcutaneous tissues. Prophylactic systemic antibiotic coverage tailored to the naso-oral flora is recommended in either situation. Uyanik and colleagues3 and Kim et al.17 suggest that these complications could be prevented by using a rubber dam, using remote exhaust or electrical motor-driven handpieces; avoiding the use of the compressed air syringe during irrigation; and avoiding the use of hydrogen peroxide while irrigating canals. Romeo et al.18 suggest that during third molar surgery, mucoperiosteal flap elevation, important for protecting soft tissues from cutting instruments, must be firmly, but gently retracted when using high-speed air turbine handpieces. Minimizing their use may also help prevent the aforementioned complications.19 Clinicians should be aware of subcutaneous periorbital emphysema as a complication of dental procedures and be able to distinguish this benign entity from the more dangerous orbital emphysema.
REFERENCES 1. Turnball A. A remarkable coincidence in dental surgery. Br Med J. 1900, 102:1131. 2. Parkar A, Medhurst C, Irbash M, et al. Periorbital oedema and surgical emphysema, an unusual complication of a dental procedure: a case report. Cases J 2009;2:8108. 3. Uyanık LO, Aydın M, Buhara O, et al. Periorbital emphysema during dental treatment: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011;112:e94–6. 4. Alexander CM, Owens TF. Periorbital emphysema related to oral surgery. Am J Ophthalmol 1968;66:1166–7. 5. Singh M, Phua VM, Sundar G. Sight-threatening orbital emphysema treated with needle decompression. Clin Experiment Ophthalmol 2007;35:386–7.
Case Reports
6. Buckley MJ, Turvey TA, Schumann SP, et al. Orbital emphysema causing vision loss after a dental extraction. J Am Dent Assoc 1990;120:421–2, 424; discussion 423. 7. Davies DE. Pneumomediastinum after dental surgery. Anaesth Intensive Care 2001;29:638–41. 8. Yang SC, Chiu TH, Lin TJ, et al. Subcutaneous emphysema and pneumomediastinum secondary to dental extraction: a case report and literature review. Kaohsiung J Med Sci 2006;22:641–5. 9. Steelman RJ, Johannes PW. Subcutaneous emphysema during restorative dentistry. Int J Paediatr Dent 2007;17:228–9. 10. Sujeet K, Shankar S. Images in clinical medicine. Prevertebral emphysema after a dental procedure. N Engl J Med 2007; 356:173. 11. Zemann W, Feichtinger M, Kärcher H. Cervicofacial and mediastinal emphysema after crown preparation: a rare complication. Int J Prosthodont 2007;20:143–4. 12. Imai T, Michizawa M, Arimoto E, et al. Cervicofacial subcutaneous emphysema and pneumomediastinum after intraoral laser irradiation. J Oral Maxillofac Surg 2009;67:428–30. 13. Kung JC, Chuang FH, Hsu KJ, et al. Extensive subcutaneous emphysema after extraction of a mandibular third molar: a case report. Kaohsiung J Med Sci 2009;25:562–6. 14. Bastion ML, Wong YC. A case of sneezing-related orbital emphysema treated by aspiration-decompression in the office. Ophthal Plast Reconstr Surg 2006;22:500–1. 15. Fleishman JA, Beck RW, Hoffman RO. Orbital emphysema as an ophthalmologic emergency. Ophthalmology 1984;91:1389–91. 16. Hunts JH, Patrinely JR, Holds JB, et al. Orbital emphysema. Staging and acute management. Ophthalmology 1994;101:960–6. 17. Kim Y, Kim MR, Kim SJ. Iatrogenic pneumomediastinum with extensive subcutaneous emphysema after endodontic treatment: report of 2 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2010;109:e114–9. 18. Romeo U, Galanakis A, Lerario F, et al. Subcutaneous emphysema during third molar surgery: a case report. Braz Dent J 2011;22:83–6. 19. Sekine J, Irie A, Dotsu H, et al. Bilateral pneumothorax with extensive subcutaneous emphysema manifested during third molar surgery. A case report. Int J Oral Maxillofac Surg 2000;29:355–7.
Mucosa-Associated Lymphoid Tissue Lymphoma Masquerading as Herniated Orbital Fat Cindy S. Hwang, M.D.*, Pedro Diaz-Marchan, M.D.†, and Douglas P. Marx, M.D.* Abstract: Lymphomas are the most common primary orbital malignancies in adults. The authors present a 62-year-old Hispanic woman with a 2-year history of slowly enlarging bilateral lower eyelid masses that the patient described as “bags.” On palpation, firm, mobile, nontender masses with associated tear trough deformities were noted. Biopsy of the left lower eyelid mass was consistent with a mucosaassociated lymphoid tissue lymphoma. Herniated orbital fat is an extremely common finding in the aging population and is often associated with a prominent tear trough. The patient with orbital lymphoma appeared to have herniated orbital fat with associated tear trough deformities. Lymphoma
Departments of *Ophthalmology and †Radiology, Baylor College of Medicine, Houston, Texas, U.S.A. Accepted for publication March 4, 2013. The authors have no financial or conflicts of interest to disclose. Address correspondence and reprint requests to Douglas P. Marx, M.D., 1977 Butler Boulevard, Baylor College of Medicine Alkek Eye Center, Houston, TX 77030. E-mail: [email protected] DOI: 10.1097/IOP.0b013e31829aedf3
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
e45
(Fig. E and F) and she was pleased with the reduced exposure and epiphora.
COMMENT Generalized eruptive keratoacanthomas involve the skin and mucous membranes.1 They are characterized by multiple small umbilicated keratinous papules histologically resembling keratoacanthoma.1 Ferguson-Smith described the most common variant of multiple keratoacanthomas, characterized by early onset of tens to hundreds of medium to large-sized lesions that usually leave deep scars.2 FergusonSmith type may or may not have familial inheritance.2 One autosomal familial subtype named multiple self-healing squamous epitheliomata has been described in people with Scottish descent.2 Grzybowski variant is rare and characterized by nonfamilial, adult-onset type of disease, with smaller lesions involving the skin of the face, eyelids, palms, and soles.2 Typically these lesions are pruritic and painful, and heal forming deep scars.2 Rare case reports exist in which Ferguson-Smith type and Grzybowski-type keratoacanthomas have existed in the same patient, so-called Witten–Zak type keratoacanthomas.2 Further multiple keratoacanthomas may be associated with cancers of meibomian gland and colon in Muir-Torre syndrome or of the genitourinary tract in keratoacanthoma visceral malignancy syndrome.2 Therapies reported to have some benefit for multiple keratoacanthomas include systemic chemotherapy, intralesional agents, surgery, and radiation.2 Synthetic retinoids like isotretinoin and etretinate have been beneficial in Ferguson-Smith type but are usually ineffective in the more generalized Grzybowski type.2 Immune modulation by agents like methotrexate and cyclophosphamide have been reported to alleviate symptoms of pruritus, involute existing keratoacanthomas and prevent appearance of new lesions.1 Mittelveifhaus3 and Consigli et al.4 described challenges from progressive cicatricial ectropions in a case of disseminated eruptive keratoacanthomas. Mittelveifhaus3 emphasized that multiple reoperations using oversized skin grafts with traction sutures and pressure patching were needed to correct the recurrent eyelid ectropion. He also detected bacterial contamination of the tear lake and suggested this might contribute to recurrent corneal infections. Our patient likewise had significant cosmetic deformity and early corneal damage from her upper and lower eyelid cicatricial ectropions; her generalized distribution of lesions limited the donor site to the dorsal foot, which may be a good source but requires significant undermining to close. As yet, her symptoms are well controlled, but if recurrences occur, further grafting may be required and suitable donor sites may be limited.
Case Reports
Subcutaneous and Periorbital Emphysema Following Dental Procedure David Fleischman, M.D., M,S., Richard M. Davis, M.D., and Lyndon B. Lee, M.D. Abstract: Subcutaneous emphysema following a dental procedure is a relatively uncommon phenomenon that may produce a dramatic clinical presentation. The advent of highpowered dental equipment that uses directed and highly pressurized air has increased the risk of this complication, but few cases have been described in the ophthalmic literature. The authors present a case of subcutaneous periorbital emphysema in a young woman following a dental procedure. Aside from a temporary ptosis, there were no other ophthalmic complications. An attempt to decompress the air produced minimal relief. The condition resolved spontaneously within 1 week.
T
he introduction of air into the subcutaneous tissues in the periorbital region is a recognized phenomenon that can occur after trauma, infection with gas-forming organisms, following head, neck, and sinus surgery, or as a factitious disorder. Isolated periorbital emphysema has also been reported as an exceedingly rare complication of dental procedures, with only 4 cases described in the literature.1–4 As an isolated condition, periorbital subcutaneous emphysema is benign and selflimited. However, if air gains access to the orbit, significant and catastrophic visual complications including blindness from congestive orbitopathy may develop.5,6 Of the few cases of dental procedure–related periorbital emphysema, only 1 has been described in the ophthalmic literature.4 Given the dramatic ophthalmic presentation and the possibility of being confused with more serious cases of orbital emphysema, the authors herein describe a case of subcutaneous periorbital and orbital emphysema following a dental procedure, with the intention of raising awareness about this procedural complication. The case presented is in compliance with the United States Health Insurance Portability and Accountability Act.
CASE REPORT A healthy 15-year-old black girl with no medical problems presented to our ophthalmology clinic with acute periorbital swelling that occurred immediately following an otherwise-uncomplicated extraction of the upper left third molar under general anesthesia. She was initially evaluated in a local emergency department where CT imaging of the head with contrast revealed subcutaneous air tracking along facial planes from the left maxilla to the left orbit (Figs. 1 and 2). She was diagnosed with subcutaneous emphysema, discharged on
REFERENCES 1. Oakley A, Ng S. Grzybowski’s generalized eruptive keratoacanthoma: remission with cyclophosphamide. Australas J Dermatol 2005;46:118–23. 2. Feldman RJ, Maize JC. Multiple keratoacanthomas in a young woman: report of a case emphasizing medical management and a review of the spectrum of multiple keratoacanthomas. Int J Dermatol 2007;46:77–9. 3. Mittelveifhaus. Cicatricial ectropion in progressive skin diseases. Orbit 2001;20:91–9. 4. Consigli JE, González ME, Morsino R, et al. Generalized eruptive keratoacanthoma (Grzybowski variant). Br J Dermatol 2000;142:800–3.
Department of Ophthalmology, University of North Carolina Hospitals, Chapel Hill, North Carolina, U.S.A. Accepted for publication April 1, 2013. The authors have no financial or conflicts of interest to disclose. Supported by the Department of Ophthalmology, University of North Carolina Chapel Hill. University of North Carolina is a recipient of an unrestricted grant from the Research to Prevent Blindness, Inc., New York City, New York. Address correspondence and reprint requests to Lyndon B. Lee, m.d.,Department of Ophthalmology, University of North Carolina Hospitals, 5151 Bioinformatics Bldg No. 7040, Chapel Hill, North Carolina 27599. E-mail: [email protected] DOI: 10.1097/IOP.0b013e318295f982
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
e43
Ophthal Plast Reconstr Surg, Vol. 30, No. 2, 2014
Case Reports
unremarkable without conjunctival chemosis or edema. Dilated fundus examination in OU was normal. Given the appearance of significant confluent air from CT imaging, an attempt was made to decompress the eyelid by inserting a 30-gauge needle on a syringe into the subcutaneous space to reduce the blepharoptosis. A small amount of air was released, but the tissue remained swollen with persistent crepitus. The patient was discharged from the clinic with prophylactic oral amoxicillin and instructions to return in 1 week. Follow-up examination on day 7 showed nearly complete resolution of the ptosis and eyelid emphysema though trace amounts of subcutaneous crepitus remained (Fig. 4). The antibiotic therapy was continued to complete a 10-day course, and the patient recovered fully.
DISCUSSION
FIG. 1. Coronal CT image reveals left periorbital and superior orbital emphysema, as well as soft tissue emphysema along the left maxilla. CT, computed tomography.
oral antibiotics, and referred to the University of North Carolina (UNC) Ophthalmology Clinic. On presentation, the patient’s visual acuity was 20/25 in the (OD) and 20/20 in the (OS). Pupils were equal, round, and reactive to light with no afferent pupillary defect. Extraocular motility was full in OU. Testing of cranial nerves III, IV, V, VI, and VII was unremarkable. Intraocular pressure measured 20 mm·Hg OD and 15 mm·Hg OS. On examination of the periorbital adnexa, the left upper eyelid was ptotic and swollen without warmth or erythema (Fig. 3). Palpation of the periorbital tissue revealed crepitus in the left upper eyelid without tenderness. The swelling and crepitus was limited to the upper eyelid. Hertel measurements with a base of 103 mm were 20 mm OD and 24 mm OS. Anterior segment examination was
FIG 2. Axial CT image remarkable for significant left eyelid and superior orbital emphysema. CT, computed tomography.
e44
In this case, subcutaneous emphysema from a dental procedure was a benign occurrence. A previous report had described, in a circumstance leading to orbital and periorbital emphysema, vision loss from a compressive neuropathy.6 Other reports of subcutaneous emphysema from dental procedures have been published in dental, oral, and maxillofacial literature, and once in the ophthalmic literature.1–4 Subcutaneous emphysema as a complication of dental surgery was first reported in 1900 following a tooth extraction and has since been described in association with root canal treatment, crown placement, and periodontal surgery.7–12 It is postulated that the high-speed, air-driven instruments used in modern dental treatments combined with a disruption of the intraoral barrier introduces pressurized air into the subcutaneous tissues and facilitates the tracking of air along the facial planes. The roots of the first, second, and third molars communicate directly with the sublingual and submandibular spaces. Furthermore, the sublingual space is in direct communication with the pterygomandibular, parapharyngeal, and retropharyngeal spaces.2 Subcutaneous air following these pathways may result in local swelling in the neck, mandible, or cheek. However, isolated periorbital involvement remains a relatively rare occurrence. In the case of upper third molar extraction, air conceivably tracks along the pterygomaxillary region to enter the periorbital tissue plane. In the patient reported here, the emphysema was limited to the subcutaneous tissues of the upper eyelid. The orbit remained uninvolved as an intact orbital septum prevented subcutaneous air from entering the orbital space. CT imaging confirmed the extent of spread. Subcutaneous emphysema may present during the dental procedure or within hours of its completion. Other important conditions to consider include allergy, angioedema, hemorrhage, and infection. More serious complications can occur when air enters the parapharyngeal and retropharyngeal spaces resulting in airway compromise through pneumomediastinum or pneumothorax. There is no consensus on the appropriate management of periorbital subcutaneous emphysema in a patient who does not appear to have vision-threatening complications.5,13 However, close observation for signs of airway involvement is warranted. Even if there are no immediate signs of compromised vision or a threatened airway, the patient should be observed until the clinician is satisfied that the path of the air has been fully demarcated and is no longer advancing.14 Imaging, while recommended, may not be necessary in the absence of worrisome clinical findings. Once a stable, nonprogressive exam has been documented, the patient can be successfully managed on an outpatient basis with close follow up. In contrast, patients with orbital signs and visual compromise have had air enter the
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
Ophthal Plast Reconstr Surg, Vol. 30, No. 2, 2014
FIG 3. Photograph of the patient reveals a proptotic left orbit. Povidone-iodine solution is present on the eyelid prior to needle decompression.
FIG. 4. One-week follow up. Near resolution of orbital and periorbital subcutaneous emphysema.
orbit and may require acute management. Imaging is warranted in these cases. If significant changes (e.g., proptosis, loss of vision, increased intraocular or intraorbital pressure) have occurred, the orbit must be decompressed. Direct decompression of the air mass with a needle-coupled syringe and indirect treatment with lateral canthotomy and cantholysis have both been used successfully.15,16 Needle decompression is not as useful in periorbital emphysema, given the diffuse spread of air in the subcutaneous tissues. Prophylactic systemic antibiotic coverage tailored to the naso-oral flora is recommended in either situation. Uyanik and colleagues3 and Kim et al.17 suggest that these complications could be prevented by using a rubber dam, using remote exhaust or electrical motor-driven handpieces; avoiding the use of the compressed air syringe during irrigation; and avoiding the use of hydrogen peroxide while irrigating canals. Romeo et al.18 suggest that during third molar surgery, mucoperiosteal flap elevation, important for protecting soft tissues from cutting instruments, must be firmly, but gently retracted when using high-speed air turbine handpieces. Minimizing their use may also help prevent the aforementioned complications.19 Clinicians should be aware of subcutaneous periorbital emphysema as a complication of dental procedures and be able to distinguish this benign entity from the more dangerous orbital emphysema.
REFERENCES 1. Turnball A. A remarkable coincidence in dental surgery. Br Med J. 1900, 102:1131. 2. Parkar A, Medhurst C, Irbash M, et al. Periorbital oedema and surgical emphysema, an unusual complication of a dental procedure: a case report. Cases J 2009;2:8108. 3. Uyanık LO, Aydın M, Buhara O, et al. Periorbital emphysema during dental treatment: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011;112:e94–6. 4. Alexander CM, Owens TF. Periorbital emphysema related to oral surgery. Am J Ophthalmol 1968;66:1166–7. 5. Singh M, Phua VM, Sundar G. Sight-threatening orbital emphysema treated with needle decompression. Clin Experiment Ophthalmol 2007;35:386–7.
Case Reports
6. Buckley MJ, Turvey TA, Schumann SP, et al. Orbital emphysema causing vision loss after a dental extraction. J Am Dent Assoc 1990;120:421–2, 424; discussion 423. 7. Davies DE. Pneumomediastinum after dental surgery. Anaesth Intensive Care 2001;29:638–41. 8. Yang SC, Chiu TH, Lin TJ, et al. Subcutaneous emphysema and pneumomediastinum secondary to dental extraction: a case report and literature review. Kaohsiung J Med Sci 2006;22:641–5. 9. Steelman RJ, Johannes PW. Subcutaneous emphysema during restorative dentistry. Int J Paediatr Dent 2007;17:228–9. 10. Sujeet K, Shankar S. Images in clinical medicine. Prevertebral emphysema after a dental procedure. N Engl J Med 2007; 356:173. 11. Zemann W, Feichtinger M, Kärcher H. Cervicofacial and mediastinal emphysema after crown preparation: a rare complication. Int J Prosthodont 2007;20:143–4. 12. Imai T, Michizawa M, Arimoto E, et al. Cervicofacial subcutaneous emphysema and pneumomediastinum after intraoral laser irradiation. J Oral Maxillofac Surg 2009;67:428–30. 13. Kung JC, Chuang FH, Hsu KJ, et al. Extensive subcutaneous emphysema after extraction of a mandibular third molar: a case report. Kaohsiung J Med Sci 2009;25:562–6. 14. Bastion ML, Wong YC. A case of sneezing-related orbital emphysema treated by aspiration-decompression in the office. Ophthal Plast Reconstr Surg 2006;22:500–1. 15. Fleishman JA, Beck RW, Hoffman RO. Orbital emphysema as an ophthalmologic emergency. Ophthalmology 1984;91:1389–91. 16. Hunts JH, Patrinely JR, Holds JB, et al. Orbital emphysema. Staging and acute management. Ophthalmology 1994;101:960–6. 17. Kim Y, Kim MR, Kim SJ. Iatrogenic pneumomediastinum with extensive subcutaneous emphysema after endodontic treatment: report of 2 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2010;109:e114–9. 18. Romeo U, Galanakis A, Lerario F, et al. Subcutaneous emphysema during third molar surgery: a case report. Braz Dent J 2011;22:83–6. 19. Sekine J, Irie A, Dotsu H, et al. Bilateral pneumothorax with extensive subcutaneous emphysema manifested during third molar surgery. A case report. Int J Oral Maxillofac Surg 2000;29:355–7.
Mucosa-Associated Lymphoid Tissue Lymphoma Masquerading as Herniated Orbital Fat Cindy S. Hwang, M.D.*, Pedro Diaz-Marchan, M.D.†, and Douglas P. Marx, M.D.* Abstract: Lymphomas are the most common primary orbital malignancies in adults. The authors present a 62-year-old Hispanic woman with a 2-year history of slowly enlarging bilateral lower eyelid masses that the patient described as “bags.” On palpation, firm, mobile, nontender masses with associated tear trough deformities were noted. Biopsy of the left lower eyelid mass was consistent with a mucosaassociated lymphoid tissue lymphoma. Herniated orbital fat is an extremely common finding in the aging population and is often associated with a prominent tear trough. The patient with orbital lymphoma appeared to have herniated orbital fat with associated tear trough deformities. Lymphoma
Departments of *Ophthalmology and †Radiology, Baylor College of Medicine, Houston, Texas, U.S.A. Accepted for publication March 4, 2013. The authors have no financial or conflicts of interest to disclose. Address correspondence and reprint requests to Douglas P. Marx, M.D., 1977 Butler Boulevard, Baylor College of Medicine Alkek Eye Center, Houston, TX 77030. E-mail: [email protected] DOI: 10.1097/IOP.0b013e31829aedf3
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
e45
