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OP.17,18 Junquera et al13 described the first case of simultaneous maxillofacial osteomyelitis secondary to OP in 2005. Since then, very few cases have been published.5,8,13Y15,18 The synchronous presence of maxillomandibular osteomyelitis associated with cutaneous fistula and purulent nasal discharge has not quite been described in the literature. In our patient, osteomyelitis was the result of multiple dental extractions performed despite knowing the underlying systemic disease. The best treatment for patients with OP seems to be the preventive management. At present, no effective medical treatment exists for OP. As dental problems such as a delayed tooth eruption, ankylosis, abscesses, cysts, and fistulas are common, routine maintenance of oral hygiene should include tooth caries prevention, fluoride application, and dietary counseling.6 Teeth should be treated endodontically, if possible, instead of being removed, because of the increased risk of infection. If the patient needs a tooth extraction, special attention must be paid to the possibility of developing a possible subsequent bone infection. The most important factors associated with the appearance of infectious complications are impaired resistance to infection due to neutropenia and reduction of vascular supply to the bone, which limits the availability of antibiotics in the infection area.17 Dental practitioners should be aware of decreased bone vascularity and damaged white cell function, which may provoke the development of osteomyelitis after dental extractions, as was observed in our case.19 The appearance of osteomyelitis in the maxilla is much rarer than in the mandible probably because the former has a thin cortical bone and a rich collateral blood supply. Osteomyelitis associated with OP is a potentially serious infection that has a prolonged course and generally resists treatment. There are very few studies with successful treatment. In many patients, osteomyelitis remains unresolved indefinitely.13,17 Ultimate treatment has not been established and remains a challenge. If an osteomyelitis occurs, the more reasonable options are long antibiotic therapy according to the antibiogram and local bone curettage. Specific antibiotic recommendations include, among others, amoxicillin and clavulanic acid, amoxicillin, metronidazole, levofloxacin, and clindamycin.19 However, antibiotics hardly reach the affected area because of decreased bone vascularity. Other treatments described include incision and drainage, sequestrectomy, tooth extraction, decortication, saucerization, bone resection, and hyperbaric oxygen.10,12,14,15 Although surgery plays a significant role in the management of this condition, the challenge is to indicate the best surgical procedure to each patient. Nevertheless, there is a tendency to treat these patients with a conservative surgical approach.7 The adult OP is a rare disease, in which to perform a clinical and radiographic study is essential to establish an accurate diagnosis and appropriate management. Our case is worthy of discussion because the maxillomandibular osteomyelitis associated with cutaneous fistula and nasal purulent discharge is very unusual. Bone infection was difficult to control despite the surgical and antibiotic treatment performed. Our case outlines how prolonged antibiotic therapy together with a conservative operative procedure was associated with no recorded acute infective exacerbations in the long term, but complete healing of the case was not achieved.

REFERENCES 1. Stark Z, Savarirayan R. Osteopetrosis. Orphanet J Rare Dis 2009;4:5 2. Tolar J, Teitelbaum SL, Orchard PJ. Osteopetrosis. N Engl J Med 2004;351:839Y849 3. Albers-Schonberg HE. Rontgenbilder einer seltenen Knochenerkrankung. Munch Med Wschr 1904;51:365 4. Karshner RG. Osteopetrosis. AJR 1926;16:405Y419 5. Krithika C, Neelakandan RS, Sivapathasundaram B, et al. Osteopetrosisassociated osteomyelitis of the jaws: a report of 4 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009;108:e56Ye65

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6. Lam DK, Sa´ndor GK, Holmes HI, et al. Marble bone disease: a review of osteopetrosis and its oral health implications for dentists. J Can Dent Assoc 2007;73:839Y843 7. de Oliveira Hdo C, Pereira Filho VA, Gabrielli MF, et al. Marginal resection for treatment of mandibular osteomyelitis associated with osteopetrosis: case report. J Craniomaxillofac Surg 2011;39:525Y529 8. Barry CP, Ryan CD, Stassen LF. Osteomyelitis of the maxilla secondary to osteopetrosis: a report of 2 cases in sisters. J Oral Maxillofac Surg 2007;65:144Y147 9. Albuquerque MA, Melo ES, Jorge WA, et al. Osteomyelitis of the mandible associated with autosomal dominant osteopetrosis: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:94Y98 10. Og˘u¨tcen-Toller M, Tek M, Sener I, et al. Intractable bimaxillary osteomyelitis in osteopetrosis: review of the literature and current therapy. J Oral Maxillofac Surg 2010;68:167Y175 11. Tohidi E, Bagherpour A. Clinicoradiological findings of benign osteopetrosis: report of two new cases. J Dent Res Dent Clin Dent Prospects 2012;6:152Y157 12. Tabrizi R, Arabi AM, Arabion HR, et al. Jaw osteomyelitis as a complication in osteopetrosis. J Craniofac Surg 2010;21:136Y141 13. Junquera L, Rodrı´guez-Recio C, Villarreal P, et al. Autosomal dominant osteopetrosis and maxillomandibular osteomyelitis. Am J Otolaryngol 2005;26:275Y278 14. Trivellato AE, Ribeiro MC, Sverzut CE, et al. Osteopetrosis complicated by osteomyelitis of the maxilla and mandible: light and electron microscopic findings. Head Neck Pathol 2009;3:320Y326 15. Va´zquez E, Lo´pez-Arcas JM, Navarro I, et al. Maxillomandibular osteomyelitis in osteopetrosis. Report of a case and review of the literature. Oral Maxillofac Surg 2009;13:105Y108 16. Yamada T, Mishima K, Imura H, et al. Osteomyelitis of the mandible secondary to infantile osteopetrosis: a case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009;107:e25Ye29 17. Barry CP, Ryan CD. Osteomyelitis of the maxilla secondary to osteopetrosis: report of a case. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2003;95:12Y15 18. Long RG, Ziccardi VB. Osteopetrosis of the maxilla. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;91:139Y140 19. Sekerci AE, Sisman Y, Ertas ET, et al. Infantile malignant osteopetrosis: report of 2 cases with osteomyelitis of the jaws. J Dent Child (Chic) 2012;79:93Y99

Successful Microsurgical Revascularization of an Almost Totally Amputated Ear Lobe by Horse Bite Mehmet Dadaci, MD, Ayse Ozlem Gundesliog˘lu, MD, Bilsev Ince, MD, Zeynep Altuntas, MD Abstract: Total or subtotal amputation of the external ear related to horse bite is an uncommon situation. In our case, we report From the Department of Plastic, Reconstructive and Aesthetic Surgery, School of Medicine, Necmettin Erbakan University, Konya, Turkey. Received May 27, 2013. Accepted for publication August 26, 2013. Address correspondence and reprint requests to Mehmet Dadaci, MD, Department of Plastic & Reconstructive and Aesthetic Surgery, School of Medicine, Necmettin Erbakan University, 42080 Meram, Konya, Turkey; E-mail: [email protected] This study conformed to the Helsinki Declaration. The authors report no conflicts of interest. Copyright * 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000405

* 2014 Mutaz B. Habal, MD

Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

The Journal of Craniofacial Surgery

& Volume 25, Number 1, January 2014

Brief Clinical Studies

successful microsurgical revascularization of almost totally amputated ear of a 75-year-old male patient caused by a horse bite. Key Words: Ear amputation, ear lobe replantation, horse bite

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otal or subtotal amputation of the external ear related to horse bite is an uncommon situation. The first-choice therapy for the reconstruction of totally or subtotally amputated ear is microsurgical replantation. Replantation of the amputated part gives the best results regarding the consistence of shape, texture, and color match, but many reasons such as the small vessel size of the amputated part, difficulty of the opposition of the vessels, being not a clean-cut laceration, and associated traumas besides the main injury and avulsions caused by human and animal bites make microsurgical repair difficult.1,2 We report a case of nearly total ear amputation caused by horse bite that was revascularized by the anastomosis of an artery only.

FIGURE 1. Ear subtotal amputation. Ear lobe had no circulation.

operation date, reduced to 11 g/dL in 16 days. Total healing was observed on the postoperative 30th day (Figs. 2 and 3). Smoking was prohibited for the postoperative 1-month period.

CLINICAL REPORT

DISCUSSION

A 75-year-old man with subtotal ear amputation was seen in the emergency department. The patient reported that his horse bit his left ear while he was taking care of it after the labor. He was brought to the hospital within 45 minutes after the injury. His physical examination in the emergency department revealed a subtotal ear amputation; only a small segment of 1 cm of distal ear helix between the ear lobe and conchal cartilage was intact. The ear lobe had no circulation (Fig. 1). After the detection of a pulsatile arterial hemorrhage from the conchal cartilage site, the patient was transferred to the operating room for revascularization. The patient was monitored. As his fasting duration was 2 hours, had blood pressure of 220/110 mm Hg, had chronic obstructive lung disease, and was a heavy smoker, general anesthesia was not applied. Infiltration anesthesia was carried out by injecting 10 mL of 2% prilocaine hydrochloride around the ear after intravascular sedation with 3 mg of intravenously administered midazolam; 1 g of sulbactam-ampicillin was administered intravenously for prophylaxis. After the proper disinfection and wrapping of the patient exploration were conducted using a surgical microscope, a microclamp was applied to the artery with the pulsatile hemorrhagia, and the vessel, which was thought to be a branch of the posterior auricular artery, was dissected under the microscope. The reciprocal ends of the artery were apposed with the aid of an approximator and sutured each other with 10-0 nylon sutures. After the repair clamps were removed, the anastomosis was observed to work. The color of the ear lobule changed to pink, and the hemorrhage from the venous circulation was observed to increase. No veins could be found for repair despite proper dissection. Cartilage was sutured with 4-0 poliglecaprone 25 (Monocryl), and the skin was sutured with 5-0 polypropylene. The operation lasted approximately 2 hours. Postoperatively, the patient received 5000 IU of heparin intravenously every 8 hours, dextran 40 at a rate of 500 mL every 8 hours for 5 days, and 1 tablet of acetylsalicylic acid (300 mg) orally per day. Sulbactam-ampicillin 1.5 g was administered intravenously every 6 hours for 7 days, and tetanus and rabies vaccines were administered for prophylaxis. Approximately 30 minutes after the surgery, the revascularized ear became congested, and a leech was applied. Postoperatively, the patient had leech application once every 4 hours for 3 days, then every 6 hours for the next 4 days, and 1 week later every 12 hours for 4 days. After the 10th day, leech was applied once in 2 days for a total of 3 times more. On the 16th day after the operation, leech application was stopped, but the patient continued to take 300 mg acetylsalicylic acid day. Sutures were removed, and the patient was discharged from the hospital at the 16th postoperative day. The hemoglobin level of the patient, which was 16.5 g/dL on the

We demonstrated in our case that the nearly totally amputated ear lobe and partially amputated ear helix were rescued totally by microvascular anastomosis of an artery only. To our knowledge, there are no cases of revascularization of an ear amputation caused by a horse bite in the literature. We had no trouble of wound infection due to the mouth flora of the horse. After the healing period, total integrity of the ear was maintained with little scar tissue. Many cases have been reported in the literature, most of which were of total or subtotal avulsions that were reconstructed by microvascular anastomosis. There are few reports of successful replantation of amputated ear lobe and lower one third of the ear. Microsurgical replantation of the auricle after traumatic amputations remains the best reconstructive option, but it is a very challenging procedure because of the small size of the vessels. Best results can be achieved with successful anastomosis of both artery and veins.1Y3 Park et al3 described the details of blood supply to the ear and proposed that the retroauricular artery, which transverses the concha, can be used for most cases of microvascular anastomosis. Our chance in this case was the observation of the pulsations of the retroauricular artery because the patient had a high blood pressure. The corresponding end of the artery was found and dissected, and the

FIGURE 2. Appearance of the ear on the postoperative 30th day.

* 2014 Mutaz B. Habal, MD

Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

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4. Safak T, Ozcan G, Kecik A, et al. Microvascular ear replantation with no vein anastomosis. Plast Reconstr Surg 1993;92:945Y948 5. Weinfeld AB, Yuksel E, Boutros S. Clinical and scientific considerations in leech therapy for the management of acute venous congestion; an updated review. Ann Plast Surg 2000;45:207Y212 6. Pao-Yuan L, Yuan-Cheng C, Ching-Hua H, et al. Microsurgical replantation and salvage procedures in traumatic ear amputation. J Trauma 2010;4:69 7. Sung WJ, Junsang L, Suk JO, et al. A review of microvascular ear replantation. J Reconstr MNcrosurg 2013;29:181Y188 8. Abrahamian FM, Goldstein EJ. Infections, microbiology of animal bite wound. Clin Microbiol Rev 2011;24:23 9. Edixhoven P, Sinha SC, Dandy DJ. Horse injuries. Injury 1981;12: 279Y282 10. Kose R, Sogut O, Mordeniz C. Management of horse and donkey bite wounds; a series of 24 cases. Plast Reconstr Surg 2010;125:251eY252e

Eagle Syndrome Pedro Costa Ferreira, MD,* Ma´rio Mendanha, MD,* Tiago Frada, MD,Þ Jorge Carvalho, MD,* A´lvaro Silva, MD,* Jose´ Amarante, MD, PhD*

FIGURE 3. Appearance of the ear on the postoperative 30th day.

vessels were anastomosed. We could not find a vein with enough diameter to be repaired upon exploration of the ear, which had active bleeding from the lower third part after revascularization. When no venous anastomosis is possible, alternative methods are applied to relieve venous congestion, such as milking, pin pricking, medical leech application, and an artery-to-vein fistula. When the venous anastomosis is technically difficult, medical leeches can relieve the venous congestion. Also, acetylsalicylic acid and heparin can be used for anticoagulation.4Y6 Pao-Yuan et al6 and Sung et al7 reported that ear amputations, which are commonly accompanied by crush or avulsion injuries, make microsurgical replantation difficult. Surgical repair in 2 cases of ear amputation caused by human bite failed because of infection and avulsion injury. Horse bites are relatively infrequent, but when they occur, they are associated with crush injuries and tissue loss. Most reports of the bacteriology of horse bite wounds in humans have revealed infections to be polymicrobial, with a mixture of aerobic and anaerobic organisms.8Y10 Our case has the importance of being an ear amputation caused by horse bite, which is successfully repaired by performing revascularization, and the ear lobe was rescued. Antibiotic prophylaxis with sulbactam-ampicillin, tetanus and rabies prophylaxis, and early repair were found to be important factors. In conclusion, in case of an ear amputation related to horse bite when it is not possible to repair the veins, successful results can be obtained by microvascular anastomosis of an artery only.

REFERENCES 1. Pennington DG, Lai MF, Pelly AD. Successful replantation of a completely avulsed ear by microvascular anastomosis. Plast Reconstr Surg 1980;65:820Y823 2. Juri J, Irigaray A, Juri C, et al. Ear replantation. Plast Reconstr Surg 1987;80:431Y435 3. Park C, Lineaweaver WC, Rumly TO, et al. Arterial supply of the anterior ear. Plast Reconstr Surg 1992;90:38Y44

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Abstract: Eagle syndrome, also known as elongated styloid process, is a condition first described by Watt Eagle in 1937. It occurs when an elongated styloid process or calcified stylohyoid ligament causes recurrent throat pain or foreign body sensation, dysphagia, or facial pain. Additional symptoms may include neck or throat pain with radiation to the ipsilateral ear. It is usually hard to diagnose because the symptoms related to this condition can be confused with those attributed to a wide variety of facial neuralgias. In this article, a case of Eagle syndrome exhibiting unilateral symptoms with bilateral elongation of styloid process is reported. Key Words: Eagle syndrome, elongated styloid process

E

agle syndrome (ES) is the term given to the symptomatic elongation of the styloid process.1 Typical styloid processes are between 2.5 and 3.0 cm in length. Lengths greater than 3 cm are considered elongated. Bilateral elongation is common, although symptoms are typically unilateral.2 It is characterized by pharyngodynia localized in the tonsillar fossa and sometimes accompanied by dysphagia, odynophagia, foreign body sensation, and temporary voice changes.3 Many important neurovascular structures lie near the tip of the styloid process. The internal carotid artery, internal jugular vein, and cranial nerves X, XI, and XII lie on its medial side. In some cases, the stylohyoid apparatus compresses the internal and/or the external

From the *Department of Plastic Reconstructive, Aesthetic, Maxillofacial Surgery and Burn Unit, Hospital de Sa˜o Joa˜o, Porto Medical School, Porto; and †Department of Orthopaedic Surgery, Hospital de Braga, and School of Health Sciences, Life and Health Sciences Research Institute, University of Minho, Braga, Portugal. Received May 27, 2013. Accepted for publication August 26, 2013. Address correspondence and reprint requests to Pedro Manuel Costa Ferreira, Rua Joa˜o Rosa n- 52, Habita0a˜o D72, 4460-189 Senhora da Hora, Portugal; E-mail: [email protected] The authors report no conflicts of interest. Copyright * 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000392

* 2014 Mutaz B. Habal, MD

Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

Successful microsurgical revascularization of an almost totally amputated ear lobe by horse bite.

Total or subtotal amputation of the external ear related to horse bite is an uncommon situation. In our case, we report successful microsurgical revas...
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