CLINICAL STUDIES
Facial Paralysis and Mediastinitis Due to Odontogenic Infection and Poor Prognosis Abdulkadir Bucak, PhD,* S¸ahin Ulu, PhD,* Serdar Kokulu, PhD,Þ Gu¨rhan O¨z, PhD,þ Okan Solak, PhD,þ Orhan Kemal Kahveci, PhD,* and Abdullah Ayc¸ic¸ek, PhD* Abstract: Cervical necrotizing fasciitis (CNF) is a rare, rapidly advancing infection that involves the skin, the subcutaneous fibrofatty tissue, as well as the superficial and deep fascia and can cause life-threatening complications. The most frequent initiating factors in the head and neck region are a primary odontogenic infection, a peritonsillar infection, as well as posttraumatic or iatrogenic skin and mucosal injuries. Necrotizing fasciitis (NF) can expand within hours, and the reported mortality rate is up to 75% with delay interference. If the patients have any risk factors, poor prognosis can be seen. In this study, 1 patient with CNF with a history of peritonsillar infection and 2 patients with CNF who had a history of odontogenic infection with spreading to the temporal region and the mediastinum were described, with information of the literature and a clinical experience that was gained from 5 patients with NF who were seen at our clinic in the recent year, despite the fact that CNF was not seen up to last year. None of the patients had any risk factors. One of them had a worse clinical state with ascending infection to the temporal region, cranial nerve paralysis, and descending necrotizing mediastinitis, but he recovered from NF. After the oral intake began, dyspnea due to aspiration was seen and he died because of sepsis and multiorgan dysfunction. We aimed to attract attention to the importance of dental pathologies and increased mortality in a healthy patient. Key Words: Cervical necrotizing fasciitis, odontogenic infection, mediastinitis, cranial nerve paralysis (J Craniofac Surg 2013;24: 1953Y1956)
T
he term necrotizing fasciitis (NF) was first used by Wilson in 1952 and was found to have no specific microorganism related to the disease.1 Cervical necrotizing fasciitis (CNF) is a rare, rapidly advancing infection that involves the skin, the subcutaneous fibrofatty tissue, as well as the superficial and deep fascia and can cause life-threatening complications. It is also known as flesh-eating bacteria syndrome.2,3 The inflammatory process of NF is believed to be collagen necrosis caused by activated proteolytic enzyme, such From the *Departments of Otolaryngology, †Anesthesiology and Reanimation, and ‡Thoracic Surgery, Faculty of Medicine, Afyon Kocatepe University, Afyonkarahisar, Turkey. Received March 6, 2013. Accepted for publication May 1, 2013. Address correspondence and reprint requests to Abdulkadir Bucak, PhD, Department of Otorhinolaryngology, Afyon Kocatepe University, Faculty of Medicine Ali Cetinkaya Kampusu T@p Fakultesi, Izmir Karayolu 8.km 03200, Afyonkarahisar, Turkey; E-mail: [email protected] The authors report no conflicts of interest. Copyright * 2013 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0b013e31829ac617
The Journal of Craniofacial Surgery
as hyaluronidase from Streptococcus and proteases from anaerobic gram negative, in the superficial and deep fascial planes, which might relate to thrombosis, ischemia, and tissue necrosis.4,5 Widespread necrosis supplies a nutritious culture environment for bacterial growth.6 The relative lack of vascularity of the relevant fascial planes has also been hypothesized as a contributing factor in NF. Necrotizing fasciitis affects mostly the abdominal wall, the groin, and the extremities, and it is rarely seen in the head and neck region. The mortality rate of CNF is very high because of the tendency to spread to the mediastinum; this condition is called descending necrotizing mediastinitis (DNM). Because the infection always exhibits a fulminant course, the key for successful treatment of CNF is early diagnosis, which, when combined with aggressive surgical debridement of necrotic tissues and administration of parenteral broad-spectrum antibiotics as early as possible, can substantially improve the outcome.4 Up to last year, the patient with CNF was not seen in our clinic, but in the recent year, the 5 patients with CNF (2 of them were presented in the same journal) were admitted to our clinic.7 Hence, we want to share the worriment about CNF that rises up and the clinical experience that was gained from patients with CNF. In this study, 3 patients with CNF (1 patient with the spreading to the temporal region and the mediastinum) are presented with information of the literature.
PATIENT 1 A 36-year-old woman presented to our department with a painful swelling in the submental region and both the submandibular regions, which was more pronounced on the right side for 3 days, dysphagia, common status failure, poor level of nourishment, and fever for 7 days. The examination results revealed diffuse swelling, sensitivity, and erythema of the right submandibular and submental areas. Trismus was initially present. The intraoral examination results revealed diffuse swelling and erythema, especially of the right peritonsillar region, a fistula, necrotized tissues in the fistula, as well as effluxing dirty-yellow, necrotic, foul-smelling fluid at the oral basement. She had no history of systemic disease or substance abuse. A cervical computed tomographic (CT) scan was obtained. The CT scan showed abscesses from the nasopharyngeal level to the inferior border of the hyoid bone with gas formation in the oral basement and another abscess in the right oropharyngeal level. The patient was thought to have CNF clinically and radiologically. Initially, the patient underwent tracheotomy with local anesthesia with sedation, followed by an urgent neck exploration under general anesthesia with bilateral modified apron incision. After passing platysma, an effluxing dirty-yellow, necrotic, foul-smelling fluid was seen at the space of the fascia. Necrotized tissues were not detected at the left side but were seen from the skull base to the superior of the clavicle reaching to the parapharyngeal region and to the paratonsillar region. The right parapharyngeal area was fistulized to the oral basement. The necrotized tissues were cleaned, and the cleaning was continued until bleeding with aggressive debridement. Intravenous imipenem (3 500 mg/d) was administered, with consultation from an infectious diseases clinic.
& Volume 24, Number 6, November 2013
Copyright © 2013 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
1953
The Journal of Craniofacial Surgery
Bucak et al
FIGURE 1. Computed tomographic scan: common air densities in the temporoparietal scalp, the infratemporal fossa, the narrowing left nasopharyngeal air column, and neck spaces.
The patient was admitted to the critical care unit. The necrotized tissues were debrided every day until bleeding. Streptococcus viridans group had been isolated in wound culture, and histopathologic findings were compatible with NF. The patient was moved out from the critical care unit on day 7 and began oral nourishment on day 18; then, the patient was discharged asymptomatic.
PATIENT 2 An 81-year-old man presented to our department with asymmetric appearance of the eyes and the mouth, painfully hard swelling in
& Volume 24, Number 6, November 2013
the left temporal region and common neck region and the left temporal area, trismus, dyspnea, dysphagia, common status failure, poor level of nourishment, as well as high fever. He had a history of asymmetric appearance of the eyes and mouth, trismus, and swelling on the neck and the temporal region for 3 days, as well as toothache for 15 days. He had no history of systemic disease. The examination results revealed diffuse painful swelling of the left common neck, buccal, and left temporal regions with paralysis of the left side of the face (compatible with Hausse-Breckmann type 4) as well as crepitation of the left temporal region; the intraoral examination results revealed a fistula on the left retromolar trigon with poor dental health. Cervical and thoracic CT scan was obtained. The CT scan showed narrowing of the left nasopharyngeal air column, gas formation, and abscesses in the left temporoparietal scalp, the infratemporal fossa, the masseter muscle, the oral basement, as well as the parapharyngeal, retropharyngeal and submandibular spaces to the thyroid gland level (Fig. 1). Pneumomediastinum and left pleural effusion were seen in the mediastinum scanning (Fig. 2). The patient was thought to have CNF and DNM. Initially, the patient underwent tracheotomy with local anesthesia with sedation, followed by an urgent neck exploration under general anesthesia. After passing platysma, an effluxing dirty-yellow, necrotic, foul-smelling fluid was seen at the space of the fascia. Necrotized tissues were not detected at the right side but were seen at the left side from the skull base to the superior of the clavicle reaching to the parapharyngeal region, the left infratemporal fossa, as well as the lateral and medial area of the mandible. A fistula, necrotized tissues in the fistula, and effluxing dirty-yellow, necrotic, foul-smelling fluid were seen on the left retromolar trigon. Another surgical debridement was performed, followed by a 6-cm excision of the temporoparietal region (Fig. 3). A new surgical debridement was performed through thoracic surgery; the debridement of the upper, posterior, inferior, and anterior mediastinum as well as the paraesophageal, paratracheal, and posterior spaces of the vena cava was performed; the tissues were also cleaned, and the cleaning was continued until bleeding with aggressive debridement (Fig. 4). Intravenous imipenem (4 250 mg/d) was administered, with consultation
FIGURE 2. Computed tomographic scan: common air densities in the left side of the trachea in the neck and the mediastinum.
1954
* 2013 Mutaz B. Habal, MD
Copyright © 2013 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery
& Volume 24, Number 6, November 2013
Facial Paralysis and Mediastinitis
DISCUSSION
FIGURE 3. Abscesses in the left temporoparietal region.
from an infectious diseases clinic. The patient was admitted to the critical care unit. The necrotized tissues were debrided every day until bleeding. A new small necrotized tissue was seen on the right submandibular region and was debrided. After day 18 of the treatment, necrotized tissues were not seen and debridement was stopped. No microorganism had been isolated in wound culture, and histopathologic findings were compatible with NF. Thus, we can say that the patient had a good response to the treatment, so oral intake was begun. However, a serious dyspnea was seen because of aspiration and the oral intake was stopped. On the following days, aspiration pneumonia developed. In our opinion, nerve disorder may be developed because of NF. While continuing the therapy at the critical care unit, Candida species had been isolated in blood culture and Acinetobacter baumannii had been isolated in culture of tracheal aspiration. Then, the patient died on day 29 because of sepsis and multiorgan dysfunction.
PATIENT 3 A 40-year-old woman presented to our department with a painful swelling in the submental and right submandibular regions for 2 days, toothache, dysphagia, common status failure, poor level of nourishment, as well as fever for 5 days. Antibiotherapy was applied by a dentist to the patient for the odontogenic infection. However, she was referred to our clinic because of her augmented symptoms. She had no history of systemic disease or of substance abuse. The examination results revealed diffuse painful swelling of the right submandiblar and submental areas. The intraoral examination results revealed diffuse swelling and erythema, especially of the right sublingual region. Cervical CT scan was obtained. The CT scan showed narrowing of the left hypopharengeal air column, gas formation, as well as abscesses in the right submandibular space and oral basement. The patient was thought to have CNF. First of all, the patient underwent tracheotomy with local anesthesia with sedation, followed by an urgent neck exploration under general anesthesia. After passing platysma, an effluxing dirty-yellow, necrotic, foul-smelling fluid was seen at the space of the fascia. Necrotized tissues were seen at the right submandibular and submental area reaching to the oral basement; the tissues were cleaned, and the cleaning was continued until bleeding with aggressive debridement. Intravenous meropenem (3 1 g/d) was administered, with consultation from an infectious diseases clinic. The patient was admitted to the critical care unit. The necrotized tissues were debrided every day until bleeding. Prevotella species had been isolated in blood culture, and histopathologic findings were compatible with NF. The patient was moved out from the critical care unit on day 3 and began oral nourishment on day 10; then, the patient was discharged asymptomatic.
Necrotizing fasciitis of the head and the neck is rare. The most frequent initiating factors for NF in the head and neck region are dental pathology, parapharyngeal or peritonsillar infections, posttraumatic or iatrogenic skin, mucosal injuries, froncule, fractures of the mandible, trauma caused by firearms, tracheotomy, and radiotherapy. The systemic disease, organ failure, drugs (eg, steroids), malnutrition, underlying malignancy, chronic alcoholism, and suppressed immunity seem to be risk factors in defining the induction, progression, and results of the disease.4,7 One of the 5 patients with CNF (including the 2 patients who were previously presented) had diabetes mellitus, but the other 4 patients were healthy and had no risk factors.7 Three of the 5 patients had an odontogenic infection, and the other one had a peritonsillar infection. Thus, we can say that peritonsillar infection and dental pathologies (especially with poor dental health) have a very important role for the development of NF. In addition, NF seems to be rising among healthy people. This condition may be related to the changed environmental conditions, misuse of antibiotics, and/or living conditions. Symptoms of NF may be nonspecific at onset and are dependent on the stage of the disease and its origin site and early stages; NF can be misdiagnosed such as cellulitis or erysipials.8 As the infection progresses, the skin becomes increasingly erythematous, tense, and smooth without prominent margins compared with normal skin. As so in our patients, reliable indicators of NF can be fistule and effluxing dirty-yellow, necrotic, foul-smelling fluid at the oral mucosa, the subcutaneous crepitus, as well as pain and common status failure that do not correlate to the local skin findings. Paralysis of the cranial nerve has been reported in the literature as an encountered complication of NF. The cranial nerve paralysis 9,10,12 could be seen in the progression of the NF.5,7,9 The patient, who was previously presented to our clinic, had paralysis of the right vocal cord.7 From a distance, one of the patients (patient 2) had no history of facial paralysis and aspiration with oral intake; however, he was found to have left facial paralysis (compatible with Hausse-Breckmann type 4), and this was thought to be caused by NF. To the best of our knowledge, a patient with facial paralysis, NF of the temporal region, and DNM has not been documented in the literature, and this condition can be related to the spreading of the
FIGURE 4. The view of the neck after debridement of the necrotic tissues.
* 2013 Mutaz B. Habal, MD
Copyright © 2013 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
1955
Bucak et al
The Journal of Craniofacial Surgery
disease. After recovery, oral intake caused aspiration and dyspnea to the same patient. This condition can be related to the involvement of the superior laryngeal nerve. Hence, we can say that the oral intake after recovery is very important for patients with cervical NF and DNM. The most important issue is the diagnosis of NF. Delay in diagnosis causes poor prognosis. The criterion standard is intravenous contrast CT scan that helps in early diagnosis and shows soft tissue gas formation. A CT scan distinguishes between abscesses, erysipelas, or cellulitis, better describes the characteristics of the lesions, and can give a hint on monitoring of the disease, recurrence of purulence, and new gas formation.7,10 In addition, it can help to establish the need, extent, and boundary of surgery. Air density between the neck fascias, the mediastinum, and the temporal region was shown in the CT scan of our patients. This situation is pathognomonic for NF. We prefer CT scan for all patients who were admitted to our clinic with deep infections in the neck. Also, in our patients, accurate and rapid diagnosis, nutritional support whether enteral or parenteral, effective intravenous broadspectrum antibiotherapy with anaerobic coverage, rapid control of the upper airway with tracheotomy, as well as early and vigorous surgical debridement of the cervical spaces and/or the mediastinum until the disappearance of the physical and radiologic findings of the necrosis and the abscess with intensive medical care were keys to successful outcome in treatment of NF.2,8 Immediate and well-balanced surgical debridement between infected and healthy tissues with wide extensive fasciotomies with exposure of all involved fascia and excision of necrotic tissues until exposure of viable bleeding tissue are crucial for patient survival.11 This should be done early not only to control the primary infectious process but also to remove the necrotic tissue that is the source of secondary infection and that of toxin production.4 Also, early surgical interference would restore adequate tissue perfusion with appropriate antibiotics diffusion, minimize loss of tissue, and prevent cosmetic disfigurement. Dermal necrosis was not seen in our patients with successful management of therapy. When the wound is opened, it should be well irrigated with betadine and hydrogen peroxide solution, and this regimen should be repeated periodically until further tissue necrosis stops and growth of the fresh viable tissue is observed.8 We used wound dressings soaked in nitrofurazone soluble ointment for daily dressings. Multiple operative sessions are frequently required for full surgical control. Descending necrotizing mediastinitis was seen in 3 of the 5 patients who had primary odontogenic infection. Because an extensive fascial communication exists between the gums, the neck, and the mediastinum, odontogenic infections can rapidly spread to the mediastinum facilitated by gravity, respiration, and negative intrathoracic pressure.12 To our knowledge, from the neck to the mediastinum, there exist 3 potential major pathways for the infection to spread along the fascial spaces in DNM. The retropharyngeal space (danger space) accounts for 70% of cases of DNM. It lies behind the esophagus (the visceral layer of the deep cervical fascia) and anterior to the prevertebral fascia, extending from the base of the skull to the posterior mediastinum.13 The development of DNM leads to fatality complication. Descending necrotizing mediastinitis has a mortality rate that is 4 times greater than that of cervical involvement only.14 The involvement of the posterior mediastinum was seen in our 2 patients with DNM. They died because of sepsis and multiorgan dysfunction, despite recovery of the neck, the temporal region, and the mediastinum of patient 2. Necrotizing fasciitis, especially of odontogenic origin, is almost always polymicrobial and often involves a combination of aerobic and anaerobic bacterias. Because of synergistic work, polymicrobial infections are more pathogenic than monoinfections are.6,15 We preferred to use the broad-spectrum antibiotherapy with anaerobic
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coverage. Except one, in our patients, no microorganism had been isolated in wound cultures. This situation can be related to antibiotherapy by local physicians before hospitalization. Other adjunctive approaches to treatment include hyperbaric oxygen therapy and intravenous immunoglobulin.6 Hyperbaric oxygen treatment can be begun if the patient is stable and can tolerate it.7 The 2 patients had a bad clinical state and the other 3 patient had a good response to the treatment, so we did not use hyperbaric oxygen treatment. As a conclusion, necrotizing fasciitis can expand within hours and the reported mortality rate is up to 75% with delay interference.16 Paralysis of the cranial nerve, such as the facial nerve and the vagal nerve branches, were seen in the progression of NF. Necrotizing fasciitis can be seen in healthy participants with increased ratio, and it should always be borne in mind when faced with deep infections of the neck.
REFERENCES 1. Wilson B. Necrotizing fasciitis. Am Surg 1952;18:416Y431 2. Lopez-Fernandez R, Ramirez-Melgoza J, Martinez-Aguilar NE, et al. Growth factor-enriched autologous plasma improves wound healing after surgical debridement in odontogenic necrotizing fasciitis: a case report. J Med Case Rep 2011;5:98 3. Yadav S, Verma A, Sachdeva A. Facial necrotizing fasciitis from an odontogenic infection. Oral Surg Oral Med Oral Pathol Oral Radiol 2012;113:e1Ye4 4. Bono G, Argo A, Zerbo S, et al. Cervical necrotizing fasciitis and descending necrotizing mediastinitis in a patient affected by neglected peritonsillar abscess: a case of medical negligence. J Forensic Leg Med 2008;15:391Y394 5. Lee JH, Choi HC, Kim C, et al. Fulminanst cerebral infarction of anterior and posterior cerebral circulation after ascending type of facial necrotizing fasciitis. J Stroke Cerebrovasc Dis [published ahead of print August 29, 2012] doi: 10.1016/j.jstrokecerebrovasdis.2012.07.013 6. Park E, Hirsch EM, Steinberg JP, et al. Ascending necrotizing fasciitis of the face following odontogenic infection. J Craniofac Surg 2012;23:e211Ye214 7. Ulu S, Ulu SM, Oz G, et al. Paralysis of cranial nerve and striking prognosis of cervical necrotizing fasciitis. J Craniofac Surg 2012;23:1812Y1814 8. Sasindran V, Joseph A. Necrotizing fasciitis: an unusual presentation. Indian J Otolaryngol Head Neck Surg 2011;63:390Y392 9. Lorenzini G, Picciotti M, Di Vece L, et al. Cervical necrotizing fasciitis of odontogenic origin involving the temporal regionVa case report. Int J Oral Maxillofac Surg 2010;39:830Y834 10. Santos Gorjo´n P, Blanco Pe´rez P, Morales Martı´n AC, et al. Deep neck infection. Review of 286 cases. Acta Otorrinolaringol Esp 2012;63:31Y41 11. Thomas AJ, Mong S, Golub JS, et al. Klebsiella pneumoniae cervical necrotizing fasciitis originating as an abscess. Am J Otolaryngol 2012;33:764Y766 12. Gonza´lez-Garcı´a R, Risco-Rojas R, Roma´n-Romero L, et al. Descending necrotizing mediastinitis following dental extraction. Radiological features and surgical treatment considerations. J Craniomaxillofac Surg 2011;39:335Y339 13. Sakamoto H, Aoki T, Kise Y, et al. Descending necrotizing mediastinitis due to odontogenic infections. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;89:412Y419 14. Weed HG, Forest LA. Deep neck infections. In: Cummings CW, Flint PW, Harker LA, Haughey BH, Richardson MA, Robbins KT, Schuller DE, Thomas JR, eds. Cummings Otolaryngology Head & Neck Surgery. Philadelphia, PA: 2005:2521Y2522 15. Boffano P, Roccia F, Pittoni D, et al. Management of 112 hospitalized patients with spreading odontogenic infections: correlation with DMFT and oral health impact profile 14 indexes. Oral Surg Oral Med Oral Pathol Oral Radiol 2012;113:207Y213 16. Danic Hadzibegovic A, Sauerborn D, Grabovac S, et al. Necrotizing fasciitis of the neck after total laryngectomy. Eur Arch Otorhinolaryngol 2013;270:277Y280
* 2013 Mutaz B. Habal, MD
Copyright © 2013 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Facial Paralysis and Mediastinitis Due to Odontogenic Infection and Poor Prognosis Abdulkadir Bucak, PhD,* S¸ahin Ulu, PhD,* Serdar Kokulu, PhD,Þ Gu¨rhan O¨z, PhD,þ Okan Solak, PhD,þ Orhan Kemal Kahveci, PhD,* and Abdullah Ayc¸ic¸ek, PhD* Abstract: Cervical necrotizing fasciitis (CNF) is a rare, rapidly advancing infection that involves the skin, the subcutaneous fibrofatty tissue, as well as the superficial and deep fascia and can cause life-threatening complications. The most frequent initiating factors in the head and neck region are a primary odontogenic infection, a peritonsillar infection, as well as posttraumatic or iatrogenic skin and mucosal injuries. Necrotizing fasciitis (NF) can expand within hours, and the reported mortality rate is up to 75% with delay interference. If the patients have any risk factors, poor prognosis can be seen. In this study, 1 patient with CNF with a history of peritonsillar infection and 2 patients with CNF who had a history of odontogenic infection with spreading to the temporal region and the mediastinum were described, with information of the literature and a clinical experience that was gained from 5 patients with NF who were seen at our clinic in the recent year, despite the fact that CNF was not seen up to last year. None of the patients had any risk factors. One of them had a worse clinical state with ascending infection to the temporal region, cranial nerve paralysis, and descending necrotizing mediastinitis, but he recovered from NF. After the oral intake began, dyspnea due to aspiration was seen and he died because of sepsis and multiorgan dysfunction. We aimed to attract attention to the importance of dental pathologies and increased mortality in a healthy patient. Key Words: Cervical necrotizing fasciitis, odontogenic infection, mediastinitis, cranial nerve paralysis (J Craniofac Surg 2013;24: 1953Y1956)
T
he term necrotizing fasciitis (NF) was first used by Wilson in 1952 and was found to have no specific microorganism related to the disease.1 Cervical necrotizing fasciitis (CNF) is a rare, rapidly advancing infection that involves the skin, the subcutaneous fibrofatty tissue, as well as the superficial and deep fascia and can cause life-threatening complications. It is also known as flesh-eating bacteria syndrome.2,3 The inflammatory process of NF is believed to be collagen necrosis caused by activated proteolytic enzyme, such From the *Departments of Otolaryngology, †Anesthesiology and Reanimation, and ‡Thoracic Surgery, Faculty of Medicine, Afyon Kocatepe University, Afyonkarahisar, Turkey. Received March 6, 2013. Accepted for publication May 1, 2013. Address correspondence and reprint requests to Abdulkadir Bucak, PhD, Department of Otorhinolaryngology, Afyon Kocatepe University, Faculty of Medicine Ali Cetinkaya Kampusu T@p Fakultesi, Izmir Karayolu 8.km 03200, Afyonkarahisar, Turkey; E-mail: [email protected] The authors report no conflicts of interest. Copyright * 2013 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0b013e31829ac617
The Journal of Craniofacial Surgery
as hyaluronidase from Streptococcus and proteases from anaerobic gram negative, in the superficial and deep fascial planes, which might relate to thrombosis, ischemia, and tissue necrosis.4,5 Widespread necrosis supplies a nutritious culture environment for bacterial growth.6 The relative lack of vascularity of the relevant fascial planes has also been hypothesized as a contributing factor in NF. Necrotizing fasciitis affects mostly the abdominal wall, the groin, and the extremities, and it is rarely seen in the head and neck region. The mortality rate of CNF is very high because of the tendency to spread to the mediastinum; this condition is called descending necrotizing mediastinitis (DNM). Because the infection always exhibits a fulminant course, the key for successful treatment of CNF is early diagnosis, which, when combined with aggressive surgical debridement of necrotic tissues and administration of parenteral broad-spectrum antibiotics as early as possible, can substantially improve the outcome.4 Up to last year, the patient with CNF was not seen in our clinic, but in the recent year, the 5 patients with CNF (2 of them were presented in the same journal) were admitted to our clinic.7 Hence, we want to share the worriment about CNF that rises up and the clinical experience that was gained from patients with CNF. In this study, 3 patients with CNF (1 patient with the spreading to the temporal region and the mediastinum) are presented with information of the literature.
PATIENT 1 A 36-year-old woman presented to our department with a painful swelling in the submental region and both the submandibular regions, which was more pronounced on the right side for 3 days, dysphagia, common status failure, poor level of nourishment, and fever for 7 days. The examination results revealed diffuse swelling, sensitivity, and erythema of the right submandibular and submental areas. Trismus was initially present. The intraoral examination results revealed diffuse swelling and erythema, especially of the right peritonsillar region, a fistula, necrotized tissues in the fistula, as well as effluxing dirty-yellow, necrotic, foul-smelling fluid at the oral basement. She had no history of systemic disease or substance abuse. A cervical computed tomographic (CT) scan was obtained. The CT scan showed abscesses from the nasopharyngeal level to the inferior border of the hyoid bone with gas formation in the oral basement and another abscess in the right oropharyngeal level. The patient was thought to have CNF clinically and radiologically. Initially, the patient underwent tracheotomy with local anesthesia with sedation, followed by an urgent neck exploration under general anesthesia with bilateral modified apron incision. After passing platysma, an effluxing dirty-yellow, necrotic, foul-smelling fluid was seen at the space of the fascia. Necrotized tissues were not detected at the left side but were seen from the skull base to the superior of the clavicle reaching to the parapharyngeal region and to the paratonsillar region. The right parapharyngeal area was fistulized to the oral basement. The necrotized tissues were cleaned, and the cleaning was continued until bleeding with aggressive debridement. Intravenous imipenem (3 500 mg/d) was administered, with consultation from an infectious diseases clinic.
& Volume 24, Number 6, November 2013
Copyright © 2013 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
1953
The Journal of Craniofacial Surgery
Bucak et al
FIGURE 1. Computed tomographic scan: common air densities in the temporoparietal scalp, the infratemporal fossa, the narrowing left nasopharyngeal air column, and neck spaces.
The patient was admitted to the critical care unit. The necrotized tissues were debrided every day until bleeding. Streptococcus viridans group had been isolated in wound culture, and histopathologic findings were compatible with NF. The patient was moved out from the critical care unit on day 7 and began oral nourishment on day 18; then, the patient was discharged asymptomatic.
PATIENT 2 An 81-year-old man presented to our department with asymmetric appearance of the eyes and the mouth, painfully hard swelling in
& Volume 24, Number 6, November 2013
the left temporal region and common neck region and the left temporal area, trismus, dyspnea, dysphagia, common status failure, poor level of nourishment, as well as high fever. He had a history of asymmetric appearance of the eyes and mouth, trismus, and swelling on the neck and the temporal region for 3 days, as well as toothache for 15 days. He had no history of systemic disease. The examination results revealed diffuse painful swelling of the left common neck, buccal, and left temporal regions with paralysis of the left side of the face (compatible with Hausse-Breckmann type 4) as well as crepitation of the left temporal region; the intraoral examination results revealed a fistula on the left retromolar trigon with poor dental health. Cervical and thoracic CT scan was obtained. The CT scan showed narrowing of the left nasopharyngeal air column, gas formation, and abscesses in the left temporoparietal scalp, the infratemporal fossa, the masseter muscle, the oral basement, as well as the parapharyngeal, retropharyngeal and submandibular spaces to the thyroid gland level (Fig. 1). Pneumomediastinum and left pleural effusion were seen in the mediastinum scanning (Fig. 2). The patient was thought to have CNF and DNM. Initially, the patient underwent tracheotomy with local anesthesia with sedation, followed by an urgent neck exploration under general anesthesia. After passing platysma, an effluxing dirty-yellow, necrotic, foul-smelling fluid was seen at the space of the fascia. Necrotized tissues were not detected at the right side but were seen at the left side from the skull base to the superior of the clavicle reaching to the parapharyngeal region, the left infratemporal fossa, as well as the lateral and medial area of the mandible. A fistula, necrotized tissues in the fistula, and effluxing dirty-yellow, necrotic, foul-smelling fluid were seen on the left retromolar trigon. Another surgical debridement was performed, followed by a 6-cm excision of the temporoparietal region (Fig. 3). A new surgical debridement was performed through thoracic surgery; the debridement of the upper, posterior, inferior, and anterior mediastinum as well as the paraesophageal, paratracheal, and posterior spaces of the vena cava was performed; the tissues were also cleaned, and the cleaning was continued until bleeding with aggressive debridement (Fig. 4). Intravenous imipenem (4 250 mg/d) was administered, with consultation
FIGURE 2. Computed tomographic scan: common air densities in the left side of the trachea in the neck and the mediastinum.
1954
* 2013 Mutaz B. Habal, MD
Copyright © 2013 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery
& Volume 24, Number 6, November 2013
Facial Paralysis and Mediastinitis
DISCUSSION
FIGURE 3. Abscesses in the left temporoparietal region.
from an infectious diseases clinic. The patient was admitted to the critical care unit. The necrotized tissues were debrided every day until bleeding. A new small necrotized tissue was seen on the right submandibular region and was debrided. After day 18 of the treatment, necrotized tissues were not seen and debridement was stopped. No microorganism had been isolated in wound culture, and histopathologic findings were compatible with NF. Thus, we can say that the patient had a good response to the treatment, so oral intake was begun. However, a serious dyspnea was seen because of aspiration and the oral intake was stopped. On the following days, aspiration pneumonia developed. In our opinion, nerve disorder may be developed because of NF. While continuing the therapy at the critical care unit, Candida species had been isolated in blood culture and Acinetobacter baumannii had been isolated in culture of tracheal aspiration. Then, the patient died on day 29 because of sepsis and multiorgan dysfunction.
PATIENT 3 A 40-year-old woman presented to our department with a painful swelling in the submental and right submandibular regions for 2 days, toothache, dysphagia, common status failure, poor level of nourishment, as well as fever for 5 days. Antibiotherapy was applied by a dentist to the patient for the odontogenic infection. However, she was referred to our clinic because of her augmented symptoms. She had no history of systemic disease or of substance abuse. The examination results revealed diffuse painful swelling of the right submandiblar and submental areas. The intraoral examination results revealed diffuse swelling and erythema, especially of the right sublingual region. Cervical CT scan was obtained. The CT scan showed narrowing of the left hypopharengeal air column, gas formation, as well as abscesses in the right submandibular space and oral basement. The patient was thought to have CNF. First of all, the patient underwent tracheotomy with local anesthesia with sedation, followed by an urgent neck exploration under general anesthesia. After passing platysma, an effluxing dirty-yellow, necrotic, foul-smelling fluid was seen at the space of the fascia. Necrotized tissues were seen at the right submandibular and submental area reaching to the oral basement; the tissues were cleaned, and the cleaning was continued until bleeding with aggressive debridement. Intravenous meropenem (3 1 g/d) was administered, with consultation from an infectious diseases clinic. The patient was admitted to the critical care unit. The necrotized tissues were debrided every day until bleeding. Prevotella species had been isolated in blood culture, and histopathologic findings were compatible with NF. The patient was moved out from the critical care unit on day 3 and began oral nourishment on day 10; then, the patient was discharged asymptomatic.
Necrotizing fasciitis of the head and the neck is rare. The most frequent initiating factors for NF in the head and neck region are dental pathology, parapharyngeal or peritonsillar infections, posttraumatic or iatrogenic skin, mucosal injuries, froncule, fractures of the mandible, trauma caused by firearms, tracheotomy, and radiotherapy. The systemic disease, organ failure, drugs (eg, steroids), malnutrition, underlying malignancy, chronic alcoholism, and suppressed immunity seem to be risk factors in defining the induction, progression, and results of the disease.4,7 One of the 5 patients with CNF (including the 2 patients who were previously presented) had diabetes mellitus, but the other 4 patients were healthy and had no risk factors.7 Three of the 5 patients had an odontogenic infection, and the other one had a peritonsillar infection. Thus, we can say that peritonsillar infection and dental pathologies (especially with poor dental health) have a very important role for the development of NF. In addition, NF seems to be rising among healthy people. This condition may be related to the changed environmental conditions, misuse of antibiotics, and/or living conditions. Symptoms of NF may be nonspecific at onset and are dependent on the stage of the disease and its origin site and early stages; NF can be misdiagnosed such as cellulitis or erysipials.8 As the infection progresses, the skin becomes increasingly erythematous, tense, and smooth without prominent margins compared with normal skin. As so in our patients, reliable indicators of NF can be fistule and effluxing dirty-yellow, necrotic, foul-smelling fluid at the oral mucosa, the subcutaneous crepitus, as well as pain and common status failure that do not correlate to the local skin findings. Paralysis of the cranial nerve has been reported in the literature as an encountered complication of NF. The cranial nerve paralysis 9,10,12 could be seen in the progression of the NF.5,7,9 The patient, who was previously presented to our clinic, had paralysis of the right vocal cord.7 From a distance, one of the patients (patient 2) had no history of facial paralysis and aspiration with oral intake; however, he was found to have left facial paralysis (compatible with Hausse-Breckmann type 4), and this was thought to be caused by NF. To the best of our knowledge, a patient with facial paralysis, NF of the temporal region, and DNM has not been documented in the literature, and this condition can be related to the spreading of the
FIGURE 4. The view of the neck after debridement of the necrotic tissues.
* 2013 Mutaz B. Habal, MD
Copyright © 2013 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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disease. After recovery, oral intake caused aspiration and dyspnea to the same patient. This condition can be related to the involvement of the superior laryngeal nerve. Hence, we can say that the oral intake after recovery is very important for patients with cervical NF and DNM. The most important issue is the diagnosis of NF. Delay in diagnosis causes poor prognosis. The criterion standard is intravenous contrast CT scan that helps in early diagnosis and shows soft tissue gas formation. A CT scan distinguishes between abscesses, erysipelas, or cellulitis, better describes the characteristics of the lesions, and can give a hint on monitoring of the disease, recurrence of purulence, and new gas formation.7,10 In addition, it can help to establish the need, extent, and boundary of surgery. Air density between the neck fascias, the mediastinum, and the temporal region was shown in the CT scan of our patients. This situation is pathognomonic for NF. We prefer CT scan for all patients who were admitted to our clinic with deep infections in the neck. Also, in our patients, accurate and rapid diagnosis, nutritional support whether enteral or parenteral, effective intravenous broadspectrum antibiotherapy with anaerobic coverage, rapid control of the upper airway with tracheotomy, as well as early and vigorous surgical debridement of the cervical spaces and/or the mediastinum until the disappearance of the physical and radiologic findings of the necrosis and the abscess with intensive medical care were keys to successful outcome in treatment of NF.2,8 Immediate and well-balanced surgical debridement between infected and healthy tissues with wide extensive fasciotomies with exposure of all involved fascia and excision of necrotic tissues until exposure of viable bleeding tissue are crucial for patient survival.11 This should be done early not only to control the primary infectious process but also to remove the necrotic tissue that is the source of secondary infection and that of toxin production.4 Also, early surgical interference would restore adequate tissue perfusion with appropriate antibiotics diffusion, minimize loss of tissue, and prevent cosmetic disfigurement. Dermal necrosis was not seen in our patients with successful management of therapy. When the wound is opened, it should be well irrigated with betadine and hydrogen peroxide solution, and this regimen should be repeated periodically until further tissue necrosis stops and growth of the fresh viable tissue is observed.8 We used wound dressings soaked in nitrofurazone soluble ointment for daily dressings. Multiple operative sessions are frequently required for full surgical control. Descending necrotizing mediastinitis was seen in 3 of the 5 patients who had primary odontogenic infection. Because an extensive fascial communication exists between the gums, the neck, and the mediastinum, odontogenic infections can rapidly spread to the mediastinum facilitated by gravity, respiration, and negative intrathoracic pressure.12 To our knowledge, from the neck to the mediastinum, there exist 3 potential major pathways for the infection to spread along the fascial spaces in DNM. The retropharyngeal space (danger space) accounts for 70% of cases of DNM. It lies behind the esophagus (the visceral layer of the deep cervical fascia) and anterior to the prevertebral fascia, extending from the base of the skull to the posterior mediastinum.13 The development of DNM leads to fatality complication. Descending necrotizing mediastinitis has a mortality rate that is 4 times greater than that of cervical involvement only.14 The involvement of the posterior mediastinum was seen in our 2 patients with DNM. They died because of sepsis and multiorgan dysfunction, despite recovery of the neck, the temporal region, and the mediastinum of patient 2. Necrotizing fasciitis, especially of odontogenic origin, is almost always polymicrobial and often involves a combination of aerobic and anaerobic bacterias. Because of synergistic work, polymicrobial infections are more pathogenic than monoinfections are.6,15 We preferred to use the broad-spectrum antibiotherapy with anaerobic
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coverage. Except one, in our patients, no microorganism had been isolated in wound cultures. This situation can be related to antibiotherapy by local physicians before hospitalization. Other adjunctive approaches to treatment include hyperbaric oxygen therapy and intravenous immunoglobulin.6 Hyperbaric oxygen treatment can be begun if the patient is stable and can tolerate it.7 The 2 patients had a bad clinical state and the other 3 patient had a good response to the treatment, so we did not use hyperbaric oxygen treatment. As a conclusion, necrotizing fasciitis can expand within hours and the reported mortality rate is up to 75% with delay interference.16 Paralysis of the cranial nerve, such as the facial nerve and the vagal nerve branches, were seen in the progression of NF. Necrotizing fasciitis can be seen in healthy participants with increased ratio, and it should always be borne in mind when faced with deep infections of the neck.
REFERENCES 1. Wilson B. Necrotizing fasciitis. Am Surg 1952;18:416Y431 2. Lopez-Fernandez R, Ramirez-Melgoza J, Martinez-Aguilar NE, et al. Growth factor-enriched autologous plasma improves wound healing after surgical debridement in odontogenic necrotizing fasciitis: a case report. J Med Case Rep 2011;5:98 3. Yadav S, Verma A, Sachdeva A. Facial necrotizing fasciitis from an odontogenic infection. Oral Surg Oral Med Oral Pathol Oral Radiol 2012;113:e1Ye4 4. Bono G, Argo A, Zerbo S, et al. Cervical necrotizing fasciitis and descending necrotizing mediastinitis in a patient affected by neglected peritonsillar abscess: a case of medical negligence. J Forensic Leg Med 2008;15:391Y394 5. Lee JH, Choi HC, Kim C, et al. Fulminanst cerebral infarction of anterior and posterior cerebral circulation after ascending type of facial necrotizing fasciitis. J Stroke Cerebrovasc Dis [published ahead of print August 29, 2012] doi: 10.1016/j.jstrokecerebrovasdis.2012.07.013 6. Park E, Hirsch EM, Steinberg JP, et al. Ascending necrotizing fasciitis of the face following odontogenic infection. J Craniofac Surg 2012;23:e211Ye214 7. Ulu S, Ulu SM, Oz G, et al. Paralysis of cranial nerve and striking prognosis of cervical necrotizing fasciitis. J Craniofac Surg 2012;23:1812Y1814 8. Sasindran V, Joseph A. Necrotizing fasciitis: an unusual presentation. Indian J Otolaryngol Head Neck Surg 2011;63:390Y392 9. Lorenzini G, Picciotti M, Di Vece L, et al. Cervical necrotizing fasciitis of odontogenic origin involving the temporal regionVa case report. Int J Oral Maxillofac Surg 2010;39:830Y834 10. Santos Gorjo´n P, Blanco Pe´rez P, Morales Martı´n AC, et al. Deep neck infection. Review of 286 cases. Acta Otorrinolaringol Esp 2012;63:31Y41 11. Thomas AJ, Mong S, Golub JS, et al. Klebsiella pneumoniae cervical necrotizing fasciitis originating as an abscess. Am J Otolaryngol 2012;33:764Y766 12. Gonza´lez-Garcı´a R, Risco-Rojas R, Roma´n-Romero L, et al. Descending necrotizing mediastinitis following dental extraction. Radiological features and surgical treatment considerations. J Craniomaxillofac Surg 2011;39:335Y339 13. Sakamoto H, Aoki T, Kise Y, et al. Descending necrotizing mediastinitis due to odontogenic infections. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;89:412Y419 14. Weed HG, Forest LA. Deep neck infections. In: Cummings CW, Flint PW, Harker LA, Haughey BH, Richardson MA, Robbins KT, Schuller DE, Thomas JR, eds. Cummings Otolaryngology Head & Neck Surgery. Philadelphia, PA: 2005:2521Y2522 15. Boffano P, Roccia F, Pittoni D, et al. Management of 112 hospitalized patients with spreading odontogenic infections: correlation with DMFT and oral health impact profile 14 indexes. Oral Surg Oral Med Oral Pathol Oral Radiol 2012;113:207Y213 16. Danic Hadzibegovic A, Sauerborn D, Grabovac S, et al. Necrotizing fasciitis of the neck after total laryngectomy. Eur Arch Otorhinolaryngol 2013;270:277Y280
* 2013 Mutaz B. Habal, MD
Copyright © 2013 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
