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Stomatococcus mucilaginosis infection leading to early cervical necrotizing fasciitis
Stomatococcus mucilaginosis infection leading to early cervical necrotizing fasciitis THOMAS R. LOWRY,
MD,
and JOSEPH A. BRENNAN,
MD, FACS,
C ervical necrotizing fasciitis is characterized by aggressive infection with connective tissue necrosis, spread along fascia planes, and frequent mortality. It is usually caused by a polymicrobial or mixed aerobicanaerobic infection, with the very early stages resembling an abscess or cellulitis.1 Although predominately a disease of the elderly and immunocompromised, it recently has been observed increasingly in young, otherwise healthy individuals with intact immunologic response and without recent surgery or trauma.2 We describe a unique case of early cervical necrotizing fasciitis in a young healthy male arising from a parotid cyst infected with Stomatococcus mucilaginosis, an infrequent opportunistic microorganism that normally resides in the oral cavity. CASE REPORT A 24-year-old Caucasian male emergency department technician presented with a 2-day history of right neck pain and swelling. The patient had no preceding upper respiratory illness, no dental carries or recent dental work, and no history of trauma. His past medical history was unremarkable. Physical exam was noteworthy for a tender, fluctuant mass over the tail of the right parotid gland with ipsilateral lymphadenopathy. No erythema of the overlying skin was noted. Laboratory evaluation including white blood cell count and HIV was normal. CT with contrast of the neck was obtained, which showed a 2- ⫻ 3-cm cystic mass with air in the tail of the right parotid gland and inflammation with obscured fat planes extending inferiorly into the parapharyngeal space. A needle aspiration of the lesion obtained 2 mL of bloody serous fluid, which was sent for culture. The patient was placed on intravenous clindamycin and admitted for observation. The following day the patient complained of worsening neck pain and was febrile at 38.5° C. A welldemarcated line of cervical erythema developed and an infectious disease consultation was made. Intravenous From Wright-Patterson AFB, OH, (Dr Lowry) and the Department of Otolaryngology–Head and Neck Surgery, Wilford Hall Medical Center, Lackland AFB, TX (Dr Brennan). EBM rating: C. Reprint requests: Thomas R. Lowry, MD, 74th MSGS/SGCXL, 4881 Sugar Maple Drive, Wright-Patterson AFB, OH 45433-5529; e-mail, [email protected]. Otolaryngol Head Neck Surg 2005;132:658-660. doi:10.1016/j.otohns.2004.05.016
658
Wright-Patterson AFB, Ohio, and Lackland AFB, Texas
ampicillin/sulbactam was added to the treatment regimen for broader antimicrobial coverage. A repeat CT of the neck demonstrated continued presence of a fluidfilled cavity within the right parotid gland. The patient was taken to the operating room for open incision and drainage of the lesion. The cystic cavity was identified within the inflamed parotid parenchyma, and 5 mL of brown serous fluid was obtained and sent for repeat cultures. The wound was closed with a Penrose drain in place. The next day the patient continued to have neck pain, increased swelling, and progression of the line of surrounding erythema extending down to the base of the neck (Fig 1). Wound cultures grew out Stomatococcus mucilaginosis. Concern over the possibility of early necrotizing fasciitis prompted urgent cervical fasciotomy in the operating room. A combined preauricular and apron incision exposed the neck. The cystic cavity within the parotid gland was re-opened, and diffuse inflammation and edema of the cervical soft tissues was present throughout the dissection. Both the superficial cervical and sternocleidomastoid muscle fascias were widely incised, and biopsies sent for pathologic evaluation demonstrated extensive acute and chronic inflammatory changes. The wound was subsequently closed about several Penrose drains. Postoperatively the patient was placed on intravenous vancomycin and clindamycin. He subsequently showed rapid improvement in his symptoms and was discharged home 3 days later. No recurrences of the patient’s symptoms or parotid cyst developed over the ensuing months, and repeat blood tests including complete blood count and HIV remained normal. DISCUSSION Necrotizing fasciitis was first recognized as a clinical entity in 1871, although the term was not coined until 1952 by Wilson, who described a necrotizing soft tissue infection in which the fascia is invariably involved.1,3 More recently, cervical necrotizing fasciitis has been described as a distinct clinical entity, with involvement of 1 or multiple fascia planes of the neck being characteristic. It occurs predominantly in immunocompromised or elderly patients, yet our case demonstrates that young healthy patients may also be affected. Sources of infection are usually odontogenic, with lower molars being the most common teeth of origin. Other etiologies noted in the literature include
Otolaryngology– Head and Neck Surgery Volume 132 Number 4
Fig 1. Clinical photograph demonstrating progressive infection extending inferiorly along anterior neck and chest.
infections of the tonsils, pharynx, and salivary glands as well as cervical lymphadenitis and trauma.1-3 The polymicrobial nature of the infection is important in that a synergism between different types of bacteria is thought to play a key role in its aggressive nature.1,3 In a recent review of 11 cases of cervical necrotizing fasciitis by Tung-Yiu,1 anaerobes were isolated in 73% of infections, with Streptococcus species being uniformly present. Other case reports and series note similar findings of mixed aerobic-anaerobic infections. Of particular interest to our case was the finding of an infrequently isolated opportunistic pathogen, Stomatococcus mucilaginosis. This gram-positive cocci is an important part of the oral flora and was first described as a human pathogen by Rubin and colleagues in 1978.4 It is a slime-producing, catalase-variable, oxidase-negative, nonhemolytic-encapsulated organism that forms gray mucoid colonies. The organism does not grow on 5% sodium chloride agar. Subsequent sparse reports of infections due to S. mucilaginosis have been seen predominantly in immunocompromised patients with serious underlying disease, neutropenia, presence of a foreign body, cardiac valve disease, or intravenous drug use.5 Infection often occurs in previously damaged tissues, with destruction of oral mucous membranes playing a possible role in the dissemination from the oral cavity.5 Our patient demonstrated no immunodeficiency and had good oral hygiene, however, the organism may have entered the parotid tissue through an unseen mucosal injury or an obstructed parotid duct. To our knowledge, this organism has never previously been implicated in salivary gland infections or the development of cervical necrotizing fasciitis. Necrotizing fasciitis is classically described in the acutely ill patient with fever. Tender, erythematous and
LOWRY and BRENNAN 659
edematous overlying skin with occasional bulla formation and soft tissue crepitence is typical. However, the clinical distinction from more benign inflammatory conditions of the neck, such as cellulitis, may be impossible at an early stage.2 Certain findings may be present that should make one suspicious of cervical necrotizing fasciitis. These include odontogenic infections that spread to the lower neck or anterior chest, abnormal accumulation of gas in the tissues, very rapid progression of the infection, and an orange-peel appearance of the involved skin.1 Our patient demonstrated rapid progression of his infection despite initial antibiotic and surgical intervention along with spread to the lower neck and chest. The complications frequently encountered with cervical necrotizing fasciitis account for its high mortality. Pneumonia, pleural effusion, mediastinitis, pericardial involvement, and disseminated intravascular coagulation leading to sepsis and shock are all potentially life-threatening sequelae. In their review of the literature, Tung-Yiu noted that the mean mortality rate of necrotizing fasciitis between 1924 and 1994 was 34% and ranged from 6% to 76%.1 The frequent development of mediastinitis is thought to contribute substantially to this high number. CT with contrast is invaluable in the diagnostic workup of patients thought to have cervical necrotizing fasciitis. Becker and colleagues2 describe 7 main diagnostic features found on neck CT. These include diffuse thickening of the cutis and subcutis and reticular enhancement of the subcutaneous fat of the face and neck (cellulitis), thickening and/or enhancement of cervical fascia (fasciitis), asymmetric thickening or enhancement of cervical muscles (myositis), and fluid collections in multiple neck spaces. Inconsistent features included gas collections in the neck, involvement of the mediastinum, and other features attributable to the inflammatory process.2 Our patient demonstrated CT findings of a fluid-filled cavity with air in the right parotid gland, thickening of the involved cervical fascia, and reticular changes with inflammation in the adjacent descending parapharyngeal fat compartments (Figs 2 and 3). The treatment for necrotizing fasciitis has seen little change over the years. Early surgical debridement of the involved cervical fascia compartments is the cornerstone of therapy, with empiric broad-spectrum antibiotics employed to cover the usual polymicrobial pathogens. Common antibiotic regimens include thirdgeneration cephalosporins combined with clindamycin or metronidazole. Surgical findings consist of extensive soft tissue necrosis with fat liquefaction, whereas histology generally shows marked tissue necrosis and foci of polymorphonuclear and mononuclear cells admixed with bacterial colonies.1 The role of adjunctive hyper-
Otolaryngology– Head and Neck Surgery April 2005
660 LOWRY and BRENNAN
Fig 2. Axial CT at level of right parotid gland showing fluid-filled collection and diffuse inflammation.
baric oxygen therapy to treat necrotizing fasciitis has been noted to decrease mortality in various studies and is advocated by some authors.3 Although the clinical diagnosis of necrotizing fasciitis is straightforward at an advanced stage, it may be very difficult to differentiate between cellulitis and necrotizing fasciitis at initial presentation. The distinction is crucial, however, because cellulitis responds to antibiotic treatment alone, whereas survival of patients with necrotizing fasciitis depends on early extensive surgical drainage and debridement in addition to antibiotic treatment, fluid management, and blood pressure support with vasopressors.2 The initial presentation of our patient was consistent with early abscess formation and cellulitis, but his poor response to antibiotics and rapid spread of infection led to our heightened concern for early necrotizing fasciitis. Additionally, Stomatococcus mucilaginosis is universally susceptible to vancomycin,5 and it was not until this antibiotic was added along with wide surgical debridement that our patient began to clinically improve. The high mortality associated with this disease process should make any patient with a progressive soft tissue cervical infection,
Fig 3. Axial CT at level of right parapharyngeal space demonstrating soft tissue reticular changes with obliteration of fat planes.
despite broad-spectrum antibiotic therapy, suspect for cervical necrotizing fasciitis. Early aggressive surgical intervention should be considered in these cases even when the classic signs of necrotizing fasciitis are not present. REFERENCES 1. Tung-Yiu W, Jehn-Shyun H, Ching-Hung C, et al. Cervical necrotizing fasciitis of odontogenic origin: a report of 11 cases. J Oral Maxillofac Surg 2000;58:1347-52. 2. Becker M, Zbaren P, Hermans R, et al. Necrotizing fasciitis of the head and neck: role of CT in diagnosis and management. Radiology 1997;202:471-6. 3. Kantu S, Har-El G. Cervical necrotizing fasciitis. Ann Otol Rhinol Laryngol 1997;106:965-70. 4. Rubin SJ, Lyons RW, Murcia AJ. Endocarditis associated with cardiac catheterization due to a gram-positive coccus designated Micrococcus mucilaginosus incertae sedis. J Clin Microbiol 1978; 7:546-8. 5. Ruoff KL. Leuconostoc, Pediococcus, Stomatococcus, and miscellaneous gram-positive cocci that grow aerobically. In: Murray PR, Baron EJ, Pfaller MA, et al., editors. Manual of clinical microbiology, 7th ed. Washington, DC: American Society for Microbiology; 1999. pp. 306-15.
MD,
and JOSEPH A. BRENNAN,
MD, FACS,
C ervical necrotizing fasciitis is characterized by aggressive infection with connective tissue necrosis, spread along fascia planes, and frequent mortality. It is usually caused by a polymicrobial or mixed aerobicanaerobic infection, with the very early stages resembling an abscess or cellulitis.1 Although predominately a disease of the elderly and immunocompromised, it recently has been observed increasingly in young, otherwise healthy individuals with intact immunologic response and without recent surgery or trauma.2 We describe a unique case of early cervical necrotizing fasciitis in a young healthy male arising from a parotid cyst infected with Stomatococcus mucilaginosis, an infrequent opportunistic microorganism that normally resides in the oral cavity. CASE REPORT A 24-year-old Caucasian male emergency department technician presented with a 2-day history of right neck pain and swelling. The patient had no preceding upper respiratory illness, no dental carries or recent dental work, and no history of trauma. His past medical history was unremarkable. Physical exam was noteworthy for a tender, fluctuant mass over the tail of the right parotid gland with ipsilateral lymphadenopathy. No erythema of the overlying skin was noted. Laboratory evaluation including white blood cell count and HIV was normal. CT with contrast of the neck was obtained, which showed a 2- ⫻ 3-cm cystic mass with air in the tail of the right parotid gland and inflammation with obscured fat planes extending inferiorly into the parapharyngeal space. A needle aspiration of the lesion obtained 2 mL of bloody serous fluid, which was sent for culture. The patient was placed on intravenous clindamycin and admitted for observation. The following day the patient complained of worsening neck pain and was febrile at 38.5° C. A welldemarcated line of cervical erythema developed and an infectious disease consultation was made. Intravenous From Wright-Patterson AFB, OH, (Dr Lowry) and the Department of Otolaryngology–Head and Neck Surgery, Wilford Hall Medical Center, Lackland AFB, TX (Dr Brennan). EBM rating: C. Reprint requests: Thomas R. Lowry, MD, 74th MSGS/SGCXL, 4881 Sugar Maple Drive, Wright-Patterson AFB, OH 45433-5529; e-mail, [email protected]. Otolaryngol Head Neck Surg 2005;132:658-660. doi:10.1016/j.otohns.2004.05.016
658
Wright-Patterson AFB, Ohio, and Lackland AFB, Texas
ampicillin/sulbactam was added to the treatment regimen for broader antimicrobial coverage. A repeat CT of the neck demonstrated continued presence of a fluidfilled cavity within the right parotid gland. The patient was taken to the operating room for open incision and drainage of the lesion. The cystic cavity was identified within the inflamed parotid parenchyma, and 5 mL of brown serous fluid was obtained and sent for repeat cultures. The wound was closed with a Penrose drain in place. The next day the patient continued to have neck pain, increased swelling, and progression of the line of surrounding erythema extending down to the base of the neck (Fig 1). Wound cultures grew out Stomatococcus mucilaginosis. Concern over the possibility of early necrotizing fasciitis prompted urgent cervical fasciotomy in the operating room. A combined preauricular and apron incision exposed the neck. The cystic cavity within the parotid gland was re-opened, and diffuse inflammation and edema of the cervical soft tissues was present throughout the dissection. Both the superficial cervical and sternocleidomastoid muscle fascias were widely incised, and biopsies sent for pathologic evaluation demonstrated extensive acute and chronic inflammatory changes. The wound was subsequently closed about several Penrose drains. Postoperatively the patient was placed on intravenous vancomycin and clindamycin. He subsequently showed rapid improvement in his symptoms and was discharged home 3 days later. No recurrences of the patient’s symptoms or parotid cyst developed over the ensuing months, and repeat blood tests including complete blood count and HIV remained normal. DISCUSSION Necrotizing fasciitis was first recognized as a clinical entity in 1871, although the term was not coined until 1952 by Wilson, who described a necrotizing soft tissue infection in which the fascia is invariably involved.1,3 More recently, cervical necrotizing fasciitis has been described as a distinct clinical entity, with involvement of 1 or multiple fascia planes of the neck being characteristic. It occurs predominantly in immunocompromised or elderly patients, yet our case demonstrates that young healthy patients may also be affected. Sources of infection are usually odontogenic, with lower molars being the most common teeth of origin. Other etiologies noted in the literature include
Otolaryngology– Head and Neck Surgery Volume 132 Number 4
Fig 1. Clinical photograph demonstrating progressive infection extending inferiorly along anterior neck and chest.
infections of the tonsils, pharynx, and salivary glands as well as cervical lymphadenitis and trauma.1-3 The polymicrobial nature of the infection is important in that a synergism between different types of bacteria is thought to play a key role in its aggressive nature.1,3 In a recent review of 11 cases of cervical necrotizing fasciitis by Tung-Yiu,1 anaerobes were isolated in 73% of infections, with Streptococcus species being uniformly present. Other case reports and series note similar findings of mixed aerobic-anaerobic infections. Of particular interest to our case was the finding of an infrequently isolated opportunistic pathogen, Stomatococcus mucilaginosis. This gram-positive cocci is an important part of the oral flora and was first described as a human pathogen by Rubin and colleagues in 1978.4 It is a slime-producing, catalase-variable, oxidase-negative, nonhemolytic-encapsulated organism that forms gray mucoid colonies. The organism does not grow on 5% sodium chloride agar. Subsequent sparse reports of infections due to S. mucilaginosis have been seen predominantly in immunocompromised patients with serious underlying disease, neutropenia, presence of a foreign body, cardiac valve disease, or intravenous drug use.5 Infection often occurs in previously damaged tissues, with destruction of oral mucous membranes playing a possible role in the dissemination from the oral cavity.5 Our patient demonstrated no immunodeficiency and had good oral hygiene, however, the organism may have entered the parotid tissue through an unseen mucosal injury or an obstructed parotid duct. To our knowledge, this organism has never previously been implicated in salivary gland infections or the development of cervical necrotizing fasciitis. Necrotizing fasciitis is classically described in the acutely ill patient with fever. Tender, erythematous and
LOWRY and BRENNAN 659
edematous overlying skin with occasional bulla formation and soft tissue crepitence is typical. However, the clinical distinction from more benign inflammatory conditions of the neck, such as cellulitis, may be impossible at an early stage.2 Certain findings may be present that should make one suspicious of cervical necrotizing fasciitis. These include odontogenic infections that spread to the lower neck or anterior chest, abnormal accumulation of gas in the tissues, very rapid progression of the infection, and an orange-peel appearance of the involved skin.1 Our patient demonstrated rapid progression of his infection despite initial antibiotic and surgical intervention along with spread to the lower neck and chest. The complications frequently encountered with cervical necrotizing fasciitis account for its high mortality. Pneumonia, pleural effusion, mediastinitis, pericardial involvement, and disseminated intravascular coagulation leading to sepsis and shock are all potentially life-threatening sequelae. In their review of the literature, Tung-Yiu noted that the mean mortality rate of necrotizing fasciitis between 1924 and 1994 was 34% and ranged from 6% to 76%.1 The frequent development of mediastinitis is thought to contribute substantially to this high number. CT with contrast is invaluable in the diagnostic workup of patients thought to have cervical necrotizing fasciitis. Becker and colleagues2 describe 7 main diagnostic features found on neck CT. These include diffuse thickening of the cutis and subcutis and reticular enhancement of the subcutaneous fat of the face and neck (cellulitis), thickening and/or enhancement of cervical fascia (fasciitis), asymmetric thickening or enhancement of cervical muscles (myositis), and fluid collections in multiple neck spaces. Inconsistent features included gas collections in the neck, involvement of the mediastinum, and other features attributable to the inflammatory process.2 Our patient demonstrated CT findings of a fluid-filled cavity with air in the right parotid gland, thickening of the involved cervical fascia, and reticular changes with inflammation in the adjacent descending parapharyngeal fat compartments (Figs 2 and 3). The treatment for necrotizing fasciitis has seen little change over the years. Early surgical debridement of the involved cervical fascia compartments is the cornerstone of therapy, with empiric broad-spectrum antibiotics employed to cover the usual polymicrobial pathogens. Common antibiotic regimens include thirdgeneration cephalosporins combined with clindamycin or metronidazole. Surgical findings consist of extensive soft tissue necrosis with fat liquefaction, whereas histology generally shows marked tissue necrosis and foci of polymorphonuclear and mononuclear cells admixed with bacterial colonies.1 The role of adjunctive hyper-
Otolaryngology– Head and Neck Surgery April 2005
660 LOWRY and BRENNAN
Fig 2. Axial CT at level of right parotid gland showing fluid-filled collection and diffuse inflammation.
baric oxygen therapy to treat necrotizing fasciitis has been noted to decrease mortality in various studies and is advocated by some authors.3 Although the clinical diagnosis of necrotizing fasciitis is straightforward at an advanced stage, it may be very difficult to differentiate between cellulitis and necrotizing fasciitis at initial presentation. The distinction is crucial, however, because cellulitis responds to antibiotic treatment alone, whereas survival of patients with necrotizing fasciitis depends on early extensive surgical drainage and debridement in addition to antibiotic treatment, fluid management, and blood pressure support with vasopressors.2 The initial presentation of our patient was consistent with early abscess formation and cellulitis, but his poor response to antibiotics and rapid spread of infection led to our heightened concern for early necrotizing fasciitis. Additionally, Stomatococcus mucilaginosis is universally susceptible to vancomycin,5 and it was not until this antibiotic was added along with wide surgical debridement that our patient began to clinically improve. The high mortality associated with this disease process should make any patient with a progressive soft tissue cervical infection,
Fig 3. Axial CT at level of right parapharyngeal space demonstrating soft tissue reticular changes with obliteration of fat planes.
despite broad-spectrum antibiotic therapy, suspect for cervical necrotizing fasciitis. Early aggressive surgical intervention should be considered in these cases even when the classic signs of necrotizing fasciitis are not present. REFERENCES 1. Tung-Yiu W, Jehn-Shyun H, Ching-Hung C, et al. Cervical necrotizing fasciitis of odontogenic origin: a report of 11 cases. J Oral Maxillofac Surg 2000;58:1347-52. 2. Becker M, Zbaren P, Hermans R, et al. Necrotizing fasciitis of the head and neck: role of CT in diagnosis and management. Radiology 1997;202:471-6. 3. Kantu S, Har-El G. Cervical necrotizing fasciitis. Ann Otol Rhinol Laryngol 1997;106:965-70. 4. Rubin SJ, Lyons RW, Murcia AJ. Endocarditis associated with cardiac catheterization due to a gram-positive coccus designated Micrococcus mucilaginosus incertae sedis. J Clin Microbiol 1978; 7:546-8. 5. Ruoff KL. Leuconostoc, Pediococcus, Stomatococcus, and miscellaneous gram-positive cocci that grow aerobically. In: Murray PR, Baron EJ, Pfaller MA, et al., editors. Manual of clinical microbiology, 7th ed. Washington, DC: American Society for Microbiology; 1999. pp. 306-15.