International Journal of Pediatric Otorhinolaryngology (2009) 73, 467—473
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CASE REPORT
Serratia marcescens causing cervical necrotizing oropharyngitis Melissa M. Statham a,*, Amit Vohra b,d, Deepak K. Mehta c, Troy Baker d, Robert Sarlay e, Michael J. Rutter a,f a
University of Cincinnati School of Medicine Department of Otolaryngology — Head and Neck Surgery, 231 Albert Sabin Way Cincinnati, OH 45267-0528, United States b The Children’s Medical Center of Dayton Division of Critical Care Medicine, One Children’s Plaza, Dayton, OH 45404, United States c Children’s Hospital of Pittsburgh of UPMC Department of Otolaryngology, 3705 Fifth Avenue Pittsburgh, PA 15213, United States d Boonshoft School of Medicine at Wright State University, Department of Pediatrics, Dayton, OH, United States e Boonshoft School of Medicine at Wright State University, Department of Emergency Medicine, 3525 Southern Blvd, Kettering, OH 45429, United States f Cincinnati Children’s Hospital Medical Center Department of Pediatric Otolaryngology — Head and Neck Surgery, ML #2018, 3333 Burnet Avenue OSB-3 Cincinnati, OH 45229, United States Received 21 July 2008; received in revised form 14 October 2008; accepted 15 October 2008 Available online 10 December 2008
KEYWORDS Necrotizing fasciitis; Necrotizing oropharyngitis; Serratia marcescens; Necrotizing pharyngitis; Necrotizing cervical fasciitis; Serratia marcescens pharyngitis; Serratia marcescens necrotizing fasciitis; Serratia marcescens cervical necrotizing fasciitis; Cervical necrotizing fasciitis
Summary Necrotizing fasciitis is a rare, life-threatening infection. We report a case of necrotizing oropharyngitis caused by Serratia marcescens in a previously immunocompetent 6-year-old male. This necrotizing infection led to a near-total defect of the oropharynx. The wound was managed with daily wound debridement of the patient’s oropharynx with 3% hydrogen peroxide, carotid artery coverage with KaltostatTM, and pharyngeal packing with iodoform ribbon gauze. Our patient’s resultant nasopharyngeal and hypopharyngeal stenoses present challenges for restoration of form and function for voicing and deglutition. We present our experience of managing this child’s hypopharyngeal stenosis with a minimally invasive double-balloon dilatation technique. # 2008 Elsevier Ireland Ltd. All rights reserved.
* Corresponding author. Tel.: +1 513 558 4198. E-mail address:
[email protected] (M.M. Statham). 0165-5876/$ — see front matter # 2008 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2008.10.019
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1. Introduction Necrotizing fasciitis is a rare, life-threatening infection, and its aggressive nature can quickly lead to systemic toxicity with a mortality rate of 30—60%. Many organisms can cause necrotizing fasciitis, of which group A beta-haemolytic streptococci and Staphylococcus aureus are the most common. We report a case of necrotizing oropharyngitis caused by Serratia marcescens in a previously immunocompetent 6-year-old male. S. marcescens is an extremely rare pathogen in oropharyngitis, and this is the first reported pediatric case of S. marcescens causing a necrotizing oropharyngitis. This index case presented to a referring hospital with upper airway obstruction, respiratory failure and septic shock. The patient survived his initial fulminant course of multiorgan dysfunction. However, the necrotizing infection caused a near-total defect of the oropharynx and great vessel exposure in the oropharynx rendering wound debridement particularly challenging and potentially life-threatening. The patient was transferred to a tertiary children’s hospital facility for our special otolaryngologic expertise. We present our experience of managing this child’s subsequent hypopharyngeal stenosis with a minimally invasive double-balloon dilatation technique.
2. Case 2.1. Medical management (at referring hospital) A previously healthy 6-year-old male presented with 5 days of odynophagia and fever. The patient had a 4-month antedating history of recurrent otitis media and pharyngitis for which he had been treated with multiple courses of antibiotics. Prior to admission, the patient was initially evaluated at an urgent care clinic and was empirically treated with Amoxicillin for presumptive streptococcal pharyngitis. The patient’s odynophagia continued to worsen over the next 3 days and he was then treated with clindamycin and corticosteroids. Progressive hoarseness and neck swelling developed, followed by the emergence of respiratory distress and stridor on the day of admission. He presented to the emergency department with nasal flaring, retractions, and intermittent inspiratory stridor. The patient had erythematous, edematous, and severely enlarged tonsils. Contrastenhanced computed tomography (CT) of the neck showed right peritonsillar phlegmom and slight deviation of the trachea (Fig. 1). Complete blood count revealed pancytopenia, with leukopenia of
Fig. 1 Initial presentation axial image CT of neck with intravenous contrast demonstrates parapharyngeal phlegmon.
300 mm 3, agranulocytosis, and anemia with hemoglobin of 7.5 g/dL. He was electively orotracheally intubated for airway management. On arrival to the pediatric intensive care unit (PICU), the patient was unstable and required mechanical ventilation, treatment for septic shock and broad-spectrum intravenous antibiotics including clindamycin, vancomycin, and cefepime. Bone marrow biopsy revealed myeloproliferative fibrosis, which was thought to be secondary to infection or possible leukemia/lymphoma, yet flow cytometry failed to demonstrate a monoclonal cell population. Blood and respiratory cultures, obtained on admission, grew Serratia marcescens. Antibiotics were changed to gentamicin and piperacillin/tazobactam. The patient required multiple red cell and platelet transfusions and also developed significant renal failure which necessitated continuous venovenous hemodiafiltration (CVVHDF). Despite aggressive medical therapy, the patient failed to demonstrate appropriate clinical improvement. His neck progressively became more swollen, erythematous, and indurated. Contrast-enhanced magnetic resonance imaging (MRI) of the neck demonstrated a retropharyngeal abscess (Fig. 2). The patient was taken for incision and drainage, but no purulence was encountered. A necrotic right tonsil was removed along with necrotic posterior soft palate. No odontogenic pathology was noted. Tissue cultures of the tonsil grew Serratia marcescens with a similar susceptibility profile as the initial blood culture. Viral cultures of the tonsil revealed herpes simplex virus. Culture-tailored antibiotics were changed to cefoxitin and vancomycin.
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Fig. 2 (a and b) Axial T1 fat-suppressed images with gadolinium demonstrates a rim-enhancing parapharyngeal fluid collection and a contralateral necrotic lymph node.
Repeat bone marrow biopsy on hospital day 10 demonstrated rejuvenating marrow with no evidence of malignancy. Complete blood count revealed resolved pancytopenia and recovery of neutrophil counts. The patient was extubated on hospital day 11, only to be reintubated for respiratory distress several hours later. On repeat exam in the operating room (OR), he was found to have necrotic tissue involving almost all of the posterior and lateral oropharynx, soft palate, and hypopharynx. On hospital day 14, the patient underwent elective tracheostomy for respiratory failure. Once medically stable from his multi-system organ dysfunction, the patient was transferred to a pediatric hospital with special expertise in otolaryngologic management.
gauze. In order to tolerate the oropharynx and hypopharynx being packed with iodoform dressing, the patient was maintained under moderate sedation and mechanical ventilation in the PICU. Treatment with hyperbaric oxygen was entertained, but the patient was too medically infirm to tolerate this therapy. The patient returned to the OR on a daily basis for 2 weeks and underwent wound debridement, irrigation with 3% hydrogen peroxide, KaltostatTM and iodoform packing replacement. He was maintained on carotid-blowout precautions for the first week of daily debridements with multiple units of packed red blood cells available for the OR. Over this course of daily debridements, the oropharynx and hypopharynx developed granulation tissue and began
2.2. Necrotizing oropharyngitis–—surgical management (at tertiary hospital) Shortly after hospital transfer, the patient returned to the OR for an airway evaluation, and he was found to have extensive necrosis of his oropharynx, involving the entire soft palate, circumferential pharyngeal constrictor erosion to the prevertebral fascia and the parapharyngeal space, which exposed both carotid arteries to infected, necrotic tissue and oropharyngeal secretions (Fig. 3). The hypopharynx was extensively necrotic as well, but the cervical esophagus was spared. The supraglottic and infraglottic structures were grossly normal other than moderate epiglottic and bilateral aryepiglottic fold edema. After extensive debridement of this necrotic tissue, the wound was irrigated with 3% hydrogen peroxide, the carotid arteries covered with KaltostatTM and the pharynx packed with iodoform ribbon
Fig. 3 Operative photograph of necrotic oropharynx from initial debridement at tertiary hospital. A nasogastric feeding tube traverses the wound (solid arrow), and the suction is resting on the prevertebral fascia. Empty arrow delineates remnant of soft palate.
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Fig. 4 Operative photograph of oropharynx after 2 weeks of daily debridements. Note the granulation tissue and mucosalization in this interval. Arrow points to remnant of soft palate.
mucosalization (Fig. 4). To overcome the loss of his soft palate, a soft palate obturator was fashioned to assist with communication and deglutition. The patient was successfully weaned from mechanical ventilation. In addition, a gastrostomy tube was placed for enteric nutrition. Though medically debilitated, the patient was able to undergo extensive in-patient physical rehabilitation. After a 6-week hospital stay at Cincinnati Children’s Hospital, the patient was discharged being able to tolerate capping of his tracheostomy tube while awake, and he was able to speak with a passy-muir valve with significant velopharyngeal insufficiency.
2.3. Functional restoration As an outpatient, the patient underwent serial airway evaluations, and as his wound healed, oro- and
M.M. Statham et al. hypopharyngeal cicatricial scar formed (Fig. 4). This evolved to include the nasopharynx, leading to nasopharyngeal stenosis. Hypopharyngeal stenosis was managed with scar lysis and hypopharyngeal dilations. Hypopharyngeal scar bands were divided with PlasmaCision (Gyrus ENT, Bartlett, TN) cautery. As there was only fibrotic tissue covering the great vessels within the pharynx, PlasmaCision was chosen because the plasma field is of lower temperature than conventional cautery (Fig. 5). Dilatation was performed via a minimally invasive technique utilizing balloon dilatation catheters (BlueMax; Boston Scientific Corp., Watertown, MA) inflated with saline, with the inflation pressure being the manufacturer-rated burst pressure of the balloon [9]. Two 5.0 endotracheal tubes were introduced into the nasal cavities and advanced to the hypopharynx. A 10-mm balloon dilator and a 12-mm were then guided, one through each of the endotracheal tubes to the hypopharynx. Both balloons were then inflated in a side-by-side fashion and remained in place for 2 min at a pressure of 8 atm. The doubleballoon dilation allowed more physiologic elliptical expansion of the hypopharynx, at higher pressures than could be achieved with dilation via a single balloon. Subsequent dilation, performed 2 weeks later, was done to a diameter of 28 mm, using two 14 mm balloons, each inflated to 8 atm of pressure. As an adjunct to scar inhibition, 5 mg of triamcinolone was injected into the dilated scar band. Serial dilations and scar band divisions were performed every 2—3 weeks for three operative sessions. This was extended to monthly intervals for three further dilations, and the patient’s hypopharynx maintained patency thereafter (Fig. 6). The patient was successfully decannulated 9 months after his initial admission. To date, no further surgical manipulation of his aerodigestive tract has been required.
Fig. 5 (a) Hypopharyngeal stenosis at 3 months from initial presentation. Arrow points to the suprahyoid epiglottis. (b) Photo of hypopharyngeal stenosis after balloon dilation and PlasmaCision (Gyrus ENT, Bartlett, TN) cautery. Solid arrow points to suprahyoid epiglottis, open arrow to lysed scar band.
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Fig. 6 Resultant hypopharyngeal stenosis 20 months after initial presentation. Solid arrow points to suprahyoid epiglottis, star delineates hypopharyngeal scar, open arrow to left arytenoid cartilage.
Outpatient speech therapy allowed the patient to return to consuming a regular diet with no evidence of aspiration. In addition, his acquired nasopharyngeal stenosis evolved such that it functioned to effectively treat his velopharyngeal insufficiency, and at last follow-up evaluation, was 5 mm 8 mm. mm. The patient is currently functioning well with good deglutition and excellent voicing with only mild velopharyngeal insufficiency.
3. Discussion Necrotizing fasciitis (NF) is a progressive infection of soft tissues which spreads along fascial planes. Treatment typically consists of early recognition, prompt treatment with antibiotics, surgical debridement of devitalized tissue, and meticulous supportive care. Broad spectrum antibiotic coverage must include anaerobic and aerobic organisms [1]. NF results from the spread of infection along fascial planes producing thrombosis of vessels, resulting in necrosis of the overlying tissues [2]. NF often follows a fulminant course resulting in mortality around 30% of affected patients [3]. NF is rare in childhood and most often caused by group A beta-haemolytic streptococci and Staphylococcus aureus. Septic shock is a common sequelae of NF. Few reports exist of NF secondary to pharyngeal infections, and many of these patients were adults with medical co-morbidities associated with compromised immune function [15]. The isolation of S. marcescens in the blood cultures and tissue cultures indicates a causative role in this patient. Severe sepsis and resultant transient
471 pancytopenia did occur in our patient but fortunately responded to medical treatment at the referring hospital. The stabilization of multi-system organ dysfunction in the patient was not accompanied by a commensurate improvement in the oropharyngeal and parapharyngeal infection. Initial removal of necrotic tonsillar and soft palate tissue and debridement, as well as recovery of the patient’s transient pancytopenia, did not arrest or reverse the necrotic process in the oropharynx and necessitated transfer to the regional center with otolaryngologic expertise. Hyperbaric oxygen therapy has been described as an adjunct to the standard medical and surgical management of NF. However, no objective evidence for its use has been shown in randomized controlled trials [4], and our patient did not receive this therapy. S. marcescens is a gram-negative, facultative anaerobe that is transmitted via direct contact as respiratory droplets. It has been associated with bacteremia, pneumonia, meningitis, endocarditis, and septic arthritis, and infection is usually found in chronically ill or immuncompromised patients [5]. S. marcescens is a rare cause of community-acquired soft tissue infection with risk factors including: previous trauma, chronic extremity ulceration, diabetes mellitus, and chronic renal failure [6]. Five previous cases of NF, caused by S. marcescens, have been reported. Four cases involved adults with lower extremity NF [1,5—7]. One reported pediatric case involved an immunocompetent patient who developed cervical NF and concomitant pancytopenia and sepsis, but unfortunately, this patient rapidly succumbed to the infection [8]. As in the case of our patient, an extensive necrotic wound in the oro- and hypopharynx poses therapeutic challenges for debridement and wound care. Management of the wound in the acute setting requires vigilant local wound care. Serial debridements with irrigation of 3% hydrogen peroxide was an effective means of removing necrotic tissue without need for external incisions, which would have lead to a pharyngocutaneous fistula in this pediatric patient. By packing the oro- and hypopharynx with KaltostatTM and iodoform packing, saliva was diverted from the carotid sheaths. Unfortunately, this patient required a prolonged intensive care stay because of sedation requirements in order to tolerate wound packing. Much of the published literature regarding management of hypopharyngeal stenosis recounts experience in adult patients after operative and/ or radiation therapy for treatment of upper aerodigestive malignancies [11,12] or in pediatric and/ or adult patients after caustic ingestion [13,14]. As in the setting of caustic ingestion, the degree of
472 significant tissue loss in our patient’s oropharynx made circumferential scarring difficult to avoid without early placement of a free tissue graft. In the setting of acute infection, placement of a free tissue graft carries significant potential morbidities in this patient. Free tissue transfer has a significant anastomotic leak rate in the hypopharynx, a significant rate of postoperative stenosis, and the possibility of a neurologically uncoordinated insensate conduit for deglutition. Donor site morbidity is another consideration if pedicled myocutaneous flaps or free tissue transfer is entertained. Though the minimally invasive technique of serial dilation can prove successful in patients with scarring after caustic ingestion, esophageal replacement may still be needed [10]. Fortunately, this patient was able to be managed completely with endoscopic techniques. By avoiding reconstruction with a pedicled myocutaneous flap or a free tissue transfer, our patient was left with a sensate pharynx which prevented aspiration with all dietary consistencies. The patient’s acquired nasopharyngeal stenosis, which was approximately 5 mm 8 mm at last follow-up examination, was significant enough to aid him in occluding his velum during speech, and as such, the patient was able to spontaneously recover from a very significant velopharyngeal insufficiency. After about a period of 3 months, this patient’s soft palate obturator was no longer needed for avoidance of nasopharyngeal reflux. Had this patient healed with a lesser degree of nasopharyngeal stenosis, performing a pharyngeal flap to treat his velopharyngeal insufficiency would have been very technically challenging.
4. Conclusion This case illustrates the importance of the role of aggressive broad-spectrum antibiotic coverage, optimizing medical care in the intensive care unit, and surgical debridement in the management of necrotizing fasciitis. Our patient’s clinical course for his necrotizing infection is atypical in that the necrosis remained focally contained to the oropharynx and parapharyngeal spaces through initial debridements performed at the admitting hospital and aggressive optimization of his multi-system organ dysfunction. Unlike the prior pediatric case report of S. marcescens NF, the patient survived the associated sepsis despite the initial delay in definitive surgical debridement due to his unstable multisystem organ dysfunction. Though the patient developed an extensive tissue loss in the oroand hypopharynx, free tissue transfer was not
M.M. Statham et al. required for reconstructive purposes. Healing by secondary intention and minimally invasive endoscopic interventions rendered this patient with a sensate laryngopharynx which functions well for voicing and deglutition.
Acknowledgements We would like to acknowledge Jonathan Singer, in the Department of Emergency Medicine at Boonshoft School of Medicine at Wright State University, and Arthur Pickoff, in the Department of Pediatrics at Dayton Children’s Hospital, for their assistance in preparation of this manuscript.
Conflict of interest None of the authors of this manuscript have any financial or personal relationships to disclose.
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