- Email: [email protected]
Neonatal retropharyngeal abscess with complications: Apnea and cervical osteomyelitis
International Journal of Pediatric Otorhinolaryngology 126 (2019) 109613
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Neonatal retropharyngeal abscess with complications: Apnea and cervical osteomyelitis
T
Noor-E-Seher Ali, Jennifer C. Alyono, Peter J. Koltai* Department of Otolaryngology – Head and Neck Surgery, Stanford University School of Medicine, Stanford, CA, USA
A R T I C LE I N FO
A B S T R A C T
Keywords: Neonate Retropharyngeal abscess Cervical osteomyelitis
Objective: To evaluate the clinical presentation and management strategies for neonatal retropharyngeal abscess (RPA). Methods: Retrospective chart review was performed, and literature reviewed. Results: We report two cases of neonatal RPA, with one complicated by cervical osteomyelitis, and the other presenting with apparent life-threatening events (ALTEs). A 6-week-old female underwent transoral drainage of an RPA, which grew methicillin sensitive Staphylococcus aureus. She had a prolonged recovery course and was found to have developed osteomyelitis of the dens and atlas. She was treated with 14 weeks of IV antibiotics and rigid collar fixation for spinal cord instability. A 2-month-old female was admitted after multiple ALTEs with episodes of apnea and pallor. Direct laryngoscopy revealed a bulging RPA, which was drained transorally. This grew multiple organisms including methicillin resistant Staphylococcus aureus, Streptococcal oralis and Prevotella species. Conclusions: Uncommon in neonates, RPA can present in this age group without fever, and are is likely to have airway complications than in older children. In cases with prolonged recovery, additional diagnostic intervention is recommended to rule out rare complications such as osteomyelitis. Emphasis in such complex cases is placed on a multidisciplinary approach to patient care, coordinating neonatologists, infectious disease specialists, neurosurgeons, and otolaryngologists.
1. Introduction RPA is relatively rare in the neonate. The average age at presentation for pediatric cases is 3.7–5.1 [ [1–5]]. Neonates can present with different symptoms than do older patients due to their immature immune systems. Localizing or neurological symptoms may also be absent, even in the presence of complications. Here we present two cases of neonatal RPA, with attention to the microbiology, and recognition and treatment of complications. 2. Case 1 A 6-week-old ex-35 week female was transferred to our tertiary care facility after initially presenting with inability to turn her head to the right, pain with neck manipulation, decreased oral intake, and a positional increased work of breathing, without fever. A CT scan at the transferring facility demonstrated a 2.4 × 1 × 1.8 cm retropharyngeal lesion with airway narrowing. The patient was intubated to ensure a secure airway for transfer, requiring intubation attempts by multiple
*
neonatologists and an otolaryngologist, during which pus was noted in the pharynx. Laboratory studies were significant for a leukocytosis of 22,000/mm3 and CRP of 0.69 mg/dL. After transfer, an MRI was obtained demonstrating that the retropharyngeal lesion was an abscess (see Fig. 1A and Fig. 1B). No enhancement of the vertebrae was noted. She underwent transoral incision and drainage (I&D) on the day after transfer, with cultures growing methicillin sensitive Staphylococcus aureus (MSSA) (see Fig. 2). She was extubated postoperative day (POD) 3. A repeat direct laryngoscopy was performed on POD 5 as the infant developed significant dysphagia with stridor; this showed a partially healing but open incision in the abscess wall, with no further purulence. She was discharged back to her local hospital POD 10 after demonstrating improved oral intake, improvement in leukocytosis count to 9.6, remaining afebrile, with plan to complete a total 3 week course of IV nafcillin. At the transferring facility, she required nutritional supplementation with nasogastric tube feeds. She was retransferred back to our facility on POD 21 after a stridorous episode and inability to tolerate oral secretions. A bedside flexible fiberoptic exam showed no evidence of RPA
Corresponding author. Department of Otolaryngology-Head and Neck Surgery, Stanford University, 801 Welch Road, Stanford, CA, 94305-5739, USA. E-mail address: [email protected] (P.J. Koltai).
https://doi.org/10.1016/j.ijporl.2019.109613 Received 10 June 2019; Accepted 27 July 2019 Available online 31 July 2019 0165-5876/ © 2019 Published by Elsevier B.V.
International Journal of Pediatric Otorhinolaryngology 126 (2019) 109613
N.-E.-S. Ali, et al.
Fig. 1. Axial (A) and sagittal (B) gadolinium enhanced fat-saturated T1 sections demonstrating retropharyngeal abscess prior to drainage following intubation. There is no enhancement of the vertebral bodies, which was noted on subsequent imaging.
weakness, hyperreflexia or discomfort, no urinary retention or difficulties with stooling. 3. Case 2 A 2-month-old ex-full-term female was admitted after multiple ALTEs associated with pallor, occurring both during sleep and while awake. Her parents reported 3 weeks of noisy breathing, 2 weeks nasal congestion, 1 week increased work of breathing with retractions, and several days of increasing spit up following feeds. On admission she was noted to have a temperature maximum of 38.1, sternal retractions, intermittent stridor, with no restriction in neck movement, drooling, or neck masses. Laboratory workup was remarkable for a leukocytosis of 33,000/mm3 with an elevated CRP of 2.8 mg/ dL. The larynx could not be visualized using bedside flexible fiberoptic nasopharyngoscopy; thus the patient was taken to the operating room for rigid endoscopy. This showed a submucosal, fluctuant, posterior pharyngeal wall lesion to the right of midline with slight displacement of the larynx to the left, consistent with a retropharyngeal abscess (see Fig. 4). After confirming that the subglottis, trachea and bronchi were clear, I&D was performed with drainage of approximately 3 mL of thick pus. The patient was started on IV ampicillin/sulbactam and vancomycin, which was narrowed to oral linezolid for a 3-week course after cultures grew methicillin-resistant Staphylococcus aureus (MRSA), Streptococcal oralis, and Prevotella species. MRI of the cervical spine and chest 3 days following I&D showed no remaining fluid collection, and no osseous abnormalities or mediastinal extension of the infection (see Fig. 5). Prior to discharge the patient was afebrile with no desaturations, no stridor, resumed normal feeding, with leukocytosis improved to 16.9, and CRP decreased to 2.2.
Fig. 2. Intraoperative photograph during direct laryngoscopy demonstrating a retropharyngeal fluid collection centered right of midline.
or fluid collection. MRI and CT performed four weeks postoperatively were remarkable for new enhancement of the C1 and C2 vertebra, with bony destruction and irregularity of C2 at the junction of the dens and body as well as an atlantoaxial offset suggestive of osteomyelitis (see Fig. 3A and Fig. 3B). Given her persistent infection and progression to osteomyelitis without fever or elevation in ESR, or CRP, a workup for immunodeficiency was performed, which was normal. She completed a 14-week course of nafcillin. Neurosurgery was consulted for spinal cord instability with both flexion and extension. Serial imaging showed severe narrowing of her cervical spinal canal with flexion and continued atlantoaxial instability at 1 year of age. Nonetheless, no myelomalacia or intrinsic signal abnormality of the spinal cord has been demonstrated on MRI. Given her young age, occipital to cervical fixation was deemed to have a high risk of failure. Thus, surgical intervention has been deferred by neurosurgery as long as possible, and the patient managed conservatively with rigid collar fixation. She has now been followed clinically and radiographically for > 10 months since initial presentation, during which she has returned to regular breathing and oral intake, with no neurologic deficits. She has maintained full spontaneous movement of all extremities without
4. Discussion The retropharyngeal space is bounded by the prevertebral and buccopharyngeal fascia. It is contiguous with the parapharyngeal space bilaterally and contains lymph nodes that receive drainage from the nasopharynx, adenoids and posterior paranasal sinuses. It is thought that suppuration of these lymph nodes leads to abscess formation [6]. RPA in the neonate is rare, and presenting symptoms are atypical. More common in older children, the mean age for presentation is 3.7–5.1 [ [1–5]]. Typical clinical findings are neck mass, sore throat, torticollis, anorexia and oropharyngeal bulge, with fever cited as the 2
International Journal of Pediatric Otorhinolaryngology 126 (2019) 109613
N.-E.-S. Ali, et al.
Fig. 3. (A) Axial gadolinium-enhanced T1-weighted MRI performed four weeks postoperatively showing enhancement of the C1 and C2 vertebra. (arrow head) (B) Sagittal CT of the cervical spine showing atlantoaxial anterior subluxation, with a predental distance of 7.5 mm, from the posterior margin of C1 to the anterior margin of the dens. There is associated bony destruction and incomplete visualization of the dens. (arrow head) No narrowing of the spinal canal was noted on either MRI or CT.
to 77–90% of pediatric patients [ [2,4,5,8]]. Additionally, neck mass or lymphadenopathy was present in only 20% of neonatal cases, as compared to in 58–85% of cases in older children [ [4,5,8]]. As was demonstrated in both our cases, retrospective studies suggest airway symptoms are more common in neonates [ [1,9,10]]. One study found that respiratory symptoms were present in 71% of those less than one year, but in 43% of those older than one [10]. Another review of 130 children with RPA found that those patients who developed airway obstruction had an average age of 1.4, whereas those without airway issues had an average age of 3.6 [1]. Another recent study looking at 540 children with deep neck space abscesses found patients less than 2 years of age with a retropharyngeal abscess were more likely to develop airway complications compared to older children [11]. Few cases of neonatal cervical osteomyelitis have been reported, and even fewer in association with retropharyngeal abscess [ [4,12–15]]. The most frequently described complications of pediatric deep neck space abscesses are mediastinitis, airway obstruction, persistent fluid collection requiring repeat drainage, pneumonia (presumably from aspiration of pus), sepsis, and jugular vein thrombosis [ [5,8,16]]. In adults, a limited number of cases of RPA complicated by vertebral osteomyelitis have also been reported [ [17–20]]. In addition to more typical symptoms of RPA such as dysphagia, sore throat and fever, patients generally presented with neurologic deficits such as extremity weakness/numbness, gait instability and bladder dysfunction—symptoms that are less reliably distinguished in neonates. Vertebral osteomyelitis is most commonly caused by Staphylococcus aureus, followed by streptococcus and pneumococcus [21]. In rare cases, gram-negative bacteria or nonpyogenic microorganisms such as Mycobacterium tuberculosis, Brucella, or fungi can be the causative agent [21]. Direct extension from a contiguous abscess, as presented here, is relatively rare, with hematogenous seeding thought to be more common [21,22]. While a survey of the American Society of Pediatric Otolaryngologists reported CT as the preferred diagnostic method in RPA [23], MRI is recommended in cases where vertebral osteomyelitis is suspected due to its early detection, good spatial resolution, and high sensitivity (82–100%) and specificity (75–96%) [24]. CT images demonstrate areas of bony erosion; and while they are less sensitive overall, are better at demonstrating bony sequestra and pathologic calcifications [21]. In terms of RPA treatment, many groups advocate early surgical intervention [ [4,7]] while others prefer medical therapy with IV antibiotics for the majority of cases, reserving surgery only for those with complications [ [2,25]]. Given the higher incidence of airway issues in neonates, earlier intervention in this subset of patients may be prudent.
Fig. 4. Direct laryngoscopy of Case 2 demonstrating a submucosal retropharyngeal fluid collection to the right of midline.
Fig. 5. Axial gadolinium enhanced T1-weighted MRI of Case 2 demonstrating inflammatory changes of the right retropharyngeal soft tissues following transoral drainage of abscess. No spinal cord or cervical vertebral abnormalities were noted.
most common symptom in multiple series [ [2,4,5,7,8]]. Neonates present a particular diagnostic challenge given their immunological immaturity, and thus atypical presentation. In a review of published neonatal RPA, only 27% of patients presented with fever [9] compared 3
International Journal of Pediatric Otorhinolaryngology 126 (2019) 109613
N.-E.-S. Ali, et al.
In cases of vertebral osteomyelitis, treatment involves addressing not only the infection itself with antibiotics and abscess drainage, but also any spinal sequelae that may result. This includes assessment for instability with flexion/extension films, and possible fixation, be it with a rigid collar or surgically.
retrospective and historical analysis, The Laryngoscope (1988). [6] B. Asmar, Bacteriology of retropharyngeal abscess in children, Pediatr. Infect. Dis. J. (1990). [7] D.J. Kirse, D.W. Roberson, Surgical management of retropharyngeal space infections in children, The Laryngoscope 111 (2001) 1413–1422. [8] S.S. Lee, R.H. Schwartz, R.S. Bahadori, Retropharyngeal abscess: epiglottitis of the new millennium, J. Pediatr. 138 (2001) 435–437. [9] Y. Kanazawa, M. Asai, Y. Adachi, et al., Retropharyngeal abscess in a neonate: a case report and literature review, Int. J. Pediatr. Otorhinolaryngol. Extra 7 (2012) 115–118. [10] M. Coulthard, D. Isaacs, Retropharyngeal abscess, Arch. Dis. Child. 66 (1991) 1227–1230. [11] A. Jain, I. Singh, R. Meher, A. Raj, P. Rajpurohit, P. Prasad, Deep neck space abscesses in children below 5 years of age and their complications, Int. J. Pediatr. Otorhinolaryngol. 109 (2018) 40–43. [12] H.S. An, R. Munk, Osteomyelitis of the posterior elements of the cervical spine in an infant, Orthopedics 16 (1993) 618–620. [13] N. Buxton, J. Punt, Osteomyelitis of the atlas in an infant, Child's Nerv. Syst. : ChNS : Off. J. Int. Soc. Pediatr. Neurosurg. 13 (1997) 175–177. [14] S.H. Ein, B. Shandling, R. Humphreys, I. Krajbich, Osteomyelitis of the cervical spine presenting as a neurenteric cyst, J. Pediatr. Surg. 23 (1988) 779–781. [15] Z. Papp, G. Czigléczki, P. Banczerowski, Multiple abscesses with osteomyelitis and destruction of both the atlas and the Axis in a 4-week-old infant, Spine (2013). [16] C.M. Baldassari, R. Howell, M. Amorn, R. Budacki, S. Choi, M. Pena, Complications in pediatric deep neck space abscesses, Otolaryngology-Head Neck Surg.: Off. J. Am. Acad. Otolaryngology-Head Neck Surg. 144 (2011) 592–595. [17] G. Barratt, C. Koopmann, S. Coulthard, Retropharyngeal abscess-a ten year experience, The Laryngoscope (1984). [18] a Faidas, J.V. Ferguson, J.E. Nelson, L.M. Baddour, Cervical vertebral osteomyelitis presenting as a retropharyngeal abscess, Clin. Infect. Dis. : Off. Publ. Infect. Dis. Soc. Am. 18 (1994) 992–994. [19] Ja Moche, K. Chopra, B. Gastman, Vascularized free fibula for cervical spine reconstruction following complicated retropharyngeal abscess, Otolaryngology-Head Neck Surg. : Off. J. Am. Acad. Otolaryngology-Head Neck Surg. 145 (2011) 178–179. [20] M. Sakamoto, K. Ichimura, N. Tayama, M. Nakamura, K. Inokuchi, Cervical vertebral osteomyelitis revisited: a case of retropharyngeal abscess and progressive muscle weakness, Otolaryngol. Head Neck Surg. 121 (1999) 657–660. [21] V. Jevtic, Vertebral infection, Eur. Radiol. 14 (Suppl 3) (2004) E43–E52. [22] S.J. Bagatell, S.M. Weimer, R.B. Van Dyke, S.V. Bracey, Neck pain, Clin. Pediatr. 46 (2007) 280–281. [23] M. Lalakea, A. Messner, Retropharyngeal abscess management in children: current practices, Otolaryngol. Head Neck Surg. 121 (1999) 398–405. [24] C. Pineda, R. Espinosa, A. Pena, Radiographic imaging in osteomyelitis : the role of plain radiography , computed tomography , ultrasonography , magnetic resonance imaging and scintigraphy, Semin. Plast. Surg. 1 (2009) 80–89. [25] J.-Y. Sichel, I. Dano, E. Hocwald, A. Biron, R. Eliashar, Nonsurgical management of parapharyngeal space infections: a prospective study, The Laryngoscope 112 (2002) 906–910.
5. Conclusion RPA is rare in the neonate, and can present with atypical symptoms, often lacking the fever or lymphadenopathy more commonly seen in older patients. Early surgical intervention should be considered given the higher prevalence of airway symptoms in neonates. Should neurologic deficits be noted or in cases where the infant demonstrates a prolonged recovery to baseline, further investigation with MRI is warranted. Clinical suggestion for vertebral osteomyelitis may be less obvious than in adults, as symptoms such as bladder dysfunction, gait instability, and sensory deficits are not as reliably distinguished. In nearly all cases of retropharyngeal abscess complicated by vertebral osteomyelitis, MRSA or MSSA was the single responsible organism. Emphasis in such complex cases is placed on taking a multidisciplinary approach to patient care, coordinating neonatologists, infectious disease specialists, neurosurgeons and otolaryngologists. Financial disclosures The authors have no relevant financial disclosures. References [1] A.M. Elsherif, A.H. Park, S.C. Alder, M.E. Smith, H.R. Muntz, F. Grimmer, Indicators of a more complicated clinical course for pediatric patients with retropharyngeal abscess, Int. J. Pediatr. Otorhinolaryngol. 74 (2010) 198–201. [2] D. Johnston, R. Schmidt, P. Barth, Parapharyngeal and retropharyngeal infections in children: argument for a trial of medical therapy and intraoral drainage for medical treatment failures, Int. J. Pediatr. Otorhinolaryngol. 73 (2009) 761–765. [3] L. Lander, S. Lu, R.K. Shah, Pediatric retropharyngeal abscesses: a national perspective, Int. J. Pediatr. Otorhinolaryngol. 72 (2008) 1837–1843. [4] N.C. Page, E.M. Bauer, J.E.C. Lieu, Clinical features and treatment of retropharyngeal abscess in children, Otolaryngology-Head Neck Surg. : Off. J. Am. Acad. Otolaryngology-Head Neck Surg. 138 (2008) 300–306. [5] J. Thompson, P. Reddk, S. Cohen, Retropharyngeal abscess in children: a
4
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Neonatal retropharyngeal abscess with complications: Apnea and cervical osteomyelitis
T
Noor-E-Seher Ali, Jennifer C. Alyono, Peter J. Koltai* Department of Otolaryngology – Head and Neck Surgery, Stanford University School of Medicine, Stanford, CA, USA
A R T I C LE I N FO
A B S T R A C T
Keywords: Neonate Retropharyngeal abscess Cervical osteomyelitis
Objective: To evaluate the clinical presentation and management strategies for neonatal retropharyngeal abscess (RPA). Methods: Retrospective chart review was performed, and literature reviewed. Results: We report two cases of neonatal RPA, with one complicated by cervical osteomyelitis, and the other presenting with apparent life-threatening events (ALTEs). A 6-week-old female underwent transoral drainage of an RPA, which grew methicillin sensitive Staphylococcus aureus. She had a prolonged recovery course and was found to have developed osteomyelitis of the dens and atlas. She was treated with 14 weeks of IV antibiotics and rigid collar fixation for spinal cord instability. A 2-month-old female was admitted after multiple ALTEs with episodes of apnea and pallor. Direct laryngoscopy revealed a bulging RPA, which was drained transorally. This grew multiple organisms including methicillin resistant Staphylococcus aureus, Streptococcal oralis and Prevotella species. Conclusions: Uncommon in neonates, RPA can present in this age group without fever, and are is likely to have airway complications than in older children. In cases with prolonged recovery, additional diagnostic intervention is recommended to rule out rare complications such as osteomyelitis. Emphasis in such complex cases is placed on a multidisciplinary approach to patient care, coordinating neonatologists, infectious disease specialists, neurosurgeons, and otolaryngologists.
1. Introduction RPA is relatively rare in the neonate. The average age at presentation for pediatric cases is 3.7–5.1 [ [1–5]]. Neonates can present with different symptoms than do older patients due to their immature immune systems. Localizing or neurological symptoms may also be absent, even in the presence of complications. Here we present two cases of neonatal RPA, with attention to the microbiology, and recognition and treatment of complications. 2. Case 1 A 6-week-old ex-35 week female was transferred to our tertiary care facility after initially presenting with inability to turn her head to the right, pain with neck manipulation, decreased oral intake, and a positional increased work of breathing, without fever. A CT scan at the transferring facility demonstrated a 2.4 × 1 × 1.8 cm retropharyngeal lesion with airway narrowing. The patient was intubated to ensure a secure airway for transfer, requiring intubation attempts by multiple
*
neonatologists and an otolaryngologist, during which pus was noted in the pharynx. Laboratory studies were significant for a leukocytosis of 22,000/mm3 and CRP of 0.69 mg/dL. After transfer, an MRI was obtained demonstrating that the retropharyngeal lesion was an abscess (see Fig. 1A and Fig. 1B). No enhancement of the vertebrae was noted. She underwent transoral incision and drainage (I&D) on the day after transfer, with cultures growing methicillin sensitive Staphylococcus aureus (MSSA) (see Fig. 2). She was extubated postoperative day (POD) 3. A repeat direct laryngoscopy was performed on POD 5 as the infant developed significant dysphagia with stridor; this showed a partially healing but open incision in the abscess wall, with no further purulence. She was discharged back to her local hospital POD 10 after demonstrating improved oral intake, improvement in leukocytosis count to 9.6, remaining afebrile, with plan to complete a total 3 week course of IV nafcillin. At the transferring facility, she required nutritional supplementation with nasogastric tube feeds. She was retransferred back to our facility on POD 21 after a stridorous episode and inability to tolerate oral secretions. A bedside flexible fiberoptic exam showed no evidence of RPA
Corresponding author. Department of Otolaryngology-Head and Neck Surgery, Stanford University, 801 Welch Road, Stanford, CA, 94305-5739, USA. E-mail address: [email protected] (P.J. Koltai).
https://doi.org/10.1016/j.ijporl.2019.109613 Received 10 June 2019; Accepted 27 July 2019 Available online 31 July 2019 0165-5876/ © 2019 Published by Elsevier B.V.
International Journal of Pediatric Otorhinolaryngology 126 (2019) 109613
N.-E.-S. Ali, et al.
Fig. 1. Axial (A) and sagittal (B) gadolinium enhanced fat-saturated T1 sections demonstrating retropharyngeal abscess prior to drainage following intubation. There is no enhancement of the vertebral bodies, which was noted on subsequent imaging.
weakness, hyperreflexia or discomfort, no urinary retention or difficulties with stooling. 3. Case 2 A 2-month-old ex-full-term female was admitted after multiple ALTEs associated with pallor, occurring both during sleep and while awake. Her parents reported 3 weeks of noisy breathing, 2 weeks nasal congestion, 1 week increased work of breathing with retractions, and several days of increasing spit up following feeds. On admission she was noted to have a temperature maximum of 38.1, sternal retractions, intermittent stridor, with no restriction in neck movement, drooling, or neck masses. Laboratory workup was remarkable for a leukocytosis of 33,000/mm3 with an elevated CRP of 2.8 mg/ dL. The larynx could not be visualized using bedside flexible fiberoptic nasopharyngoscopy; thus the patient was taken to the operating room for rigid endoscopy. This showed a submucosal, fluctuant, posterior pharyngeal wall lesion to the right of midline with slight displacement of the larynx to the left, consistent with a retropharyngeal abscess (see Fig. 4). After confirming that the subglottis, trachea and bronchi were clear, I&D was performed with drainage of approximately 3 mL of thick pus. The patient was started on IV ampicillin/sulbactam and vancomycin, which was narrowed to oral linezolid for a 3-week course after cultures grew methicillin-resistant Staphylococcus aureus (MRSA), Streptococcal oralis, and Prevotella species. MRI of the cervical spine and chest 3 days following I&D showed no remaining fluid collection, and no osseous abnormalities or mediastinal extension of the infection (see Fig. 5). Prior to discharge the patient was afebrile with no desaturations, no stridor, resumed normal feeding, with leukocytosis improved to 16.9, and CRP decreased to 2.2.
Fig. 2. Intraoperative photograph during direct laryngoscopy demonstrating a retropharyngeal fluid collection centered right of midline.
or fluid collection. MRI and CT performed four weeks postoperatively were remarkable for new enhancement of the C1 and C2 vertebra, with bony destruction and irregularity of C2 at the junction of the dens and body as well as an atlantoaxial offset suggestive of osteomyelitis (see Fig. 3A and Fig. 3B). Given her persistent infection and progression to osteomyelitis without fever or elevation in ESR, or CRP, a workup for immunodeficiency was performed, which was normal. She completed a 14-week course of nafcillin. Neurosurgery was consulted for spinal cord instability with both flexion and extension. Serial imaging showed severe narrowing of her cervical spinal canal with flexion and continued atlantoaxial instability at 1 year of age. Nonetheless, no myelomalacia or intrinsic signal abnormality of the spinal cord has been demonstrated on MRI. Given her young age, occipital to cervical fixation was deemed to have a high risk of failure. Thus, surgical intervention has been deferred by neurosurgery as long as possible, and the patient managed conservatively with rigid collar fixation. She has now been followed clinically and radiographically for > 10 months since initial presentation, during which she has returned to regular breathing and oral intake, with no neurologic deficits. She has maintained full spontaneous movement of all extremities without
4. Discussion The retropharyngeal space is bounded by the prevertebral and buccopharyngeal fascia. It is contiguous with the parapharyngeal space bilaterally and contains lymph nodes that receive drainage from the nasopharynx, adenoids and posterior paranasal sinuses. It is thought that suppuration of these lymph nodes leads to abscess formation [6]. RPA in the neonate is rare, and presenting symptoms are atypical. More common in older children, the mean age for presentation is 3.7–5.1 [ [1–5]]. Typical clinical findings are neck mass, sore throat, torticollis, anorexia and oropharyngeal bulge, with fever cited as the 2
International Journal of Pediatric Otorhinolaryngology 126 (2019) 109613
N.-E.-S. Ali, et al.
Fig. 3. (A) Axial gadolinium-enhanced T1-weighted MRI performed four weeks postoperatively showing enhancement of the C1 and C2 vertebra. (arrow head) (B) Sagittal CT of the cervical spine showing atlantoaxial anterior subluxation, with a predental distance of 7.5 mm, from the posterior margin of C1 to the anterior margin of the dens. There is associated bony destruction and incomplete visualization of the dens. (arrow head) No narrowing of the spinal canal was noted on either MRI or CT.
to 77–90% of pediatric patients [ [2,4,5,8]]. Additionally, neck mass or lymphadenopathy was present in only 20% of neonatal cases, as compared to in 58–85% of cases in older children [ [4,5,8]]. As was demonstrated in both our cases, retrospective studies suggest airway symptoms are more common in neonates [ [1,9,10]]. One study found that respiratory symptoms were present in 71% of those less than one year, but in 43% of those older than one [10]. Another review of 130 children with RPA found that those patients who developed airway obstruction had an average age of 1.4, whereas those without airway issues had an average age of 3.6 [1]. Another recent study looking at 540 children with deep neck space abscesses found patients less than 2 years of age with a retropharyngeal abscess were more likely to develop airway complications compared to older children [11]. Few cases of neonatal cervical osteomyelitis have been reported, and even fewer in association with retropharyngeal abscess [ [4,12–15]]. The most frequently described complications of pediatric deep neck space abscesses are mediastinitis, airway obstruction, persistent fluid collection requiring repeat drainage, pneumonia (presumably from aspiration of pus), sepsis, and jugular vein thrombosis [ [5,8,16]]. In adults, a limited number of cases of RPA complicated by vertebral osteomyelitis have also been reported [ [17–20]]. In addition to more typical symptoms of RPA such as dysphagia, sore throat and fever, patients generally presented with neurologic deficits such as extremity weakness/numbness, gait instability and bladder dysfunction—symptoms that are less reliably distinguished in neonates. Vertebral osteomyelitis is most commonly caused by Staphylococcus aureus, followed by streptococcus and pneumococcus [21]. In rare cases, gram-negative bacteria or nonpyogenic microorganisms such as Mycobacterium tuberculosis, Brucella, or fungi can be the causative agent [21]. Direct extension from a contiguous abscess, as presented here, is relatively rare, with hematogenous seeding thought to be more common [21,22]. While a survey of the American Society of Pediatric Otolaryngologists reported CT as the preferred diagnostic method in RPA [23], MRI is recommended in cases where vertebral osteomyelitis is suspected due to its early detection, good spatial resolution, and high sensitivity (82–100%) and specificity (75–96%) [24]. CT images demonstrate areas of bony erosion; and while they are less sensitive overall, are better at demonstrating bony sequestra and pathologic calcifications [21]. In terms of RPA treatment, many groups advocate early surgical intervention [ [4,7]] while others prefer medical therapy with IV antibiotics for the majority of cases, reserving surgery only for those with complications [ [2,25]]. Given the higher incidence of airway issues in neonates, earlier intervention in this subset of patients may be prudent.
Fig. 4. Direct laryngoscopy of Case 2 demonstrating a submucosal retropharyngeal fluid collection to the right of midline.
Fig. 5. Axial gadolinium enhanced T1-weighted MRI of Case 2 demonstrating inflammatory changes of the right retropharyngeal soft tissues following transoral drainage of abscess. No spinal cord or cervical vertebral abnormalities were noted.
most common symptom in multiple series [ [2,4,5,7,8]]. Neonates present a particular diagnostic challenge given their immunological immaturity, and thus atypical presentation. In a review of published neonatal RPA, only 27% of patients presented with fever [9] compared 3
International Journal of Pediatric Otorhinolaryngology 126 (2019) 109613
N.-E.-S. Ali, et al.
In cases of vertebral osteomyelitis, treatment involves addressing not only the infection itself with antibiotics and abscess drainage, but also any spinal sequelae that may result. This includes assessment for instability with flexion/extension films, and possible fixation, be it with a rigid collar or surgically.
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5. Conclusion RPA is rare in the neonate, and can present with atypical symptoms, often lacking the fever or lymphadenopathy more commonly seen in older patients. Early surgical intervention should be considered given the higher prevalence of airway symptoms in neonates. Should neurologic deficits be noted or in cases where the infant demonstrates a prolonged recovery to baseline, further investigation with MRI is warranted. Clinical suggestion for vertebral osteomyelitis may be less obvious than in adults, as symptoms such as bladder dysfunction, gait instability, and sensory deficits are not as reliably distinguished. In nearly all cases of retropharyngeal abscess complicated by vertebral osteomyelitis, MRSA or MSSA was the single responsible organism. Emphasis in such complex cases is placed on taking a multidisciplinary approach to patient care, coordinating neonatologists, infectious disease specialists, neurosurgeons and otolaryngologists. Financial disclosures The authors have no relevant financial disclosures. References [1] A.M. Elsherif, A.H. Park, S.C. Alder, M.E. Smith, H.R. Muntz, F. Grimmer, Indicators of a more complicated clinical course for pediatric patients with retropharyngeal abscess, Int. J. Pediatr. Otorhinolaryngol. 74 (2010) 198–201. [2] D. Johnston, R. Schmidt, P. Barth, Parapharyngeal and retropharyngeal infections in children: argument for a trial of medical therapy and intraoral drainage for medical treatment failures, Int. J. Pediatr. Otorhinolaryngol. 73 (2009) 761–765. [3] L. Lander, S. Lu, R.K. Shah, Pediatric retropharyngeal abscesses: a national perspective, Int. J. Pediatr. Otorhinolaryngol. 72 (2008) 1837–1843. [4] N.C. Page, E.M. Bauer, J.E.C. Lieu, Clinical features and treatment of retropharyngeal abscess in children, Otolaryngology-Head Neck Surg. : Off. J. Am. Acad. Otolaryngology-Head Neck Surg. 138 (2008) 300–306. [5] J. Thompson, P. Reddk, S. Cohen, Retropharyngeal abscess in children: a
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