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Skull base osteomyelitis following a mastoidectomy
Auris Nasus Larynx 40 (2013) 584–586
Contents lists available at SciVerse ScienceDirect
Auris Nasus Larynx journal homepage: www.elsevier.com/locate/anl
Skull base osteomyelitis following a mastoidectomy Chang-Woo Kim *, Jong Joo Lee, Hyeon-Seong Kim, Joong-Yeon Won Department of Otorhinolaryngology-Head & Neck Surgery, College of Medicine, Hallym University, Seoul, South Korea
A R T I C L E I N F O
A B S T R A C T
Article history: Received 18 October 2012 Accepted 11 January 2013 Available online 8 February 2013
Skull base osteomyelitis is a rare but life-threatening condition that is characterized by osteitis of the temporal bone and skull base. Although skull base osteomyelitis is recognized as a rare complication of malignant external otitis or middle ear infection, it may also occur following a mastoidectomy. We present a case of an 81-year-old woman who suffered severe otalgia with normal-looking EAC and tympanic membrane that developed six weeks following a canal wall up mastoidectomy. Otalgia was the only abnormal feature to suspect occurrence of skull base osteomyelitis. ß 2013 Published by Elsevier Ireland Ltd.
Key words: Skull base Osteomyelitis Otitis media Mastoidectomy
1. Introduction Skull base osteomyelitis is a rare but life-threatening condition that is characterized by osteitis of the temporal bone and the skull base. This disease is frequently seen in elderly diabetics or immunocompromised patients. It usually begins as an infection of the external auditory canal (EAC) and progresses to involve the temporal bone. Although skull base osteomyelitis is recognized as a rare complication of malignant external otitis or middle ear infections, it may also occur as a complication of a mastoidectomy. Patel reported a case of skull base osteomyelitis that presented as an aural polyp with associated external otitis and facial cellulitis following a mastoidectomy [1]. This was the only case report that could be found in the English literature, and it showed typical findings of an EAC infection. Here, we describe a patient with skull base osteomyelitis, with a normal-looking EAC and tympanic membrane, who suffered severe otalgia that developed following a canal wall up mastoidectomy. Otalgia was the only abnormal feature, which led to the suspected occurrence of skull base osteomyelitis.
2. Case presentation An 81-year-old insulin-dependent diabetic woman presented with intermittent otalgia and otorrhea in her left ear. She denied
* Corresponding author at: Department of Otorhinolaryngology-Head & Neck Surgery, Hallym University College of Medicine, 445 Gil-Dong, Gangdong-gu, Seoul 134-701, South Korea. Tel.: +82 2 2224 2279; fax: +82 2 482 2279. E-mail address: [email protected] (C.-W. Kim). 0385-8146/$ – see front matter ß 2013 Published by Elsevier Ireland Ltd. http://dx.doi.org/10.1016/j.anl.2012.12.002
tinnitus, dizziness, prior surgery, and trauma. A physical examination using an endoscope revealed a large perforation of the tympanic membrane, with purulent discharge and a swelling of her left ear canal. Audiological examination revealed normal hearing on the right and mixed hearing loss on the left. The fourfrequency pure tone average was 55 dB for air conduction and 30 dB for bone conduction. Aerobic bacterial and fungal cultures were obtained and were found to be negative. There was no visible bacteria on gram staining. A random blood glucose level was 182 mg/dl and HbA1c was 5.8%. The erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP) were 30 mm/h and 4 mg/l, respectively. Computerized tomography (CT) revealed thickened skin in the left ear canal and soft tissue density lesions within the middle ear cavity and the mastoid antrum, with an intact clival cortical bone (Fig. 1A). Magnetic resonance imaging (MRI) showed no inflammation of the skull base (Fig. 2A). The patient underwent a canal wall up mastoidectomy and type I tympanoplasty using the temporalis fascia. The postoperative course was uneventful. Six weeks after the operation, she developed intermittent mild pain around the left auricle; however, the tympanic membrane and the skin of the ear canal healed with no evidence of infection. Three months after the operation, the patient was admitted with severe otalgia in her left ear, which manifested itself as throbbing pain deep inside her head that was exacerbated at night. The tympanic membrane and the ear canal both appeared healthy and did not exhibit any discharge. A neurological examination revealed weakness of the cranial nerve X with hoarseness. The ESR and CRP were elevated at 110 mm/h and 34 mg/l, respectively. A CT scan showed soft tissue density lesions within the mastoid cavity and the epitympanum, and exhibited bony erosion on the left side of the clivus (Fig. 1B). The MRI revealed loss of the normal fatty
C.-W. Kim et al. / Auris Nasus Larynx 40 (2013) 584–586
585
Fig. 1. Axial computed tomography scan images. (A) This preoperative image demonstrates the intact cortical bone of the left clivus (arrows). (B) This postoperative image demonstrates cortical bone destruction of the left clivus (arrows). A bony defect (arrowhead) on the left mastoid area following a mastoidectomy is noted.
marrow signal in both the left petrous apex and the clivus on the T1-weighted image and enhancement of the left prevertebral muscles and areas around the jugular bulb (Fig. 2B–D). The left ear and the mastoid cavity were both explored. The mastoid cavity was filled with fibrous tissue and cholesterol granuloma. Histopathological results revealed scar tissue formation. Aerobic bacterial and fungal cultures were again tested and found to be negative. The patient commenced treatment with intravenous ceftazidime (2 g twice per day) and amikacin (250 mg three times per day for two weeks). Three weeks later, the patient developed dysphargia, which it might have been due to her impaired ability to elevate and pull her larynx forward. Left side vocal cord palsy was remained, but there was no aspiration. Antibiotic therapy was continued until the ESR decreased to a normal range. The patient was discharged following four months of intravenous therapy with ceftazidime; she continued oral ciprofloxacin for an additional three months as an out-patient. At present, the patient’s pain has disappeared, but she continues to have dysphargia and vocal cord palsy. 3. Discussion Skull base osteomyelitis has classically been considered a complication of necrotizing external otitis [2]. Typically, granulation tissue is seen on the floor of the EAC at the bony-cartilaginous junction [3]. From there, infection spreads to the venous channels and fascial planes within the temporal bone and along the dural sinuses, eventually reaching the petrous apex and the skull base. Infection of the EAC may spread to the skull base via the Santorini fissures and the tympanomastoid suture to involve the stylomastoid foramen and the jugular foramen [4]. However some cases may show atypical presentation due to partial or inadequate treatment of the local infection of the EAC [5,6]. Severe otalgia or persistent unilateral headaches are a characteristic symptoms, and otorrhea, aural fullness, and hearing loss may be present as well. Cranial nerves may also become involved; infection spreading to the stylomastoid foramen and the jugular foramen results in palsies of the facial nerve (VII) and glossopharyngeal (IX), vagus, and spinal accessory nerve (XI) [3,7]. Intracranial complications include meningitis and cerebral abscess [3]. In our case, dysphargia developed later; generally, this is thought to be due to paralysis of the glossopharyngeal nerve because it innervates the stylopharyngeus muscle, which elevates the larynx and pulls it forward
during the pharyngeal phase of swallowing. Bacterial culture and sensitivity analyses should be performed on otorrhea and tissue biopsies [4]. Pseudomonas aerugiona is the most common pathogen of skull base osteomyelitis. Sometimes previous treatment with topical or oral antibiotics leads to negative cultures of the EAC. In such a case, anti-pseudomonal antibiotics are effective [8]. Computed tomography is useful to detect bony destruction, and MRI is valuable to identify dural enhancement and the involvement of the bone’s medullary space [9]. Gallium-67 bone scanning is the best method for monitoring a patient’s therapeutic progress. Typically, elevated ESR normalizes and otalgia disappears after successful treatment [10]. Management of skull base osteomyelitis consists of the exploration of the ear and the mastoid cavity and long-term treatment with high-dose antibiotics. Skull base osteomyelitis is an extremely rare complication following a mastoidectomy. Patel reported a case of skull base osteomyelitis developing after this procedure [1]. In this case, physical examination showed an aural polyp with associated external otitis and facial cellulitis. It seemed that localized external otitis progressed to a more invasive disease process and showed similar features to those typically found in cases of skull base osteomyelitis. Exploratory operation findings revealed severe inflammation with extensive granulations in the ear canal and a congested mastoid bone. However, in our case, the exploratory operation revealed fibrous tissue in the mastoid cavity without active infection of the middle ear or the EAC, and histopathological results revealed was compatible with scar tissue formation. The route of the infection to the skull base was not clear. Although skull base osteomyelitis occurred long time after the mastoidectomy and no evidence of causal relationship between the mastoidectomy procedure and osteomyelitis was found during the second exploratory surgery, it was clear that just an otalgia was the sign of this life-threatening condition. Perhaps, quiescent infection within the temporal bone had been reactivated after discontinuation of postoperative administration of antibiotics. We think it is important to suspect the development of postoperative problems, including skull base osteomyelitis, when otalgia has either been prolonged or progressed even in cases when postoperative recovery is uneventful. Careful examination, including a temporal bone CT, is recommended when postoperative otalgia has not subsided after
C.-W. Kim et al. / Auris Nasus Larynx 40 (2013) 584–586
586
Fig. 2. Axial MRI images. (A) This preoperative T1-weighted post-gadolium contrast image demonstrates normal prevertebral muscles and aeras around the jugular bulb. (B) This postoperative T1-weighted post-gadolinium contrast image demonstrates intense contrast uptake in the left prevertebral muscles and around the jugular bulb (arrow). (C) This postoperative T2-weighted image demonstrates high signal intensity around the jugular bulb (arrow). (D) This postoperative T1-weighted non-contrast axial MRI shows low signal intensity in the left clivus (arrow).
analgesics have been taken. Administration of antibiotics such as oral quinolone should be considered when otalgia is prolonged, even when postoperative imaging studies are negative. Conflict of interest None. References [1] Patel SK, McPartlin DW, Philpott JM, Abramovich S. A case of malignant otitis externa following mastoidectomy. J Laryngol Otol 1999;113:1095–7. [2] Benecke JE. Management of osteomyelitis of the skull base. Laryngoscope 1989;99:1220–3. [3] Slattery WH, Brackmann DE. Skull base osteomyelitis: malignant external otitis. Otolaryngol Clin North Am 1996;29:795–806.
[4] Sreepada GS, Kwartler JA. Skull base osteomyelitis secondary to malignant otitis externa. Curr Opin Otolaryngol Head Neck Surg 2003;11: 316–23. [5] Sie KC, Glenn MG, Hillel AH, Cummings CW. Osteomyelitis of the skull base, etiology unknown. Otolaryngol Head Neck Surg 1991;104:252–6. [6] Singh A, Khabori MA, Hyder MJ. Skull base osteomyelitis: diagnostic and therapeutic challenges in atypical presentation. Otolaryngol Head Neck Surg 2005;133:121–5. [7] Nadol JB. Histopathology of pseudomonas osteomyelitis of the temporal bone starting as malignant external otitis. Am J Otolaryngol 1980;1: 359–71. [8] Djalilian HR, Shamloo B, Thakkar KH, Najme-Rahim M. Treatment of culturenegative skull base osteomyelitis. Otol Neurotol 2006;27:250–5. [9] Grandis JR, Curton HD, Yu VL. Necrotizing (malignant) external otitis: prospective comparison of CT and MR imaging in diagnosis and follow-up. Radiology 1995;196:499–504. [10] Magliulo G, Varacalli S, Ciofalo A. Osteomyelitis of the skull base with atypical onset and evolution. Ann Otol Rhinol Laryngol 2000;109:326–30.
Contents lists available at SciVerse ScienceDirect
Auris Nasus Larynx journal homepage: www.elsevier.com/locate/anl
Skull base osteomyelitis following a mastoidectomy Chang-Woo Kim *, Jong Joo Lee, Hyeon-Seong Kim, Joong-Yeon Won Department of Otorhinolaryngology-Head & Neck Surgery, College of Medicine, Hallym University, Seoul, South Korea
A R T I C L E I N F O
A B S T R A C T
Article history: Received 18 October 2012 Accepted 11 January 2013 Available online 8 February 2013
Skull base osteomyelitis is a rare but life-threatening condition that is characterized by osteitis of the temporal bone and skull base. Although skull base osteomyelitis is recognized as a rare complication of malignant external otitis or middle ear infection, it may also occur following a mastoidectomy. We present a case of an 81-year-old woman who suffered severe otalgia with normal-looking EAC and tympanic membrane that developed six weeks following a canal wall up mastoidectomy. Otalgia was the only abnormal feature to suspect occurrence of skull base osteomyelitis. ß 2013 Published by Elsevier Ireland Ltd.
Key words: Skull base Osteomyelitis Otitis media Mastoidectomy
1. Introduction Skull base osteomyelitis is a rare but life-threatening condition that is characterized by osteitis of the temporal bone and the skull base. This disease is frequently seen in elderly diabetics or immunocompromised patients. It usually begins as an infection of the external auditory canal (EAC) and progresses to involve the temporal bone. Although skull base osteomyelitis is recognized as a rare complication of malignant external otitis or middle ear infections, it may also occur as a complication of a mastoidectomy. Patel reported a case of skull base osteomyelitis that presented as an aural polyp with associated external otitis and facial cellulitis following a mastoidectomy [1]. This was the only case report that could be found in the English literature, and it showed typical findings of an EAC infection. Here, we describe a patient with skull base osteomyelitis, with a normal-looking EAC and tympanic membrane, who suffered severe otalgia that developed following a canal wall up mastoidectomy. Otalgia was the only abnormal feature, which led to the suspected occurrence of skull base osteomyelitis.
2. Case presentation An 81-year-old insulin-dependent diabetic woman presented with intermittent otalgia and otorrhea in her left ear. She denied
* Corresponding author at: Department of Otorhinolaryngology-Head & Neck Surgery, Hallym University College of Medicine, 445 Gil-Dong, Gangdong-gu, Seoul 134-701, South Korea. Tel.: +82 2 2224 2279; fax: +82 2 482 2279. E-mail address: [email protected] (C.-W. Kim). 0385-8146/$ – see front matter ß 2013 Published by Elsevier Ireland Ltd. http://dx.doi.org/10.1016/j.anl.2012.12.002
tinnitus, dizziness, prior surgery, and trauma. A physical examination using an endoscope revealed a large perforation of the tympanic membrane, with purulent discharge and a swelling of her left ear canal. Audiological examination revealed normal hearing on the right and mixed hearing loss on the left. The fourfrequency pure tone average was 55 dB for air conduction and 30 dB for bone conduction. Aerobic bacterial and fungal cultures were obtained and were found to be negative. There was no visible bacteria on gram staining. A random blood glucose level was 182 mg/dl and HbA1c was 5.8%. The erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP) were 30 mm/h and 4 mg/l, respectively. Computerized tomography (CT) revealed thickened skin in the left ear canal and soft tissue density lesions within the middle ear cavity and the mastoid antrum, with an intact clival cortical bone (Fig. 1A). Magnetic resonance imaging (MRI) showed no inflammation of the skull base (Fig. 2A). The patient underwent a canal wall up mastoidectomy and type I tympanoplasty using the temporalis fascia. The postoperative course was uneventful. Six weeks after the operation, she developed intermittent mild pain around the left auricle; however, the tympanic membrane and the skin of the ear canal healed with no evidence of infection. Three months after the operation, the patient was admitted with severe otalgia in her left ear, which manifested itself as throbbing pain deep inside her head that was exacerbated at night. The tympanic membrane and the ear canal both appeared healthy and did not exhibit any discharge. A neurological examination revealed weakness of the cranial nerve X with hoarseness. The ESR and CRP were elevated at 110 mm/h and 34 mg/l, respectively. A CT scan showed soft tissue density lesions within the mastoid cavity and the epitympanum, and exhibited bony erosion on the left side of the clivus (Fig. 1B). The MRI revealed loss of the normal fatty
C.-W. Kim et al. / Auris Nasus Larynx 40 (2013) 584–586
585
Fig. 1. Axial computed tomography scan images. (A) This preoperative image demonstrates the intact cortical bone of the left clivus (arrows). (B) This postoperative image demonstrates cortical bone destruction of the left clivus (arrows). A bony defect (arrowhead) on the left mastoid area following a mastoidectomy is noted.
marrow signal in both the left petrous apex and the clivus on the T1-weighted image and enhancement of the left prevertebral muscles and areas around the jugular bulb (Fig. 2B–D). The left ear and the mastoid cavity were both explored. The mastoid cavity was filled with fibrous tissue and cholesterol granuloma. Histopathological results revealed scar tissue formation. Aerobic bacterial and fungal cultures were again tested and found to be negative. The patient commenced treatment with intravenous ceftazidime (2 g twice per day) and amikacin (250 mg three times per day for two weeks). Three weeks later, the patient developed dysphargia, which it might have been due to her impaired ability to elevate and pull her larynx forward. Left side vocal cord palsy was remained, but there was no aspiration. Antibiotic therapy was continued until the ESR decreased to a normal range. The patient was discharged following four months of intravenous therapy with ceftazidime; she continued oral ciprofloxacin for an additional three months as an out-patient. At present, the patient’s pain has disappeared, but she continues to have dysphargia and vocal cord palsy. 3. Discussion Skull base osteomyelitis has classically been considered a complication of necrotizing external otitis [2]. Typically, granulation tissue is seen on the floor of the EAC at the bony-cartilaginous junction [3]. From there, infection spreads to the venous channels and fascial planes within the temporal bone and along the dural sinuses, eventually reaching the petrous apex and the skull base. Infection of the EAC may spread to the skull base via the Santorini fissures and the tympanomastoid suture to involve the stylomastoid foramen and the jugular foramen [4]. However some cases may show atypical presentation due to partial or inadequate treatment of the local infection of the EAC [5,6]. Severe otalgia or persistent unilateral headaches are a characteristic symptoms, and otorrhea, aural fullness, and hearing loss may be present as well. Cranial nerves may also become involved; infection spreading to the stylomastoid foramen and the jugular foramen results in palsies of the facial nerve (VII) and glossopharyngeal (IX), vagus, and spinal accessory nerve (XI) [3,7]. Intracranial complications include meningitis and cerebral abscess [3]. In our case, dysphargia developed later; generally, this is thought to be due to paralysis of the glossopharyngeal nerve because it innervates the stylopharyngeus muscle, which elevates the larynx and pulls it forward
during the pharyngeal phase of swallowing. Bacterial culture and sensitivity analyses should be performed on otorrhea and tissue biopsies [4]. Pseudomonas aerugiona is the most common pathogen of skull base osteomyelitis. Sometimes previous treatment with topical or oral antibiotics leads to negative cultures of the EAC. In such a case, anti-pseudomonal antibiotics are effective [8]. Computed tomography is useful to detect bony destruction, and MRI is valuable to identify dural enhancement and the involvement of the bone’s medullary space [9]. Gallium-67 bone scanning is the best method for monitoring a patient’s therapeutic progress. Typically, elevated ESR normalizes and otalgia disappears after successful treatment [10]. Management of skull base osteomyelitis consists of the exploration of the ear and the mastoid cavity and long-term treatment with high-dose antibiotics. Skull base osteomyelitis is an extremely rare complication following a mastoidectomy. Patel reported a case of skull base osteomyelitis developing after this procedure [1]. In this case, physical examination showed an aural polyp with associated external otitis and facial cellulitis. It seemed that localized external otitis progressed to a more invasive disease process and showed similar features to those typically found in cases of skull base osteomyelitis. Exploratory operation findings revealed severe inflammation with extensive granulations in the ear canal and a congested mastoid bone. However, in our case, the exploratory operation revealed fibrous tissue in the mastoid cavity without active infection of the middle ear or the EAC, and histopathological results revealed was compatible with scar tissue formation. The route of the infection to the skull base was not clear. Although skull base osteomyelitis occurred long time after the mastoidectomy and no evidence of causal relationship between the mastoidectomy procedure and osteomyelitis was found during the second exploratory surgery, it was clear that just an otalgia was the sign of this life-threatening condition. Perhaps, quiescent infection within the temporal bone had been reactivated after discontinuation of postoperative administration of antibiotics. We think it is important to suspect the development of postoperative problems, including skull base osteomyelitis, when otalgia has either been prolonged or progressed even in cases when postoperative recovery is uneventful. Careful examination, including a temporal bone CT, is recommended when postoperative otalgia has not subsided after
C.-W. Kim et al. / Auris Nasus Larynx 40 (2013) 584–586
586
Fig. 2. Axial MRI images. (A) This preoperative T1-weighted post-gadolium contrast image demonstrates normal prevertebral muscles and aeras around the jugular bulb. (B) This postoperative T1-weighted post-gadolinium contrast image demonstrates intense contrast uptake in the left prevertebral muscles and around the jugular bulb (arrow). (C) This postoperative T2-weighted image demonstrates high signal intensity around the jugular bulb (arrow). (D) This postoperative T1-weighted non-contrast axial MRI shows low signal intensity in the left clivus (arrow).
analgesics have been taken. Administration of antibiotics such as oral quinolone should be considered when otalgia is prolonged, even when postoperative imaging studies are negative. Conflict of interest None. References [1] Patel SK, McPartlin DW, Philpott JM, Abramovich S. A case of malignant otitis externa following mastoidectomy. J Laryngol Otol 1999;113:1095–7. [2] Benecke JE. Management of osteomyelitis of the skull base. Laryngoscope 1989;99:1220–3. [3] Slattery WH, Brackmann DE. Skull base osteomyelitis: malignant external otitis. Otolaryngol Clin North Am 1996;29:795–806.
[4] Sreepada GS, Kwartler JA. Skull base osteomyelitis secondary to malignant otitis externa. Curr Opin Otolaryngol Head Neck Surg 2003;11: 316–23. [5] Sie KC, Glenn MG, Hillel AH, Cummings CW. Osteomyelitis of the skull base, etiology unknown. Otolaryngol Head Neck Surg 1991;104:252–6. [6] Singh A, Khabori MA, Hyder MJ. Skull base osteomyelitis: diagnostic and therapeutic challenges in atypical presentation. Otolaryngol Head Neck Surg 2005;133:121–5. [7] Nadol JB. Histopathology of pseudomonas osteomyelitis of the temporal bone starting as malignant external otitis. Am J Otolaryngol 1980;1: 359–71. [8] Djalilian HR, Shamloo B, Thakkar KH, Najme-Rahim M. Treatment of culturenegative skull base osteomyelitis. Otol Neurotol 2006;27:250–5. [9] Grandis JR, Curton HD, Yu VL. Necrotizing (malignant) external otitis: prospective comparison of CT and MR imaging in diagnosis and follow-up. Radiology 1995;196:499–504. [10] Magliulo G, Varacalli S, Ciofalo A. Osteomyelitis of the skull base with atypical onset and evolution. Ann Otol Rhinol Laryngol 2000;109:326–30.