[No abstract available]734576Benecke Jr., J.A., Management of osteomyelitis of the skull base (1989) Laryngoscope, 99 (12), pp. 1220-1223Boringa, J.B., Hoekstra, O.S., Roos, J.W., Bertelsmann, F.W., Multiple cranial nerve palsy after otitis externa: A case report (1995) Clin Neurol Neurosurg, 97, pp. 332-335Rubin, J., Yu, V.L., Malignant external otitis: Insights into pathogenesis, clinical manifestations and therapy (1998) Am J Med, 85, pp. 391-39

De Andrade N.A.

Fonseca A.S.

Neto M.L.A.

Santos V.M.D.M.

English

Repositorio da Producao Cientifica e Intelectual da Unicamp

576
BRAZILIAN JOURNAL OF OTORHINOLARYNGOLOGY 73 (4) JULY/AUGUST 2007
http://www.rborl.org.br  /  e-mail: revista@aborlccf.org.br
CASE REPORT
Bilateral hipoglossal nerve 
palsy in necrotizing otitis 
externa
Adriano Santana Fonseca 1, Nilvano Alves de Andrade 2, 
Miguel Leal Andrade Neto 3, Vyrna Medeiros de Moura 
Santos 4
INTRODUCTION
Necrotizing otitis externa is a potentially 
lethal infection that starts in the external audi-
tory canal and may progress to the skull base. 
It happens in elderly diabetic patients and is 
associated to a high morbi-mortality rate.1  The 
major causal agent is Pseudomonas aeruginosa, 
in 96 to 98% of the cases2. 
The infection extends from the osteo-
cartilaginous junction to the temporal bone by 
means of the Santorini fissures. The infection 
may progress towards the skull base and affect 
the facial, glossopharyngeal, vagus and acces-
sory nerves. It may occasionally affect the hypo-
glossal, abducens and trigeminal nerves.3
Symptoms such as otalgia, headache, 
hypoacusis, otorrhea, in diabetic or immunosu-
pressed patients are very relevant. Laboratory 
investigation reveals high ESR, with normal 
or mildly high white cell count. Clinical signs 
include ulceration on the floor of the EAC. CT 
scan shows bone destruction, and MRI shows 
both the location and extension of the infection, 
intracranial invasion and cranial nerve involve-
ment. Scintigraphy with technetium and gallium 
has been used in order to assess cure criteria. 
Technetium scintigraphy is useful to diagnose 
osteitis, which is positive in cases of acute or 
chronic osteomyelitis, or trauma. It bears low 
specificity and may remain positive for one 
year2. Galium-67 scintigraphy is used in the 
follow up and check of the therapeutic response, 
since galium has great affinity for acute phase 
leucocytes and proteins.2
Germ culture is necessary for proper 
treatment, however one should not wait for its 
result in order to start treatment. Two empirical 
antibiotics are used: aminoglycosides together 
with ciprofloxacin of cephtazidime. Alternative 
drugs are cefepime, cefoperazone, imipenem 
and aztreonam. Treatment lasts between 4 and 
6 weeks.1 Otalgia reduction or cessation is an 
important control parameter. Most patients 
may be clinically treated, and surgery bears 
controversies, such as: progressive pain, cranial 
neuropathy and granulation persistence in the 
EAC. Mastoidectomy may be carried out, howe-
ver in some cases it does not prevent skull base 
disease extension.3
CASE REPORT
AAB, 71 years old, male, from Salvador, 
hypertensive and diabetic, came to us in August 
20, 2004, complaining of two months of right 
side otalgia. He had used antibiotics (cephalexin, 
moxifloxacin e gatifloxacin) without improve-
ment, and started having very sharp pain. The 
exam showed signs of otitis media, with EAC 
thickening, without ulceration. CT scan (July 
2004) showed right side mastoid and middle 
ear opacification. Audiometry showed mixed 
hypoacusis. 
Thinking of mastoiditis, the patient 
was treated with ciprofloxacin for 21 days and 
evolved with symptoms remission.
In October of 2004, otalgia returned and 
a new CT scan showed a rupture in the mastoid’s 
external cortical bone.
The patient was submitted to mastoidec-
tomy and the anatomical study revealed  chronic 
unspecific inflammation and positive culture for 
Pseudomonas. The patient was started on cipro-
floxacin 1500 mg/daily for 6 weeks.
Diabetes compensation, pain control 
and epithelization of the canal wall down cavity 
was the treatment result. That is why we did not 
order scintigraphy for initial control. 
He returned after 4 months complaining 
of headache and tongue paralysis (Figure 1). 
He was admitted and a new CT scan revealed 
contrast uptake from the petrous apex to the 
retropharynx, and opacification of the canal 
wall down cavity. Technetium scintigraphy was 
non-conclusive.
Right side mastoid cavity biopsy and 
culture, together with that from the retropharynx 
through nasal endoscopy revealed unspecific in-
flammatory process in a pseudomonas culture.
The patient remained in the hospital 
with cefepime and amycacin for 8 weeks. He 
was discharged symptomless, with normal CT 
scan and controlled glucose levels.
10 weeks afterwards, he came back 
with headaches, worsening in his dysphagia and 
contralateral hypoacusis and otorrhea. A new CT 
scan showed bilateral mastoiditis and retropha-
rynx mass extending to the oropharynx. 
Blood culture and left ear secretion cul-
ture revealed Pseudomonas and a biopsy of the 
oropharynx muscles showed unspecific chronic 
inflammatory process. The patient remained in 
the hospital for one week and was discharged 
with a central line for outpatient use of cefepime 
4g/daily for 6 months. The patient was treated 
and is under follow up for 10 months now, with 
signs of tongue reinnervation. 
DISCUSSION
Necrotizing otitis externa, for the first 
time described by Chandler, in 1968, is an  ag-
gressive infection of the external ear, involving 
the skin, cartilage and temporal bone. 
Intravenous antibiotics is the treatment 
of choice, as long as there is no osteomyelitis 
- in these cases surgery is indicated.1-3
It is very rare to see NOE involving the 
neck muscles and the contralateral mastoid.3 A 
benign outcome after skull base osteomyelitis 
is also statistically very rare, since the mortality 
rate in these cases is of 60-80%.3
REFERENCES
1. Benecke JA Jr. Management of osteomye-
litis of the skull base. Laryngoscope 
1989;99(12):1220-23.
2. Boringa JB, Hoekstra OS, Roos JW, Bertels-
mann FW. Multiple cranial nerve palsy after 
otitis externa: a case report. Clin Neurol 
Neurosurg 1995;97:332-5.
3. Rubin J, Yu VL. Malignant external otitis: insi-
ghts into pathogenesis, clinical manifestations 
and therapy. Am J Med 1998;85:391-8. 
Keywords: otitis externa, necrotizing otitis, nerve palsy.
1 Specialist in Otorhinolaryngology and Head and Neck Surgery - HC/UNICAMP; Assistant Professor of Head and Neck Surgery - Santa Casa de Salvador - Santa Izabel Hospital. Head of the Otorhi-
nolaryngology Clinics of the Naval Hospital in Salvador. Dysphagologist and Head and Neck Surgeon - NOEV/Hospital - Bahia.
2 M.S. in Surgery -UFBA and PhD in Otorhinolaryngology - USP; Otorhinolaryngology and Head and Neck Surgery Residency Program Coordinator - Santa Izabel Hospital and Head of the Santa 
Izabel Hospital - Santa Casa de Misericórdia da Bahia.
3 Resident Physician in Otorhinolaryngology - Santa Izabel Hospital - Santa Casa de Misericórdia da Bahia.
4 Resident Physician in Otorhinolaryngology - Santa Izabel Hospital - Santa Casa de Misericórdia da Bahia.
Mailing Address: Unidade de Otorrinolaringologia - Hospital Santa Izabel Praça Almeida Couto 500 Nazaré Salvador BA 40050-410.
Paper submitted to the ABORL-CCF SGP (Management Publications System) on December 17th, 2005 and accepted for publication on JuLY 20th, 2006. cod. 1652.
Rev Bras Otorrinolaringol
2007;73(4):576.
Figure 1. Tongue protrusion attempt with an infection in the 
retropharyngeal area. 


Bilateral Hipoglossal Nerve Palsy In Necrotizing Otitis Externa

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Bilateral Hipoglossal Nerve Palsy In Necrotizing Otitis Externa

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