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| CASE REPORT |
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| Year : 2015 | Volume
: 21
| Issue : 1 | Page : 61-63 |
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Mucormycosis of the temporal bone with facial nerve palsy: A rare case report
KN Sathish Kumar, Nishan
Department of ENT, MMCRI, Mysore, Karnataka, India
| Date of Web Publication | 10-Mar-2015 |
Correspondence Address:
Dr. K N Sathish Kumar
S/O Nirvanaiah, Rtd., Tahsildar, Panchavati, 5th Cross, Adarsha Nagar, Hassan - 573 201, Karnataka
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/0971-7749.152870
Mucormycosis is a serious, relatively uncommon invasive fungal infection and one of the most aggressive and lethal invasive mycoses. In the region of head and neck, it usually invades nose, paranasal sinuses, orbit, intracranial structures. Mucormycosis of temporal bone with facial palsy is by far very rare and till now only one case has been reported in the literature. [1] Our experience with a diabetic patient who developed this disease and the treatment aspects in the form of surgical debridement and amphotericin B are discussed here. Keywords: Amphotericin, Diabetes, Facial palsy, Mucormycosis
How to cite this article:
Sathish Kumar K N, Nishan. Mucormycosis of the temporal bone with facial nerve palsy: A rare case report. Indian J Otol 2015;21:61-3 |
| Introduction | |  |
Mucormycosis of temporal bone is a very rare entity. Review of literature shows only one case of facial nerve paralysis secondary to tympanic mucormycosis has been reported. But extensive involvement of pinna and temporal bone with facial nerve palsy as in our patient may be the first case report till now. For this reason, it is very unique in its aspect and it will be the first such case to be reported in the literature.
| Case Report | | |
A 50-year-old elderly lady presented with h/o left ear discharge since 1-year scanty, mucopurulent, foul smelling. She also had dull aching pain and blackish discoloration [Figure 1] of the left ear since 2 months and facial asymmetry since 3 weeks. Diagnosed as diabetic 15 years back but not taking medication since 5 years. | Figure 1: Necrosed left pinna and external auditory canal filled with slough
Click here to view |
On examination, her left pinna was necrosed with slough [Figure 2] and external auditory canal was filled with blackish debris obscuring the vision of tympanic membrane with left facial nerve palsy, lower motor neuron type of HB grade 4 [Figure 3]. Tuning fork test showed conductive hearing loss on the left side. Nose and paranasal sinuses and oral cavity were normal on examination.
Laboratory data on admission were as follows: HB - 8.6 g%, total count - 13,438/mm 3 , random blood sugar - 312 mg/dl, fasting blood sugar - 220 mg/dl, X-ray mastoid showed relatively decreased pneumatization on left side: X-ray chest and paranasal sinuses were normal. High-resolution computed tomography temporal bone showed soft tissue attenuation in the left middle ear and mastoid with erosion of facial bony canal near 2 nd genu. Intracranial structures and orbit were normal.
Patient was started with higher antibiotics and her blood sugar was controlled with human insulin. After 2 days, surgical debridement of the wound followed by modified radical mastoidectomy and facial nerve decompression was done.
Intraoperative findings include brownish, soft mass filling middle ear, and mastoid [Figure 4]. There was erosion of facial bony canal near 2 nd genu. All the ossicles were absent except the footplate of stapes.
Modified radical mastoidectomy was completed with debridement of all devitalized tissue completely [Figure 5]. | Figure 5: Complete disease removal both from mastoid and middle ear before performing facial nerve decompression
Click here to view |
There was a significant improvement in eye closure immediately after surgery [Figure 6].
Histopathological report showed thin-walled, broad, nonseptate, haphazardly branching at right angled with bullous tip on periodic acid Schiff stain [Figure 7]. | Figure 7: Broad, nonseptate hyphae, branching at right angled (PAS stain)
Click here to view |
Liposomal amphotericin-B was started 15 mg/kg/day. Regular monitoring of creatinine clearance and hemogram was done. Blood sugar level was under control. After 2 weeks of antibiotics and antifungal treatment, patient was referred to the plastic surgeon for reconstruction of pinna.
| Discussion | | |
Mucormycosis of the temporal bone is a very rare entity. Review of literature shows only one case of facial nerve paralysis secondary to tympanic mucormycosis has been reported. But extensive involvement of pinna and temporal bone with facial nerve palsy as in our patient may be the first case report till now.
We reviewed various sources to find out exact pathogenesis of primary temporal bone mucormycosis but none of them gives reliable pathogenesis. Direct spread through the Eustachian tube More Details or through the vascular channels may be considered to explain the possible pathway of spread into temporal bone since the fungus might be carried in the nose and nasopharynx of a healthy person. The spread of the fungus is unique in that it has a great affinity for arteries. It penetrates their tough muscular walls, grows within the lumina and stimulates acute arteritis or thrombosis. [2],[3] Later, it invades veins and lymphatics and sets in the characteristic lesions of a combination of infarction and inflammation. In diabetes, [4] especially with elevated blood sugar levels and acidemia, there will be a favorable environment for the spores to germinate and hyphae production. Facial nerve was subsequently compromised by this invasive infection.
Without affecting nose and paranasal sinuses how the infection involved only temporal bone? Whether it is a cutaneous mucormycosis of external ear invaded the middle ear and mastoid or evolution of new subtypes? Answer for these questions are still uncertain. With the use of Amphotericin B, incidence of morbidity and mortality associated with this life-threatening infection has come down significantly. [5]
Three factors are key to a successful outcome of therapy for mucormycosis: [6]
- Reversal of the underlying predisposition
- Aggressive surgical debridement
- Aggressive antifungal therapy with early initiation and high drug doses.
| Acknowledgment | | |
Dr. K. M. Govinde Gowda, Professor and HOD, Department of ENT, MMCRI Hospital staff, PG students, Family and Friends and Almighty God.
| References | | |
| 1. | Yun MW, Lui CC, Chen WJ. Facial paralysis secondary to tympanic mucormycosis: Case report. Am J Otol 1994;15:413-4.  |
| 2. | Bodenstein NP, Mcintosh WA, Vlantis AC. Clinical signs of orbital ischemia in rhino-orbitocerebral mucormycosis. Laryngoscope 1993;103:1357-61. |
| 3. | Bhattacharyya AK, Deshpande AR, Nayak SR, Kirtane MV, Ingle MV, Vora IM. Rhinocerebral mucormycosis: An unusual case presentation. J Laryngol Otol 1992;106:48-9. |
| 4. | Petrikkos G, Skiada A, Lortholary O. Epidemiology and clinical manifestations of mucormycosis. Oxf J Med Clin Infect Dis 2012;54 Suppl 1:S23-34. |
| 5. | Hazarika P, Zachariah J, Victor J, John M, Devi C, Abraham P. Mucormycosis of the middle ear: a case report with review of literature. Indian J Otolaryngol Head Neck Surg 2012;64:90-4. |
| 6. | Sugar AM. Mucormycosis. Harrison's Principles of Internal Medicine 17 th ed., Ch. 198. p. 1261-2. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]
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