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Journal of Neurological Sciences (Turkish) | |||||
2005, Volume 22, Number 3, Page(s) 304-308 | |||||
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Sarcoidosis: A Cause For Bilateral Facial Palsy | |||||
Kadir KOTIL, Bekir KILINÇ | |||||
Haseki Educational and Research Hospital, Neurosurgery, İSTANBUL, Türkiye | |||||
Summary | |||||
Sarcoidosis is a systemic, chronic, granulomatous disease and it mainly affects lung. Bilateral facial nerve involvoment due to occult sarcoidosis is an extremely rare phenomenon that can be the presenting complaint in a wide spectrum of the disease.Case report. A 38 year-old man presented to our clinic with facial diplegia. His general, neurologic (except facial diplegia), ocular examination and laboratory tests were normal. There was no respiratory system symptom. Chest X-ray revealed bilateral hilar and paratracheal lymphadenopathy without atelectesia. In sarcoidosis, definitive diagnosis was made by bronco-alveoalar leavage. Corticosteroid therapy was not applied, because, he had have stage I sarcoidosis. First left sided and later right sided facial palsy detoriated and almost recovered in 7 weeks from admission. The abnormalities seen on chest x-ray resolved slowly over this period. He has been followed up in our out-patient clinic without any medication after discharge and no recurrent sign or symptoms have appeared so far. This paper describes a spesific unique case of occult facial diplegia occuring in a patient having stage I occult sarcoidosis. We believe, this phenomenon proved that facial diplegia can be created by the infiltration just only the seventh cranial nerves and the most important point to be stressed in this phenomenon should be the investigation of sarcoidosis among the facial diplegic cases | |||||
Introduction | |||||
Facial nerve paralysis ( prosopoplgia ) is a facial neuropathy commonly seen with Bells palsy having an annual incidence rate of 23 to 25 patients per 100,000 population 5 , 6 , 8 , 14 - 19. Bilateral form occurs in only 0.3 2.0 % of patients with idiopathic facial paralysis, and it differs from the more common recurrent or alternating palsy 4 , 14 - 20. Facial diplegia might rarely be the sole presenting sign of sarcoidosis a multisystemic chronic granulomatous disease of unknown etiology frequently affecting individuals 20 - 40 years of age 1 , 3 , 5 , 7 , 9 , 10 , 11 - 22. Although sarcoidosis emerges primarily in the lungs causing infiltration as a response to immune system disorder , it can affect any organ other than the lungs. Neurological manifestations occur in approximately 5 % of patients with sarcoidosis 2 , 16 and 50 % of these patients suffer from peripheral facial palsy 14. Another clinical study records that 4 % of sarcoidosis patients have neuro logical involvements, half of these have facial nerve involvement and one-sixth have facial diplegia and this contributes approximately 7 cases per 1,000 patients 2. Although cranial nerve palsy is the most common complication, its mechanism remains unclear 2 , 14. However, it is thought to be infiltration of nerve by sarcoid granuloma 15. This paper describes a spesific case of facial diplegia occuring in a patient having stage 1 occult sarcoidosis. Another dazzling point in this case is that facial diplegia was created by the infiltration just only in the seventh cranial nerve. | |||||
Case Presentation | |||||
A 38 year-old man presented to our clinic with bilateral facial diplegia. Developing on the left side one week before, and on the right three days prior to admission. He was able to close both of his eyes almost completely and demonstrated bilateral Bells phenomenon. There was no both nasal reflux of fluids on swallowing and no palatal paresis. His general examination and laboratory tests were normal. Serum angiotensin converting enzyme (ACE) level was normal. There is no other neural involvement. There was no respiratory symptom. Magnetic resonace imaging (MRI) of the brain was normal. Cerebrospinal fluid examination showed no abnnormalitiy. Chest X-ray revealed bilateral hilar and paratracheal lymphadenopathy without atelectesia (Fig. 1), proving typical characteristics for the presentation of stage I sarcoidosis. The tracheal aspiration was performed, and it was diagnosed as sarcoidosis by histopathological examination. Histopathologic examination of mediastinoscopic biopsy samples disclosed noncaseating granulomas composed of epithelioid cells with scattered lympohcytes and Langhans' type giant cells. No Schaumann bodies, asteroid bodies or calcium oxalate crystals were found (Fig 2 A&B)
Since he had dry eyes bilaterally so that eyes closed therapy with artificial tears and vitamin replacement were commenced. Corticosteroid therapy wasnt applied. First left sided and later right sided palsy further detoriated and almost recovered in 7 weeks from admission. The abnormalities seen on chest X-ray resolved slowly over this period. He has been followed up in our out-patient clinic without medication after discharge and no recurrent sign or symptoms have appeared so far. | |||||
Discussion | |||||
Leprosy, syphilis, bacterial meningitis, leukemia, sarcoidosis and Guillan-Barre syndrome are the diseases, most commonly associated with bilateral facial paralysis 5. Over all, the cranial nerves are involved in 5 % of cases of sarcoid 3 , 10. Multiple cranial nerve palsies are often present 3 with facial nerve, being the most frequently involved 2 , 3 , 10 , 16 - 21. Unilateral facial nerve involvement occurs 22-66 % of cases and bilateral involvement occurs in 11 % of cases 3 , 10 , 17. In most cases, the paralysis is only temporarily affected from the lower motor neuron origin 11 , 15. The pathogenesis is thought to be infiltration of nerve by sarcoid granuloma 11. The onset of the palsy is often sudden and the palsy may be incomplete 10. The response to steroids is thought to be favorable 3 and facial nerve decompression is indispensable only if there is no response to steroids 3,9-11,15-17. Although the course is variable, the palsy usullay resolves spontenously 1,5,7,10,16-22, as in our case report. In a case of facial diplegia, the unique most important diagnostic tool is a history and a careful full examination, as this may suggest to definitive diagnosis. Elevated serum ACE level may not elevated all patients, for thus it is not diagnostic tool 1,5,7,15-18. In conclusion, this report obtains evidence that facial diplegia in occult sarcoidosis: a) starts in suddenly, b) may fully reversible without steroid therapy, c) this phenomenon proved that facial diplegia can be created by the infiltration just only the seventh cranial nerves and the most important point to be stressed in this phenomenon should be the investigation of sarcoidosis among the facial diplegic cases. | |||||
References | |||||
2) Colover J. Sarcoidosis with involvement of the nervous system. Brain 1948;71:451-454 9) James DG, Sharma OP. Neurological complications of sarcoidosis. J R Soc Med 1967;62:1169-1170 11) Lambert V, Richards SH. Facial palsy in Heerfordts syndrome. J Laryng Otol 1964;78:684-693. 13) Matthews WB. Sarcoidosis of the nervous system. J Neurol Neurosurg Psychiatry 1965;28 :23-9. 16) Rontal E, Sigel ME. Bilateral facial pralysis. Laryngoscope 1971; 82: 607-616. 17) Scott TF. Neurosarcoidosis: progress and clinical aspects. Neurology 1993;43 (1):8-12 21) Silverstein A, Feuer MM, Siltzbach LE. Neurologic sarcoidosis. Arch Neurol 1965;12:1-11. Recived by: Aug 29.2005 | |||||
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