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Journal of Neurological Sciences (Turkish)
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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

  • Top
  • Summary
  • Introduction
  • Case Presentation
  • Discussion
  • References
  • Introduction
    Facial nerve paralysis ( prosopoplgia ) is a facial neuropathy commonly seen with Bell’s 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.
  • Top
  • Summary
  • Introduction
  • Case Presentation
  • Disscussion
  • References
  • 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 Bell’s 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)


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    Figure 1: Direct postero-anterior chest x-ray performed at presentation revealing hilar, mediastinal and paratracheal lymphadenopathy (arrows).


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    Figure 2: A Many small, well circumscribed granulomas (arrow) (H&E x100). B. Typical noncaseating hard granuloma with Langhans' type giant cell (H&E x400)

    Since he had dry eyes bilaterally so that eyes closed therapy with artificial tears and vitamin replacement were commenced. Corticosteroid therapy wasn’t 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.

  • Top
  • Summary
  • Introduction
  • Case Presentation
  • Disscussion
  • References
  • 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.

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  • Summary
  • Introduction
  • Case Presentation
  • Discussion
  • References
  • References

    1) Chapelon C, Ziza JM, Piette JC. Neurosarcoidosis: signs, course and treatment in 35 confirmed cases. Medicine 1990;69:261-276.

    2) Colover J. Sarcoidosis with involvement of the nervous system. Brain 1948;71:451-454

    3) Delany P: Neurologic manifestations of sarcoid : review of the literature with a review of 23 cases. Ann Intern Med 1977;87(3):336-45

    4) Friedman JH, Challenor Y, Brust JCM . Bilateral facial paralysis associated with Stevens-Jhonson syndrome. Neurol 1979;29 :1304-1306.

    5) George MK, Pahor AL. Sarcoidosis: a cause for bilateral facial palsy. Ear Nose Throat J. 1991:70;492-3.

    6) Hauser WA, Karnes WE, Annis J. Incidence and prognosis of Bell’s Palsy in the population of Rochester, Minnesota. Mayo Clin Proc 1971;46:258-264.

    7) Haydar AA, Hujairi NM, Tawil A, Savaya R. Bilateral facial paralysis: what's the cause? MJA 2003; 179 (10):553.

    8) Hoitsma E, Faber CG, Drent M, Sharma OP. Neurosarcoidosis: a clinical dilemma. Lancet Neurol. 2004;3(7):397-407

    9) James DG, Sharma OP. Neurological complications of sarcoidosis. J R Soc Med 1967;62:1169-1170

    10) Katusic SK, Beard CM, Wiederholt WC. Incidence clinical features, and prognosis in Bell’s palsy, Rochester, Minnesota,1968-1982. Ann Neurol 1986;20:622- 627.

    11) Lambert V, Richards SH. Facial palsy in Heerfordt’s syndrome. J Laryng Otol 1964;78:684-693.

    12) Lower EE, Broderick JP, Brott TG. Baughman RP. Diagnosis and management of neurological sarcoidosis.Arch Intern Med. 1997;157(16):1864-8.

    13) Matthews WB. Sarcoidosis of the nervous system. J Neurol Neurosurg Psychiatry 1965;28 :23-9.

    14) McIntosh WE, Brenner JF, Aschenbrenner JE. Bilateral facial paralysis as the sole presenting feature of sarcoidosis: report of a case. J Am Osteopath Assoc. 1987 Mar; 87(3) 245-7

    15) Oki M, Takizawa S, Ohnuki Y. MRI findings of VIIth cranial nerve involvement in sarcoidosis. Br J Radiol 1997;70:859-861

    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

    18) Sharma OP, Sharma AM. Sarcoidosis of the nervous system. A clinical Approach. Arch Intern Med 1991;151(7):1317-21

    19) Sharma SK, Mohan A. Uncommon manifestations of sarcoidosis. J Assoc Physicians India. 2004 Mar;52:210-4.

    20) Sharma OP. Neurosarcoidosis: a personal perspective based on the study of 37 patients. Chest 1997;112(1):220-8.

    21) Silverstein A, Feuer MM, Siltzbach LE. Neurologic sarcoidosis. Arch Neurol 1965;12:1-11.

    22) Stern BJ, Krumholz A, Johns C. Sarcoidosis and its neurological manifestations. Arch Neurol. 1985;42(9):909-17.

    Recived by:  Aug 29.2005
    Revised by: Sept 02.2005
    Accepted:   Sept 06.2005

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  • Summary
  • Introduction
  • Case Presentation
  • Discussion
  • References
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