Adie's tonic pupil as a manifestation of Sjögren's syndrome

Abstract

We here report two cases of Adie's tonic pupil, associated with clinical sensory polyneuropathy and Sjögren's syndrome, in one of whom it actually heralded the onset of the syndrome. Electrophysiology studies indicated absent H reflexes but normal peripheral nerve conductions, thus suggesting an involvement of the dorsal roots or spinal ganglion that would be in line with previously published reports of dorsal ganglionitis as the primary neuropathological lesion in Sjögren's syndrome. We suggest that all cases of tonic pupils should be screened for polyneuropathy and Sjögren's syndrome.

Sommario

Descriviamo qui due casi di pupilla tonica associati con una polineuropatia sensitiva clinica e sindrome di Sjögren. Gli studi elettrofisiologici documentavano un'assenza dei riflessi H con conduzioni periferiche conservate, e suggerivano una lesione a livello delle radici dorsali o del ganglio spinale, come riscontrato nei rari studi neuropatologici dimonstranti ganglionite dorsale in corso di sindrome di Sjögren.

Suggeriamo che tutti i casi di pupilla tonica isolata siano esaminati per polineuropatia e sindrome di Sjögren.

This is a preview of subscription content, access via your institution.

References

  1. [1]

    Anzai T, Uematsu D, Takahashi K, Katayama T.Guillain-Barrè syndrome with bilateral tonic pupils. Intern Med, 1994; 33: 248–251.

    PubMed  Google Scholar 

  2. [2]

    Denislic M, Meh D.Early asymmetric neuropathy in primary Sjögren's syndrome. J Neurol, 1997; 244: 383–387.

    PubMed  Google Scholar 

  3. [3]

    Gemignani F, Marbini A, Pavesi G et al.Peripheral neuropathy associated with primary Sjögren's syndrome. J Neurol Neurosurg Psychiatry, 1994; 57: 983–986.

    PubMed  Google Scholar 

  4. [4]

    Gemignani F, Manganelli P, Pavesi G, Marbini A.Polyneuropathy in Sjögren's syndrome. A case of prevalently autonomic neuropathy with tonic pupil and hypohidrosis. Funct Neurol, 1988; 3: 337–348.

    PubMed  Google Scholar 

  5. [5]

    Griffin JW, Cornblath DR, Alexander E et al.Ataxic sensory neuropathy and dorsal root ganglionitis associated with Sjörgren's syndrome. Ann Neurol, 1990: 27: 304–315.

    PubMed  Google Scholar 

  6. [6]

    Harriman DGF, Garland H.The pathology of Adie's syndrome. Brain, 1968; 91: 401–418.

    PubMed  Google Scholar 

  7. [7]

    Kennet RP, Harding AE.Peripheral neuropathy associated with the sicca syndrome. J Neurol Neurosurg Psychiatry, 1986; 49: 90–92.

    PubMed  Google Scholar 

  8. [8]

    Malinow K, Yannakakis GD, Glusman SM et al.Subacute sensory neuronopathy secondary to dorsal root ganglionitis in primary Sjögren's syndrome. Ann Neurol, 1986; 20: 535–537.

    PubMed  Google Scholar 

  9. [9]

    Manthorpe R, Oxholm P, Prause JU, Schiodt M.The Copenhagen criteria for Sjögren's syndrome. Scand J Rheumatol, 1986; suppl. 61: 19–21.

    Google Scholar 

  10. [10]

    Mellgren SI, Conn DL, Stevens JC, Dyck PJ.Peripheral neuropathy in primary Sjögren's syndrome. Neurology, 1989; 39: 390–394.

    PubMed  Google Scholar 

  11. [11]

    Salama J, Hauw JJ, Dubas F, Giroux C, Frison A, Delaporte P.Adie's syndrome and polyneuropathy: a propos of a new case and a case of pupillotonia, polyneuropathy and generalized amyloidosis sparing the nerve. Ann Med Interne, 1984; 135: 39–42.

    Google Scholar 

  12. [12]

    Satake M, Yoshimura T, Iwaki T et al.Anti-dorsal root ganglion neuron antibody in a case of dorsal root ganglionitis associated with Sjögren's syndrome. J. Neurol Sci, 1995; 132: 122–125.

    PubMed  Google Scholar 

  13. [13]

    Sobue G, Yasuda T, Kachi T et al.Chronic progressive sensory ataxic neuropathy: clinicopathological features of idiopathic and Sjögren's syndrome-associated cases. J. Neurol, 1993; 240: 1–7.

    PubMed  Google Scholar 

  14. [14]

    Takase Y, Takahashi K, Takada K, Tatsumi H, Tabuchi Y.Hereditary motor and sensory neuropathy type I (HMNS I) associated with cranial neuropathy: an autopsy case report. Acta Neurol Scand, 1990; 82: 368–373.

    PubMed  Google Scholar 

  15. [15]

    Ulrich J.Morphological basis of Adie's syndrome. Eur Neurol, 1980; 19: 390–395.

    PubMed  Google Scholar 

  16. [16]

    Vitali C, Bombardieri S, Moutsopulos HM et al.Preliminary criteria for the classification of Sjögren's syndrome. Results of a prospective concerted action supported by the Europeen Community. Arthritis Rheum, 1993; 36: 340–347.

    PubMed  Google Scholar 

  17. [17]

    Waterschoot JM, Guerit JM, Lambert M, De Barsy T.Bilateral tonic pupil and polyneuropathy in Sjögren's syndrome: a common pathophysiological mechanism? Eur Neurol, 1991; 31: 114–116.

    PubMed  Google Scholar 

Download references

Author information

Affiliations

Authors

Rights and permissions

Reprints and Permissions

About this article

Cite this article

Vetrugno, R., Liguori, R., Cevoli, S. et al. Adie's tonic pupil as a manifestation of Sjögren's syndrome. Ital J Neuro Sci 18, 293–295 (1997). https://doi.org/10.1007/BF02083307

Download citation

Key Words

  • Adie's tonic pupil
  • Sjögren's syndrome
  • Polyneuropathy