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Holmes-Adie syndrome and Lyme disease

2001; Elsevier BV; Volume: 357; Issue: 9258 Linguagem: Inglês

10.1016/s0140-6736(05)71234-4

1474-547X

Raphael B. Stricker, Edward E. Winger,

Sympathectomy and Hyperhidrosis Treatments

Paolo Martinelli (Nov 18, p 1760)1Martinelli P Holmes-Adie syndrome.Lancet. 2000; 356: 1760-1761Summary Full Text Full Text PDF PubMed Scopus (35) Google Scholar provides an excellent clinical and historical overview of Holmes-Adie syndrome (tonic pupil and areflexia), more commonly known in the USA as Adie syndrome. Although the syndrome has not been associated with “infection of conventional bacterial or viral origin”, it is occasionally linked to early syphilis, parvovirus B19, and herpes simplex virus infections.2Yasaki S Ohshima J Yonekura J Takahash Y Someya K A case of early syphilis presenting general paresis-like symptoms and bilateral tonic pupils.Rinsho Shinkeigaku. 1992; 32: 994-999PubMed Google Scholar, 3Corridan PG Laws DE Morrell AJ Murray PI Tonic pupils and human parvovirus (B19) infection.J Clin Neuroophthalmol. 1991; 11: 109-110PubMed Google Scholar, 4Anzai T Uematsu D Takahashi K Katayama T Guillain-Barre syndrome with bilateral tonic pupils.Intern Med. 1994; 33: 248-251Crossref PubMed Scopus (19) Google Scholar We now report an association with neurological Lyme disease.In a referral practice of about 140 patients with Lyme disease, we have seen three patients with predominant neurological symptoms who presented with Holmes-Adie syndrome (table). The diagnosis of Lyme disease was based on a history of tickbite, presence of an erythema migrans rash and positive serology for the spirochaete Borrelia burgdorferi. The duration of Lyme disease symptoms ranged from 2–16 years, and each patient developed a unilateral tonic pupil before the diagnosis of Lyme disease was made. All three patients had significant neuropsychiatric and cognitive defects, hyporeflexia, and facial dysaesthesia without anhydrosis. Rapid plasma reagin testing was negative in each case. Two patients had abnormal brain magnetic resonance imaging with white-matter lesions consistent with neurological Lyme disease. A decreased concentration of CD57 lymphocytes characteristic of chronic Lyme disease5Stricker RB, Winger EE. Decreased CD57 lymphocyte subset in patients with chronic Lyme disease. Immunol Lett (in press).Google Scholar was found in two patients before antibiotic therapy. Of note, Holmes-Adie syndrome persisted in each case despite intravenous antibiotic therapy and partial resolution of other neurological symptoms of Lyme disease.TableCharacteristics of patients with Lyme disease and Holmes-Adie syndromePatientAge/sexDuration of Lyme disease (years)Topic pupilCD57 (cells/μL)Brain MRIAntibiotic treatment139 F16Unilateral49*Before antibiotics.NegativeIV242 F8Unilateral31*Before antibiotics.PositiveIV339 F2Unilateral111†On antibiotics. Normal CD57 range 60–360 cells/μL.PositiveIVMRI=magnetic resonance imaging; IV=intravenous ceftriaxone.* Before antibiotics.† On antibiotics. Normal CD57 range 60–360 cells/μL. Open table in a new tab Neurological Lyme disease has been associated with various cranial nerve, meningeal, and neuropsychiatric abnormalities.5Stricker RB, Winger EE. Decreased CD57 lymphocyte subset in patients with chronic Lyme disease. Immunol Lett (in press).Google Scholar Based on our clinical observation, it seems that Holmes-Adie syndrome might represent an early and unrecognised manifestation of Lyme disease. We suggest that patients presenting with this unusual neuro-ophthalmological syndrome should be tested for the Lyme disease spirochaete. Paolo Martinelli (Nov 18, p 1760)1Martinelli P Holmes-Adie syndrome.Lancet. 2000; 356: 1760-1761Summary Full Text Full Text PDF PubMed Scopus (35) Google Scholar provides an excellent clinical and historical overview of Holmes-Adie syndrome (tonic pupil and areflexia), more commonly known in the USA as Adie syndrome. Although the syndrome has not been associated with “infection of conventional bacterial or viral origin”, it is occasionally linked to early syphilis, parvovirus B19, and herpes simplex virus infections.2Yasaki S Ohshima J Yonekura J Takahash Y Someya K A case of early syphilis presenting general paresis-like symptoms and bilateral tonic pupils.Rinsho Shinkeigaku. 1992; 32: 994-999PubMed Google Scholar, 3Corridan PG Laws DE Morrell AJ Murray PI Tonic pupils and human parvovirus (B19) infection.J Clin Neuroophthalmol. 1991; 11: 109-110PubMed Google Scholar, 4Anzai T Uematsu D Takahashi K Katayama T Guillain-Barre syndrome with bilateral tonic pupils.Intern Med. 1994; 33: 248-251Crossref PubMed Scopus (19) Google Scholar We now report an association with neurological Lyme disease. In a referral practice of about 140 patients with Lyme disease, we have seen three patients with predominant neurological symptoms who presented with Holmes-Adie syndrome (table). The diagnosis of Lyme disease was based on a history of tickbite, presence of an erythema migrans rash and positive serology for the spirochaete Borrelia burgdorferi. The duration of Lyme disease symptoms ranged from 2–16 years, and each patient developed a unilateral tonic pupil before the diagnosis of Lyme disease was made. All three patients had significant neuropsychiatric and cognitive defects, hyporeflexia, and facial dysaesthesia without anhydrosis. Rapid plasma reagin testing was negative in each case. Two patients had abnormal brain magnetic resonance imaging with white-matter lesions consistent with neurological Lyme disease. A decreased concentration of CD57 lymphocytes characteristic of chronic Lyme disease5Stricker RB, Winger EE. Decreased CD57 lymphocyte subset in patients with chronic Lyme disease. Immunol Lett (in press).Google Scholar was found in two patients before antibiotic therapy. Of note, Holmes-Adie syndrome persisted in each case despite intravenous antibiotic therapy and partial resolution of other neurological symptoms of Lyme disease. MRI=magnetic resonance imaging; IV=intravenous ceftriaxone. Neurological Lyme disease has been associated with various cranial nerve, meningeal, and neuropsychiatric abnormalities.5Stricker RB, Winger EE. Decreased CD57 lymphocyte subset in patients with chronic Lyme disease. Immunol Lett (in press).Google Scholar Based on our clinical observation, it seems that Holmes-Adie syndrome might represent an early and unrecognised manifestation of Lyme disease. We suggest that patients presenting with this unusual neuro-ophthalmological syndrome should be tested for the Lyme disease spirochaete.