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Paroxysmal tonic upgaze: physiopathological considerations in three additional cases.
J Child Neurol. 2000 Jan; 15(1):15-8.JC

Abstract

Paroxysmal tonic upgaze of childhood has been described as a benign distinctive syndrome of abnormal ocular movement, with or without concomitant ataxia. After the first observation of four children, a further 29 patients have been reported with a wide spectrum of neurologic abnormalities such as ataxia, unsteady of gait, learning disabilities and mental retardation at follow-up. Electroencephalograms were normal in all the subjects and magnetic resonance imaging showed deficient myelination in only one patient. Recently it has been suggested that paroxysmal tonic upgaze could be a heterogeneous syndrome, ranging from a simply age-dependent manifestation to a clinical appearance of a variety of disorders affecting the corticomesencephalic loop of vertical eye movement. Moreover, it also could be an early sign of more widespread neurologic dysfunction. We describe three patients who presented paroxysmal tonic upgaze; in one, ataxia was present; in the second child, ataxia and language disorder also were observed; and in the third patient paroxysmal tonic upgaze was associated with loss of muscle tone (drop-attack-like events). On magnetic resonance imaging, a pinealoma compressing the dorsal mesencephalic region was detected. On the basis of our observations, we suggest that any insult with periaqueductal mesencephalic gray-matter involvement could be considered the basic condition for this peculiar clinical manifestation.

Authors+Show Affiliations

Pediatric Department, University La Sapienza, Rome, Italy.No affiliation info availableNo affiliation info available

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

10641603

Citation

Spalice, A, et al. "Paroxysmal Tonic Upgaze: Physiopathological Considerations in Three Additional Cases." Journal of Child Neurology, vol. 15, no. 1, 2000, pp. 15-8.
Spalice A, Parisi P, Iannetti P. Paroxysmal tonic upgaze: physiopathological considerations in three additional cases. J Child Neurol. 2000;15(1):15-8.
Spalice, A., Parisi, P., & Iannetti, P. (2000). Paroxysmal tonic upgaze: physiopathological considerations in three additional cases. Journal of Child Neurology, 15(1), 15-8.
Spalice A, Parisi P, Iannetti P. Paroxysmal Tonic Upgaze: Physiopathological Considerations in Three Additional Cases. J Child Neurol. 2000;15(1):15-8. PubMed PMID: 10641603.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Paroxysmal tonic upgaze: physiopathological considerations in three additional cases. AU - Spalice,A, AU - Parisi,P, AU - Iannetti,P, PY - 2000/1/21/pubmed PY - 2000/3/4/medline PY - 2000/1/21/entrez SP - 15 EP - 8 JF - Journal of child neurology JO - J Child Neurol VL - 15 IS - 1 N2 - Paroxysmal tonic upgaze of childhood has been described as a benign distinctive syndrome of abnormal ocular movement, with or without concomitant ataxia. After the first observation of four children, a further 29 patients have been reported with a wide spectrum of neurologic abnormalities such as ataxia, unsteady of gait, learning disabilities and mental retardation at follow-up. Electroencephalograms were normal in all the subjects and magnetic resonance imaging showed deficient myelination in only one patient. Recently it has been suggested that paroxysmal tonic upgaze could be a heterogeneous syndrome, ranging from a simply age-dependent manifestation to a clinical appearance of a variety of disorders affecting the corticomesencephalic loop of vertical eye movement. Moreover, it also could be an early sign of more widespread neurologic dysfunction. We describe three patients who presented paroxysmal tonic upgaze; in one, ataxia was present; in the second child, ataxia and language disorder also were observed; and in the third patient paroxysmal tonic upgaze was associated with loss of muscle tone (drop-attack-like events). On magnetic resonance imaging, a pinealoma compressing the dorsal mesencephalic region was detected. On the basis of our observations, we suggest that any insult with periaqueductal mesencephalic gray-matter involvement could be considered the basic condition for this peculiar clinical manifestation. SN - 0883-0738 UR - https://www.unboundmedicine.com/medline/citation/10641603/Paroxysmal_tonic_upgaze:_physiopathological_considerations_in_three_additional_cases_ L2 - https://journals.sagepub.com/doi/10.1177/088307380001500103?url_ver=Z39.88-2003&rfr_id=ori:rid:crossref.org&rfr_dat=cr_pub=pubmed DB - PRIME DP - Unbound Medicine ER -