Dados do Trabalho

Título

Clarithromycin Treatment in Kleine-Levin Syndrome: two case reports.

Introdução

Kleine-Levin syndrome is a rare syndrome characterized by recurrent episodes of hypersomnia lasting from days to weeks, associated with cognitive and behavioral dysfunction, hyperphagia, and hypersexuality, interspersed with periods of normal functioning. There is no definitive treatment established. The use of clarithromycin was previously suggested, due to the GABA-A antagonistic properties.

Objetivo

Report of the effect of clarithromycin on two patients with Kleine-Levin syndrome.

Métodos

Retrospective chart review.

Resultados

Both patients were evaluated at HC-FMUSP-RP. Case 1: first episode of hypersomnia and hyperphagia occurred at 16 years old. The clarithromycin was started on 9th day of symptoms. There was gradual clinical improvement with suspension of medication after 32 days. Three years later there was a new episode, associated with hypersomnia, hyperphagia and mental confusion, with spontaneous improvement after 2 days. After 3 years, a new hypersomnia episode was associated to cognitive dysfunction. Clarithromycin was started on day 13, with gradual symptomatic improvement. Clarithromycin was maintained for 60 days until resolution of symptoms. Case 2: first episode at age 16, with monthly recurrent episodes of hypersomnia associated with hyperphagia, cognitive dysfunction, and anterograde amnesia. After introduction of methylphenidate, the patient remained symptom-free for 1.5 years, when he had a new episode after methylphenidate withdrawal. Due to the lack of response to methylphenidate during this new episode, it was stopped on day 8. After 13 days period, the condition spontaneously resolved. Two months later, a new episode of hypersomnolence, psychomotor slowing and hyperphagia was registered. Clarithromycin was started on the 7th day, with total improvement after the third dose. The patient discontinued it the next day. After one month, methylphenidate was re-started and patient is asymptomatic until the present date.

Conclusões

In our cases the response to clarithromycin was not clear-cut as previously observed. There was a response variability to clarithromycin between symptomatic episodes in case 1 and from patient-to-patient. Drug-response evaluation may be hithered by the self-limited nature of the symptomatic episodes of Kleine-Levin Syndrome. It is noteworthy the methylphenidate positive effect on symptom prevention in case 2, despite no benefit during the symptomatic period.

Palavras -chave

Kleine-Levin; hypersomnia; clarithromycin; methylphenidate.