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RELATO DE CASO
Cefaléia hipnica
Hypnic headache
Roldão Faleiro de Almeida, Inês Alice Teixeira Leão, João Bosco de Lima Gomes
Headache Outpatient Unit, Hospital da Polícia Militar de Minas Gerais,
Belo Horizonte, Minas Gerais, Brasil
Roldão FA, Leão IAT, Gomes JBL. Cefaléia hipnica: relato de caso
Migrâneas cefaléias 2007;10(1):20-23
RESUMO
Introdução
Introdução. Cefaléia hipnica é uma rara e benigna cefaléia
da terceira idade, relacionada ao sono, descrita por Raskin
em 1988. Embora a média de idade para início deste tipo de
cefaléia seja 60 anos, relataremos o caso de um homem de
36 anos de idade, portador de distúrbio bipolar e de lipoma
intraventricular, cuja cefaléia teve início quando ele possuía
29 anos. Objetivos
Objetivos. Este artigo tem como objetivo descrever um caso de cefaléia hipnica em um homem de 36 anos
de idade, portador de distúrbio bipolar e de lipoma intraventricular, e discutir as opções terapêuticas.. Métodos
Métodos. Um
caso é relatado e a literatura disponível é revisada.. Conclusão
clusão. Embora a cefaléia hipnica seja descrita como uma
modalidade de dor de cabeça exclusiva da terceira idade, há
relatos de sua ocorrência em adultos e até mesmo em crianças.
Na maioria dos casos relatados, a resposta ao carbonato de
lítio é completa ou quase completa. Outras drogas como
indometacina, prednisona e até mesmo cafeína poderiam ser
consideradas alternativas válidas. Neste caso, nós acreditamos que o fato do paciente ser portador de lipoma intraventricular seja um achado fortuito. Embora distúrbio bipolar
e cefaléia hípnica sejam considerados transtornos fásicos,
após realizar pesquisa pela PubMed, não encontramos relação entre estas duas condições.
Palavras
alavras-- chave: Cefaléia hípnica; cefaléia em salvas; distúrbio bipolar; migrânea cíclica; doença encéfalo-vascular;
lipoma intraventricular; indometacina; prednisona; carbonato
de lítio; cafeína.
ABSTRACT
Background
Background: Hypnic headache is a rare, benign and sleeprelated recurrent headache disorder of the elderly described
by Raskin in 1988. Although the mean age of onset for this
type of headache is 60 years, we report the case of a 36-year20
old man with bipolar disorder and an intraventricular lipoma
whose headache began when he was 29 years old.
Objectives: To describe a case of hypnic headache in a 36year-old man with bipolar disorder and an intraventricular
lipoma and to discuss treatment of the headache. Methods:
A case is reported and the relevant literature reviewed.
Conclusion: Although described as a headache disorder of
the elderly, this type of headache can be found in adults and
even children. Most cases report complete or nearly complete
relief following treatment with lithium carbonate, but other drugs
such as indomethacin, prednisone and even caffeine are valid
alternatives. The finding of an intraventricular lipoma in our
case may have been a chance one, and while the disorder,
like hypnic headache, is a phasic one, there is to our knowledge
(based on a search of Pub-Med) no relationship between the
two conditions.
Key words: Hypnic headache; cluster headache; bipolar
disorder; cyclical migraine; stroke; intraventricular lipoma;
indomethacin; prednisone; lithium carbonate; caffeine.
INTRODUCTION
Hypnic headache is a rare, benign and non-familial
recurrent headache disorder of the elderly that occurs only
during sleep and was described by Raskin in 1988.1 Since
then, more cases have been reported. We describe the
case of a 36-year-old male patient with an intraventricular
lipoma whose hypnic headache began when he was 29
years old. To our knowledge, this is the first case of hypnic
headache in Brazil in this age group.
Migrâneas cefaléias, v.10, n.1, p.20-23, jan./fev./mar. 2007
CEFALÉIA HIPNICA
CASE REPORT
DISCUSSION
A 36-year-old man visited our headache outpatient
unit with a seven-year history of nocturnal headaches that
began when he was 29. He described the headache as
daily, bilateral and pounding with moderate to severe
intensity. He was usually awakened by the attacks (1 or 2
per night) between 2 to 4 h after falling asleep. Identical
attacks were also reported during daytime naps.
Autonomic symptoms were denied by the patient, who
could not relate the attacks to his dreams. The patient
had not been taking any medication because the pain
improved spontaneously within 15-30 minutes.
Results of general and neurological examinations and
standard laboratory evaluation were normal, but we found
a small lesion on CT scan and MRI of the brain suggestive
of intraventricular lipoma (posterior horn of the left lateral
ventricle).
The patient had been taking oxcarbazepine (600 mg
/ day) since the age of 29 years as treatment for bipolar
disorder.
Lithium carbonate was prescribed (300 mg at
bedtime) and there was initially a good response, but
the headaches returned to their original pattern after one
month. The patient is still under our care and is taking
lithium carbonate (600 mg / day) with a partial
response.
Hypnic headache is a rare, benign headache disorder
of the elderly described by Raskin in 1988,1 who reported
the disorder in six patients (1 female and 5 males) between
the ages of 65 and 77 years. The estimated prevalence
of the disorder is 0.07%.2
This type of headache was first described as occurring
exclusively in the elderly and affecting both sexes equally.3
However, Evers et al found a majority of cases in women.4
According to their demographic data, the mean age at
onset was 63 ± 11 years (range 36 to 83 years).4 A case
of hypnic headache was recently described in a 9-yearold girl whose disorder resolved spontaneously without
treatment.5
According to The International Headache Society
criteria, during hypnic headache attacks the patient is
always awoken by a dull head pain that occurs > 15
times a month and lasts e” 15 minutes after waking. The
pain is usually mild to moderate, but severe pain is
reported by 20% of patients. These attacks are not
associated with autonomic symptoms and usually last 15
to 180 minutes, although longer-lasting attacks have been
described. The IHS criteria also state that for a headache
to be classified as hypnic it cannot be attributed to another
disorder and that intracranial disorders must be excluded.
Caffeine and lithium were effective treatments in several
reported cases.5
A case of hypnic headache was recently described in
a 9-year-old girl whose disorder resolved spontaneously
without treatment6 and in a 71-year-old man whose
headache developed two years after a stroke (pontine
infarction). In the latter case, the authors suggested that
the lesion within the pontine reticular formation might have
altered the relationship between the REM sleep generator
and the chronobiological center and caused the hypnic
headache in their patient.7
In 1997 in Brazil, Queiroz LP et al reported a case of
hypnic headache in a 59-year-old man whose headache
began when he was 57.8 We believe this was the first
Brazilian case of hypnic headache to be reported. Also in
Brazil, de Souza Carvalho D et al9 reported eight patients
(seven females and one male) with an average age of
58 years (range 51-74 years) in whom hypnic headache
was diagnosed on average six years after the headache
started.
With regard to our patient, three aspects are noteworthy: the age at onset of the headache (29 years), the
fact that the patient had bipolar disorder and the finding
Figura. MRI of the brain showing a small mass suggestive of lipoma
of the posterior horn of the left lateral ventricle
Migrâneas cefaléias, v.10, n.1, p.20-23, jan./fev./mar. 2007
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ROLDÃO FALEIRO DE ALMEIDA ET AL
of a small intraventricular lipoma. Intracranial lipomas
are rare congenital malformations associated with central nervous system anomalies such as intracranial
calcifications, agenesis, hypotrophy and even
hypertrophy of the corpus callosum, and clinical findings
such as headache and epilepsy, the most commonly
associated neurologic symptoms. 10 They more
commonly occur in the corpus callosum, with lipoma of
the cerebellopontine angle being a very rare
malformation.11 Intracranial lipomas rarely require
surgical intervention. We believe this lipoma was a chance
finding, in view of its size and topographic localization.
With regard to bipolar disorder, a search of Pub-Med
failed to reveal any relationship between this condition
and hypnic headache.
The pathophysiological mechanism of hypnic
headache is still unknown. Raskin suggested that the pain
could be associated with REM sleep and be caused by
some alteration of the biological pacemaker, which is
modulated by the serotoninergic system.1 According to
Dodick, advancing age is accompanied by cellular loss
and reduction of the hypothalamic-pineal axis activity. As
a result, nocturnal melatonin secretion is lower in the
elderly.2
The therapeutic value of lithium carbonate in treating
phasic disorders such as bipolar disorder, cluster
headache, cyclical migraine and even hypnic headache
suggests a chronobiological disorder that could be
caused by reduced melatonin secretion,2 as lithium
indirectly increases the level of this hormone. Most cases
of hypnic headache reported complete or nearly complete relief in response to lithium carbonate,4,12 but use
of this drug is limited due to its significant side effects in
the elderly.2
Flunarizine13 and indomethacin14,15 may be effective
treatments for hypnic headache and can be used as
alternatives. Their mechanism, however, is still unclear.
Prednisone is an effective and valid option because it
provides rapid and long-lasting resolution of headaches.16
A good response to melatonin and pizotifene is described
in one case report.17 Caffeine or a cup of coffee at
bedtime may also be useful.2
CONCLUSION
Although initially described as a headache disorder
occurring exclusively in the elderly, some case reports
describe hypnic headaches in adults and even children.
To our knowledge, this is the first Brazilian case of hypnic
22
headache in a male under the age of 50 years. Our patient
was a 36-year-old man whose headache began when
he was 29 years old. We believe that the intraventricular
lipoma was a chance finding, and to our knowledge
there is no relationship between hypnic headache and
bipolar disorder. Lithium carbonate is the most effective
drug, but other valid therapies include flunarizine,
indomethacin, prednisone and caffeine. In our case there
was a partial response to lithium carbonate.
ACKNOWLEDGEMENTS
The authors would like to thank Dr. Deusvenir de
Souza Carvalho (São Paulo ), Dr. Pedro André Kowacs
(Curitiba ) and Dr. Antônio Lúcio Teixeira (Belo Horizonte) for their great encouragement and guidance.
REFERENCES
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3. Newman LC, Lipton RB, Solomon S. The hypnic headache
syndrome: a benign headache disorder of the elderly. Neurology
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4. Evers S, Goadsby PJ. Hypnic headache: clinical features,
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Hypnic headache syndrome: clinical aspects of eight patients in
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10. Piovesam EJ, Tatsui CE, Kowacs PA, Prazeres RF, Lange MC,
Antoniuk SA, Werneck LC. Lipoma of the corpus callosum
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the cerebellopontine angle: case report. Arq Neuropsiquiatr
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zones: a case report. Cephalalgia 2001;21:928-931.
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CEFALÉIA HIPNICA
13. Morales AF, Mauri JA, Iniguez C, Espada F, Mostacero E. The
hypnic headache syndrome: report of three new cases.
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First italian case of hypnic headache, with literature review and
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15. Ivanez V, Solar R, Barreiro P. Hypnic headache syndrome: a case
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16. Relja J, Zorzon M, Locatelli L, Carraro N, Antonello RM, Cazzato
G. Rapid and long-lasting response to prednisone in two new
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2001;21:505-506.
NB: The patient signed an informed-consent form after receiving
verbal guidance and reading the contents of the document, which
was approved by the Ethics Committee of the Hospital Militar.
Recebido: 07/03/2007
Aceito: 29/03/2007
Endereço para correspondência
Dr. Roldão Faleiro de Almeida
Rua Castelo de Guimarães, 471 / 401, Bairro Castelo
31330-250 – Belo Horizonte, MG, Brasil
E-mail: [email protected]
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