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Migraine and tension headache in children under 6 years of age
European Journal of Pain 8 (2004) 307–314 www.EuropeanJournalPain.com
Migraine and tension headache in children under 6 years of age Umberto Balottin
a,*
, Francesca Nicoli a, Giovanni Pitillo a, Oreste Ferrari Ginevra b, Renato Borgatti c, Giovanni Lanzi d
a
d
Child Neuropsychiatry Unit, University of Insubria, Macchi Foundation Hospital, Varese 21100, Italy b University of Pavia, IRCCS Fondazione C. Mondino, Pavia, Italy c IRCCS E. Medea, Bosisio Parini, Italy Department of Child Neuropsychiatry, University of Pavia, IRCCS Fondazione C. Mondino, Pavia, Italy Received 5 May 2003; accepted 15 October 2003 Available online 14 November 2003
Abstract Objective. To investigate the clinical features of idiopathic headache with early onset, whose presence is probably underestimated by parents and physicians and the influence of environmental and psychological factors on headache in children. Methods. We report on a prospective longitudinal evaluation of 35 consecutive children referred to the Neuropsychiatry Departments of the Universities of Varese and Pavia (mean age at the first observation: 4 years and 7 months, range: 12 months–6 years; mean age at onset: 4 years and 2 months, range: 10 months–6 years) presenting with headache symptomatology. Mean duration of clinical follow-up: 9.5 months. The diagnosis based on the IHS criteria was then compared to the intuitive clinical diagnosis made in accordance with alternative case definitions. We examined our patients for the presence of early developmental disorders and interictal somatic disorders. We also studied the role of psychosocial factors at the onset and in the course of headache. Results. Diagnosis: migraine without aura in two cases, episodic tension headache in four cases, migrainous disorders not fulfilling above criteria in eight cases, headache of the tension-type not fulfilling above criteria in 12 cases and headache not classifiable in nine cases. Clinical features of headache are described in the text. Early developmental disorders (0–2 years), such as eating difficulties and sleep disorders, were detected in 18/35 children. Among patients older than 2 years, we also detected interictal somatic disorders (20 cases) such as sleep disorders, eating difficulties, enuresis and idiopathic vomiting. In 14/35 subjects, we identified psychosocial components playing a significant role at the onset of, and during, the headache. Conclusions. A better clinical definition of the disorder would make it easier to identify very young affected children and consequently to plan more specific therapeutic interventions, taking into account environmental and psychological factors. A diagnosis of idiopathic headache becomes particularly significant: according to our cases, despite their being limited in number, migraine and tension headache can be considered also as indices of individual or family related problems requiring appropriate psychiatric or psychological intervention. This stresses the need for a multidisciplinary team of specialists that would include a psychologist/ psychiatrist or headache specialist with specific training in psychiatry. Ó 2003 European Federation of Chapters of the International Association for the Study of Pain. Published by Elsevier Ltd. All rights reserved. Keywords: Migraine; Idiopathic headache; Children; Follow-up
1. Introduction The prevalence of headache ranges from 4% to 20% in preschool age, and from 38 to 50% in school-age children (Bille, 1962; Lanzi et al., 1982; Mortimer et al., 1992; Sillanpaa et al., 1991; Zuckermann et al., 1987); in *
Corresponding author. Fax: +39-332-299381. E-mail address: [email protected] (U. Balottin).
particular, the occurrence of migraine headache increases with age, its prevalence ranging from 1.4% to 3.2% in children up to the age of 7, and from 4% to 11% in school-age subjects (Lipton, 1997; Sillanpaa and Anttila, 1996). However, cases of migraine have also been reported in the early years, and even months, of life (Barlow, 1994; Chu and Shinnar, 1992; Elser and Woody, 1990; Vahlquist and Hackzell, 1949). Various considerations,
1090-3801/$30 Ó 2003 European Federation of Chapters of the International Association for the Study of Pain. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.ejpain.2003.10.004
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which include the inability of a child in preverbal age to provide an adequate description of his symptomatology, and the rarity with which migraine onset is accompanied – in the first few years of life – by the classic signs and symptoms of the disorder, suggest that the prevalence of migraine in infancy and early childhood may in fact be underestimated. Migraine onset in children can occur with a semeiology similar to the picture of the so-called ‘‘childhood periodic syndrome’’, whose symptoms include recurrent abdominal pain, cyclic vomiting, episodes of fever not apparently linked to the presence of an infectious disease process, joint pain and other symptoms (Hockaday, 1989; Lanzi et al., 1983, 1997; Li et al., 1999). Even in cases in which the ‘‘headache’’ is, as in adult sufferers, the expression of migraine, its clinical characteristics present certain peculiarities. In early childhood, in fact, the pain is often short-lasting, its location is prevalently frontal-median or bilateral, and it is often described as having a tightening-pressing quality (Prensky and Sommer, 1979; Silberstein, 1990; Winner and Martinez, 1995; Wober-Bingol et al., 1995). Furthermore, in very young children, migraine without aura is often accompanied by a less intense headache symptomatology and by a more marked gastrointestinal one: abdominalgia, nausea and vomiting (Hockaday, 1989; Barlow, 1984). Most studies of migraine in infancy are retrospective investigations, and thus present a number of methodological difficulties (Chu and Shinnar, 1992; Vahlquist and Hackzell, 1949). In our opinion, greater significance can be attributed to the two longitudinal studies (Barlow, 1994; Elser and Woody, 1990) which we found in the course of our review of the literature, particularly ElserÕs (Elser and Woody, 1990) study which was conducted on six children aged 5–42 months. This author describes – as equivalents of the adult symptomatology – manifestations such as sleep and behavioural disorders, irritability, motor difficulties, such as ataxia of gait, pallor and recurrent abdominal pain. Some authors maintain that early-onset idiopathic headaches are often present (Sillanpaa et al., 1991; Chu and Shinnar, 1992; Chaitel et al., 1995; Maytal et al., 1995; Medina et al., 1997). In contrast, others stress the prominent importance of a neuroradiological diagnostic approach, and consider idiopathic headaches very rare in this age group (Honing and Charney, 1992). Nevertheless different studies highlight the importance – from infancy – of psychological factors influencing both the onset and course of the headache or the onset of other somatic symptoms. All these are pathologies that are widely regarded as psychosomatic (Balottin et al., 1992; Kreisler et al., 1981; Sirol, 1995; Kreisler, 1995; Zeanah, 1993). We set out to study the clinical features of idiopathic headache (migraine and tension headache) with early onset (in infancy and early childhood). As the pain is short-lasting, idiopathic headache is probably underes-
timated by parents first and foremost, and by physicians too. A better clinical definition of this disorder would make it easier to identify affected children and to plan more specific therapeutic interventions. With reference to the diagnostic process, we underline the importance of emotional-psychological aspects (Kreisler et al., 1981; Sirol, 1995).
2. Methods 2.1. Subjects’ selection and follow-up Our population consisted of 35 children, under 6 years of age, consecutively referred because of headache symptomatology to the Departments of Child and Adolescent Neuropsychiatry of the Macchi Varese Hospital (University of Insubria-Varese, Italy) and IRCCS Fondazione C. Mondino (University of Pavia, Italy) between July 1996 and late July 2000. To exclude a symptomatic headache, the diagnosis was based in the first instance on the clinical evaluation, history and neurological examination of the patient. To allow more in-depth diagnostic evaluation, some of the patients received the following investigations: ophthalmologic investigation (5/35), computed tomographic scan (6/35), magnetic resonance imaging (2/35), ear, nose and throat specialistic examination (2/35), electrocardiogram (2/35), visual evoked cortical potentials (1/35), echocardiogram (1/35) and electroencephalogram (22/35). The clinical follow-up lasted on average 9.5 months and included at least four consultations consisting of clinical evaluations and diagnostic sessions performed by the same child neuropsychiatrist. 2.2. Clinical features of headache and classification The headache diagnosis and subclassifications (to one- and two-digit levels) were made according to the IHS criteria (International Headache Society, 1988). We also developed alternative case definitions (Table 6) for paediatric migraine without aura and tension headache. In particular, with regard to migraine without aura, we proposed the following criteria: (A) at least five attacks fulfilling criteria B and C; (B) the headache attacks last between few minutes and several hours; (C) at least three of the following characteristics: unilateral location; pulsating quality; moderate or severe intensity; aggravation by routine physical activity; nausea and/or vomiting; phonophobia and photophobia. As far as tension headache is concerned, we proposed a modification of IHS criterion B as follows: duration of attacks ranged from few minutes to days. The diagnosis made using the IHS criteria was then compared to the intuitive clinical diagnosis made in accordance with alternative case definitions developed
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to minimize the instances of misclassification and to increase the relevance of the IHS criteria to children. The clinical diagnosis was considered to be completely in line with the IHS criteria if agreement extended to the two-digit level, partially concordant in the presence of agreement at the one-digit level, and at complete variance if there was disagreement at the onedigit level.
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context or interpersonal conflicts, inappropriate social support or other context-related problems where the child showed some problem behaviour (Psychosocial and Environmental Problems: problems with the primary support group, problems linked to the social environment, educational and socio-economic problems).
3. Results 2.3. Other disorders (early somatic disorders and interictal somatic disorders) During the investigation of past medical history, our patients received a semi-structured interview investigating the presence of early developmental disorders (0–2 years) such as sleep and eating difficulties. We also investigated the presence of interictal somatic symptoms after two years; in particular: (1) sleep disorders defined as difficulty to fall asleep and pathological behaviour during sleep such as pavor, nightmares and sleepwalking; (2) eating disorders such as regurgitation, functional vomiting and early-onset anorexia; (3) alterations in sphincter control: enuresis, encopresis and intractable constipation; (4) ‘‘periodic syndrome’’: recurrent abdominal pain, cyclic vomiting, episodes of fever not apparently linked to the presence of an infectious disease process, joint pain and other symptoms. We distinguished between early disorders (0–2 years) and later symptoms as they are more easily influenced by primary-relation disorders. 2.4. Affective functioning styles in children We assessed the affective functioning style of children during at least two playing sessions, two clinical observations and interviews with parents. Psychological disorders were classified according to ICD-10 criteria (ICD-10 Classification of Mental and Behavioural Disorders, 1992): 10th revision of the international classification of syndromes and psychic and behavioural disorders. The ICD-10 diagnostic criteria are structured in such a way to ensure some flexibility in diagnosis even when the clinical picture is not totally clear or information is incomplete. 2.5. Psychosocial stressors Emotional stressors were investigated in each single patient according to DSM-IV criteria, that is the multiaxial assessment system of the various mental disorders (Diagnostic and Statistical Manual, 1994–1995). Stressors are defined as environmental and psychosocial factors occurring during the year preceding consultation, which can influence diagnosis, treatment and prognosis of a disorder. Therefore, they can correspond to environmental difficulties or deprivation, family-
3.1. General features A total of 35 children (18 females and 17 males) suffering from headache were studied. The mean age at the first observation was 4 years and 7 months (range: 12 months–6 years). Age at onset of the headache ranged from 10 months to 6 years (mean: 4 years and 2 months). Examination of the patientsÕ histories revealed substantially normal development, in both the pre- and post-natal periods. There was one case of perinatal distress (short-lasting transitory neonatal respiratory distress) without long-term consequences; two children were born preterm (one of them was of a twin birth). Investigation of the subjectsÕ past medical history revealed the presence, in 10 cases, of disorders embraced by the so-called ‘‘childhood periodic syndrome’’, in particular attacks of cyclic vomiting in one case, car sickness in three cases, recurrent abdominal pain in three cases and joint pain in three patients (Table 3). In one case a past diagnosis of bronchial asthma was reported, while another child, aged 10 months, had suffered an isolated episode of asthmatic bronchitis. Twenty-two subjects presented a family history of headache: maternal in 13 cases and paternal in four; both maternal and paternal in five cases. 3.2. Clinical features of headache All the children presented a headache symptomatology which was verbally expressed even by the youngest child in the sample (12 months) who, while indicating the site of pain, used simple expressions like ‘‘hurt’’ to communicate the symptoms she felt to her mother. The prevalent early expressions of symptomatology were: headache (17/35), headache and vomiting (5/35), headache and nausea (3/35), and headache and abdominal pain (4/35). Migraine sufferers presented at least five attacks, while the patients with episodic tension headache referred at least 10 previous headache episodes (less than 15 days per month). Nine out of 35 children were unable to provide a complete description of their symptomatology. In 10/35 subjects, who were clinically considered to have migraine without aura, the site of pain was mainly
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aura (IHS 1.1) in two cases and tension headache (2.1) in four cases. One case diagnosed as migraine without aura presented – in addition to the headache symptomatology – brief episodes of diplopia not associated with headache. These episodes could be interpreted as expression of an aura without migraine. Migrainous disorders not fulfilling above criteria (IHS 1.7) were reported in eight cases; headache of the tension-type not fulfilling above criteria (IHS 2.3) were referred in 12 cases, and headache not classifiable was found in nine cases. A comparison of the clinical diagnosis with IHS criteria showed complete agreement in 12 cases: six cases were diagnosed as headache not classifiable in accordance with both clinical and IHS criteria. In the remaining six cases, the intuitive clinical diagnosis was completely in line with the IHS criteria-based diagnosis (two migraine and four tension headache). Partial agreement with IHS criteria was found in 17 out of 35 subjects (six migraine and 11 tension headache). In particular, the six children with migraine fulfilled all but one of the IHS criteria for a diagnosis of migraine without aura (1.1.B). They could be classified as having a migrainous disorder not fulfilling above criteria (IHS code 1.7), thus reflecting agreement at the
bilateral (6/10 cases) and in 6/10 cases the pain was described as throbbing. In eight cases, migraine lasted less than 2 h (few minutes to 1 h); duration of the symptoms varied from 2 to 48 h in only two cases. In 6/10 cases, phonophobia and photophobia were co-present, while in 6/10 cases, migraine sufferers reported nausea or vomiting. Sixteen children were clinically considered to have tension headache: in 9/16 cases pain was described as pressing or tightening. In eight cases, headache lasted less than 30 min. Nausea was referred in 3/16 cases, while vomiting was reported in two cases. The last two cases, presenting vomiting, were clinically considered to have tension headache as they did not met completely the alternative criteria for migraine without aura (Table 6). In one case (migraine without aura), short-lasting episodes (2–3 min) of diplopia were reported, not associated with headache attacks. 3.3. Intuitive clinical diagnosis vs. IHS diagnosis In accordance with the IHS criteria (International Headache Society, 1988), all our subjects were classified as suffering from idiopathic headache: migraine without
Table 1 Clinical features of headache in children less than 6 years Site
Type
Intensity
Exercise
Duration
Phono/photophobia
Nausea/vomiting
IHS diagnosis
Patients with migraine without aura 1. ) ) 2. ) + 3. ) ) 4. + + 5. ) ) 6. ) + 7. + + 8. + 9. + ) 10. ) +
+ + + ) + + + + + +
+ + + ) + ) + + + +
+ + ) ) ) ) ) ) ) )
+ + ) + + ) + + ) )
) + + ) + + + + ) )
1.1 1.1 1.7 1.7 1.7 1.7 1.7 1.7 13 13
Patients with episodic tension headache 11. + + 12. + ) 13. + + 14. + + 15. + + 16. + ) 17. + ) 18. + ) 19. + + 20. + ) 21. + + 22. + + 23. + ) 24. + ) 25. + + 26. ) +
+ + ) + + ) + + + ) + + ) + ) +
) + ) ) + + + + + ) + + + ) ) +
+ + + + ) + ) ) + ) + + ) ) ) )
+ + + + + + + + + + ) ) + + + +
+ + + + + ) + + ) + ) + + + + +
2.1 2.1 2.1 2.1 2.3 2.3 2.3 2.3 2.3 13 2.3 2.3 2.3 2.3 2.3 2.3
Code IHS 1.1, Migraine without aura; IHS 1.7, Migrainous disorder not fulfilling above criteria; IHS 2.1, Episodic tension headache; IHS 2.3, Headache of the tension-type not fulfilling above criteria; IHS 13, Headache not classifiable; +, IHS criterion fulfilled; and ), IHS criterion not fulfilled.
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one-digit level. The 11 children with tension headache fulfilled all but one of the criteria for tension headache (2.1.D in four cases and 2.1 B in seven cases); they thus met the IHS requirements for headache of the tensiontype not fulfilling above criteria (2.3). In six children, agreement was not even found at the one-digit level. In three cases, headache was clinically considered as not classifiable; in the other three cases headache was classified as follows: migraine without aura (two cases) and episodic tension headache (one case), while according to IHS criteria, the symptoms shown by these three children met the criteria for headache not classifiable (code 13). Table 1 summarizes the clinical features of headache in 10 children with migraine without aura and in 16 children with episodic tension headache. 3.4. Follow-up information The children were monitored over time through clinical follow-ups lasting, on average, 9.5 months. The course of the headache over the study period was the following: 22/35 subjects presented spontaneous remission as early as the period following the first consultation; 5/35 presented a complete remission of the disorder in the course of the clinical follow-up (in one case, in particular, following pharmacological treatment: pizotifen). In two cases, at the last clinical evaluation, onset of bronchial asthma was reported: in one case asthma attacks appeared following a complete remission of the headache symptomatology, while in the other, they accompanied sporadic headache episodes. In 5/35 cases there was a drop-out and in 1/35 case we observed a variation of the clinical characteristics of headache, shifting from migraine without aura to tension headache. 3.5. Other disorders (early somatic disorders and interictal somatic disorders) The various interviews and our own observation of the patientsÕ behaviour allowed us to focus on several aspects of their clinical history: in 18/35 subjects early developmental disorders (0–2 years) were detected; in particular, manifestations of primary-relation disorder such as disturbed sleep-wake rhythms (10/35), idiopathic vomiting (5/35), eating problems (3/35), idiopathic colic (2/35), weaning anorexia (1/35) and affective spasms (1/35) (Table 2). Furthermore, a past diagnosis of bronchial asthma was reported in one case, while another patient had suffered an isolated episode of asthmatic bronchitis at age ten months. In many cases (20/33), in children older than 2 years (33/35), headache was not the only symptom presented by the child but was associated with sleep disorders (7/35
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Table 2 Early somatic disorders (onset before 2 years) Early somatic disorders (<2 years)
No. of cases
Idiopathic colic Idiopathic vomiting Affective spasms Eating difficulties Sleep disorders Allergic diathesis Asthmatic bronchitis Weaning anorexia P1 disorder
2/35 5/35 1/35 3/35 10/35 2/35 2/35 1/35 18/35
Table 3 Associated interictal somatic disorders (after 2 years) Somatic disorders in children >2 years (33/35)
No. of cases
Cyclic vomiting Eating difficulties Sleep disorders Recurrent abdominal pain Joint pain Car sickness Nocturnal enuresis P1 disorder
1/33 9/33 11/33 3/33 3/33 3/33 6/33 20/33
cases), eating problems (4/35), enuresis (2/35), and enuresis and sleep disorders (1/35), enuresis and idiopathic vomiting (1/35), and enuresis and eating difficulties (2/35). In 3/35 cases, the headache was accompanied both by sleep disorders and eating difficulties, such as anorexia, ‘‘faddy’’ eating and idiopathic vomiting (Table 3). 3.6. Emotional functioning (psychological disorders) On the basis of our clinical observation, play sessions and interviews with parents, we were also able to detect, in general terms, the presence of mental and behavioural disorders (ICD-10 Classification of Mental and Behavioural Disorders, 1992) (23/35 cases). Eight out of 35 children exhibited a ‘‘social anxiety disorder of childhood’’ (F93.2): a marked social inhibition and very shy and withdrawn behaviour which appeared excessive and did not lessen throughout the observation. These children were described by their parents as unwilling to talk to adults, and equally unwilling to play with their own contemporaries. In two cases we detected the presence of an ‘‘adjustment disorder with depressed mood’’ (F43.21). In both cases, depressive traits were linked to the separation of the childÕs parents. In particular, one patient presented sleep and eating disorders, extreme passivity and an insufficiently developed imagination, as reflected by the poor quality of the subjectÕs play and drawing. The other child expressed passivity, depressed mood and feelings of abandonment as shown
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by drawings. In 2/35 cases, ‘‘oppositional defiant disorder’’ was observed (F91.3), that is a tendency to exercise despotic control over parents, particularly the mother, as well as a low frustration tolerance threshold: frequent fits of anger and crying in response to limits imposed by parents; aggressive behaviour in reaction to, or rejection of, rules. A ‘‘separation anxiety syndrome’’ (F93.0) emerged in 4/35 cases: manifestations of separation anxiety when starting nursery school, trouble falling asleep and sleep disorders: Many of the children slept in the same room as their parents or often woke at night. In 2/35 cases we found a ‘‘phobic anxiety disorder of childhood’’ (F 93.1). Last, 5/35 cases exhibited the features of ‘‘other childhood emotional disorders’’ (F93.8), namely a tendency to exercise despotic control over parents in 3/35 subjects and a low frustration tolerance threshold in 2/35 cases (Table 4). 3.7. Psychosocial stressors The past medical history of 14/35 cases showed the presence of psychosocial and environmental stressors (Diagnostic and Statistical Manual, 1994–1995) (Table 5). In four cases, the onset of the disease was associated with situations of conflict within the family; in one case, in particular, it was a situation which led to separation of the parents, an event which was followed by a daily intensification of headache attacks. Complete remission of the childÕs headache was observed when he began Table 4 Mental and behavioural disorders (ICD-10) Mental and behavioural disorders
No. of cases
Social anxiety disorder of childhood (F93.2) Separation anxiety disorder of childhood (F93.0) Phobic anxiety disorder of childhood (F93.1) Oppositional defiant disorder (F91.3) Other childhood emotional disorders (F93.8) Adjustment disorder with depressed mood (F43.21) P1 disorder
8/35 4/35 2/35 2/35 5/35 2/35 23/35
Table 5 Psychosocial stressors Psychosocial stressors
No. of cases
ParentsÕ separation ParentsÕ separation + anorexia of the sister ParentsÕ separation + depression in the mother Depression in the mother New nursery school + birth of sibling Difficulties adapting to nursery school Disease of the younger sister Anorexia of the older sister ParentsÕ relational difficulties with the child (in particular of the mother) P1 stressor
2/35 1/35 1/35 2/35 1/35 1/35 1/35 1/35 4/35 14/35
Table 6 Alternative case definition Migraine without aura (A) There have been at least five attacks fulfilling criteria B and C (B) The headache attacks last between few minutes and several hours (C) At least three of the following characteristics are present: 1. unilateral location 2. pulsating quality 3. moderate or severe intensity 4. aggravation by routine physical activity 5. nausea and/or vomiting 6. phonophobia and photophobia Tension-type headache (A) There have been at least 10 attacks fulfilling criteria B and D (B) The headache attacks last from few minutes to days (C) At least two of the following characteristics: 1. pressing or tightening quality 2. mild or moderate intensity 3. bilateral location 4. no aggravation by routine physical activity (D) Both of the following apply: 1. There is no nausea or vomiting 2. There is no phonophobia and photophobia occurring together
spending the day with a neighbour. In two cases, the parentsÕ separation was associated to anorexia of the older sister, on the one hand, and depression of the mother, on the other. In another case, the headache onset occurred 6 months after the patient had moved to a new nursery school, an event which coincided with the birth of a sibling. This latter event was followed by clear manifestations of jealousy by the child who, even prior to it, had already presented separation difficulties (from his mother). In one case the onset of the disease seemed to be closely linked to the childÕs considerable difficulties adapting to nursery school. Finally, in another case headache onset occurred after the younger sister had had an infectious disease. In seven cases, family difficulties emerged which were linked to the onset of the disease; in two cases these difficulties were related to the presence of depression in the mother; in one case they concerned the presence of anorexia in the patientÕs older sister, while in four cases we detected the presence of parentsÕ relational difficulties with the child (in particular, the motherÕs). 4. Discussion and conclusions Although migraine and tension headache are frequent disorders, their presence is still underestimated in the earliest phases of childhood (Hockaday, 1989; Prensky and Sommer, 1979; Silberstein, 1990; Winner and Martinez, 1995; Wober-Bingol et al., 1995; Barlow, 1984). Equally frequent is the idiopathic nature of these
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manifestations (Sillanpaa et al., 1991; Chu and Shinnar, 1992; Chaitel et al., 1995; Maytal et al., 1995; Medina et al., 1997). Among our 35 headache sufferers, the intuitive clinical diagnosis was found to conform fully to the IHS criteria-based diagnosis in six cases; partial agreement was found in 17 cases, while absolute disagreement was found in six cases. In a sample of 72 children referred for headache, Seshia et al. (1994) found full agreement between the IHS criteria-based diagnosis and the intuitive clinical diagnosis in 61% and partial agreement in 31%. In accordance with this study, we found that partial or absolute disagreement between the intuitive clinical diagnosis and the IHS-criteria-based diagnosis can be attributable to several factors: (1) the childÕs inability to describe the features of the headache, (2) failure to meet the criterion for minimum duration and (3) the presence of migraine-like symptoms in some patients with tension headache. This feature was observed by Wober-Bingol et al., particularly in patients with headache of the tension-type not fulfilling above criteria (IHS code: 2.3) (Wober-Bingol et al., 1995). This suggests that tension and migraine-type headaches in childhood are at the extreme ends of a symptom spectrum rather than being individual entities (Viswanathan et al., 1998). On the basis of these considerations, we can conclude that the IHS criteria are probably too restrictive for the classification of headache in children. We also examined our patients for the presence of important psychopathological symptoms: past and present psychopathological disorders were found in many patients (23/35), as shown in Table 4. The frequent co-presence of other somatic disorders or psychological disorders, mainly primary mood disorders such as anxiety, depression and attachment disorders (we did not detect psychotic disorders, and only in one case we diagnosed a conduct disorder), as well as other psychosocial stressors triggering the onset of the disorder or of single attacks, led us to consider headache and migraine as pathologies in which psychological factors can play an important role. In our patients, the atmosphere at home, often overloaded of psychosocial stressors, was not conducive to parental management of psychological distress, that is maternal depression and conflict relations between the parents or their separation. The importance of psychopathological factors, although mostly of slight entity, is confirmed by the high proportion (65.7%) of psychological disorders found in our sample compared to general population. Actually, epidemiological studies conducted across several countries have shown a 20% prevalence rate for one or more psychiatric disturbances (DSM III disorders) in children aged 4–16 years, with higher rates for older patients (Anderson et al., 1987; Offord et al., 1989; Offord, 1995; Costello et al., 1988; Costello, 1989; Lavigne et al., 1996). There is only one study reporting higher preva-
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lence rates for psychopathological disorders in the general population. Forty-eight percent (48%) of participants showed at least a disorder, while about 54% of them displayed two or more psychopathological disorders. Dysthymic disorder was found to be the most prevalent disorder, followed by tic disorders, oppositional defiant disorder and conduct disorder (Langsford et al., 2001). With regard to family history in 22 patients, familiarity for headache resulted positive: maternal in 13 cases and paternal in four; both maternal and paternal in five cases. The prevalence of a family history of migraine in migraine sufferers is approximately 50–77% (Chu and Shinnar, 1992; Svensson et al., 1999; Antilla et al., 2000). Therefore, a controlled study to assess the import of familiarity as well as that of other data is necessary. Unfortunately, there is one limitation to our study, that is the small number of children comprising our sample. Also, the fact that the study population consists of patients referred because of headache to a child neuropsychiatry department may result in a biased sample which, probably, represents the severe end of the clinical spectrum and not the average child with headache. Nevertheless, our study also has some positive methodological aspects as our patients were monitored over time through a clinical follow-up. In fact, our review of the literature brought only few longitudinal studies of migraine in infancy to light. In our view, our findings confirm that even at a very early age headache can be often the expression of psychosocial distress and not necessarily a sign of an organic pathology. Nevertheless, it is necessary a carefully assessment of the possible presence of an underlying neurological condition (e.g., neoplasm, vascular disorder, systemic or focal infection, hypertensive disease (Medina et al., 1997)). Some clinical studies actually confirm that the initial neurological examination can result completely normal in subjects with neoplasm, particularly mid-line tumors, above all in children less than 4–5 years of age (Straussberg et al., 1993). In most cases, however, migraine and tension headache can also be considered as indices of an individual or family related problem requiring appropriate psychological intervention. This highlights the need for a multidisciplinary assessment carried out by a team of specialists that would include a psychologist or headache specialist with specific training in psychiatry, naturally after the assessment of the benignity of the symptom.
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Li BU, Murray RD, Heitlinger LA, Robbins JL, Hayes JR. Is cyclic vomiting syndrome related to migraine? J Pediatr 1999;134:567–72. Lipton RB. Diagnosis and epidemiology of pediatric migraine. Curr Opin Neurol 1997;10:231–6. Maytal J, Bienkowski RS, Patel M, Evital L. The value of brain neuroimaging in children with headaches. Pediatrics 1995;96(3 Part 1):413–6. Medina LS, Pinter JD, Zurakowski D, Davis RG, Kuban K, Barnes PD. Children with headache: clinical predictors of surgical space occupying lesions and the role of neuroimaging. Radiology 1997;202(3):819–24. Mortimer MJ, Kay J, Jaron A. Epidemiology of headache and childhood migraine in an urban general practice using Ad Hoc, Vahlquist and IHS criteria. Dev Med Child Neurol 1992;34:1095– 101. Offord DR, Boyle MH, Fleming JE, Blum HM, Grant NI. Ontario Child Health Study. Summary of selected results. Can J Psychiatr 1989;34(6):483–91. Offord DR. Child psychiatric epidemiology: current status and future prospects. Can J Psychiatr 1995;40(6):284–8. Prensky AL, Sommer D. Diagnosis and treatment of migraine in children. Neurology 1979;29:506–10. Seshia SS, Wolstein JR, Adams C, Booth FA, Reggin JD. International Headache Society criteria and childhood headache. Develop Med Child Neurol 1994;36:419–28. Silberstein SD. Twenty questions about headaches in children and adolescents. Headache 1990;30:716–24. Sillanpaa M, Piekkala P, Kero P. Prevalence of headaches at preschool age in an unselected child population. Cephalalgia 1991;11:239–42. Sillanpaa M, Anttila P. Increasing prevalence of headache in 7-yearold school children. Headache 1996;36:466–70. Sirol F. Les cephalees recurrents de la grande enfance et de lÕadolescence. In: Lebovici S, Diatkine R, Soule M, editors. Nouveau Traite de psychiatrie de lÕenfante et de lÕadolescent. Presses Universitaires de France: Tome II; 2nd edition, Paris, 1995, p. 1783–98. Straussberg R, Amir J, Tiqva P. Headache in children younger than 7 years: are they really benign? Arch Neurol 1993;50:130. Svensson DA, Larsson B, Bille B, Lichtenstein P. Genetic and environmental influences on recurrent headache in eight to nineyears-old twins. Cephalalgia 1999;19:866–72. The ICD-10 Classification of Mental and Behavioural Disorders (clinical description and diagnostic guidelines). World Health Organization; 1992. Vahlquist B, Hackzell G. Migraine of early onset: a study of thirty-one cases in which the disease first appeared between one and four years of age. Acta Paediatr (Stockh) 1949;38:622–36. Viswanathan V, Bridges SJ, Whitehouse W, Newton RW. Childhood headaches: discrete entities or continuum? Dev Med Child Neurol 1998;40:544–50. Winner P, Martinez W. Classification of pediatric migraine: proposed revision to the IHS criteria. Headache 1995;35:407–10. Wober-Bingol C, Wober C, Karwautz A, et al. Diagnosis of headache in childhood and adolescence: a study in 437 patients. Cephalalgia 1995;15:13–21. Zeanah CH. In: Handbook of infant mental health. The Guilford Press; 1993. p. 23. Zuckermann B, Stevenson J, Bailey V. Stomachaches and headaches in a community sample of preschool children. Pediatrics 1987;79:677– 82.
Migraine and tension headache in children under 6 years of age Umberto Balottin
a,*
, Francesca Nicoli a, Giovanni Pitillo a, Oreste Ferrari Ginevra b, Renato Borgatti c, Giovanni Lanzi d
a
d
Child Neuropsychiatry Unit, University of Insubria, Macchi Foundation Hospital, Varese 21100, Italy b University of Pavia, IRCCS Fondazione C. Mondino, Pavia, Italy c IRCCS E. Medea, Bosisio Parini, Italy Department of Child Neuropsychiatry, University of Pavia, IRCCS Fondazione C. Mondino, Pavia, Italy Received 5 May 2003; accepted 15 October 2003 Available online 14 November 2003
Abstract Objective. To investigate the clinical features of idiopathic headache with early onset, whose presence is probably underestimated by parents and physicians and the influence of environmental and psychological factors on headache in children. Methods. We report on a prospective longitudinal evaluation of 35 consecutive children referred to the Neuropsychiatry Departments of the Universities of Varese and Pavia (mean age at the first observation: 4 years and 7 months, range: 12 months–6 years; mean age at onset: 4 years and 2 months, range: 10 months–6 years) presenting with headache symptomatology. Mean duration of clinical follow-up: 9.5 months. The diagnosis based on the IHS criteria was then compared to the intuitive clinical diagnosis made in accordance with alternative case definitions. We examined our patients for the presence of early developmental disorders and interictal somatic disorders. We also studied the role of psychosocial factors at the onset and in the course of headache. Results. Diagnosis: migraine without aura in two cases, episodic tension headache in four cases, migrainous disorders not fulfilling above criteria in eight cases, headache of the tension-type not fulfilling above criteria in 12 cases and headache not classifiable in nine cases. Clinical features of headache are described in the text. Early developmental disorders (0–2 years), such as eating difficulties and sleep disorders, were detected in 18/35 children. Among patients older than 2 years, we also detected interictal somatic disorders (20 cases) such as sleep disorders, eating difficulties, enuresis and idiopathic vomiting. In 14/35 subjects, we identified psychosocial components playing a significant role at the onset of, and during, the headache. Conclusions. A better clinical definition of the disorder would make it easier to identify very young affected children and consequently to plan more specific therapeutic interventions, taking into account environmental and psychological factors. A diagnosis of idiopathic headache becomes particularly significant: according to our cases, despite their being limited in number, migraine and tension headache can be considered also as indices of individual or family related problems requiring appropriate psychiatric or psychological intervention. This stresses the need for a multidisciplinary team of specialists that would include a psychologist/ psychiatrist or headache specialist with specific training in psychiatry. Ó 2003 European Federation of Chapters of the International Association for the Study of Pain. Published by Elsevier Ltd. All rights reserved. Keywords: Migraine; Idiopathic headache; Children; Follow-up
1. Introduction The prevalence of headache ranges from 4% to 20% in preschool age, and from 38 to 50% in school-age children (Bille, 1962; Lanzi et al., 1982; Mortimer et al., 1992; Sillanpaa et al., 1991; Zuckermann et al., 1987); in *
Corresponding author. Fax: +39-332-299381. E-mail address: [email protected] (U. Balottin).
particular, the occurrence of migraine headache increases with age, its prevalence ranging from 1.4% to 3.2% in children up to the age of 7, and from 4% to 11% in school-age subjects (Lipton, 1997; Sillanpaa and Anttila, 1996). However, cases of migraine have also been reported in the early years, and even months, of life (Barlow, 1994; Chu and Shinnar, 1992; Elser and Woody, 1990; Vahlquist and Hackzell, 1949). Various considerations,
1090-3801/$30 Ó 2003 European Federation of Chapters of the International Association for the Study of Pain. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.ejpain.2003.10.004
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which include the inability of a child in preverbal age to provide an adequate description of his symptomatology, and the rarity with which migraine onset is accompanied – in the first few years of life – by the classic signs and symptoms of the disorder, suggest that the prevalence of migraine in infancy and early childhood may in fact be underestimated. Migraine onset in children can occur with a semeiology similar to the picture of the so-called ‘‘childhood periodic syndrome’’, whose symptoms include recurrent abdominal pain, cyclic vomiting, episodes of fever not apparently linked to the presence of an infectious disease process, joint pain and other symptoms (Hockaday, 1989; Lanzi et al., 1983, 1997; Li et al., 1999). Even in cases in which the ‘‘headache’’ is, as in adult sufferers, the expression of migraine, its clinical characteristics present certain peculiarities. In early childhood, in fact, the pain is often short-lasting, its location is prevalently frontal-median or bilateral, and it is often described as having a tightening-pressing quality (Prensky and Sommer, 1979; Silberstein, 1990; Winner and Martinez, 1995; Wober-Bingol et al., 1995). Furthermore, in very young children, migraine without aura is often accompanied by a less intense headache symptomatology and by a more marked gastrointestinal one: abdominalgia, nausea and vomiting (Hockaday, 1989; Barlow, 1984). Most studies of migraine in infancy are retrospective investigations, and thus present a number of methodological difficulties (Chu and Shinnar, 1992; Vahlquist and Hackzell, 1949). In our opinion, greater significance can be attributed to the two longitudinal studies (Barlow, 1994; Elser and Woody, 1990) which we found in the course of our review of the literature, particularly ElserÕs (Elser and Woody, 1990) study which was conducted on six children aged 5–42 months. This author describes – as equivalents of the adult symptomatology – manifestations such as sleep and behavioural disorders, irritability, motor difficulties, such as ataxia of gait, pallor and recurrent abdominal pain. Some authors maintain that early-onset idiopathic headaches are often present (Sillanpaa et al., 1991; Chu and Shinnar, 1992; Chaitel et al., 1995; Maytal et al., 1995; Medina et al., 1997). In contrast, others stress the prominent importance of a neuroradiological diagnostic approach, and consider idiopathic headaches very rare in this age group (Honing and Charney, 1992). Nevertheless different studies highlight the importance – from infancy – of psychological factors influencing both the onset and course of the headache or the onset of other somatic symptoms. All these are pathologies that are widely regarded as psychosomatic (Balottin et al., 1992; Kreisler et al., 1981; Sirol, 1995; Kreisler, 1995; Zeanah, 1993). We set out to study the clinical features of idiopathic headache (migraine and tension headache) with early onset (in infancy and early childhood). As the pain is short-lasting, idiopathic headache is probably underes-
timated by parents first and foremost, and by physicians too. A better clinical definition of this disorder would make it easier to identify affected children and to plan more specific therapeutic interventions. With reference to the diagnostic process, we underline the importance of emotional-psychological aspects (Kreisler et al., 1981; Sirol, 1995).
2. Methods 2.1. Subjects’ selection and follow-up Our population consisted of 35 children, under 6 years of age, consecutively referred because of headache symptomatology to the Departments of Child and Adolescent Neuropsychiatry of the Macchi Varese Hospital (University of Insubria-Varese, Italy) and IRCCS Fondazione C. Mondino (University of Pavia, Italy) between July 1996 and late July 2000. To exclude a symptomatic headache, the diagnosis was based in the first instance on the clinical evaluation, history and neurological examination of the patient. To allow more in-depth diagnostic evaluation, some of the patients received the following investigations: ophthalmologic investigation (5/35), computed tomographic scan (6/35), magnetic resonance imaging (2/35), ear, nose and throat specialistic examination (2/35), electrocardiogram (2/35), visual evoked cortical potentials (1/35), echocardiogram (1/35) and electroencephalogram (22/35). The clinical follow-up lasted on average 9.5 months and included at least four consultations consisting of clinical evaluations and diagnostic sessions performed by the same child neuropsychiatrist. 2.2. Clinical features of headache and classification The headache diagnosis and subclassifications (to one- and two-digit levels) were made according to the IHS criteria (International Headache Society, 1988). We also developed alternative case definitions (Table 6) for paediatric migraine without aura and tension headache. In particular, with regard to migraine without aura, we proposed the following criteria: (A) at least five attacks fulfilling criteria B and C; (B) the headache attacks last between few minutes and several hours; (C) at least three of the following characteristics: unilateral location; pulsating quality; moderate or severe intensity; aggravation by routine physical activity; nausea and/or vomiting; phonophobia and photophobia. As far as tension headache is concerned, we proposed a modification of IHS criterion B as follows: duration of attacks ranged from few minutes to days. The diagnosis made using the IHS criteria was then compared to the intuitive clinical diagnosis made in accordance with alternative case definitions developed
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to minimize the instances of misclassification and to increase the relevance of the IHS criteria to children. The clinical diagnosis was considered to be completely in line with the IHS criteria if agreement extended to the two-digit level, partially concordant in the presence of agreement at the one-digit level, and at complete variance if there was disagreement at the onedigit level.
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context or interpersonal conflicts, inappropriate social support or other context-related problems where the child showed some problem behaviour (Psychosocial and Environmental Problems: problems with the primary support group, problems linked to the social environment, educational and socio-economic problems).
3. Results 2.3. Other disorders (early somatic disorders and interictal somatic disorders) During the investigation of past medical history, our patients received a semi-structured interview investigating the presence of early developmental disorders (0–2 years) such as sleep and eating difficulties. We also investigated the presence of interictal somatic symptoms after two years; in particular: (1) sleep disorders defined as difficulty to fall asleep and pathological behaviour during sleep such as pavor, nightmares and sleepwalking; (2) eating disorders such as regurgitation, functional vomiting and early-onset anorexia; (3) alterations in sphincter control: enuresis, encopresis and intractable constipation; (4) ‘‘periodic syndrome’’: recurrent abdominal pain, cyclic vomiting, episodes of fever not apparently linked to the presence of an infectious disease process, joint pain and other symptoms. We distinguished between early disorders (0–2 years) and later symptoms as they are more easily influenced by primary-relation disorders. 2.4. Affective functioning styles in children We assessed the affective functioning style of children during at least two playing sessions, two clinical observations and interviews with parents. Psychological disorders were classified according to ICD-10 criteria (ICD-10 Classification of Mental and Behavioural Disorders, 1992): 10th revision of the international classification of syndromes and psychic and behavioural disorders. The ICD-10 diagnostic criteria are structured in such a way to ensure some flexibility in diagnosis even when the clinical picture is not totally clear or information is incomplete. 2.5. Psychosocial stressors Emotional stressors were investigated in each single patient according to DSM-IV criteria, that is the multiaxial assessment system of the various mental disorders (Diagnostic and Statistical Manual, 1994–1995). Stressors are defined as environmental and psychosocial factors occurring during the year preceding consultation, which can influence diagnosis, treatment and prognosis of a disorder. Therefore, they can correspond to environmental difficulties or deprivation, family-
3.1. General features A total of 35 children (18 females and 17 males) suffering from headache were studied. The mean age at the first observation was 4 years and 7 months (range: 12 months–6 years). Age at onset of the headache ranged from 10 months to 6 years (mean: 4 years and 2 months). Examination of the patientsÕ histories revealed substantially normal development, in both the pre- and post-natal periods. There was one case of perinatal distress (short-lasting transitory neonatal respiratory distress) without long-term consequences; two children were born preterm (one of them was of a twin birth). Investigation of the subjectsÕ past medical history revealed the presence, in 10 cases, of disorders embraced by the so-called ‘‘childhood periodic syndrome’’, in particular attacks of cyclic vomiting in one case, car sickness in three cases, recurrent abdominal pain in three cases and joint pain in three patients (Table 3). In one case a past diagnosis of bronchial asthma was reported, while another child, aged 10 months, had suffered an isolated episode of asthmatic bronchitis. Twenty-two subjects presented a family history of headache: maternal in 13 cases and paternal in four; both maternal and paternal in five cases. 3.2. Clinical features of headache All the children presented a headache symptomatology which was verbally expressed even by the youngest child in the sample (12 months) who, while indicating the site of pain, used simple expressions like ‘‘hurt’’ to communicate the symptoms she felt to her mother. The prevalent early expressions of symptomatology were: headache (17/35), headache and vomiting (5/35), headache and nausea (3/35), and headache and abdominal pain (4/35). Migraine sufferers presented at least five attacks, while the patients with episodic tension headache referred at least 10 previous headache episodes (less than 15 days per month). Nine out of 35 children were unable to provide a complete description of their symptomatology. In 10/35 subjects, who were clinically considered to have migraine without aura, the site of pain was mainly
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aura (IHS 1.1) in two cases and tension headache (2.1) in four cases. One case diagnosed as migraine without aura presented – in addition to the headache symptomatology – brief episodes of diplopia not associated with headache. These episodes could be interpreted as expression of an aura without migraine. Migrainous disorders not fulfilling above criteria (IHS 1.7) were reported in eight cases; headache of the tension-type not fulfilling above criteria (IHS 2.3) were referred in 12 cases, and headache not classifiable was found in nine cases. A comparison of the clinical diagnosis with IHS criteria showed complete agreement in 12 cases: six cases were diagnosed as headache not classifiable in accordance with both clinical and IHS criteria. In the remaining six cases, the intuitive clinical diagnosis was completely in line with the IHS criteria-based diagnosis (two migraine and four tension headache). Partial agreement with IHS criteria was found in 17 out of 35 subjects (six migraine and 11 tension headache). In particular, the six children with migraine fulfilled all but one of the IHS criteria for a diagnosis of migraine without aura (1.1.B). They could be classified as having a migrainous disorder not fulfilling above criteria (IHS code 1.7), thus reflecting agreement at the
bilateral (6/10 cases) and in 6/10 cases the pain was described as throbbing. In eight cases, migraine lasted less than 2 h (few minutes to 1 h); duration of the symptoms varied from 2 to 48 h in only two cases. In 6/10 cases, phonophobia and photophobia were co-present, while in 6/10 cases, migraine sufferers reported nausea or vomiting. Sixteen children were clinically considered to have tension headache: in 9/16 cases pain was described as pressing or tightening. In eight cases, headache lasted less than 30 min. Nausea was referred in 3/16 cases, while vomiting was reported in two cases. The last two cases, presenting vomiting, were clinically considered to have tension headache as they did not met completely the alternative criteria for migraine without aura (Table 6). In one case (migraine without aura), short-lasting episodes (2–3 min) of diplopia were reported, not associated with headache attacks. 3.3. Intuitive clinical diagnosis vs. IHS diagnosis In accordance with the IHS criteria (International Headache Society, 1988), all our subjects were classified as suffering from idiopathic headache: migraine without
Table 1 Clinical features of headache in children less than 6 years Site
Type
Intensity
Exercise
Duration
Phono/photophobia
Nausea/vomiting
IHS diagnosis
Patients with migraine without aura 1. ) ) 2. ) + 3. ) ) 4. + + 5. ) ) 6. ) + 7. + + 8. + 9. + ) 10. ) +
+ + + ) + + + + + +
+ + + ) + ) + + + +
+ + ) ) ) ) ) ) ) )
+ + ) + + ) + + ) )
) + + ) + + + + ) )
1.1 1.1 1.7 1.7 1.7 1.7 1.7 1.7 13 13
Patients with episodic tension headache 11. + + 12. + ) 13. + + 14. + + 15. + + 16. + ) 17. + ) 18. + ) 19. + + 20. + ) 21. + + 22. + + 23. + ) 24. + ) 25. + + 26. ) +
+ + ) + + ) + + + ) + + ) + ) +
) + ) ) + + + + + ) + + + ) ) +
+ + + + ) + ) ) + ) + + ) ) ) )
+ + + + + + + + + + ) ) + + + +
+ + + + + ) + + ) + ) + + + + +
2.1 2.1 2.1 2.1 2.3 2.3 2.3 2.3 2.3 13 2.3 2.3 2.3 2.3 2.3 2.3
Code IHS 1.1, Migraine without aura; IHS 1.7, Migrainous disorder not fulfilling above criteria; IHS 2.1, Episodic tension headache; IHS 2.3, Headache of the tension-type not fulfilling above criteria; IHS 13, Headache not classifiable; +, IHS criterion fulfilled; and ), IHS criterion not fulfilled.
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one-digit level. The 11 children with tension headache fulfilled all but one of the criteria for tension headache (2.1.D in four cases and 2.1 B in seven cases); they thus met the IHS requirements for headache of the tensiontype not fulfilling above criteria (2.3). In six children, agreement was not even found at the one-digit level. In three cases, headache was clinically considered as not classifiable; in the other three cases headache was classified as follows: migraine without aura (two cases) and episodic tension headache (one case), while according to IHS criteria, the symptoms shown by these three children met the criteria for headache not classifiable (code 13). Table 1 summarizes the clinical features of headache in 10 children with migraine without aura and in 16 children with episodic tension headache. 3.4. Follow-up information The children were monitored over time through clinical follow-ups lasting, on average, 9.5 months. The course of the headache over the study period was the following: 22/35 subjects presented spontaneous remission as early as the period following the first consultation; 5/35 presented a complete remission of the disorder in the course of the clinical follow-up (in one case, in particular, following pharmacological treatment: pizotifen). In two cases, at the last clinical evaluation, onset of bronchial asthma was reported: in one case asthma attacks appeared following a complete remission of the headache symptomatology, while in the other, they accompanied sporadic headache episodes. In 5/35 cases there was a drop-out and in 1/35 case we observed a variation of the clinical characteristics of headache, shifting from migraine without aura to tension headache. 3.5. Other disorders (early somatic disorders and interictal somatic disorders) The various interviews and our own observation of the patientsÕ behaviour allowed us to focus on several aspects of their clinical history: in 18/35 subjects early developmental disorders (0–2 years) were detected; in particular, manifestations of primary-relation disorder such as disturbed sleep-wake rhythms (10/35), idiopathic vomiting (5/35), eating problems (3/35), idiopathic colic (2/35), weaning anorexia (1/35) and affective spasms (1/35) (Table 2). Furthermore, a past diagnosis of bronchial asthma was reported in one case, while another patient had suffered an isolated episode of asthmatic bronchitis at age ten months. In many cases (20/33), in children older than 2 years (33/35), headache was not the only symptom presented by the child but was associated with sleep disorders (7/35
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Table 2 Early somatic disorders (onset before 2 years) Early somatic disorders (<2 years)
No. of cases
Idiopathic colic Idiopathic vomiting Affective spasms Eating difficulties Sleep disorders Allergic diathesis Asthmatic bronchitis Weaning anorexia P1 disorder
2/35 5/35 1/35 3/35 10/35 2/35 2/35 1/35 18/35
Table 3 Associated interictal somatic disorders (after 2 years) Somatic disorders in children >2 years (33/35)
No. of cases
Cyclic vomiting Eating difficulties Sleep disorders Recurrent abdominal pain Joint pain Car sickness Nocturnal enuresis P1 disorder
1/33 9/33 11/33 3/33 3/33 3/33 6/33 20/33
cases), eating problems (4/35), enuresis (2/35), and enuresis and sleep disorders (1/35), enuresis and idiopathic vomiting (1/35), and enuresis and eating difficulties (2/35). In 3/35 cases, the headache was accompanied both by sleep disorders and eating difficulties, such as anorexia, ‘‘faddy’’ eating and idiopathic vomiting (Table 3). 3.6. Emotional functioning (psychological disorders) On the basis of our clinical observation, play sessions and interviews with parents, we were also able to detect, in general terms, the presence of mental and behavioural disorders (ICD-10 Classification of Mental and Behavioural Disorders, 1992) (23/35 cases). Eight out of 35 children exhibited a ‘‘social anxiety disorder of childhood’’ (F93.2): a marked social inhibition and very shy and withdrawn behaviour which appeared excessive and did not lessen throughout the observation. These children were described by their parents as unwilling to talk to adults, and equally unwilling to play with their own contemporaries. In two cases we detected the presence of an ‘‘adjustment disorder with depressed mood’’ (F43.21). In both cases, depressive traits were linked to the separation of the childÕs parents. In particular, one patient presented sleep and eating disorders, extreme passivity and an insufficiently developed imagination, as reflected by the poor quality of the subjectÕs play and drawing. The other child expressed passivity, depressed mood and feelings of abandonment as shown
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by drawings. In 2/35 cases, ‘‘oppositional defiant disorder’’ was observed (F91.3), that is a tendency to exercise despotic control over parents, particularly the mother, as well as a low frustration tolerance threshold: frequent fits of anger and crying in response to limits imposed by parents; aggressive behaviour in reaction to, or rejection of, rules. A ‘‘separation anxiety syndrome’’ (F93.0) emerged in 4/35 cases: manifestations of separation anxiety when starting nursery school, trouble falling asleep and sleep disorders: Many of the children slept in the same room as their parents or often woke at night. In 2/35 cases we found a ‘‘phobic anxiety disorder of childhood’’ (F 93.1). Last, 5/35 cases exhibited the features of ‘‘other childhood emotional disorders’’ (F93.8), namely a tendency to exercise despotic control over parents in 3/35 subjects and a low frustration tolerance threshold in 2/35 cases (Table 4). 3.7. Psychosocial stressors The past medical history of 14/35 cases showed the presence of psychosocial and environmental stressors (Diagnostic and Statistical Manual, 1994–1995) (Table 5). In four cases, the onset of the disease was associated with situations of conflict within the family; in one case, in particular, it was a situation which led to separation of the parents, an event which was followed by a daily intensification of headache attacks. Complete remission of the childÕs headache was observed when he began Table 4 Mental and behavioural disorders (ICD-10) Mental and behavioural disorders
No. of cases
Social anxiety disorder of childhood (F93.2) Separation anxiety disorder of childhood (F93.0) Phobic anxiety disorder of childhood (F93.1) Oppositional defiant disorder (F91.3) Other childhood emotional disorders (F93.8) Adjustment disorder with depressed mood (F43.21) P1 disorder
8/35 4/35 2/35 2/35 5/35 2/35 23/35
Table 5 Psychosocial stressors Psychosocial stressors
No. of cases
ParentsÕ separation ParentsÕ separation + anorexia of the sister ParentsÕ separation + depression in the mother Depression in the mother New nursery school + birth of sibling Difficulties adapting to nursery school Disease of the younger sister Anorexia of the older sister ParentsÕ relational difficulties with the child (in particular of the mother) P1 stressor
2/35 1/35 1/35 2/35 1/35 1/35 1/35 1/35 4/35 14/35
Table 6 Alternative case definition Migraine without aura (A) There have been at least five attacks fulfilling criteria B and C (B) The headache attacks last between few minutes and several hours (C) At least three of the following characteristics are present: 1. unilateral location 2. pulsating quality 3. moderate or severe intensity 4. aggravation by routine physical activity 5. nausea and/or vomiting 6. phonophobia and photophobia Tension-type headache (A) There have been at least 10 attacks fulfilling criteria B and D (B) The headache attacks last from few minutes to days (C) At least two of the following characteristics: 1. pressing or tightening quality 2. mild or moderate intensity 3. bilateral location 4. no aggravation by routine physical activity (D) Both of the following apply: 1. There is no nausea or vomiting 2. There is no phonophobia and photophobia occurring together
spending the day with a neighbour. In two cases, the parentsÕ separation was associated to anorexia of the older sister, on the one hand, and depression of the mother, on the other. In another case, the headache onset occurred 6 months after the patient had moved to a new nursery school, an event which coincided with the birth of a sibling. This latter event was followed by clear manifestations of jealousy by the child who, even prior to it, had already presented separation difficulties (from his mother). In one case the onset of the disease seemed to be closely linked to the childÕs considerable difficulties adapting to nursery school. Finally, in another case headache onset occurred after the younger sister had had an infectious disease. In seven cases, family difficulties emerged which were linked to the onset of the disease; in two cases these difficulties were related to the presence of depression in the mother; in one case they concerned the presence of anorexia in the patientÕs older sister, while in four cases we detected the presence of parentsÕ relational difficulties with the child (in particular, the motherÕs). 4. Discussion and conclusions Although migraine and tension headache are frequent disorders, their presence is still underestimated in the earliest phases of childhood (Hockaday, 1989; Prensky and Sommer, 1979; Silberstein, 1990; Winner and Martinez, 1995; Wober-Bingol et al., 1995; Barlow, 1984). Equally frequent is the idiopathic nature of these
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manifestations (Sillanpaa et al., 1991; Chu and Shinnar, 1992; Chaitel et al., 1995; Maytal et al., 1995; Medina et al., 1997). Among our 35 headache sufferers, the intuitive clinical diagnosis was found to conform fully to the IHS criteria-based diagnosis in six cases; partial agreement was found in 17 cases, while absolute disagreement was found in six cases. In a sample of 72 children referred for headache, Seshia et al. (1994) found full agreement between the IHS criteria-based diagnosis and the intuitive clinical diagnosis in 61% and partial agreement in 31%. In accordance with this study, we found that partial or absolute disagreement between the intuitive clinical diagnosis and the IHS-criteria-based diagnosis can be attributable to several factors: (1) the childÕs inability to describe the features of the headache, (2) failure to meet the criterion for minimum duration and (3) the presence of migraine-like symptoms in some patients with tension headache. This feature was observed by Wober-Bingol et al., particularly in patients with headache of the tension-type not fulfilling above criteria (IHS code: 2.3) (Wober-Bingol et al., 1995). This suggests that tension and migraine-type headaches in childhood are at the extreme ends of a symptom spectrum rather than being individual entities (Viswanathan et al., 1998). On the basis of these considerations, we can conclude that the IHS criteria are probably too restrictive for the classification of headache in children. We also examined our patients for the presence of important psychopathological symptoms: past and present psychopathological disorders were found in many patients (23/35), as shown in Table 4. The frequent co-presence of other somatic disorders or psychological disorders, mainly primary mood disorders such as anxiety, depression and attachment disorders (we did not detect psychotic disorders, and only in one case we diagnosed a conduct disorder), as well as other psychosocial stressors triggering the onset of the disorder or of single attacks, led us to consider headache and migraine as pathologies in which psychological factors can play an important role. In our patients, the atmosphere at home, often overloaded of psychosocial stressors, was not conducive to parental management of psychological distress, that is maternal depression and conflict relations between the parents or their separation. The importance of psychopathological factors, although mostly of slight entity, is confirmed by the high proportion (65.7%) of psychological disorders found in our sample compared to general population. Actually, epidemiological studies conducted across several countries have shown a 20% prevalence rate for one or more psychiatric disturbances (DSM III disorders) in children aged 4–16 years, with higher rates for older patients (Anderson et al., 1987; Offord et al., 1989; Offord, 1995; Costello et al., 1988; Costello, 1989; Lavigne et al., 1996). There is only one study reporting higher preva-
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lence rates for psychopathological disorders in the general population. Forty-eight percent (48%) of participants showed at least a disorder, while about 54% of them displayed two or more psychopathological disorders. Dysthymic disorder was found to be the most prevalent disorder, followed by tic disorders, oppositional defiant disorder and conduct disorder (Langsford et al., 2001). With regard to family history in 22 patients, familiarity for headache resulted positive: maternal in 13 cases and paternal in four; both maternal and paternal in five cases. The prevalence of a family history of migraine in migraine sufferers is approximately 50–77% (Chu and Shinnar, 1992; Svensson et al., 1999; Antilla et al., 2000). Therefore, a controlled study to assess the import of familiarity as well as that of other data is necessary. Unfortunately, there is one limitation to our study, that is the small number of children comprising our sample. Also, the fact that the study population consists of patients referred because of headache to a child neuropsychiatry department may result in a biased sample which, probably, represents the severe end of the clinical spectrum and not the average child with headache. Nevertheless, our study also has some positive methodological aspects as our patients were monitored over time through a clinical follow-up. In fact, our review of the literature brought only few longitudinal studies of migraine in infancy to light. In our view, our findings confirm that even at a very early age headache can be often the expression of psychosocial distress and not necessarily a sign of an organic pathology. Nevertheless, it is necessary a carefully assessment of the possible presence of an underlying neurological condition (e.g., neoplasm, vascular disorder, systemic or focal infection, hypertensive disease (Medina et al., 1997)). Some clinical studies actually confirm that the initial neurological examination can result completely normal in subjects with neoplasm, particularly mid-line tumors, above all in children less than 4–5 years of age (Straussberg et al., 1993). In most cases, however, migraine and tension headache can also be considered as indices of an individual or family related problem requiring appropriate psychological intervention. This highlights the need for a multidisciplinary assessment carried out by a team of specialists that would include a psychologist or headache specialist with specific training in psychiatry, naturally after the assessment of the benignity of the symptom.
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