[The use of Pantogam syrup in treating small children's breath holding spells]. / Primenenie preparata pantogam sirop v terapii affektivno-respiratornykh paroksizmov u detei rannego vozrasta.

AIM: To study the efficacy of pantogam syrup 10% (hopantenic acid) in the treatment of breath-holding spells (BHS). MATERIAL AND METHODS: Sixty children, aged from 2 to 4 years, with BHS were studied. The evaluation of clinical manifestations and anxiety level was performed. Results of neurophysiological examination (long-term video-EEG-monitoring) were analyzed. Children were divided into 2 groups: main, in which the patients received pantogam syrup, and control group, in which only psychological methods were used. RESULTS AND CONCLUSION: The results of the clinical and neurophysiological studies performed after the treatment, showed the clinical improvement in 73.3% of patients of the main group compared with similar data from the children in the control group (16.7%). The anxiety level significantly decreased after the neuroprotective therapy. A comparative analysis of electroencephalographic indicators demonstrated a significant (р<0.05) reduction in the power range of slow rhythms and manifestations of functional immaturity of the brain in patients of the main group. These results give evidence for the high efficacy of pantogam syrup in the treatment of children with BHS.

Piracetam in severe breath holding spells.

BACKGROUND: Breath holding spells (BHS) are apparently frightening events occurring in otherwise healthy children. Generally, no medical treatment is recommended and parental reassurance is believed to be enough, however, severe BHS can be very stressful for the parents and a pharmacological agent may be desired in some of these children. OBJECTIVE: In this prospective study aim was to determine the usefulness of piracetam as prophylactic treatment for severe BHS. METHODS: Children were recruited from Neurology Clinic in Children's Hospital, Islamabad between January 2002 to December 2004. Diagnosis of BHS was based on characteristic history and normal physical examination. Piracetam was prescribed to those children who were diagnosed as severe BHS in a dose ranging from 50-100 mg/kg/day. Iron supplements were added if hemoglobin was less than 10 gm%. Patients were seen at 2-4 weeks interval and follow-up was continued until 3 months after the cessation of drug therapy. RESULTS: Fifty-two children were enrolled in the study, 34 boys and 18 girls. Ages ranged from 4 weeks to 5 years with mean age of 17 months. In 81% of children, spells disappeared completely and in 9% frequency was reduced to less than one per month and of much lesser intensity. Prophylaxis was given for 3-6 months (mean 5) duration. CONCLUSIONS: Piracetam is an effective prophylactic treatment for severe BHS.

Cyanotic episodes in a male child with fragile X syndrome.

A 9-year-old male with a diagnosis of fragile X syndrome (FXS) was evaluated for cyanotic episodes of unknown etiology. Clinical observation revealed frequent episodes of hyperventilation lasting several minutes, only while the patient was awake. This was followed by apnea associated with cyanosis and oxygen desaturation. Polysomnogram confirmed episodic central apnea temporally associated with hypocapnia, only during the awake state. Extensive evaluation failed to reveal other neurological, cardiac, gastrointestinal, or pulmonary etiologies for the events. The clinical observations and investigations allowed us to conclude that the patient's cyanotic episodes were caused by primary behavioral hyperventilation in the awake state. Similar behaviors have been reported in children with a variety of diagnoses but to our knowledge have not been previously reported in children with FXS. Treatment for this unusual behavior in FXS consists of reassurance and behavior modification to decrease the frequency and severity of the cyanotic episodes.

[Voluntary apnea of maximum duration as a model of short-term asphyxia in psycho- and electrophysiological studies].

We evaluated the test with arbitrary external respiratory standstill (AEKS) of maximum duration as a model of short-term asphyxia. The experiments were carried out in volunteer students. The level of oxyhemoglobin saturation and tissue oxygen tension that we registered in a group of sportsmen in the course of AEKS of maximum duration demonstrated that asphyxia developed in some of the probationers within 100-120 seconds. Taking into consideration the fact that registration of many psychophysiological and electrophysiological parameters takes place within the interval from several hundreds of milliseconds to several tens of seconds AEKS of maximum duration may be regarded as an adequate model of short-term asphyxia.

Cardiorespiratory events detected by home memory monitoring and one-year neurodevelopmental outcome.

OBJECTIVE: To determine if infants with cardiorespiratory events detected by home memory monitoring during early infancy have decreased neurodevelopmental performance. STUDY DESIGN: Infants (n = 256) enrolled in the Collaborative Home Infant Monitoring Evaluation also completed the Bayley Scales of Infant Development II at 92 weeks' postconceptional age. Infants were classified as having 0, 1 to 4, or 5+ cardiorespiratory events. Events were defined as apnea >or=20 seconds or heart rate <60 to 80 bpm or <50 to 60 bpm, for >or=5 to 15 seconds, depending on age. RESULTS: For term infants, having 0, 1 to 4, and 5+ cardiorespiratory events was associated with unadjusted mean Mental Developmental Index (MDI) values (+/-SD) of 103.6 (10.6), 104.2 (10.7), and 97.7 (10.9), respectively, and mean Psychomotor Developmental Index (PDI) values of 109.5 (16.6), 105.8 (16.5), and 100.2 (17.4). For preterm infants, having 0, 1 to 4, and 5+ cardiorespiratory events was associated with unadjusted mean MDI values of 100.4 (10.3), 96.8 (11.5), and 95.8 (10.6), respectively, and mean PDI values of 91.7 (19.2), 93.8 (15.5), and 94.4 (17.7). The adjusted difference in mean MDI scores with 5+ events compared with 0 events was 5.6 points lower in term infants ( P = .03) and 4.9 points lower in preterm infants ( P = .04). CONCLUSIONS: Having 5+ conventional events is associated with lower adjusted mean differences in MDI in term and preterm infants.

Breath-holding spells in somatoform disorder.

Breath-holding spells (BHS) are commonly seen in childhood. However, there are no case reports of BHS occurring in adolescents or young adults. We report two young adult cases and discuss the pathogensis, both physically and psychologically. BHS occurred for 1-2 minutes after hyperventilation accompanied by cyanosis in both cases. Oxygen saturation was markedly decreased. Each patient had shown distress and a regressed state psychologically. These cyanotic BHS occurred after hyperventilation, and we considered that a complex interplay of hyperventilation followed by expiratory apnea increased intrathoracic pressure and respiratory spasm. Breath-holding spells can occur beyond childhood.

Isolated mental developmental delay in very low birth weight infants: association with prolonged doxapram therapy for apnea.

OBJECTIVE: We investigated factors associated with isolated mental delay in infants weighing < 1250 g at birth. STUDY DESIGN: With a case-control design, matching variables for 40 cases included gestation, birth weight, sex, grade of intraventricular hemorrhage, and socioeconomic status. Case subjects had a mental developmental index < 70, and controls had a mental developmental index > or = 85, according to the Bayley Scales of Infant Development II at 18 months' corrected age. RESULTS: There were no differences between the case and control subjects for neonatal complications and antenatal or postnatal steroid use. There was a marked difference in the cumulative dosage and duration of doxapram therapy used for apnea of prematurity (total dose 2233 +/- 1927 mg vs 615 +/- 767 mg, P < .001; duration 45.2 +/- 32.5 days vs 19.4 +/- 23.4 days, P < .001 for case subjects and control subjects, respectively). Multivariate analysis did not identify additive predictive variables. CONCLUSION: Isolated mental delay in infants weighing < 1250 g at birth was associated with the total dosage and duration of doxapram therapy for severe apnea. Although this may be a marker for cerebral dysfunction manifesting as apnea of prematurity, possible adverse effects of doxapram or its preservative, benzyl alcohol, on the developing brain deserve further study.

Prolonged generalized epileptic seizures triggered by breath-holding spells.

We report a 3-year-old female with anoxic-epileptic seizures. Beginning at 11 months of age, she had repeated breath-holding spells with transition into generalized tonic-clonic seizures or status epilepticus. Interictal electroencephalography exhibited no abnormalities. A multidisciplinary diagnostic approach revealed a severely disturbed mother-daughter relationship that was the trigger of the breath-holding spells. Psychotherapy for the mother and daughter led to cessation of the breath-holding spells and, consequently, of the anoxic-epileptic seizures. Her further development was largely normal. We discuss the etiology and treatment of anoxic-epileptic seizures. This case is the first reported case of anoxic epileptic seizures that responded to psychologic rather than antiepileptic treatment. We advocate an initial psychologic assessment to help determine the appropriate treatment in children with recurrent anoxic-epileptic seizures.

Assessment of stress in mothers of children with severe breath-holding spells.

BACKGROUND: Severe breath-holding spells (BHS) in children consist of a stereotypical sequence of provocation to cry, noiseless expiration, color change, and loss of consciousness. Parenting a child who exhibits BHS is likely more stressful than parenting a healthy child, owing to this additional medical concern. It is also likely more stressful than parenting a child with a convulsive seizure disorder (SD), because it is often not recognized or misdiagnosed as behavioral rather than medical. Mothers of children with BHS were hypothesized to suffer significantly greater stress than mothers of children with SD or mothers of children without any significant medical conditions. OBJECTIVES: To examine maternal stress in parenting a child who exhibits BHS, and to identify the specific areas of stress impact, compared with mothers of children with a convulsive SD and mothers of control children. METHODS: The Parenting Stress Index and questions regarding how mothers coped were individually administered to 34 mothers of children with BHS, 16 mothers of children with SD, and 16 mothers of children with no medical conditions (controls). RESULTS: Mothers of children with BHS or SD experience more overall stress and disruption in their attachment or understanding of their child, compared with control mothers. These groups of mothers (BHS and SD) also perceived their child as more distractible/hyperactive, less adaptive, and more demanding than did control mothers. However, mothers of the BHS group alone showed significant disruption in their sense of competence as a parent, maintaining self-identity, and receiving positive reinforcement from their child. Mothers of children with SD showed a similar trend, but it was not significant. These findings were not related to maternal health or feelings of depression/isolation, insufficient spousal support, child's mood, or other life stresses. CONCLUSION: Parenting a child with BHS or SD impacts a greater degree of life stress on mothers of these children than does parenting control children, although the stress is greater for the BHS group than for the SD group.

Air swallowing in Rett syndrome.

The possible causes of excessive swallowing of air leading to bloating, which is common in Rett syndrome (RS), were investigated during feeding and at rest. Seven individuals with RS aged between 4 and 33 years (three with air bloat) underwent feeding videoflouroscopy and concurrent respiration monitoring. The results were compared with a randomly selected group of 11 individuals, aged between 2 and 16 years, with quadriplegic cerebral palsy and feeding problems, some of whom had mild air bloat. All individuals from both groups had isolated pharyngeal swallows and several mouth breathed; this may account for some air swallowing but not the severe air bloat characteristic of RS. Thirty-three individuals with RS aged between 3 and 44 years were monitored for nasal respiration, chest movements, swallowing, and vocal cord position at rest (between feeding). Twenty had air bloat, 17 of whom swallowed air during breath-holding in the same way, and three gulped air during hyperventilation. Of the 13 without air bloat, eight did not have recurrent breath-holding and five did, but without concurrent air swallowing. Several methods for reducing air swallowing in apnoea were investigated. The most successful was a dummy with an air leak, but this was poorly tolerated and could only be used for short periods of time. Apnoeas and air bloat are often worse when individuals are distressed and may in some individuals be reduced by anxiolytic medications.

[Hypochromic anemia in children with affective breath-holding spells]. / Hipohromna anemija kod djece sa respiratornim afektivnim krizama.

Breath holding attacks are most common in children aged 6 months to 6 years, in 76% of cases between 6 and 18 months of age. Very often they are misinterpreted as tonic epileptic seizures. They are provoked by frustration, anger or sudden injury. Child starts to cry, then holds the breath at the end of expirium. After a few seconds it becomes cyanotic, and losses consciousness. It is usually floppy, but sometimes stiffness, and clonic seizures can be present, and child can be diagnosed as having epilepsy. The form in which child is pale is less frequent, and crying is usually brief or even absent in this type. Breath holding attacks usually do not last more then one to three minutes. Good heteroanamnesis is essential for diagnosis, revealing provoking factors for each attack. Interictal EEG registration is usually normal. Attacks often spontaneously cease after 5 or 6 years of age, and do not require any medical treatment. In more severe cases behavioral therapy has shown good results. It has been noticed that those children in adolescence have syncope more frequent then rest of population. Seventeen children (12 male and 5 female) were investigated at Pediatric Hospital in Sarajevo as breath holding attacks in period from June 1997 to June 2000. Age of patients was between 5 months and 5.5 years (median was 11 months). Hypochromic anemia was present in 12 patients (76%), with average hemoglobin value of 8.2 g/dl (5.9-11.0 g/dl). All children had normal EEG recording. Iron therapy gave positive response in 8 out of 9 patients that were followed (88.9%). Three patients had not come for follow up. It is concluded that hypochromic anemia is often a part of clinical presentation of breath holding attacks in children, and iron therapy can stop them.

Parenting of at-risk infants in the face of uncertainty: home apnea monitoring of subsibs.

This article describes the experiences of a group of parents in New Zealand who lost infants to sudden infant death syndrome (SIDS) and who monitored their subsequent infants or subsibs (infants born after the death of an infant due to SIDS) at home for signs of apnea. Their caregiving experiences are explored within the framework of the substantive theory developed by Cohen (1993) that describes how another group of parents, those caring for children with chronic life-threatening illnesses, copes with living under conditions of sustained uncertainty. Attention is drawn to the similarities in both the grieving processes and coping strategies used by both groups of parents in these parallel situations.

Voluntary breath holding in panic and generalized anxiety disorders.

OBJECTIVE: Because breath holding causes arterial pCO2 to increase, we used it to test the hypothesis that in panic disorder (PD) a biological suffocation monitor is pathologically sensitive. METHOD: Nineteen patients with PD, 17 with generalized anxiety disorder (GAD), and 22 normal controls took deep breaths on signal and held them until a release signal was given 30 seconds later. This was repeated 12 times separated by 60-second normal breathing periods. RESULTS: PD patients reported having had in the past more symptoms of shortness of breath when anxious, and more frequent frightening suffocation experiences than the other groups. However, increases in self-rated anxiety between periods of normal breathing and periods of breath holding were similar in all three groups. Skin conductance, blood pressure, and T-wave amplitude reactions to breath holdings were also similar, but heart rate acceleration upon taking a deep breath was greater in GAD patients. Before and after individual breath holdings, end-tidal pCO2 was lower in PD patients than in normal controls; GAD patients were intermediate. Inspiratory flow rate did not differ between groups. CONCLUSIONS: Our physiological results provide no direct support for an overly sensitive suffocation alarm system in PD. Lower pCO2 may be due to anxiety causing hyperventilation in patients prone to panic.