Revista Colombiana de Psiquiatría (English ed.)
Review ArticleNeuroleptic malignant syndrome in children and adolescents: Systematic review of case reportsSíndrome neuroléptico maligno en niños y adolescentes: revisión sistemática de reportes de caso☆
Introduction
The symptoms associated with neuroleptic malignant syndrome (NMS) were first described in 1956 by Ayd, and defined as such by Delay and Deniker in 1968, when they reported an unusual response to haloperidol.1 Initially, the term neuroleptic was used to describe those psychotropic drugs that controlled psychotic symptoms and produced extrapyramidal symptoms as a side effect — in other words, first-generation antipsychotics.2 Thus, NMS was described as a set of signs and symptoms that present as a potentially severe adverse reaction, secondary to the use of first-generation antipsychotics. However, over time it has been seen that it can also occur secondary to the use of atypical antipsychotics, mood stabilisers and even medication other than psychotropic drugs.
Initially, a prevalence of around 0.2%–3% was described in 1993,3 but in recent years a decrease in this has been observed, to 0.01%–0.02%,4 possibly due to greater care in the prescription and titration of medication. Four main symptoms are usually considered for its diagnosis: hyperthermia, muscular rigidity, altered consciousness and autonomic dysfunction. Added to these are diaphoresis, tremors, incontinence, leukocytosis and elevated creatine kinase (CK). Despite this, to date there is no consensus on universal diagnostic criteria.5
Although this adverse reaction can occur at any stage of life, in any gender and in people with both psychiatric and non-psychiatric illness, some risk factors associated with its appearance have been identified, such as advanced age, male gender, polypharmacy, dehydration, malnutrition, rapid administration of antipsychotics, structural brain damage and affective disorders.1, 5 Among the main associated complications are the development of rhabdomyolysis, respiratory failure, kidney failure and sepsis, with a mortality of 5.6%. Among the predictive factors of mortality are advanced age, respiratory, renal and cardiovascular failure.6 It is, then, a widely described and studied syndrome, with various fatal complications. Despite this, there are still few studies in the child–adolescent population. Apparently, it is postulated that the course and treatment in children and adolescents is usually the same as in adults. However, more studies are needed in this regard.1, 2 For this reason, the present work aims to describe the clinical and laboratory characteristics and the treatment of child and adolescent patients with NMS, and we will also analyse the grouping of the symptoms.
Section snippets
Methods
The guidelines of the PRISMA Statement7 guide were followed. A search was carried out in the MEDLINE/PubMed search engine of all the reported cases of NMS from 1 January 2000 to 3 November 2018, entering the terms: (children OR child OR paediatric OR pediatric OR school child OR adolescents OR adolescence OR teenage) AND (case OR report OR case report OR case reports) AND (neuroleptic malignant syndrome). We selected the articles that met the following characteristics: articles written in
Results
The initial search yielded a total of 118 articles. Of these, 59 articles that were not case reports or series were discarded, leaving 59 articles with reports of cases of NMS in children and adolescents published between 1 January 2000 and 3 November 2018.1, 3, 8, 9, 10, 11, 12, 13, 14, 15, 16, 17, 18, 19, 20, 21, 22, 23, 24, 25, 26, 27, 28, 29, 30, 31, 32, 33, 34, 35, 36, 37, 38, 39, 40, 41, 42, 43, 44, 45, 46, 47, 48, 49, 50, 51, 52, 53, 54, 55, 56, 57, 58, 59, 60, 61, 62, 63, 64
From the
Discussion
The current state of knowledge of NMS is based on case reports and series, few of which were carried out in children and adolescents. This study found that more cases were reported in males, similarly to Silva et al.,2 who found a higher frequency of NMS in males (63.63%) in case reports of children and adolescents. This is probably due to this population being more frequently exposed to antipsychotics, since many of the diseases in child and adolescent psychiatry predominantly affect males.65
Conclusions
NMS in this sample was more frequent in males. The most frequently reported symptoms were: muscle rigidity, autonomic instability and fever. According to the factor analysis of symptoms, NMS could be of three types: catatonic, extrapyramidal and autonomically unstable.
Funding
Self-financed.
Conflicts of interest
The authors have no conflicts of interest to declare.
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Please cite this article as: León-Amenero D, Huarcaya-Victoria J. Síndrome neuroléptico maligno en niños y adolescentes: revisión sistemática de reportes de caso. Rev Colomb Psiquiat. 2021;50:290–300.