ABSTRACT
BACKGROUND: Obsessive-Compulsive Disorder (OCD) is a common and chronic psychiatric disorder with significant morbidity characterized by intrusive, uncontrollable and reoccurring thoughts (i.e., obsessions) and/or ritualistic behaviours (i.e., compulsions). Conradi-Hünerman-Happle Syndrome (CHHS) is a rare inherited X-linked dominant variant of chondrodysplasia punctata, a heterogeneous group of rare bone dysplasias characterized by punctate epiphyseal calcifications of complex etiology and pathophysiology that remain to be defined. Available literature reveals a lacuna in regards to the coexistence of the entities with no clinical reports described. CASE PRESENTATION: A 12 year old female patient with diagnosis of CHHS, presents to psychiatric consultation due to aggravation of her OCD clinical picture, with aggravation of hand-washing frequency during the Covid-19 pandemic with significant functional impact. Psychopharmacological treatment aimed at OCD with Selective Serotonin Reuptake Inhibitor (SSRI) and antipsychotic was instituted with favourable, albeit partial response. CONCLUSIONS: The authors aim to describe a clinical case in which the patient presents with Conradi-Hünerman-Happle Syndrome and Obsessive-Compulsive Disorder. Clinical descriptions of CHHS and OCD are not available in the literature. Through this case description the authors aim to present a rare case as well as discuss an eventual association between etiology and/or pathophysiology of the two disorders.
Subject(s)
COVID-19 , Chondrodysplasia Punctata , Obsessive-Compulsive Disorder , Humans , Female , Child , Pandemics , Obsessive-Compulsive Disorder/complications , Obsessive-Compulsive Disorder/diagnosis , Obsessive-Compulsive Disorder/epidemiology , Compulsive Behavior/psychologyABSTRACT
O haloperidol é um antipsicótico de primeira geração e um dos medicamentos mais usados dentro dessa categoria. É um potente antagonista de ação central dos recetores de dopamina tipo 2, com baixa atividade anti alfa 1 adrenérgica e sem atividade anti-histamínica ou anticolinérgica, regularmente utilizado em casos de agitação. O angioedema é um efeito colateral especialmente grave, caracterizado por edema acentuado do tecido subcutâneo ou submucoso, e pode afetar várias partes do corpo, podendo até culminar, nos casos mais graves, em morte. Já foi descrito com administração de vários antipsicóticos, como clozapina, risperidona, ziprazidona e clorpromazina, no entanto é raro como resultado da administração de haloperidol, ocorrendo em <1% dos casos. O objetivo deste artigo é descrever o desenvolvimento de angioedema em um paciente com síndrome confusional agudo após a segunda administração de haloperidol IM para controle sintomático da agitação em contexto de serviço de urgência. [au]
Haloperidol is a first-generation antipsychotic and one of the most commonly used drugs within this category. It is a potent centrally acting antagonist of type 2 dopamine receptors, with low anti-alpha-1-adrenergic activity and no antihistaminic or anticholinergic activity, regularly used in cases of agitation. Angioedema is an especially serious side effect, characterized by marked edema of the subcutaneous or submucosal tissue, and can affect various parts of the body, and may even culminate, in the most severe cases, in death. It has already been described with the administration of several antipsychotics, such as clozapine, risperidone, ziprasidone, and chlorpromazine, but it is rare as a result of administration of haloperidol, occurring in <1% of cases. This article aims to describe the development of angioedema in a patient with acute confusional syndrome after the second administration of IM haloperidol for symptomatic control of agitation in an emergency department setting. [au]