Psychiatry Research 246 (2016) 821–826 Contents lists available at ScienceDirect Psychiatry Research journal homepage: www.elsevier.com/locate/psychres “I swear it is Tourette's!”: On functional coprolalia and other tic-like vocalizations crossmark ⁎ Christos Ganosa,b, Mark J. Edwardsc, Kirsten Müller-Vahld, a Department of Neurology, University Medical Center Hamburg-Eppendorf (UKE), Hamburg, Germany b Sobell Department of Motor Neuroscience and Movement Disorders, UCL Institute, UK c Department of Cell Sciences, St George's University of London, Cranmer Terrace, London, UK d Clinic of Psychiatry, Socialpsychiatry and Psychotherapy, Hannover Medical School, Carl-Neuberg-Str. 1, d-30625, Hannover, Germany ARTICLE INFO ABSTRACT Keywords: Coprolalia in neuropsychiatry is typically associated with tic disorders, in particular Gilles de la Tourette Coprolalia syndrome. To date, there has been no report of functional coprolalia. Here, we provide the clinical Functional neurological symptoms characteristics of 13 adolescent and adult patients with coprolalic and other functional tic-like complex Gilles de la Tourette syndrome vocalizations who, on the basis of these symptoms, were misdiagnosed with a primary tic disorder, most commonly Gilles de la Tourette syndrome. We describe similarities and highlight the differences from primary tic disorders in order to provide a pragmatic list of clinical clues that will facilitate correct diagnostic labeling and thereby treatment. Finally, we emphasize that the distinction between a primary and a functional tic disorder should rely on a combination of neuropsychiatric symptoms and signs and not on the presence of single, however striking, abnormal behaviors, such as coprolalia. 1. Introduction post-ictal phenomenon (Panunzi et al., 2013). Although the functional neuroanatomical basis of coprolalia remains unclear, the limbic Involuntary vocalizations are a well-recognised feature of tic circuitry has been suggested to play an important role in the patho- disorders. Indeed, coprolalia, the occurrence of obscene and socially physiology of coprolalic behaviors (Van Lancker and Cummings, 1999). inappropriate vocalizations without intent is seen as almost pathogno- Here we report 13 patients who presented with prominent vocaliza- monic of Gilles de la Tourette syndrome (GTS). Coprolalia has tions including coprolalia, palilalia and echolalia, some with additional significant notoriety amongst the public, even though it is a fairly movement disorders, but where specific features of clinical history and uncommon feature of GTS (lifetime prevalence of less than 20% examination are in our view not compatible with the diagnosis of GTS, (Freeman et al., 2009)). Coprolalia in GTS is independently associated and where investigations did not reveal a secondary cause. We propose with poor quality of life, tic severity, as well as a range of further that the diagnosis of these patients is most likely a functional neuropsychiatric problems, such as increased anxiety, sexually inap- neurological disorder, and we discuss the problems and potential propriate and also non-obscene socially inappropriate behaviors benefits of making this diagnosis in people with tic-like vocalizations (NOSI), obsessive-compulsive and attention-deficit hyperactivity dis- and movements. order (Eapen et al., 2016; Eddy and Cavanna, 2013a, 2013b; Freeman et al., 2009; Kobierska et al., 2014). 2. Methods However, repetitive involuntary vocalizations, including coprolalia, are not exclusively encountered in primary tic disorders. Coprolalia has All patients presented at the GTS referral clinic of one of the been reported in patients with structural brain lesions and in patients authors (KMV; Clinic of Psychiatry, Socialpsychiatry and with neurodegenerative and autoimmune disorders (Singer, 1997). Psychotherapy, Hannover Medical School) during the period of Coprolalia, alongside other complex vocal tic behaviors, such as 1995–2015. Among those, patients with predominant complex voca- palilalia, echolalia and klazomania (compulsive shouting) has been lizations, such as coprolalia and/or other prominent vocal tic-like documented in patients with post-encephalitic neuropsychiatric syn- behaviors (i.e. palilalia/echolalia/NOSI) were selected and their clinical dromes (Lees, 1985). Coprolalia has also been described as an ictal or characteristics were extracted. We selected 13 patients who in our view ⁎ Corresponding author. E-mail address: [email protected] (K. Müller-Vahl). http://dx.doi.org/10.1016/j.psychres.2016.10.021 Received 24 February 2016; Received in revised form 11 October 2016; Accepted 16 October 2016 Available online 17 October 2016 0165-1781/ © 2016 The Authors. Published by Elsevier Ireland Ltd. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by/4.0/). C. Ganos et al. Table 1 Clinical characteristics of patients with functional coprolalia and other functional complex tic-like vocalizations. GTS=Gilles de la Tourette syndrome; NOSI=Non-obscene socially inappropriate behaviors; ADHD = Attention-deficit hyperactivity disorder; OCD = Obsessive-compulsive disorder. Case Age/ Age at Modality of Symptoms Simple Complex vocalizations on Blocking Other Urge Suggestibility/ Stereotyped Other Previous Previous Number Sex onset of onset/ at onset vocalizations presentation phenomena movements on Distractibility/ nature/ functional Treatments/ Diagnosis functional Precipitants on presentation Suppressibility Fluctuations symptoms Improvement tic-like presentation Coprolalia Echolalia/ symptoms Palilalia/NOSI 1 42/ 33 Abrupt/Mild Episodic “Eh”“Ficken” Palilalia: Nonsense No Blinking, Head Yes No/Yes/Yes Yes/Yes Urge to hit None GTS M accident hand tremor “Kacken” words (“tiff”, “taff”, banging, Hitting himself “Kackeficken” “piff”) head with hand, against Episodic hand objects, tremor functional hyposmia, episodic whole-body shivering 2 10/ 5 Abrupt/ Stuttering Snuffling, “Arsch”, Palilalia: “Hilfe” (up Speech Multifocal jerky No Yes/Yes/No Yes/Yes Episodic None GTS M School coughing, “Hurensohn” to 15 fold) blocks movements double mobbing engine noise, “Vollidiot”, “fick vision moaning dich” 3 35/ 26 Abrupt/ Snuffing, None "ich will dich in Echolalia while Speech Rapid multifocal Yes No/Yes/Yes Yes/Yes Urge to look Botulinum toxin GTS M Change of blinking den Arsch watching TV blocks jerking, urge to into sun, in vocal cords/ work ficken", "du bist exert pressure on dizzy spells, Improvement due hässlich", "ich arteries and functional to hoarseness; kann dich nicht genitals hypaesthesia Aripiprazole, leiden" Quetiapine, 822 Fluoxetine/No improvement 4 17/F 14 Abrupt/ Repetition of Screaming, "fick dich", “du Immediate ambient Slurring Jerky limb Yes Yes/Yes/Yes Yes/Yes None Risperidone, GTS plus Familial a single word syllables ("ja", Hurentochter", echolalia; Palilalia: movements, Fluvoxamine/ ADHD conflict (“Korb”) "he", "paha", "du abgefickte "meine", "ok", pulling on the None "lalalalala", Schlampe", "man", "Kevin", "ich curtain (only "eh", "äh", "Hure", "Fotze", bin fertig damit", when listening to "ehm") Dreckschwein", "leider nein" songs), pinching "Hurensohn", ("Bolle" (nonsense others, knocking "fick dich", word)) against the wall, "Scheiße", peace sign, "Schlange", showing the Nutte", "ich middle finger liebe dich", "ich (only in the will ein Kind presence of von dir” others) 5 19/ 13 Abrupt, while Sticking out Gagging, Schwein, Sau, No No Bizarre and Yes Yes/Yes/Yes Yes/ Yes None Sulpiride, GTS plus M watching TV his tongue groaning Arschloch, complex whole- Lorazepam, ADHD Psychiatry Research246(2016)821–826 Noises Hure, Ficker, body movements Clonazepam, similar to Fotze, Pisskopf, with twisting and Chlorprothixene, gagging Schwuchtel, backward arching Methylphenidate, Hurensohn, of trunk Aripiprazole – all Ficklippe, without effect, but adverse effects (e. g. not able to speak under treatment with Aripiprazole ) Symptom-free when smoking (continued on next page) C. Ganos et al. Table 1 (continued) Case Age/ Age at Modality of Symptoms Simple Complex vocalizations on Blocking Other Urge Suggestibility/ Stereotyped Other Previous Previous Number Sex onset of onset/ at onset vocalizations presentation phenomena movements on Distractibility/ nature/ functional Treatments/ Diagnosis functional Precipitants on presentation Suppressibility Fluctuations symptoms Improvement tic-like presentation Coprolalia Echolalia/ symptoms Palilalia/NOSI cannabis. 6 19/F 16 Abrupt, Head “hm”, ba, ne, Fotze. “Heil Echolalia: No Copropraxia: Yes Yes/Yes/Yes Yes/Yes None Nabiximols GTS plus during jerking miaowing, he, Hitler du “Katzengulasch” Middle finger sign improves noises ADHD; inpatient ja, eo, bababa, Fotze”, “Ich (upon meeting (she can say by 30–40% No admission in hab dich nicht patient No 8) exactly that this effect with adolescent beleidigt du “Policracker”“so occurs once a Atomoxetin, psychiatry Fickfotze”, viel Sand und week) Aripiprazole, due to “Bullshit” keine Förmchen” Hitting her Tiaprid, depression, Palilalia: hand against Risperidone panic “Interessiert her head and attacks, and keinen”, “Guck chest (copied self- mal du Frettchen” upon seeing injurious other GTS behavior . patients performing these actions), head jerking, hand jerking, stamping feet on the ground 823 7 41/ 33 Abrupt, Whole body None “Schwule Sau”, NOSI: “man bist du No Rarely grimacing, Yes, Yes/Yes/Yes No/Yes None Aripiprazole/tics GTS plus M during jerks “fette Sau”, hässlich”, “ich bin