Background: Conversion disorder refers to functional bodily impairments that can be precipitated by high stress situations including trauma and surgery. Symptoms of conversion disorder may mimic or complicate otolaryngology diseases in the pediatric population. Case presentation: In this report, the authors describe 3 cases of conversion disorder that presented to a pediatric otolaryngology-head and neck surgery practice. This report highlights a unique population of patients who have not previously been investigated. The clinical presentation and management of these cases are discussed in detail. Non-organic otolaryngology symptoms of conversion disorder in the pediatric population are reviewed. In addition, we discuss the challenges faced by clinicians in appropriately identifying and treating these patients and present an approach to management of their care. Conclusion: In this report, the authors highlight the importance of considering psychogenic illnesses in patients with atypical clinical presentations of otolaryngology disorders. Keywords: Pediatrics, Conversion disorders, Otolaryngology, Misdiagnosis Background severity of a patient’s impairment. In pediatric patients, The prevalence of mental illness is estimated to be the presentations of conversion disorder tend to be com- 10–20% amongst children and adolescents worldwide, plex, and multiple conversion symptoms are the norm making it the leading cause of disability in young people [3–5]. As it has been found to be associated with bodily . Furthermore, treatments (both behavioral and pharma- stress, it is imperative that surgeons are aware of this cological) of mental illness and the demand for them for disorder in the post-operative setting [5–9]. Developing children and adolescents has increased significantly in an approach to this issue requires an appreciation for thepastdecade. Untreated psychiatric disorders the multifactorial nature of its etiology. can impair a child’s development and limit educational It is prudent that clinicians be informed about the achievement . prevalence of mental illness in their patient population Conversion disorders refer to body dysfunction char- and its implications. Misdiagnosis or delayed diagnosis acterized by neurological symptoms, either sensory or can have a significant impact on patients and creates a motor, that cannot be explained by a medical condition. burden not only on the healthcare system, but also on Given their somatosensory nature, they typically require the patient and their family members. In this article, the a medical assessment and the diagnosis of conversion authors discuss 3 pediatric cases referred for otolaryngo- disorder can only be established after organic diseases logic complaints that were complicated by conversion have been excluded or if they fail to account for the disorder. We discuss the implications of conversion disorder for the diagnosis and treatment for the oto- laryngologist - head and neck surgeon and the need for * Correspondence: email@example.com Faculty of Medicine, University of Ottawa, Ottawa, ON, Canada an awareness of the impact of conversion disorders on Department of Otolaryngology-Head and Neck Surgery, The Ottawa presentation, treatment and recovery. Hospital, Ottawa, ON, Canada Full list of author information is available at the end of the article © The Author(s). 2018 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Caulley et al. Journal of Otolaryngology - Head and Neck Surgery (2018) 47:39 Page 2 of 5 Case presentations as an unconscious avoidant coping mechanism. While an Patient 1 was a previously healthy 11-year-old girl who inpatient, she was followed by a multidisciplinary team presented to hospital with a 2-week history of “dizziness”. including Psychiatry, Psychology and Physiotherapy. The Her symptoms were described as disequilibrium precipi- focus of mental health interventions was on communica- tated by standing and sitting and relieved by lying flat. Her tion and expression of emotion, while Physiotherapy pro- symptoms were unaffected by eye opening. Her symptoms vided exercises to improve her symptoms and validation of were debilitating and she had difficulty ambulating. Her her psychological distress. She improved significantly over symptoms were unresponsive to antiemetics and she pre- the course of hospitalization and was discharged to sented to the Children’s Hospital of Eastern Ontario emer- outpatient follow-up through Mental Health Services for gency room where she was diagnosed with vestibular continued intervention and support. neuronitis. When her symptoms persisted, she was admit- Patient 3 was a 13-year-old boy who underwent an ted to hospital and assessed by the Otolaryngology-Head adenoidectomy. He had a past medical history of signifi- and Neck Surgery service. In addition to a history and cant nasal obstruction due to adenoid hypertrophy. His focused head and neck examination, an oto-neurological post-operative course was complicated by recurrent examination was performed including evaluation of adenoid bleeds. One month post-operatively, the patient cranial nerves, voluntary saccades, spontaneous and began to complain of daily headaches. Over the two gaze-evoked nystagmus, rapid head thrust and dix-hall weeks following, he reported daily nausea and disequilib- pike maneuver. She did not demonstrate any clinical rium. He returned to the emergency department when he findings suggestive of vestibular neuronitis, migraine developed complete paralysis of the lower limbs, essen- variant nor benign positional paroxysmal variant. tially rendering him paraplegic and disabled. A complete Routine laboratory investigations were within normal head and neck examination including oto-neurologic limits. Magnetic resonance imaging of the brain was examination was within normal limits. Routine laboratory non-contributory. She was admitted to hospital for investigations were within normal limits. An MRI and MR 4 days. She received instructions for daily strengthen- venogram of the brain failed to reveal evidence of any ing exercises from the physiotherapy service. These intracranial pathology. He remained in hospital for 17 days. interventions validated her experiences and offered a His gait progressively improved with physiotherapy until mechanism for symptom resolution that was psycho- he returned to baseline. The Mental Health service identi- logically and emotionally acceptable to the patient and fied pre-existing significant traumatic events and psycho- her family, which resulted in a complete resolution of social stressors for the entire family associated with her symptoms. A previous diagnosis of social anxiety multiple moves and living in a refugee camp for a year some 5 years ago may have been a relevant risk factor and a half prior to immigrating to Canada. In addition, the in the development of her symptoms. patient was found to have some evidence of anxiety and Patient 2 was an 11-year-old girl who presented to perfectionism that were exacerbated by his inability to hospital with a history of head trauma while somersault- participate in school, secondary to the surgery. ing 3 weeks prior to her presentation. She described progressive headaches, disequilibrium, choreiform move- Discussion ments and ataxia following the mild traumatic head To our knowledge, this is the first reported case series of injury. Her symptoms were debilitating and she was un- pediatric psychiatric disorders that presented for consider- able to sit upright or ambulate. She had no significant ation of otolaryngology-related pathology. Mental health past medical history. She was admitted to the medical ser- disorders are misdiagnosed as organic diseases more vice for 23 days, during which she was assessed by multiple frequently than clinicians expect due to several disease, subspecialties including the Otolaryngology – Head and patient and clinician factors. Furthermore, the ability to Neck Surgery service. After a complete oto-neurological accurately diagnose patients with a psychiatric illness may examination, she was found to have no evidence of a often fall outside of the scope of practice for the average vestibular pathology to account for her symptoms, and a Otolaryngologist – Head and Neck Surgeon. However, as head computed tomography (CT) and magnetic resonance highlighted in the following section, it is crucial that clini- imaging (MRI) were normal. Routine laboratory investiga- cians keep psychiatric illnesses on the differential diagno- tions were within normal limits. Further assessment from sis, especially for patients who present with atypical or Mental Health services identified the following contribut- contradictory physical signs and symptoms. In addition, ing factors: post concussive symptomatology including an approach to the management of these patients is high anxiety, high family expectations in the presence of provided as a resource for clinicians. limited communication and sibling rivalry, and the pres- Functional disorders have been linked in the adult lit- ence of a pre-existing significant traumatic event. A diag- erature with a wide breadth of head and neck com- nosis of conversion disorder was made, and conceptualized plaints, such as hearing loss, anosmia, stridor, Caulley et al. Journal of Otolaryngology - Head and Neck Surgery (2018) 47:39 Page 3 of 5 dysphonia, and vision loss [10–14]. In children and ado- including audiometry, electronystagmography and rota- lescents, the most recent literature has reported symp- tional chair testing are essential to rule out organic path- toms arising from disorders such as pseudohypacusis ology. Ancillary studies to detect non-organic pathology and functional upper airway obstruction [15–17]. Para- in children, including the Stenger Test, can be consid- doxical vocal cord motion, or psychogenic stridor, refers ered to identify pseudohypoacusis. The authors encour- to the inappropriate adduction of the vocal cords during age consideration of psychological stressors as factors to the respiratory cycle, and remains a common and fre- be considered, which may be associated with conversion, quently misdiagnosed functional disorder in the or other, disorders encountered in severe disease and in pediatric population. Over 50% of patients with paradox- the postoperative period as demonstrated in the pre- ical vocal cord motion are diagnosed with conversion sented cases. Early diagnosis and therapy can signifi- disorder . The differential diagnosis in the pediatric cantly improve health outcomes in these patients , population is challenging, given the high base rates of and the prognosis is considered to be excellent (with pediatric mental health disorders including conversion roughly 95% of affected individuals experiencing spon- disorder, adjustment disorder and autism spectrum dis- taneous resolution of their symptoms within one month order. This has the potential to be mistakenly diagnosed of diagnosis) . as primary otolaryngologic disease, as observed in this case series. A diagnostic challenge Conversion disorder (CD), or functional neurological Understanding the patient symptom disorder, is characterized by disturbances in With the support of online resources and media publicity, body function that are inconsistent with known anatomy patients now present with a plethora of computer-generated or pathophysiology . The Diagnostic and Statistical differential diagnoses and planned diagnostic investi- Manual of Mental Disorders V defines the conversion gations independent of their physician’sinput . disorder as the presence of “one or more symptoms of However, the new age of knowledgeable and autono- altered voluntary motor or sensory function” in the ab- mous patients poses both benefits and challenges to sence of any identifiable neurological or medical cause. clinicians. While the symptoms are psychogenic in origin, conver- Patients may find it challenging to accept a psychiatric sion disorder distinguishes itself from malingering and diagnosis, which is based heavily on clinical judgment, factitious disorder as the CD patient is not intentionally when they have initially become invested in the concept experiencing these symptoms [18, 19]. The patient of an organic disease as the etiology of their symptoms. population affected can be characterized as having per- The agnostic approach, where possible diagnoses and fectionist tendencies with high expectations regarding explanations are equally valid, is used by clinicians to achievements and high levels of anxiety associated with manage relaying a lack of diagnosis to a family . This illness [20–25]. There is no clear etiology of conversion approach may avoid questions about the authenticity of disorder. However, in general, theories focus on the symptoms that can contribute to a hostile physician-patient management of affect and stress [5, 18, 20–22, 24, 25]. relationship [20, 29]. Clinicians should be aware of the very There are no bedside tests or investigations to estab- sensitive and frank discussion that needs to take place with lish the diagnosis of conversion disorder. The diagnosis patients regarding the nature of their illness. Establishing a is made after organic disease has been ruled out [5, 18]. forum for discussion will also assist in providing guidance As such, diagnosing conversion disorder can put the around patient-specific therapy for this disorder . This clinician in the difficult position of having to communi- discussion, validating both physical and mental symptoms, cate the presence of a non-organic illness as the source optimally could include an open invitation for the value of of the patient’s severe disability, while validating the mental health professionals as part of the health care team. authenticity of their symptoms . However, this com- munication combined with appropriate intervention as- sists in validating symptoms and provides a mechanism Superfluous diagnostic testing for their resolution. Diagnosis of mental illness continues to rely heavily on Although it is rare for the practicing surgical specialist clinical judgment and judicious use of diagnostic testing. to encounter this disorder, its atypical sensory and motor This means that clinicians must balance patient perspec- manifestations make it a potential diagnosis in any spe- tives and values with clinical practice guidelines and cialist’s practice. For instance, these patients may find professional expertise. This can be challenging in the themselves under the care of an Otolaryngologist-Head context of unexplained symptoms and patients’ conceptu- and Neck Surgeon to rule out organic hearing loss or alizations about the symptoms, but optimally can be an op- peripheral vertigo as the etiology of their symptoms . portunity to assist the patient in accessing evidence-based Objective tests of hearing and vestibular dysfunction, resources and care. Caulley et al. Journal of Otolaryngology - Head and Neck Surgery (2018) 47:39 Page 4 of 5 The patient-physician relationship is founded heavily Table 1 Approach to the atypical ENT patient in trust from both parties. This may invoke some re- Establish broad differential diagnoses straint on the part of the clinician in opposing patient Organic diseases requests for non-beneficial investigations and procedures Non-organic diseases Pseudohypacusis . However, clinicians must be wary of the fact that Functional upper airway obstruction both patients as well as physicians can introduce biases Conversion disorder into clinical decision-making that can complicate care Adjustment disorder and increase health care costs . Brett and McCullogh [28, 31] described an approach to clinical practice in the Autism spectrum disorders context of differing interests to facilitate patient-physician Patient evaluation Thorough history and physical examination including flexible nasolaryngoscopy relationships. The authors recommend that a patient’s preference for a diagnostic or therapeutic intervention Audiometry and vestibular testing should be performed to rule out organic pathology as dictate medical decision-making only when there is a indicated by the presenting complaint modicum of potential clinical benefit. It is only if Consultation with relevant specialists including they meet this criterion that physicians proceed with neurology and psychiatry patient-selected interventions [28, 31]. Consider neuroimaging to rule out structural In addition to their patients, clinicians also have a pathology fiscal responsibility to the health care system on a soci- Treatment overview Assessment of goals with staff, patient and family etal level to ensure its sustainability and judicious use of Confirm belief in presenting symptoms its resources [28, 32]. Although cost should not prevent Avoid accusation patients from receiving optimal medical care, the fact remains that non-beneficial interventions have implica- Ensure patient and family is connected with appropriate community resources including tions for individual patients and society as a whole and physical or psychological rehabilitation should be considered essential to professional integrity Arrange follow-up visits [28, 33–35]. Eliminating waste in diagnostic interven- This should be a resource utilized by clinicians in appropriate cases where an tions, including duplicate and non-beneficial testing, can organic pathology has been ruled out reduce a significant cost burden on the health care sys- tem . At a certain threshold, challenging a patient’s request when supported by clinical evidence should not oto-neurologic examination and test of vestibular func- be misconstrued as a denial of patient’s perspective, but tion. Rehabilitation should be prescribed based on results rather a professional responsibility to ensure cost-effective of screening and diagnostic testing. A consultation with medical care . This is best managed through transpar- Neurology and Mental Health services should be contem- ent and collaborative dialogue, focused on the value of plated early, and where appropriate, an integrated team further investigations in improving the understanding of approach considered. The final care plan for these patients the evolving clinical picture and/or changing the clinical should commence with an understanding of the patient management. and family’s goals, validation of the patient’ssympto- mology and appropriate plan of care. Approach to the atypical otolaryngology-head and neck surgery patient The appropriate management of pediatric patients with Conclusion atypical symptoms can be a challenging task for Otolaryn- Delayed identification of mental illness can result in gologists. This report should stand as a resource for significant medical and psychological consequences for clinicians who encounter difficult cases in this context patients, increase the burden of care, and can impact (Table 1). Open dialogue should be maintained with the their faith in the health care system. Furthermore, it cre- patient and their family throughout the patient inter- ates a significant economic burden for the health-care action. In challenging cases, such as those presented in system as a whole. In this case series, the authors present this article, it is important for clinicians to broaden their 3 patients in whom presentation and management of differentials to include non-organic etiologies of otolaryn- otolaryngology-related concerns were confounded by gology disorders. Evaluation of these patients should be underlying conversion disorder. initiated with a thorough history and should note any While most patients referred to an Otolaryngology-Head potential risk factors for mental health and psycho- and Neck Surgeon will have organic explanations for their social factors which may impact the resiliency of the symptoms, it is important for the clinician to keep psycho- patient to cope with medical interventions, including genic causes and contributors on the differential, especially surgery. Physical examination should include a complete in patients with atypical clinical presentations. The Caulley et al. Journal of Otolaryngology - Head and Neck Surgery (2018) 47:39 Page 5 of 5 diagnostic approach to these patients requires a compre- 8. de Lange FP, Toni I, Roelofs K. Altered connectivity between prefrontal and sensorimotor cortex in conversion paralysis. Neuropsychologia. 2010;48(6): hensive assessment of the contributing factors, including 1782–8. the features of converson disorder associated with otolaryn- 9. Voon V, Brezing C, Gallea C, Ameli R, Roelofs K, LaFrance WC Jr, Hallett M. gology diseases, and impact the relationship between pa- Emotional stimuli and motor conversion disorder. Brain. 2010;133(Pt 5): 1526–36. tients and clinicians. This article is intended to stimulate 10. Rintelmann WF, Schwan SA, Blakley BW. Pseudohypacusis. Otolaryngol Clin discussion between patients and clinicians regarding safe N Am. 1991;24(2):381–90. and efficient diagnosis of challenging clinical cases. 11. Kumpf W. Auscultatory detection of respiratory responses to olfactory stimuli. Approach to feigned anosmia (author's transl). Laryngol Rhinol Otol (Stuttg). 1978;57(9):830–3. Abbreviations 12. Lacy TJ, McManis SE. Psychogenic stridor. Gen Hosp Psychiatry. 1994;16(3): CD: Conversion disorder; CT: Computed tomography; MRI: Magnetic 213–23. resonance imaging 13. Norton A, Roberton G. Functional upper airway obstruction. Anaesth Intensive Care. 1998;26(2):216–8. Availability of data and materials 14. Pula J. Functional vision loss. Curr Opin Ophthalmol. 2012;23(6):460–5. Patient records and information available on request. 15. Pracy JP, Walsh RM, Mepham GA, Bowdler DA. Childhood pseudohypacusis. Int J Pediatr Otorhinolaryngol. 1996;37(2):143–9. Authors’ contributions 16. Psarommatis I, Kontorinis G, Kontrogiannis A, Douniadakis D, Tsakanikos M. Conceptualization: MB. Methodology: LC, MB. Data curation: LC, SK, JO, HG, Pseudohypacusis: the most frequent etiology of sudden hearing loss in MB. Writing original draft preparation: LC, SK, JO, HG, MB. Writing review and children. Eur Arch Otorhinolaryngol. 2009;266(12):1857–61. editing: LC, SK, JO, HG, MB. All authors read and approved the final 17. Tomoda A, Kinoshita S, Korenaga Y, Mabe H. Pseudohypacusis in childhood manuscript No authors have any financial or non-financial competing and adolescence is associated with increased gray matter volume in the interests in regards to this manuscript. medial frontal gyrus and superior temporal gyrus. Cortex. 2012;48(4):492–503. 18. American Psychiatric Association. DSM-5 Task Force. Diagnostic and Ethics approval and consent to participate statistical manual of mental disorders : DSM-5. 5th ed. Arlington, VA: Not required. American Psychiatric Association; 2013. 19. Krasnik C, Grant C. Conversion disorder: not a malingering matter. Paediatr Consent for publication Child Health. 2012;17(5):246. Written informed consent was obtained from the patients’ legal guardians 20. Kozlowska K. Good children with conversion disorder: breaking the silence. for publication of this case series. A copy of the written consent is available Clinical Child Psychology and Psychiatry. 2003;8(1):73–90. for review by the Editor-in-Chief of this journal. 21. Grattan-Smith P, Fairley M, Procopis P. Clinical features of conversion disorder. Arch Dis Child. 1988;63(4):408–14. Competing interests 22. Leslie SA. Diagnosis and treatment of hysterical conversion reactions. Arch The authors declare that they have no competing interests. Dis Child. 1988;63(5):506–11. 23. Garralda ME. A selective review of child psychiatric syndromes with a somatic presentation. The British journal of psychiatry : the journal of Publisher’sNote mental science. 1992;161:759–73. Springer Nature remains neutral with regard to jurisdictional claims in 24. Bass C, Benjamin S. The management of chronic somatisation. The British published maps and institutional affiliations. journal of psychiatry : the journal of mental science. 1993;162:472–80. 25. Kozlowska K. Good children presenting with conversion disorder. Clinical Author details Child Psychology and Psychiatry. 2001;6:575–91. 1 2 Faculty of Medicine, University of Ottawa, Ottawa, ON, Canada. Department 26. Nicholson TR, Stone J, Kanaan RA. Conversion disorder: a problematic of Otolaryngology-Head and Neck Surgery, The Ottawa Hospital, Ottawa, ON, diagnosis. J Neurol Neurosurg Psychiatry. 2011;82(11):1267–73. Canada. Division of Otolaryngology – Head and Neck Surgery, Children’s 27. Carlson ML, Archibald DJ, Gifford RH, Driscoll CL. Conversion disorder: a Hospital of Eastern Ontario, 400 Smyth Road, Ottawa, ON K1H 8L1, Canada. missed diagnosis leading to cochlear reimplantation. Otol Neurotol. 2011; Department of Psychiatry, Children’s Hospital of Eastern Ontario, University 32(1):36–8. of Ottawa, Ottawa, ON, Canada. Department of Psychology, Children’s 28. Brett AS, McCullough LB. Addressing requests by patients for nonbeneficial Hospital of Eastern Ontario, University of Ottawa, Ottawa, ON, Canada. interventions. Jama. 2012;307(2):149–50. 29. Miller E. Defining hysterical symptoms. Psychol Med. 1988;18(2):275–7. Received: 30 October 2017 Accepted: 21 May 2018 30. Crosskerry P, Nimmo GR. Better clinical decision making and reducing diagnostic error. Journal of the Royal College of Physicians and Edinburgh. 2011;41(2):155–62. References 31. Brett A, McCullough LB. When patients request specific interventions: 1. Organization WH: Child and adolescent mental health.. 2015. defining the limits of the physician's obligations. N Engl J Med. 1986; 2. Olfson M, Druss BG, Marcus SC. Trends in mental health care among 315(21):1347–51. children and adolescents. N Engl J Med. 2015;372(21):2029–38. 32. American College of Physicians. How Can Our Nation Conserve and Distribute 3. Ani C, Reading R, Lynn R, Forlee S, Garralda E. Incidence and 12-month Health Care Resources Effectively and Efficiently? Philadelphia: American outcome of non-transient childhood conversion disorder in the U.K. and College of Physicians; 2011: Policy Paper. (Available from American College of Ireland. Br J Psychiatry. 2013;202:413–8. Physicians, 190 N. Independence Mall West, Philadelphia, PA 19106.). https:// 4. Kozlowska K, Nunn KP, Rose D, Morris A, Ouvrier RA, Varghese J. Conversion www.acponline.org/system/files/documents/advocacy/current_policy_papers/ disorder in Australian pediatric practice. J Am Acad Child Adolesc assets/health_care_resources.pdf. Psychiatry. 2007;46(1):68–75. 33. Reuben DB, Cassel CK. Physician stewardship of health care in an era of 5. Kozlowska K, Palmer DM, Brown KJ, Scher S, Chudleigh C, Davies F, Williams finite resources. Jama. 2011;306(4):430–1. LM. Conversion disorder in children and adolescents: a disorder of cognitive 34. Brody H. Medicine's ethical responsibility for health care reform–the top five control. J Neuropsychol. 2014;9:87–108. https://doi.org/10.1111/jnp.12037. list. N Engl J Med. 2010;362(4):283–5. 6. van der Kruijs SJ, Bodde NM, Vaessen MJ, Lazeron RH, Vonck K, Boon P, 35. Newman-Toker DE, McDonald KM, Meltzer DO. How much diagnostic safety Hofman PA, Backes WH, Aldenkamp AP, Jansen JF. Functional connectivity can we afford, and how should we decide? A health economics of dissociation in patients with psychogenic non-epileptic seizures. J Neurol perspective. BMJ Qual Saf. 2013;22:ii11–20. Neurosurg Psychiatry. 2012;83(3):239–47. 7. Cojan Y, Waber L, Carruzzo A, Vuilleumier P. Motor inhibition in hysterical conversion paralysis. Neuroimage. 2009;47(3):1026–37.
Journal of Otolaryngology -Head & Neck Surgery
– Springer Journals
Published: May 29, 2018