Oral cenesthopathy superimposed on burning mouth syndrome treated with aripiprazole: A case report with a phenomenological overview

Abstract

Background Burning mouth syndrome (BMS) is an idiopathic condition that presents with chronic pain and/or burning sensations in the oral structures. The syndrome mostly affects elderly women with hormonal changes and/or with a diagnosis of comorbid psychiatric disorder. In some rare conditions, the clinical appearance of BMS may also overlap with oral cenestopathy (OC), which is defined in the literature as a special form of delusional disorder of somatic type. Patients with OC may complain about abnormal experiences such as melting, feeling of stickiness, as well as extremely strange feelings of wires, metal coils, etc being present in their mouths. Case Presentation We present an elderly woman whose ongoing symptoms of BMS (burning in the mouth and tongue, taste alterations etc) were, over time, superimposed by cenesthopatic delusions that her gums had melted, and her palate had totally dissolved. We believe that the case is clinically striking and demonstrative for the understanding of complex nosology of BMS and OC, given that (a) the patient exhibited a relatively rare example of overlapping BMS and OS symptoms, which both are not sufficiently recognised by clinicians, (b) OC symptoms have disappeared with low‐dose aripiprazole and psychoeducation‐based cognitive therapy, which resulted in significant improvement in the patient's quality of life. Conclusion Clinicians are required to be aware of BMS and OC, two syndromes with multifactorial aetiology and highly heterogeneous presentation, in order to determine the most appropriate treatment options from a multidisciplinary perspective, as well as to avoid unnecessary medical interventions.

Case report: Remission of chronic low back pain and oral dysesthesia comorbid with attention deficit/hyperactivity disorder by treatment with atomoxetine and pramipexole

“…The results of this case and a previous report that used aripiprazole (a dopamine partial agonist) showed improved OD (6) and suggest that dopamine agonists may be effective for OD. Furthermore, as the combination of ATX and PPX effectively treated the coexistence of low back pain and OD in the case presented herein, the improvement of both prefrontal and limbic neurotransmission may be necessary in the treatment of patients with overlapping idiopathic pain conditions, such as COPCs.…”

“…Furthermore, COPCs, such as OD, BMS, and chronic low back pain, have a similar tendency of coexisting with psychiatric disorders such as anxiety and depression, suggesting that these COPCs may have a central nervous system (CNS) dysfunction that makes the corresponding patients vulnerable to both pain and psychiatric disorders (1)(2)(3)(4)(5). CNS dysfunction is thought to be attributable to dysfunction of the dopaminergic nervous system (2,4,6). However, COPCs, such as OD, BMS, and chronic low back pain, are often refractory to treatment, and the development of novel treatment modalities tailored to their pathophysiology is expected (2,5,7,8).…”

Collaboration between Psychiatrists and Other Allied Medical Specialists for the Treatment of Delusional Disorders

“…Chronic pain (rheumatic pains, referred pain, fibromyalgia, neuropathies) induces the development of delusional explanations for the pain, but pain can also result from primary delusions [ 62 ]. Burning Mouth Syndrome and oral cenesthopathy are specific examples of clinical pain conditions that require collaboration between psychiatrists and pain specialists [ 63 ].…”

“…Karakus and Bulut [ 63 ] reported a case of overlapping OC delusions and burning mouth syndrome in an elderly woman. OC symptoms responded to a combination of low-dose aripiprazole and psychoeducation-based cognitive therapy.…”

The similarities and differences between oral cenesthopathy and burning mouth syndrome in the elderly

“…The similarities and differences between oral cenesthopathy and burning mouth syndrome in the elderly We read Dr Ibrahim Hakki Karakus and Dr Necati Serkut Bulut's article entitled "Oral cenesthopathy superimposed on burning mouth syndrome treated with Aripiprazole: A case report with a phenomenological overview 1 " with deep empathy.…”