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Burning mouth syndrome to oral cenesthopathy: A spectrum of neuropsychiatric and sensory complications in neurodegenerative parkinsonism?

Published:September 23, 2022DOI:https://doi.org/10.1016/j.parkreldis.2022.09.010
      Sensory disturbances are well known nonmotor manifestations in Parkinson disease (PD), with growing data on orofacial pain. Oral pain may include burning mouth syndrome (BMS), likely an under-reported phenomenon [
      • Yu X.X.
      • Hogue O.
      • Rasameesoraj T.
      • Spilsbury J.
      • Fernandez H.
      Burning mouth syndrome –a potentially under-recognized nonmotor symptom of Parkinson disease?.
      ], described as a burning sensation of the oral cavity without any detectable clinical changes in the mucosa or organic cause [
      • Bonenfant D.
      • Rompré P.
      • Rei N.
      • Jodoin N.
      • Soland V.
      • Rey V.
      • Brefel-Courbon C.
      • Ory-Magne F.
      • Rascol O.
      • Blanchet P.
      Characterization of burning mouth syndrome in patients with Parkinson's disease.
      ]. Cenesthetic hallucinations, on the other hand, are strange sensations characterized by distortions of body awareness such as abdominal organs being extruded from a patient's limbs reported in a patient with PD [
      • Jimenez-Jimenez F.J.
      • Orti-Pareja M.
      • Gasalla T.
      • Tallon-Barranco A.
      • Cabrera-Valdivia F.
      • Fernandez-Lliria A.
      Cenesthetic hallucinations in a patient with Parkinson's disease.
      ]. These may occur in any part of the body. When localized in the oral cavity, it is termed oral cenesthopathy [
      • Umezaki Y.
      • Miura A.
      • Watanabe M.
      • Takenoshita M.
      • Uezato A.
      • Toriihara A.
      • Nishikawa T.
      • Toyofuku A.
      Oral cenesthopathy.
      ] and may include an additional feature of sensation of distortions such as a squeezing-pulling sensation in the mouth, a slimy sensation, or a feeling of coils or wires within the oral region [
      • Umezaki Y.
      • Miura A.
      • Watanabe M.
      • Takenoshita M.
      • Uezato A.
      • Toriihara A.
      • Nishikawa T.
      • Toyofuku A.
      Oral cenesthopathy.
      ]. Whether or not this is a subtype of a tactile hallucination that is confined to the oral cavity remains to be elucidated. Despite absence of mucosal abnormalities, patients report extreme discomfort, prompting consults with dentists and otolaryngologists but less often to neurologists or psychiatrists. Determining the exact etiology, however, may present a challenge when the patient has both parkinsonism and comorbid psychiatric conditions, especially when oral cenesthopathy is present without diagnosed schizophrenia. We present this case as an example.
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