Objective: To report a PD patient with severe BMS refractory to different medications with significant improvement from LCIG infusion.

Background: BMS is an often distressing but uncommonly described non-motor condition associated with PD characterized by painful burning sensation in the mouth. Dysregulation in the nigrostriatal dopaminergic system has been implicated in its pathophysiology. Treatment of BMS is often unsatisfactory.

Methods: Case report.

Results: A 73 year-old woman was diagnosed with PD at age 68. She was initially treated with oral carbidopa-levodopa with good response. Over time, she developed motor fluctuations and other non-motor symptoms including anxiety and back pain. On her first visit with us, she was on carbidopa-levodopa 25/100 1.5 tablets 6 times daily. She reported severe burning sensation in her tongue, cheeks and lips in the past year. She had extensive dental and ENT workup including lip biopsy and autoimmune panel which were all unremarkable. Because the onset of her BMS coincided with an increase in her daily levodopa dose from 600 to 900mg, levodopa was reduced without improvement in BMS. As she was experiencing anxiety, we tried her on duloxetine and clonazepam which are typically used for BMS, without significant relief. We also tried other neuropathic pain medications such as gabapentin and pregabalin without benefit. Because she was experiencing motor fluctuations, she was tried on Rytary with some improvement noted in motor symptoms, but her BMS persisted. She continued to have significant motor and non-motor fluctuations prompting evaluation for deep brain stimulation (DBS). Due to cognitive impairment, DBS was not recommended. She was then started on LCIG treatment. After 1 week of careful dose titration, on 13.9 ml morning bolus and 2.8 ml/hr continuous dose, patient reported complete resolution of BMS for 5 weeks along with significant improvement in back pain, anxiety and motor fluctuations. At the latest follow up, she had occasional recurrence but much milder and more manageable symptoms of BMS.

Conclusions: This is the first documented case of BMS in a PD patient responsive to LCIG treatment. Our case report reinforces the occurrence of significant non-motor symptoms associated with motor fluctuations. While non-motor symptoms such as BMS can be refractory to oral therapies, LCIG may be considered as a treatment option.

« Back to 2016 International Congress

MDS Abstracts - https://www.mdsabstracts.org/abstract/first-documented-case-of-burning-mouth-syndrome-bms-responsive-to-levodopa-carbidopa-intestinal-gel-lcig-infusion-in-parkinsons-disease-pd/