Tongue Biting Event in Patients with Sleep-Related Facial Mandibular Myoclonus: A Case Series Study

Figures & data

Figure 1 SRFMM tongue ulcer in patient 2. (A) shows a small ulcer at 8 months of age. (B) shows multiple ulcers at 1.2 year of age.

Table 1 Clinical and Electrophysiological Characteristics of Patients with SRFMM

#	Age	Sex	Primary Complaint	Age of Disease Onset	Brain MRI	Type	Sleep Stage	Frequency	Comorbidities	Medication/Prognosis
1	8 months	M	Tongue biting in sleep for 1 month	6 months	Normal	Single	NREM 2/3/early drowsiness	2–3 times/night	–	-/remission for 1 year
2	10 months	M	Tongue biting in sleep for 4 months	6 months	Normal	Single	NREM 1/2	5–20 times/night	Head and facial trauma (1 week before symptom onset)	CLO for 1 month/remission for 6 years
3	1.2 y	M	Tongue biting in sleep for 6 months	6 months	Normal	Single	NREM 1/2	1–2 times/night	-	CLO for 2 months /remission for 0.5 year
4	2 y	M	Tongue biting in sleep for 2 months	2 y	Normal	Single	Sleep	4–5 times/night	-	-/remission for 0.5 year
5	20y	M	Poor sleep	20y	Normal	Single	Sleep	1 time/week	Insomnia	-/sporadic
6	38y	F	Difficulty in falling asleep	36y	Normal	Single	Sleep	1–2 times/month	-	-/sporadic
7	42y	M	Tongue biting in sleep after aripiprazole	42y	Normal	Single	NREM 1/2	1–5 times/night	Bipolar disorder, OSA	Withdrawal of aripiprazole/remission for 0.5 year
8	45y	M	Tongue biting in sleep	45y	Normal	Single/clustered	NREM 2	4 times in total	-	-/remission for 5 years
9	47y	M	Tongue biting in sleep	45y	Normal	Single/clustered	Sleep	1–2 times/night	OSA, PLMS	-/remission for 1 year (ventilation treatment)
10	47y	M	Tongue biting in sleep	46y	Normal	Single	Sleep	3–4 times/year	-	-/remission for 3 years
11	50y	F	Tongue biting in sleep	50y	Normal	Single	Sleep	3–4 times/month	-	-/remission for 4 years
12	50y	M	Tongue biting in sleep	50y	Normal	Single	Sleep	1–2 times/night	OSA, PLMS	-/sporadic
13	53y	M	Tongue biting in sleep	52y	Normal	Single	Sleep	4–5 times/year	-	-/sporadic
14	56y	F	Tongue biting in sleep	56y	Lacunar infarction	Single	NREM 2/REM	2 times/night-1 time/week	OSA, Hypertension, history of cerebral infarction	Intermittent CLO/sporadic
15	57y	M	Tongue biting in sleep	57y	Midbrain abnormal signals	Single	Sleep	5 times in February	OSA, acute brainstem infarction	-/remission
16	57y	F	Tongue biting in sleep	56y	Normal	Single	NREM 2	1–2 times/week	Anxiety	-/remission for 2 years
17	58y	M	Tongue biting in sleep and wakefulness	57y	Normal	Single	NREM	2–3 times/day	PLMS	Intermittent CLO/sporadic
18	58y	M	Lips and Tongue biting in sleep	54y	Lacunar infarction	Single/clustered	NREM 2	1–4 times/night	Hypertension, OSA	-/1–4 times/night
19	58y	M	Lips and Tongue biting in sleep	58y	Normal	Single	NREM 1/2	1–3 times/night	OSA, PLMS	-/remission for 0.5 year
20	63y	M	Tongue biting in sleep	63y	Normal	Single	REM/NREM 1/2	4–5 times/night	Head trauma (20 days before symptom onset), OSA	CLO for 2 months/sporadic
21	65y	M	Tongue biting in sleep	59y	Normal	Single	NREM 1	3–5 times/month	OSA, PLMS, hypertension, CHD, colon tumor	-/sporadic
22	68y	M	Tongue biting in sleep	67y	Unknown	Single	NREM 1/2	1–2 times/night	OSA	CLO for 2 months/remission for 0.5 year
23	89y	M	Tongue biting in sleep	84y	Brain atrophy	Single	Sleep	Once/several months to twice/night	-	-/sporadic

Notes: “-” means none; “#” means number.

Abbreviations: CHD, coronary heart disease; CLO, clonazepam; NREM, non-rapid eye movement; OSA, obstructive sleep apnea; PLMS, periodic limb movement syndrome; REM, rapid eye movement.

Figure 2 Electroclinical features of SRFMM in patient 1. (A) showed a sudden EMG burst of bilateral masticatory muscle followed by arousals at NREM stage 3 period during video-EEG monitoring. Red arrow indicates EMG bursts over bilateral masseter. (B-D) showed clinical manisfestations of crying and limbs lifting during SRFMM episode.

Table 2 Comparisons of Electrophysiological Features of the Patient 20 Before and After Clonazepam Treatment

	Total Sleep Time (min)	Sleep Efficiency (%)	No of SRFMM	No of Awakenings	WASO (Min)
Before	349	75.6%	23	35	112.5
After	405	81.5%	0	21	76.8

Abbreviations: SRFMM, sleep-related facial mandibular myoclonus; WASO, wakefulness after sleep onset; No, number.