Introduction
Miller Fisher syndrome (MFS) is a type of acute inflammatory polyneuropathy, a disease characterized by abnormal muscle coordination, absence of tendon reflexes, and paralysis of the eye muscle. Collier first described it in 1932 as a variant of Guillain-Barre syndrome, and in 1956, Miller Fischer described it as an independent disease with 3 main symptoms [1]. It is reported that in two thirds of MFS patients, respiratory or gastro-intestinal infection precedes, and in an average of 10 days, nervous symptoms start to appear [2]. Diagnosis of MFS is based on 3 characteristic clinical symptoms with examination of serum anti-GQ1b IgG antibody [3]. This study reports on one MFS patient who did not show improvement in eye movement disorder, diplopia, and dizziness upon 5-days of high-dosage steroids, however symptom improvement was observed after CKMT. This study was exempt from IRB deliberation (IRB No.: DJDSKH-18-E-03-1).
Case Report
A 24-year-old male suffered from enteritis from September, 9, 2017, followed after 5 days by diplopia, eye movement disorder, and dizziness. He showed gait ataxia, deep tendon reflex (DTR) hypoactive, and limited eye movement in all directions. Brain magnetic resonance imaging and cerebrospinal fluid examination did not show any abnormalities, and the blood test showed Anti-GQ1b Ab of 1:1,600, diagnosing him as having MFS. During his admission period in OO hospital, high-dosage steroid administration for 5 days did not improve his symptoms. He then visited OO Korean medicine hospital and received combined Korean medicine treatment (CKMT) as an inpatient for 40 days.
Treatment methods
For 6 weeks, the patient received acupuncture, electroacupuncture, pharmacopuncture, herbal medicine, and physical therapy treatment. Acupuncture needles were inserted at 12 acupoints for 15 minutes: Bilateral BL2, Ex-HN4, TE23, Ex-HN5, TE17, and ST2. Details of the acupuncture treatments are provided in Table 1 (Table 1). Electroacupuncture treatment was administered 3 times a week, taking turns with pharmacopuncture. Selected electroacupuncture points were bilateral BL2- TE23, Ex-HN5-ST2. The machine for electroacupuncture was STN-110 set with stimulation of 3Hz, continuous for 20 minutes. Pharmacopuncture used was SBV10 (extracted and refined from bee toxin and mixed to 99.9% pure melittin 0.1mg/mL) manufactured in extramural herbal dispensaries. 1mL/cc syringe was used for 1mL administration of single sessions, and each dose administered 0.1mL intramuscularly. For physical therapy, ICT (interferential current therapy) was administered on the neck, and SSP-C (silver spike point) and manual therapy was administered on the facial region. Herbal medicine was given 3 times a day. Two kinds of herbal medicine were given (Table 2).
Evaluation
To measure the range of movement of adduction, abduction, up and down movement of both eyes, a ruler was used to measure in unit of mm with the patient’s pupil set as the center. Also, eye movements in 9 cardinal positions were recorded in photographs 2 times a week. For evaluation of diplopia, a straight distance from the patient’s glabella to where diplopia started to occur to where diplopia disappeared was measured. Also, this case report used the diplopia questionnaire that Jonathan et al [4, 5] had developed replacing the Goldmann test to evaluate the severity of diplopia (Appendix 1). For evaluation of the patient’s subjective feeling of dizziness and diplopia, VAS scale was utilized. Evaluation was done by a resident trainee in acupuncture and moxibustion medicine.
Evaluation of range of eye movement
Primary eye measurements were taken on October 16, 2017. The right eye showed horizontal movement of 9 mm and vertical movement of 7 mm, while the left eye showed horizontal movement of 8 mm and vertical movement of 6 mm. Final measurements were obtained on November 23, 2017, which showed the right eye with horizontal movement of 17 mm and vertical movement of 11 mm while the left eye showed horizontal movement of 17 mm and vertical movement of 11 mm (Fig. 1).
Measurement of distance of diplopia
The primary measurement of the straight distance from the patient’s glabella to where diplopia started to occur, and to where diplopia disappeared was recored in all distance, while measurement on November, 23, 2017 was recorded in distance ranging from 59 cm to 67 cm (Table 3).
Evaluation of diplopia questionnaire
Primary score on the questionnaire performed on October, 16, 2017 was 25 where diplopia occurred in “always” in the 7 gaze positions. In the questionnaire performed finally on November, 23, 2017 and during admission, a score of 25 was recorded with frequency of diplopia recorded as “always”, showing no difference to the primary score.
VAS evaluation for diplopia and dizziness
Primary VAS scores evaluated on October, 16, 2017 for diplopia and dizziness were 9.2 and 1.8 respectively. Final scores on the 6 week of admission were 4.3 and 0.2 respectively, showing a reduction in VAS scores (Fig. 5).
Discussion
MFS has a male to female occurrence in a 2:1 ratio, with an average age of about 43 years old, and with pediatric patients consisting of 14% of the total patients [2]. Anti-GQ1b antibody induces a secretory disorder of acetylcholine on the end of motor nerves, which is thought to be the etiological cause of eye movement disorder from muscle weakness in MFS [6]. The most common treatment uses anti-GQ1b IgG antibody that helps inhibit this auto-immune reaction [7].
Of the three main symptoms of MFS, ophthalmoplegia shows the slowest recovery with the recovery period being from 1 week to 20 weeks [8]. In this case report, the up and down eye movement started to show rapid recovery in the 2nd week of admission, reaching near complete recovery by the 4th week. The abduction and adduction also showed rapid recovery from the 1st week of admission reaching near complete recovery on the 4th week. There were also notable reductions in the distance that diplopia appears and VAS evaluation scores for diplopia and dizziness. In this study, the recovery period was shortened compared to recovery periods of MFS in previous studies. However, the patient showed diplopia in all gaze positions in equivalent frequency according to the questionnaire which suggests no improvement on diplopia.
This case report suggests that CKMT may have a role in treating eye movement disorders of MFS patients. Although the mechanism of CKMT remains unclear, one of the possible therapeutic mechanisms in this study is stimulation of acupuncture points near to the orbital wall. Acupuncture generates microinjury which increases local blood flow and facilitates healing [9]. Acupuncture points were adopted from previous studies. You et al [10] treated diplopia on acupuncture points like GV20, BL2, Ex-HN4, TE23, ST2, TE17, LI04, LR03, and ST36. Electroacupuncture promotes regeneration of nerve fibers and motor function recovery by decreasing the production of free radicals and regulating neuropeptide secretion [11]. And it is reported that bee venom has anti-inflammatory properties for disorders including atherosclerosis, diabetes-related endothelial damage, and autoimmune diseases [12,13,14]. It might have a similar effect on this disease. Lastly, Lycium-Chrysanthemum blood-nourishing decoction has been reported to have an effect on neuromyelitis optica and vision disorder [15]. According to our clinical judgement, the function of the eye may be improved by increasing the nourishing blood supply.
The limit of this study may be the uncertainty in determining which treatment had the main effect in this study because the patient received various CKMT therapies. Also, it is difficult to estimate the exact therapeutic effect of CKMT, because the duration of treatment in this case is within the period of MFS’s natural recovery. Most symptoms of MFS start to recover within 3 weeks of onset and reach complete recovery within six months [8]. There are few studies comparing natural recovery with CKMT for treating MFS. This study is also limited as it is a retrospective studies. Prospective studies are required to examine the beneficial effect of CKMT for MFS in larger number of cases.
This case report suggests that CKMT may have a role in treating eye movement disorders and dizziness of MFS patients.
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