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).
Photographs of eye movements in 9 cardinal positions
Photographs of eye movements taken on November 23, 2017 on 9 cardinal positions showed difference compared to photographs taken on October 16, 2017 (
Figs 2-
4).
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 2
nd week of admission, reaching near complete recovery by the 4
th week. The abduction and adduction also showed rapid recovery from the 1st week of admission reaching near complete recovery on the 4
th 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.
Conflict of Interest
The authors have no conflicts of interest to declare.
Fig. 1.
Fig. 2.
Photographs of eye movement on 9 cardinal positions taken on October, 16, 2017.
Fig. 3.
Photographs of eye movement on 9 cardinal positions taken on October, 30, 2017.
Fig. 4.
Photographs of eye movement on 9 cardinal positions taken on November, 23, 2017.
Fig. 5.
VAS for diplopia and dizziness.
Table 1.
Standards for Reporting Intervention in Clinical Trials of Acupuncture.
Item |
Detail |
Details |
1. Acupuncture rationale |
1a) Style of acupuncture |
Acupuncture treatments were based on Korean medicine theory. |
|
1b) Reasoning for treatment provided |
Textbook of Acupuncture and Moxibustion Medicine |
|
1c) Extent to which treatment was varied |
Treatment was almost the same. |
|
2. Details of needling |
2a) Number of needles inserted |
12 needles per session |
|
2b) Names of points used |
Bilateral BL2, Ex-HN4, TE23, Ex-HN5, TE17, and ST2 |
|
2c) Depth of insertion |
0.5 to 1.0 cm on BL2, Ex-HN4, TE23, Ex-HN5, TE17, and ST2 points |
|
2d) Response sought |
Subjective De-qi sensation |
|
2e) Needle stimulation |
Manual stimulation of rotation, lifting and thrusting |
|
2f) Needle retention time |
0 ~ 15minutes |
|
2g) Needle type |
Dong-bang stainless steel disposable acupuncture needle 0.20 × 30 mm |
|
3. Treatment regimen |
3a) Number of sessions |
80 sessions |
|
3b) Frequency/ duration |
Twice a day for 40 days |
|
4. Other components of treatment |
4a) Details of other interventions |
electro-acupuncture, pharmacopuncture, herbal medicine, and physical therapy treatment |
|
5. Practitioner background |
5) Description of participating acupuncturists |
2 Korean medicine doctors. 1) Resident trainee in acupuncture and moxibustion medicine with 2 years of experience |
2) Specialist of a acupuncture and moxibusion medicine with more than 20 years of experience |
Table 2.
Herbal Compositions of 2 Herbal Medicines for Daily Dosage.
Pinellia, Atractylodis Mcrocephalae, and Gastrodia Decoction (2017.10.16-2017.11.06) |
|
Lycium-Chrysanthemum blood-nourishing decoction (2017.11.07-2017.11.23) |
|
Pinellia ternate |
6g |
Rehmanniae Radix Preparata |
12g |
Citrus tachibana |
6g |
Angelica gigas Nakai |
6g |
Hordeum vulgare Linné |
6g |
Paeoniae Radix Alba |
6g |
Atractylodes macrocephala Koidzumi |
4g |
Lycium chinense Miller |
6g |
Massa Medicata Fermentata |
4g |
Cnidium officinale Makino |
4g |
Atractylodes japonica KOIDZ |
2g |
Chrysanthemum indicum |
4g |
Panax ginseng C. A. Meyer |
2g |
Liriope platyphylla F.T.Wang & T.Tang |
4g |
Astragalus membranaceus |
2g |
Tribulus terrestris L |
4g |
Gastrodia elata BLUME |
2g |
Ledebouriella seseloides WOLFF |
4g |
Poria cocas Wolf. |
2g |
Schizonepeta tenuifolia var. japonica |
2g |
Alisma canaliculatum A.Br. & Bouche |
2g |
Mentha arvensis var. piperascens Malinv. ex Holmes |
2g |
Zingiberis Rhizoma |
1g |
Glycyrrhiza uralensis |
2g |
Phellodendri Cortex |
1g |
|
|
Table 3.
Date |
Distance (cm) |
October, 16, 2017 |
All distance |
October, 19, 2017 |
All distance |
October, 23, 2017 |
All distance |
October, 26, 2017 |
All distance |
October, 30, 2017 |
All distance |
November, 2, 2017 |
18-112 |
November, 6, 2017 |
21-81 |
November, 9, 2017 |
30-78 |
November, 13, 2017 |
44-75 |
November, 16, 2017 |
60-78 |
November, 20, 2017 |
65-75 |
November, 23, 2017 |
59-67 |
References
1. Fischer M. An unusual variant of acute idiopathic polyneuritis (syndrome of ophthalmoplegia, ataxia and areflexia). N Engl J Med. 1956;255:57–65.
2. Berlit P, Rakicky J. Miller Fischer syndrome. Review of the literature. J Clin Neurophthalmol. 1992;12:57–63.
3. Lo YL. Clinical and immunological spectrum of the Miller Fisher syndrome. Muscle Nerve. 2007;36:615–627.
4. Jonathan MH, Leske DA, Kupersmith MJ. New Methods for Quantifying Diplopia. Ophthalmology. 2005;112:2035–2039.
6. Buchwald B, Weisjaupt A, Toyka KV, Dudel J. Pre-and postsynaptic blockade of neuromuscular transmission by Miller Fisher syndrome IgG at mouse motor nerve terminals. Eur J Neurosci. 1998;10:281–290.
8. Son MG, Ahn HS. Miller Fisher syndrome. J Korean Ophthalmol Soc. 1997;38:1470–1479. [in Korean].
9. Cheng KJ. Neurobiological Mechanisms of Acupuncture for Some Common Illnesses: A Clinician’s Perspective. J Acupunct Meridian Stud. 2014;7:105–114.
10. Ryu JY, Lee KW, Cho MK, Cho HK, Yoo HR, Seol IC, et al. Case report: Miller Fisher Syndrome. J Int Korean Med. 2016;37:661–668. [in Korean].
11. Li YF, Li T, Zhang DW, Xue H, Chen D, Li C, et al. The comprehensive Therapy of Electroacupunture Promotes Regeneration of Nerve Fibers and Motor Function Recovery in Rats after Spinal Cord Injury. Evid Based Complement Alternat Med. 2018;2018:7568697.
12. Lee WR, Kim SJ, Park JH. Bee venom reduces atherosclerotic lesion formation via anti-inflammatory mechanism. Am J Chin Med. 2010;38:1077–1092.
13. Behroozi J, Divsalar A, Saboury AA. Honey bee venom decreases the complications of diabetes by preventing hemoglobin glycation. J Mol Liq. 2014;199:371–375.
14. Wesselius T, Heersema DJ, Mostert JP. A randomized crossover study to bee sting therapy for multiple sclerosis. Neurology. 2005;65:1764–1768.
15. Woo SJ, Shin JW, Jang WS, Baek KM. A Case Report of Treatment of a patient with Neuromyelitis Optica and Suffering from vision Disorder and Quadriplegia with Korean Traditional Medicine. J Intern Korean Med. 2017;38:658–667.
Appendices
Appendix 1.
Questionnaire for Evaluation of Diplopia.
Gaze position |
Always |
Sometimes |
Never |
Score |
Straight ahead in distance |
6 |
3 |
0 |
|
Up |
2 |
1 |
0 |
|
Down |
4 |
2 |
0 |
|
Right |
4 |
2 |
0 |
|
Left |
4 |
2 |
0 |
|
Reading |
4 |
2 |
0 |
|
Any position |
1 |
1 |
0 |
|