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Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (2017) xxx–xxx

Contents lists available at ScienceDirect

Journal of Oral and Maxillofacial Surgery,


Medicine, and Pathology
journal homepage: www.elsevier.com/locate/jomsmp

Case report

Peripheral facial palsy after set-forward orthognathic surgery: A case


report and review of literature
Eiji Mitate a,b,∗ , Jun-Nosuke Hayashida b,e , Takeshi Toyoshima b,c,e , Takeshi Noguchi d,e ,
Toru Kitahara d,e , Ichiro Takahashi d,e , Tetsuro Ikebe a , Seiji Nakamura b
a
Department of Oral and Maxillofacial Surgery, Fukuoka Dental College, 2-15-1, Tamura, Sawara-ku, Fukuoka 814-0193, Japan
b
Section of Oral and Maxillofacial Oncology, Division of Maxillofacial Diagnostic and Surgical Sciences, Faculty of Dental Science, Kyushu University, 3-1-1,
Maidashi, Higashi-ku, Fukuoka 812-8582, Japan
c
Medical corporation Toyoshima dental clinic, 5-1, Furujinmachi, Takamatsu, Kagawa 760-0025, Japan
d
Section of Orthodontics and Dentofacial Orthopedics, Graduate School of Dental Sciences, Kyushu University, 3-1-1, Maidashi, Higashi-ku, Fukuoka
812-8582, Japan
e
Dental and Maxillofacial Center, Kyushu University Hospital, 3-1-1, Maidashi, Higashi-ku, Fukuoka 812-8582, Japan

a r t i c l e i n f o a b s t r a c t

Article history: The complications of orthognathic surgery may include but are not limited to postoperative unexpected
Received 11 May 2017 bleeding, abnormal fractures, infections, and nerve damages. Peripheral facial palsy is rare. We report
Received in revised form 23 October 2017 a case of 23-year-old woman with signs of malocclusion, including pectus excavatum, wrist sign, and
Accepted 8 November 2017
thumb sign for Marfan syndrome (MFS) underwent peripheral facial palsy after orthognathic surgery
Available online xxx
with mandibular advancement. Because cardiovascular, thoracic, and ocular abnormalities were absent,
she was suspected of having MFS but not definitively diagnosed. On the other hand, she was diagnosed
Keywords:
with a maxillary protrusion with mandibular retrognathism. After presurgical orthodontic treatment,
Facial nerve palsy
Orthognathic surgery
we performed Le Fort I osteotomy and 8 mm advancement by bilateral sagittal split ramus osteotomy.
Complication Because the periosteum is vulnerable, we checked the bleeding more are fully and frequently during the
Set-forward operation than usual. The day after the operation, peripheral facial palsy with inability to wrinkle the
Tissue vulnerability forehead, blink, and grimace were found. The patient was administered mecobalamin, linear polarized
infrared irradiation, and stellate ganglion blocks. The patient was also administered valacyclovir for 6 days
and steroid pulse therapy for 7 days. Six months after the operation, the facial palsy had disappeared
completely.
In 58 previous reports, the facial palsy was induced by the reasons, including the operative proce-
dure, postoperative hematoma, edema, perioperative stress, and tissue vulnerability. In the present case,
because of the suspected MFS, tissue vulnerability was considered to be one of the major cause of facial
palsy, which should be paid attention more carefully in addition to general causes.
© 2017 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.夽

1. Introduction 2. Case report

The common complications of orthognathic surgery are unex- In February 2012, a 23-year-old woman was introduced to
pected bleeding, abnormal fractures, and nerve injuries. In Kyushu University Hospital from a private dental clinic with a chief
particular, maxillary and mandibular trigeminal nerve injuries are complaint of malocclusion. (Fig. 1A, B).
often seen. Few reports of facial nerve injury can be found, and most She was 167 cm in height and 42 kg in weight. The thumb sign,
of them are setback cases of bilateral sagittal split ramus osteotomy the wrist sign, and pectus excavatum were evident. (Fig. 2A, B).
(SSRO). Here, we report an extremely rare case of facial palsy after However, there were no past, social, and familial histories in par-
mandibular advancement with SSRO. ticular. The cardiovascular surgeon and the ophthalmologist found
no cardiovascular disease and no ectopia lentis. Finally, she was
diagnosed on suspicion of having Marfan Syndrome (MFS).
夽 The gist of this paper was presented at the 26th congress of the Japanese Society Angle Class II, division 2 malocclusion was observed as follows:
for Jaw Deformities (24–25, June 2016, Tokyo). +1 mm of overjet and +6 mm of overbite, and +1 mm deflection of
∗ Corresponding author at: Department of Oral and Maxillofacial Surgery, the mandible to the left side (Fig. 3A–G). The result of cepharometric
Fukuoka, Dental College, 2-15-1, Tamura, Sawara-ku, Fukuoka 814-0193, Japan. analysis is indicated as follows; convexity: +3 SD, SNB: −3 SD, ANB:
E-mail address: mitate@college.fdcnet.ac.jp (E. Mitate).

https://doi.org/10.1016/j.ajoms.2017.11.002
2212-5558/© 2017 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.夽

Please cite this article in press as: Mitate E, et al. Peripheral facial palsy after set-forward orthognathic surgery: A case report and review
of literature. J Oral Maxillofac Surg Med Pathol (2017), https://doi.org/10.1016/j.ajoms.2017.11.002
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Fig. 1. Facial photographs at pretreatment (A and B).

+3 SD, ramus plane to SN: +3 SD, ramus plane to Frankfurt plane: after the operation, the enzyme immunoassay (EIA) level of herpes
+2 SD, intergonial angle: −2 SD, occlusal plane to SN: +2 SD. (Figs. simplex virus (HSV) in serum IgG was 49, and that of varicella zoster
4A, B, 5, and 6 ). The orthognathic operation with 3 mm upward of virus (VZV) was 17. The EIA levels of both serum IgMs were within
the ANS with Le Fort I osteotomy and 8 mm advancement of the normal limits. There seemed to be a possibility of herpes zoster
mandible with SSRO was planned. from perioperative stress, we consulted to otorhinolaryngologist,
After preoperative orthodontic treatment flaring upper incisors 3000 mg/day of valacyclovir for 6 days were administered. From
to the normal inclination without extraction, Le Fort I osteotomy the 6th to 34th days, total 14 times of linear polarized infrared irra-
and SSRO were carried out in November 2014 at age 25. Since the diation (Super Lizer; Tokyo Iken Co., Ltd, Tokyo) were administered
periosteum was so vulnerable in both maxilla and mandible, there to the stellate ganglion. As the recovery was so slow, additionally
were some difficulties to operate the periosteum and stop bleed- 13 times stellate ganglion block (SGB) were administered from the
ing during the operation. The length of general anesthesia was 6 h 11th day to the 75th day after operation. The score on the Yanag-
57 min. The length of operation was 4 h 59 min. There was 327 g of ihara’s 40-point grading scale was 24 points at discharge (23 days
bleeding with 280 ml of autotransfusion. No problems in cardiores- after operation). At 6 months after the operation, the score was fully
piratory dynamics occurred in the perioperative period. On the day 40 points, and the facial palsy had fully recovered. (Figs. 7, 8A–G,
after the operation, the patient was diagnosed as peripheral facial 9A, B, 10, 11 ).
palsy on the right side. She could not wrinkle the forehead, blink,
close the eyes tightly, wrinkle the nose, or depress the lower lip
3. Discussion
on the right side. The score on the Yanagihara’s 40-point grading
scale was 14 points. It was considered to be caused by a facial nerve
Dendy [1] was the first to report a case of peripheral facial
compression, injury, and/or edema of the surrounding tissue. One
palsy after SSRO. The incidence of peripheral facial palsy has been
thousand five hundred mg/day of mecobalamin was administered
reported from 0.14 to 1% [24]. The reasons for palsy indicated in pre-
immediately. Otorhinolaryngologist suggested steroid pulse treat-
vious reports were as follows: (a) compression by anterior segment
ment with 40 mg/day of prednisolone for one week. On the 9th day
following SSRO, (b) fracture of the styloid process, (c) instrumen-

Fig. 2. Walker-Murdoch wrist sign (A) and Steinberg thumb sign (B).

Please cite this article in press as: Mitate E, et al. Peripheral facial palsy after set-forward orthognathic surgery: A case report and review
of literature. J Oral Maxillofac Surg Med Pathol (2017), https://doi.org/10.1016/j.ajoms.2017.11.002
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Fig. 3. Oral photographs at pretreatment. Angle Class two occlusion with +1 mm of overjet and +6 mm of overbite was found. The mandible was shifted 1 mm to the left.
(A–G).

tation of retractors behind the ramus, (d) traction or compression changes, pain, and intermaxillary fixation are stressors that may
by postoperative hematoma and edema, (e) anatomical variation cause HSV and VZV reactivation perioperatively. Paired serum sam-
of the facial nerve [5], and (f) HSV and VZV reactivation. ples are needed to determine HSV and VZV reactivation. In this
Out of these reasons, compression by anterior segment (a) is case, HSV and VZV reactivation was not found in the paired serum
the most frequently reported in set-back movement of SSRO. Since samples.
we advanced the mandible by SSRO, this did not seem to suit. Dendy and de Vries [1,2] reported that the distance between
In addition, fracture of the styloid process (b) was not found in the ascending ramus and the facial nerve is less than 1 cm in the
postoperative computed tomography. Further, occlusal and airway open-mouth position. Therefore, introduction of retractors behind

Fig. 4. Frontal and lateral cepharograms at pretreatment (A and B).

Please cite this article in press as: Mitate E, et al. Peripheral facial palsy after set-forward orthognathic surgery: A case report and review
of literature. J Oral Maxillofac Surg Med Pathol (2017), https://doi.org/10.1016/j.ajoms.2017.11.002
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Fig. 5. Panoramic radiograph at pretreatment.

Fig. 6. Cepharometric analysis. Convexity: +3 SD, SNB: −3 SD, ANB: +3 SD, Ramus plane to SN: +3 SD, Ramus plane to Frankfurt plane: +2 SD, Intergonial angle: −2 SD,
Occlusal plane to SN: +2 SD.

Fig. 7. Time course of treatment. Mecobalamin and steroid pulse treatment were administered the next day after operation (Day 1). 3000 mg/day of valacyclovir was
administered from Day 9 to Day 14. Linearly polarized near infrared ray irradiation was applied from Day 6 to Day 34 (12 times). Stellate ganglion block was administered
from Day 12 to Day 76 (14 times).

the ascending ramus is a considerable cause for palsy. Traction or excavatum, and maxillary protrusion were found. Because she did
compression by postoperative hematoma and edema, and anatom- not consent to further inspection for MFS, no definitive diagno-
ical variation of the facial nerve are also likely considerable. Since sis was obtained in the present case. However, tissue vulnerability
the present case was suspected of having MFS, tissue vulnerability seen in the present case under traction or compression by retrac-
was also considered. MFS is a systemic disorder of connective tissue tors and/or postoperative hematoma and edema is supposed to be
caused by mutations in fibrillin-1 [6]. Clinical manifestations occur one of the causes of the facial palsy.
in the cardiovascular, skeletal, ocular, and pulmonary systems. In In this report, we used Yanagihara’s facial nerve grading system
this case, Walker-Murdoch wrist & Steinberg thumb signs, pectus to assess facial palsy [7]. This system is a method for clinical assess-

Please cite this article in press as: Mitate E, et al. Peripheral facial palsy after set-forward orthognathic surgery: A case report and review
of literature. J Oral Maxillofac Surg Med Pathol (2017), https://doi.org/10.1016/j.ajoms.2017.11.002
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Fig. 8. Time course of paralysis recovery. Yanagihara’s 40-point grading system shows complete recovery from paralysis at Day 160.

Fig. 9. Oral photographs at six months after operation. The occlusion is maintained as expected. (A–G).

ment of facial motor function. The 10 discrete stages are (1) facial cases (74.6%) were detected within 4 days after operation. Because
symmetry at rest, (2) wrinkling of the forehead, (3) blinking, (4) the amount of bleeding and the length of the operation time were
closure of the eyes lightly, (5) closure of the eyes tightly, (6) clo- not reported in most of the cases, we could not discuss these points.
sure of the eye on the involved side only, (7) wrinkling of the nose, The details of facial nerve paralysis were an inability to wrinkle
(8) whistling, (9) grinning, (10) depression of the lower lip. Each (37 cases), close the eye (49 cases), grin (34 cases), depresses the
aspect was scored as 0 (complete palsy), 2 (partial paralysis), or 4 lower lip (41 cases), and whistle (33 cases). Salivary secretion and
(almost normal). The maximum score is 40, and we diagnosed full stapes reflex were normal in all of the cases. These results suggested
recovery between 36 and 40 points. In this case, the score was 14 at that the facial nerve was damaged at the peripheral side of the
the onset. Mecobalamin, valacyclovir, steroid pulse treatment, lin- stylomastoid foramen.
early polarized near infrared ray irradiation, and stellate ganglion The treatments variations for peripheral facial palsy were
blocks were administered; the score was 24 at discharge. 40 points. administration of vitamins (28 cases), physiotherapy (27 cases),
(Fig. 8). adenosine triphosphate formulation (21 cases), steroids (12 cases),
We summarized our case and 58 previously reported cases of and SGB (7 cases). In 49 cases (83.0%), patients were almost or fully
facial palsy after orthognathic surgery [1–5,8–37]. The data con- recovered in 22.6 (± 21.3) weeks with a range from 10 days [10] to
sisted of 26 males and 33 females (average age was 23.4 years old). 36 months [11]. The palsy remained in 7 cases (11.2%).
Eleven cases (20.3%) were detected on the day of operation, and 44

Please cite this article in press as: Mitate E, et al. Peripheral facial palsy after set-forward orthognathic surgery: A case report and review
of literature. J Oral Maxillofac Surg Med Pathol (2017), https://doi.org/10.1016/j.ajoms.2017.11.002
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Fig. 10. Frontal and lateral cepharograms at 6 months after operation (A and B).

Fig. 11. Panoramic radiograph at 6 months after operation.

Therefore, safer manipulation during orthognathic surgery


(especially SSRO cases) is required. If tissue vulnerability was
expected previously, we have to explain the risk of facial palsy pre-
operatively, take care gently instrumentation intra-operatively,
and observe diligently.

Conflict of interest

The authors declare no conflict of interest.

Acknowledgements
Fig. 12. Summary of case reports (including our case). Ten cases were Le Fort I
osteotomy with sagittal split ramus osteotomy (SSRO). 48 cases were SSRO only. I would like to express my gratitude to Dr. Sei Yoshida, Dr.
Eight cases were set-forward of SSRO. Only Le Fort I osteotomy case report was not Kazuhiko Kubo (Otorhinolaryngology, Kyushu University Hospital),
found. and Dr. Eiji Sakamoto (Dental Anesthesiology, Kyushu University
Hospital).
In the 59 cases (including our case), 10 cases were Le Fort I
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Please cite this article in press as: Mitate E, et al. Peripheral facial palsy after set-forward orthognathic surgery: A case report and review
of literature. J Oral Maxillofac Surg Med Pathol (2017), https://doi.org/10.1016/j.ajoms.2017.11.002
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Please cite this article in press as: Mitate E, et al. Peripheral facial palsy after set-forward orthognathic surgery: A case report and review
of literature. J Oral Maxillofac Surg Med Pathol (2017), https://doi.org/10.1016/j.ajoms.2017.11.002

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