Oromandibular dystonia is a focal dystonia involving sustained or intermittent contractions of the masticatory, lingual, and/or other stomatognathic system muscles. 1 David Marsden published a study concerning blepharospasm‐oromandibular dystonia, suggesting that it be called Brueghel's syndrome. 2 Marsden cited “De Gaper” by Pieter Brueghel the Elder, which depicts involuntary jaw opening and eye closure (Fig. 1A), assuming Brueghel had observed this syndrome. However, other researchers have been less convinced, seeing the subject caught facial expression during yawning to exhibit the artist's dexterity. 3 , 4 Garcia‐Ruiz et al 5 suggested that a sculpture of Xipe Totec (300–900 AD) with closed eyes, opened jaw, and tongue protrusion (Fig. 1B) represents classical Meige syndrome. Nevertheless, in Aztec mythology, Xipe Totec refers to the god of life, death, and rebirth. The statues of the god have a typical pattern of closed eyes, mouth opening, and tongue protrusion, making it difficult to imagine them as representative illnesses.
FIG. 1.
(A) “Yawning Man” or “De Gaper” (Musées Royaux des Beaux‐Arts, Bruxelles), by Pieter Brueghel the Elder (1525–1569), around 1560. (B) Sculpture of Xipe‐Totec made of terracotta (Vera‐cruz Culture heritage, Mexico, 300–900 AD). Reproduced from Garcia‐Ruiz et al. 5 with permission of the publisher.
“Yamai no Soshi,” which means scroll of diseases, is a picture scroll painted in the 12th century, and comprises a simple narrative with one illustration (Fig. 2). It describes the symptoms and treatments of various illnesses, including rare diseases and deformities. The authors of the illustrations and text are unknown. Nine of the remaining 21 sections are preserved at the Kyoto National Museum and designated as National Treasures. The nine sections include; a man with “fuubyo” (Fig. 2), man with tongue tumor, man with very severe ileus, hermaphrodite man, man with cataracts, man with periodontal disease, man with anal fistula, man infected with hair lice, and woman with extremely severe diarrhea and vomiting.
FIG. 2.
Overall view “Fuubyo no Otoko (a man with wind disease)” and close‐up of the face. “Yamai no Soshi” (Kyoto National Museum, Kyoto), 12th century, National Treasure.
The man with “fuubyo” is depicted with involuntary movements of the fingers and face (Fig. 2). The inscription reads (Fig. 2), “The man was shivering and shaking as if he were naked in the bitter cold.” Before, “fuubyo,” which means wind disease, was believed to be caused by the poison of the wind. It is a general term for illnesses accompanied by symptoms, such as headaches, pain, abnormal sensations in the limbs, speech disorders, and limb movement disorders. The man playing “go” had a distorted face, his mouth was open, and his mandible deviated to the left. His fingers tremble and are unable to move steadily as he plays with the “go” stones. Women playing with him giggled as they watched him. Trembling fingers are suggested to be tremors and jaw opening can be oromandibular dystonia (jaw opening or deviation dystonia). 1
“De Gaper” (Fig. 1A) and the statue of Xipe Totec (Fig. 1B) are postulated as cranial dystonia from only facial expressions. Conversely, “Yamai no Soshi” is a scroll depicting rare diseases, and from the inscription, the man definitely suffered from a movement disorder. “Yamai no Soshi” is the oldest description of oromandibular dystonia worldwide.
Author Roles
(1) Research project: A. Conception, B. Organization, C. Execution; (2) Statistical Analysis: A. Design, B. Execution, C. Review and Critique; (3) Manuscript Preparation: A. Writing of the First Draft, B. Review and Critique.
K.Y.: 1A, 1B, 1C, 2A, 2B, 2C, 3A, 3B
Disclosures
Ethical Compliance Statement: Approval of an Institutional Review Board and patient consent were not required. I confirm that I have read the Journal's position on issues involved in ethical publication and affirm that this work is consistent with those guidelines.
Funding Sources and Conflicts of Interest: This study was supported by the Japan Society for the Promotion of Science (JSPS) and KAKENHI (JP22K10091). The author declares no conflict of interest.
Financial Disclosures for the Previous 12 Months: The author declares that there are no conflicts of interest relevant to this work.
Acknowledgment
None.
Relevant disclosures and conflict of interest are listed at the end of this article.
Data Availability Statement
The data that support the findings of this study are available from the corresponding author upon reasonable request.
References
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Associated Data
This section collects any data citations, data availability statements, or supplementary materials included in this article.
Data Availability Statement
The data that support the findings of this study are available from the corresponding author upon reasonable request.