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A holistic approach to stylalgia
Int. J. Oral Maxillofac. Surg. 1986: 15: 549-552 (Key words: stylalgia; syndrome. styloid; syndrome. Eagle's; pain, atypical facial)
A holistic approach to stylalgia GORAN HAMPF, VEIKKO AALBERG, ARVI TASANEN AND CHRISTIAN NYMAN Departments of Maxillo-Facial Surgery and Psychiatry, Helsinki University Central Hospital, Helsinki, Finland
A follow-up study was performed on 20 patients surgically treated because of stylalgia (Eagle's Syndrome). The aim of the study was to investigate why somatic treatment was often ineffective; furthermore, if there was a psychosomatic background to the complaints. The results of a psychiatric interview in which II patients took part clearly showed that most of them were suffering primarily from a psychiatric disorder. The question of stylalgia only as a form of atypical facial pain is discussed.
ABSTRACT -
(Accepted for publication 25 October /985)
Styloid syndrome was clinically described 80 years ago by Dwight and Kyle 3,1I. Their patients suffered from neckache, headache, dysphagia, otalgia and/or TMJ pain. They also suffered from various pains and discomfort in the face, mouth and throat. During the years 1930-1950 Eagle, an English otologist, described more than 200 cases of stylalgia, and because of his special interest in this syndrome, it acquired his name'. Later, it has also been called stylalgia'", styloid syndrome and stylohyoid syndrome. The styloid process develops from the second branchial arch of the Reichert 'cartilage, which is divided into 4 parts': I. the tympanohyalic, which later on fuses almost completely with the petrosus bone; 2. the stylohyalic, which develops into the main part; 3. the ceratohyalic, which usually forms the continuation of the styloid process and is known as the stylohyoideal ligament;
4. the hypohyalic, which forms the minor horn of the hyalic bone, even though the hyalic bone itself develops from the third branchial arch. These different parts may develop differences in ossification or fibrosis. The styloid process goes down and forward from the base of the cranium, normally to a length of 2-3 em, The stylohyoideal ligament continues to the minor horn of the hyalic bone from the distal part of the process and is situated between the external and the internal carotid arteries just behind the pharynx wall in the area of the tonsillar fossa. Three muscles are attached to it: the 11/. styloglossus (supplied by 11. hypoglossus), the 11/. stylopharyngealis (11. glossopharyngeusj and the m. stylohyoideus tn.facialisi. The 11. glossopharyngeus rounds the styloid process from where it sends some motor fibres, the 11. stylopharyngeus, to the soft palate, to the tonsillar region and to the palatal arch.
550
HAMPF, AALBERG, TASANEN AND NYMAN
The sensory part of 11. glossopharyngeus continues to the middle ear and to the tuba acustica. Graf7 pays great attention to this nerve and its role in the syndrome. According to him, patients with glossopharyngicic may suffer from syncope, convulsions, and even cardial arrest. The pain sensation may be due to the stimulation of the sensory nerve endings in the pharynx of the facial nerve, the trigeminal nerve, and the hypoglossal nerve. Recently these clinical features have also been connected with atypical facial pain s,9,12,13,14,16. Most patients suffer from different kinds of dysesthesia in the facial area and from pain when swallowing, radiating to the TMJ region. The pain may take the form of a dull ache or a sharp, shooting, glossopharyngic tic-like pain . This may result in the patient being referred for tonsillectomy, treatment of the TMJ, or other treatment in the region, which seldom helps the patient and may simply have a placebo effects. Difficulty in swallowing and the sensation of'a foreign body in the throat might be explained by the rigidity of the styloideal system in the case of extreme pathological ossification, 'although the same symptoms arc also common in atypical facial pain . The normal styloid process is about 3 em long and is easily seen on x-rays, though it is usually not palpable. The prolonged process is palpable behind the pharynx arch in the tonsillar fossa if it is more than 7.5 em long. Elogated processes arc found in about 4% of the normal population and 4% of these show symptoms of stylalgia'. The following conditions should be kept in mind as possible differential diagnoses: impacted wisdom tooth, adenosis of the submandibular gland, pharyngitis, tonsillitis, tumour of the tongue base, cervical arthrosis, TMJ dysfunction or arthrosis of the TMJ, specific neuralgias, and last but not least, a previous psychiatric disorder-". The aim of this study was to investigate
why somatic treatment was often ineffective, and why the diagnosis "stylalgia" was so difficult to make, so diffuse, and so misleading.
Material and methods The material consisted of 20 patients with signs and symptoms of stylalgia, 19 females and I male between 22 and 71 years of age (mean 47). They had been suffering from stylalgic types of pain for a period ranging from 2 months to 20 years (mean 3.5 years). All the patients had been examined and surgically treated in the Department of Maxilla-Facial Surgery of the Helsinki University Central Hospital during the previous 10 years. The diagnosis was based on the complaints and/or the palpatory findings (Tables I and 2),
Table I. Anamnestic findings in 20 patients suffering from stylalgia Pain in the styloideus region 10 Pain in the TMJ region 8 Referred pain - on the temple 9 - in the masseteric region 6 5 - in the infraorbital region 4 - in front of the ear - behind the ear 4 - in the neek 4 - superficially on the mandible I Swelling sensation in the mouth 4 Pain on biting 4 Dysphagia 3 Glossodynia I Tinnitus I
Table 2. Palpatory findings in 20 patients suffering from stylalgia EXTRAORALLY 2 Pain in front of the ear Pain in the mandibular angulus 2 Pain in the masseteric muscle I Pain behind the ear I INTRAORALLY Styloid process palpable - pain on palpation - no pain Pain in the retromolar region Pain in the coronoidal region
14 13 I I I
EAGLE'S SYNDROME The treatment was surgical resection of the processus styloldeus or of the calcified or fibrotic ligament under general anaesthesia, in one case intraorally, and in 19 cases extraorally. In some cases, the resectioned processes were sent for histological examination. In the follow-up study, the patients were reexamined, new X-rays taken, and the patients referred for a psychiatric consultation based on a semi-structured psychodynamic interview of about 1.5 h duration. In the interview, the DSM III criteria were used',
551
Table 3. Psychiatric disorders of20 patients treated for stylalgia Atypical psychosis Major affective psychosis with psychotic features, in remission I Paranoid personality disorder I Histrionic personality disorder I Hypochondriasis 1 Dysthymic disorder 2 Post-traumatic stress disorder 1 No diagnosis (mentally healthy) 3 Diagnosis unknown (patient refused consultation) 9
Results The x-rays revealed a prolonged process in 14 cases out of 20, despite the fact that the diagnosis "stylalgia" was made according to other symptoms described in Tables I and 2. Increased calcification of the styloid ligament was seen in eight cases, a normal finding was noted in nine cases, and the remaining three X-rays were not available prior to operation. The primary results of the operations were fairly good in half of the patients. Later results showed that one of the patients was free from pain, while 12 had less pain than before the operation. Six patients experienced no relief from pain, and in one case the pain was worse than before the operation. A slight dysfunction of the lower facial nerve was noted in two cases and a fistula of long duration appeared in one case. Nine of the patients agreed to a psychiatric consultation, and, in two cases, earlier psychiatric records were found. Table 3 shows the psychiatric diagnoses and includes the two patients for whom earlier psychiatric diagnoses were available. In the psychiatric investigation of seven patients, a traumatic childhood was found; in nine cases, the patients developed stylalgia simultaneously with stress factors. Even the patients with no psychiatric diagnosis (mentally healthy) were under some stress at the
moment of onset of pain. In two cases, there was a facial injury prior to the appearance of stylalgia. Nine of the patients refused the interview (diagnosis "unknown").
Discussion Technically, most of the operations were successful, yet many of the patients experienced a return of pain and were given auxiliary treatment (Table 4). Most of the patients had undergone at least some treatment or examination before the operation (Table 5). According to the psychiatric investigation, most of the patients were suffering primarily from a psychiatric disorder. Even the patients who refused the psychiatric interview seemed to be at least as mentally disturbed as those who consented to the interview.
Table 4. Auxiliary treatment of 10 of 20 patients after styloideus resection Injection of glucocorticoids into the TMJ 2 Condylotomy 2 Dental prosthesis renewed 1 Revisio mandibulae 1 Resectio cristae mylohyoidea I Drug therapy 1 Lower lip training 1 TMJ physiotherapy 1 TMJ arthroplasty I Psychiatric treatment 1
552
HAMPF, AALBERG, TASANEN AND NYMAN
Table 5. Treatment and examination of 17 of 20 patients before styloideus resection Condylotomy Drug therapy Injection of glucocorticoids into the TMJ Physiotherapy Dental prosthesis renewed Injection of local anaesthetics Failed styloideus resection Neuroplexia of the mental nerve Carotid artery angiography Other examinations and/or treatments
7 5 4 4 4 3 2 I I 3
The diagnosis of atypical facial pain had probably been correct in many of the cases; the same clinical features have been described by several authors 5.9•12,14.16 and diagnosed as atypical facial pain. The sex and age distributions of the present patients correspond with some earlier reports of stylalgia or atypical facial pain5.1o.12. The findings in Tables I and 2 demonstrate that there was a strong behavioral or psychosomatic background to the complaints despite the fact that none of the patients were aware of mental disturbance or felt ·the need for psychiatric treatment.
Conclusion According to the findings of this study, patients with pain typical of stylalgia should be sent for a psychiatric consultation in order to exclude a mental disorder before surgical or other treatment is administered.
2. BADDOUR, H. M .: Eagle's syndrome. Oral Surg , 1978: 46: 486-494. 3. DWIGHT, T.: Styloid ossification. Ann. Surg, 1907: 46: 721-735. 4. EAGLE, W. W.: Elongated Styloid Process; symptoms and treatment. Arch. Otolaryngol. 1958: 67: 172-176. 5. ENGEL, G. L.: Primary atypical facial neuralgia, an hysterical conversion symptom. Psychosom, Med. 1951: 13: 375-396. 6. GOSSMAN, J. R.: The styloid-stylohyoid syndrome. J. Oral Surg, 1977: 35: 555-560. 7. GRAF, C. J.: Glossopharyngeal neuralgia and ossification of the styloid ligament. J. Neurosurg, 1959: 16: 448-453. 8. HAMPF, G.: Dilemma in treatment of patients suffering from orofacial dysesthesia. Manuscript. 9. HARRIS, M.: Psychogenic aspects of facial pain. Sr. Dent . J. 1974: 136: 199-202. 10. HARMA, R.: Stylalgia. Acta Otolaryngol. Suppl. 1966: 224: 149-155. I I. KYLE, J. J.: Anatomy and diseases of the styloid epiphysis. Ann. Otol. Rhinal. Laryngal. 1909: 18: 128-134 . 12. LASCELLES, R. G.: Atypical facial pain and depression. Sr. J. Psychiat, 1966: 112: 651-659. 13. MOULTON, R. E.: Emotional Factors in NonOrganic Temporomandibular Joint Pain. Dent .:Clin. North Am. 1966: 10: 651-659. 14. MOULTON, R. E.: -Psychiatric considerations in maxillofacial pain. J. Am. Dent. Assoc. 1955: 51: 408-414. 15. RUSSELL, T. E.: Eagle's syndrome: diagnostic considerations and report of a case. J. Am. Dent. Assoc. 1977: 94: 548-550. 16. SPECULAND, B., Goss, A. N., HALLETT, E. & SPENCE, N . D.: Intractable facial pain. Sr. J. Oral Surg, 1979-1980: 17: 166-178.
Address:
References l. AMERICAN PSYCHlATRIC ASSOCIATION (APA): Diagnostic and Statistical Manual of Mental Disorders, 3rd ed. Washington, D.C. 1980.
G. Hampf Department of Oral Surgery University of Helsinki Mannerheimintie 172 00280 Helsinki 28. Finland
A holistic approach to stylalgia GORAN HAMPF, VEIKKO AALBERG, ARVI TASANEN AND CHRISTIAN NYMAN Departments of Maxillo-Facial Surgery and Psychiatry, Helsinki University Central Hospital, Helsinki, Finland
A follow-up study was performed on 20 patients surgically treated because of stylalgia (Eagle's Syndrome). The aim of the study was to investigate why somatic treatment was often ineffective; furthermore, if there was a psychosomatic background to the complaints. The results of a psychiatric interview in which II patients took part clearly showed that most of them were suffering primarily from a psychiatric disorder. The question of stylalgia only as a form of atypical facial pain is discussed.
ABSTRACT -
(Accepted for publication 25 October /985)
Styloid syndrome was clinically described 80 years ago by Dwight and Kyle 3,1I. Their patients suffered from neckache, headache, dysphagia, otalgia and/or TMJ pain. They also suffered from various pains and discomfort in the face, mouth and throat. During the years 1930-1950 Eagle, an English otologist, described more than 200 cases of stylalgia, and because of his special interest in this syndrome, it acquired his name'. Later, it has also been called stylalgia'", styloid syndrome and stylohyoid syndrome. The styloid process develops from the second branchial arch of the Reichert 'cartilage, which is divided into 4 parts': I. the tympanohyalic, which later on fuses almost completely with the petrosus bone; 2. the stylohyalic, which develops into the main part; 3. the ceratohyalic, which usually forms the continuation of the styloid process and is known as the stylohyoideal ligament;
4. the hypohyalic, which forms the minor horn of the hyalic bone, even though the hyalic bone itself develops from the third branchial arch. These different parts may develop differences in ossification or fibrosis. The styloid process goes down and forward from the base of the cranium, normally to a length of 2-3 em, The stylohyoideal ligament continues to the minor horn of the hyalic bone from the distal part of the process and is situated between the external and the internal carotid arteries just behind the pharynx wall in the area of the tonsillar fossa. Three muscles are attached to it: the 11/. styloglossus (supplied by 11. hypoglossus), the 11/. stylopharyngealis (11. glossopharyngeusj and the m. stylohyoideus tn.facialisi. The 11. glossopharyngeus rounds the styloid process from where it sends some motor fibres, the 11. stylopharyngeus, to the soft palate, to the tonsillar region and to the palatal arch.
550
HAMPF, AALBERG, TASANEN AND NYMAN
The sensory part of 11. glossopharyngeus continues to the middle ear and to the tuba acustica. Graf7 pays great attention to this nerve and its role in the syndrome. According to him, patients with glossopharyngicic may suffer from syncope, convulsions, and even cardial arrest. The pain sensation may be due to the stimulation of the sensory nerve endings in the pharynx of the facial nerve, the trigeminal nerve, and the hypoglossal nerve. Recently these clinical features have also been connected with atypical facial pain s,9,12,13,14,16. Most patients suffer from different kinds of dysesthesia in the facial area and from pain when swallowing, radiating to the TMJ region. The pain may take the form of a dull ache or a sharp, shooting, glossopharyngic tic-like pain . This may result in the patient being referred for tonsillectomy, treatment of the TMJ, or other treatment in the region, which seldom helps the patient and may simply have a placebo effects. Difficulty in swallowing and the sensation of'a foreign body in the throat might be explained by the rigidity of the styloideal system in the case of extreme pathological ossification, 'although the same symptoms arc also common in atypical facial pain . The normal styloid process is about 3 em long and is easily seen on x-rays, though it is usually not palpable. The prolonged process is palpable behind the pharynx arch in the tonsillar fossa if it is more than 7.5 em long. Elogated processes arc found in about 4% of the normal population and 4% of these show symptoms of stylalgia'. The following conditions should be kept in mind as possible differential diagnoses: impacted wisdom tooth, adenosis of the submandibular gland, pharyngitis, tonsillitis, tumour of the tongue base, cervical arthrosis, TMJ dysfunction or arthrosis of the TMJ, specific neuralgias, and last but not least, a previous psychiatric disorder-". The aim of this study was to investigate
why somatic treatment was often ineffective, and why the diagnosis "stylalgia" was so difficult to make, so diffuse, and so misleading.
Material and methods The material consisted of 20 patients with signs and symptoms of stylalgia, 19 females and I male between 22 and 71 years of age (mean 47). They had been suffering from stylalgic types of pain for a period ranging from 2 months to 20 years (mean 3.5 years). All the patients had been examined and surgically treated in the Department of Maxilla-Facial Surgery of the Helsinki University Central Hospital during the previous 10 years. The diagnosis was based on the complaints and/or the palpatory findings (Tables I and 2),
Table I. Anamnestic findings in 20 patients suffering from stylalgia Pain in the styloideus region 10 Pain in the TMJ region 8 Referred pain - on the temple 9 - in the masseteric region 6 5 - in the infraorbital region 4 - in front of the ear - behind the ear 4 - in the neek 4 - superficially on the mandible I Swelling sensation in the mouth 4 Pain on biting 4 Dysphagia 3 Glossodynia I Tinnitus I
Table 2. Palpatory findings in 20 patients suffering from stylalgia EXTRAORALLY 2 Pain in front of the ear Pain in the mandibular angulus 2 Pain in the masseteric muscle I Pain behind the ear I INTRAORALLY Styloid process palpable - pain on palpation - no pain Pain in the retromolar region Pain in the coronoidal region
14 13 I I I
EAGLE'S SYNDROME The treatment was surgical resection of the processus styloldeus or of the calcified or fibrotic ligament under general anaesthesia, in one case intraorally, and in 19 cases extraorally. In some cases, the resectioned processes were sent for histological examination. In the follow-up study, the patients were reexamined, new X-rays taken, and the patients referred for a psychiatric consultation based on a semi-structured psychodynamic interview of about 1.5 h duration. In the interview, the DSM III criteria were used',
551
Table 3. Psychiatric disorders of20 patients treated for stylalgia Atypical psychosis Major affective psychosis with psychotic features, in remission I Paranoid personality disorder I Histrionic personality disorder I Hypochondriasis 1 Dysthymic disorder 2 Post-traumatic stress disorder 1 No diagnosis (mentally healthy) 3 Diagnosis unknown (patient refused consultation) 9
Results The x-rays revealed a prolonged process in 14 cases out of 20, despite the fact that the diagnosis "stylalgia" was made according to other symptoms described in Tables I and 2. Increased calcification of the styloid ligament was seen in eight cases, a normal finding was noted in nine cases, and the remaining three X-rays were not available prior to operation. The primary results of the operations were fairly good in half of the patients. Later results showed that one of the patients was free from pain, while 12 had less pain than before the operation. Six patients experienced no relief from pain, and in one case the pain was worse than before the operation. A slight dysfunction of the lower facial nerve was noted in two cases and a fistula of long duration appeared in one case. Nine of the patients agreed to a psychiatric consultation, and, in two cases, earlier psychiatric records were found. Table 3 shows the psychiatric diagnoses and includes the two patients for whom earlier psychiatric diagnoses were available. In the psychiatric investigation of seven patients, a traumatic childhood was found; in nine cases, the patients developed stylalgia simultaneously with stress factors. Even the patients with no psychiatric diagnosis (mentally healthy) were under some stress at the
moment of onset of pain. In two cases, there was a facial injury prior to the appearance of stylalgia. Nine of the patients refused the interview (diagnosis "unknown").
Discussion Technically, most of the operations were successful, yet many of the patients experienced a return of pain and were given auxiliary treatment (Table 4). Most of the patients had undergone at least some treatment or examination before the operation (Table 5). According to the psychiatric investigation, most of the patients were suffering primarily from a psychiatric disorder. Even the patients who refused the psychiatric interview seemed to be at least as mentally disturbed as those who consented to the interview.
Table 4. Auxiliary treatment of 10 of 20 patients after styloideus resection Injection of glucocorticoids into the TMJ 2 Condylotomy 2 Dental prosthesis renewed 1 Revisio mandibulae 1 Resectio cristae mylohyoidea I Drug therapy 1 Lower lip training 1 TMJ physiotherapy 1 TMJ arthroplasty I Psychiatric treatment 1
552
HAMPF, AALBERG, TASANEN AND NYMAN
Table 5. Treatment and examination of 17 of 20 patients before styloideus resection Condylotomy Drug therapy Injection of glucocorticoids into the TMJ Physiotherapy Dental prosthesis renewed Injection of local anaesthetics Failed styloideus resection Neuroplexia of the mental nerve Carotid artery angiography Other examinations and/or treatments
7 5 4 4 4 3 2 I I 3
The diagnosis of atypical facial pain had probably been correct in many of the cases; the same clinical features have been described by several authors 5.9•12,14.16 and diagnosed as atypical facial pain. The sex and age distributions of the present patients correspond with some earlier reports of stylalgia or atypical facial pain5.1o.12. The findings in Tables I and 2 demonstrate that there was a strong behavioral or psychosomatic background to the complaints despite the fact that none of the patients were aware of mental disturbance or felt ·the need for psychiatric treatment.
Conclusion According to the findings of this study, patients with pain typical of stylalgia should be sent for a psychiatric consultation in order to exclude a mental disorder before surgical or other treatment is administered.
2. BADDOUR, H. M .: Eagle's syndrome. Oral Surg , 1978: 46: 486-494. 3. DWIGHT, T.: Styloid ossification. Ann. Surg, 1907: 46: 721-735. 4. EAGLE, W. W.: Elongated Styloid Process; symptoms and treatment. Arch. Otolaryngol. 1958: 67: 172-176. 5. ENGEL, G. L.: Primary atypical facial neuralgia, an hysterical conversion symptom. Psychosom, Med. 1951: 13: 375-396. 6. GOSSMAN, J. R.: The styloid-stylohyoid syndrome. J. Oral Surg, 1977: 35: 555-560. 7. GRAF, C. J.: Glossopharyngeal neuralgia and ossification of the styloid ligament. J. Neurosurg, 1959: 16: 448-453. 8. HAMPF, G.: Dilemma in treatment of patients suffering from orofacial dysesthesia. Manuscript. 9. HARRIS, M.: Psychogenic aspects of facial pain. Sr. Dent . J. 1974: 136: 199-202. 10. HARMA, R.: Stylalgia. Acta Otolaryngol. Suppl. 1966: 224: 149-155. I I. KYLE, J. J.: Anatomy and diseases of the styloid epiphysis. Ann. Otol. Rhinal. Laryngal. 1909: 18: 128-134 . 12. LASCELLES, R. G.: Atypical facial pain and depression. Sr. J. Psychiat, 1966: 112: 651-659. 13. MOULTON, R. E.: Emotional Factors in NonOrganic Temporomandibular Joint Pain. Dent .:Clin. North Am. 1966: 10: 651-659. 14. MOULTON, R. E.: -Psychiatric considerations in maxillofacial pain. J. Am. Dent. Assoc. 1955: 51: 408-414. 15. RUSSELL, T. E.: Eagle's syndrome: diagnostic considerations and report of a case. J. Am. Dent. Assoc. 1977: 94: 548-550. 16. SPECULAND, B., Goss, A. N., HALLETT, E. & SPENCE, N . D.: Intractable facial pain. Sr. J. Oral Surg, 1979-1980: 17: 166-178.
Address:
References l. AMERICAN PSYCHlATRIC ASSOCIATION (APA): Diagnostic and Statistical Manual of Mental Disorders, 3rd ed. Washington, D.C. 1980.
G. Hampf Department of Oral Surgery University of Helsinki Mannerheimintie 172 00280 Helsinki 28. Finland