1427
MALINS AND FARROW
J Oral
Maxillo?ac Surg
49:1127-1129.1991
Due t Cystic %.,,I.MALBNS, FDSRCS, FRCS,* AND A. FARRQW, B5S-f
Adenoid cystic carckroma comprises some 5% of ah salivary tumors of the major glands, the parotid being the major gland most often affected. Adenoid cystic carcinomas grow slowly and because of their propensity for infiltration of nerves, pain or other neurological symptoms may be present. Two cases are presented in which facial pain predated the diagnosis of a parotid tumor. In the first case there was remarkedly long interval of 7 years, and in the second 18 months, between the onset of pain and the detection of the tumor. The possibility of occult neoplasia should always be considered when examining a patient with facial pam. In both cases, combined surgery and radiotherapy resulted in control of the disease and relief of pain
Case 1 A 44-year-old white woman initially presented to an otorhinohu-yngologist in 1981 complaining of pain in the right mastoid region. She described a burning pain over the mastoid, which was constantly present, radiated over the temporomandibular joint (TMJ), and was exacerbated by eating and psychological stress. Her symptoms had been present for 3 years. Following clinical and radiologic examination, a diagnosis of cervical spondyiosis was made. Subsequent physiotherapy, however, had little effect on her symptoms. In 1982, the patient was examined by a general surgeon who sought the opinion of another ENT surgeon. With the exception of slight tenderness anterior to the mastoid process, there were no findings of note. Radiographs of the mastoid were normal. In 1983 the pain was diagnosed
Received from the Department of Oral and Maxillofacial Surgery, Westminster Hospital, London. * Senior Registrar. t House Officer. Address correspondence and reprint requests to Dr Malins: Department of Oral and Maxillofacial Surgery, Westminster Hospital, Horseferry Rd, London SWlP 2AP, England.
0 1991 American Association of Oral and Maxillofacial Surgeons 0278-2391/91/4909-0048$3.OO/O
as idiopathic trigemina? neuralgia by a physician and treated with carbamazepine, 400 mg daily, with no effect. In 19&5 the patient was referred to the National Hospita1 for Nervous Diseases. Examination by a neurologist revealed no neurologic signs; however, she was noted to be psychologically depressed. There was tenderness over the right TMJ. In addition, a small, indurated area was noted in the retromandibular region. She was started on amitriptyline, 25 mg, and referred to the surgeon who had examined her in 1982. On this occasion she was noted to have a 1.5-cm nonmobile swelling in the retromandibular region. A snbsequent computed tomography (CT) scan revealed a well circumscribed, low-attenuation lesion in the superficial lobe of the right parotid gland. In March $986, a superficial parotidectomy was performed. Histologic examination (Fig I) showed a poorly circumscribed adenoid cystic carcinoma with a narrow excision margin. The patient received postoperative radiotherapy, 6,400 cGy, to the parotid region. IIer pain resolved postoperatively. She remains well, with no furth.er pain or recurrence of the tumor.
Case 2 A 33-year-old white woman was initially seen in an oral surgery department in 1983 regarding bouts of tenderness in the right temporomandibular region. On clinical examination she was noted to be tender over the right condyle, which was also prominent and appeared to subluxate on wide opening. She was diagnosed as suB?ering from TMJ dysfunction syndrome and treated with analgesics. At review 1 year later, her symptoms were no better aml she was referred for another opinion. In 1984, following a further examination by an oral surgeon, the diagnosis was confirmed and she received physiotherapy. Two months later, on reexamination, she was noted to have a swelling over the right condyle, which was tender. A parotid sialogram was noncontributory and, therefore, it was decided to explore the region, At operation, a l-cm mass overlying the capsule was encountered. The lesion was excised and histologic examination confirmed the presence of an adenoid cystic carcinoma (Fig 2). In view of uncertainty regarding complete removal, the patient subsequently had an extended superfkial parotidectomy with sacrifice of the temporal branch of the facial nerve and temporalis muscle. This was folkwed by radiotherapy, 6,000 cGy, to the area and to the skull base. Postoperatively her pain resolved and she remains well and disease free.
1128
FACIAL PAIN DUE TO OCCULT PARBTID TUMOR.
FIGURE 1, Phqtorqicrograph of adenoid cystic carcinoma from case 1 showing perineural tumor invasion @hematoxylin-eosin staik, original magnification X 125).
Disscwssiem The diagnosis of facial pain remains an area with many pitfalls for the unwary. The care of patients with facial pain may include many specialities and the danger arises that each specialist may presume a cause without considering all the differential diagnoses. In addition, each specialty develops clinical skills in examination relevant to their own special-
FIGURE 2. Photomicrograph of adenoid cystic carcinoma from case 2 showing extensive perineural tumor invasion (hematoxylin-eosin stain, original magnification X 100).
ity, which may overlook some of the less obvious causes. Both TMJ dysfunction and idiopathic trigeminal neuralgia are conditions routinely diagnosed and treated by oral and maxillofacial surgeons. The coexistence of TMJ pain and parotid neoplasms has been previously reported.‘T2 In the experience of Shapshay et al* with occult tumors, five of seven patients with infratemporal fossa neoplasms were
WOLF, KESSLER,
%129
AND HGROVITZ
initially given the diagnosis of TMJ dysfunction. Two of this group actually had parotid neoplasms. Extraoral neoplasms also can cause pain that resembles trigeminal neuralgia. The pain is usually more persistent and there may be other neurologic signs and symptoms, including sensory changes. Differential diagnoses in patients with facial pain and sensory changes include tumors of the infratemporal fossa, gasserian ganglion, pontine tumors, acoustic neuroma, glioma, meningioma, cholesteatoma, arterial abnormalities, and cervical spondylosis. Systemic conditions include the connective tissue disorders, eg, polyarteritis nodosa, multiple sclerosis, neuritis secondary to sarcoidosis, and syphilis. The importance of numbness as a significant finding has been previously emphasized.3,4 Atypical facial neuralgia is a common form of facial pain. In Rusthton’s survey5 of patients initially diagnosed as having atypical facial pain, more than 50% of the cases were placed under the category of psychiatric disease. Common organic causes were vasodilating facial pain, dental disease, neuritis, and neoplasms. In the last group were two cases of adenoid cystic carcinoma. Chronic pain may pre-
J Oral Maxillofac
cipitate psychological depression; most of the patients in Shapsey et al’s series’ had such severe symptoms. Bn the other hand, the chnician must avoid the temptation to consider the pain as being a manifestation of depression. The key to the management of patients with facial pain is an accurate and expeditious diagnosis. Delay in diagnosis allows the disease to progress, successful treatment may have to be more radical, and the prognosis may be significantly altered. Thorough ear, nose, throat, dental, and neurologic examination should be performed, and repeated, should %he symptoms persist. References 1. Grace EG, North AF: Temporomandibular joint dysfunction and orofacial pain caused by parotid gland malignancy: Report of a case. J Am Dent Assoc 115:348, 1988 2. Shapsey SM, Elber E, Strong SM: Occult tumors of the infratemporal fossa. Arch Otolaryngol 102535, 1976 3. Thrush DC, Small M: How benign a symptom is facial numbness? Lancet 2% 1, 1970 4. Roistacher SL: Numbness-A significant finding. Oral Surg Oral Med Oral Path01 36:22, 1973 5. Rushton JG, Gibilisen JA, Goldstein SP: Aty.pical facial pain. JAMA 171545, 19.59
Surg
49:1129-1131,199l
OLF, MD,* ALEXANDER
KESSLER,
Diagnosis of a solitary mass of the neck is a challenge for the clinician. Nearly 80% of cervical masses are neoplastic, increasing to 90% over the age of 90. Eighty-five percent of cervical masses in patients under the age of 21 are benign and include Received from the Chaim-Sheba Medical Center, TelHashomer, and Sackler School of Medicine, Tel-Aviv, Israel. * Department of Otorhinolaryngology. I Department of Gtorhinolaryngology. f Department of Pathology. Address correspondence and reprint requests to Dr Wolf: Department of Otorhinolaryngology, Chaim-Sheba Medical Center, Tel-Hashomer, 52621, Israel. 0 1991 American geons
Association of Oral and Maxillofacial
Sur-
MDJ
AND ADA WOR
mainly reactive lymph nodes and congenital cysts or malformations.’ Castelman’s disease (CD>, more commonly known as benign angiofohicular lymph node hyperplasia, is a rare condition that involves a localized lymphatic enlargement with characteristic microscopic features. It commomy invdves the mediastinal and ptdmonary lymph nodes, with neck involvement in only 15% to 20% of ca~es.~‘~ There are two primary variants: the hyaline-vascular and plasma cell types. The hyaline-vascular type comprises 80% of cases and is usually asymptomatic, unless there is impingement on adjacent structures. The plasma cell type is often associated with systemic manifestations, the most common of which are fever, anemia, elevated erythrocyte sedimentation rate, and altered plasma proteins levels4 A
MALINS AND FARROW
J Oral
Maxillo?ac Surg
49:1127-1129.1991
Due t Cystic %.,,I.MALBNS, FDSRCS, FRCS,* AND A. FARRQW, B5S-f
Adenoid cystic carckroma comprises some 5% of ah salivary tumors of the major glands, the parotid being the major gland most often affected. Adenoid cystic carcinomas grow slowly and because of their propensity for infiltration of nerves, pain or other neurological symptoms may be present. Two cases are presented in which facial pain predated the diagnosis of a parotid tumor. In the first case there was remarkedly long interval of 7 years, and in the second 18 months, between the onset of pain and the detection of the tumor. The possibility of occult neoplasia should always be considered when examining a patient with facial pam. In both cases, combined surgery and radiotherapy resulted in control of the disease and relief of pain
Case 1 A 44-year-old white woman initially presented to an otorhinohu-yngologist in 1981 complaining of pain in the right mastoid region. She described a burning pain over the mastoid, which was constantly present, radiated over the temporomandibular joint (TMJ), and was exacerbated by eating and psychological stress. Her symptoms had been present for 3 years. Following clinical and radiologic examination, a diagnosis of cervical spondyiosis was made. Subsequent physiotherapy, however, had little effect on her symptoms. In 1982, the patient was examined by a general surgeon who sought the opinion of another ENT surgeon. With the exception of slight tenderness anterior to the mastoid process, there were no findings of note. Radiographs of the mastoid were normal. In 1983 the pain was diagnosed
Received from the Department of Oral and Maxillofacial Surgery, Westminster Hospital, London. * Senior Registrar. t House Officer. Address correspondence and reprint requests to Dr Malins: Department of Oral and Maxillofacial Surgery, Westminster Hospital, Horseferry Rd, London SWlP 2AP, England.
0 1991 American Association of Oral and Maxillofacial Surgeons 0278-2391/91/4909-0048$3.OO/O
as idiopathic trigemina? neuralgia by a physician and treated with carbamazepine, 400 mg daily, with no effect. In 19&5 the patient was referred to the National Hospita1 for Nervous Diseases. Examination by a neurologist revealed no neurologic signs; however, she was noted to be psychologically depressed. There was tenderness over the right TMJ. In addition, a small, indurated area was noted in the retromandibular region. She was started on amitriptyline, 25 mg, and referred to the surgeon who had examined her in 1982. On this occasion she was noted to have a 1.5-cm nonmobile swelling in the retromandibular region. A snbsequent computed tomography (CT) scan revealed a well circumscribed, low-attenuation lesion in the superficial lobe of the right parotid gland. In March $986, a superficial parotidectomy was performed. Histologic examination (Fig I) showed a poorly circumscribed adenoid cystic carcinoma with a narrow excision margin. The patient received postoperative radiotherapy, 6,400 cGy, to the parotid region. IIer pain resolved postoperatively. She remains well, with no furth.er pain or recurrence of the tumor.
Case 2 A 33-year-old white woman was initially seen in an oral surgery department in 1983 regarding bouts of tenderness in the right temporomandibular region. On clinical examination she was noted to be tender over the right condyle, which was also prominent and appeared to subluxate on wide opening. She was diagnosed as suB?ering from TMJ dysfunction syndrome and treated with analgesics. At review 1 year later, her symptoms were no better aml she was referred for another opinion. In 1984, following a further examination by an oral surgeon, the diagnosis was confirmed and she received physiotherapy. Two months later, on reexamination, she was noted to have a swelling over the right condyle, which was tender. A parotid sialogram was noncontributory and, therefore, it was decided to explore the region, At operation, a l-cm mass overlying the capsule was encountered. The lesion was excised and histologic examination confirmed the presence of an adenoid cystic carcinoma (Fig 2). In view of uncertainty regarding complete removal, the patient subsequently had an extended superfkial parotidectomy with sacrifice of the temporal branch of the facial nerve and temporalis muscle. This was folkwed by radiotherapy, 6,000 cGy, to the area and to the skull base. Postoperatively her pain resolved and she remains well and disease free.
1128
FACIAL PAIN DUE TO OCCULT PARBTID TUMOR.
FIGURE 1, Phqtorqicrograph of adenoid cystic carcinoma from case 1 showing perineural tumor invasion @hematoxylin-eosin staik, original magnification X 125).
Disscwssiem The diagnosis of facial pain remains an area with many pitfalls for the unwary. The care of patients with facial pain may include many specialities and the danger arises that each specialist may presume a cause without considering all the differential diagnoses. In addition, each specialty develops clinical skills in examination relevant to their own special-
FIGURE 2. Photomicrograph of adenoid cystic carcinoma from case 2 showing extensive perineural tumor invasion (hematoxylin-eosin stain, original magnification X 100).
ity, which may overlook some of the less obvious causes. Both TMJ dysfunction and idiopathic trigeminal neuralgia are conditions routinely diagnosed and treated by oral and maxillofacial surgeons. The coexistence of TMJ pain and parotid neoplasms has been previously reported.‘T2 In the experience of Shapshay et al* with occult tumors, five of seven patients with infratemporal fossa neoplasms were
WOLF, KESSLER,
%129
AND HGROVITZ
initially given the diagnosis of TMJ dysfunction. Two of this group actually had parotid neoplasms. Extraoral neoplasms also can cause pain that resembles trigeminal neuralgia. The pain is usually more persistent and there may be other neurologic signs and symptoms, including sensory changes. Differential diagnoses in patients with facial pain and sensory changes include tumors of the infratemporal fossa, gasserian ganglion, pontine tumors, acoustic neuroma, glioma, meningioma, cholesteatoma, arterial abnormalities, and cervical spondylosis. Systemic conditions include the connective tissue disorders, eg, polyarteritis nodosa, multiple sclerosis, neuritis secondary to sarcoidosis, and syphilis. The importance of numbness as a significant finding has been previously emphasized.3,4 Atypical facial neuralgia is a common form of facial pain. In Rusthton’s survey5 of patients initially diagnosed as having atypical facial pain, more than 50% of the cases were placed under the category of psychiatric disease. Common organic causes were vasodilating facial pain, dental disease, neuritis, and neoplasms. In the last group were two cases of adenoid cystic carcinoma. Chronic pain may pre-
J Oral Maxillofac
cipitate psychological depression; most of the patients in Shapsey et al’s series’ had such severe symptoms. Bn the other hand, the chnician must avoid the temptation to consider the pain as being a manifestation of depression. The key to the management of patients with facial pain is an accurate and expeditious diagnosis. Delay in diagnosis allows the disease to progress, successful treatment may have to be more radical, and the prognosis may be significantly altered. Thorough ear, nose, throat, dental, and neurologic examination should be performed, and repeated, should %he symptoms persist. References 1. Grace EG, North AF: Temporomandibular joint dysfunction and orofacial pain caused by parotid gland malignancy: Report of a case. J Am Dent Assoc 115:348, 1988 2. Shapsey SM, Elber E, Strong SM: Occult tumors of the infratemporal fossa. Arch Otolaryngol 102535, 1976 3. Thrush DC, Small M: How benign a symptom is facial numbness? Lancet 2% 1, 1970 4. Roistacher SL: Numbness-A significant finding. Oral Surg Oral Med Oral Path01 36:22, 1973 5. Rushton JG, Gibilisen JA, Goldstein SP: Aty.pical facial pain. JAMA 171545, 19.59
Surg
49:1129-1131,199l
OLF, MD,* ALEXANDER
KESSLER,
Diagnosis of a solitary mass of the neck is a challenge for the clinician. Nearly 80% of cervical masses are neoplastic, increasing to 90% over the age of 90. Eighty-five percent of cervical masses in patients under the age of 21 are benign and include Received from the Chaim-Sheba Medical Center, TelHashomer, and Sackler School of Medicine, Tel-Aviv, Israel. * Department of Otorhinolaryngology. I Department of Gtorhinolaryngology. f Department of Pathology. Address correspondence and reprint requests to Dr Wolf: Department of Otorhinolaryngology, Chaim-Sheba Medical Center, Tel-Hashomer, 52621, Israel. 0 1991 American geons
Association of Oral and Maxillofacial
Sur-
MDJ
AND ADA WOR
mainly reactive lymph nodes and congenital cysts or malformations.’ Castelman’s disease (CD>, more commonly known as benign angiofohicular lymph node hyperplasia, is a rare condition that involves a localized lymphatic enlargement with characteristic microscopic features. It commomy invdves the mediastinal and ptdmonary lymph nodes, with neck involvement in only 15% to 20% of ca~es.~‘~ There are two primary variants: the hyaline-vascular and plasma cell types. The hyaline-vascular type comprises 80% of cases and is usually asymptomatic, unless there is impingement on adjacent structures. The plasma cell type is often associated with systemic manifestations, the most common of which are fever, anemia, elevated erythrocyte sedimentation rate, and altered plasma proteins levels4 A
