749
RUBIN AND SANFILIPPO
terior joint space into the anterior joint space difficult, as well as limit the surgeon’s ability to perform the eminentia release and capsular stretch procedures’ required to reverse the effects of capsular fibrosis. References 1. Holmlund A, Hellsing G: Arthroscopy of the temporomandibular joint. Int J Oral Surg 14169, 1985
J Oral Maxillofac
2. Saunders B, Buoncristini R: Diagnostic and surgical arthroscopy of the temooromandibular ioint. Clinical exnerienee with 137 procedures over a 2-year pehod. J Craniomand Disorders Fat Oral Pain 1:202-213, 1986 3. Murakami K, Ono T: Temporomandibular arthroscopy by inferolateral approach. Int J Oral Maxillofac Surg 15:410, 1986 4. McCain J. De la Rua H: Princiules and practice of operative arthro&opy of the human temporomandibular joint. Oral Maxillofac Surg Clin North Am 1:135, 1989 5. Moses J. Topper D: A functional approach to the treatment of temporomandibular joint internal derangement. J Craniomand Disorders Fat Oral Pain 5: 19, 199 1
Surg
50:749-751.1992
Lymphoma of the Paranasal Sinuses Presenting as Cavernous Sinus Syndrome MITCHELL
M. RUBIN, DMD,* AND ROSS J. SANFILIPPO,
Cavernous sinus syndrome (CSS) is characterized by unilateral opthalmoplegia involving the oculomotor, trochlear, and abducent nerves; loss of sensation in the areas supplied by the ophthalmic and maxillary branches of the trigeminal nerve; unilateral exopthalmos; and signs of oculosympathetic nerve paresis.’ Thomas and Yoss2 reviewed 102 cases of cavernous sinus syndrome and found 69% were caused by neoplasms, 19% by vascular disorders, and 13% by inflammation. We report the second case of CSS due to infiltration of the structures of the lateral wall of the cavernous sinus by non-Hodgkin’s lymphoma in the adjacent paranasal sinuses3 and the first reported case in a patient with acquired immunodeficiency syndrome (AIDS).
DMDt
carinii pneumonia, and insulin-dependent diabetes mellitus secondary to pentamidine treatment. She reported an allergy to trimethoprim. Her current medications included ethambutol, rifampin, azathioprine, dapsone, clotrimazole, and pyrazinamide. Ophthalmology consultation was requested, and orbital and sinus computerized tomograms (CT scans) were obtained. On clinical examination, the patient was in moderate distress. Her lacrimal and parotid glands were enlarged bilaterally. The left eye was proptotic, with marked periorbital edema and ptosis. Extraocular movements were limited in all directions, with lateral gaze most notably affected (Fig 1).
Case Report On July 9, 1991 a 3%year-old black woman presented to the Nassau County Medical Center with a complaint of left
eye pain and decreased vision that she had first noticed 3 days before. Her medical history was positive for human immunodeficiency virus (HIV), tuberculosis, pneumocystis
Received from the Department of Oral and Maxillofacial Surgery. Nassau County Medical Center, East Meadow, NY. * Attending Surgeon; in private practice, Rockville Centre, NY. t Chief Resident. Address correspondence and reprint requests to Dr Rubin: South Shore Oral Surgery Associates, P.C., Maple Medical Center, 24 Maple Ave, Rockville Centre, NY 11570. 0 1992 American
Association
0278-2391/92/5007-0016$3.00/O
of Oral and Maxillofacial
Surgeons
FIGURE I. Photograph showing left eye ptosis, proptosis, periorbital edema, and inability to move left eye laterally.
LYMPHOMA OF SINUSES
FIGURE 2. Axial CT scan showing opacification of the maxillary sinuses, greater on the right than the left side.
sulted for immediate sinus decompression. A CT scan of the cavernous sinus showed no evidence of cavernous sinus involvement. The patient underwent bilateral maxillary sinus debridement and nasal antrostomy. The sinus contents were submitted for routine cultures and histologic evaluation. The antibiotic regimen was continued, with the addition of ceftriaxone. The cultures demonstrated growth of a-hemolytic streptococcus species. The histologic specimen showed diffuse large-cell type, B-cell malignant lymphoma of the maxillary sinus (Fig 4). At this time, a provisional diagnosis of cavernous sinus syndrome was made, based on infiltration of the structures of the lateral wall of the cavernous sinus by lymphoma in the adjacent paranasal sinuses. Neurologic consultation supported this diagnosis on clinical findings alone. The hematology-oncology service suggested radiotherapy. She subsequently received 4,000 cGray to the retro-orbital and paranasal sinus region. The proptosis and ptosis resolved within 1 month of treatment. Extraocular movements returned. Her blindness, however, remained unchanged. She is currently being observed as an outpatient by the medical service; her prognosis remains poor.
Discussion Vision in the left eye was poor on initial examination. The right eye had normal vision and extraocular movements were intact. The patient was afebrile. Vital signs were stable and values of her laboratory tests were unremarkable. She was admitted
to the medical service and placed on intravenous nafcillin and gentamycin. Over the next 24 hours she developed complete ophthalmoplegia and blindness of the left eye. The CT scan showed opacification of the right and left maxillary sinuses (Fig 2), as well as the ethmoid and sphenoid sinuses (Fig 3). The oral and maxillofacial surgery service was con-
The manifestations of non-Hodgkin’s lymphoma are highly variable, the most common being painless enlargement of the lymph nodes. It may, however, occasionally develop in extranodal tissues. Lymphoma of the paranasal sinuses, although uncommon, has been well recognized. Non-Hodgkin’s lymphoma accounts for the majority of cases. The common signs and symptoms of lymphoma of the paranasal sinuses are local facial swelling, pain, nasal obstruction, nasal discharge, epistaxis, and proptosis.3
FIGURE 3. Coronal CT scan showing opacification of the ethmoid sinuses.
751
RUBIN AND SANFILIPPO
FIGURE 4. Photomicrograph showing diffuse large-cell type, Bcell malignant lymphoma (hematoxylin-eosin, original magnification X400).
The case definition of AIDS has recently been revised to include patients with non-Hodgkin’s lymphoma of a high-grade pathologic type diagnosed by biopsy, as long as the result of the serologic or virologic tests are positive for HIV.4 These lymphomas generally occur in extranodal sites in the advanced stages, and have a much worse prognosis than those seen in the general population. They are clinically very aggressive and often display an incomplete response to chemotherapy or radiotherapy.5 The oral and maxillofacial surgeon should be alert to the trend for the development of aggressive non-Hodgkin’s lymphoma with unusual sites of extranodal involvement in patients with AIDS.6 Neuro-opthalmologic abnormalities are rare in patients with lymphoma and, if they occur, usually result from direct invasion of the orbital and ocular structures. Lesions in the cavernous sinus producing oculovisual manifestations are most unusual, with 10 cases reported in the literature. Delpassand and Kirkpatrick’ reviewed the 10 reported cases of CSS caused by malignant lymphoma and found that sharp retro-orbital pain, paresthesia around the orbit, and complete sixth nerve palsy were the dominant presenting symptoms. There
were no characteristic presenting symptoms to differentiate lymphoma from other causes of CSS. Symptoms reflected location of the lesion rather than its cause. The mean survival time after the first symptom was 8.7 months. During the early stages of the disease, the CT scan was abnormal in only 20% of the cases.’ Julien et al* also reported that the CT scan, which has been the crucial investigation detecting lymphomas in the cavernous sinus, is often normal in the early stages of the disease. This was found in most of the cases reported by Kuri and Mitsumito as we11.9 The involvement of the cavernous sinus can be the first sign of a lymphoma and the neurological signs can appear suddenly or progressively. Abnormalities that can be detected by CT scan often appear later than the clinical signs, and so the correct diagnosis can be missed.’ In this context, the high incidence of mass lesions in the production of CSS would seem well worth remembering,* especially to the oral and maxillofacial surgeon who is more familiar with infectious causes of the syndrome. References I. Bosley TM. Schatz NJ: Clinical diagnosis of cavernous sinus syndromes. Nemo1 Clin 1:929, 1983 2. Thomas JE. Yoss RE: The parasellar syndrome: Problems in determinine etioloav. Mavo Clin Proc 45:6 17, 1970 3. Koh CS, Tan”CT. AThady SF: Cavernous sinus syndrome, a manifestation of non-Hodgkin’s lymphoma of the ethmoid sinus. Med J Aust 2:45 1, 1983 4. Centers for Disease Control: Revision of case definition of acquired immunodeficiency syndrome. MMWR 34:373, 1985 5. Hommel DJ, Brown ML, Kinzie JJ: Response to radiotherapy of head and neck tumors in AIDS patients. Am J Surg 154: 443, 1987 6. Rubin MM, Gatta CG, Cozzi GM: Non-Hodgkin’s lymphoma of the buccal ainaiva as the initial manifestation of AIDS. J Oral Maxillof& Surg 47: 131 I, 1989 7. Delpassano ES, Kirkpatrick JB: Cavernous sinus syndrome as the presentation of malignant lymphoma: Case report and review of the literature. Neurosurgery 23:50 I. I988 8. Julien J, Ferrer X, Drouillard J. et al: Cavernous sinussyndrome due to lymphoma. J Neural Neurosurg Psychiatry 47:558. 1984 9. Kori SH. Mitsumoto H: Cavernous sinus involvement in lymphoma patients. Neurology 32:2. 1982
RUBIN AND SANFILIPPO
terior joint space into the anterior joint space difficult, as well as limit the surgeon’s ability to perform the eminentia release and capsular stretch procedures’ required to reverse the effects of capsular fibrosis. References 1. Holmlund A, Hellsing G: Arthroscopy of the temporomandibular joint. Int J Oral Surg 14169, 1985
J Oral Maxillofac
2. Saunders B, Buoncristini R: Diagnostic and surgical arthroscopy of the temooromandibular ioint. Clinical exnerienee with 137 procedures over a 2-year pehod. J Craniomand Disorders Fat Oral Pain 1:202-213, 1986 3. Murakami K, Ono T: Temporomandibular arthroscopy by inferolateral approach. Int J Oral Maxillofac Surg 15:410, 1986 4. McCain J. De la Rua H: Princiules and practice of operative arthro&opy of the human temporomandibular joint. Oral Maxillofac Surg Clin North Am 1:135, 1989 5. Moses J. Topper D: A functional approach to the treatment of temporomandibular joint internal derangement. J Craniomand Disorders Fat Oral Pain 5: 19, 199 1
Surg
50:749-751.1992
Lymphoma of the Paranasal Sinuses Presenting as Cavernous Sinus Syndrome MITCHELL
M. RUBIN, DMD,* AND ROSS J. SANFILIPPO,
Cavernous sinus syndrome (CSS) is characterized by unilateral opthalmoplegia involving the oculomotor, trochlear, and abducent nerves; loss of sensation in the areas supplied by the ophthalmic and maxillary branches of the trigeminal nerve; unilateral exopthalmos; and signs of oculosympathetic nerve paresis.’ Thomas and Yoss2 reviewed 102 cases of cavernous sinus syndrome and found 69% were caused by neoplasms, 19% by vascular disorders, and 13% by inflammation. We report the second case of CSS due to infiltration of the structures of the lateral wall of the cavernous sinus by non-Hodgkin’s lymphoma in the adjacent paranasal sinuses3 and the first reported case in a patient with acquired immunodeficiency syndrome (AIDS).
DMDt
carinii pneumonia, and insulin-dependent diabetes mellitus secondary to pentamidine treatment. She reported an allergy to trimethoprim. Her current medications included ethambutol, rifampin, azathioprine, dapsone, clotrimazole, and pyrazinamide. Ophthalmology consultation was requested, and orbital and sinus computerized tomograms (CT scans) were obtained. On clinical examination, the patient was in moderate distress. Her lacrimal and parotid glands were enlarged bilaterally. The left eye was proptotic, with marked periorbital edema and ptosis. Extraocular movements were limited in all directions, with lateral gaze most notably affected (Fig 1).
Case Report On July 9, 1991 a 3%year-old black woman presented to the Nassau County Medical Center with a complaint of left
eye pain and decreased vision that she had first noticed 3 days before. Her medical history was positive for human immunodeficiency virus (HIV), tuberculosis, pneumocystis
Received from the Department of Oral and Maxillofacial Surgery. Nassau County Medical Center, East Meadow, NY. * Attending Surgeon; in private practice, Rockville Centre, NY. t Chief Resident. Address correspondence and reprint requests to Dr Rubin: South Shore Oral Surgery Associates, P.C., Maple Medical Center, 24 Maple Ave, Rockville Centre, NY 11570. 0 1992 American
Association
0278-2391/92/5007-0016$3.00/O
of Oral and Maxillofacial
Surgeons
FIGURE I. Photograph showing left eye ptosis, proptosis, periorbital edema, and inability to move left eye laterally.
LYMPHOMA OF SINUSES
FIGURE 2. Axial CT scan showing opacification of the maxillary sinuses, greater on the right than the left side.
sulted for immediate sinus decompression. A CT scan of the cavernous sinus showed no evidence of cavernous sinus involvement. The patient underwent bilateral maxillary sinus debridement and nasal antrostomy. The sinus contents were submitted for routine cultures and histologic evaluation. The antibiotic regimen was continued, with the addition of ceftriaxone. The cultures demonstrated growth of a-hemolytic streptococcus species. The histologic specimen showed diffuse large-cell type, B-cell malignant lymphoma of the maxillary sinus (Fig 4). At this time, a provisional diagnosis of cavernous sinus syndrome was made, based on infiltration of the structures of the lateral wall of the cavernous sinus by lymphoma in the adjacent paranasal sinuses. Neurologic consultation supported this diagnosis on clinical findings alone. The hematology-oncology service suggested radiotherapy. She subsequently received 4,000 cGray to the retro-orbital and paranasal sinus region. The proptosis and ptosis resolved within 1 month of treatment. Extraocular movements returned. Her blindness, however, remained unchanged. She is currently being observed as an outpatient by the medical service; her prognosis remains poor.
Discussion Vision in the left eye was poor on initial examination. The right eye had normal vision and extraocular movements were intact. The patient was afebrile. Vital signs were stable and values of her laboratory tests were unremarkable. She was admitted
to the medical service and placed on intravenous nafcillin and gentamycin. Over the next 24 hours she developed complete ophthalmoplegia and blindness of the left eye. The CT scan showed opacification of the right and left maxillary sinuses (Fig 2), as well as the ethmoid and sphenoid sinuses (Fig 3). The oral and maxillofacial surgery service was con-
The manifestations of non-Hodgkin’s lymphoma are highly variable, the most common being painless enlargement of the lymph nodes. It may, however, occasionally develop in extranodal tissues. Lymphoma of the paranasal sinuses, although uncommon, has been well recognized. Non-Hodgkin’s lymphoma accounts for the majority of cases. The common signs and symptoms of lymphoma of the paranasal sinuses are local facial swelling, pain, nasal obstruction, nasal discharge, epistaxis, and proptosis.3
FIGURE 3. Coronal CT scan showing opacification of the ethmoid sinuses.
751
RUBIN AND SANFILIPPO
FIGURE 4. Photomicrograph showing diffuse large-cell type, Bcell malignant lymphoma (hematoxylin-eosin, original magnification X400).
The case definition of AIDS has recently been revised to include patients with non-Hodgkin’s lymphoma of a high-grade pathologic type diagnosed by biopsy, as long as the result of the serologic or virologic tests are positive for HIV.4 These lymphomas generally occur in extranodal sites in the advanced stages, and have a much worse prognosis than those seen in the general population. They are clinically very aggressive and often display an incomplete response to chemotherapy or radiotherapy.5 The oral and maxillofacial surgeon should be alert to the trend for the development of aggressive non-Hodgkin’s lymphoma with unusual sites of extranodal involvement in patients with AIDS.6 Neuro-opthalmologic abnormalities are rare in patients with lymphoma and, if they occur, usually result from direct invasion of the orbital and ocular structures. Lesions in the cavernous sinus producing oculovisual manifestations are most unusual, with 10 cases reported in the literature. Delpassand and Kirkpatrick’ reviewed the 10 reported cases of CSS caused by malignant lymphoma and found that sharp retro-orbital pain, paresthesia around the orbit, and complete sixth nerve palsy were the dominant presenting symptoms. There
were no characteristic presenting symptoms to differentiate lymphoma from other causes of CSS. Symptoms reflected location of the lesion rather than its cause. The mean survival time after the first symptom was 8.7 months. During the early stages of the disease, the CT scan was abnormal in only 20% of the cases.’ Julien et al* also reported that the CT scan, which has been the crucial investigation detecting lymphomas in the cavernous sinus, is often normal in the early stages of the disease. This was found in most of the cases reported by Kuri and Mitsumito as we11.9 The involvement of the cavernous sinus can be the first sign of a lymphoma and the neurological signs can appear suddenly or progressively. Abnormalities that can be detected by CT scan often appear later than the clinical signs, and so the correct diagnosis can be missed.’ In this context, the high incidence of mass lesions in the production of CSS would seem well worth remembering,* especially to the oral and maxillofacial surgeon who is more familiar with infectious causes of the syndrome. References I. Bosley TM. Schatz NJ: Clinical diagnosis of cavernous sinus syndromes. Nemo1 Clin 1:929, 1983 2. Thomas JE. Yoss RE: The parasellar syndrome: Problems in determinine etioloav. Mavo Clin Proc 45:6 17, 1970 3. Koh CS, Tan”CT. AThady SF: Cavernous sinus syndrome, a manifestation of non-Hodgkin’s lymphoma of the ethmoid sinus. Med J Aust 2:45 1, 1983 4. Centers for Disease Control: Revision of case definition of acquired immunodeficiency syndrome. MMWR 34:373, 1985 5. Hommel DJ, Brown ML, Kinzie JJ: Response to radiotherapy of head and neck tumors in AIDS patients. Am J Surg 154: 443, 1987 6. Rubin MM, Gatta CG, Cozzi GM: Non-Hodgkin’s lymphoma of the buccal ainaiva as the initial manifestation of AIDS. J Oral Maxillof& Surg 47: 131 I, 1989 7. Delpassano ES, Kirkpatrick JB: Cavernous sinus syndrome as the presentation of malignant lymphoma: Case report and review of the literature. Neurosurgery 23:50 I. I988 8. Julien J, Ferrer X, Drouillard J. et al: Cavernous sinussyndrome due to lymphoma. J Neural Neurosurg Psychiatry 47:558. 1984 9. Kori SH. Mitsumoto H: Cavernous sinus involvement in lymphoma patients. Neurology 32:2. 1982
