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Intracochlear vestibular schwannoma: A potential source for recurrence following translabyrinthine resection
P210
Otolaryngology Head and Neck Surgery August 1996
Scientific Posters
169
Klkuchl-Fujlmoto Disease: A Case Report and Review of the Literature PEARLINE V. MCKENZIE GARNER, MD, CPT-USA, and ELIZABETH A. BLAIR, MD, MAJ-USA, Washington, D.C.
Kikuchi-Fujimoto disease (KFD), also called histiocytic necrotizing lymphadenitis, is a rare condition that was first described in Japan 24 years ago. Clinically, it presents with cervical adenopathy that may or may not be painful. The patients are predominantly of Asian ancestry and female. This adenopathy can be associated with a prodromal upper respiratory illness to include fever and malaise. Occasionally, hepatosplenomegaly and weight loss are seen. Symptoms usually resolve 1 to 4 months after onset. However, if the disease is multicentric and widespread, death can occur. In the literature KFD has been associated with the EpsteinBarr virus, herpes virus, and systemic lupus erythematosus. Its etiology, however, remains unclear. Histologically, KFD patterns its clinical course in three stages. It begins with a proliferative histiocytic phase, evolves into frank necrosis and karyolysis, and then resolves. Unfortunately, during its proliferative phase KFD can be misdiagnosed as malignant lymphoma. A recent case of KFD in a young woman is presented and used to discuss the clinical course and histopathologic and radiologic workup needed to prevent misdiagnosis. An understanding of KFD is important for the otolaryngologist treating patients with cervical adenopathy. 170
Recurrent Oral Ulcers In a Patient Wlth Hemochromatosls and Hepatitis C Infection: A Case Report LENHANH P. TRAN, MD, ELIZABETHA. BLAIR, MD, ZIAD E. DEEB, MD, and ANA A. CARDENAS, Silver Spring, Md., and Washington, D.C.
Recurrent oral lesions in adults often present a diagnostic dilemma for the physician. Lesions in this region may be developmental, traumatic, infectious, neoplastic, or inflammatory. They may be localized to the oral cavity or are representative of a systemic disorder. Frequently a biopsy is required to confirm the clinical impression. Unfortunately, definitive diagnosis using histopathologic analysis may be difficult. We present a 47-year-old man with hepatitis C with a 3year history of recurrent, painful oral ulcers, who was subsequently found to have cutaneous lichen planus and hemochromatosis. A previous excisional biopsy of the oral lesion had revealed chronic inflammation without evidence of malignancy. The patient returned 2 years later with the same complaint of recurrent oral ulcers. Excisional biopsy of the oral lesions was repeated to rule out oral lichen planus and malignancy. The procedure was complicated with intraoperative spontaneous epistaxis. He was subsequently
found to have a coagulopathy secondary to hepatic disease. Hemochromatosis was first suggested by the history of NIDDM, arthropathy, physical findings of lower extremities, cutaneous hyperpigmentation, and hypogonadism and then later was confirmed by elevated plasma iron level, total iron binding capacity, percent transferrin saturation, and serum ferritin. Mucocutaneous lesions have been described in patients with chronic hepatic disease. The liver disease in our patient may be due to both hepatitis C and hemochromatosis. Diagnosis and management of hemochromatosis are reviewed. Systemic diseases such as hemochromatosis and hepatitis should be considered in the evaluation of intraoral lesions. 171
Laryngeal Obstruction Following Inhalation of a Coin Via a Metered Dose Inhaler STEVEN H. DAYAN, MD, LOUIS G. PORTUGAL, MD, and DAVID L. WALNER, MD, Chicago, III.
Laryngeal foreign-body impaction is a rare, life-threatening event that most commonly occurs in the pediatric population between the ages of 6 months and 3 years. Such events are most frequently a result of oral manipulation of the foreign body before aspiration. We present an unusual case in which a penny inhaled from a metered-dose inhaler became impacted in the laryngeal ventricles, causing near-complete airway obstruction. The diagnosis was established on anteroposterior and lateral neck films and confirmed by flexible nasolaryngoscopy. Progressive airway obstruction necessitated a tracheotomy before removal of the foreign body. This case highlights the potential for aspiration of foreign bodies with the use of metered-dose inhalers. The case is described in detail and presented with photographic documentation. The discussion will focus on the diagnosis and the management of laryngeal foreign bodies. 172
Intracochlear Vestlbular Schwannoma: A Potentlal Source for Recurrence Following Translabyrinthlne Resection J. DOUGLAS GREEN, Jr, MD, CHARLES W. BEATTY, MD, RONALD REIMER, MD, and JAMES E, BENECKE, MD, Jacksonville, Fla., Rochester, Minn., and St. Louis, Mo. Objective: Clinical experience with two patients with intracochlear involvement of a vestibular schwannoma is presented in hopes of increasing awareness of the potential for recurrence when tumor is identified in this area on preoperative imaging studies. Illustrative cases: A 55-year-old woman was found to have an intracanalicular vestibular schwannoma. The patient subsequently underwent translabyrinthine tumor resection. Serial MRI scans showed a small enhancing area within the fundus of the internal auditory canal and in the basal turn of
Otolaryngology Head and Neck Surgery Volume 115 Number 2
the cochlea that was initially thought to represent postoperative changes. This area continued to enlarge and was suspicious for recurrent tumor. The patient subsequently had the recurrent tumor removed through a transotic approach. Tumor was found filling the entire basal turn of the cochlea, vestibule, and internal auditory canal. A 68-year-old woman was found to have a small, intracanalicular vestibular schwannoma with intracochlear extension on MRI. Because of refractory episodic vertigo, the patient had a translabyrinthine resection of the vestibular schwannoma with tumor left in the basal turn of the cochlea. This patient has been observed for 42 months without evidence of tumor growth on serial MRI scans. Discussion: These patients on close examination of the preoperative and postoperative scans had vestibular schwannoma within the basal turn of the cochlea. The tumor within the basal turn of the cochlea was not removed at the initial translabyrinthine surgical resection because the basal turn could not be adequately accessed through this approach. Conclusion: Vestibular schwannoma recurrence may occur after translabyrinthine resection due to residual tumor within the basal turn of the cochlea. This area should be examined on the preoperative MRI scans with consideration given to the transotic approach if tumor is identified there. Patients with evidence of intracochlear vestibular schwannoma who have already had translabyrinthine tumor removal should be followed up closely with serial MRI scans.
173
Intrathecal Fluoresceln Usage In CSF Fistula FIDELIA YUAN-SHIN BUTT, MD, and HILARY BRODIE, MD, PhD, Sacramento and Stockton, Calif.
Cerebrospinal fluid (CSF) otorrhea and rhinorrhea commonly present to the otolaryngologist as a result of head trauma; iatrogenic otologic, rhinologic, and neurosurgical procedures; congenital anomalies; and erosive disease processes involving the skull base. Identification and localization of the CSF fistula are crucial in the management of these patients. Many of the techniques are available to assist in the identification of CSF leaks including CT scanning with intrathecal contrast, radionuclide scans, various intrathecal dyes, and analysis of obtained fluid samples for 13-2 transferrin. Intrathecal fluorescein dye has been demonstrated to be a very sensitive and effective means of CSF fistula identification. The fluorescein dye fluoresces brightly with a Woods lamp and can be easily identified on micropledgets placed strategically within the nasal cavity and adjacent to the torus tubarius. The dye also effectively localized fistula intraoperatively. Enthusiasm for the use of intrathecal fluorescein has been dampened by reported complications, including transient hemiparesis and grand mal seizures. These complications occurred with high doses of the fluorescein.
Scientific Posters
P211
However, with appropriate dosing of 0.5 ml 5% solution mixed with 10 ml CSF, the risk of neurologic complications is quite low. The literature is reviewed for the relative sensitivity of intrathecal fluorescein to identify CSF fistula compared.with the other available techniques, as well as the relative safety.
174
Oro-Rhino-Orbltal-Cerebral Mucormycosis Following Dental Extraction JAMES K. FORTSON, MD, TIENCHIN HO, MD, and JONE KIM, DDS, MS, Fresno, Calif.
The purpose of this paper is to present a case report of mucormycosis in 57-year-old truck driver with uncontrolled diabetes. The patient had refused insulin therapy because he would lose his occupation. He opted to take oral hyperglycemic agents. Two days before admission he was seen by his dentist for increasing teeth pain. He underwent extraction of his second and third left maxillary molar teeth. He began receiving Keflex for facial and cheek swelling. Over the next 24 hours the patient noticed increased swelling with erythema and numbness of his left cheek and eye. He noted progressive visual loss in his left eye. He presented to Valley Medical Center with blindness in his left eye and swelling with ecchymosis of left forehead, periorbital area, and cheek. His blood glucose level was 407, and he admitted to poor compliance with his oral hypoglycemic agents. Physical examination at the time of admission revealed left-sided orbital, cheek, and facial cellulitis. He had paralysis of his third, fourth, fifth, sixth, and seventh nerves. There was no vision in his left eye. Ophthalmoplegia, chemosis, and proptosis were present. The nasal examination revealed a deviated septum with scant secretions from the middle meatus. The oral cavity had a dark area with clot at the extraction site. The rest of the physical examination was essentially normal. He was admitted and began receiving intravenous antibiotics. Laboratory values revealed an elevated white blood cell count, anemia, low pH, and elevated glucose and creatinine. Consultations were obtained by internal medicine, neurology, infectious disease, otolaryngology, and ophthalmology. A CT scan revealed a mild sinusitis but no obvious masses. The patient underwent endoscopic sinusotomies with biopsies and cultures. He began receiving high-dose broadspectrum antibiotics and amphotericin B. His clinical course rapidly deteriorated. He developed black spots on his face and hard and soft palates. Biopsies were positive for mucormycosis. The patient's Glasgow coma scale began to deteriorate, and he died 72 hours after admission. Patients with diabetes have considerable risk during surgical procedures. If the diabetes is uncontrolled, catastrophic consequences may result. A thorough history should precede any surgical procedure by physician or dentist. Diabetes as well as other medical problems should be corrected before surgery.
Otolaryngology Head and Neck Surgery August 1996
Scientific Posters
169
Klkuchl-Fujlmoto Disease: A Case Report and Review of the Literature PEARLINE V. MCKENZIE GARNER, MD, CPT-USA, and ELIZABETH A. BLAIR, MD, MAJ-USA, Washington, D.C.
Kikuchi-Fujimoto disease (KFD), also called histiocytic necrotizing lymphadenitis, is a rare condition that was first described in Japan 24 years ago. Clinically, it presents with cervical adenopathy that may or may not be painful. The patients are predominantly of Asian ancestry and female. This adenopathy can be associated with a prodromal upper respiratory illness to include fever and malaise. Occasionally, hepatosplenomegaly and weight loss are seen. Symptoms usually resolve 1 to 4 months after onset. However, if the disease is multicentric and widespread, death can occur. In the literature KFD has been associated with the EpsteinBarr virus, herpes virus, and systemic lupus erythematosus. Its etiology, however, remains unclear. Histologically, KFD patterns its clinical course in three stages. It begins with a proliferative histiocytic phase, evolves into frank necrosis and karyolysis, and then resolves. Unfortunately, during its proliferative phase KFD can be misdiagnosed as malignant lymphoma. A recent case of KFD in a young woman is presented and used to discuss the clinical course and histopathologic and radiologic workup needed to prevent misdiagnosis. An understanding of KFD is important for the otolaryngologist treating patients with cervical adenopathy. 170
Recurrent Oral Ulcers In a Patient Wlth Hemochromatosls and Hepatitis C Infection: A Case Report LENHANH P. TRAN, MD, ELIZABETHA. BLAIR, MD, ZIAD E. DEEB, MD, and ANA A. CARDENAS, Silver Spring, Md., and Washington, D.C.
Recurrent oral lesions in adults often present a diagnostic dilemma for the physician. Lesions in this region may be developmental, traumatic, infectious, neoplastic, or inflammatory. They may be localized to the oral cavity or are representative of a systemic disorder. Frequently a biopsy is required to confirm the clinical impression. Unfortunately, definitive diagnosis using histopathologic analysis may be difficult. We present a 47-year-old man with hepatitis C with a 3year history of recurrent, painful oral ulcers, who was subsequently found to have cutaneous lichen planus and hemochromatosis. A previous excisional biopsy of the oral lesion had revealed chronic inflammation without evidence of malignancy. The patient returned 2 years later with the same complaint of recurrent oral ulcers. Excisional biopsy of the oral lesions was repeated to rule out oral lichen planus and malignancy. The procedure was complicated with intraoperative spontaneous epistaxis. He was subsequently
found to have a coagulopathy secondary to hepatic disease. Hemochromatosis was first suggested by the history of NIDDM, arthropathy, physical findings of lower extremities, cutaneous hyperpigmentation, and hypogonadism and then later was confirmed by elevated plasma iron level, total iron binding capacity, percent transferrin saturation, and serum ferritin. Mucocutaneous lesions have been described in patients with chronic hepatic disease. The liver disease in our patient may be due to both hepatitis C and hemochromatosis. Diagnosis and management of hemochromatosis are reviewed. Systemic diseases such as hemochromatosis and hepatitis should be considered in the evaluation of intraoral lesions. 171
Laryngeal Obstruction Following Inhalation of a Coin Via a Metered Dose Inhaler STEVEN H. DAYAN, MD, LOUIS G. PORTUGAL, MD, and DAVID L. WALNER, MD, Chicago, III.
Laryngeal foreign-body impaction is a rare, life-threatening event that most commonly occurs in the pediatric population between the ages of 6 months and 3 years. Such events are most frequently a result of oral manipulation of the foreign body before aspiration. We present an unusual case in which a penny inhaled from a metered-dose inhaler became impacted in the laryngeal ventricles, causing near-complete airway obstruction. The diagnosis was established on anteroposterior and lateral neck films and confirmed by flexible nasolaryngoscopy. Progressive airway obstruction necessitated a tracheotomy before removal of the foreign body. This case highlights the potential for aspiration of foreign bodies with the use of metered-dose inhalers. The case is described in detail and presented with photographic documentation. The discussion will focus on the diagnosis and the management of laryngeal foreign bodies. 172
Intracochlear Vestlbular Schwannoma: A Potentlal Source for Recurrence Following Translabyrinthlne Resection J. DOUGLAS GREEN, Jr, MD, CHARLES W. BEATTY, MD, RONALD REIMER, MD, and JAMES E, BENECKE, MD, Jacksonville, Fla., Rochester, Minn., and St. Louis, Mo. Objective: Clinical experience with two patients with intracochlear involvement of a vestibular schwannoma is presented in hopes of increasing awareness of the potential for recurrence when tumor is identified in this area on preoperative imaging studies. Illustrative cases: A 55-year-old woman was found to have an intracanalicular vestibular schwannoma. The patient subsequently underwent translabyrinthine tumor resection. Serial MRI scans showed a small enhancing area within the fundus of the internal auditory canal and in the basal turn of
Otolaryngology Head and Neck Surgery Volume 115 Number 2
the cochlea that was initially thought to represent postoperative changes. This area continued to enlarge and was suspicious for recurrent tumor. The patient subsequently had the recurrent tumor removed through a transotic approach. Tumor was found filling the entire basal turn of the cochlea, vestibule, and internal auditory canal. A 68-year-old woman was found to have a small, intracanalicular vestibular schwannoma with intracochlear extension on MRI. Because of refractory episodic vertigo, the patient had a translabyrinthine resection of the vestibular schwannoma with tumor left in the basal turn of the cochlea. This patient has been observed for 42 months without evidence of tumor growth on serial MRI scans. Discussion: These patients on close examination of the preoperative and postoperative scans had vestibular schwannoma within the basal turn of the cochlea. The tumor within the basal turn of the cochlea was not removed at the initial translabyrinthine surgical resection because the basal turn could not be adequately accessed through this approach. Conclusion: Vestibular schwannoma recurrence may occur after translabyrinthine resection due to residual tumor within the basal turn of the cochlea. This area should be examined on the preoperative MRI scans with consideration given to the transotic approach if tumor is identified there. Patients with evidence of intracochlear vestibular schwannoma who have already had translabyrinthine tumor removal should be followed up closely with serial MRI scans.
173
Intrathecal Fluoresceln Usage In CSF Fistula FIDELIA YUAN-SHIN BUTT, MD, and HILARY BRODIE, MD, PhD, Sacramento and Stockton, Calif.
Cerebrospinal fluid (CSF) otorrhea and rhinorrhea commonly present to the otolaryngologist as a result of head trauma; iatrogenic otologic, rhinologic, and neurosurgical procedures; congenital anomalies; and erosive disease processes involving the skull base. Identification and localization of the CSF fistula are crucial in the management of these patients. Many of the techniques are available to assist in the identification of CSF leaks including CT scanning with intrathecal contrast, radionuclide scans, various intrathecal dyes, and analysis of obtained fluid samples for 13-2 transferrin. Intrathecal fluorescein dye has been demonstrated to be a very sensitive and effective means of CSF fistula identification. The fluorescein dye fluoresces brightly with a Woods lamp and can be easily identified on micropledgets placed strategically within the nasal cavity and adjacent to the torus tubarius. The dye also effectively localized fistula intraoperatively. Enthusiasm for the use of intrathecal fluorescein has been dampened by reported complications, including transient hemiparesis and grand mal seizures. These complications occurred with high doses of the fluorescein.
Scientific Posters
P211
However, with appropriate dosing of 0.5 ml 5% solution mixed with 10 ml CSF, the risk of neurologic complications is quite low. The literature is reviewed for the relative sensitivity of intrathecal fluorescein to identify CSF fistula compared.with the other available techniques, as well as the relative safety.
174
Oro-Rhino-Orbltal-Cerebral Mucormycosis Following Dental Extraction JAMES K. FORTSON, MD, TIENCHIN HO, MD, and JONE KIM, DDS, MS, Fresno, Calif.
The purpose of this paper is to present a case report of mucormycosis in 57-year-old truck driver with uncontrolled diabetes. The patient had refused insulin therapy because he would lose his occupation. He opted to take oral hyperglycemic agents. Two days before admission he was seen by his dentist for increasing teeth pain. He underwent extraction of his second and third left maxillary molar teeth. He began receiving Keflex for facial and cheek swelling. Over the next 24 hours the patient noticed increased swelling with erythema and numbness of his left cheek and eye. He noted progressive visual loss in his left eye. He presented to Valley Medical Center with blindness in his left eye and swelling with ecchymosis of left forehead, periorbital area, and cheek. His blood glucose level was 407, and he admitted to poor compliance with his oral hypoglycemic agents. Physical examination at the time of admission revealed left-sided orbital, cheek, and facial cellulitis. He had paralysis of his third, fourth, fifth, sixth, and seventh nerves. There was no vision in his left eye. Ophthalmoplegia, chemosis, and proptosis were present. The nasal examination revealed a deviated septum with scant secretions from the middle meatus. The oral cavity had a dark area with clot at the extraction site. The rest of the physical examination was essentially normal. He was admitted and began receiving intravenous antibiotics. Laboratory values revealed an elevated white blood cell count, anemia, low pH, and elevated glucose and creatinine. Consultations were obtained by internal medicine, neurology, infectious disease, otolaryngology, and ophthalmology. A CT scan revealed a mild sinusitis but no obvious masses. The patient underwent endoscopic sinusotomies with biopsies and cultures. He began receiving high-dose broadspectrum antibiotics and amphotericin B. His clinical course rapidly deteriorated. He developed black spots on his face and hard and soft palates. Biopsies were positive for mucormycosis. The patient's Glasgow coma scale began to deteriorate, and he died 72 hours after admission. Patients with diabetes have considerable risk during surgical procedures. If the diabetes is uncontrolled, catastrophic consequences may result. A thorough history should precede any surgical procedure by physician or dentist. Diabetes as well as other medical problems should be corrected before surgery.