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Pediatric subglottic granular cell myoblastoma
Otolaryngology– Head and Neck Surgery Volume 129 Number 2
P189 Intracranial Complications of Sinusitis: A Five-Year Review of Three Cases Fabio De Rezende Pinna MD (presenter); Fabrizio Ricci Romano MD; Richard L Voegels MD Sao Paulo Brazil; Sao Paulo Brazil; Sao Paulo Brazil
Objectives: Despite improvements in antibiotic therapies and surgical techniques, sinusitis still carries a risk of serious and potentially fatal complications. In this study, we describe our findings in the patients who had intracranial complications. Methods: We examined the charts of 38 patients who had been admitted to the “Hospital das Clı´nicas da Faculdade de Medicina da USP,” Sa˜ o Paulo, Brazil, between January 1, 1997, and March 31, 2002, for treatment of sinusitis complications. Of these 38 patients, 35 had orbital complications and 3 had intracranial complications. Results: We found one case of epidural abscess, one case of cavernous sinus thrombosis, and one case of frontal osteomyelitis and cerebral abscess. The last two patients did not take antibiotics properly before admitted to our service. All of them had no central signs or symptoms such as seizures, hemiparesia, weakness, and mental confusion. The diagnosis of intracranial complications was only possible by the use of computed tomography. Those patients were treated with intravenous antibiotics and drainage of the affected sinuses by endoscopic surgery, which has been helping to improve outcomes. Neurosurgery was not necessary in any of these, and neither the use of anticoagulant therapy in the case of cavernous sinus thrombosis. Conclusion: Intracranial infections secondary to rhinosinusitis occur sporadically, and although it appears that this cannot be prevented, recognition and management of these patients should be undertaken immediately and are best achieved via a multidisciplinary approach.
P273
tragnathic examples include the temporal bone, concha bullosa, and sphenoid. Previous case reports have raised the issue of this lesion as a pregnancy-dependent tumor. Methods: We present the case of a 16-year-old pregnant female diagnosed with a rapidly progressive, expansile growth of the left maxilla during her first trimester. Results: Fine needle aspiration biopsy was nondiagnostic, and incisional biopsy via the Caldwell-Luc approach provided the diagnosis. Definitive management consisted of wide local excision. Conclusion: Differential diagnosis, pathology, and treatment options are discussed. P191 Presentation and Management of Pseudoaneurysm of the Superficial Temporal Artery Jason G Cundiff MD (presenter); Benjamin A Bassichis MD; Tim Malisch MD Chicago IL; Chicago IL; Chicago IL
Maxillary Giant Cell Reparative Granuloma Presenting during Pregnancy
Objectives: In 1740 Thomas Bartholin reported the first case of a temporal artery pseudoaneurysm secondary to blunt trauma. Since that time, nearly 200 cases of face and temple pseudoaneurysm secondary to trauma have been described; however, this phenomenon has been rarely described in the otolaryngology literature. In this report, we describe an atypical case secondary to penetrating trauma and a non-surgical treatment option by interventional radiology and endovascular embolization. Methods: Case report and literature review of superficial temporal artery pseudoaneurysm. Results: Superficial temporal artery pseudoaneurysms typically result from blunt trauma to the temple. These pseudoaneurysms present as a gradually enlarging pulsatile mass with a bruit on auscultation. Pseudoaneurysms result from total or partial rupture of the arterial wall with formation of a hematoma and a new wall of perivascular tissue. Definitive diagnosis can be made with ultrasound, CT scan with contrast, or angiogram (gold standard). In the past these were typically treated surgically with ligation of the affected artery and resection of the pseudoaneurysm. This case was successfully managed with endovascular embolization of pseudoaneurysm with regression of the facial mass and no complications. Conclusion: Superficial temporal artery pseudoaneurysms can be successfully treated by endovascular embolization avoiding the risks involved with surgery and the scar from incision.
Patricia L Bell MD (presenter); Robert P Zitsch III MD Columbia MO; Columbia MO
P192
P190
Objectives: Giant cell reparative granuloma can manifest in the head and neck as a locally aggressive, destructive process. It can present in several locations of the head and neck but is most commonly diagnosed in the mandible. Ex-
Pediatric Subglottic Granular Cell Myoblastoma Steven E Sobol MD (presenter); Daniel Sharya Samadi MD; Ralph F Wetmore MD Philadelphia PA; Philadelphia PA; Philadelphia PA
POSTERS
Conclusion: The use of antibiotics for otitis media has significantly reduced the incidence of petrous apicitis. In this report, we describe an atypical case of bilateral petrous apicitis resulting from an anaerobic infection (Fusobacterium necrophorum). The treatment of the second side needs to be performed urgently to prevent bilateral carotid or jugular thrombosis, or cranial nerve palsies.
Scientific Posters
Scientific Posters
Objectives: Granular cell myoblastoma is a rare, benign tumor of unknown origin that most commonly presents in the oral cavity or larynx of adult patients. There have only been several reports in the literature of this tumor presenting in the pediatric population. The objective of this study is to report a case of a granular cell myoblastoma presenting in the subglottic airway of a child. Methods: Retrospective chart review. Results: A 7-year-old male presented with a history of a barking cough, shortness of breath on exertion, and progressively worsening biphasic stridor over a 3-month period. Physical examination was significant for biphasic stridor and was otherwise within normal limits. Computed tomography and magnetic resonance imaging demonstrated a 2-cm, round, solid, soft-tissue mass between the upper trachea and esophagus, which appeared to be extrinsic to the airway lumen. The patient underwent a direct laryngoscopy, bronchoscopy, and esophagoscopy, which revealed a rubbery submucosal mass in the posterior subglottic space. A laryngofissure was performed, and the mass was resected by elevating posterior subglottic mucosal flaps. Histopathological evaluation was consistent with granular cell myoblastoma. Conclusion: Although rare, granular cell myoblastoma should be considered in the differential diagnosis of subglottic lesions in children.
P193 Case Report on Sentinel Lymph Node Use in Merkel Cell Carcinoma Richard J Barnett MD (presenter); D Michael Rose MD; Timothy F Ragsdale MD; Mark E Augspurger MD New Orleans LA; Keesler AFB MS; Ocean Springs MS; Keesler AFB MS
Objectives: Merkel cell carcinoma (MCC) is a highly aggressive primary neuroendocrine tumor. The literature reveals that delayed diagnoses and poor survival are common with MCC. The role of sentinel lymph node has been shown to be effective in the evaluation of patients with melanoma and breast cancer. We present a case report on the use of sentinel lymph node biopsy in a patient with Merkel cell carcinoma of the tragus. Methods: Preoperative lymphoscintigraphy was performed on the morning of surgery by injecting radiolabeled colloid intradermally. Static images demonstrated uptake in two nodes in the same region. In the operating room, lymphazurine blue dye was injected intradermally around the primary Merkel cell carcinoma. Two sentinel lymph nodes were found to have blue dye as well as increased radioactive counts on intraoperative mapping. The nodes were removed and sent for permanent section.
Results: Histologic examination of the primary site revealed clear margins, and both nodes were negative for metastases. The patient received radiation to the primary site, ipsilateral parotid nodes, and superior cervical chain. The patient remained free of disease at a 7-month follow-up appointment. Conclusion: Merkel cell carcinoma of the skin has a poor prognosis and is difficult to study due to its rarity. Sentinel lymph node biopsy may prove to be an invaluable tool to decrease morbidity associated with elective nodal dissections, provide actual anatomic drainage patterns, and supply staging and prognostic information to the patient and physician. Further therapeutic options incorporating systemic therapy are needed to decrease the mortality rate.
P194 Langerhans Cell Histiocytosis Presenting as Bilateral Temporal Bone Lesions Chester Park Barton III MD (presenter); Drew M Horlbeck MD San Antonio TX; Boerne TX
Objectives: Langerhans cell histiocytosis, commonly referred to as histiocytosis X, is an idiopathic condition characterized by a proliferation of histiocytes. It is generally considered to include a spectrum of disorders (eosinophilic granuloma, Hand-Schuller-Christian disease, Letterer-Siwe disease) which vary in the degree of systemic involvement and aggressive nature of the disease. Eosinophilic granuloma, the most benign of the three conditions, is characterized by osteolytic bone lesions involving the skull, ribs, pelvis, or scapula. In most instances, these lesions are solitary; however, there are rare reports of bilaterally symmetrical lesions of the temporal bones. We present one such case. Methods: Case report. Results: A 14-month-old male presented with a 2-month history of left-sided, bloody otorrhea and a polypoid mass in the left external meatus. Prior biopsy of the aural mass revealed acute and chronic granulation tissue. Computed tomography indicated osteolytic lesions in the temporal bones bilaterally. A plain-film KUB showed a possible abdominal mass, thus raising the concern for malignancy. Differential diagnosis initially included neuroblastoma, lymphoma, sarcoma, and LCH. Subsequent bone marrow aspirate was normal, and an abdominal ultrasound revealed no mass. A biopsy of the temporal bone lesion indicated a proliferation of mononuclear histiocytes, consistent with Langerhans cell histiocytosis. The patient was treated with steroids and a single-drug chemotherapy regimen (Vinblastine). Complete response was noted at a recent follow-up. Conclusion: This case illustrates an unusual presentation of eosinophilic granuloma, a condition which should be considered in the diagnostic workup of bony lesions in children and young adults.
POSTERS
P274
Otolaryngology– Head and Neck Surgery August 2003
P189 Intracranial Complications of Sinusitis: A Five-Year Review of Three Cases Fabio De Rezende Pinna MD (presenter); Fabrizio Ricci Romano MD; Richard L Voegels MD Sao Paulo Brazil; Sao Paulo Brazil; Sao Paulo Brazil
Objectives: Despite improvements in antibiotic therapies and surgical techniques, sinusitis still carries a risk of serious and potentially fatal complications. In this study, we describe our findings in the patients who had intracranial complications. Methods: We examined the charts of 38 patients who had been admitted to the “Hospital das Clı´nicas da Faculdade de Medicina da USP,” Sa˜ o Paulo, Brazil, between January 1, 1997, and March 31, 2002, for treatment of sinusitis complications. Of these 38 patients, 35 had orbital complications and 3 had intracranial complications. Results: We found one case of epidural abscess, one case of cavernous sinus thrombosis, and one case of frontal osteomyelitis and cerebral abscess. The last two patients did not take antibiotics properly before admitted to our service. All of them had no central signs or symptoms such as seizures, hemiparesia, weakness, and mental confusion. The diagnosis of intracranial complications was only possible by the use of computed tomography. Those patients were treated with intravenous antibiotics and drainage of the affected sinuses by endoscopic surgery, which has been helping to improve outcomes. Neurosurgery was not necessary in any of these, and neither the use of anticoagulant therapy in the case of cavernous sinus thrombosis. Conclusion: Intracranial infections secondary to rhinosinusitis occur sporadically, and although it appears that this cannot be prevented, recognition and management of these patients should be undertaken immediately and are best achieved via a multidisciplinary approach.
P273
tragnathic examples include the temporal bone, concha bullosa, and sphenoid. Previous case reports have raised the issue of this lesion as a pregnancy-dependent tumor. Methods: We present the case of a 16-year-old pregnant female diagnosed with a rapidly progressive, expansile growth of the left maxilla during her first trimester. Results: Fine needle aspiration biopsy was nondiagnostic, and incisional biopsy via the Caldwell-Luc approach provided the diagnosis. Definitive management consisted of wide local excision. Conclusion: Differential diagnosis, pathology, and treatment options are discussed. P191 Presentation and Management of Pseudoaneurysm of the Superficial Temporal Artery Jason G Cundiff MD (presenter); Benjamin A Bassichis MD; Tim Malisch MD Chicago IL; Chicago IL; Chicago IL
Maxillary Giant Cell Reparative Granuloma Presenting during Pregnancy
Objectives: In 1740 Thomas Bartholin reported the first case of a temporal artery pseudoaneurysm secondary to blunt trauma. Since that time, nearly 200 cases of face and temple pseudoaneurysm secondary to trauma have been described; however, this phenomenon has been rarely described in the otolaryngology literature. In this report, we describe an atypical case secondary to penetrating trauma and a non-surgical treatment option by interventional radiology and endovascular embolization. Methods: Case report and literature review of superficial temporal artery pseudoaneurysm. Results: Superficial temporal artery pseudoaneurysms typically result from blunt trauma to the temple. These pseudoaneurysms present as a gradually enlarging pulsatile mass with a bruit on auscultation. Pseudoaneurysms result from total or partial rupture of the arterial wall with formation of a hematoma and a new wall of perivascular tissue. Definitive diagnosis can be made with ultrasound, CT scan with contrast, or angiogram (gold standard). In the past these were typically treated surgically with ligation of the affected artery and resection of the pseudoaneurysm. This case was successfully managed with endovascular embolization of pseudoaneurysm with regression of the facial mass and no complications. Conclusion: Superficial temporal artery pseudoaneurysms can be successfully treated by endovascular embolization avoiding the risks involved with surgery and the scar from incision.
Patricia L Bell MD (presenter); Robert P Zitsch III MD Columbia MO; Columbia MO
P192
P190
Objectives: Giant cell reparative granuloma can manifest in the head and neck as a locally aggressive, destructive process. It can present in several locations of the head and neck but is most commonly diagnosed in the mandible. Ex-
Pediatric Subglottic Granular Cell Myoblastoma Steven E Sobol MD (presenter); Daniel Sharya Samadi MD; Ralph F Wetmore MD Philadelphia PA; Philadelphia PA; Philadelphia PA
POSTERS
Conclusion: The use of antibiotics for otitis media has significantly reduced the incidence of petrous apicitis. In this report, we describe an atypical case of bilateral petrous apicitis resulting from an anaerobic infection (Fusobacterium necrophorum). The treatment of the second side needs to be performed urgently to prevent bilateral carotid or jugular thrombosis, or cranial nerve palsies.
Scientific Posters
Scientific Posters
Objectives: Granular cell myoblastoma is a rare, benign tumor of unknown origin that most commonly presents in the oral cavity or larynx of adult patients. There have only been several reports in the literature of this tumor presenting in the pediatric population. The objective of this study is to report a case of a granular cell myoblastoma presenting in the subglottic airway of a child. Methods: Retrospective chart review. Results: A 7-year-old male presented with a history of a barking cough, shortness of breath on exertion, and progressively worsening biphasic stridor over a 3-month period. Physical examination was significant for biphasic stridor and was otherwise within normal limits. Computed tomography and magnetic resonance imaging demonstrated a 2-cm, round, solid, soft-tissue mass between the upper trachea and esophagus, which appeared to be extrinsic to the airway lumen. The patient underwent a direct laryngoscopy, bronchoscopy, and esophagoscopy, which revealed a rubbery submucosal mass in the posterior subglottic space. A laryngofissure was performed, and the mass was resected by elevating posterior subglottic mucosal flaps. Histopathological evaluation was consistent with granular cell myoblastoma. Conclusion: Although rare, granular cell myoblastoma should be considered in the differential diagnosis of subglottic lesions in children.
P193 Case Report on Sentinel Lymph Node Use in Merkel Cell Carcinoma Richard J Barnett MD (presenter); D Michael Rose MD; Timothy F Ragsdale MD; Mark E Augspurger MD New Orleans LA; Keesler AFB MS; Ocean Springs MS; Keesler AFB MS
Objectives: Merkel cell carcinoma (MCC) is a highly aggressive primary neuroendocrine tumor. The literature reveals that delayed diagnoses and poor survival are common with MCC. The role of sentinel lymph node has been shown to be effective in the evaluation of patients with melanoma and breast cancer. We present a case report on the use of sentinel lymph node biopsy in a patient with Merkel cell carcinoma of the tragus. Methods: Preoperative lymphoscintigraphy was performed on the morning of surgery by injecting radiolabeled colloid intradermally. Static images demonstrated uptake in two nodes in the same region. In the operating room, lymphazurine blue dye was injected intradermally around the primary Merkel cell carcinoma. Two sentinel lymph nodes were found to have blue dye as well as increased radioactive counts on intraoperative mapping. The nodes were removed and sent for permanent section.
Results: Histologic examination of the primary site revealed clear margins, and both nodes were negative for metastases. The patient received radiation to the primary site, ipsilateral parotid nodes, and superior cervical chain. The patient remained free of disease at a 7-month follow-up appointment. Conclusion: Merkel cell carcinoma of the skin has a poor prognosis and is difficult to study due to its rarity. Sentinel lymph node biopsy may prove to be an invaluable tool to decrease morbidity associated with elective nodal dissections, provide actual anatomic drainage patterns, and supply staging and prognostic information to the patient and physician. Further therapeutic options incorporating systemic therapy are needed to decrease the mortality rate.
P194 Langerhans Cell Histiocytosis Presenting as Bilateral Temporal Bone Lesions Chester Park Barton III MD (presenter); Drew M Horlbeck MD San Antonio TX; Boerne TX
Objectives: Langerhans cell histiocytosis, commonly referred to as histiocytosis X, is an idiopathic condition characterized by a proliferation of histiocytes. It is generally considered to include a spectrum of disorders (eosinophilic granuloma, Hand-Schuller-Christian disease, Letterer-Siwe disease) which vary in the degree of systemic involvement and aggressive nature of the disease. Eosinophilic granuloma, the most benign of the three conditions, is characterized by osteolytic bone lesions involving the skull, ribs, pelvis, or scapula. In most instances, these lesions are solitary; however, there are rare reports of bilaterally symmetrical lesions of the temporal bones. We present one such case. Methods: Case report. Results: A 14-month-old male presented with a 2-month history of left-sided, bloody otorrhea and a polypoid mass in the left external meatus. Prior biopsy of the aural mass revealed acute and chronic granulation tissue. Computed tomography indicated osteolytic lesions in the temporal bones bilaterally. A plain-film KUB showed a possible abdominal mass, thus raising the concern for malignancy. Differential diagnosis initially included neuroblastoma, lymphoma, sarcoma, and LCH. Subsequent bone marrow aspirate was normal, and an abdominal ultrasound revealed no mass. A biopsy of the temporal bone lesion indicated a proliferation of mononuclear histiocytes, consistent with Langerhans cell histiocytosis. The patient was treated with steroids and a single-drug chemotherapy regimen (Vinblastine). Complete response was noted at a recent follow-up. Conclusion: This case illustrates an unusual presentation of eosinophilic granuloma, a condition which should be considered in the diagnostic workup of bony lesions in children and young adults.
POSTERS
P274
Otolaryngology– Head and Neck Surgery August 2003