The Journal of Craniofacial Surgery • Volume 25, Number 5, September 2014
of recurrence of paraganglioma. Three months after second surgical intervention, the patient died because of respiratory insufficiency that was associated with poor general conditions. The study was exempt from IRB approval as a case report. We followed Helsinki Declaration guidelines.
DISCUSSION Vagal paraganglioma may develop everywhere along the course of the vagal nerve.1 The terms glomus tumor and chemodectoma are often used for such neoplasm too.2 The literature reveals a predominance of female cases. Most cases display a lateral-cervical mass with a low growth pattern, thus being often asymptomatic whereas less than half present hoarseness or dysphonia (vagal dysfunction caused by paralysis of the vocal cords). A minor fraction of the paragangliomas are synthesizing and releasing catecholamines, producing a clinical situation similar to the pheochromocytoma (tachyarrhythmia, flushing, and hypertension).4,5 Malignancy in vagal paragangliomas has been rarely reported, with the metastatic involvement of the lungs, skull, vertebral bodies, cervical lymph nodes, heart, liver, pancreas, pleura, dura mater, and skin. The only acceptable parameter for characterizing malignancy is the presence of metastatic disease, either extension to regional lymph nodes or distant metastasis, more than histological findings.1–3 Therefore, our case could be considered as malignant. Vagal paragangliomas frequently arise from the inferior nodose ganglion, but they may occur at any point along the course of the cervical vagal nerve. Therefore, different symptoms can be associated with this lesion, such as a painless mass in the lateral neck, dysphagia, cranial nerve deficits, and Horner syndrome.1,2 Precise determination of tumor sites and their relation to adjacent structures are essential to plan an appropriate treatment. Both CT and MR depict these highly vascular, soft tissue masses equally well on contrast-enhanced images. The displacement of the internal carotid artery and external carotid artery may be observed in some cases.1,2 Finally, scintigraphy with octreotide and PET-CT can be useful devices to aid the search for multicentric locations.1–3 Vagal paragangliomas and pheochromocytomas are made up of 2 cell types: chief cells and sustentacular cells. Paraganglial tumors derive from the chief cells and are benign neoplasms in the majority of cases. Few paragangliomas are malignant, and there are no histopathological or immunohistochemical criteria for the diagnosis of a malignant paraganglial tumor. Therefore, a diagnosis of malignant vagal paraganglioma can be performed when there is metastasis to non-neuroendocrine tissue, as in our case. Most frequently, metastasis involve cervical lymph nodes, bone, liver, and lung.1–4 A correct diagnosis of vagal paraganglioma by fine-needle aspiration biopsy is extremely difficult, although it remains an important tool for the pre-therapeutic diagnosis. The differential diagnosis of a nontender lateral neck mass includes lymphadenopathies, branchial cleft cysts, salivary gland tumors, and neurogenic tumors.1–6 Surgery is still the therapy of choice in patients with malignant vagal paraganglioma with resection of all metastases whenever possible. During surgical intervention, the vagal nerve is at risk to be sacrificed, whereas if possible the hypoglossal and glossopharyngeal nerves should be spared.1–6 Vagal nerve resection leads to a weak and hoarse voice due to paralysis of the vocal cord and aspiration, partially due to incomplete closure of the larynx, but also because the sensory innervation of the laryngeal mucosa is lost.5,6 A preoperative embolization is not necessary, but it can be useful to decrease loss of blood. Furthermore, if surgery is not feasible, because of poor medical conditions, a therapeutic embolization may be an option too. Instead, the use of radiotherapy and chemotherapy in the treatment of vagal paraganglioma is controversial.1–6
Brief Clinical Studies
Anyway, a long time follow-up is needed for patients affected by malignant vagal paragangliomas. All patients have to undergo a thorough examination of the head and neck. When a recurrence is suspected, a scintigraphy or a PET can be prescribed.1–6
CONCLUSION Malignant vagal paraganglioma is a rare and challenging lesion that has to be carefully assessed to plan an appropriate treatment. The presence of metastases is the only parameter of malignancy and suggests a poor prognosis. A strict follow-up is fundamental for an early diagnosis of eventual recurrences.
REFERENCES 1. Cakmakci H, Usal C, Guneri A. Metastatic hypervascular lymph nodes in malignant glomus vagale tumor: angiography findings. Clin Imaging 2001;25:167–170 2. Boedeker CC. Paragangliomas and paraganglioma syndromes. GMS Curr Top Otorhinolaryngol Head Neck Surg 2011;10:1–26 3. Carlsen CS, Godballe C, Krogdahl AS, et al. Malignant vagal paraganglioma: report of a case treated with embolization and surgery. Auris Nasus Larynx 2003;30:443–446 4. Paal E, Chung EM. Head and neck pathology-radiology classics: vagal paraganglioma. Head Neck Pathol 2007;1:35–37 5. Lozano FS, Gómez JL, Mondillo MC, et al. Surgery of vagal paragangliomas: six patients and review of literature. Surg Oncol 2008;17:281–287 6. Bradshaw JW, Jansen JC. Management of vagal paraganglioma: is operative resection really the best option? Surgery 2005;137:225–228
Complicated Giant Mucoceles of the Frontal Sinus: Approach to Endoscopic Marsupialization Technique Sedat Aydin, MD, Mehmet Gökhan Demir, MD Abstract: A number of external and radical procedures have been the only surgical approaches for the treatment of frontal sinus mucoceles. However, these procedures have major complications including high surgical morbidity, esthetic concerns of postoperative scar formation, and difficulty in radiologic diagnosis of recurrence after obliteration. In this clinical report, we demonstrated an endoscopic marsupialization technique that could easily be applied to a very large mucocele that has extended to the intracranial region, without any serious complications.
From the ENT Department, Dr. Lutfi Kirdar Kartal Education and Research Hospital, Istanbul, Turkey. Received February 4, 2014. Accepted for publication April 20, 2014. Address correspondence and reprint requests to Sedat Aydin, MD, İstasyon Caddesi Merdivenli Sokak, No 5 D 6, Kartal-Istanbul, Turkey; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001034
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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The Journal of Craniofacial Surgery • Volume 25, Number 5, September 2014
Brief Clinical Studies
Key Words: Mucocele, frontal sinus, marsupialization, endoscopy
M
ucocele was first described by Rollet in 1896 and histopathologically defined by Onodi in 1901. Mucoceles of paranasal sinuses are mucus-filled cavities that primarily result from the obstructed sinus outflow and also can result from trauma, chronic sinusitis, neoplasms (eg, osteoma), after sinus surgery, or allergic reactions.1,2 Mucocele, a pseudostratified ciliated columnar epitheliallined, mucous-containing sac, completely fills the paranasal sinus and is capable of expansion by virtue of dynamic process of bone resorption and new bone formation3 that will eventually erode through the bony wall and then protrude to the surrounding structures.4–6 Mucoceles tend to occur between the fourth and seventh decades of life, and both the women and men are affected at the same ratio.7 Frontal sinus is the most common site of mucoceles.1 Clinically, mucoceles are represented with headaches, diplopia, and swelling of the upper eyelids. If a mucocele erodes the posterior wall, it can then lead to meningitis, meningoencephalitis, and cerebrospinal fluid fistula formation.8 Classification of mucoceles is based on the radiologic examination.9
PATIENT An 89-year-old woman who was admitted to our outpatient clinic had a left-sided exoftalmia and a left-sided swelling on the left eye. Reviewing her medical history, she was operated on because of a left-sided pain beyond her eye and diplopia 2 years ago in 2011. In 2013, she was seen by an opthalmologist when her complains recurred and was then referred to an otolaryngologist. On the medical examination, her left middle concha was paradoxically curved and was in close contact with the septum, and this side of the mucosa was edematous (Fig. 1). Furthermore, her left eye movement was restricted, exoftalmus was detected, and the upper corner of the left orbital rim showed Pott puffy tumor sign. On the paranasal sinus computed tomography (CT), expansile smooth contured multiple giant mucoceles were detected on the left side of the frontal sinus, and mucoceles eroded the sinus wall integrity (Fig. 2). The informed consent was obtained from the patient. After the diagnosis, an endoscopic marsupialization of the mucoceles was performed under local anesthesia via transnasal route in the operating room without any postoperative complications. In 1-year follow-up, the endoscopic marsupialization site has been still patent (Fig. 3), and we did not detect any complications or recurrence in the coronal view of the paranasal sinus CT (Fig. 2).
DISCUSSION Mucoceles are mucous-filled, pseudostratified columnar epitheliumlined cystic benign lesions. They mostly localize to the frontal sinuses with an equal frequency in men and women. Mucoceles occur at any age but have been more common between the forth and seventh decades of life. The least common side is the maxillary sinus region.10 Although the complete etiology is not clear, mucoceles
FIGURE 1. Preoperative endoscopic examination of the left nasal cavity. On the left side, curved middle concha is seen (right side) and, on the left oedemotous mucosa, is seen lateral to it (left side).
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FIGURE 2. Preoperative (right side) and postoperative (left side) CT examinations of the giant mucoceles are seen.
are mostly seen after surgey, trauma, chronic sinusitis, or neoplasms. In a retrospective review by Busaba and Salman,2 it has been suggested that ethmoid mucoceles occur as a late complication of endoscopic ethmoidectomy; however, in another study, the recurrence rate of mucoceles after surgery was reported as 0%.11 Clinical presentation of mucoceles varies from asymptomatic to headache and visual disturbances as proptosis and diplopia. Physical examination usually presents with periorbital tenderness, swelling, chemosis, decreased visual acuity, eye movement restriction, and asymetry of the face. Evaluating a CT image, findings that are required for the diagnosis of mucoceles include homogenous isodence mass, clearly defined margin, and patchy osteolysis around the mass.12–14 Marginal sclerosis and sinus wall erosion are also other common indicators for diagnosis. Mucoceles tend to appear fairly bright on T1-weighted images compared with the brain and appear isohyperintense on T2-weighted images.14 Surgical treatment of mucoceles includes endoscopic or nonendoscopic methods. The choice of a surgical approach depends on the patient’s pathology. The nonendoscopic methods are trephination procedures and osteoplastic flap procedure with obliteration. The endoscopic techniques are marsupialization or modified endoscopic lothrop procedure. Both endoscopic techniques can be used for the treatment of complicated frontal mucoceles. Khong et al15 have reported that, in 21 patients with frontal mucoceles and a follow-up time of 16 months, only 5 patients had additional surgery and 4 patients had minor complications such as epistaxis and adhesions. In another report by Chiu and Vaughan,16 10 patients had undergone computer-aided endoscopic surgical drainage, and 9 patients were symptom free in a median time of 25 months. In our case, although our patient had a complicated mucocele, we were able to endoscopically drain the mucocele and constructed a huge sinus ostium, and there was no recurrence in the follow-up. It should also be noted that the patient’s old age and other medical problems have been additional risk factors for major operations such as ostoeplastic flap technique. Thus, we believe that a minimal surgical intervention can be performed endoscopically and is adequate for the management of this patient. The use of endoscopic techniques shifted the surgical manegement of mucoceles. In 1989, Kennedy et al17 described the successful use of endoscopic approach in 15 of the 18 mucocele patients with a median followup of 17.4 months. In a study by Har-El,11 108 mucocele patients were treated with endoscopic methods, and only 5 patients had recurrent disease. Mucoceles that involve the skull base and orbital
FIGURE 3. Endoscopic site of the marsipulization postoperatively after 1 month (right side) and 1 year (left side).
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 25, Number 5, September 2014
lesions were reduced successfully with the endoscopic procedures. Lund and Milroy3 have shown that mucocele remnant retains respiratory epithelium with normal mucociliary clearence after marsupialization, which allows physiologic sinonasal function. However, the need for a complete mucosal cavity removal is still controversial. In summary, the endoscopic approach has several benefits, which are mainly better cosmetic outcomes and and lower complication rates. However, in a review of 250 cases of osteoplastic flap technique for the treatment of mucoceles by Hardy and Montgomery,18 19% of patients had complications including 13 patients with abdominal infection and 7 patients with cerebrospinal fluid leaks.
CONCLUSIONS Endoscopic procedures for mucoceles have a number of advantages including the lower rate of morbidity. This technique can be performed easily in an outpatient clinic under local anesthesia; thus, the patients with an underlying medical problem or with a high risk for general anesthesia are good candidates for the endoscopic procedure. Morever, this technique allows the better visualization of the surgical area both preoperatively and postoperatively and also provides a better preservation of the anatomic structures and nasal mucosa. If performed by experienced surgeons, an endoscopic surgery should be considered as an only procedure, specifically for the treatment of complicated mucocele cases.
REFERENCES 1. Thio D, Phelps PD, Bath AP. Maxillary sinus mucocele presenting as late complication of a maxillary advancement procedure. J Laryngol Otol 2003;117:402–403 2. Busaba NY, Salman SD. Ethmoid mucocele as a late complication of endoscopic ethmoidectomy. Otolaryngol Head Neck Surg 2003;128:517–522 3. Lund VJ, Milroy CM. Fronto-ethmoidal mucocele a histopathological analysis. J Laryngol Otol 1991;105:921–923 4. Lund VJ. Anatomical considerations in the aetiology of fronto-ethmoidal mucoceles. Rhinology 1987;25:83–88 5. Lund VJ. The complications of sinusitis. In: Kerr AG, Mackay IS, Bull TR, eds. Scott-Brown’s Otolaryngology-Rhinology. 6th ed. Oxford, UK: Butterworth- Heinemann; 1997 6. Serrano E, Klossek JM, Percodani J, et al. Surgical management of paranasal sinus mucoceles: A long term study of 60 cases. Otolaryngol Head Neck Surg 2004;131:131–140 7. Lai PC, Liao SL, Jou JR, et al. Transcaruncular approach for the management of frontoethmoid mucoceles. Br J Ophthalmol 2004;88:725 8. Chiarini L, Nocini PF, Bedogni A, et al. Intracranial spread of giant frontal mucocele: a case report. Br J Oral Maxillofac Surg 2000;38:637–640 9. Har-El G. Transnasal endoscopic management of frontal mucoceles. Otolaryngol Clin North Am 2001;34:243–251 10. Conboy PJ, Jones NS. The place of endoscopic sinus surgery in the treatment of paranasal sinus mucoceles. Clin Otolaryngol 2003;28:207–210. 11. Har-El G. Endoscopic management of 108 sinus mucoceles. Laryngoscope 2001;111:2131–2134 12. Tan CS, Yong VK, Yip LW, et al. An unusual presentation of a giant frontal sinus mucocele manifesting with a subcutaneous forehead mass. Ann Acad Med Singapore 2005;34:397–398 13. Suri A, Mahapatra AK, Gaikwad S, et al. Giant mucoceles of the frontal sinus: a series and review. J Clin Neurosci 2004;11:214–218 14. Edelman RR, Hesselink JR, Zlatkin MB, et al. Clinical magnetic resonance imaging. 3rd ed. Philadelphia: Elsevier; 2006:2035–2037 15. Khong JJ, Malhorta R, Selva D, et al. Efficacy of endoscopic sinus surgery for paranasal sinus mucocele including modified endoscopic Lothrop procedure for frontal sinus mucocele. J Laryngol Otol 2004;118:352–356
Brief Clinical Studies
16. Chiu AG, Vaughan WC. Management of the lateral frontal sinus lesion and the supraorbital cell mucocele. Am J Rhinol 2004;18:83–86 17. Kennedy DW, Josephson JS, Zinreich SJ, et al. Endoscopic sinus surgery for mucoceles: a viable alternative. Laryngoscope 1989;99:885–895 18. Hardy JM, Montgomery WW. Osteoplastic frontal sinusotomy: an analysis of 250 operations. Ann Otol Rhinol Laryngol 1976;85:523–532
Comprehensive Treatment and Rehabilitation of a Patient With Maxillary Arteriovenous Malformation Hossein Behnia, DMD, MS,* Mohammad Jafarian, DMD, MS,† Nima Dehghani, DMD,* Siavash Dehghani, MD,‡ Kaveh Seyedan, MD§ Abstract: Arteriovenous malformations (AVMs) of the maxilla are rare and potentially life-threatening conditions that can pose a therapeutic dilemma. We reported the first case of maxillary AVM in a 15-year-old girl who was treated by marginal hemimaxillectomy including overlying palatal mucosa and immediate replantation of the segment after removing the AVM tissues and teeth and covering by a full-thickness pedicled temporal muscle flap rotated into the mouth. Then, this preserved bone underwent distraction osteogenesis and dental implant rehabilitation successfully. This method was previously used for the definitive treatment of mandibular AVMs, and in this case, we applied this method for the first time in maxillary AVMs. In conclusion, this surgical method may be considered as a safe, convenient, and effective treatment and reconstructive modality for such vascular malformations in the maxilla and restores function and symmetry of the jaws while obviating the need for bone harvesting and future major reconstructive operations. Key Words: Vascular malformations, arteriovenous malformations, maxilla, rehabilitation
A
rteriovenous malformations (AVM) of the jaws are rare and potentially life-threatening conditions due to bleeding, which can be massive and difficult to control.1–4 The AVM arises because of From the *Department of Oral and Maxillofacial Surgery, Taleghani Hospital, Shahid Beheshti University of Medical Sciences, Tehran, Iran; †Dentofacial Deformity Research Center, Research Institute of Dental Sciences, Department of Oral and Maxillofacial Surgery, Shahid Beheshti University of Medical Sciences, Tehran, Iran; ‡Department of Neurosurgery, Poursina Hospital, Guilan University of Medical Sciences, Rasht, Iran; and §Department of Proesthetics, Dental School, Shahid Beheshti University of Medical Sciences, Tehran, Iran. Received February 15, 2014. Accepted for publication April 23, 2014. Address correspondence and reprint requests to Nima Dehghani, DMD, Department of Oral and Maxillofacial Surgery, Taleghani Hospital, Shahid Beheshti University of Medical Sciences, Parvaneh Avenue, Velenjak Street, Tehran, Iran 1985711151; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001054
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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of recurrence of paraganglioma. Three months after second surgical intervention, the patient died because of respiratory insufficiency that was associated with poor general conditions. The study was exempt from IRB approval as a case report. We followed Helsinki Declaration guidelines.
DISCUSSION Vagal paraganglioma may develop everywhere along the course of the vagal nerve.1 The terms glomus tumor and chemodectoma are often used for such neoplasm too.2 The literature reveals a predominance of female cases. Most cases display a lateral-cervical mass with a low growth pattern, thus being often asymptomatic whereas less than half present hoarseness or dysphonia (vagal dysfunction caused by paralysis of the vocal cords). A minor fraction of the paragangliomas are synthesizing and releasing catecholamines, producing a clinical situation similar to the pheochromocytoma (tachyarrhythmia, flushing, and hypertension).4,5 Malignancy in vagal paragangliomas has been rarely reported, with the metastatic involvement of the lungs, skull, vertebral bodies, cervical lymph nodes, heart, liver, pancreas, pleura, dura mater, and skin. The only acceptable parameter for characterizing malignancy is the presence of metastatic disease, either extension to regional lymph nodes or distant metastasis, more than histological findings.1–3 Therefore, our case could be considered as malignant. Vagal paragangliomas frequently arise from the inferior nodose ganglion, but they may occur at any point along the course of the cervical vagal nerve. Therefore, different symptoms can be associated with this lesion, such as a painless mass in the lateral neck, dysphagia, cranial nerve deficits, and Horner syndrome.1,2 Precise determination of tumor sites and their relation to adjacent structures are essential to plan an appropriate treatment. Both CT and MR depict these highly vascular, soft tissue masses equally well on contrast-enhanced images. The displacement of the internal carotid artery and external carotid artery may be observed in some cases.1,2 Finally, scintigraphy with octreotide and PET-CT can be useful devices to aid the search for multicentric locations.1–3 Vagal paragangliomas and pheochromocytomas are made up of 2 cell types: chief cells and sustentacular cells. Paraganglial tumors derive from the chief cells and are benign neoplasms in the majority of cases. Few paragangliomas are malignant, and there are no histopathological or immunohistochemical criteria for the diagnosis of a malignant paraganglial tumor. Therefore, a diagnosis of malignant vagal paraganglioma can be performed when there is metastasis to non-neuroendocrine tissue, as in our case. Most frequently, metastasis involve cervical lymph nodes, bone, liver, and lung.1–4 A correct diagnosis of vagal paraganglioma by fine-needle aspiration biopsy is extremely difficult, although it remains an important tool for the pre-therapeutic diagnosis. The differential diagnosis of a nontender lateral neck mass includes lymphadenopathies, branchial cleft cysts, salivary gland tumors, and neurogenic tumors.1–6 Surgery is still the therapy of choice in patients with malignant vagal paraganglioma with resection of all metastases whenever possible. During surgical intervention, the vagal nerve is at risk to be sacrificed, whereas if possible the hypoglossal and glossopharyngeal nerves should be spared.1–6 Vagal nerve resection leads to a weak and hoarse voice due to paralysis of the vocal cord and aspiration, partially due to incomplete closure of the larynx, but also because the sensory innervation of the laryngeal mucosa is lost.5,6 A preoperative embolization is not necessary, but it can be useful to decrease loss of blood. Furthermore, if surgery is not feasible, because of poor medical conditions, a therapeutic embolization may be an option too. Instead, the use of radiotherapy and chemotherapy in the treatment of vagal paraganglioma is controversial.1–6
Brief Clinical Studies
Anyway, a long time follow-up is needed for patients affected by malignant vagal paragangliomas. All patients have to undergo a thorough examination of the head and neck. When a recurrence is suspected, a scintigraphy or a PET can be prescribed.1–6
CONCLUSION Malignant vagal paraganglioma is a rare and challenging lesion that has to be carefully assessed to plan an appropriate treatment. The presence of metastases is the only parameter of malignancy and suggests a poor prognosis. A strict follow-up is fundamental for an early diagnosis of eventual recurrences.
REFERENCES 1. Cakmakci H, Usal C, Guneri A. Metastatic hypervascular lymph nodes in malignant glomus vagale tumor: angiography findings. Clin Imaging 2001;25:167–170 2. Boedeker CC. Paragangliomas and paraganglioma syndromes. GMS Curr Top Otorhinolaryngol Head Neck Surg 2011;10:1–26 3. Carlsen CS, Godballe C, Krogdahl AS, et al. Malignant vagal paraganglioma: report of a case treated with embolization and surgery. Auris Nasus Larynx 2003;30:443–446 4. Paal E, Chung EM. Head and neck pathology-radiology classics: vagal paraganglioma. Head Neck Pathol 2007;1:35–37 5. Lozano FS, Gómez JL, Mondillo MC, et al. Surgery of vagal paragangliomas: six patients and review of literature. Surg Oncol 2008;17:281–287 6. Bradshaw JW, Jansen JC. Management of vagal paraganglioma: is operative resection really the best option? Surgery 2005;137:225–228
Complicated Giant Mucoceles of the Frontal Sinus: Approach to Endoscopic Marsupialization Technique Sedat Aydin, MD, Mehmet Gökhan Demir, MD Abstract: A number of external and radical procedures have been the only surgical approaches for the treatment of frontal sinus mucoceles. However, these procedures have major complications including high surgical morbidity, esthetic concerns of postoperative scar formation, and difficulty in radiologic diagnosis of recurrence after obliteration. In this clinical report, we demonstrated an endoscopic marsupialization technique that could easily be applied to a very large mucocele that has extended to the intracranial region, without any serious complications.
From the ENT Department, Dr. Lutfi Kirdar Kartal Education and Research Hospital, Istanbul, Turkey. Received February 4, 2014. Accepted for publication April 20, 2014. Address correspondence and reprint requests to Sedat Aydin, MD, İstasyon Caddesi Merdivenli Sokak, No 5 D 6, Kartal-Istanbul, Turkey; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001034
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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The Journal of Craniofacial Surgery • Volume 25, Number 5, September 2014
Brief Clinical Studies
Key Words: Mucocele, frontal sinus, marsupialization, endoscopy
M
ucocele was first described by Rollet in 1896 and histopathologically defined by Onodi in 1901. Mucoceles of paranasal sinuses are mucus-filled cavities that primarily result from the obstructed sinus outflow and also can result from trauma, chronic sinusitis, neoplasms (eg, osteoma), after sinus surgery, or allergic reactions.1,2 Mucocele, a pseudostratified ciliated columnar epitheliallined, mucous-containing sac, completely fills the paranasal sinus and is capable of expansion by virtue of dynamic process of bone resorption and new bone formation3 that will eventually erode through the bony wall and then protrude to the surrounding structures.4–6 Mucoceles tend to occur between the fourth and seventh decades of life, and both the women and men are affected at the same ratio.7 Frontal sinus is the most common site of mucoceles.1 Clinically, mucoceles are represented with headaches, diplopia, and swelling of the upper eyelids. If a mucocele erodes the posterior wall, it can then lead to meningitis, meningoencephalitis, and cerebrospinal fluid fistula formation.8 Classification of mucoceles is based on the radiologic examination.9
PATIENT An 89-year-old woman who was admitted to our outpatient clinic had a left-sided exoftalmia and a left-sided swelling on the left eye. Reviewing her medical history, she was operated on because of a left-sided pain beyond her eye and diplopia 2 years ago in 2011. In 2013, she was seen by an opthalmologist when her complains recurred and was then referred to an otolaryngologist. On the medical examination, her left middle concha was paradoxically curved and was in close contact with the septum, and this side of the mucosa was edematous (Fig. 1). Furthermore, her left eye movement was restricted, exoftalmus was detected, and the upper corner of the left orbital rim showed Pott puffy tumor sign. On the paranasal sinus computed tomography (CT), expansile smooth contured multiple giant mucoceles were detected on the left side of the frontal sinus, and mucoceles eroded the sinus wall integrity (Fig. 2). The informed consent was obtained from the patient. After the diagnosis, an endoscopic marsupialization of the mucoceles was performed under local anesthesia via transnasal route in the operating room without any postoperative complications. In 1-year follow-up, the endoscopic marsupialization site has been still patent (Fig. 3), and we did not detect any complications or recurrence in the coronal view of the paranasal sinus CT (Fig. 2).
DISCUSSION Mucoceles are mucous-filled, pseudostratified columnar epitheliumlined cystic benign lesions. They mostly localize to the frontal sinuses with an equal frequency in men and women. Mucoceles occur at any age but have been more common between the forth and seventh decades of life. The least common side is the maxillary sinus region.10 Although the complete etiology is not clear, mucoceles
FIGURE 1. Preoperative endoscopic examination of the left nasal cavity. On the left side, curved middle concha is seen (right side) and, on the left oedemotous mucosa, is seen lateral to it (left side).
e462
FIGURE 2. Preoperative (right side) and postoperative (left side) CT examinations of the giant mucoceles are seen.
are mostly seen after surgey, trauma, chronic sinusitis, or neoplasms. In a retrospective review by Busaba and Salman,2 it has been suggested that ethmoid mucoceles occur as a late complication of endoscopic ethmoidectomy; however, in another study, the recurrence rate of mucoceles after surgery was reported as 0%.11 Clinical presentation of mucoceles varies from asymptomatic to headache and visual disturbances as proptosis and diplopia. Physical examination usually presents with periorbital tenderness, swelling, chemosis, decreased visual acuity, eye movement restriction, and asymetry of the face. Evaluating a CT image, findings that are required for the diagnosis of mucoceles include homogenous isodence mass, clearly defined margin, and patchy osteolysis around the mass.12–14 Marginal sclerosis and sinus wall erosion are also other common indicators for diagnosis. Mucoceles tend to appear fairly bright on T1-weighted images compared with the brain and appear isohyperintense on T2-weighted images.14 Surgical treatment of mucoceles includes endoscopic or nonendoscopic methods. The choice of a surgical approach depends on the patient’s pathology. The nonendoscopic methods are trephination procedures and osteoplastic flap procedure with obliteration. The endoscopic techniques are marsupialization or modified endoscopic lothrop procedure. Both endoscopic techniques can be used for the treatment of complicated frontal mucoceles. Khong et al15 have reported that, in 21 patients with frontal mucoceles and a follow-up time of 16 months, only 5 patients had additional surgery and 4 patients had minor complications such as epistaxis and adhesions. In another report by Chiu and Vaughan,16 10 patients had undergone computer-aided endoscopic surgical drainage, and 9 patients were symptom free in a median time of 25 months. In our case, although our patient had a complicated mucocele, we were able to endoscopically drain the mucocele and constructed a huge sinus ostium, and there was no recurrence in the follow-up. It should also be noted that the patient’s old age and other medical problems have been additional risk factors for major operations such as ostoeplastic flap technique. Thus, we believe that a minimal surgical intervention can be performed endoscopically and is adequate for the management of this patient. The use of endoscopic techniques shifted the surgical manegement of mucoceles. In 1989, Kennedy et al17 described the successful use of endoscopic approach in 15 of the 18 mucocele patients with a median followup of 17.4 months. In a study by Har-El,11 108 mucocele patients were treated with endoscopic methods, and only 5 patients had recurrent disease. Mucoceles that involve the skull base and orbital
FIGURE 3. Endoscopic site of the marsipulization postoperatively after 1 month (right side) and 1 year (left side).
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 25, Number 5, September 2014
lesions were reduced successfully with the endoscopic procedures. Lund and Milroy3 have shown that mucocele remnant retains respiratory epithelium with normal mucociliary clearence after marsupialization, which allows physiologic sinonasal function. However, the need for a complete mucosal cavity removal is still controversial. In summary, the endoscopic approach has several benefits, which are mainly better cosmetic outcomes and and lower complication rates. However, in a review of 250 cases of osteoplastic flap technique for the treatment of mucoceles by Hardy and Montgomery,18 19% of patients had complications including 13 patients with abdominal infection and 7 patients with cerebrospinal fluid leaks.
CONCLUSIONS Endoscopic procedures for mucoceles have a number of advantages including the lower rate of morbidity. This technique can be performed easily in an outpatient clinic under local anesthesia; thus, the patients with an underlying medical problem or with a high risk for general anesthesia are good candidates for the endoscopic procedure. Morever, this technique allows the better visualization of the surgical area both preoperatively and postoperatively and also provides a better preservation of the anatomic structures and nasal mucosa. If performed by experienced surgeons, an endoscopic surgery should be considered as an only procedure, specifically for the treatment of complicated mucocele cases.
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Brief Clinical Studies
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Comprehensive Treatment and Rehabilitation of a Patient With Maxillary Arteriovenous Malformation Hossein Behnia, DMD, MS,* Mohammad Jafarian, DMD, MS,† Nima Dehghani, DMD,* Siavash Dehghani, MD,‡ Kaveh Seyedan, MD§ Abstract: Arteriovenous malformations (AVMs) of the maxilla are rare and potentially life-threatening conditions that can pose a therapeutic dilemma. We reported the first case of maxillary AVM in a 15-year-old girl who was treated by marginal hemimaxillectomy including overlying palatal mucosa and immediate replantation of the segment after removing the AVM tissues and teeth and covering by a full-thickness pedicled temporal muscle flap rotated into the mouth. Then, this preserved bone underwent distraction osteogenesis and dental implant rehabilitation successfully. This method was previously used for the definitive treatment of mandibular AVMs, and in this case, we applied this method for the first time in maxillary AVMs. In conclusion, this surgical method may be considered as a safe, convenient, and effective treatment and reconstructive modality for such vascular malformations in the maxilla and restores function and symmetry of the jaws while obviating the need for bone harvesting and future major reconstructive operations. Key Words: Vascular malformations, arteriovenous malformations, maxilla, rehabilitation
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rteriovenous malformations (AVM) of the jaws are rare and potentially life-threatening conditions due to bleeding, which can be massive and difficult to control.1–4 The AVM arises because of From the *Department of Oral and Maxillofacial Surgery, Taleghani Hospital, Shahid Beheshti University of Medical Sciences, Tehran, Iran; †Dentofacial Deformity Research Center, Research Institute of Dental Sciences, Department of Oral and Maxillofacial Surgery, Shahid Beheshti University of Medical Sciences, Tehran, Iran; ‡Department of Neurosurgery, Poursina Hospital, Guilan University of Medical Sciences, Rasht, Iran; and §Department of Proesthetics, Dental School, Shahid Beheshti University of Medical Sciences, Tehran, Iran. Received February 15, 2014. Accepted for publication April 23, 2014. Address correspondence and reprint requests to Nima Dehghani, DMD, Department of Oral and Maxillofacial Surgery, Taleghani Hospital, Shahid Beheshti University of Medical Sciences, Parvaneh Avenue, Velenjak Street, Tehran, Iran 1985711151; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001054
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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