- Email: [email protected]
Endoscopic management of an inverted nasal papilloma in a child
876 COOTER et al.
Otolaryngology– Head and Neck Surgery June 1998
Endoscopic management of an inverted nasal papilloma in a child MICHAEL S. COOTER, MD, SCOTT A. CHARLTON, MD, DENIS LAFRENIERE, MD, and JEFFREY SPIRO, MD, Farmington, Connecticut
S
inonasal papillomas are rare, benign tumors that arise from ectodermally derived nasal and paranasal sinus mucosa, known classically as the Schneiderian membrane. Anecdotal reports of these tumors date as far back as 1854, but it was not until 1971 that Hyams,1 in a large series reported from the Armed Forces Institute of Pathology, formally described these lesions in histologic terms and subclassified them into inverted, exophytic, and columnar cell types. The inverted papilloFrom the Division of Otolaryngology, Department of Surgery, University of Connecticut School of Medicine. Presented at the Spring Meeting of the New England Otolaryngology Society, Boston, Mass., April 12, 1996. Reprint requests: Denis Lafreniere, MD, Division of OtolaryngologyMC 3955, University of Connecticut School of Medicine, 263 Farmington Ave., Farmington, CT 06030. Otolaryngol Head Neck Surg 1998;118:876-9. Copyright © 1998 by the American Academy of OtolaryngologyÐ Head and Neck Surgery Foundation, Inc. 0194-5998/98/$5.00 + 0 23/4/79027
ma (IP) is the most common type, accounting for 60% to 70% of all Schneiderian papillomas.2 Overall, however, IPs represent only 0.5% to 4% of all nasal tumors.3 Inverted papillomas are found predominantly in men in the 5th or 6th decades of life. Fewer than thirty cases have been reported in children or adolescents. Inverted papillomas have a tendency to recur if not completely excised, can be locally destructive, and are associated with malignancy.4 These characteristics have made complete surgical excision the mainstay of treatment for IP. Wide excision via lateral rhinotomy or midfacial degloving is currently the accepted Ògold standardÓ for management of this lesion. Recently, however, a number of authors have proposed endoscopic surgery as an option for definitive management of carefully selected tumors. An uncommon case of IP in a child that was treated with primary endoscopic excision is described. The evaluation and management of this lesion are discussed in the context of the pediatric patient.
Otolaryngology– Head and Neck Surgery Volume 118 Number 6
COOTER et al. 877
Fig. 1. Preoperative CT scan demonstrating right-sided nasal mass with medial bowing of the lateral nasal wall and opacification of the right maxillary sinus.
CASE REPORT
A 10-year-old boy presented to the emergency department for evaluation of lacerations sustained after falling through a plate glass window. On physical examination, an intranasal mass filling the right nasal cavity was observed. Upon questioning, the patient disclosed a 2-month history of right nasal obstruction with associated clear nasal discharge. On further evaluation by the otolaryngologyÐhead and neck surgery service, the patient denied epistaxis, facial pain, vision changes, or systemic complaints. The past history of the patient was significant for minor trauma, with no prior hospitalizations, and no history of environmental allergies or cystic fibrosis. On examination, the boy was found to have a fleshy mass filling the right nasal cavity. The full extent of the mass was difficult to evaluate because the patient refused a flexible nasopharyngoscope examination at the time. Notably, no middle ear effusion or nasal bone splaying were present. Visual acuity, extraocular movements, and facial sensation were intact. A computed tomography (CT) scan (Fig. 1) showed a mass filling the right nasal cavity with associated medial bulging of the lateral nasal wall and opacification of the right maxillary sinus. There was no obvious bone destruction or extension of the mass into the orbit or intracranially.
The patient underwent an examination under general anesthesia in the operating room. The left nasal cavity and nasopharynx were carefully evaluated and determined to be free of disease. The findings of a biopsy of the right nasal mass were consistent with a diagnosis of IP. The diagnosis and the surgical management options were discussed with the family. An endoscopic approach was advocated for definitive management of the tumor, with the option to convert to an open procedure if the tumor proved too extensive to be fully excised in this manner. At the time of the definitive procedure, the mass was found to originate from the posterior border of the maxillary os, with its base extending toward, but not involving, the ethmoid bulla. The medial maxillary sinus wall and a portion of the inferior turbinate were involved. The maxillary sinus was filled with postobstructive mucus. Complete excision of the mass was accomplished by performance of a wide maxillary antrostomy, anterior ethmoidectomy, and partial inferior turbinectomy. A pathologic examination of the specimen showed respiratory and squamous epithelium inverting into the underlying stroma, with an intact basement membrane (Fig. 2). No malignant changes were present. The patient had an uneventful recovery, and on routine postoperative office visits has shown no evidence of recurrence at the 1-year follow up evaluation.
878 COOTER et al.
Otolaryngology– Head and Neck Surgery June 1998
Fig. 2. Photomicrograph of section of nasal mass demonstrating inversion of epithelium into underlying stroma. (Hematoxylin and eosin stain; original magnification 3 100).
DISCUSSION The most common presenting complaints in patients with IP are nonspecific and include, in decreasing order of frequency, nasal obstruction, rhinorrhea, facial pain, and epistaxis.4 Other intranasal lesions in the pediatric age group that can present with these symptoms, and therefore should be considered in the differential diagnosis of a pediatric nasal mass, are inflammatory polyps, hemangiomas, lesions of minor salivary glands, neural tumors, pyogenic granulomas, and rhabdomyosarcoma.5 Findings on physical examination can supplement the history, with IP typically presenting as a unilateral nasal mass originating from the lateral nasal wall. Imaging plays a vital role in the workup of suspected or biopsy-proven IPs. Computed tomography can identify bone erosion and can suggest sinus involvement, guiding therapeutic intervention. Sinus involvement is variable but includes, in decreasing order of frequency, ethmoid, maxillary, frontal, and sphenoid sinuses.4 As demonstrated in this case, distinguishing sinus involvement with tumor from postobstructive mucus on CT scans can be difficult. In this instance, magnetic resonance imaging (MRI) may play a role in the differentiation of soft tissue from mucus.6 Surgical excision is the treatment of choice for IP. Historically, limited local excision was advocated for the management of this lesion, but because of the high recurrence rates this approach was supplanted by techniques such as medial maxillectomy, which allow for a wide resection of all involved mucosa.3 With the advent of improved endoscopic techniques and technology, however, a conservative surgical
approach may again be considered in certain carefully selected cases. Recent studies have demonstrated results and recurrence rates using endoscopic surgery that are comparable to those of more extensive procedures. Kamel7 was one of the first authors to demonstrate the excellent results achievable with the endoscopic approach, reporting no recurrences in 3 patients followed up for an average of 23 months. Waitz and Wigand8 also noted comparable recurrence rates of 17% (6/35 patients) after endoscopic management versus 19% (3/16 patients) after extranasal resection of IPs. More recent studies by Lawson et al.,3 McCary et al.,9 and Stankiewicz and Girgis6 have documented similar success with endoscopic surgical management of IP. Comparisons of these techniques in pediatric patients are understandably lacking because of the infrequent occurrence of IP in this group. The morbidity of endoscopic surgery should be weighed against that of more extensive procedures in the management of the pediatric patient with IP. When compared with a medial maxillectomy via a lateral rhinotomy or midfacial degloving approach, endoscopic surgical management of IP has the benefit of the lack of an external scar, less blood loss, and a shorter hospital stay.9 Furthermore, the surgeon should consider the effects that any procedure might have on future facial growth in the pediatric patient. This is a controversial yet important consideration that has yet to be fully elucidated in the literature. Currently, most clinical data on this subject area provided by studies on the effects of trauma on the developing facial skeleton. Midfacial development is considered a complex process regulated not only by bony structures such as the nasal septum, but also by overlying nasal
Otolaryngology– Head and Neck Surgery Volume 118 Number 6
mucosa and soft tissues.10 Intuitively, then, major alterations of the structural components of the midface, as in medial maxillectomy for wide excision of an IP, could affect further development in these areas. In fact, even a more conservative procedure may have deleterious effects, as suggested by Mair et al.11 in a recent study on midfacial development in newborn piglets after endoscopic sinus surgery. An attempt at endoscopic resection of IP may be warranted in carefully selected pediatric cases. These selected patients may benefit from a more conservative procedure as opposed to initially performing an extensive procedure such as medial maxillectomy. Waitz and Wigand8 emphasize the importance that careful evaluation of tumor extent plays in this management decision. If endoscopic surgery is used, the lesion should be adequately visualized to allow complete resection of the involved mucosa. This requirement would preclude from consideration for endoscopic surgery those tumors that are excessively large or those with extensive sinus involvement. These tumors are better managed with the more traditional techniques, which offer unparalleled exposure of structures less accessible to endoscopic surgery. Therefore, a low threshold for converting to an open procedure should be maintained, because it is important to ensure adequate initial resection of all involved mucosa. This important caveat should be emphasized in preoperative discussions with the patient and in the informed consent for the definitive procedure when endoscopic surgical excision of IP is considered. CONCLUSION The case of a 10-year-old child with an IP was described. The tumor of this pediatric patient was definitively managed with endoscopic surgery, and he has remained free of recur-
COOTER et al. 879
rence to date. Although en bloc resection remains the mainstay of surgical therapy for IP, a more conservative endoscopic approach may be indicated in the management of carefully selected, limited lesions in pediatric patients. REFERENCES 1. Hyams VJ. Papillomas of the nasal cavity and paranasal sinuses: a clinicopathological study of 315 cases. Ann Otol Rhinol Laryngol 1971;80:192-206. 2. Buchwald C, Franzmann MB, Tos M. Sinonasal papillomas: a report of 82 cases in Copenhagen County, including a longitudinal epidemiological and clinical study. Laryngoscope 1995;105:72-9. 3. Lawson W, Ho BT, Shaari CM, Biller HF. Inverted papilloma: a report of 112 cases. Laryngoscope 1995;105:282-8. 4. Vrabec DP. The inverted Schneiderian papilloma: a 25-year study. Laryngoscope 1994;104:582-605. 5. Limaye AP, Mirani N, Kwartler J, Raz S. Inverted schneiderian papilloma of the sinonasal tract in children. Pediatric Pathol Lab Med 1989;9:583-90. 6. Stankiewicz JA, Girgis SJ. Endoscopic surgical treatment of nasal and paranasal sinus inverted papilloma. Otolaryngol Head Neck Surg 1993;109(6):988-95. 7. Kamel RH. Conservative endoscopic surgery in inverted papilloma: preliminary report. Arch Otolaryngol Head Neck Surg 1992;118:649-53. 8. Waitz G, Wigand E. Results of endoscopic sinus surgery for the treatment of inverted papillomas. Laryngoscope 1992;102:91722. 9. McCary WS, Gross CW, Reibel JF, Cantrell RW. Preliminary report: endoscopic versus external surgery in the management of inverted papilloma. Laryngoscope 1994;104:415-9. 10. Koltai PJ. Maxillofacial injuries in children. In: Smith JD, Bumsted RM, editors. Pediatric facial plastic and reconstructive surgery. New York: Raven Press Ltd; 1993. p. 283-316. 11. Mair EA, Bolger WE, Breisch EA. Sinus and facial growth after pediatric endoscopic sinus surgery. Arch Otolaryngol Head Neck Surg 1995;121(5):547-52.
Otolaryngology– Head and Neck Surgery June 1998
Endoscopic management of an inverted nasal papilloma in a child MICHAEL S. COOTER, MD, SCOTT A. CHARLTON, MD, DENIS LAFRENIERE, MD, and JEFFREY SPIRO, MD, Farmington, Connecticut
S
inonasal papillomas are rare, benign tumors that arise from ectodermally derived nasal and paranasal sinus mucosa, known classically as the Schneiderian membrane. Anecdotal reports of these tumors date as far back as 1854, but it was not until 1971 that Hyams,1 in a large series reported from the Armed Forces Institute of Pathology, formally described these lesions in histologic terms and subclassified them into inverted, exophytic, and columnar cell types. The inverted papilloFrom the Division of Otolaryngology, Department of Surgery, University of Connecticut School of Medicine. Presented at the Spring Meeting of the New England Otolaryngology Society, Boston, Mass., April 12, 1996. Reprint requests: Denis Lafreniere, MD, Division of OtolaryngologyMC 3955, University of Connecticut School of Medicine, 263 Farmington Ave., Farmington, CT 06030. Otolaryngol Head Neck Surg 1998;118:876-9. Copyright © 1998 by the American Academy of OtolaryngologyÐ Head and Neck Surgery Foundation, Inc. 0194-5998/98/$5.00 + 0 23/4/79027
ma (IP) is the most common type, accounting for 60% to 70% of all Schneiderian papillomas.2 Overall, however, IPs represent only 0.5% to 4% of all nasal tumors.3 Inverted papillomas are found predominantly in men in the 5th or 6th decades of life. Fewer than thirty cases have been reported in children or adolescents. Inverted papillomas have a tendency to recur if not completely excised, can be locally destructive, and are associated with malignancy.4 These characteristics have made complete surgical excision the mainstay of treatment for IP. Wide excision via lateral rhinotomy or midfacial degloving is currently the accepted Ògold standardÓ for management of this lesion. Recently, however, a number of authors have proposed endoscopic surgery as an option for definitive management of carefully selected tumors. An uncommon case of IP in a child that was treated with primary endoscopic excision is described. The evaluation and management of this lesion are discussed in the context of the pediatric patient.
Otolaryngology– Head and Neck Surgery Volume 118 Number 6
COOTER et al. 877
Fig. 1. Preoperative CT scan demonstrating right-sided nasal mass with medial bowing of the lateral nasal wall and opacification of the right maxillary sinus.
CASE REPORT
A 10-year-old boy presented to the emergency department for evaluation of lacerations sustained after falling through a plate glass window. On physical examination, an intranasal mass filling the right nasal cavity was observed. Upon questioning, the patient disclosed a 2-month history of right nasal obstruction with associated clear nasal discharge. On further evaluation by the otolaryngologyÐhead and neck surgery service, the patient denied epistaxis, facial pain, vision changes, or systemic complaints. The past history of the patient was significant for minor trauma, with no prior hospitalizations, and no history of environmental allergies or cystic fibrosis. On examination, the boy was found to have a fleshy mass filling the right nasal cavity. The full extent of the mass was difficult to evaluate because the patient refused a flexible nasopharyngoscope examination at the time. Notably, no middle ear effusion or nasal bone splaying were present. Visual acuity, extraocular movements, and facial sensation were intact. A computed tomography (CT) scan (Fig. 1) showed a mass filling the right nasal cavity with associated medial bulging of the lateral nasal wall and opacification of the right maxillary sinus. There was no obvious bone destruction or extension of the mass into the orbit or intracranially.
The patient underwent an examination under general anesthesia in the operating room. The left nasal cavity and nasopharynx were carefully evaluated and determined to be free of disease. The findings of a biopsy of the right nasal mass were consistent with a diagnosis of IP. The diagnosis and the surgical management options were discussed with the family. An endoscopic approach was advocated for definitive management of the tumor, with the option to convert to an open procedure if the tumor proved too extensive to be fully excised in this manner. At the time of the definitive procedure, the mass was found to originate from the posterior border of the maxillary os, with its base extending toward, but not involving, the ethmoid bulla. The medial maxillary sinus wall and a portion of the inferior turbinate were involved. The maxillary sinus was filled with postobstructive mucus. Complete excision of the mass was accomplished by performance of a wide maxillary antrostomy, anterior ethmoidectomy, and partial inferior turbinectomy. A pathologic examination of the specimen showed respiratory and squamous epithelium inverting into the underlying stroma, with an intact basement membrane (Fig. 2). No malignant changes were present. The patient had an uneventful recovery, and on routine postoperative office visits has shown no evidence of recurrence at the 1-year follow up evaluation.
878 COOTER et al.
Otolaryngology– Head and Neck Surgery June 1998
Fig. 2. Photomicrograph of section of nasal mass demonstrating inversion of epithelium into underlying stroma. (Hematoxylin and eosin stain; original magnification 3 100).
DISCUSSION The most common presenting complaints in patients with IP are nonspecific and include, in decreasing order of frequency, nasal obstruction, rhinorrhea, facial pain, and epistaxis.4 Other intranasal lesions in the pediatric age group that can present with these symptoms, and therefore should be considered in the differential diagnosis of a pediatric nasal mass, are inflammatory polyps, hemangiomas, lesions of minor salivary glands, neural tumors, pyogenic granulomas, and rhabdomyosarcoma.5 Findings on physical examination can supplement the history, with IP typically presenting as a unilateral nasal mass originating from the lateral nasal wall. Imaging plays a vital role in the workup of suspected or biopsy-proven IPs. Computed tomography can identify bone erosion and can suggest sinus involvement, guiding therapeutic intervention. Sinus involvement is variable but includes, in decreasing order of frequency, ethmoid, maxillary, frontal, and sphenoid sinuses.4 As demonstrated in this case, distinguishing sinus involvement with tumor from postobstructive mucus on CT scans can be difficult. In this instance, magnetic resonance imaging (MRI) may play a role in the differentiation of soft tissue from mucus.6 Surgical excision is the treatment of choice for IP. Historically, limited local excision was advocated for the management of this lesion, but because of the high recurrence rates this approach was supplanted by techniques such as medial maxillectomy, which allow for a wide resection of all involved mucosa.3 With the advent of improved endoscopic techniques and technology, however, a conservative surgical
approach may again be considered in certain carefully selected cases. Recent studies have demonstrated results and recurrence rates using endoscopic surgery that are comparable to those of more extensive procedures. Kamel7 was one of the first authors to demonstrate the excellent results achievable with the endoscopic approach, reporting no recurrences in 3 patients followed up for an average of 23 months. Waitz and Wigand8 also noted comparable recurrence rates of 17% (6/35 patients) after endoscopic management versus 19% (3/16 patients) after extranasal resection of IPs. More recent studies by Lawson et al.,3 McCary et al.,9 and Stankiewicz and Girgis6 have documented similar success with endoscopic surgical management of IP. Comparisons of these techniques in pediatric patients are understandably lacking because of the infrequent occurrence of IP in this group. The morbidity of endoscopic surgery should be weighed against that of more extensive procedures in the management of the pediatric patient with IP. When compared with a medial maxillectomy via a lateral rhinotomy or midfacial degloving approach, endoscopic surgical management of IP has the benefit of the lack of an external scar, less blood loss, and a shorter hospital stay.9 Furthermore, the surgeon should consider the effects that any procedure might have on future facial growth in the pediatric patient. This is a controversial yet important consideration that has yet to be fully elucidated in the literature. Currently, most clinical data on this subject area provided by studies on the effects of trauma on the developing facial skeleton. Midfacial development is considered a complex process regulated not only by bony structures such as the nasal septum, but also by overlying nasal
Otolaryngology– Head and Neck Surgery Volume 118 Number 6
mucosa and soft tissues.10 Intuitively, then, major alterations of the structural components of the midface, as in medial maxillectomy for wide excision of an IP, could affect further development in these areas. In fact, even a more conservative procedure may have deleterious effects, as suggested by Mair et al.11 in a recent study on midfacial development in newborn piglets after endoscopic sinus surgery. An attempt at endoscopic resection of IP may be warranted in carefully selected pediatric cases. These selected patients may benefit from a more conservative procedure as opposed to initially performing an extensive procedure such as medial maxillectomy. Waitz and Wigand8 emphasize the importance that careful evaluation of tumor extent plays in this management decision. If endoscopic surgery is used, the lesion should be adequately visualized to allow complete resection of the involved mucosa. This requirement would preclude from consideration for endoscopic surgery those tumors that are excessively large or those with extensive sinus involvement. These tumors are better managed with the more traditional techniques, which offer unparalleled exposure of structures less accessible to endoscopic surgery. Therefore, a low threshold for converting to an open procedure should be maintained, because it is important to ensure adequate initial resection of all involved mucosa. This important caveat should be emphasized in preoperative discussions with the patient and in the informed consent for the definitive procedure when endoscopic surgical excision of IP is considered. CONCLUSION The case of a 10-year-old child with an IP was described. The tumor of this pediatric patient was definitively managed with endoscopic surgery, and he has remained free of recur-
COOTER et al. 879
rence to date. Although en bloc resection remains the mainstay of surgical therapy for IP, a more conservative endoscopic approach may be indicated in the management of carefully selected, limited lesions in pediatric patients. REFERENCES 1. Hyams VJ. Papillomas of the nasal cavity and paranasal sinuses: a clinicopathological study of 315 cases. Ann Otol Rhinol Laryngol 1971;80:192-206. 2. Buchwald C, Franzmann MB, Tos M. Sinonasal papillomas: a report of 82 cases in Copenhagen County, including a longitudinal epidemiological and clinical study. Laryngoscope 1995;105:72-9. 3. Lawson W, Ho BT, Shaari CM, Biller HF. Inverted papilloma: a report of 112 cases. Laryngoscope 1995;105:282-8. 4. Vrabec DP. The inverted Schneiderian papilloma: a 25-year study. Laryngoscope 1994;104:582-605. 5. Limaye AP, Mirani N, Kwartler J, Raz S. Inverted schneiderian papilloma of the sinonasal tract in children. Pediatric Pathol Lab Med 1989;9:583-90. 6. Stankiewicz JA, Girgis SJ. Endoscopic surgical treatment of nasal and paranasal sinus inverted papilloma. Otolaryngol Head Neck Surg 1993;109(6):988-95. 7. Kamel RH. Conservative endoscopic surgery in inverted papilloma: preliminary report. Arch Otolaryngol Head Neck Surg 1992;118:649-53. 8. Waitz G, Wigand E. Results of endoscopic sinus surgery for the treatment of inverted papillomas. Laryngoscope 1992;102:91722. 9. McCary WS, Gross CW, Reibel JF, Cantrell RW. Preliminary report: endoscopic versus external surgery in the management of inverted papilloma. Laryngoscope 1994;104:415-9. 10. Koltai PJ. Maxillofacial injuries in children. In: Smith JD, Bumsted RM, editors. Pediatric facial plastic and reconstructive surgery. New York: Raven Press Ltd; 1993. p. 283-316. 11. Mair EA, Bolger WE, Breisch EA. Sinus and facial growth after pediatric endoscopic sinus surgery. Arch Otolaryngol Head Neck Surg 1995;121(5):547-52.