The Journal of Craniofacial Surgery
Brief Clinical Studies
& Volume 25, Number 4, July 2014
If the correct diagnosis is made, the excision process is not difficult. Because it is a well-encapsulated mass, it is easily excised from its surroundings, and thus, reports of recurrence have been very rare. Buyukbabani et al9 reported 2 cases of recurrence of cutaneous angiomyolipoma, but it is suspected that the excision during surgery in these cases was perhaps incomplete. We report this case of cutaneous angiomyolipoma, which we believe to be another category of skin and soft tissue tumors. FIGURE 3. A, Well-encapsulated cutaneous angiomyolipoma; B, hematoxylin-eosinYstained section shows variably sized blood vessels, smooth muscle bundles, and mature adipose tissue (100).
FIGURE 4. Immunochemistry stain. A, HMB-45: negative; B, smooth muscle myosin heavy chain: positive.
was first reported in 1990, by Fitzpatrick et al,6 who introduced the term angiolipoleiomyoma.3 With 23 previously reported cases, 39% (9/23) of which occurred in areas around the ear, the lesions are mostly solitary, asymptomatic, noninvasive, and nonrecurring.4,5 The lesion is found mostly in adults, with patients’ ages ranging from 16 to 77 years.4 However, according to Ammanagi et al,2 there was a recent case of angiomyolipoma found in a 3-year-old. The difference between cutaneous angiomyolipoma and renal angiomyolipoma is that the former has no link to tuberous sclerosis and is found predominantly in males, is noninvasive and rarely recurring, and tests negative to HMB-45 immunochemistry stain (Table 1).4 Cutaneous angiomyolipoma contains no epithelioid cell component that stains with HMB-45 and, for this reason, tests negative to this stain.6 Cutaneous angiomyolipoma is generally not known to be a skin tumor and thus is generally diagnosed as an epidermal inclusion cyst, lipoma, or vascular tumor.7,8 Before we performed surgery, we suspected that this lesion could be an epidermal inclusion cyst or a lymph node, but because the CT scan revealed a high-density lump, we began to suspect a vascular tumor and performed surgery. Because angiomyolipomas include a convoluted thick-walled blood vessel, during the contrast-enhanced CT, it is possible to misdiagnose this lesion as a vascular tumor such as a hemangioma. It is helpful to perform magnetic resonance imaging before the operation to obtain information that may aid in the diagnosis.
TABLE 1. Comparison of Renal AML and Cutaneous AML
Sex Tuberous sclerosis Aggressiveness Recurrence Number HMB-45 AML, angiomyolipoma.
e344
Renal AML
Cutaneous AML
Male predominance Associated Invasive Common Solitary or multiple Positive
Female predominance Not associated Noninvasive Rare Solitary Negative
REFERENCES 1. Singh K, Pai RR, Kini H, et al. Cutaneous angiomyolipoma. Indian J Pathol Microbiol 2009;52:242Y243 2. Ammanagi AS, Dombale VD, Shindholimath VV. Cutaneous angiomyolipoma. Indian Dermatol Online J 2012;3:40Y41 3. Blute ML, Malek PS, Segura JW. Angiomyolipoma: clinical metamorphosis and concepts for management. J Urol 1988;139:20Y24 4. Shin JU, Lee KY, Roh MR. A case of a cutaneous angiomyolipoma. Ann Dermatol 2009;21:217Y220 5. Mikoshiba Y, Murata H, Ashida A, et al. Case of a cutaneous angiomyolipoma in the ear. J Dermatol 2012;39:808Y809 6. Beer TW. Cutaneous angiomyolipomas are HMB45 negative, not associated with tuberous sclerosis, and should be considered as angioleiomyomas with fat. Am J Dermatopathol 2005;27:418Y421 7. Fitzpatrick JE, Mellete JR Jr, Hwang RJ, et al. Cutaneous angiolipoleiomyoma. J Am Acad Dermatol 1990;23:1093Y1098 8. Hatori M. Angiomyolipoma in the knee: a case report. UPS J Med Sci 2005;110:245Y249 9. Buyukbabani N, Tetikkurt S, Ozturk AS. Cutaneous angiomyolipomas: report of two cases with emphasis on HMB-45 utility. J Eur Acad Dermatol Venereol 1998;11:151Y154.
Central Giant Cell Granuloma in Pediatric Maxilla: Surgical Management Leonardo Perez Faverani, DDS, PhD, Sabrina Ferreira, DDS, Gabriel Ramalho Ferreira, DDS, MS, Juliana Zorzi Cole´te, DDS, Alessandra Marcondes Aranega, DDS, PhD, Idelmo Rangel Garcia, Ju´nior, DDS, PhD Abstract: Central giant cell granuloma (CGCG) is an intraosseous lesion consisting of fibrous cellular tissue that contains multiple foci of hemorrhage, multinucleated giant cells, and occasional trabeculae of woven bone. An 8-year-old boy presented himself complaining of From the Division of Oral and Maxillofacial Surgery, Department of Surgery and Integrated Clinic, Aracatuba Dental School, Univ Estadual Paulista Y Julio de Mesquita Filho Y UNESP, Ara0atuba, Brazil. Received October 9, 2013. Accepted for publication January 7, 2014. Address correspondence and reprint requests to Leonardo Perez Faverani, DDS, PhD, Departamento de Cirurgia e Clı´nica Integrada, Rua Jose´ Bonifa´cio, 1193 Vila Mendon0a, CEP: 16015-050 Ara0atuba, SP, Brazil; 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.0000000000000750
* 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 4, July 2014
a painless swelling in the left maxilla that had started 1 year. Computed tomography (CT) scan confirmed a poorly defined multilocular radiolucent lesion in the left maxilla crossing the midline. The patient underwent enucleation through an intraoral approach of the lesion. The biopsy revealed multinucleated giant cells in a fibrous stroma. A CT was taken approximately 1 year postoperatively. There was no clinical or radiographic evidence of recurrence. Therefore, surgical treatment of CGCG can be performed, trying to preserve the surrounding anatomic structures, which can be maintained in case the lesion does not show an aggressive clinical behavior, avoiding large surgical defects which are undesirable in children.
Brief Clinical Studies
FIGURE 2. Histopathological image revealing multinucleated giant cells in a fibrous stroma.
CGCG of the jaws are more commonly found in the mandible than in the maxilla and in females more often than in males. These lesions occur before the age of 30 years.3 However, in the current case, the
patient was 8 years old at the time of the first examination when the maxilla was affected. Clinical behavior of CGCG is variable. Rapidly growing expansive lesions with an aggressive appearance are at one end of the spectrum and small asymptomatic slow-growing lesions are at the other end.4 Clinical findings of the child of the present study were asymptomatic swelling of the maxilla with slow-growing tooth displacement as well as eruption failures. The tomographic examination in this case demonstrated a radiolucent unilocular area involving the maxilla on the right side, crossing the midline, with a well-defined defect which is consistent with the literature.5 Decreased nasal cavity and septal deviation were also observed on CT, however, without suggestive areas of nasal floor disruption. Histopathological examination showed multinucleated giant cells in a background of proliferative ovoid to spindle-shaped mesenchymal cells. Giant cell granuloma forms a lobulated mass of proliferative vascular connective tissue packed with giant cells.6 These giant cells are seen lying in vascular stroma with signs of bleeding into the mass, and deposits of hemosiderin are frequently observed.6 The procedures recommended in the literature vary according to the lesion. With aggressive or recurrent lesions, bone resection, including healthy bone, is an option, but it results in large surgical defects which are undesirable in children or young adults.7 For the remaining lesions, the treatments indicated are simple curettage, curettage accompanied by peripheral osteotomy, or cryotherapy with liquid nitrogen.8 Histopathological examination showed multinucleated giant cells in a fibrocellular proliferative mesenchyme, with ovoid to spindleshaped mesenchymal cells that seem like fibroblasts. Hemorrhagic outbreaks and hemosiderin deposits were also present. This fact is consistent with relevant literature.9 The histological and radiographic appearance of CGCG is similar to brown tumors observed in hyperparathyroidism, which should be excluded by performing differential diagnostic laboratory analysis.2 They were ruled out based on normal serum levels of calcium, phosphorus, alkaline phosphatase, and good renal function. Therefore, surgical treatment of CGCG can be performed trying to preserve the surrounding anatomic structures.
FIGURE 1. CT scan showing the presence of a poorly defined multilocular radiolucent lesion in the left maxilla crossing the midline.
FIGURE 3. One year CT postoperative showing new bone formation.
Key Words: Giant cell, granuloma, children, jaws, treatment
C
entral giant cell granuloma (CGCG) is a nonodontogenic disease that represents about 10% of all benign maxilla bone lesions.1 The World Health Organization has defined CGCG as an intraosseous lesion consisting of fibrous cellular tissue that contains multiple foci of hemorrhage, aggregations of multinucleated giant cells, and occasional trabeculae of woven bone with an incidence rate of 1.1/million population/year.2 Management of CGCG has traditionally been achieved by surgical removal of the lesion.
CLINICAL REPORT An 8-year-old boy presented himself complaining of a painless swelling in the left maxilla that had started 1 year ago. CT scan confirmed the presence of a poorly defined multilocular radiolucent lesion in the left maxilla crossing the midline (Fig. 1). The lesion was greater than 4 cm and the overlying mucosa was normal in color and texture. Incisional biopsy revealed multinucleated giant cells in a fibrous stroma (Fig. 2). Clinical, radiographic, and histopathological findings were consistent with the presence of a CGCG. Hyperparathyroidism was excluded as laboratory investigations showed that parathyroid hormone, alkaline phosphates, and calcium levels were all within normal limits. The patient underwent enucleation through an intraoral approach of the lesion under general anesthesia, and histopathological findings were consistent with a CGCG. A CT was performed approximately 1 year postoperatively (Fig. 3). There was no clinical or radiographic evidence of recurrence.
DISCUSSION
* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
e345
The Journal of Craniofacial Surgery
Brief Clinical Studies
REFERENCES 1. Nogueira RL, Teixeira RC, Cavalcante RB, et al. Intralesional injection of triamcinolone hexacetonide as an alternative treatment for central giant-cell granuloma in 21 cases. Int J Oral Maxillofac Surg 2010;39:1204Y1210 2. Teixeira RC, Horz HP, Damante JH, et al. SH3BP2-encoding exons involved in cherubism are not associated with central giant cell granuloma. Int J Oral Maxillofac Surg 2011;40:851Y855 3. Eisenbud L, Stern M, Rothberg M, et al. Central giant cell granuloma of the jaws: experiences in the management of thirty-seven cases. J Oral Maxillofac Surg 1988;46:376Y384 4. Schutz P, El-Bassuoni KH, Munish J, et al. Aggressive central giant cell granuloma of the mandible. J Oral Maxillofac Surg 2010;68:2537Y2544 5. O’Connell JE, Kearns GJ. Aggressive giant cell granuloma of the jaws treated with interferon alpha: a report of two cases. Ir J Med Sci 2013;182:163Y170 6. Kumar KA, Humayun S, Kumar BP, et al. Reparative giant cell granuloma of the maxilla. Ann Maxillofac Surg 2011;1:181Y186 7. da Silva Sampieri MB, Yaedu RY, Santos PS, et al. Central giant cell granuloma: treatment with calcitonin, triamcinolone acetonide, and a cystic finding 3 years and 6 months after the primary treatment. Oral Maxillofac Surg 2013;17:229Y234 8. de Lange J, van den Akker HP, van den Berg H. Central giant cell granuloma of the jaw: a review of the literature with emphasis on therapy options. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;104:603Y615 9. Nicolai G, Lore B, Mariani G, et al. Central giant cell granuloma of the jaws. J Craniofac Surg 2010;21:383Y386
A Rare Cause of Dysphagia in Children: Lymphangiomatous Polyp of the Palatine Tonsil Emre Gunbey, MD,* Hediye Pinar Gu¨nbey, MD,Þ Yasemin Do¨lek, MD,þ Yasemin Yuyucu Karabulut, MDþ Abstract: Oropharyngeal and tonsillar masses are a very rare cause of dysphagia in children. In this article, we describe a rare cause of dysphagia in children, a lymphangiomatous lesion of the palatine tonsil, and discuss the clinical and histopathologic features of these lesions. Key Words: Dysphagia, tonsillar masses, lymphangiomatous polyp
From the *Department of Otorhinolaryngology, Ondokuz Mayis University School of Medicine; †Department of Radiology, Samsun Training and Research Hospital, Samsun; and ‡Department of Pathology, Cankiri State Hospital, Ankara, Turkey. Received November 15, 2013. Accepted for publication January 17, 2014. Address correspondence and reprint requests to Emre Gunbey, MD, Department of Otorhinolaryngology, Ondokuz Mayis University School of Medicine, Kurupelit, Samsun, Turkey 55139; 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.0000000000000773
e346
& Volume 25, Number 4, July 2014
N
ervous system disorders (eg, cerebral palsy, meningitis, encephalopathy), gastrointestinal conditions (eg, reflux), conditions affecting the airway, head and neck abnormalities and masses, heart disease, respiratory difficulties, infectious disease of the throat, as well as problems with parent-child interactions at meal times are the major etiological factors causes to dysphagia and swallowing problems in children. Oropharyngeal and tonsillar masses are a very rare cause of dysphagia in children.1 Benign tumors of the tonsils are rarely seen, compared with malignancies.2,3 The polypoid masses tend to have varying degrees of fibrous, adipose, and lymphoid components, and they tend to ocur most commonly in the lower gastrointestinal tracts.4 Lymphangiomatous polypoid lesions also arise from the lymphatic and vascular systems but are histologically different than a typical lymphatic malformation.2,5 Lymphangiomatous polypoid lesions of the head and neck are very rare, and case reports of these tumors arising from the palatine tonsils in the literature are sparse.2,6Y10 Here, in this article, we describe a rare cause of dysphagia in children, a lymphangiomatous polypoid lesion of the palatine tonsil, and discuss the clinical and histopathologic features of these lesions.
CLINICAL REPORT A 14-year-old girl was presented to the otolaryngology clinic for evaluation of dysphagia and foreign body sensation in the throat for 6 months. She had no other local or systemic symptoms. Results of physical examination of her oral cavity revealed a long, pale mass lesion extending from the left palatine tonsil to the root of the tongue (Fig. 1). The other otorhinolaryngologic examination was unremarkable, and there was no palpable cervical lymphadenopathy. To get detailed information about the origin and extention of the mass, a preoperative gadolinium magnetic resonance imaging (MRI) scan of the neck was performed. On the MRI scan, a lesion extending from the left tonsillar fossa to the pharynx with hypointense signal on T1 weighted images and slightly hyperintense signal on T2 weighted images showed supression on T1 weighted short tau inversion recovery images, suggesting an intensive and fatty content (Fig. 2). The patient underwent a left tonsillectomy. Surgical procedure and postoperative follow-up period were unremarkable. The symptoms of dysphagia gradually improved without any other interventions after 48 hours postoperatively. In the histopathologic examination, the left tonsil specimen was measured macroscopically 2.5 1.9 0.6 cm in diameter. A firm, smooth polypoid lesion, 2.5 0.8 0.6 cm in diameter, was observed in the surface of the tonsil tissue (Fig. 3). Histologically, its surface was covered with parakeratotic squamous epithelium and its stroma was composed of loose fibrous tissue including numerous dilated lymphatic space and aggregates of lymphoid tissue (Fig. 4). In light of these pathologic findings, the diagnosis of lymphangiomatous polyp was confirmed. After surgical excision, the
FIGURE 1. Physical examination of the oral cavity revealed a long, pale mass lesion extending from the left palatine tonsil to the root of the tongue.
* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Brief Clinical Studies
& Volume 25, Number 4, July 2014
If the correct diagnosis is made, the excision process is not difficult. Because it is a well-encapsulated mass, it is easily excised from its surroundings, and thus, reports of recurrence have been very rare. Buyukbabani et al9 reported 2 cases of recurrence of cutaneous angiomyolipoma, but it is suspected that the excision during surgery in these cases was perhaps incomplete. We report this case of cutaneous angiomyolipoma, which we believe to be another category of skin and soft tissue tumors. FIGURE 3. A, Well-encapsulated cutaneous angiomyolipoma; B, hematoxylin-eosinYstained section shows variably sized blood vessels, smooth muscle bundles, and mature adipose tissue (100).
FIGURE 4. Immunochemistry stain. A, HMB-45: negative; B, smooth muscle myosin heavy chain: positive.
was first reported in 1990, by Fitzpatrick et al,6 who introduced the term angiolipoleiomyoma.3 With 23 previously reported cases, 39% (9/23) of which occurred in areas around the ear, the lesions are mostly solitary, asymptomatic, noninvasive, and nonrecurring.4,5 The lesion is found mostly in adults, with patients’ ages ranging from 16 to 77 years.4 However, according to Ammanagi et al,2 there was a recent case of angiomyolipoma found in a 3-year-old. The difference between cutaneous angiomyolipoma and renal angiomyolipoma is that the former has no link to tuberous sclerosis and is found predominantly in males, is noninvasive and rarely recurring, and tests negative to HMB-45 immunochemistry stain (Table 1).4 Cutaneous angiomyolipoma contains no epithelioid cell component that stains with HMB-45 and, for this reason, tests negative to this stain.6 Cutaneous angiomyolipoma is generally not known to be a skin tumor and thus is generally diagnosed as an epidermal inclusion cyst, lipoma, or vascular tumor.7,8 Before we performed surgery, we suspected that this lesion could be an epidermal inclusion cyst or a lymph node, but because the CT scan revealed a high-density lump, we began to suspect a vascular tumor and performed surgery. Because angiomyolipomas include a convoluted thick-walled blood vessel, during the contrast-enhanced CT, it is possible to misdiagnose this lesion as a vascular tumor such as a hemangioma. It is helpful to perform magnetic resonance imaging before the operation to obtain information that may aid in the diagnosis.
TABLE 1. Comparison of Renal AML and Cutaneous AML
Sex Tuberous sclerosis Aggressiveness Recurrence Number HMB-45 AML, angiomyolipoma.
e344
Renal AML
Cutaneous AML
Male predominance Associated Invasive Common Solitary or multiple Positive
Female predominance Not associated Noninvasive Rare Solitary Negative
REFERENCES 1. Singh K, Pai RR, Kini H, et al. Cutaneous angiomyolipoma. Indian J Pathol Microbiol 2009;52:242Y243 2. Ammanagi AS, Dombale VD, Shindholimath VV. Cutaneous angiomyolipoma. Indian Dermatol Online J 2012;3:40Y41 3. Blute ML, Malek PS, Segura JW. Angiomyolipoma: clinical metamorphosis and concepts for management. J Urol 1988;139:20Y24 4. Shin JU, Lee KY, Roh MR. A case of a cutaneous angiomyolipoma. Ann Dermatol 2009;21:217Y220 5. Mikoshiba Y, Murata H, Ashida A, et al. Case of a cutaneous angiomyolipoma in the ear. J Dermatol 2012;39:808Y809 6. Beer TW. Cutaneous angiomyolipomas are HMB45 negative, not associated with tuberous sclerosis, and should be considered as angioleiomyomas with fat. Am J Dermatopathol 2005;27:418Y421 7. Fitzpatrick JE, Mellete JR Jr, Hwang RJ, et al. Cutaneous angiolipoleiomyoma. J Am Acad Dermatol 1990;23:1093Y1098 8. Hatori M. Angiomyolipoma in the knee: a case report. UPS J Med Sci 2005;110:245Y249 9. Buyukbabani N, Tetikkurt S, Ozturk AS. Cutaneous angiomyolipomas: report of two cases with emphasis on HMB-45 utility. J Eur Acad Dermatol Venereol 1998;11:151Y154.
Central Giant Cell Granuloma in Pediatric Maxilla: Surgical Management Leonardo Perez Faverani, DDS, PhD, Sabrina Ferreira, DDS, Gabriel Ramalho Ferreira, DDS, MS, Juliana Zorzi Cole´te, DDS, Alessandra Marcondes Aranega, DDS, PhD, Idelmo Rangel Garcia, Ju´nior, DDS, PhD Abstract: Central giant cell granuloma (CGCG) is an intraosseous lesion consisting of fibrous cellular tissue that contains multiple foci of hemorrhage, multinucleated giant cells, and occasional trabeculae of woven bone. An 8-year-old boy presented himself complaining of From the Division of Oral and Maxillofacial Surgery, Department of Surgery and Integrated Clinic, Aracatuba Dental School, Univ Estadual Paulista Y Julio de Mesquita Filho Y UNESP, Ara0atuba, Brazil. Received October 9, 2013. Accepted for publication January 7, 2014. Address correspondence and reprint requests to Leonardo Perez Faverani, DDS, PhD, Departamento de Cirurgia e Clı´nica Integrada, Rua Jose´ Bonifa´cio, 1193 Vila Mendon0a, CEP: 16015-050 Ara0atuba, SP, Brazil; 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.0000000000000750
* 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 4, July 2014
a painless swelling in the left maxilla that had started 1 year. Computed tomography (CT) scan confirmed a poorly defined multilocular radiolucent lesion in the left maxilla crossing the midline. The patient underwent enucleation through an intraoral approach of the lesion. The biopsy revealed multinucleated giant cells in a fibrous stroma. A CT was taken approximately 1 year postoperatively. There was no clinical or radiographic evidence of recurrence. Therefore, surgical treatment of CGCG can be performed, trying to preserve the surrounding anatomic structures, which can be maintained in case the lesion does not show an aggressive clinical behavior, avoiding large surgical defects which are undesirable in children.
Brief Clinical Studies
FIGURE 2. Histopathological image revealing multinucleated giant cells in a fibrous stroma.
CGCG of the jaws are more commonly found in the mandible than in the maxilla and in females more often than in males. These lesions occur before the age of 30 years.3 However, in the current case, the
patient was 8 years old at the time of the first examination when the maxilla was affected. Clinical behavior of CGCG is variable. Rapidly growing expansive lesions with an aggressive appearance are at one end of the spectrum and small asymptomatic slow-growing lesions are at the other end.4 Clinical findings of the child of the present study were asymptomatic swelling of the maxilla with slow-growing tooth displacement as well as eruption failures. The tomographic examination in this case demonstrated a radiolucent unilocular area involving the maxilla on the right side, crossing the midline, with a well-defined defect which is consistent with the literature.5 Decreased nasal cavity and septal deviation were also observed on CT, however, without suggestive areas of nasal floor disruption. Histopathological examination showed multinucleated giant cells in a background of proliferative ovoid to spindle-shaped mesenchymal cells. Giant cell granuloma forms a lobulated mass of proliferative vascular connective tissue packed with giant cells.6 These giant cells are seen lying in vascular stroma with signs of bleeding into the mass, and deposits of hemosiderin are frequently observed.6 The procedures recommended in the literature vary according to the lesion. With aggressive or recurrent lesions, bone resection, including healthy bone, is an option, but it results in large surgical defects which are undesirable in children or young adults.7 For the remaining lesions, the treatments indicated are simple curettage, curettage accompanied by peripheral osteotomy, or cryotherapy with liquid nitrogen.8 Histopathological examination showed multinucleated giant cells in a fibrocellular proliferative mesenchyme, with ovoid to spindleshaped mesenchymal cells that seem like fibroblasts. Hemorrhagic outbreaks and hemosiderin deposits were also present. This fact is consistent with relevant literature.9 The histological and radiographic appearance of CGCG is similar to brown tumors observed in hyperparathyroidism, which should be excluded by performing differential diagnostic laboratory analysis.2 They were ruled out based on normal serum levels of calcium, phosphorus, alkaline phosphatase, and good renal function. Therefore, surgical treatment of CGCG can be performed trying to preserve the surrounding anatomic structures.
FIGURE 1. CT scan showing the presence of a poorly defined multilocular radiolucent lesion in the left maxilla crossing the midline.
FIGURE 3. One year CT postoperative showing new bone formation.
Key Words: Giant cell, granuloma, children, jaws, treatment
C
entral giant cell granuloma (CGCG) is a nonodontogenic disease that represents about 10% of all benign maxilla bone lesions.1 The World Health Organization has defined CGCG as an intraosseous lesion consisting of fibrous cellular tissue that contains multiple foci of hemorrhage, aggregations of multinucleated giant cells, and occasional trabeculae of woven bone with an incidence rate of 1.1/million population/year.2 Management of CGCG has traditionally been achieved by surgical removal of the lesion.
CLINICAL REPORT An 8-year-old boy presented himself complaining of a painless swelling in the left maxilla that had started 1 year ago. CT scan confirmed the presence of a poorly defined multilocular radiolucent lesion in the left maxilla crossing the midline (Fig. 1). The lesion was greater than 4 cm and the overlying mucosa was normal in color and texture. Incisional biopsy revealed multinucleated giant cells in a fibrous stroma (Fig. 2). Clinical, radiographic, and histopathological findings were consistent with the presence of a CGCG. Hyperparathyroidism was excluded as laboratory investigations showed that parathyroid hormone, alkaline phosphates, and calcium levels were all within normal limits. The patient underwent enucleation through an intraoral approach of the lesion under general anesthesia, and histopathological findings were consistent with a CGCG. A CT was performed approximately 1 year postoperatively (Fig. 3). There was no clinical or radiographic evidence of recurrence.
DISCUSSION
* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
e345
The Journal of Craniofacial Surgery
Brief Clinical Studies
REFERENCES 1. Nogueira RL, Teixeira RC, Cavalcante RB, et al. Intralesional injection of triamcinolone hexacetonide as an alternative treatment for central giant-cell granuloma in 21 cases. Int J Oral Maxillofac Surg 2010;39:1204Y1210 2. Teixeira RC, Horz HP, Damante JH, et al. SH3BP2-encoding exons involved in cherubism are not associated with central giant cell granuloma. Int J Oral Maxillofac Surg 2011;40:851Y855 3. Eisenbud L, Stern M, Rothberg M, et al. Central giant cell granuloma of the jaws: experiences in the management of thirty-seven cases. J Oral Maxillofac Surg 1988;46:376Y384 4. Schutz P, El-Bassuoni KH, Munish J, et al. Aggressive central giant cell granuloma of the mandible. J Oral Maxillofac Surg 2010;68:2537Y2544 5. O’Connell JE, Kearns GJ. Aggressive giant cell granuloma of the jaws treated with interferon alpha: a report of two cases. Ir J Med Sci 2013;182:163Y170 6. Kumar KA, Humayun S, Kumar BP, et al. Reparative giant cell granuloma of the maxilla. Ann Maxillofac Surg 2011;1:181Y186 7. da Silva Sampieri MB, Yaedu RY, Santos PS, et al. Central giant cell granuloma: treatment with calcitonin, triamcinolone acetonide, and a cystic finding 3 years and 6 months after the primary treatment. Oral Maxillofac Surg 2013;17:229Y234 8. de Lange J, van den Akker HP, van den Berg H. Central giant cell granuloma of the jaw: a review of the literature with emphasis on therapy options. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;104:603Y615 9. Nicolai G, Lore B, Mariani G, et al. Central giant cell granuloma of the jaws. J Craniofac Surg 2010;21:383Y386
A Rare Cause of Dysphagia in Children: Lymphangiomatous Polyp of the Palatine Tonsil Emre Gunbey, MD,* Hediye Pinar Gu¨nbey, MD,Þ Yasemin Do¨lek, MD,þ Yasemin Yuyucu Karabulut, MDþ Abstract: Oropharyngeal and tonsillar masses are a very rare cause of dysphagia in children. In this article, we describe a rare cause of dysphagia in children, a lymphangiomatous lesion of the palatine tonsil, and discuss the clinical and histopathologic features of these lesions. Key Words: Dysphagia, tonsillar masses, lymphangiomatous polyp
From the *Department of Otorhinolaryngology, Ondokuz Mayis University School of Medicine; †Department of Radiology, Samsun Training and Research Hospital, Samsun; and ‡Department of Pathology, Cankiri State Hospital, Ankara, Turkey. Received November 15, 2013. Accepted for publication January 17, 2014. Address correspondence and reprint requests to Emre Gunbey, MD, Department of Otorhinolaryngology, Ondokuz Mayis University School of Medicine, Kurupelit, Samsun, Turkey 55139; 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.0000000000000773
e346
& Volume 25, Number 4, July 2014
N
ervous system disorders (eg, cerebral palsy, meningitis, encephalopathy), gastrointestinal conditions (eg, reflux), conditions affecting the airway, head and neck abnormalities and masses, heart disease, respiratory difficulties, infectious disease of the throat, as well as problems with parent-child interactions at meal times are the major etiological factors causes to dysphagia and swallowing problems in children. Oropharyngeal and tonsillar masses are a very rare cause of dysphagia in children.1 Benign tumors of the tonsils are rarely seen, compared with malignancies.2,3 The polypoid masses tend to have varying degrees of fibrous, adipose, and lymphoid components, and they tend to ocur most commonly in the lower gastrointestinal tracts.4 Lymphangiomatous polypoid lesions also arise from the lymphatic and vascular systems but are histologically different than a typical lymphatic malformation.2,5 Lymphangiomatous polypoid lesions of the head and neck are very rare, and case reports of these tumors arising from the palatine tonsils in the literature are sparse.2,6Y10 Here, in this article, we describe a rare cause of dysphagia in children, a lymphangiomatous polypoid lesion of the palatine tonsil, and discuss the clinical and histopathologic features of these lesions.
CLINICAL REPORT A 14-year-old girl was presented to the otolaryngology clinic for evaluation of dysphagia and foreign body sensation in the throat for 6 months. She had no other local or systemic symptoms. Results of physical examination of her oral cavity revealed a long, pale mass lesion extending from the left palatine tonsil to the root of the tongue (Fig. 1). The other otorhinolaryngologic examination was unremarkable, and there was no palpable cervical lymphadenopathy. To get detailed information about the origin and extention of the mass, a preoperative gadolinium magnetic resonance imaging (MRI) scan of the neck was performed. On the MRI scan, a lesion extending from the left tonsillar fossa to the pharynx with hypointense signal on T1 weighted images and slightly hyperintense signal on T2 weighted images showed supression on T1 weighted short tau inversion recovery images, suggesting an intensive and fatty content (Fig. 2). The patient underwent a left tonsillectomy. Surgical procedure and postoperative follow-up period were unremarkable. The symptoms of dysphagia gradually improved without any other interventions after 48 hours postoperatively. In the histopathologic examination, the left tonsil specimen was measured macroscopically 2.5 1.9 0.6 cm in diameter. A firm, smooth polypoid lesion, 2.5 0.8 0.6 cm in diameter, was observed in the surface of the tonsil tissue (Fig. 3). Histologically, its surface was covered with parakeratotic squamous epithelium and its stroma was composed of loose fibrous tissue including numerous dilated lymphatic space and aggregates of lymphoid tissue (Fig. 4). In light of these pathologic findings, the diagnosis of lymphangiomatous polyp was confirmed. After surgical excision, the
FIGURE 1. Physical examination of the oral cavity revealed a long, pale mass lesion extending from the left palatine tonsil to the root of the tongue.
* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
