Vol. 117 No. 6 June 2014
CLINICOPATHOLOGIC CONFERENCE
An asymptomatic tumor on the dorsal tongue Eleni-Marina Kalogirou, DDS,a Konstantinos I. Tosios, DDS, PhD,b Vassileios Petsinis, MD, DDS, MSc, PhD,c Ioulia Chatzistamou, MD, PhD,d and Alexandra Sklavounou, DDS, MSc, PhDe National and Kapodistrian University of Athens and Evaggelismos General Hospital, Athens, Greece
(Oral Surg Oral Med Oral Pathol Oral Radiol 2014;117:659-662)
CLINICAL PRESENTATION A 42-year-old man was referred by his dentist for diagnosis and management of a painless nodule on the posterior dorsal surface of the tongue. According to the patient, the lesion was present for at least 2 months and developed after a fishbone injury 5 months earlier. An otorhinolaryngology specialist diagnosed it as a “hyperplastic lingual papilla”; an internist, as a “normal structure of the tongue.” It had “slightly shrunk” during the past week and did not cause any discomfort or functional disturbance. The patient’s medical history was noncontributory. Clinical examination revealed a well-circumscribed hemispherical tumor on the midline of the dorsal surface of the tongue, anterior to the lingual V (Figure 1). It measured approximately 0.6 cm, was covered by normal-appearing mucosa, and on palpation was painless, firm, and adherent to the overlying mucosa. The rest of the oral mucosa was within normal limits. No palpable lymph nodes were present.
Among foreign bodies, fish bones are those most commonly implanted in the upper aerodigestive tract, although they usually involve the base of the tongue and only rarely its mobile part.1 In contrast to the lesion in our patient, they are usually tender on palpation. Trauma in children or young adults commonly precedes the development of a mucocele, the most common reactive lesion of the oral soft tissues, but a mucocele typically presents as a translucent and fluctuant mass.2 In a study of 1715 mucoceles,2 none was reported to arise on the dorsal tongue, possibly owing to the scarcity of minor salivary glands in this area.3 Trauma is also associated with the development of pyogenic granuloma, but this tumor is typically erythematous and bleeding. In a recent review,4 only 7% of 1127 cases of pyogenic granuloma were located in the tongue. Finally, an irritation fibroma should be included in the differential diagnosis, because it is the most common intraoral tumor.
DIFFERENTIAL DIAGNOSIS An asymptomatic tumor on the dorsal tongue may represent an inflammatory-reactive, developmental, or neoplastic lesion. The indolent growth and good demarcation were against a diagnosis of malignancy.
Developmental The posterior dorsal tongue is a common site for developmental lesions. Lingual thyroid is usually found in the median posterior tongue, but it is situated between the foramen cecum and the epiglottis.5 The remnants of the thyroglossal duct tract that arises from the foramen cecum may form a thyroglossal duct cyst, which predominantly affects females.6 It usually involves the neck, although in 2% to 3% it may be intralingual and involve the inferior half of the tongue. The midline of the dorsal tongue anterior to the lingual V is the typical site of occurrence of median rhomboid glossitis, which has a striking male predilection.7 Apart from its typical presentation as an atrophic area, median rhomboid glossitis may rarely manifest as an exophytic mass, but it is usually red with a fissured or lobulated surface and hard consistency.7 The oral lymphoepithelial cyst usually affects adults and presents as a soft to firm mass of yellow-white discoloration.8 In a study of 120 intraoral lymphoepithelial cysts, Yang et al.8 found that 50% of the cases were located in the tongue, but only one case was found in the dorsum. The dermoid cyst predominantly presents in children or young adults as a soft to dough-like mass that is rare in the tongue and common in the floor of the mouth.9 Lingual choristomas are not unusual in the posterior third of the
Reactive The history of a fishbone trauma 5 months earlier was strongly suggestive of a foreign body granuloma. a Postgraduate Student, Department of Oral Pathology and Surgery, Faculty of Dentistry, National and Kapodistrian University of Athens. b Assistant Professor, Department of Oral Pathology and Surgery, Faculty of Dentistry, National and Kapodistrian University of Athens. c Lecturer, Department of Oral Pathology and Surgery, Faculty of Dentistry, National and Kapodistrian University of Athens; Department of Oral and Maxillofacial Surgery, Evaggelismos General Hospital. d Lecturer, Department of Basic Biomedical Sciences, Faculty of Dentistry, National and Kapodistrian University of Athens. e Professor, Department of Oral Pathology and Surgery, Faculty of Dentistry, National and Kapodistrian University of Athens. Received for publication Jul 17, 2013; returned for revision Sep 11, 2013; accepted for publication Oct 11, 2013. Ó 2014 Elsevier Inc. All rights reserved. 2212-4403/$ - see front matter http://dx.doi.org/10.1016/j.oooo.2013.10.004
659
CLINICOPATHOLOGIC CONFERENCE 660 Kalogirou et al.
Fig. 1. Clinical view of a well-circumscribed hemispherical mass on the midline posterior tongue, anterior to the lingual V (circumvallate papillae).
tongue and most are of the osseous type.10 They typically manifest as well-defined, painless, tumor-like masses covered by normal-appearing mucosa, are hard on palpation, and are more common in young females. Leiomyomatous hamartomas may also develop in the midline of the tongue dorsum, although rarely.11 Finally, a subgemmal neurogenous plaque may have a clinical presentation similar to the case described herein, but it is extremely rare.12 Benign connective tissue neoplasms Benign connective tissue tumors usually present as asymptomatic nodules covered by normal-appearing mucosa. The tongue is the most common site for the granular cell tumor, which has rubbery consistency and affects patients aged between 40 and 60 years, with a predilection for women.5 Clinically, it manifests with a whitish discoloration of the mucosa. The dorsal surface of the tongue is a common site for neurilemmoma (schwannoma), which affects patients of any age; neurofibromas typically arise in young adults.12 Most oral leiomyomas are of the vascular subtype and have a red to blue color, but leiomyomas arising in the posterior tongue are usually of the solid (nonvascular) type and are covered by normalappearing mucosa.11 The tongue is the second most common location for rhabdomyomas, which are usually found in the floor of the mouth of adult males.13 In a study of 125 lipomas of the oral and maxillofacial region, 15 (12%) were localized in the tongue, and most of them were of the classic subtype.14 Lipomas are expected to be compressible tumors with yellow discoloration. The color of the present lesion was also not consistent with a vascular tumor, such as a vascular malformation or a lymphangioma, which are typically seen in children as red to bluish tumors.5,15
OOOO June 2014
Fig. 2. Mucosal fragment infiltrated by cystic and solid nests of neoplastic cells. Most nests are cystic, and the lesion extends to the deep margin of the sample (hematoxylin-eosin, original magnification 25).
Benign salivary neoplasm Minor salivary gland tumors should also be included in the differential diagnosis, although they are extremely rare in the tongue. In a series of 737 cases presented by Wang et al.,16 only 5 cases of adenomas were found in the tongue, and, in striking contrast to other intraoral locations, none of them was a pleomorphic adenoma. On the other hand, lingual salivary gland tumors are more likely to be malignant, with adenoid cystic carcinoma being the most common.16,17 Finally, the tongue is the oral site most commonly involved by amyloidosis, whose localized form, amyloid tumor, may have a presentation similar to that of the lesion described herein.18 In such cases, the medical history may not be contributory, because the patient may not have systemic involvement.
DIAGNOSIS AND MANAGEMENT With the provisional diagnosis of a granular cell tumor, the lesion was excised under local anesthesia. Grossly, it measured 1.0 0.9 0.5 cm, with a solid, gray cut surface. Microscopic examination of 5-mm-thick formalin-fixed and paraffin-embedded tissue sections stained with hematoxylin-eosin revealed a mucosal fragment infiltrated by cystic and solid nests of neoplastic cells that extended to the deep margin of the specimen (Figure 2). The neoplastic cells exhibited phenotypic characteristics of mucous, epidermoid, and intermediate cells. Overall, there was a clear predominance of cystic structures and mucous cells (Figure 3). There was no pronounced anaplasia, mitotic figures were
CLINICOPATHOLOGIC CONFERENCE
An asymptomatic tumor on the dorsal tongue Eleni-Marina Kalogirou, DDS,a Konstantinos I. Tosios, DDS, PhD,b Vassileios Petsinis, MD, DDS, MSc, PhD,c Ioulia Chatzistamou, MD, PhD,d and Alexandra Sklavounou, DDS, MSc, PhDe National and Kapodistrian University of Athens and Evaggelismos General Hospital, Athens, Greece
(Oral Surg Oral Med Oral Pathol Oral Radiol 2014;117:659-662)
CLINICAL PRESENTATION A 42-year-old man was referred by his dentist for diagnosis and management of a painless nodule on the posterior dorsal surface of the tongue. According to the patient, the lesion was present for at least 2 months and developed after a fishbone injury 5 months earlier. An otorhinolaryngology specialist diagnosed it as a “hyperplastic lingual papilla”; an internist, as a “normal structure of the tongue.” It had “slightly shrunk” during the past week and did not cause any discomfort or functional disturbance. The patient’s medical history was noncontributory. Clinical examination revealed a well-circumscribed hemispherical tumor on the midline of the dorsal surface of the tongue, anterior to the lingual V (Figure 1). It measured approximately 0.6 cm, was covered by normal-appearing mucosa, and on palpation was painless, firm, and adherent to the overlying mucosa. The rest of the oral mucosa was within normal limits. No palpable lymph nodes were present.
Among foreign bodies, fish bones are those most commonly implanted in the upper aerodigestive tract, although they usually involve the base of the tongue and only rarely its mobile part.1 In contrast to the lesion in our patient, they are usually tender on palpation. Trauma in children or young adults commonly precedes the development of a mucocele, the most common reactive lesion of the oral soft tissues, but a mucocele typically presents as a translucent and fluctuant mass.2 In a study of 1715 mucoceles,2 none was reported to arise on the dorsal tongue, possibly owing to the scarcity of minor salivary glands in this area.3 Trauma is also associated with the development of pyogenic granuloma, but this tumor is typically erythematous and bleeding. In a recent review,4 only 7% of 1127 cases of pyogenic granuloma were located in the tongue. Finally, an irritation fibroma should be included in the differential diagnosis, because it is the most common intraoral tumor.
DIFFERENTIAL DIAGNOSIS An asymptomatic tumor on the dorsal tongue may represent an inflammatory-reactive, developmental, or neoplastic lesion. The indolent growth and good demarcation were against a diagnosis of malignancy.
Developmental The posterior dorsal tongue is a common site for developmental lesions. Lingual thyroid is usually found in the median posterior tongue, but it is situated between the foramen cecum and the epiglottis.5 The remnants of the thyroglossal duct tract that arises from the foramen cecum may form a thyroglossal duct cyst, which predominantly affects females.6 It usually involves the neck, although in 2% to 3% it may be intralingual and involve the inferior half of the tongue. The midline of the dorsal tongue anterior to the lingual V is the typical site of occurrence of median rhomboid glossitis, which has a striking male predilection.7 Apart from its typical presentation as an atrophic area, median rhomboid glossitis may rarely manifest as an exophytic mass, but it is usually red with a fissured or lobulated surface and hard consistency.7 The oral lymphoepithelial cyst usually affects adults and presents as a soft to firm mass of yellow-white discoloration.8 In a study of 120 intraoral lymphoepithelial cysts, Yang et al.8 found that 50% of the cases were located in the tongue, but only one case was found in the dorsum. The dermoid cyst predominantly presents in children or young adults as a soft to dough-like mass that is rare in the tongue and common in the floor of the mouth.9 Lingual choristomas are not unusual in the posterior third of the
Reactive The history of a fishbone trauma 5 months earlier was strongly suggestive of a foreign body granuloma. a Postgraduate Student, Department of Oral Pathology and Surgery, Faculty of Dentistry, National and Kapodistrian University of Athens. b Assistant Professor, Department of Oral Pathology and Surgery, Faculty of Dentistry, National and Kapodistrian University of Athens. c Lecturer, Department of Oral Pathology and Surgery, Faculty of Dentistry, National and Kapodistrian University of Athens; Department of Oral and Maxillofacial Surgery, Evaggelismos General Hospital. d Lecturer, Department of Basic Biomedical Sciences, Faculty of Dentistry, National and Kapodistrian University of Athens. e Professor, Department of Oral Pathology and Surgery, Faculty of Dentistry, National and Kapodistrian University of Athens. Received for publication Jul 17, 2013; returned for revision Sep 11, 2013; accepted for publication Oct 11, 2013. Ó 2014 Elsevier Inc. All rights reserved. 2212-4403/$ - see front matter http://dx.doi.org/10.1016/j.oooo.2013.10.004
659
CLINICOPATHOLOGIC CONFERENCE 660 Kalogirou et al.
Fig. 1. Clinical view of a well-circumscribed hemispherical mass on the midline posterior tongue, anterior to the lingual V (circumvallate papillae).
tongue and most are of the osseous type.10 They typically manifest as well-defined, painless, tumor-like masses covered by normal-appearing mucosa, are hard on palpation, and are more common in young females. Leiomyomatous hamartomas may also develop in the midline of the tongue dorsum, although rarely.11 Finally, a subgemmal neurogenous plaque may have a clinical presentation similar to the case described herein, but it is extremely rare.12 Benign connective tissue neoplasms Benign connective tissue tumors usually present as asymptomatic nodules covered by normal-appearing mucosa. The tongue is the most common site for the granular cell tumor, which has rubbery consistency and affects patients aged between 40 and 60 years, with a predilection for women.5 Clinically, it manifests with a whitish discoloration of the mucosa. The dorsal surface of the tongue is a common site for neurilemmoma (schwannoma), which affects patients of any age; neurofibromas typically arise in young adults.12 Most oral leiomyomas are of the vascular subtype and have a red to blue color, but leiomyomas arising in the posterior tongue are usually of the solid (nonvascular) type and are covered by normalappearing mucosa.11 The tongue is the second most common location for rhabdomyomas, which are usually found in the floor of the mouth of adult males.13 In a study of 125 lipomas of the oral and maxillofacial region, 15 (12%) were localized in the tongue, and most of them were of the classic subtype.14 Lipomas are expected to be compressible tumors with yellow discoloration. The color of the present lesion was also not consistent with a vascular tumor, such as a vascular malformation or a lymphangioma, which are typically seen in children as red to bluish tumors.5,15
OOOO June 2014
Fig. 2. Mucosal fragment infiltrated by cystic and solid nests of neoplastic cells. Most nests are cystic, and the lesion extends to the deep margin of the sample (hematoxylin-eosin, original magnification 25).
Benign salivary neoplasm Minor salivary gland tumors should also be included in the differential diagnosis, although they are extremely rare in the tongue. In a series of 737 cases presented by Wang et al.,16 only 5 cases of adenomas were found in the tongue, and, in striking contrast to other intraoral locations, none of them was a pleomorphic adenoma. On the other hand, lingual salivary gland tumors are more likely to be malignant, with adenoid cystic carcinoma being the most common.16,17 Finally, the tongue is the oral site most commonly involved by amyloidosis, whose localized form, amyloid tumor, may have a presentation similar to that of the lesion described herein.18 In such cases, the medical history may not be contributory, because the patient may not have systemic involvement.
DIAGNOSIS AND MANAGEMENT With the provisional diagnosis of a granular cell tumor, the lesion was excised under local anesthesia. Grossly, it measured 1.0 0.9 0.5 cm, with a solid, gray cut surface. Microscopic examination of 5-mm-thick formalin-fixed and paraffin-embedded tissue sections stained with hematoxylin-eosin revealed a mucosal fragment infiltrated by cystic and solid nests of neoplastic cells that extended to the deep margin of the specimen (Figure 2). The neoplastic cells exhibited phenotypic characteristics of mucous, epidermoid, and intermediate cells. Overall, there was a clear predominance of cystic structures and mucous cells (Figure 3). There was no pronounced anaplasia, mitotic figures were
