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Lingual hamartoma in a newborn with chromosome 2q terminal deletion
International Journal of Pediatric Otorhinolaryngology Extra (2008) 3, 109—112
www.elsevier.com/locate/ijporl
CASE REPORT
Lingual hamartoma in a newborn with chromosome 2q terminal deletion Nobuhiko Seki a,*, Tomoko Shintani a, Hiroshi Tsubota a, Kazumasa Watanabe a, Noriko Ogasawara a, Yu-uki Ueda b, Tetsuo Himi a a b
Department of Otolaryngology, Sapporo Medical University, Japan Kitami Red Cross Hospital, Japan
Received 18 October 2007; accepted 4 December 2007 Available online 16 January 2008
KEYWORDS Lingual hamartoma; Chromosome 2q terminal deletion
Summary Hamartoma, defined as tumour-like overgrowths of tissue, may occur in any organ in the body and is commonly seen in liver, spleen, kidney and lung, however, lingual hamartoma is a rare entity. Terminal deletion of chromosome 2q is a chromosomal abnormality causing various craniofacial malformations, and no reports can be obtained about patients who have chromosomal disorder presenting with lingual hamartoma. We describe the first case of lingual hamartoma in a patient with terminal deletion of chromosome 2, and summarize the clinical management and the histogenesis of lingual hamartoma from the literature. A 9-month-old girl who had chromosome 2q terminal deletion consulted Sapporo Medical University hospital for evaluation of upper airway obstruction. She had retractive breathing soon after birth and required treatment with continuous positive airway pressure (CPAP). Physical examination showed craniofacial abnormalities, such as round face, high palate, aural atresia, anotia and so on. Fiberoptic examination and CT scan revealed a mass on the right side of the tongue base. She underwent surgical resection and the tumour was pathologically diagnosed as hamartoma. After surgery, her respiratory condition improved to the extent where CPAP was withdrawn. There has been no tumour recurrence during the 1-year follow-up period. # 2007 Elsevier Ireland Ltd. All rights reserved.
1. Introduction In 1904, hamartoma was first reported and defined as a focal, tumour-like congenital malformation in which one or several normal tissue components of * Corresponding author at: Department of Otolaryngology, Sapporo Medical University, S1 W16, Chuo-ku, Sapporo 060-8543, Japan. Tel.: +81 11 611 2111x3491; fax: +81 11 615 5405. E-mail address: [email protected] (N. Seki).
a organ are abnormally arranged [1], possibly occurring in any organ of the body. In the head and neck region, several cutaneous involvements such as pigmented nevus or port-wine stain are relatively common lesions considered as hamartomatous, however, hamartomas in other areas of the head and neck are uncommon [2]. Terminal deletion of chromosome 2q is a chromosomal abnormality causing various craniofacial malformations, but no reports can be obtained about chromosomal disorders in association
1871-4048/$ — see front matter # 2007 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.pedex.2007.12.002
110
N. Seki et al.
with lingual hamartoma. We report the first case of lingual hamartoma, in a newborn, with chromosome 2q terminal deletion.
2. Case report A 9-month-old girl was referred to Sapporo Medical University hospital for evaluation of respiratory disturbance, dysphagia and hearing loss. She was born after a normal term pregnancy and vaginal delivery but retractive breathing was detected immediately after birth. There was no known family history of birth defects. Physical examination was remarkable for dysmorphic features including round face, prominent forehead, depressed nasal bridge, micrognathia, high palate, aural atresia and anotia. Chromosome analysis with G-banding revealed chromosome 2q terminal deletion (46XX del (2)(q37.1)). She required treatment with continuous positive airway pressure (CPAP) and tubal feeding. Flexible endoscopic examination confirmed a mass on the right side of the lingual base (Fig. 1). The surface was almost the same as the tongue, so it looked just like a ‘‘lobulated tongue’’. Nonenhanced computed tomographic (CT) scan showed a homogenous soft tissue density mass measuring 10 mm 10 mm 15 mm in size (Fig. 2). Normally detected thyroid on the CT excluded ectopic lingual thyroid. The remainder of the examination revealed no other lesions. The lingual tumour was surgically resected with YAG laser under general anesthesia. We had no difficulty in inserting an endotracheal tube and there was little intraoperative bleeding. Postoperative course was uneventful. The surgically resected specimen was a soft mass measuring 11 mm 15 mm, the macroscopic appearance of which is shown in Fig. 3. Histopathology (Fig. 4) showed a tumour-like mass covered with stratified
Fig. 2 Axial non-enhanced CT scan shows a homogenous soft tissue mass (arrows, 10 mm 10 mm 15 mm in size) arising from base of the tongue.
squamous epithelium and composed of randomly oriented striated muscle fibers and minor salivary glands in a connective stroma, and the tumour was diagnosed as hamartoma of the tongue. Now she has no difficulty in swallowing and CPAP is not required except when the respiratory status is extremely worsened. There has been no tumour recurrence during the 1-year follow-up period.
3. Discussion Hamartoma is a tumour-like malformation, which is commonly seen in liver, spleen, kidney and lung, and the occurrence on tongue is rare. As far as we know, 25 case reports of lingual hamartoma [2—24], sum-
Fig. 1 Endoscopic appearance of lingual hamartoma. PW, posterior wall of pharynx; SP, soft palate; Tm, tumour; arrow, feeding tube.
Lingual hamartoma in a newborn
Fig. 3
111
Gross appearance of the resected tumour.
marized in Table 1, have been published in the English literature since Stamm et al. reported the first patient in 1945. The patients comprised 7 males and 18 females, and the ages at presentation ranged from birth to 60 years (median, 20 months; mean 9.51 years). The commonest site was the base of the tongue (15 cases). The most important symptom of lingual hamartoma was dysphagia, however symptoms were often absent (15 cases, 60%).
Fig. 4 Histopathology of the lingual hamartoma (hematoxylin-eosin stain, scale bar = 20 mm).
The histogenesis of hamartoma has yet to be revealed, but the etiology on the base of tongue can be explained as follows [3,4,6,7,12]: in the developmental processes of the tongue, the anterior two thirds of tongue and the posterior third are formed separately, and they fuse together making a V-shaped structure (terminal sulcus). Overproduc-
Table 1 Summary of the reported cases of lingual hamartoma Year
Author
Sex
Age at presentation
Site
Symptoms
1945 1956 1963 1968
Stamm Perri Hinshaw Ishii
F F F F
Birth 34 years 4 years 4 months
Dysphagia Globus None None
1981 1984 1985 1985
Demuth Becker Cutchavaree Takato
1986 1989 1991 1992 1995 1995 1998 1999 2001 2001 2001
Herzog Takimoto Miyamoto Owen Yoshihara Goldsmith Ide de la Rosa-Garcia Halfpenny Kobayashi Wallace
M M F F F F F F F F M F M F M F
2 months Birth 24 years 7 years 2 years 6 weeks 6 years 21 months 9 months 8 months 16 months 60 years 6 years 4 years 3 months 41 years
Base Base Base Base and anterior tongue (4 masses) Base Base Mid-dorsum Base Base Base Base Bifurcation Base Base Base Lateral border Tip of tongue Anterior tongue Dorsum Base
2003 2004 2006 2006 2007
Gillett Steele Ameh Noguchi Seki
F M M F F
32 years 12 years 5 months Birth Birth
Postero-lateral border Lateral border Dorsum Dorsum Base
Dysphagia Dysphagia Dysphagia Dysphagia None None None None None Dysphagia Choking on swallowing None None None None Stridor, voice change, sleep apnea None None None None Respiratory distress
112
N. Seki et al.
tion of tissue can easily occur around the terminal sulcus, which is the juncture of several embryologic processes, resulting in formation of a hamartoma. Craniofacial malformations like cleft palate are often seen in patients with lingual hamartoma [6,7,13,24,25], so occurrence of these abnormalities may be associated with formation of lingual hamartoma. To our knowledge, this is the first case of lingual hamartoma in a patient with terminal deletion of chromosome 2. It was reported that patients with 2q terminal deletion often presented various craniofacial malformations, for example, prominent forehead, depressed nasal bridge, dysmorphic ears and nose, cleft palate and so on [26]. As stated above, our patient had characteristic craniofacial and integumentary features as well, leading to morphological narrowing of the pharynx. The respiratory disturbance immediately after birth was probably caused by upper airway obstruction owing to the congenital pharyngeal narrowing as well as the lingual tumour. This is why CPAP is still necessary even after surgery. The differential diagnosis of a solid lingual mass in children includes lingual thyroid, hamartoma, neurofibroma, fibromatosis, rhabdomyoma, rhabdomyosarcoma and granular cell myoblastoma [23,25,27]. Preoperative diagnosis is usually difficult because they can only be distinguished by histopathology. Complete excision with a small surgical margin is the treatment of choice. To date, there have been no reports of recurrent cases or malignant change in lingual hamartoma after resection, so the prognosis is good in general.
References [1] H. Albrecht, Ueber Hamartome. Verh Deutsch Ges. Path. (1904) 153—157. [2] G. Owen, J. Berry, P. Bicknell, Hamartoma of the tongue, J. Laryngol. Otol. 107 (1993) 363—367. [3] C. Stamm, R. Tauber, Hamartoma of the tongue, Laryngoscope 55 (1945) 140—146. [4] F.A. Perri, Myoepithelial hamartoma of tongue, AMA Arch. Otolaryngol. 64 (1956) 289—290. [5] C.T. Hinshaw Jr., Unusual lesions of the tongue: hamartoma, J. Kans. Med. Soc. 64 (1963) 154—157. [6] T. Ishii, S. Takemori, J.I. Suzuki, Hamartoma of the tongue. Report of a case, Arch. Otolaryngol. 88 (1968) 171—173. [7] R.J. Demuth, D.F. Johns, Recurrent aspiration pneumonitis in a cleft palate child with hamartoma of the tongue, Cleft Palate J. 18 (1981) 299—303. [8] G. Becker, R. Ridolfi, C. Ingber, Lingual hamartoma in a newborn., Otolaryngol. Head Neck Surg. 92 (1984) 357—359.
[9] A. Cutchavaree, S. Rungruxsirivorn, S. Suphanakorn, Hamartoma of the tongue, J. Med. Assoc. Thai. 68 (1985) 216—219. [10] T. Takato, O. Fukuda, Y. Ohhara, A. Yanai, S. Hirabayashi, T. Nakatsuka, Hamartoma of the tongue: two case reports, Plast. Reconstr. Surg. 75 (1985) 258—262. [11] S. Herzog, J. Bressman, J.A. Giglio, Case 61: tongue mass in an infant, J. Oral Maxillofac. Surg. 44 (1986) 463—466. [12] T. Takimoto, T. Yoshizaki, R. Umeda, Hamartoma of the tongue, Int. J. Pediatr. Otorhinolaryngol. 18 (1989) 157— 161. [13] Y. Miyamoto, M. Nagayama, Y. Hayashi, A cleft palate child with lobulated tongue and lingual hamartoma: report of a case, J. Oral Maxillofac. Surg. 49 (1991) 644—646. [14] P. Goldsmith, J.V. Soames, D. Meikle, Leiomyomatous hamartoma of the posterior tongue: a case report, J. Laryngol. Otol. 109 (1995) 1190—1191. [15] T. Yoshihara, A. Oda, Y. Takahashi, T. Ishii, Y. Yaku, Fibrous hamartoma of the tongue: report of a case with immunohistochemical and ultrastructural studies, Int. J. Pediatr. Otorhinolaryngol. 33 (1995) 171—178. [16] F. Ide, T. Shimoyama, N. Horie, Angiomyolipomatous hamartoma of the tongue, Oral Surg. Oral Med. Oral Pathol. Oral Radiol. Endod. 85 (1998) 581—584. [17] E. de la Rosa-Garcia, A. Mosqueda-Taylor, Leiomyomatous hamartoma of the anterior tongue: report of a case and review of the literature, Int. J. Paediatr. Dent. 9 (1999) 129— 132. [18] W. Halfpenny, E.W. Odell, P.D. Robinson, Cystic and glial mixed hamartoma of the tongue, J. Oral Pathol. Med. 30 (2001) 368—371. [19] A. Kobayashi, T. Amagasa, N. Okada, Leiomyomatous hamartoma of the tongue: case report, J. Oral Maxillofac. Surg. 59 (2001) 337—340. [20] H. Wallace, A. Davis, A. Spedding, Tongue-base hamartoma in tuberous sclerosis, J. Laryngol. Otol. 115 (2001) 149—150. [21] D. Gillett, F. Fahmy, J.W. Eveson, J.C. Shotton, Intramuscular capillary hamartoma of the tongue, J. Laryngol. Otol. 117 (2003) 734—735. [22] J.C. Steele, A. Triantafyllou, E.A. Field, Lingual striated muscle hamartoma or herniation? J. Oral Pathol. Med. 33 (2004) 454—455. [23] E.A. Ameh, P.M. Mshelbwala, A.H. Rafindadi, Lingual harmatoma in an infant, Ann. Trop. Paediatr. 26 (2006) 137— 139. [24] T. Noguchi, Y. Jinbu, H. Itoh, K. Matsumoto, O. Sakai, M. Kusama, Epignathus combined with cleft palate, lobulated tongue, and lingual hamartoma: report of a case, Oral Surg. Oral Med. Oral Pathol. Oral Radiol. Endod. 101 (2006) 481— 486. [25] R. Hanna, Z.B. Argenyi, J.A. Benda, Hamartoma of the tongue in an infant with a primary diagnosis of ectrodactyly-ectodermal dysplasia-cleft lip and palate syndrome, J. Cutan. Pathol. 21 (1994) 173—178. [26] K.A. Casas, T.K. Mononen, C.N. Mikail, S.J. Hassed, S. Li, J.J. Mulvihill, et al., Chromosome 2q terminal deletion: report of 6 new patients and review of phenotype-breakpoint correlations in 66 individuals, Am. J. Med. Genet. A 130 (2004) 331—339. [27] F.T. Velcek, D.H. Klotz, C.H. Hill, L.E. Ladogana, P.K. Kottmeier, Tongue lesions in children, J. Pediatr. Surg. 14 (1979) 238—246.
Available online at www.sciencedirect.com
www.elsevier.com/locate/ijporl
CASE REPORT
Lingual hamartoma in a newborn with chromosome 2q terminal deletion Nobuhiko Seki a,*, Tomoko Shintani a, Hiroshi Tsubota a, Kazumasa Watanabe a, Noriko Ogasawara a, Yu-uki Ueda b, Tetsuo Himi a a b
Department of Otolaryngology, Sapporo Medical University, Japan Kitami Red Cross Hospital, Japan
Received 18 October 2007; accepted 4 December 2007 Available online 16 January 2008
KEYWORDS Lingual hamartoma; Chromosome 2q terminal deletion
Summary Hamartoma, defined as tumour-like overgrowths of tissue, may occur in any organ in the body and is commonly seen in liver, spleen, kidney and lung, however, lingual hamartoma is a rare entity. Terminal deletion of chromosome 2q is a chromosomal abnormality causing various craniofacial malformations, and no reports can be obtained about patients who have chromosomal disorder presenting with lingual hamartoma. We describe the first case of lingual hamartoma in a patient with terminal deletion of chromosome 2, and summarize the clinical management and the histogenesis of lingual hamartoma from the literature. A 9-month-old girl who had chromosome 2q terminal deletion consulted Sapporo Medical University hospital for evaluation of upper airway obstruction. She had retractive breathing soon after birth and required treatment with continuous positive airway pressure (CPAP). Physical examination showed craniofacial abnormalities, such as round face, high palate, aural atresia, anotia and so on. Fiberoptic examination and CT scan revealed a mass on the right side of the tongue base. She underwent surgical resection and the tumour was pathologically diagnosed as hamartoma. After surgery, her respiratory condition improved to the extent where CPAP was withdrawn. There has been no tumour recurrence during the 1-year follow-up period. # 2007 Elsevier Ireland Ltd. All rights reserved.
1. Introduction In 1904, hamartoma was first reported and defined as a focal, tumour-like congenital malformation in which one or several normal tissue components of * Corresponding author at: Department of Otolaryngology, Sapporo Medical University, S1 W16, Chuo-ku, Sapporo 060-8543, Japan. Tel.: +81 11 611 2111x3491; fax: +81 11 615 5405. E-mail address: [email protected] (N. Seki).
a organ are abnormally arranged [1], possibly occurring in any organ of the body. In the head and neck region, several cutaneous involvements such as pigmented nevus or port-wine stain are relatively common lesions considered as hamartomatous, however, hamartomas in other areas of the head and neck are uncommon [2]. Terminal deletion of chromosome 2q is a chromosomal abnormality causing various craniofacial malformations, but no reports can be obtained about chromosomal disorders in association
1871-4048/$ — see front matter # 2007 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.pedex.2007.12.002
110
N. Seki et al.
with lingual hamartoma. We report the first case of lingual hamartoma, in a newborn, with chromosome 2q terminal deletion.
2. Case report A 9-month-old girl was referred to Sapporo Medical University hospital for evaluation of respiratory disturbance, dysphagia and hearing loss. She was born after a normal term pregnancy and vaginal delivery but retractive breathing was detected immediately after birth. There was no known family history of birth defects. Physical examination was remarkable for dysmorphic features including round face, prominent forehead, depressed nasal bridge, micrognathia, high palate, aural atresia and anotia. Chromosome analysis with G-banding revealed chromosome 2q terminal deletion (46XX del (2)(q37.1)). She required treatment with continuous positive airway pressure (CPAP) and tubal feeding. Flexible endoscopic examination confirmed a mass on the right side of the lingual base (Fig. 1). The surface was almost the same as the tongue, so it looked just like a ‘‘lobulated tongue’’. Nonenhanced computed tomographic (CT) scan showed a homogenous soft tissue density mass measuring 10 mm 10 mm 15 mm in size (Fig. 2). Normally detected thyroid on the CT excluded ectopic lingual thyroid. The remainder of the examination revealed no other lesions. The lingual tumour was surgically resected with YAG laser under general anesthesia. We had no difficulty in inserting an endotracheal tube and there was little intraoperative bleeding. Postoperative course was uneventful. The surgically resected specimen was a soft mass measuring 11 mm 15 mm, the macroscopic appearance of which is shown in Fig. 3. Histopathology (Fig. 4) showed a tumour-like mass covered with stratified
Fig. 2 Axial non-enhanced CT scan shows a homogenous soft tissue mass (arrows, 10 mm 10 mm 15 mm in size) arising from base of the tongue.
squamous epithelium and composed of randomly oriented striated muscle fibers and minor salivary glands in a connective stroma, and the tumour was diagnosed as hamartoma of the tongue. Now she has no difficulty in swallowing and CPAP is not required except when the respiratory status is extremely worsened. There has been no tumour recurrence during the 1-year follow-up period.
3. Discussion Hamartoma is a tumour-like malformation, which is commonly seen in liver, spleen, kidney and lung, and the occurrence on tongue is rare. As far as we know, 25 case reports of lingual hamartoma [2—24], sum-
Fig. 1 Endoscopic appearance of lingual hamartoma. PW, posterior wall of pharynx; SP, soft palate; Tm, tumour; arrow, feeding tube.
Lingual hamartoma in a newborn
Fig. 3
111
Gross appearance of the resected tumour.
marized in Table 1, have been published in the English literature since Stamm et al. reported the first patient in 1945. The patients comprised 7 males and 18 females, and the ages at presentation ranged from birth to 60 years (median, 20 months; mean 9.51 years). The commonest site was the base of the tongue (15 cases). The most important symptom of lingual hamartoma was dysphagia, however symptoms were often absent (15 cases, 60%).
Fig. 4 Histopathology of the lingual hamartoma (hematoxylin-eosin stain, scale bar = 20 mm).
The histogenesis of hamartoma has yet to be revealed, but the etiology on the base of tongue can be explained as follows [3,4,6,7,12]: in the developmental processes of the tongue, the anterior two thirds of tongue and the posterior third are formed separately, and they fuse together making a V-shaped structure (terminal sulcus). Overproduc-
Table 1 Summary of the reported cases of lingual hamartoma Year
Author
Sex
Age at presentation
Site
Symptoms
1945 1956 1963 1968
Stamm Perri Hinshaw Ishii
F F F F
Birth 34 years 4 years 4 months
Dysphagia Globus None None
1981 1984 1985 1985
Demuth Becker Cutchavaree Takato
1986 1989 1991 1992 1995 1995 1998 1999 2001 2001 2001
Herzog Takimoto Miyamoto Owen Yoshihara Goldsmith Ide de la Rosa-Garcia Halfpenny Kobayashi Wallace
M M F F F F F F F F M F M F M F
2 months Birth 24 years 7 years 2 years 6 weeks 6 years 21 months 9 months 8 months 16 months 60 years 6 years 4 years 3 months 41 years
Base Base Base Base and anterior tongue (4 masses) Base Base Mid-dorsum Base Base Base Base Bifurcation Base Base Base Lateral border Tip of tongue Anterior tongue Dorsum Base
2003 2004 2006 2006 2007
Gillett Steele Ameh Noguchi Seki
F M M F F
32 years 12 years 5 months Birth Birth
Postero-lateral border Lateral border Dorsum Dorsum Base
Dysphagia Dysphagia Dysphagia Dysphagia None None None None None Dysphagia Choking on swallowing None None None None Stridor, voice change, sleep apnea None None None None Respiratory distress
112
N. Seki et al.
tion of tissue can easily occur around the terminal sulcus, which is the juncture of several embryologic processes, resulting in formation of a hamartoma. Craniofacial malformations like cleft palate are often seen in patients with lingual hamartoma [6,7,13,24,25], so occurrence of these abnormalities may be associated with formation of lingual hamartoma. To our knowledge, this is the first case of lingual hamartoma in a patient with terminal deletion of chromosome 2. It was reported that patients with 2q terminal deletion often presented various craniofacial malformations, for example, prominent forehead, depressed nasal bridge, dysmorphic ears and nose, cleft palate and so on [26]. As stated above, our patient had characteristic craniofacial and integumentary features as well, leading to morphological narrowing of the pharynx. The respiratory disturbance immediately after birth was probably caused by upper airway obstruction owing to the congenital pharyngeal narrowing as well as the lingual tumour. This is why CPAP is still necessary even after surgery. The differential diagnosis of a solid lingual mass in children includes lingual thyroid, hamartoma, neurofibroma, fibromatosis, rhabdomyoma, rhabdomyosarcoma and granular cell myoblastoma [23,25,27]. Preoperative diagnosis is usually difficult because they can only be distinguished by histopathology. Complete excision with a small surgical margin is the treatment of choice. To date, there have been no reports of recurrent cases or malignant change in lingual hamartoma after resection, so the prognosis is good in general.
References [1] H. Albrecht, Ueber Hamartome. Verh Deutsch Ges. Path. (1904) 153—157. [2] G. Owen, J. Berry, P. Bicknell, Hamartoma of the tongue, J. Laryngol. Otol. 107 (1993) 363—367. [3] C. Stamm, R. Tauber, Hamartoma of the tongue, Laryngoscope 55 (1945) 140—146. [4] F.A. Perri, Myoepithelial hamartoma of tongue, AMA Arch. Otolaryngol. 64 (1956) 289—290. [5] C.T. Hinshaw Jr., Unusual lesions of the tongue: hamartoma, J. Kans. Med. Soc. 64 (1963) 154—157. [6] T. Ishii, S. Takemori, J.I. Suzuki, Hamartoma of the tongue. Report of a case, Arch. Otolaryngol. 88 (1968) 171—173. [7] R.J. Demuth, D.F. Johns, Recurrent aspiration pneumonitis in a cleft palate child with hamartoma of the tongue, Cleft Palate J. 18 (1981) 299—303. [8] G. Becker, R. Ridolfi, C. Ingber, Lingual hamartoma in a newborn., Otolaryngol. Head Neck Surg. 92 (1984) 357—359.
[9] A. Cutchavaree, S. Rungruxsirivorn, S. Suphanakorn, Hamartoma of the tongue, J. Med. Assoc. Thai. 68 (1985) 216—219. [10] T. Takato, O. Fukuda, Y. Ohhara, A. Yanai, S. Hirabayashi, T. Nakatsuka, Hamartoma of the tongue: two case reports, Plast. Reconstr. Surg. 75 (1985) 258—262. [11] S. Herzog, J. Bressman, J.A. Giglio, Case 61: tongue mass in an infant, J. Oral Maxillofac. Surg. 44 (1986) 463—466. [12] T. Takimoto, T. Yoshizaki, R. Umeda, Hamartoma of the tongue, Int. J. Pediatr. Otorhinolaryngol. 18 (1989) 157— 161. [13] Y. Miyamoto, M. Nagayama, Y. Hayashi, A cleft palate child with lobulated tongue and lingual hamartoma: report of a case, J. Oral Maxillofac. Surg. 49 (1991) 644—646. [14] P. Goldsmith, J.V. Soames, D. Meikle, Leiomyomatous hamartoma of the posterior tongue: a case report, J. Laryngol. Otol. 109 (1995) 1190—1191. [15] T. Yoshihara, A. Oda, Y. Takahashi, T. Ishii, Y. Yaku, Fibrous hamartoma of the tongue: report of a case with immunohistochemical and ultrastructural studies, Int. J. Pediatr. Otorhinolaryngol. 33 (1995) 171—178. [16] F. Ide, T. Shimoyama, N. Horie, Angiomyolipomatous hamartoma of the tongue, Oral Surg. Oral Med. Oral Pathol. Oral Radiol. Endod. 85 (1998) 581—584. [17] E. de la Rosa-Garcia, A. Mosqueda-Taylor, Leiomyomatous hamartoma of the anterior tongue: report of a case and review of the literature, Int. J. Paediatr. Dent. 9 (1999) 129— 132. [18] W. Halfpenny, E.W. Odell, P.D. Robinson, Cystic and glial mixed hamartoma of the tongue, J. Oral Pathol. Med. 30 (2001) 368—371. [19] A. Kobayashi, T. Amagasa, N. Okada, Leiomyomatous hamartoma of the tongue: case report, J. Oral Maxillofac. Surg. 59 (2001) 337—340. [20] H. Wallace, A. Davis, A. Spedding, Tongue-base hamartoma in tuberous sclerosis, J. Laryngol. Otol. 115 (2001) 149—150. [21] D. Gillett, F. Fahmy, J.W. Eveson, J.C. Shotton, Intramuscular capillary hamartoma of the tongue, J. Laryngol. Otol. 117 (2003) 734—735. [22] J.C. Steele, A. Triantafyllou, E.A. Field, Lingual striated muscle hamartoma or herniation? J. Oral Pathol. Med. 33 (2004) 454—455. [23] E.A. Ameh, P.M. Mshelbwala, A.H. Rafindadi, Lingual harmatoma in an infant, Ann. Trop. Paediatr. 26 (2006) 137— 139. [24] T. Noguchi, Y. Jinbu, H. Itoh, K. Matsumoto, O. Sakai, M. Kusama, Epignathus combined with cleft palate, lobulated tongue, and lingual hamartoma: report of a case, Oral Surg. Oral Med. Oral Pathol. Oral Radiol. Endod. 101 (2006) 481— 486. [25] R. Hanna, Z.B. Argenyi, J.A. Benda, Hamartoma of the tongue in an infant with a primary diagnosis of ectrodactyly-ectodermal dysplasia-cleft lip and palate syndrome, J. Cutan. Pathol. 21 (1994) 173—178. [26] K.A. Casas, T.K. Mononen, C.N. Mikail, S.J. Hassed, S. Li, J.J. Mulvihill, et al., Chromosome 2q terminal deletion: report of 6 new patients and review of phenotype-breakpoint correlations in 66 individuals, Am. J. Med. Genet. A 130 (2004) 331—339. [27] F.T. Velcek, D.H. Klotz, C.H. Hill, L.E. Ladogana, P.K. Kottmeier, Tongue lesions in children, J. Pediatr. Surg. 14 (1979) 238—246.
Available online at www.sciencedirect.com