Yi-Chun Carol Liu1,2, Michael Shih3, M John Hicks4,5, Matthew S Sitton1,2. 1. Division of Pediatric Otolaryngology, Texas Children's Hospital, Houston, Texas, U.S.A. 2. Department of Otolaryngology-Head and Neck Surgery, Baylor College of Medicine, Houston, Texas, U.S.A. 3. Baylor College of Medicine, Houston, Texas, U.S.A. 4. Department of Pathology, Texas Children's Hospital, Houston, Texas, U.S.A. 5. Department of Pathology and Immunology, Baylor College of Medicine, Houston, Texas, U.S.A.
Abstract
OBJECTIVES/HYPOTHESIS: To further clinically characterize lingual hamartomas, and to consolidate literature by analyzing clinical presentation, diagnostic evaluation, treatment, and outcomes. STUDY DESIGN: Retrospective chart review and literature review. METHODS: Case series from 1989 to 2020 at a tertiary pediatric center; follow-up ranging from 2 months to 12 years. Patient demographics, clinical presentations, physical examination findings, surgical pathology, operative methods, pre-operative imaging, and follow-up. A review of English-language literature from 1945 to 2020 was conducted. RESULTS: Seven patients (four male, three female) with nine lingual hamartomas were identified. Average age at surgical excision was 7.9 months (SD 3.5 months, range 3 to 14 months). Follow-up ranged from 2 months to 12 years, with all patients having no recurrence. Physical examination identified five pedunculated hamartomas and two sessile hamartomas. One patient had a neck CT, one had neck ultrasound, and two had facial MRIs. CT and ultrasounds confirmed presence of cervical thyroid. MRI suggested no muscular invasion for one patient, whereas motion artifact obscured findings for the other patient. Most patients who presented with dysphagia or poor weight gain achieved post-surgical improvement. The present cases combined with the literature review identified 57 patients with 63 lingual hamartomas. CONCLUSIONS: Lingual hamartomas appear as hypovascular pedunculated masses near the foramen cecum. There is no demographic predisposition, and most are identified during infancy. Imaging is not necessary, unless there is concern for lingual thyroid, in which case thyroid ultrasound should be performed. Surgical excision is curative, with dysphagia resolution and low likelihood for recurrence. LEVEL OF EVIDENCE: 4 Laryngoscope, 131:E2080-E2088, 2021.
OBJECTIVES/HYPOTHESIS: To further clinically characterize lingual hamartomas, and to consolidate literature by analyzing clinical presentation, diagnostic evaluation, treatment, and outcomes. STUDY DESIGN: Retrospective chart review and literature review. METHODS: Case series from 1989 to 2020 at a tertiary pediatric center; follow-up ranging from 2 months to 12 years. Patient demographics, clinical presentations, physical examination findings, surgical pathology, operative methods, pre-operative imaging, and follow-up. A review of English-language literature from 1945 to 2020 was conducted. RESULTS: Seven patients (four male, three female) with nine lingual hamartomas were identified. Average age at surgical excision was 7.9 months (SD 3.5 months, range 3 to 14 months). Follow-up ranged from 2 months to 12 years, with all patients having no recurrence. Physical examination identified five pedunculated hamartomas and two sessile hamartomas. One patient had a neck CT, one had neck ultrasound, and two had facial MRIs. CT and ultrasounds confirmed presence of cervical thyroid. MRI suggested no muscular invasion for one patient, whereas motion artifact obscured findings for the other patient. Most patients who presented with dysphagia or poor weight gain achieved post-surgical improvement. The present cases combined with the literature review identified 57 patients with 63 lingual hamartomas. CONCLUSIONS:Lingual hamartomas appear as hypovascular pedunculated masses near the foramen cecum. There is no demographic predisposition, and most are identified during infancy. Imaging is not necessary, unless there is concern for lingual thyroid, in which case thyroid ultrasound should be performed. Surgical excision is curative, with dysphagia resolution and low likelihood for recurrence. LEVEL OF EVIDENCE: 4 Laryngoscope, 131:E2080-E2088, 2021.