Leiomyomatous Hamartomas of the Oral Cavity: Clinicopathological and Immunohistochemical Features of 4 Cases and Literature Review

Sánchez‐Romero, Celeste; Bonan, Paulo Rogério Ferreti; Pires, Fábio Ramôa; Silva-Júnior, Geraldo Oliveira; Azañero, Wilson Delgado; Almeida, Oslei Paes de; Perez, Danyel Elias da Cruz

doi:10.1177/1066896919843337

Cited by 6 publications

(14 citation statements)

References 28 publications

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“…Four patients were at least 4 years of age, whereas one patient was 5 months old. 3,14,19 These cases similarly did not report lesion recurrence.…”

Section: Discussionmentioning

confidence: 60%

“…More recently, Sanchez‐Romero et al reported an additional four cases of oral leiomyomatous hamartomas with literature review through March 2019. Their work characterized 42 oral hamartomas by location, size, and morphology; however, diagnostic workup, treatment, and management recommendations remained vague 3 . Case reports have reported a variety diagnostic evaluations of lingual hamartomas, including magnetic resonance imaging (MRI), computed tomography (CT), neck ultrasonography, and thyroid uptake scans.…”

Section: Introductionmentioning

confidence: 99%

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Lingual Hamartomas: Clinical Characteristics, Diagnostic Evaluation, Treatment, and Outcomes

et al. 2020

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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, preoperative 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.

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“…Four patients were at least 4 years of age, whereas one patient was 5 months old. 3,14,19 These cases similarly did not report lesion recurrence.…”

Section: Discussionmentioning

confidence: 60%

Section: Introductionmentioning

confidence: 99%

Lingual Hamartomas: Clinical Characteristics, Diagnostic Evaluation, Treatment, and Outcomes

et al. 2020

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“…Per our literature review to date, there are a total of 28 cases of lingual leiomyomatous hamartomas, or smooth muscle-predominant hamartomas, reported in the literature, with 19 of them being on the dorsum of the tongue, as in our present case ( Table 1 ) [ 5 – 25 ]. Other locations of these hamartomatous lesions in the oral cavity include the anterior maxilla and the hard palate [ 24 , 27 ].…”

Section: Discussionmentioning

confidence: 99%

A Case of an Infantile Lingual Leiomyomatous Hamartoma

Gao

Schafer

Althubaiti

et al. 2022

Case Reports in Otolaryngology

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Lingual leiomyomatous hamartomas are rare lesions of the tongue with largely unknown mechanisms of formation. These lesions are often asymptomatic, though they may present with symptoms, particularly relating to swallow function. Workup should include imaging of the head and neck, and diagnosis should be made histologically. Treatment is surgical excision. This case is a report of a 4-week-old female who presented for evaluation of an asymptomatic 1 × 1 cm dorsal midline tongue mass discovered at birth. The patient was monitored until the age of 9 months, at which time the mass was surgically excised. The patient had an uncomplicated postoperative course. Pathological analysis yielded a diagnosis of leiomyomatous hamartoma.

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“…Most of the oral hamartomas occurred at the tongue [33]. At least 50% leiomyomatous hamartoma were known to be present at birth and the most common age of diagnosis was under 12months [34]. The mass of oral hemartoma mostly are asymptomatic.…”

Section: Hamartomamentioning

confidence: 99%

Clinical Features and Management of Oral Nonodontogenic Masses in Children

Zhang¹,

Zhou²,

Shen³

2020

Preprint

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Background: There are numerous clinical reports of oral tumors in children. However, the clinical features and management of oral nonodontogenic masses in children were rare reported. The aim of this article is to present a large series of oral nonodontogenic masses in children, analyzing the clinical characteristics of such masses and reviewing the relevant procedures for treatment.Methods: We conducted an observational retrospective study, reviewing medical records of 171 patients who were treated for oral nonodontogenic masses between 2014 and 2019 at the Department of Pediatric Surgery, Children’s Hospital of Nanjing Medical University. Data collected included age, gender, site, pathologic diagnosis and treatment strategy.Results: All patients were hospitalized in our department. Of the 171 cases, all of them were benign, however, only 1 case diagnosed as inflammatory myofibroblastic tumor showed a malignant process. The most frequent type was hemangioma (63.7%), followed by lymphangioma (16.4%), ranula (7.6%). The most common location of oral masses in the buccal mucosa. The second common location was in tongue. 16 cases of hemangioma were located in two or more sites in cavity. And 29 patients (26.6%) of hemangioma and 11 patients (39.3%) lymphangioma of were found other extraoral lesions. With regard to some cases of hemangioma, the other patients commonly undergone surgical resections or sclerotherapy. The follow-up period ranged from 1 to 5 years. The recurrence rate of hemangioma and lymphangioma were 8.3%, 17.9%, respectively.Conclusion: Most of oral nonodontogenic masses are benign, few oral masses are malignant or mimic a malignant process. Surgical resections are the most common treatment with excellent success.

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Leiomyomatous Hamartomas of the Oral Cavity: Clinicopathological and Immunohistochemical Features of 4 Cases and Literature Review

Cited by 6 publications

References 28 publications

Lingual Hamartomas: Clinical Characteristics, Diagnostic Evaluation, Treatment, and Outcomes

Lingual Hamartomas: Clinical Characteristics, Diagnostic Evaluation, Treatment, and Outcomes

A Case of an Infantile Lingual Leiomyomatous Hamartoma

Clinical Features and Management of Oral Nonodontogenic Masses in Children

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