Transient infantile lingual leukoplakia: An underrecognized cause of white tongues in infancy.

We have observed a distinct phenomenon of transient oral lingual leukoplakia in infancy and report 22 healthy infants with gray-white plaques on the dorsal tongue with sparing of the tip from four medical centers in three countries. The onset of the eruption ranged from 1 week to 7 months of life and resolved in 19 patients (86%, with 3 patients lost to follow-up). None of the eight patients examined at 1 year of age had residual findings. We believe this is a common entity that can be distinguished from oral candidiasis on clinical and/or laboratory examination and name this entity "transient infantile lingual leukoplakia."

Single ulcers on the tongue dorsum: differential diagnosis between paracoccidioidomycosis and squamous cell carcinoma.

BACKGROUND: Paracoccidioidomycosis (PCM) is the leading cause of death among systemic mycoses in Brazil. On the other hand, oral squamous cell carcinoma (OSCC) is the most prevalent malignant neoplasm of the mouth. Both lesions rarely affect the tongue dorsum and may share similar clinical characteristics. This study aimed to retrieve cases of single oral ulcers diagnosed as PCM or OSCC. MATERIAL AND METHODS: A cross-sectional retrospective study was conducted. All patients who had a single ulcer on dorsum of the tongue and confirmed diagnosis of PCM or OSCC were evaluated. RESULTS: A total of 9 patients (5 women and 4 men) were evaluated, 5 patients had OSCCs (mean age = 69,8 years old), and 4 patients PCM (mean age = 51 years old). Most of the lesions were infiltrated and indurated in the palpation exam. Duration ranged from 1 to 12 months (mean time of 5.2 months and 4.7 months for OSCC and PCM, respectively). OSCC was the main clinical diagnosis hypothesis. CONCLUSIONS: Although uncommon, PCM and OSCC should be considered as a diferential diagnosis hypothesis in infiltrated ulcers on the tongue dorsum. Iincisional biopsy is mandatory to confirm the diagnosis and indicate the appropriate treatment.

Surgical and Speech Therapy Evaluation of Lingual Frenulum.

Interdisciplinary evaluation is essential to diagnose and define clinical conduct for children and adults presenting with an altered lingual frenulum; however, there are few reports on the subject in the literature. In this context, the following study exemplifies a proposed protocol for the surgical and SLT treatment of a lingual frenulum based on a review of the literature and the experience of speech and language therapists and maxillofacial surgeons from hospitals in Santiago de Chile. After its application, a history of breastfeeding with difficulties and a maintained preference for soft foods was reported. Upon anatomic examination, the lingual apex was heart-shaped, and the lingual frenulum was fixed in the upper third of the ventral side of the tongue, with a pointed shape, submerged up to the apex, and of adequate thickness. Meanwhile, upon functional examination, the tongue was descended at rest, performed tongue protrusion with restrictions (raising and clicking), did not achieve attachment or vibration, and presented distortion of the sounds / r/ and /rr/. With this information, an altered lingual frenulum was diagnosed, with the indication for surgery and postoperative speech and language therapy. The constructed instrument allowed for the standardization of the evaluation in different teams but should be validated in future research.

Cartilaginous choristoma on the lateral surface of the tongue: a case report.

A cartilaginous choristoma is a nonneoplastic nodular growth of histologically normal cartilage in an abnormal site. This report describes a case of a cartilaginous choristoma on the lateral surface of the tongue in a 65-year-old woman. The entire lesion was excised, and histologic examination revealed mature cartilaginous tissue surrounded by dense connective tissue. Choristomas are rare findings in the oral cavity, easily confused with proliferative processes or soft tissue neoplasms. Nevertheless, choristomas may be part of the differential diagnosis for lesions similar to the one described in this case report.

[Morsicatio related white lesions of the tongue borders and lower lip: a case report]. / Witte veranderingen van de onderlip en de tongranden als gevolg van morsicatio.

A 28-year-old woman was referred by her dentist to an oral and maxillofacial surgeon for white lesions of the lower lip and lateral tongue borders on both sides. The diagnoss morsicatio labiorum and linguarum was made. Cheek, tongue, and lip biting occurs with some regularity and often patients are unaware of this habit. The diagnosis of morsicatio is based on clinical appearance and additional diagnostics are unnecessary. The condition belongs to a subcategory of obsessive-compulsive disorders. Treatment is not always necessary, but may consist of making the patient aware of the cause, possibly combined with symptomatic and/or causal therapy, for instance with an occlusal splint or cognitive behavioral therapy.

Congenital fibrous hamartoma of the tip of the tongue: A novel peculiar entity.

Congenital fibrous hamartoma of the tip of the tongue, a peculiar and novel entity, consists of one or two asymptomatic pearly or yellowish nodules, not exceeding 0.5 cm in maximum diameter, at the tip of the tongue, ventrally or dorsally. Unlike other localizations, congenital fibrous hamartoma of the tip of the tongue is not associated with cleft lip or palate, or with feeding problems. Surgical excision should be avoided, reserved only for dubious cases, since the lesions are benign and stable over time.

Bifid Tongue, Congenital Tongue Tumor, and Wide Bilateral Complete Cleft Palate.

ABSTRACT: Congenital tumor of tongue is not rare, but associated with cleft palate and bifid tongue is unusual. The authors describe a case of 7-year-old patient having bifid tongue, congenital tongue tumor, and cleft palate. Clinical features, physical and pathological examinations, possible diagnosis, and surgical treatment have been elaborated in this case report.

Riga-Fede Disease: A Case of Sublingual Trauma Not Associated With Abuse.

OBJECTIVES: The following case presents a pediatric patient with Riga-Fede disease (RFD), a rare disorder in children under 2 years of age characterized by sublingual ulceration of the tongue due to trauma from repeated rubbing against the primary teeth. Riga-Fede disease is well reported in dental literature but is relatively unknown to the general pediatric community. It can be confused with nonaccidental trauma (NAT) from forced feeding or other abusive trauma because it presents with injury of oral structures including the sublingual frenulum and often results in problems with growth. This case highlights the importance for physicians to recognize RFD as a source of isolated oral injury that is distinct from NAT. Although approximately 35 cases of RFD have been reported in the literature, this is the first case to describe the process of distinguishing this disease from child abuse. METHODS: We present a case of an 8-month-old female infant who presented with extensive injury of her ventral tongue from RFD who underwent a full workup for NAT. We review the literature for guidance on diagnosing RFD and discuss how to distinguish it from abuse. RESULTS: Infants with characteristic ulceration of the ventral tongue opposing new teeth and with no other medical, social, or developmental concerns do not need to undergo further workup. Clinical examination and thorough history are sufficient to make the diagnosis. CONCLUSIONS: Traumatic intraoral injury in nonmobile children is highly concerning for child abuse. However, RFD is a well described cause of sublingual ulceration in infants that is not associated with abuse.

Amyloidoma of the Tongue: Case Report, Surgical Management, and Review of the Literature.

PURPOSE: Localized amyloidosis of the tongue is a benign condition in which surgical management may be considered. The aim of the study was to review the current literature and report a case. MATERIALS AND METHODS: We searched the PubMed database for all relevant articles reporting cases of localized tongue amyloidosis published between 1980 and February 2020. In addition, we updated 1 case diagnosed and treated in our department. RESULTS: A 49-year-old male patient presented with an asymptomatic tongue nodule of the dorsum mimicking median rhomboid glossitis. The results of an incisional biopsy showed an amyloid on Congo red staining and positive findings for the κ light chain by immunohistochemical analysis. The findings of the systemic workup were negative. Therefore, a diagnosis of localized κ light-chain amyloidosis was made. The patient underwent a resection of the lesion, and no recurrence or progression was observed during a period of 18 months. The literature review showed 12 reports describing 21 patients (11 men, 52.3%) with localized tongue amyloidosis. The most common clinical presentation was nodular with a single lesion of the tongue dorsum (15 patients, 71.4%). All cases showed positive findings on Congo red staining. Immunohistochemical analysis findings were available for only 9 patients (42.8%) and showed light-chain amyloidosis. No case showed any systemic involvement or the development of systemic disease. Surgical excision was performed in 9 cases, with recurrence at the site of operation in 2 cases. CONCLUSIONS: Localized amyloidosis of the tongue is a rare disease in which surgical excision may be therapeutic when a multidisciplinary evaluation does not show any systemic disease. We recommend an excision when the lesion is persistent or shows an enlargement or when discomfort is reported. In the case of any further local recurrence, resection may be repeated.

[Pseudotumoral sublingual leishmaniasis caused by Leishmaniainfantum]. / Leishmaniose sublinguale pseudotumorale à Leishmaniainfantum.

INTRODUCTION: Mucosal leishmaniasis is endemic in certain parts of Latin America and are usually absent in Morocco. Herein we report a case of Leishmaniainfantum in a Moroccan patient. PATIENTS AND METHODS: A 61-year-old male patient working as a tourist bus driver presented with a sublingual endobuccal tumor. He reported a history of treated cutaneous leishmaniasis of the lower lip in 2009 and had presented the sublingual oral tumor since December 2011. The histopathological findings as well as the species-specific PCR analysis confirmed the diagnosis of sublingual mucosal leishmaniasis due to L. infantum. HIV serology was negative. Our patient was then treated with intra-muscular meglumine antimoniate for 25 days, resulting in complete disappearance of the oral lesion. CONCLUSION: Our case thus has several peculiarities: the strictly mucosal character of the lesion, occurring in an immunocompetent subject, the unusual pseudotumoral form, and the causative agent, L. infantum, not known for its mucosal tropism in our country.

Lymphogranuloma venereum presenting as an ulcer on the tongue.

Oropharyngeal lymphogranuloma venereum (LGV) is an uncommon presentation and mainly consists of either asymptomatic infections or enlarged cervical lymphadenopathy. According to cross-sectional studies, the prevalence of pharyngeal LGV among men who have sex with men (MSM) seems to be very low. Tissue tropism could explain the difference between rectal and oral mucosa infection frequencies. This paper reports the singular case of an oral ulcer caused by LGV in an MSM who had also an asymptomatic rectal infection. Given the increasing number of cases of LGV, unusual presentations such as oral ulceration may be seen more frequently.

Development of a standardized evaluation of endobuccal adverse events induced by repeated tongue protrusion with both a dedicated questionnaire and an endobuccal examination.

INTRODUCTION: A new approach to treat obstructive sleep apnea (OSA) is upper airway stimulation therapy (UAS). Electrical pulses applied to the hypoglossal nerve induce tongue protrusion, increase airway patency and decrease the frequency of apneic and hypopneic events. Thus, the main objective of this study was to design a standardized evaluation of endobuccal adverse events induced by repeated tongue protrusion with both a dedicated questionnaire and an endobuccal examination. METHOD: This study has designed the Tongue Adverse Event and Satisfaction Questionnaire (TAESQ) and an endobuccal examinations divided into an endobuccal lesion examination (ELE) and an endobuccal risk factor examination (ERFE). Evaluations were conducted at month 6 post-implantation. RESULTS: The study population after implantation of UAS device consisted of ten Caucasian males with a mean age of 51.9 ± 11.8 years, and a mean BMI of 28.6 ± 3.3. The AHI of the ten participants ranged from 46.7 ± 12.2/h at baseline to 14.5 ± 8.9/h with the Inspire therapy at the 6-month follow-up. The TAESQ revealed pain (30%), followed by less tongue sensitivity (20%) and tongue weakness (10%). The ELE did not reveal any lesions. The ERFE revealed that some participants had tissue and dental risk factors but not associated to more adverse events. CONCLUSION: The TAESQ, ELE and ERFE have been designed and studied on a small number of participants. These evaluations could systematically be included in the care pathway of patients treated by UAS to better investigate tongue discomfort and tongue lesion for patients treated with this technology.

Hemilingual Angioedema after Thrombolysis in a Patient on an Angiotensin II Receptor Blocker.

Orolingual angioedema (OLAE) is a rare adverse effect of alteplase. Previous studies have associated the occurrence of OLAE with thrombolysed patients maintained on angiotensin converting enzyme inhibitors. We report a case of a 60-year-old male presenting with hyperacute ischemic stroke developing hemilingual edema after thrombolysis. He was previously maintained on an angiotensin II receptor blocker (ARB), losartan. The swelling resolved over 2 days with immediate administration of intravenous steroids and antihistamine drugs. Our case is the third documented case of OLAE occurring in a thrombolysed patient concurrently taking an ARB. The presence of hemilingual edema in a post-thrombolysis patient maintained on losartan suggests a possible association between OLAE and ARBs.

Asymptomatic and painful ulcerated lesions on the lip and tongue.

The presence of multiple primary oral squamous cell carcinomas (SCCs) in a single patient is rare in dental practice. A 63-year-old man was referred for evaluation of a single tongue lesion. No lymphadenopathy was detected on extraoral examination. However, multiple asymptomatic ulcerated and crusted lesions were observed on the vermilion border of the lower lip. The intraoral examination revealed an extensive, painful endophytic ulcer of 3 months' duration on the right lateral border of the tongue. No additional findings were observed. Incisional biopsies were performed, and the histopathologic analysis confirmed the diagnosis of SCCs. The patient was referred for oncologic therapy. Surgery was the treatment of choice for both the extraoral and intraoral lesions. No disease was found after 5 months of follow-up. Although uncommon, multiple oral SCC lesions can appear to be clinically similar to deep infectious disease. Metachronous tumors were diagnosed in this patient in light of the probable course of onset of the lesions. A rigorous clinical examination that includes the lips is essential to make an accurate diagnosis and refer the patient for appropriate treatment.

[Temporal arteritis, sometimes oral symptoms are the first manifestation]. / Arteriitis temporalis, orale verschijnselen zijn soms de eerste uiting.

Sometimes it is difficult to diagnose temporal arteritis because the complaints may vary, change in intensity and are not always characteristic. The condition is a cranial form of giant-cell arteritis affecting large and medium-sized arteries. The first manifestation of temporal arteritis can be a sore tongue that does not manifest any abnormalities during a clinical investigation. In a later stage patients sometimes develop ulceration or necrosis of a part of one side of the tongue. Other symptoms can be a recently developed headache, jaw claudication and acute loss of vision. To diagnose temporal arteritis, histological investigation of a biopsy of the temporal artery is carried out. The treatment consists of long-term use of corticosteroids. A patient in your practice with inexplicable pain on one side of the tongue, without clinical abnormalities, or an ulceration of the tongue without an immediately apparent cause may have temporal arteritis.

Neck-Tongue syndrome: A systematic review.

Objective Neck-Tongue syndrome (NTS) is characterized by brief attacks of neck or occipital pain, or both, brought out by abrupt head turning and accompanied by ipsilateral tongue symptoms. As the disorder is rare, we undertook a systematic review of the literature to identify all reported cases in order to phenotype clinically the disorder and subsequently inform clinical diagnostic criteria. Methods Two electronic databases were searched using the search term "neck tongue syndrome". All English language references were reviewed in full. Cases were abstracted using a standardized abstraction form and the references of the retrieved articles were reviewed by hand to identify additional references and cases. Conference proceedings from recent headache meetings were searched. We also report six new cases from our centers. Results There were 39 primary cases, 56% of which were female. Mean age (SD) at onset was 16 (12) years. Twenty (53%) experienced neck pain, seven (18%) occipital pain, and 11 (29%) both. Pain was most often sharp or stabbing and severe, lasting several seconds to several minutes. Eleven experienced numbness and/or tingling in the neck/occiput following the pain. Thirty-six had an accompanying tongue sensory disturbance and three a motor/posture disturbance; five had both. Thirteen had other headaches, and four a family history of Neck-Tongue syndrome. Conclusions Neck-Tongue syndrome typically has pediatric or adolescent onset, suggesting that ligamentous laxity during growth and development may facilitate transient subluxation of the lateral atlantoaxial joint with sudden head turning. Familial cases suggest a genetic predisposition in some individuals. Neck-Tongue syndrome should be re-instated in the International Classification of Headache Disorders.