Challenging diagnosis and successful treatment of localised Mycobacterium avium subsp. hominissuis glossitis in a dog on long-term immunomodulatory therapy.

CASE HISTORY: A 3-year-old, intact female mixed-breed dog, weighing 7 kg, was presented with generalised swelling of the tongue, leading to impaired deglutition and episodes of dyspnoea. From the age of 2 years, the dog had been under immunosuppressive therapy due to atopic dermatitis. CLINICAL FINDINGS AND TREATMENT: Multiple nodular lesions at the apex of the tongue were noted as well as mandibular and retropharyngeal lymph node enlargement. Serum biochemistry results showed inflammatory changes. The results of several biopsies taken over 7 months indicated persistent pyogranulomatous and necrotising glossitis despite ongoing antimicrobial treatment, first with amoxicillin/clavulanic acid and then pradofloxacin. No foreign material, acid-fast bacteria or fungal hyphae were detected throughout. The final diagnosis of Mycobacterium avium subsp. hominissuis (Mah) was reached after PCR and bacterial culture were carried out on the third biopsy sample. Therapy was initiated with rifampicin, clarithromycin and doxycycline, leading to complete remission of the lesions. DIAGNOSIS: Severe chronic pyogranulomatous and necrotising glossitis associated with infection by Mah. CLINICAL RELEVANCE: This report describes challenges in the diagnosis and therapy of a localised Mah infection in an iatrogenically immunocompromised dog. Successful treatment was only achieved with a specific combination of antibiotics administered long-term. ABBREVIATIONS: AF: Acid-fast; ALP: Alkaline phosphatase; CT: Computed tomography; MAC: Mycobacterium avium complex; Mah: Mycobacterium avium subsp. hominissuis.

Effective vitamin B12 treatment can reduce serum antigastric parietal cell antibody titer in patients with oral mucosal disease.

BACKGROUND/PURPOSE: Patients with serum antigastric parietal cell antibody (GPCA) positivity may have vitamin B12 deficiency and some oral symptoms. This study assessed the changes of serum GPCA titer in GPCA-positive patients after effective vitamin B12 treatment. METHODS: Two hundred and ten GPCA-positive oral mucosal disease patients became oral symptom free (complete response) after 1.0-67.1 months of treatment with regular and continuous intramuscular injection of vitamin B12 once per week. The changes of serum GPCA titers after treatment were evaluated in these 210 patients. RESULTS: We found a significant drop of the GPCA positive rate from 100% to 42.9% in our 210 complete response patients after effective vitamin B12 treatment (p < 0.001). When 210 patients were further divided into seven subgroups according to the low to high serum GPCA titers, we noted that the higher serum GPCA titers decreased to significantly lower levels after treatment in all seven subgroups (all p < 0.001). However, serum GPCA titers increased to significantly higher levels in 46 GPCA-positive control patients receiving only oral administration of two vitamin BC capsules (containing 10 µg of vitamin B12) plus deficient hematinic supplements per day after a follow-up period of 2.7-27 months. A maintenance vitamin B12 treatment once a month could retain the GPCA-negative status in 87% of treated-to GPCA-negative patients compared with those (10%) without further maintenance vitamin B12 treatment. CONCLUSION: Regular and continuous effective vitamin B12 treatment can reduce the relatively higher serum GPCA titers to significantly lower or undetectable levels in GPCA-positive patients.

Novel submicronized rebamipide liquid with moderate viscosity: significant effects on oral mucositis in animal models.

This study aimed at developing a novel rebamipide liquid for an effective treatment of oral mucositis. The healing effects of a variety of liquids comprising submicronized rebamipide crystals were investigated using a rat cauterization-induced oral ulcer model. Whereas 2% rebamipide liquid comprising micro-crystals did not exhibit significant curative effect, 2% rebamipide liquids comprising submicronized crystals with moderate viscosities exhibited healing effects following intra-oral administration. The 2% and 4% optimized rebamipide liquids showed significant healing effects in the rat oral ulcer model (p<0.01). In addition, in the rat radiation-induced glossitis model, whereby the injury was caused to the tongue by exposing only around the rat's snout to a 15 Gy of X-irradiation, the 2% optimized rebamipide liquid significantly reduced the percent area of ulcerated injury (p<0.05). In conclusion, the submicronized rebamipide liquid with moderate viscosity following intra-oral administration showed better both healing effect in the rat oral ulcer model and preventive effect in the rat irradiation-induced glossitis model.

[Glossitis by mixed infection of cytomegalovirus and herpes simplex virus during therapy for pure red cell aplasia complicated with Good syndrome].

A 64-year-old man with a 10-year history of Good syndrome had been treated with periodic replacement of γ-globulin. He also had a 6-year history of lichen planus of the tongue. In 2009, the patient was diagnosed as having pure red cell aplasia (PRCA) based on bone marrow aspiration. Thymectomy was not effective. Then, immunosuppressive therapy with PSL and cyclosporine was initiated. Twenty days after treatment painful ulcer appeared on the left side of the tongue. Biopsy specimen of the ulcer demonstrated cells infected with cytomegalovirus and herpes simplex virus. Cytomegalovirus antigenemia was also positive. The tongue ulcer promptly improved after gancyclovir administration for a few weeks. Viral glossitis should be considered as part of the differential diagnoses of oral lesions not only in patients with HIV infection but also in those under immunosuppressive therapy.

Herpetic geometric glossitis: acyclovir resistant case in a patient with acute myelogenous leukemia.

Herpes simplex virus (HSV) infections in an immunocompromised host may be atypical in location and morphology. Lesions are more extensive and aggressive, slow healing or nonhealing and extremely painful. Intraoral lesions are ulcerative and may involve any intraoral, oropharyngeal, or esophageal site. Herpetic geometric glossitis is a recently described form of lingual HSV infection in an immunocompromised patient. It was described as ulcer on the dorsum of the tongue sensitive for acyclovir therapy. A patient is presented with acute myelogenous leukemia that developed herpetic geometric glossitis which was acyclovir resistant.

Pyostomatitis vegetans associated with inflammatory bowel disease--report of two cases.

Pyostomatitis vegetans (PV) is a rare, chronic mucocutaneous disorder associated with inflammatory bowel disease (IBD). Oral lesions of PV are distinct and present as multiple white or yellow pustules with an erythematous base that coalesce and undergo necrosis to form a typical "snail tracks" appearance. Two cases of PV associated with IBD--one with Crohn's disease (CD) and the other with ulcerative colitis (UC) are reported. In the first case, adalimumab therapy brought the oral and gastrointestinal manifestations to complete remission. In the second case, the remission was achieved with systemic steroid therapy, but the disease relapsed after therapy discontinuation. Azathioprine was added leading to sustained remission of PV. Because of persistent active intestinal manifestation of UC, in spite of immunosuppressive therapy, infliximab was introduced. With the therapy remission of intestinal manifestation of UC was achieved as well. Our cases confirm previously reported good experience with immunomodulators and biologics in the treatment of PV. But, before using them we have to exclude an infectious etiology of oral lesions.

Herpetic geometric glossitis in an immunocompetent patient with pneumonia.

Herpetic geometric glossitis is an uncommon, clinically distinctive presentation of oral herpes simplex virus infection that affects the tongue. All published reports have been in patients who are immunocompromised. We present an immunocompetent woman with viral pneumonia who developed painful linear fissures on the back of the tongue suggestive of herpetic geometric glossitis. The diagnosis was confirmed by both biopsy specimen and immunohistochemistry. As in other cases, the patient promptly responded to treatment with antiviral therapy. The morphology, laboratory findings, and similarities to herpetic corneal dendrites are discussed. Herpetic geometric glossitis should no longer be considered as a diagnosis only in immunosuppressed individuals, but in immunocompetent persons as well.

Primary tuberculous glossitis in an immunocompetent patient.

Tuberculous glossitis is a rare entity that has been described sporadically. Primary tuberculous glossitis, as described in this case report, is still exceptional. A 25-year-old male with no known immunosuppressive disorder presented with a tuberculoma at the base of his tongue. This was confirmed by tongue biopsy and a positive polymerase chain reaction response to the mycobacterium. The patient had a favourable response to anti-tubercular treatment. This highlights the importance of considering tuberculosis in the differential diagnosis of chronic tongue lesions, even in the absence of pulmonary tuberculosis.

[Burning mouth syndrome]. / Zungenbrennen.

Burning mouth syndrome (BMS) is characterized by oral dysesthesia, xerostomia and dysgeusia without visible alterations of oral mucosa. While secondary BMS results from an underlying general condition such as diabetes or iron deficiency, no causal disorder can be identified in primary BMS. The estimated prevalence is 1 - 2%, postmenopausal women are substantially more frequently affected than men. Current etiologic concepts assume a focal peripheral and central neuropathy. Only few controlled drug trials have yet been conducted. Thioctic acid appears the medical treatment of choice due to its comparatively good evidence for efficacy and low incidence of adverse reaction. Gabapentin and pregabalin are modern GABA-analogue anticonvulsants, which are also efficient in the treatment of peripheral neuropathies. Also conceptually appropriate for BMS treatment, current evidence for efficacy in BMS is insufficient. In two trials, local oral treatment with clonazepam has been beneficial in BMS. The efficacy of antidepressants is equivocal.

Rhomboid glossitis in atypical location: case report and differential diagnosis.

Median rhomboid glossitis (MRG) is an uncommon benign abnormality of the tongue, most frequently affecting men. It is typically located around the midline of the dorsum of the tongue, anterior to the lingual "V", appearing as a reddish, rhomboid area, depapillated, flat maculate or mamillated and raised by 2 - 5 mm. This paper reports a case of rhomboid glossitis in a 61-year-old man who consulted for a painless raised lesion on the dorsum of the tongue, in left paramedial (not medial) location. Histopathological findings were compatible with rhomboid glossitis. Other diagnoses considered but ruled out on the basis of the clinical and histopathological findings were haemangioma, pyogenic granuloma, amyloidosis, granular cell tumour, and squamous cell carcinoma. This case confirms that rhomboid glossitis may occur in paramedial locations.

Oral candidosis and the therapeutic use of antifungal agents in dentistry.

This paper reviews the current concepts of mycology and candidal infections as they relate to the oral cavity. Proposed classification for the presentation of oral candidosis is outlined as are examples of these topical infections, such as erythematous, pseudomembranous and hyperplastic candidosis, as well as angular chelitis and median rhomboid glossitis. The diagnosis and principles of management of oral candidosis are discussed, the therapeutic agents available for the management of these infections are presented and a treatment protocol for the management of patients with oral candidosis is given.

Granulomatous glossitis: a case report.

A 50-year-old man was admitted to our clinic with a complaint of lingual enlargement. Detection of non-caseous epithelioid granuloma on histopathological examination led to a diagnosis of a granulomatous glossitis. Extensive investigation for the presence of associated disorders yielded negative results. Metranidazole and clofazimine were totally ineffective and tetracycline led to a minimal improvement. No associated disorder was detected at a 4-year follow-up examination. The position of granulomatous glossitis within the spectrum of orofacial granulomatous conditions is discussed.

Effect of pyridoxine or riboflavin supplementation on plasma homocysteine levels in women with oral lesions.

BACKGROUND: A moderate increase in plasma homocysteine level has been reported to be involved in neural tube defects, which can be prevented with folic acid supplementation. Folic acid, vitamins B6- and B12-dependent enzymes are required to metabolize homocysteine. A study in rats showed higher tissue homocysteine levels in riboflavin as well as pyridoxine deficiency. We studied the effect of treatment with pyridoxine or riboflavin on plasma total homocysteine concentration in women with clinical and biochemical deficiencies of riboflavin and pyridoxine. METHODS: Plasma total homocysteine concentrations were measured in 20 women with glossitis and angular stomatitis before and after supplementation with pyridoxine or riboflavin. RESULTS: Pyridoxine treatment significantly reduced plasma homocysteine concentration while riboflavin treatment did not have a significant effect. CONCLUSIONS: Plasma total homocysteine levels tended to be higher in women with clinical and biochemical deficiency of vitamin B6 and therapy with pyridoxine reduced its level significantly. Riboflavin supplementation did not have a significant impact on plasma homocysteine concentration in women with glossitis and angular stomatitis.

Herpetic geometric glossitis in a pediatric patient with acute myelogenous leukemia.

Herpetic geometric glossitis, a recently described form of lingual herpes simplex virus type 1 (HSV-1) infection, has been reported in 6 human immunodeficiency virus (HIV) patients and 1 cardiac transplant patient who was receiving immunosuppressant therapy. An HIV-seronegative immunocompromised pediatric patient with acute myelogenous leukemia who developed herpetic geometric glossitis is described. Herpetic geometric glossitis can present in both adult and pediatric immunocompromised patients. The symptoms, morphology, laboratory findings and treatment of this infection are summarized. The possible consequences of untreated herpetic glossitis include superinfection and undernourishment. Although previously described patients responded to 1000 mg per day (divided in 5 doses) or oral acyclovir, with complete resolution of fissures, this patient developed herpetic geometric glossitis while receiving acyclovir and required higher doses of oral antiviral therapy (acyclovir, 3000 mg/day divided in 5 doses) to treat his HSV-1 lingual infection. Empiric treatment of an immunocompromised patient who has newly acquired painful tongue fissures or furrows with systemic acyclovir should be considered.

[Glossitis granulomatosa symptom of oligosymptomatic Melkersson-Rosenthal syndrome]. / Glossitis granulomatosa-Symptom eines oligosymptomatischen Melkersson-Rosenthal-Syndroms.

Atypical forms of Melkersson-Rosenthal syndrome are difficult to diagnose, especially when occurring in uncommon facial locations. The clinical and histological characteristics of these forms are illustrated in a typical case. We report our experiences with a 59-year-old white male who had increasing swelling of his tongue for years and a recent onset of perioral swelling. A plicated tongue was present, while symptoms of facial and trigeminal nerve involvement were absent. Minor symptoms present were speech impediments, dysgeusia, episodes of hypersalivation and burning sensations on eating. Biopsies revealed sarcoid and lymphonodular-plasmocytic granulomatous infiltrates. These findings were consistent with a diagnosis of an oligosymptomatic Melkersson-Rosenthal syndrome. Swelling of the tongue and that of the perioral region was controlled with clofazimine therapy.

Granulomatous glossitis as an unusual manifestation of Melkersson-Rosenthal syndrome.

The case of a 59-year-old man with granulomatous glossitis is presented. The patient complained of increasing swelling of his tongue for years and a recent onset of perioral edema. Because of sarcoid-like granulomas found in a first lingual biopsy specimen, the disease was initially misdiagnosed as a localized form of sarcoidosis. A second biopsy specimen from the tongue revealed lymphonodular-plasmocytic granuloma-like infiltrates, which, in connection with the labial swelling, led to the diagnosis of Schuermann's glossitis granulomatosa as a peculiar manifestation of Melkersson-Rosenthal syndrome. The surface of the tongue showed deep furrows characteristic of lingua plicata. However, no history or signs of facial or trigeminal nerve paralysis were present; thus the patient did not show the complete triad of symptoms typical of Melkersson-Rosenthal syndrome. After treatment with clofazimine the perioral and lingual swelling disappeared within two weeks. Recurrences have been suppressed with a reduced maintenance dosage of this agent.

Relationship between biochemical and clinical indices of B-vitamin deficiency. A study in rural school boys.

1. A study amongst schoolboys in villages around Hyderabad, India, showed that almost all the boys had riboflavin deficiency, 61% had pyridoxine deficiency, and 9.4% had thiamin deficiency as judged by enzymic tests. 2. The prevalence of angular stomatitis was 41.3% and that of glossitis was 18.2%. Biochemical deficiency of riboflavin and pyridoxine was marginally higher in children with angular stomatitis with or without associated glossitis, than in children without oral lesions. 3. Treatment with B-complex vitamins (containing 4 mg riboflavin and 10 mg pyridoxine) daily for 1 month produced significant reduction in the prevalence of glossitis but had no effect on angular stomatitis. The latter responded to topical application of gentian violet. 4. Small but significant changes in erythrocyte enzymes occurred over the period of 1 month even without vitamin supplements. 5. Results suggest that while glossitis is a relatively early manifestation of riboflavin or pyridoxine deficiency or both, angular stomatitis has a more complex aetiology perphaps associated with infection.