Characteristics of the salivary microbiota in cheilitis granulomatosa

BACKGROUND: Cheilitis granulomatosa (CG) is a disturbing and persistent idiopathic lip swelling. The cause and treatment has not been wholly elucidated. Some reports infer that CG is mainly associated with dental infection but no firm or reliable microbiological evidence has been provided for a causative organism. This study aimed to evaluate whether microorganisms contribute to the etiology of CG in order to inform appropriate treatment op-tions in clinic. MATERIAL AND METHODS: Unstimulated saliva was collected from 15 CG patients who were diagnosed clinically and pathologically and 15 healthy controls (HC). DNA was extracted from the precipitate of the centrifuged saliva for 16s rRNA high-throughput sequencing using the Miseq PE300 platform. The distribution of the microbiome between the two groups was compared. RESULTS: CG patients had a greater microbial flora that was more diverse than the HC. Prevotella, Alloprevotella, Porphyromonas, Actinomyces, Rothia, Fusobacterium, Haemophilus, and Aggregatibacter had a significantly higher abundance in CG patients. In contrast, Streptococcus and Campylobacter were the most abundant genera in HC with a mean relative abundance of 63% and 2%, respectively. The microbiological network indicated that most of the bacteria that were enriched at greater levels in CG patients were likely to be Prevotella, Actinomyces, and Rothia. These have been shown to co-exist with other bacteria. CONCLUSIONS: The composition and structure of bacterial communities in CG patients were different from HC. Most of the genera observed in CG patients were associated with periodontitis and pulp infection. These findings might be helpful in understanding the etiology of CG. Further study will be needed to confirm these findings and explore the underlying pathological mechanism

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Possible role of Mycobacterium tuberculosis complex in Melkersson-Rosenthal syndrome demonstrated with Gen-Probe amplified Mycobacterium tuberculosis direct test.

Melkersson-Rosenthal (MRS) syndrome is characterized by a classical triad of recurrent or persistent orofacial swelling, peripheral facial nerve paralysis and lingua plicata. Granulomatous cheilitis (GC) is regarded as a monosymptomatic form of MRS. The exact aetiologies of MRS and GC are unknown. In this study we investigated the possible role of mycobacteria in these two conditions. A ribosomal RNA amplification-based Gen-Probe amplified Mycobacterium tuberculosis direct test was used to investigate the presence of M. tuberculosis complex in paraffin-embedded skin biopsy specimens from five patients with MRS and one patient with GC. Three of the six specimens were shown to be positive using this system; one of the positive specimens also showed positive Ziehl-Neelsen staining. These results suggest a possible mycobacterial aetiology for MRS and GC.

Spirochetes--the possible etiological factor of the cheilitis granulomatosa.

OBJECTIVE: To detect spirochetes in sections and to study the therapeutic effect of penicillin in cheilitis granulomatosa (CG) and Melkersson-Rosenthal syndrome (MRS) and the relationship between the spirochetes infection and CG and MRS. METHODS: Routine HE sections and Warthin-Starry special staining were carried out in 20 cases of CG and 6 cases of MRS. Meanwhile there were 9 cases of CG and 2 caes of MRS were treated by penicillin (12 000 000u, i.v., per day) for two courses (14 days). RESULTS: A kind of spirochete was discovered in the sections of all cases of the CG and MRS. The CG and MRS could be divided into two types histopathologically, that is granuloma type and interstitial inflammatory type (non-granuloma type), those morphological changes tallied with spirochetosis. After treatment by penicillin, the facial and labial swelling of the 11 cases of CG and MRS were abated. CONCLUSION: CG and MRS probably are infectious diseases caused by spirochetes.

Granulomatous cheilitis and Borrelia burgdorferi: polymerase chain reaction and serologic studies in a retrospective case series of 12 patients.

BACKGROUND: Granulomatous cheilitis (GC) is a chronic granulomatous inflammation of the lips of unknown etiology, which may be associated with peripheral facial nerve paralysis and/or lingua plicata (Melkersson-Rosenthal syndrome [MRS]). Borrelia burgdorferi is a spirochete that causes Lyme borreliosis, a multisystemic infectious disease with frequent occurrence of facial nerve paralysis. An etiologic role of B burgdorferi in various granulomatous diseases has been suggested. The present study was performed to examine a possible causative role of B burgdorferi for GC/MRS by B burgdorferi-specific polymerase chain reaction analysis of biopsy specimens from affected lip tissue and determination of B burgdorferi IgG and IgM serum antibodies using enzyme-linked immunosorbent assay and immunoblot tests. OBSERVATIONS: We examined a retrospective case series of 12 patients with GC/MRS from a Lyme borreliosis endemic area (median duration of disease, 8 months [range, 3-348 months]). Borrelia burgdorferi-specific DNA could not be amplified by polymerase chain reaction in any of the 12 patients. One (13%) of 8 patients tested had a serum B burgdorferi IgG response on enzyme-linked immunosorbent assay, and 2 patients (25%) had an IgM response, but immunoblot testing yielded negative results in all 8 patients. CONCLUSION: The results of the present study do not indicate that B burgdorferi has an etiologic role in GC/MRS.

Ulcerous granulomatous cheilitis with lymphatic invasion caused by Scopulariopsis brevicaulis infection.

A 43-year-old man with ulcerous granulomatous cheilitis and submaxillary lymph node invasion caused by Scopulariopsis brevicaulis infection is described. Administration of itraconazole produced a dramatic and definitive cure. Reported cases of deep mycosis caused by S. brevicaulis infection are also reviewed.

[Relationship between Melkersson-Rosenthal syndrome and spirochetes infection].

By using Warthin-Starry spirochete special stain method and a transmission electron microscope for the first time, we detected spirochetes separately in lesions of 23 cases of MRS and 5 cases of MRS. Their shapes and distributive places were described. 11 cases were treated by high dose of penicillin, and 10 were responsive. This result provided further basis for a conjecture that the attack of MRS may be related to the infection of spirochetes.

[A study on the relationship between cheilitis granulomatosa and Melkersson-Rosenthal syndrome].

Serum anti Borrelia Burgdorferi (BB) antibody of 18 patients with cheilitis granulomatosa (CG) and 5 patients with Melkersson-Rosenthal syndrome (MRS) have been examined and positive results are present in 14 patients with CG(77.8%) and 4 patients with MRS(80%). According to histopathological features the lesions show characteristics of spirochetes diseases. It suggests that CG and MRS are all caused by spirochetes.

[Spirochetes in the cheilits granulomatosa and sarcoidosis].

Homogenate of seven cases of cheilits granulomatosa (CG), one case of Melkersson-Rosenthal syndrome (MRS) and one case of sarcoidosis (Sar) were found to have Borrelia underdark field microscope. CG culture of lip tissue of 2 cases showed Borrelia. Detection of anti-Borrelia Burgdorferi (BB)-antibody in serum showed that 8 (82%) of 11 cases of CG were positive, 3 (75%) of 4 cases of MRS and one case of Sar were positive. Histopathological changes in all cases were consistent with the pathological changes caused by spirochetes infection. The results confirmed that there is Borrelia in CG, MRS and Sar, which are spirochetes infected diseases.