Langerhans Cell Histiocytosis

Giovannetti, Filippo; Aboh, Ikenna Valentine; Chisci, Glauco; Gennaro, Paolo; Gabriele, Guido; Cascino, Flavia; Curzio, Paolo Di; Iannetti, Giorgio

doi:10.1097/scs.0000000000000668

Cited by 6 publications

(6 citation statements)

References 9 publications

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“…At the beginning of the case study, a differential diagnosis was made between possible syndromes involving the oral cavity [ 20 , 21 , 22 , 23 ]. This patient’s characteristic phenotype and clinical findings enabled a suspected diagnosis of CFCS to be established, which ultimately was confirmed with the genetic study [ 9 ].…”

Section: Discussionmentioning

confidence: 99%

Analysis of the Oral Microbiome in a Patient with Cardiofaciocutaneous Syndrome and Severe Periodontal Disease: Impact of Systemic Antibiotic Therapy

et al. 2022

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An 8-year-old girl diagnosed with cardiofaciocutaneous syndrome presented to our department with gingival pain, inflammation, and bleeding. Her medical history included hypoplasia of the corpus callosum, intellectual disability, trichothiodystrophy, global developmental delay, myopia, laryngomalacia, hypothyroidism, and osteoporosis. A diagnosis was reached of “periodontitis as a direct manifestation of systemic diseases”. During 9 years of follow-up, there were exacerbation episodes with spontaneous gum bleeding, ulcers in the interdental papilla, tooth mobility, and progressive tooth loss. Some of these exacerbation episodes resolved clinically with the administration of amoxicillin and metronidazole. We therefore proposed an oral microbiome study (subgingival and saliva samples) before and after antibiotic therapy. The most abundant genera at the subgingival level before administering antibiotics were Prevotella, Streptococcus, Fusobacterium, Leptotrichia, and Aggregatibacter. Of the 94 genera sequenced, 57 were less abundant in the post-treatment state than at baseline, particularly certain Gram-negative periodontal pathogens such as Porphyromonas, Treponema, Aggregatibacter, Fusobacterium, and Campylobacter. In contrast, other genera related to oral health, such as Haemophilus, Granulicatella, and Abiotrophia, showed an increase after administering the antibiotic. In conclusion, periodontitis exacerbations as a direct manifestation of systemic disease can occasionally be controlled exclusively with systemic antibiotics, without the need for performing mechanical periodontal therapy. This clinical recovery is correlated to substantial changes in the oral microbiome, which lead to the recovery of eubiosis of the microbiota.

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Section: Discussionmentioning

confidence: 99%

Analysis of the Oral Microbiome in a Patient with Cardiofaciocutaneous Syndrome and Severe Periodontal Disease: Impact of Systemic Antibiotic Therapy

et al. 2022

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“…They are found in 7.4% to 77% of cases [1,6,10,11]. Clinical signs mainly reported are pain, swelling, ulcerations, gingival bleeding, dental mobility, premature tooth loss and lack of mucosa healing after tooth extraction [1,4,10,12]. Differential diagnosis include periodontal diseases, periapical diseases, odontogenic cyst, oral cancers, lymphoma, myeloma or ameloblastoma [1,4,6,7].…”

Section: Discussionmentioning

confidence: 99%

“…Of oral manifestations, 67% affect bone tissue, the posterior mandible in particular [1,4,7,12,13]. They can be isolated or multiple and moderate to severe.…”

Section: Discussionmentioning

confidence: 99%

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Diagnosis, treatment and recurrence of a mandibular Langerhans cell histiocytosis: a three-year follow-up case report

Morcel¹,

Keribin

Quenel

et al. 2022

J Oral Med Oral Surg

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Introduction: Langerhans cell histiocytosis (LCH) is an abnormal clonal proliferation of Langerhans cells secondary to immune process, mutation of oncogene or genetic predispositions. It preferentially affects bone, lung and skin. The incidence is 2–6 cases per million per year. Prognosis is variable and depends on number and location of lesions, and impact of the initial treatment. Oral lesions may be the first sign of LCH as illustrated by the present case. Observation: A 24-year-old male consulted first for severe gingival inflammation, teeth mobilities and alveolar bone loss with a suspicion of LCH. A pulmonary involvement was secondarily revealed by tomodensitometry. Histological examination, from gingival biopsy, confirmed the diagnostic of LCH, showing cells positive for the anti-CD1A antibody. The patient was managed by oral surgery and chemotherapy approaches. Alveolar bone loss significantly reduced. But 2 years and a half after the diagnosis, a recurrence was noted and managed by surgical approach. After a three-year follow-up, no recurrence was noted. Conclusion: Oral lesions can be inaugural manifestations of LCH. The dentist has an essential role in the early detection of these lesions.

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“…Because of this, young children are usually followed up by a multidisciplinary team. Unfortunately, dental and maxillofacial professionals are not often included, even though dental and maxilla–mandibular anomalies are described as common long-term side effects of childhood cancer treatment [ 4 , 5 , 6 , 7 ].…”

Section: Introductionmentioning

confidence: 99%

Oligomicrodontia in a Pediatric Cancer Survivor after Chemotherapy: A Case Report

et al. 2022

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Chemotherapy used on pediatric patients especially those below 3 years of age causes disturbances in dental development. The aim of this case report was to present the late dental effects of chemotherapy in a patient treated for anaplastic ependymoma (WHO III) at an early age. Radiographic findings at the age of 9 years showed oligomicrodontia of six teeth, maxillary lateral incisors, and maxillary and mandibular first premolars. Pediatric cancer survivors after chemotherapy have an increased risk of one or more dental development disorders. To ensure proper dental care and to assess the long-term effects on oral health, tooth development, and occlusion, the involvement of a dentist is crucial. Adequate diagnosis and well-planned treatment of the dental defect can significantly improve patient oral health-related quality of life.

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Langerhans Cell Histiocytosis

Cited by 6 publications

References 9 publications

Analysis of the Oral Microbiome in a Patient with Cardiofaciocutaneous Syndrome and Severe Periodontal Disease: Impact of Systemic Antibiotic Therapy

Analysis of the Oral Microbiome in a Patient with Cardiofaciocutaneous Syndrome and Severe Periodontal Disease: Impact of Systemic Antibiotic Therapy

Diagnosis, treatment and recurrence of a mandibular Langerhans cell histiocytosis: a three-year follow-up case report

Oligomicrodontia in a Pediatric Cancer Survivor after Chemotherapy: A Case Report

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