Evidence map›Paper›PMID 40197712›Full record

ArticleSpecial care in dentistry : official publication of the American Association of Hospital Dentists, the Academy of Dentistry for the Handicapped, and the American Society for Geriatric Dentistry

Overlap Diagnostic Odyssey and Full Mouth Rehabilitation of a Juvenile Patient With IFIH1-Related Disorder: A Case of Aicardi-Goutières and Singleton Merten Syndromes Overlap.

Clive Friedman, Timucin Ari, Gürkan Ünsal, Tugce Balci, Matthew Morrison, Nilgun Ari

Erratum issuedAbstract readCase Reports
In one paragraph

Article in Special care in dentistry : official publication of the American Association of Hospital Dentists, the Academy of Dentistry for the Handicapped, and the American Society for Geriatric Dentistry. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

6 authors.

Clive FriedmanDepartment of Paediatric Dentistry, Schulich School of Medicine & Dentistry, Western University, London, Ontario, Canada.
Timucin AriDepartment of Paediatric Dentistry, Schulich School of Medicine & Dentistry, Western University, London, Ontario, Canada.
Gürkan ÜnsalDivision of Oral and Maxillofacial Radiology, Schulich School of Medicine & Dentistry, Western University, London, Ontario, Canada.
Tugce BalciDepartment of Paediatrics, Schulich School of Medicine & Dentistry, Western University, London, Ontario, Canada.ORCID 0000-0002-5409-8387
Matthew MorrisonDepartment of Oral Surgery, Schulich School of Medicine & Dentistry, Western University, London, Ontario, Canada.
Nilgun AriDepartment of Prosthodontics, Schulich Medicine and Dentistry, Western University, London, Ontario, Canada.ORCID 0000-0001-6632-4698

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

objectiveThis case report describes the prosthetic rehabilitation of a 15-year-old female patient with a rare IFIH1-related interferonopathy, presenting features from both Aicardi-Goutières Syndrome (AGS) and Singleton-Merten Syndrome (SMS). The report highlights the unique dental and maxillofacial challenges, and the multidisciplinary treatment approach required in such patients.

methodsThe patient underwent a comprehensive prosthetic rehabilitation, which included two stages of extractions, followed by the fabrication of a complete upper denture and a two-implant-retained mandibular overdenture. Pre- and post-treatment assessments, including panoramic radiographs and cone beam computed tomography (CBCT), were utilized to guide treatment.

resultsInitial assessments revealed root formation anomalies, multiple impacted teeth, internal resorption, and associated dental anomalies. Following extractions and ridge augmentation, two freestanding implants were successfully placed in the anterior mandible, with no complications during osseointegration. The patient reported satisfaction with the function, esthetics, and stability of her dental prostheses.

conclusionsThis case underscores the importance of a multidisciplinary approach for managing dental anomalies and bone fragility in patients with IFIH1-related disorders. Early genetic diagnosis and thorough treatment planning are critical for successful prosthetic rehabilitation and optimal patient outcomes.

Indexed as

Autoimmune Diseases of the Nervous SystemHeart Defects, CongenitalMouth RehabilitationNervous System MalformationsAdolescentAortic DiseasesCone-Beam Computed TomographyDental Enamel HypoplasiaDental Prosthesis, Implant-SupportedFemaleHumansMetacarpusMuscular DiseasesOdontodysplasiaOsteoporosisRadiography, PanoramicAicardi‐Goutières syndromedental anomaliesdental implantsgenetic disordersIFIH1interferonopathyprosthetic rehabilitationroot resorptionSingleton‐Merten syndrome

Identifiers

PMID40197712
PMCPMC11977043

What OpenQuestion holds

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.