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Record W2002095991 · doi:10.1007/s10227-005-0146-8

Familial Bullous Lichen Planus (FBLP): Pedigree Analysis and Clinical Characteristics

2005· article· en· W2002095991 on OpenAlexaff
Changzheng Huang, Siyuan Chen, Zhixiang Liu, Juan Tao, Chunsen Wang, Youwen Zhou

Bibliographic record

VenueJournal of Cutaneous Medicine and Surgery · 2005
Typearticle
Languageen
FieldDentistry
TopicOral Health Pathology and Treatment
Canadian institutionsUniversity of British Columbia
Fundersnot available
KeywordsMedicinePenetranceDermatologyMucocutaneous zoneOral lichen planusAge of onsetPedigree chartRare diseaseFamily aggregationDiseasePathologyGenetics

Abstract

fetched live from OpenAlex

Familial bullous lichen planus (FBLP) is a rare condition. The clinical features dearly have been described. We report the largest patient series of FBLP and describe its clinical characteristics and inheritance pattern. In this retrospective chart review, we analyzed nine consecutive familial pedigrees of FBLP with 36 affected individuals who presented to the Department of Dermatology at the Wuhan Union Hospital, a tertiary referral hospital in central China. Parameters analyzed include age of onset, gender predilection, lesional distribution, nail and mucosal involvement, clinical course, and inheritance pattern. Thirty-six of 85 individuals in the nine families were affected (42.4%). Females were more likely to be affected than males (58.3% vs 35.7%, Ġ(χ 2 = 3.99. P < 0.05). A bimodal disease onset was found, with one peak at 1–3 years and another at 13–17 years. The shin is the most commonly affected area (97%) followed by the upper limbs and the thighs. Involvement of the torso is relatively rare. Only a minority of cases involves the oral mucosa. The disease tends to follow a chronic and progressive course. The inheritance pattern is autosomal dominant with variable penetrance. Familial bullous lichen planus is a chronic, progressive bullous eruption of the lower and upper extremities. Compared with non familial bullous lichen planus, it has an earlier onset and wider disease distribution. It may be inherited as an autosomal dominant condition with variable penetrance. Le lichen plan bulleux familial (LPBF) est une condition rare. Les descriptions de ses manifestations cliniques sont limitées. Nous faisons état de la plus importante série de LPFB, et décrivons ses caractéristiques cliniques ainsi que ses schémas de transmission. Dans cette étude rétrospective, nous analysons neuf lignées familiales de LPFB auprès de 36 personnes atteintes qui se sont présentées au service de dermatologie de 1’hôpital Wuhan, un hôpital de recommandation tertiaire dans le centre de la Chine. Les paramèters analysés comprennent: 1’âge au déclenchement, le sexe, la distribution des lésions, l’atteinte des ongles et des muqueuses, 1’évolution clinique et les schémas de transmission. Trente six des 85 personnes appartenant aux neuf families étaient affectées, soit 42,4%. La condition touchait les femmes davantage que les homines (58,3% et 35,7% respectivement. χ 2 = 3,99, P < 0,05). On a remarqué un déclenchement bimodal de la maladie, avec un point culminant après 1 à 3 ans et un autre après 13 à 17 ans. Le tibia est la zone la plus fréquemment affectée (97%), suivie des membres supérieurs et des cuisses. L’affection du torse est plutôt rare. Une minorité de cas concernent la muqueuse buccale. La maladie est chronique et progressive. Elle se transmet en mode dominant avec une pénétrance variable. Le lichen plan bulleux familial est une éruption bulleuse chronique et progressive des extrémités inférieures et supérieures. L’âge au déclenchement est moins avancé que celui du lichen plan bulleux non familial; et sa distribution est plus étendue. Son mode de transmission est autosomique dominant avec pénétrance variable.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.001
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.088
Threshold uncertainty score0.478

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0010.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0000.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.046
GPT teacher head0.358
Teacher spread0.312 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one teacher head, not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designObservational
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations7
Published2005
Admission routes1
Has abstractyes

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