Evidence map›Paper›PMID 38872283›Full record

ArticleThe American journal of case reports2024

Morbihan Disease: A Rare Case of Periorbital Bilateral Edema with Histopathological Findings of Chronic Inflammation and Demodex Localization.

Alessandra Di Maria, Filippo Confalonieri, Gianmaria Barone, Sofia Manara, Vanessa Ferraro

Abstract readCase Reports
In one paragraph

Article in The American journal of case reports, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

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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

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3 · Its place in the literature

Who cites it

2 citing papers in PubMed.

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4 · The record

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5 · Who and what money

Authors and funding

5 authors.

Alessandra Di MariaDepartment of Ophthalmology, IRCCS Humanitas Research Hospital, Milan, Italy.
Filippo ConfalonieriDepartment of Ophthalmology, IRCCS Humanitas Research Hospital, Milan, Italy.
Gianmaria BaroneDepartment of Ophthalmology, IRCCS Humanitas Research Hospital, Milan, Italy.
Sofia ManaraDepartment of Pathology, IRCCS Humanitas Research Hospital, Milan, Italy.
Vanessa FerraroDepartment of Ophthalmology, IRCCS Humanitas Research Hospital, Milan, Italy.ORCID 0000-0002-5018-1853

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

BACKGROUND Morbihan disease, also known as Morbihan syndrome, is a rare medical condition characterized by chronic facial edema predominantly affecting the upper two-thirds of the face. Despite being recognized in medical literature for decades, its true prevalence and underlying pathophysiology remain poorly understood. Various hypotheses, including impaired lymphatic drainage, abnormal vascular permeability, immune dysregulation, and inflammatory reactions to demodex infestation, have been proposed to explain the etiology. CASE REPORT We present a case of a 61-year-old man with organized periocular edema of the upper third of the face, ultimately leading to Morbihan disease diagnosis. The patient underwent a midface lift, allowing for tissue retrieval for histopathological examination of the eyelid edematous skin, which revealed chronic inflammation, ectasia of small lymphatic vessels, and features of demodex intrafollicular localization. These findings were not specific, but consistent with the diagnostic hypothesis. The patient was referred to a rheumatologist for further evaluation and treatment. He did not respond well to systemic corticosteroids and immunosuppressive therapy. Rather, this resulted in extension of the edema to the upper eyelid. The patient opted not to undergo further treatment. CONCLUSIONS Morbihan disease is often misdiagnosed due to its rarity and overlapping clinical features with other facial conditions. Its management is challenging and can require a combination of medical and surgical interventions. Systemic corticosteroids, immunosuppressive agents, and topical treatments have had varying success. Surgical procedures, such as blepharoplasty or laser therapy, can be considered in severe cases. Early recognition and appropriate management are crucial to improving patient outcomes and quality of life.

Indexed as

EdemaMite InfestationsAnimalsChronic DiseaseEyelid DiseasesHumansInflammationMaleMiddle Aged

Identifiers

PMID38872283
PMCPMC11185837

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