Syndrome in question

Rebellato, Priscila Regina Orso; Rezende, Camila Makino; Battaglin, Eveline Roesler; Lima, Brunno Zeni de; Neto, José Fillus

doi:10.1590/abd1806-4841.20153818

Cited by 8 publications

(5 citation statements)

References 10 publications

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“…2 Doxycycline in doses of 200 mg/day for 4-12 months was successful in five patients 3,4 but was ineffective in short duration treatment schedules irrespective of the histological presence of mast cell infiltration. 5,6 The small number and intrafollicular location of Demodex mites in our case could not validate the diagnosis of granulomatous rosacea-like demodicidosis. 7 The subantimicrobial dose of 40 mg/d doxycycline monohydrate is not associated with the side-effects of long-term high doses or the risk of resistance.…”

mentioning

confidence: 61%

“…5 We submitted the sample to immunohistochemistry using anti-COX-2 antibody (ST21; Cell Marque, Rocklin, USA; 1 : 1000; 60') and performed a semiquantitative analysis (0%score 0, negative; <20%score 1, low; 20-50%score 2, moderate; >50%score 3, high). 6 Pearson's chi-square, Fisher exact and nonparametric Mann-Whitney tests were used for statistical analysis, with a 5% level of significance.…”

Section: Editormentioning

confidence: 99%

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Morbihan disease: successful treatment with slow‐releasing doxycycline monohydrate

Chaidemenos¹,

Apalla²,

Sidiropoulos

2017

Acad Dermatol Venereol

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confidence: 61%

Section: Editormentioning

confidence: 99%

Morbihan disease: successful treatment with slow‐releasing doxycycline monohydrate

Chaidemenos¹,

Apalla²,

Sidiropoulos

2017

Acad Dermatol Venereol

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“…It can inhibit mast cell degranulation during the early stages of inflammation. It suppresses pathological edema and fibrosis by reducing the release of inflammatory factors IgE, tumor necrosis factor-α, interleukin (IL)-4, and IL-13[ 17 , 18 ]. The success of the combination therapy of minocycline and ketotifen in our patient may be a good option that reduces the duration of antibiotic administration, especially in the early stages of the disease.…”

Section: Discussionmentioning

confidence: 99%

Morbihan disease misdiagnosed as senile blepharoptosis and successfully treated with short-term minocycline and ketotifen: A case report

R¹,

Wu²

2023

World J Clin Cases

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BACKGROUND Morbihan disease is a rare skin condition with diagnostic and therapeutic challenges. Facial nonpitting erythematous edema is usually considered to be a characteristic manifestation and diagnostic clue for the Morbihan disease. Treatment of Morbihan disease remains a dilemma due to its long course, poor response, and high recurrence rate. CASE SUMMARY We report the case of a 69-year-old man with Morbihan disease. The patient presented ptosis as the first and main symptom. There was no obvious edema or other skin lesions. The patient was misdiagnosed with senile blepharoptosis based on eyelid performance and no treatment was administered to him. After continuous progressive asthenia of eye-opening and ptosis for more than one year, a skin biopsy was recommended. Histopathological analysis showed edema in the dermis, lymphatic hyperplasia and dilatation, and perivascular lymphocytic infiltration. An obvious increase in toluidine blue-stained mast cells was observed. The patient was finally diagnosed with Morbihan disease. Minocycline and ketotifen were prescribed based on the increase of mast cells in skin tissue slices. The patient experienced rapid relief seven days later and complete remission after 40 d from the commencement of the treatment. CONCLUSION Ptosis without obvious swelling could be the only or main clinical manifestation of Morbihan disease in rare conditions. An increase of mast cells was an important therapeutic clue to the rapid and remarkable efficiency of the combination therapy of minocycline and antihistamine.

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“…On the contrary, combination therapy with minocycline and tranilast given in a patient with no presence of mast cell infiltrates in histology remained ineffective (Nagasaka et al, ). Promising results have been demonstrated after long‐term isotretinoin monotherapy or systemic combination of isotretinoin with ketotifen (Smith & Cohen, ; Kabuto, Fujimoto, Honda, & Tanaka, ; Mazzatenta, Giorgino, Rubegni, Aloe, & Fimiani, ; Rebellato, Rezende, Battaglin, Lima, & Fillus Neto, ). Isotretinoin's mechanism of action in MD is postulated to be correlated with its anti‐inflammatory and immunomodulatory properties that make it also useful in treatment of rosacea (Smith & Cohen, ).…”

Section: Discussionmentioning

confidence: 99%

Efficacy of long-term intralesional triamcinolone in Morbihan's disease and its possible association with mast cell infiltration

Tsiogka

Koller

2018

Dermatologic Therapy

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Morbihan's disease is characterized by chronic persistent facial edema of the upper half of the face, absence of typical diagnostic findings, and refractoriness to treatment. A 44‐year‐old man was diagnosed with Morbihan's disease based on clinical signs and histopathology, which showed dermal edema in upper dermis, discrete lymphocytic infiltrate without granulomatous reaction, and mast cell infiltration. After long‐term therapy with intralesional triamcinolone a remarkable objective and subjective clinical response was observed. Reported cases of Morbihan's disease are reviewed, with respect to their treatment and histopathological findings. Mast cell infiltration has been observed on histopathology in most patients who responded to intralesional triamcinolone, suggesting a possible marker of response. The long‐lasting response seen in our case indicates the efficacy of intralesional triamcinolone in this rare condition.

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Syndrome in question

Cited by 8 publications

References 10 publications

Morbihan disease: successful treatment with slow‐releasing doxycycline monohydrate

Morbihan disease: successful treatment with slow‐releasing doxycycline monohydrate

Morbihan disease misdiagnosed as senile blepharoptosis and successfully treated with short-term minocycline and ketotifen: A case report

Efficacy of long-term intralesional triamcinolone in Morbihan's disease and its possible association with mast cell infiltration

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