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morbihan syndrome is a rare entity characterized clinically by chronic erythematous edema localized exclusively on the forehead , glabella , eyelids , and cheeks .
oral isotretinoin is considered the therapy of first choice , although not all patients improve .
a 60-year - old caucasian man was admitted to our department because of a dermatitis on the face .
the patient stated that he was in good general health , except for obesity ( 120 kg ) , and that he was not in therapy with systemic drugs .
the patient also stated that the dermatitis appeared approximately 4 years before and that it was unsuccessfully treated with oral corticosteroids .
dermatological examination revealed a severe erythematous edema involving the forehead , glabella and both eyelids , because of which the patient was not able to open his eyes completely .
furthermore , erythema and telangiectasiae were visible on the nose and cheeks [ figures 1 and 2 ] .
erythematous edema of the forehead , glabella , eyelids , and cheeks close up of same patient general physical examination did not reveal anything pathological .
all laboratory and instrumental examinations ( echography of periorbital area , carotids , and thyroid , chest x - rays , electrocardiogram , echocardiogram , and echography of the abdomen ) were within normal ranges or negative .
histopathological examination showed dermal edema , perivascular and periadnexal lympho - histiocytic infiltrate , and sebaceous gland hyperplasia .
the patient was treated with oral isotretinoin [ starting daily dosage : 0.5 mg / kg ( = 60 mg / day ) for 4 weeks ) ] .
morbihan syndrome was possibly first described in 1956 by schimpf , who published four case reports on this subject from 1956 to 1960.[24 ] unfortunately , none of these articles had any clinical photographs .
furthermore , the histopathological picture of these patients was characterized by granulomas , that are rarely present in morbihan syndrome . in 1972 ,
merklen , et al . named this entity as  infiltration massive persistante du front avec fort dme palpbral ,  i.e. , massive persistent infiltration of the forehead with severe edema of the eyelids . in 1991 ,
this name was due to the fact that the first french patient was observed in 1957 by degos in a farmer who came from the region of morbihan ( north - western france ) .
however , we do believe that the name of  morbihan syndrome ,  in consideration of possible different etiopathogenetic factors , is more correct .
caucasian adults of both sexes are affected : to our knowledge , only one black and one indian patient were reported .
morbihan syndrome is characterized clinically by the slow appearance of erythematous edema localized exclusively on the forehead , glabella , eyelids , and cheeks .
, it is a clinical variety or a complication of acne or rosacea . in our patient ,
the age , the clinical picture ( erythema and telangiectasiae on the nose and cheeks ) , as well as the histopathological picture ( sebaceous gland hyperplasia ) , support a correlation with rosacea .
furthermore , according to some authors , morbihan syndrome may be caused by lymph vessel abnormalities .
histopathological examination is nonspecific : it is characterized by dermal edema , perivascular and periadnexal lympho - histiocytic infiltrate , with more or less numerous mast cells , and dilated lymphatic vessels .
sebaceous gland hyperplasia may be observed in patients with previous or coexisting acne or rosacea .
biopsy for histopathological examination must be always performed because morbihan syndrome enters into differential clinical diagnosis with metastases to the periorbital area .
other differential diagnoses include congenital , infectious , inflammatory , neoplastic , and miscellaneous diseases [ table 1 ] .
furthermore , barbiturates , chlorpromazine , diltiazem , and even isotretinoin can induce clinical manifestations similar to morbihan syndrome .
differential diagnoses of morbihan syndrome * several systemic drugs were used for the therapy of morbihan syndrome .
. however , only isotretinoin , alone or associated with ketotifen , seems to be really effective .
it has to be used at high doses and for long periods of time . as previously mentioned , in our patient , isotretinoin , despite the long duration of the therapy ( 26 weeks ) , was ineffective .
oral isotretinoin is ineffective : in these patients , the clinical course of morbihan syndrome is uncertain , the disease can become chronic or can relapse .
we report a case of severe morbihan syndrome ( chronic erythematous edema of the upper portion of the face ) in a 60-year - old man . 
 the syndrome was characterized clinically by erythematous edema involving the forehead , glabella , and both eyelids , because of which the patient was not able to open completely his eyes . 
 furthermore , erythema and telangiectasiae were visible on the nose and cheeks . 
 laboratory and instrumental examinations were within normal ranges or negative . 
 histopathological examination showed dermal edema , perivascular and periadnexal lympho - histiocytic infiltrate , and sebaceous gland hyperplasia . 
 oral isotretinoin was ineffective despite the relatively long duration of the therapy ( 26 weeks ) .