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scleromyxedema is a rare , chronic idiopathic disorder characterized by the appearance of numerous papules and areas of induration that are due to dermal mucin deposition in association with an increase in dermal collagen .
this entity must be distinguished from localized variants of lichen myxedematosus where skin is the sole site of involvement .
there are , however , occasional patients who have an atypical constellation of findings and fall between scleromyxedema and localized lichen gentleman who had findings of scleromyxedema and associated peripheral eosinophilia , but no monoclonal gammopathy .
a 59-year - old gentleman from punjab , india , presented with severe generalized pruritus and closely studded papules all over the body for 7 years with an exacerbation for 15 days .
the rash initially began as erythematous papules over the face , then progressed to involve the trunk and limbs .
general physical examination revealed cervical and axillary lymphadenopathy with 1 cm - sized , mobile , firm and multiple lymph nodes .
systemic examination was normal , apart from a palpable liver ; 6 cm below the costal margin .
cutaneous examination revealed numerous , 3 - 4 mm sized , closely spaced papules , which were firm and waxy , arranged in a widespread symmetrically distributed pattern all over the body including the scalp , and sparing only the mucosae and genitalia .
the glabella was typically involved with deep longitudinal furrowing and madarosis , giving the appearance of leonine facies .
skin over the flexures was thrown into folds , however mobility was not restricted . overlying the proximal interphalangeal joints , a doughnut sign was visible as a central depression surrounded by an elevated rim of infiltration due to skin thickening [ figure 1 ] .
infiltration of the face resulting in leonine facies , coalescing sheets of papules all over the body with thedoughnut sign visible on the fingers and the skin of the flexures thrown into folds abnormal investigations included a high total leukocyte count of 36,800/mm , with an absolute eosinophil count of 31,000/mm .
high - resolution computed tomography ( hrct ) of chest showed mediastinal and axillary lymphadenopathy .
the upper and mid dermis showed a moderately dense chronic inflammatory cell infiltrate of lymphocytes , plasma cells and histiocytes . in the mid dermis , collagen bundles showed fragmentation and splitting surrounded by loose myxoid stroma and plump fibroblastic cells , consistent with the diagnosis of scleromyxedema [ figure 2 ] .
collagen bundles with fragmentation and splitting surrounded by loose myxoid stroma and plump fibroblastic cells all over the mid - dermis , hematoxylin and eosin 10
scleromyxedema , also known as arndt gottron syndrome or generalized lichen myxedematosus , is a rare disease , etiology of which is not yet known .
it is a chronic and progressive disease , in which response to therapy has been inconsistent .
diagnostic criteria of scleromyxedema include a generalized papular eruption and sclerodermoid features , histological features of mucin deposition , fibroblast proliferation and fibrosis , monoclonal gammopathy on serum protein electrophoresis and the absence of thyroid disease .
accentuation of facial folds and creases may distort the features giving the appearance of leonine facies .
as the condition progresses , skin stiffening , sclerodactyly and decreased mobility of joints is seen .
nail fold and mat - like telangiectasias , and calcinosis , as seen in systemic sclerosis are absent . the skin in scleromyxedema moves over the subcutaneous tissue , unlike in scleroderma .
the monoclonal gammopathy is usually igg with lambda light chains seen in 80% patients . the paraproteinemia is said to either stimulate the production of hyaluronic acid and prostaglandin e by fibroblasts or represents an immunologic response to dermal mucin .
the patients who do not have paraproteinemia should be followed up for its presence for years .
patients of scleromyxedema can have a number of internal manifestations , in particular- muscular , neurologic , rheumatologic , pulmonary , renal and cardiovascular involvement .
staging of the disease has been proposed , with stage i being limited cutaneous papular mucinosis , stage ii is generalized cutaneous mucinosis and/or extracutaneous manifestation(s ) and stage iii generalized cutaneous mucinosis and/or extracutaneous manifestation(s ) and disease related karnofsky ps < 50% .
the characteristic histopathologic triad includes diffuse dermal mucin deposition , increased collagen and numerous irregularly shaped fibroblasts .
treatment is difficult ; melphalan is the most commonly used therapeutic agent ; high doses have been recommended for stage ii and iii patients .
other therapies include steroids , methotrexate , cyclophosphamide , thalidomide , cyclosporine , oral retinoids , extracorporeal photopheresis and plasmapheresis .
iv immunoglobulins can be considered as a well tolerated and efficacious second line therapy and also as first line therapy in cases of acutely worsening neurologic function .
high - dose melphalan along with autologous stem cell therapy was found to be effective .
beam regimen ( bcnu , etoposide , cytarabine and melphalan ) has been recommended as the conditioning prior to autologous stem cell transplantation ( asct ) for prolonged remission . the level of evidence ascribed to all these therapies is level iii , as this is a rare disease .
scleromyxedema can also present with severe pruritus , scalp involvement , unrestricted mobility and associated eosinophilia without paraproteinemia .
scleromyxedema is a rare , chronic and persistent idiopathic disorder characterized by a generalized papular eruption due to dermal mucin deposition with an increase in dermal collagen . 
 patients usually have associated paraproteinemia . 
 we describe the case of a 59-year - old gentleman with features of scleromyxedema , who had severe pruritus , scalp involvement , unrestricted mobility and associated peripheral eosinophilia , but no monoclonal gammopathy .