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cutaneous plasmacytosis ( cp ) is a very rare , recently described entity in world literature .
there are < 60 cases reported worldwide , no case reported from india till date .
we report a case of primary cp in an adult female with site predilection of apocrine gland - bearing areas which has not been described previously in world literature .
a 46-year - old married female presented with asymptomatic , multiple , reddish - brown lesions in axillae , mammary area , lower abdomen , perineum , and upper inner thighs since last 2 years .
they started as small pea - sized lesions with a gradual progression in size and number . on general examination
cutaneous examination revealed multiple well defined erythematous skin to brown colored succulent - looking papulo - plaque lesions in the above - mentioned sites ranging from 0.5 to 10 cm in diameter [ figure 1 ] .
the vulva showed mainly papular lesions with a predominance of plaques in medial thighs which had atrophy as well .
initially , a presumptive clinical diagnosis of hidradenitis suppurativa ( hs ) was made observing the distribution areas , and she was put on antibiotics and topical benzoyl peroxide 5% , but the lesions did not show regression . after 2 weeks , she was put on oral isotretinoin 1 mg / kg for a period of further 2 weeks , but still there were no signs of regression in the lesions . moreover , new lesions kept on emerging .
hence , a biopsy was performed , and it revealed acanthosis , parakeratosis , mature polyclonal plasma cells with small uniformly sized nuclei present up to the deep dermis and fibrosis [ figure 2 ] .
interestingly , the folliculosebaceous apparatus was seen to be uninvolved at places with plasma cell infiltrates in dermis , excluding the diagnosis of hs .
( a - d ) multiple erythematous , red - brown infiltrated plaques distributed in apocrine gland bearing areas but not strictly bilateral .
well defined fibrosed plaques seen in upper medial thighs ( a ) perivascular and periadnexal patchy infiltrate of mature typical plasma cells admixed with some lymphocytes and histiocytes ( h and e , 10 ) .
( c ) lambda stain ( immunostain , 10 ) immunohistochemical examination revealed a polyclonal staining pattern with kappa to lambda ratio of 3:1 , with cd138 being positive [ figure 3 ] .
serum protein electrophoresis revealed polyclonality with levels of igg as 2300 mg / dl ( reference range 8801800 mg / dl ) , iga as 762 mg / dl ( reference range 126517 mg / dl ) , igm as 486 mg / dl ( reference range 52270 mg / dl ) , and ige as 980 mg / dl ( reference range 0450 mg / dl ) .
chest x - ray , ultrasound whole abdomen and pelvis and computed tomography scan did not reveal any significant abnormality .
bone marrow examination revealed a normocellular marrow with normal myeloid to erythroid ratio and plasma cells being 4% of marrow cellularity .
since no internal organ was involved and also there was no history of any previous cutaneous disease , the final diagnosis was confirmed as primary cp .
( a ) sparing of hair follicle with perifollicular plasmacytic infiltrate ( h and e , 400 ) .
( d ) lambda positivity ( immunostain , 400 ) she was put on prednisolone 40 mg / day and after 8 weeks of treatment , there was moderate effect in that there were no new lesions and size and elevation of old lesions regressed .
we have currently put her on monthly follow - up to monitor progression to systemic plasmacytosis ( pc ) , and it has been 1 year now she has not developed systemic involvement .
cutaneous and systemic plasmacytosis ( csp ) is a rare disorder with approximately 60 cases being reported worldwide , mostly from japan ; none from the indian subcontinent .
it consists of a triad of cutaneous lesions , superficial lymphadenopathy , and polyclonal hypergammaglobulinemia .
it was first reported in 1976 , with the first case in english literature being reported by watanabe et al . , in 1986 .
cutaneous lesions are characterized by disseminated reddish - brown macules and plaques distributed mainly on the trunk
. there may be systemic involvement with hepatosplenomegaly and constitutional symptoms , but the term systemic plasmacytosis is used in such cases .
extracutaneous involvement of lymph nodes , lungs , liver and spleen is also seen . among all the systemic sites ,
histologically , uhara et al . stated that the most characteristic histopathological change in cp is perivascular and periadnexal patchy infiltrate of mature typical plasma cells admixed with some lymphocytes and histiocytes .
cutaneous plasma cell infiltration of the skin can be due to various reasons ranging from neoplasms to infections .
the differential diagnoses to be considered are malignancies ( cutaneous plasmacytoma , marginal zone b - cell lymphoma , follicle center b - cell lymphoma ) , systemic lupus erythematosus , scleroderma , infections ( bacterial , fungal , mycobacterial , syphilis ) , plasma cell vulvitis , systemic plasmacytosis , nodular amyloidosis , and cutaneous rosai  dorfman disease . in this case , hs / acne inversa ( ai ) is an additional diagnosis to be considered . here , although significant fibrosis was present and the igg4 levels were normal , we favor the diagnosis of cp because : 
 absence of ruptured cysts , follicular involvement clinically in this patient , which characterize aiin the vulval area , predominant areas involved were upper medial thighs and not vulva proper .
there were erythematous succulent and fibrosed asymmetrical plaques in upper medial thighs , unlike the lesions of ai .
also , there was no predominant areolar area involvementthe infiltrate is predominantly non - specific lymphohistiocytic , plasma cells , mononuclear infiltrate , and neutrophilic granulocytes in ai , which is lacking here . no sinus tracts or late apocrine gland involvement
absence of ruptured cysts , follicular involvement clinically in this patient , which characterize ai in the vulval area , predominant areas involved were upper medial thighs and not vulva proper .
there were erythematous succulent and fibrosed asymmetrical plaques in upper medial thighs , unlike the lesions of ai .
also , there was no predominant areolar area involvement the infiltrate is predominantly non - specific lymphohistiocytic , plasma cells , mononuclear infiltrate , and neutrophilic granulocytes in ai , which is lacking here . no sinus tracts or late apocrine gland involvement was seen .
basically , the inciting event leading to proliferation of plasma cells has been unknown till date .
kodama et al . , detected increase in serum il-6 in systemic plasmacytosis and intralesional injection of corticosteroids has led to decrease in lesions in few patients .
we also are of the view that maceration and friction in these areas in predisposed individuals may lead to the development of cp in patients as seen in our patient .
uhara et al . , reported that serum immunoglobulin levels more than 5000 mg / dl as well as plasma cell counts in bone marrow more than 6.9% are associated with the more severe clinical course .
fatal outcomes are seen with csp , where renal , t - cell lymphoma , or interstitial pneumonia have been the cause of death . regarding treatment options available for cp ,
psoralen and ultraviolet a , radiotherapy , systemic chemotherapy ( cyclophosphamide , epirubicin , vincristine , and prednisone ) , intralesional steroids have all been tried with variable success rates . recently , topical 5-aminolaevulinic acid photodynamic therapy using long - pulsed ruby laser has been described as a novel treatment option .
we present the case due to its rarity and its unique distribution of lesions . moreover , on a closer look
, we can appreciate well- to ill - defined plaque lesions which partly involve areola but mainly the peri - areolar skin .
similarly , in vulval region , although the lesion distribution simulates hs , these types of lesions are different .
hence , a very meticulous observation of the lesions is very important as is very well highlighted in this case .
whenever cases of cp are encountered , search for underlying diseases leading to plasma cell infiltration as well as systemic plasmacytosis is warranted .
we report the first case from indian region with lesions of cutaneous plasmacytosis masquerading as hidradentis suppurativa , which has not been previously reported .
we report the first case from indian region with lesions of cutaneous plasmacytosis masquerading as hidradentis suppurativa , which has not been previously reported .
we report the first case from indian region with lesions of cutaneous plasmacytosis masquerading as hidradentis suppurativa , which has not been previously reported .
isolated cutaneous plasmacytosis ( cp ) is a rare entity with few cases reported in world literature . 
 cp masquerading as hidradenitis suppurativa like presentation is a unique case with some features differentiating it clinically from it which were further confirmed by histopathology and immunostaining . 
 our case showed hyperplasia of mature plasma cells and polyclonal hypergammaglobulinemia , immunostaining for cd138 positivity and kappa : lambda ratio more than 3:1 . 
 extensive clinical and laboratory investigations failed to reveal any underlying pathology , presence of any underlying disease accompanying the hypergammaglobulinemia and/or plasma cell proliferation .