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the tumor was named aggressive due to its characteristically slow and insidious growth as well as carrying a moderate - to - high risk of local relapse .
usually , it presents as a vulval polyp clinically and is diagnosed only on histopathology .
it is a rare local mesenchymal tumor of unknown etiology usually affecting vulva , perineal region , buttocks , or pelvis of women in reproductive age .
considering its nature of aggression and chance of local relapse , appropriate management and long - term follow - up are necessary to diagnose early recurrence .
no single modality of treatment of recurrence has been found to be of proven benefit till now .
radiotherapy and chemotherapy have been used as adjunctive therapies but are unlikely to be useful as it has few mitotic activity . despite the availability of many options of treatment , recurrence of aa
a 40-year - old female para 2 presented with a swelling on the right labia majora with a duration of 3 years growing slowly throughout and increased in size for 6 months [ figure 1 ] . pedunculated large , partly encapsulated growth from vulva there was no history of any vulval discharge , bleeding , sexual difficulty , or pain , except a sensation of weight hanging while standing .
menstrual cycles were regular with a normal flow . local examination revealed a well - circumscribed pedunculated fleshy polypoidal mass measuring 18 cm  10 cm .
ultrasonography ( usg ) of the swelling revealed heterogeneous hyperechoic areas with peripheral vascularity , thick echoes , and nonvascular central areas .
computed tomography ( ct ) scan of pelvis revealed no pelvic disease inside and had similar findings of the mass like usg . with a clinical diagnosis of a vulvar fibroepithelial polyp or lipofibroma , she underwent local excision of the tumor with ligation of the stalk [ figure 2 ] .
the cut surface revealed a glistening , gelatinous , and soft homogeneous appearance [ figure 3 ] . on histopathology ,
the tumor was composed of spindle- and stellate - shaped cells scattered in a myxoid background [ figures 4 and 5 ] .
, there would be spindle cells in fatty background with minimum of vessels unlike aa .
there were many variable - sized thin - walled and thick - walled vascular channels in the histopathological specimen , which were diagnostic of aa .
a 6 monthly follow - up has been done for more than 2 years now without any specific therapy for the prevention of recurrence , yet showing no sign of any relapse so far .
cut open specimen of angiomyxoma showing homogeneous white area high power picture of angiomyxoma , ( h and e , 40 ) immunohistochemistry picture of angiomyxoma , cd34 ( low power ) postoperative picture of vulva after resection of angiomyxoma
the aa tumor commonly presents as an asymptomatic mass in the genital area of women in their reproductive life , but is occasionally reported in men ( male - to - female ration being 1:6 ) . the term  aggressive  denotes its propensity for local aggression and recurrence after excision .
clinically , aa may misdiagnosed as bartholin cyst , lipoma , labial cyst , gartner duct cyst , etc .
superficial angiomyxoma , angiomyofibroblastoma , cellular angiofibroma , and smooth muscle tumors also need to be considered in the differential diagnosis of a polypoidal mass in the perineum .
in addition , aa has thick - walled vessels , which are less numerous than thin - walled vessels in angiomyofibroblastoma .
on ct scan , these tumors have a well - defined margin with attenuation less than that of muscle .
the attenuation on ct and high signal intensity on magnetic resonance imaging ( mri ) are likely to be due to the presence of loose myxoid matrix and high water content of aa .
usually , this tumor does not metastasize , but there are reports of multiple metastases in women treated initially by excision and ultimately succumbing to it .
this hormonally responsive tumor is believed to arise from specialized mesenchymal cells of the pelvic  perineal region or from the multipotent perivascular progenitor cells , which often display variable myofibroblastic and fibroblastic features .
immunohistochemically , most aa express different combinations of estrogen and progesterone receptors , vimentin , desmin , smooth muscle actin cd34 , and cd44 , but all are invariably negative for s-100 , carcinoembryonic antigen , and keratin .
recent cytogenetic and molecular studies have revealed a variety of genetic alterations , involving the chromosome 12 , in the region 12q13 - 15 . a gene in this region , called high - mobility group protein isoform i - c ( hmgi - c ) , which encodes protens involved in the transcriptional regulation , appears to have a role in the pathogenesis of this tumor .
detection of inappropriate hmgi - c expression using the immunoperoxidase technique with anti - hmgi - c antibody may potentially be a useful marker for microscopic residual disease .
unlike most aas , our case was completely encapsulated without any breach in continuity and without any projections into the neighboring tissues .
this may be the reason why there was no recurrence in the past 2 years or so .
considering her very poor economic status and the hpe report , we decided not to start any preventive therapy , i.e. , gnrh agonist , etc .
, in her case . on h and e staining , the tissue resembled a typical aa , and immunohistochemically , it showed positive for cd34 , desmin , and vimentin , hence proving beyond any doubt that it was a case of vulval aa .
our patient required no additional treatment or any investigations postoperatively till date and has been asymptomatic and enjoying good health . despite this fact , aa is notorious for local recurrence in approximately 70% of the cases after a period of 2 years postoperatively , and it has been reported even 20 years after surgery as well .
proposed the following guideline for treating aa : ( 1 ) complete excision of the lesion when possible , avoiding mutilating surgery , ( 2 ) adjunct therapy using arterial embolization and/or hormonal treatment needed in case of partial resection of the tumor , and ( 3 ) radiotherapy is reserved for cases that are resistant to embolization and/or hormonal therapy and still symptomatic .
there are no specific guidelines for postoperative management of vulvar aa ; however , due to high recurrence rate and potential morbidity associated with undiagnosed recurrences , several authors recommend a periodic evaluation with physical examination and mri up to 15 years after excision .
this case report illustrates the challenges that a physician might face when dealing with a vulvar mass which may be an aa .
though it is a rare entity , it should always be considered , especially when it is an insidious painless lesion , particularly in premenopausal women in their third to fourth decades of life .
all relevant hematological and radiological studies including mri or ct scan should help in reducing the number of misdiagnosed cases of aa preoperatively .
once its anatomical location and extension , if any , are defined , any vulvar tumor  particularly aacan be optimally treated by surgical excision only , while avoiding any mutilating surgery .
if complete resection is possible under the circumstances , one should expect lowest recurrence rate .
aa is rarely life - threatening , and therefore one can afford to have a partial resection when high operative morbidity is anticipated .
irrespective of treatment modalities instituted postsurgery , it is evident that aa requires close and long - term follow - up .
aggressive angiomyxoma is a rare , locally invasive mesenchymal tumor predominantly presenting in women of reproductive age and also having a moderate - to - high risk for local relapse . 
 hence , it needs to be differentiated from other mesenchymal tumors occurring in this region . 
 we present here a case of a 40-year - old female presenting with a large , fleshy , pedunculated mass on the right labia majora .