myxoglobulosis , a term coined by von hansemann in 1914 , is described as a variant of mucocele .
it is associated with the formation of opaque globules varying in diameter from 0.1 cm - 1.0 cm and lying in a clear mucous .
mucous retention cyst is a true cyst , pathogenesis of which can be due to ductal dilatation , secondary to ductal obstruction , leading to increased intraluminal pressure .
extravasation mucocele is a common lesion of the oral mucosa that results from rupture of a salivary gland duct , often the result of local trauma , and spillage of mucin into the surrounding connective tissue .
it is most frequently seen in association with the minor salivary glands with a site predilection for the lower lip .
the lumen contains extravasated mucin and is surrounded by granulation tissue wall with no epithelial lining .
our present case report reveals extravasation mucocele with unique intraluminal globules resembling those usually seen in appendix 
a 24-year - old female patient presented an asymptomatic swelling of lower lip which had gradually increased over a period of 6 months and measured about 1 cm  2 cm .
the swelling was dome - shaped , smooth - surfaced , soft in consistency and without discharge of any fluid .
histopathological examination of the excised tissue revealed a well demarcated cystic lumen in the sub - mucosa , surrounded by compressed granulation tissue forming the wall of the lumen with overlying atrophic epithelium [ figure 1 ] .
the cystic lumen showed a few muciphages and numerous globules ( approximately 100 in number ) .
the size of each globule varied , ranging from small to large ( 88 m to 416 m ) .
each globule exhibited a central , eosinophilic , amorphous and acellular core , surrounded by mucinous substance [ figures 2 and 3 ] .
the peripheral globules of the cystic lumen were in contact with the surrounding compressed granulation tissue wall , while some globules were also seen within the granulation tissue wall and the latter were more cellular compared to the centrally located globules in the lumen [ figure 4 ] .
the granulation tissue forming the cystic wall was vascular , fibrous and cellular , comprising of fibroblasts , endothelial cells and chronic inflammatory cells , of which , muciphages were predominant .
the adjacent minor mucous salivary glands showed varying stages of degeneration like loss of architecture of acini , and mild chronic sialoadenitis [ figure 5 ] .
photomicrograph showing cystic lumen in the submucosa with numerous globules , surrounded by granulation tissue wall and overlying epithelium ( h and e stain , 15 ) photomicrograph showing cystic lumen with globules having central eosinophilic , amorphous and acellular core surrounded by mucinous substance ( h and e stain , 200 ) photomicrograph showing cystic lumen , containing numerous globular organizations , spherical to polygonal in shape , with central eosinophilic , amorphous and acellular core surrounded by mucinous substance ( h and e stain , 100 ) photomicrograph showing some peripheral globules of the cystic lumen in contact with surrounding granulation tissue wall , and some within the granulation tissue wall .
the globules within the granulation tissue wall are more cellular ( h and e stain , 100 ) photomicrograph showing cystic lumen , containing globular organizations surrounded by the granulation tissue wall , and minor mucous salivary glands showing varying stages of degeneration like loss of architecture of acini , and mild chronic sialoadenitis ( h and e stain , 100 ) a 40-year - old male patient reported to our hospital with a chief complaint of painless swelling of lower lip near the right angle of mouth of 5 months duration with a history of local trauma .
no relevant medical history was elicited . on clinical examination , the swelling measured approximately 2 cm  1.5 cm in size , was pale in color , sessile , dome - shaped , soft in consistency , smooth - surfaced and fluctuant .
treatment included surgical excision of the lesion under local anesthesia and its submission for routine processing .
histopathological examination of the tissue revealed a cystic lumen in the submucosa containing mucin , few muciphages and a few unique mucinous globular organizations .
these globules were spherical to polyhedral in shape , 5 in number , with each globule having a central , amorphous , acellular area surrounded by mucin . in 3 of these globules ,
two of these globules were connected to the surrounding granulation tissue wall that formed the capsule .
the granulation tissue was highly cellular , comprising of chronic inflammatory cells ( lymphocytes , macrophages and muciphages ) , plump fibroblasts , proliferating endothelial cells and numerous dilated and congested vascular channels [ figures 6 and 7 ] .
photomicrograph showing cystic lumen containing globular organization that are spherical with surrounding cellular and vascular granulation tissue wall ( h and e stain , 100 ) photomicrograph showing cystic lumen containing globular organizations in contact with surrounding granulation tissue wall(h and e stain , 100 )
a 24-year - old female patient presented an asymptomatic swelling of lower lip which had gradually increased over a period of 6 months and measured about 1 cm  2 cm .
the swelling was dome - shaped , smooth - surfaced , soft in consistency and without discharge of any fluid .
histopathological examination of the excised tissue revealed a well demarcated cystic lumen in the sub - mucosa , surrounded by compressed granulation tissue forming the wall of the lumen with overlying atrophic epithelium [ figure 1 ] .
the cystic lumen showed a few muciphages and numerous globules ( approximately 100 in number ) .
the size of each globule varied , ranging from small to large ( 88 m to 416 m ) .
each globule exhibited a central , eosinophilic , amorphous and acellular core , surrounded by mucinous substance [ figures 2 and 3 ] .
the peripheral globules of the cystic lumen were in contact with the surrounding compressed granulation tissue wall , while some globules were also seen within the granulation tissue wall and the latter were more cellular compared to the centrally located globules in the lumen [ figure 4 ] .
the granulation tissue forming the cystic wall was vascular , fibrous and cellular , comprising of fibroblasts , endothelial cells and chronic inflammatory cells , of which , muciphages were predominant .
the adjacent minor mucous salivary glands showed varying stages of degeneration like loss of architecture of acini , and mild chronic sialoadenitis [ figure 5 ] .
photomicrograph showing cystic lumen in the submucosa with numerous globules , surrounded by granulation tissue wall and overlying epithelium ( h and e stain , 15 ) photomicrograph showing cystic lumen with globules having central eosinophilic , amorphous and acellular core surrounded by mucinous substance ( h and e stain , 200 ) photomicrograph showing cystic lumen , containing numerous globular organizations , spherical to polygonal in shape , with central eosinophilic , amorphous and acellular core surrounded by mucinous substance ( h and e stain , 100 ) photomicrograph showing some peripheral globules of the cystic lumen in contact with surrounding granulation tissue wall , and some within the granulation tissue wall .
the globules within the granulation tissue wall are more cellular ( h and e stain , 100 ) photomicrograph showing cystic lumen , containing globular organizations surrounded by the granulation tissue wall , and minor mucous salivary glands showing varying stages of degeneration like loss of architecture of acini , and mild chronic sialoadenitis ( h and e stain , 100 )
a 40-year - old male patient reported to our hospital with a chief complaint of painless swelling of lower lip near the right angle of mouth of 5 months duration with a history of local trauma .
the swelling measured approximately 2 cm  1.5 cm in size , was pale in color , sessile , dome - shaped , soft in consistency , smooth - surfaced and fluctuant .
treatment included surgical excision of the lesion under local anesthesia and its submission for routine processing .
histopathological examination of the tissue revealed a cystic lumen in the submucosa containing mucin , few muciphages and a few unique mucinous globular organizations .
these globules were spherical to polyhedral in shape , 5 in number , with each globule having a central , amorphous , acellular area surrounded by mucin . in 3 of these globules ,
two of these globules were connected to the surrounding granulation tissue wall that formed the capsule .
the granulation tissue was highly cellular , comprising of chronic inflammatory cells ( lymphocytes , macrophages and muciphages ) , plump fibroblasts , proliferating endothelial cells and numerous dilated and congested vascular channels [ figures 6 and 7 ] .
photomicrograph showing cystic lumen containing globular organization that are spherical with surrounding cellular and vascular granulation tissue wall ( h and e stain , 100 ) photomicrograph showing cystic lumen containing globular organizations in contact with surrounding granulation tissue wall(h and e stain , 100 )
mucoceles are the common non - neoplastic lesions of the oral minor salivary glands , of which , the majority are mucous extravasation cysts .
mucoceles develop due to the disruption of the flow of saliva from the secretory apparatus of the salivary glands .
the lesions are most often associated with extravasation of mucus into the adjacent soft tissues caused by a traumatic ductal insult . in our present cases , the clinical features and histopathological findings are suggestive of an extravasation mucocele .
although diagnosis of extravasation mucoceles is non - challenging to the pathologists , the intraluminal globular organizations of mucin surrounded by the granulation tissue capsule are histologically very unusual .
the etiology and pathogenesis of these globular organizations are still unclear . probstein and lassar stated that in myxoglobulosis of the intestine or appendix , the speculative etiological factors are bacteria and necrotic epithelial debris which may act as nidus for concentric deposition of mucin .
lubin and berle proposed that the core of the globules represents an organizing mass of mucin by granulation tissue originated from the appendical wall .
li et al . stated that the globules represent an attempt to organize the mucin by the granulation tissue capsule , which is then extruded into the lumen due to chronic frictional or mechanical forces .
ide and kusama stated that localized changes in the microenvironment may act as predisposing factors for formation of these florid globules .
myxoglobulosis of salivary mucocele probably occurs as a consequence of exuberant reparative process of the capsular granulation tissue wall in response to intraluminal accumulation of mucin .
lubin and berle reported 2 cases of myxoglobulosis of the appendix , which is a rare variant of extravasation mucocele .
histologically , the lesions consist of intraluminal globules which measure about 0.2 cm to 1.0 cm in diameter , and consist of granulation tissue , mucin , varying degrees of secondary focal calcifications and cholesterol clefts . in our present cases ,
the globules are similar to myxoglobulosis of appendix , except for their smaller size ( ranging from 88 m to 416 m ) , absence of calcification and cholesterol clefts .
the globules in both of our cases are almost similar in size , shape and contents , but the number of globules seen in case 1 are more and the surrounding granulation tissue wall is moderately cellular and vascular .
but in our case 2 , the globules are fewer in number and the surrounding granulation tissue wall is highly cellular and vascular . in the present case reports , the globules in the granulation tissue wall are cellular but the globules in the lumen are acellular , which is in accordance with ka shah , who stated that it is a time related phenomenon , with earlier globules being more cellular .
we conclude that these present cases represent a unique organization of mucin by granulation tissue wall as an exaggerated reparative response and release of the globules into the lumen due to chronic mechanical forces .
a somewhat similar analogy is seen in collagenous spherulosis that is frequently seen in benign and malignant salivary gland tumors , proliferative lesions of breast ductal epithelium , chondroid syringomas and schwannomas .
differentiating collagenous spherulosis from myxoglobulosis is comparatively easy , as the globules in collagenous spherulosis are seen at the tissue level , are myoepithelial in origin , acellular in nature , and almost always seen in association with epithelial and myoepithelial cells .
they are generally positive for collagen , p63 and basement membrane , and generally negative for mucin stains , whereas globules in myxoglobulosis are seen mostly in the lumen , are hyalinized , discrete , of variable cellularity and associated with granulation tissue wall .
their origin is from connective tissue and stains positive for acidic mucin and periodic acid schiff stain .
thus , it can be said that collagenous spherulosis and myxoglobulosis are the only morphologically related reaction patterns .
prognosis of this variant of extravasation mucocele will be better than other types of mucoceles with low recurrence rate due to good host response and globular organizations of mucin .
however , follow - up of these cases and more such cases is required to confirm the prognosis .
study of many cases is needed to confirm the etiology , pathogenesis and biologic nature of this variant .
 in this paper , we present two case reports of myxoglobulosis , in a 24-year - old female and a 40-year - old male patient who came to our hospital with a chief complaint of painless swelling of the lower lip of approximately 6 months duration . 
 a study of two case reports has been given here . 
 in these patients , histological examination of the surgically excised tissue was carried out . 
 histopathological examination showed an extravasation mucocele with the lumen exhibiting unique globular organizations of mucin surrounded by granulation tissue capsule and lacking an epithelial lining . 
 our two cases are possibly an analogue of myxoglobulosis , a rare variant of the appendical mucocele . 
 thus , though rare , the possibility of occurrence of myxoglobulosis in cystic lesions of the lip should be considered . 
 the prognosis is regarded better as compared to the other types of mucoceles with low recurrence rate due to good host response and globular organizations of mucin . 
 however , follow- up of these cases and more such cases is required to confirm the prognosis . 
 the need for study of many such cases to confirm the etiology , pathogenesis and biologic nature of this variant is being felt . 
INTRODUCTION
CASE REPORTS
Case 1
Case 2
DISCUSSION