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Home , Fordyce spots, Gingival cyst, Hairy leukoplakia, Lipochrome

Original Article Brunei Int Med J. 2013; 9 (5): 290-301

Yellow of the oral cavity: diagnostic appraisal and management strategies

Faraz MOHAMMED 1, Arishiya THAPASUM 2, Shamaz MOHAMED 3, Halima SHAMAZ 4, Ramesh KUMARASAN 5 1 Department of Oral & Maxillofacial , Dr Syamala Reddy Dental College Hospital & Research Centre, Bangalore, India 2 Department of Oral Medicine & Radiology, Dr Syamala Reddy Dental College Hospital & Research Centre, Bangalore, India 3 Department of Community & Public Health , Faculty of Dentistry, Amrita University, Cochin, India 4 Amrita center of Nanosciences, Amrita University, Cochin, India 5 Oral and Maxillofacial Surgery, Faculty of Dentistry, AIMST University, Kedah, Malaysia

ABSTRACT Yellow lesions of the oral cavity constitute a rather common group of lesions that are encountered during routine clinical dental practice. The process of clinical diagnosis and treatment planning is of great concern to the patient as it determines the nature of future follow up care. There is a strong need for a rational and functional classification which will enable better understanding of the basic process, as well as in formulating a differential diagnosis. Clinical diagnostic skills and good judgment forms the key to successful management of yellow lesions of the oral cavity.

Keywords: Yellow lesions, oral cavity, diagnosis, management

INTRODUCTION INTRODUCTI Changes in colour have been traditionally low lesions have a varied prognostic spec- used to register and classify mucosal and soft trum. The yellowish colouration may be tissue pathology of the oral cavity. Thus, the- caused by (the pigment of fat). It se lesions have been categorised as white, may also be the result of other causes such red, white and red, blue and/or purple, as accumulation of pus, aggregation of lym- brown, grey and/or black and yellow. 1-5 Yel- phoid tissue, exudation of serum, degenera- tion of blood pigments, lipid containing struc- tures, and extrinsic stains. Most Correspondence: Faraz Mohammed # 471, 14 th Cross, 8 th Main Wilson Garden, are harmless and do not require any treat- Bangalore – 560030 ment other than reassurance. But still a mi- Karnataka, India. E mail: [email protected] nority of these lesions are potentially danger- MOHAMMED et al. Brunei Int Med J. 2013; 9 (5): 291 ous if left untreated. Thus there is a need for a rational and functional classification which Table 1: Classification of lesions in the oral cavity with yellow colour. will enable better understanding of the basic disease process as well as in formulating a Nature of the Pathology disease differential diagnosis on a clinical basis. Such a classification will go a long way in improving Neoplasias Lipoma/liposarcoma the diagnostic acumen of the general dentist, Dermoid and epidermoid cysts Cysts and at the same time providing care for the Lymphoepithelial cyst Hyperplastic general population at large. The classification reactions for lesions/conditions in the dis- Infectious playing a ‘yellow colour’ is based upon nature Jaundice 5 Pigmental is as shown in Table 1. This article review deposits Carotenemia the various yellow lesions encountered in the Amyloidosis Metabolic oral cavity. disorders Hyalinosis cutis et mucosae Accessory lymphoid Lipoma aggregates Developmental Lipomas are the most common soft tissue alterations Yellow hairy benign tumours that are composed chiefly of adipose tissue. 6 Approximately 20% occur in the head and neck region and oral lipomas or with histology. However, it can resemble account for only between 1 and 4% of all cas- other conditions such submucous cysts es. 7 In the oral cavity, it is most commonly (lymphoid and lymphoepithelial cysts). Histol- located in the cheeks, floor of the mouth and ogy typically shows the presence of mature the tongue (Figure 1a). The first description adipose tissue. 10-12 of such was provided by Roux in 1848 in a review of alveolar masses which he re- Irrespective of location, the treatment ferred to it as a "yellow ". 8 When locat- is simple surgical excision, including a cuff of ed in the superficial plane, there is a yellow surrounding tissue to prevent local recurrenc- surface discolouration. The tumour has a less es. Advantages of suction-assisted lipectomy dense and more uniform appearance than for medium sized (4 to 10 cm) or large lipo- surrounding fibrovascular tissues when it is mas (>10 cm) have been reported. An infil- transilluminated. 9 Magnetic resonance imag- trating lipoma must be ‘de-bulked’ where a ing (MRI) scans are very useful in diagnosis portion of the infiltrating fat is deliberately left whereas computed tomography (CT) and ul- untouched in order to preserve as much nor- trasound scans are less reliable. mal tissue as possible. 13 Malignant transfor- mations or recurrences in the oral and maxil- Lipoma is composed predominantly of lofacial regions are rare. 14 Intramuscular li- mature adipocytes, possibly admixed with pomas have a higher recurrence rate because collagenic streaks, and is often well demar- of their infiltrative growth pattern, but this cated from the surrounding connective tis- variant is rare in oral and maxillofacial region. sues. It can be easily distinguished clinically Cao et al . reported recurrences in young pa- MOHAMMED et al. Brunei Int Med J. 2013; 9 (5): 292 tients (under 18 years old) and development They constitute between 1.6% and 6.9% of all of liposarcoma after several recurrences. cystic in the head and neck region. 30 The epi- Therefore, long-term follow-up is necessary in dermoid cyst, often mistakenly referred to as patients under 18 years old. 15 a sebaceous cyst or wen, is a very common skin lesion which arises from traumatic en- Liposarcoma trapment of surface . The epider- Liposarcoma, the malignancy of adipocytes, moid cyst of the oral mucosa is usually locat- was first described by Virchow in the 1860s. 16 ed on the attached gingiva, where it has tradi- It is a bulky yellow tumour (Figure 1b)similar tionally been called of adult. 30, 31 to a lipoma, but generally more complex and The oral dermoid cysts tend to be simple with contains areas of prominent sclerosis. 17 It is skin adnexa in the wall. In most cases, these the most common soft tissue sarcoma, com- cysts are treated by enucleation. 32 Akao and prising approximately 17% of all sarcomas, colleagues believed that intraoral approach and 3% of all liposarcomas occurring in the must be attempted first, even when dealing head and neck region. Early diagnosis and with a large cyst. This approach leads to good complete resection plays a key role in the cosmetic and functional results. 33, 34 Marsupi- treatment of liposarcoma. Liposarcoma alisation has also been proposed as an alter- demonstrates considerable microscopic varia- native treatment especially for giant cysts. 32 bility and the biological behaviour of the tu- When intraoral access is complicated, a com- mour is greatly dependent on the exact histo- bined intraoral and extraoral approach should pathologic appearance. In fact, Enzinger and be considered. Surgical excision is normally Weiss have stated that a diagnosis of liposar- achieved without major complications and coma without further qualification as to the prognosis is very good. 33, 34 exact histological type is meaningless. 18 Lymphoepithelial cyst The treatment of choice for liposarco- Oral lymphoepithelial cyst (so-called branchi- mas is surgical excision 19, 20 and the frequent al) is a rare lesion, which manifests as a pink- presence of satellite nodules means that wide ish to yellowish, small, asymptomatic, submu- surgical excision is necessary for adequate cosal mass (Figure 1c). Most cases occur in removal of the tumour. Despite aggressive regions of the oral cavity presenting lymphoid surgery, between 50 and 70% of tumours aggregates, such as the floor of the mouth recur locally. 21, 22 It has also been reported (65.3%) and the lateral and ventral surfaces that non-surgical treatments have limited val- of the tongue (13.7%). 35 However, they may ue, 23-25 but the role of radiotherapy have occur in the parotid gland. 36 Histologically, been report to reduce local recurrence after lymphoepithelial cysts exhibit a cystic cavity surgery. 26-28 Adjuvant chemotherapy has not lined by a parakeratinised stratified squamous been shown to be of value. 29 epithelium. The luminal space is filled with sloughed epithelial cells and the epithelial lin- Dermoid and Epidermoid cysts ing is devoid of rete ridges. Characteristically, Dermoid and epidermoid cysts are cystic mal- the fibrous capsule shows a dense lymphoid formations lined with squamous epithelium. tissue, with germinal centres. No treatment is MOHAMMED et al. Brunei Int Med J. 2013; 9 (5): 293

a b

c d

e f

g h

Figs. 1: a) Giant intramuscular lipoma of the tongue, b) Liposarcoma of the tongue, c) Lymphoepithelial cyst in buccal mucosa, d) Verruciform xanthoma, e) Pyostomatitis vegetans with gingival edema and pustules, f) in amyloidosis, g) Hyalinosis cutis et mucosae - Yellowish white papular deposits near the frenum, h) Fordyce spots, and i i) Yellow hairy tongue MOHAMMED et al. Brunei Int Med J. 2013; 9 (5): 294 usually required for oral lymphoepithelial cyst proximately 37 cases have been documented unless its location results in it frequently trau- in the literature. 43 Subsequently McCarthy matised leading to symptoms. However, in considered that this disorder to be a variant of most cases, the lesions are removed by con- pyodermatitis with oral location. This condi- servative surgical excision in order to arrive at tion is an unusual oral expression of inflam- a definitive diagnosis. 35 There is no malignant matory bowel disease. 44 It is a highly specific potential but the lymphoid stroma, as with all marker for inflammatory bowel disease and its lymphoid tissues, can become involved with correct recognition may lead to the diagnosis an extra nodal . There are currently of ulcerative colitis or Crohn’s disease. 43 no reports of neoplastic transformation or re- Thus, a presumptive diagnosis of pyostomati- currence after surgical excision. 37, 38 tis vegetans should include a complete gastro- intestinal investigation. Verruciform Xanthoma Verruciform xanthoma is a rare slow-growing The of pyostomatitis benign lesion of the skin and mucosa. It is vegetans is as yet unknown, although immu- characterised by a granular (verruciform) sur- nological and microbial factors have been sug- . Clinically, most are asymptomatic. To gested as possible aetiological factors. Pyosto- date, only 160 cases involving the oral cavity matitis vegetans is characterised by erythem- particularly on the have been reported. 39 atous, thickened oral mucosa with multiple It is yellowish-red or grey in colour and up to yellow or yellowish white friable pustules and 2 cm in diameter (Figure 1d). Histologically, a superficial erosions. 44 Buccal mucosa shows papillary and/or verrucous proliferation of the ‘cobble-stone’ appearance. A peripheral eosin- squamous epithelium with hyperparakeratosis ophilia has been observed in most cases re- and numerous foam cells is present. These ported. Histology shows epithelial acanthosis cells are predominantly located within the pa- and superficial ulceration with intraepithelial pillae of the lamina propria. In the differential and/or subepithelial abscesses containing diagnosis, other papillomatous and verrucous large numbers of eosinophils. The underlying lesions such as verrucous carcinomas or squa- connective tissue exhibits neutrophil and eo- mous cell carcinomas need to be ruled out. 39 sinophil infiltration, with miliary abscesses in Surgical excision is the treatment of choice. 40 some cases. Treatment of pyostomatitis vege- To date, only three cases of recurrence have tans focuses on control of the underlying gas- been reported. 41, 42 Malignant transformation trointestinal . 43 Surgical treatments in has not been reported in the literature. severe cases of inflammatory bowel disease involve total colectomy and have resulted in Pyostomatitis vegetans permanent remission of the oral lesions. 45 Pyostomatitis vegetans is a rare condition Oral lesions can be managed with local thera- characterised by pustules that affect the oral pies utilising antiseptic mouthwashes such as mucosa (Figure 1e). The name pyostomatitis chlorhexidine, and topical corticosteroids such vegetans was originally proposed by McCarthy as triamcinolone acetonide paste or betame- in 1949 after having observed that this lesion thasone mouthwash. Topical steroid therapy is isolated to the oral cavity. Since then, ap- has limited success. Strategic treatment ini- MOHAMMED et al. Brunei Int Med J. 2013; 9 (5): 295 tially consists of systemic steroid therapy but it can also be associated with the inges- aimed aimed at resolving and controlling the tion of many other yellow and green vegeta- lesions. 46, 47 A recent study reported that bles and citrus fruits. Carotene is a lipochrome three infliximab injections followed with that normally adds yellow colour to the skin. maintenance therapy with methotrexate can With elevated blood levels of carotene, the cause a rapid and complete regression of prominence of this yellowing is increased. 53 pyostomatitis vegetans. 48 Awareness of carotenemia is important to avoid confusion with jaundice and unneces- Jaundice sary diagnostic studies. Its clinical presenta- Excess bilirubin in the blood results in the ac- tion is as a yellowish pigmentation in the pal- cumulation of bilirubin in tissues, including the ate and, occasionally, in palms, soles and na- oral mucosa, producing a yellow discoloura- solabial fold. The absence of sclerae pigmen- tion. 49 The severity of the yellow discoloura- tation and the carotene serum level permit a tion depends on the blood concentration of differential diagnosis with jaundice. 54 bilirubin and the duration of the problem. Be- cause bilirubin has an affinity for elastin, the Amyloidosis mobile oral tissues with higher elastin content, Amyloidosis represents a group of conditions such as the lingual frenum and the soft pal- in which there is extracellular deposition of ate, are more severely affected. 50 Moreover amorphous fibrillar proteins, termed amyloid. the soft and the sublingual region are Clinically amyloidosis has the widest spectrum often first to reveal a yellow hue due to its of tissue and organ involvement. Amyloidosis loose and thin mucosa. 51 A yellowish to affecting the oral cavity tends to involve the greenish pigmentation (biliverdin deposition) buccal mucosa, tongue and gingiva (Figure occurs in the teeth of children with hyperbili- 1f). Oral involvement of amyloidosis has been rubinemia during calcification, as may be seen reported in 40% of patients with general amy- in the primary teeth of biliary atresia patients. loidosis. 55, 56 Oral localised amyloidosis is This is not seen in adults who develop liver quite uncommon. To our knowledge, only 13 disease after the enamel on the teeth has al- cases of oral localised amyloidosis have been ready calcified. reported to date. The nature of amyloid depo- sition in the oral cavity has long been the sub- Gastroenterologists may examine the ject of controversy. 57, 58 A final diagnosis of oral tissues to help in the clinical assessment oral amyloidosis is usually made on the basis of the extent of jaundice. However, care of clinical presentation else ware in the body. should be taken in assessing a yellowish dis- 59 The most common oral manifestation of colouration of the in patients re- amyloidosis is , which occurs in ceiving or eating large amounts of vitamin A, 20% of patients. The enlarged tongue demon- which is stored in the fat of the soft palate. 52 strates lateral ridging due to teeth indenta- tion. Grossly, the tongue may be firm and Carotenemia appear relatively normal or it may have yellow Carotenemia is a common finding in children, nodules on the lateral surface. 60 The submu- mainly due to the excessive intake of carrots, cosal masses of amyloid will be reflected clini- MOHAMMED et al. Brunei Int Med J. 2013; 9 (5): 296 ically as pale yellow hyperkeratotic excres- However, it was first described in 1908 by cences, especially in the cheek and lip areas. Seibenmann, a professor of otolaryngology in Interference with taste has also been reported Basel, Switzerland. Although this disease can in some patients, and hyposalivation may re- be found worldwide, almost a quarter of re- sult from amyloid deposition in the salivary ported diagnoses are from South Africa. 65 The glands. Submandibular swelling occurs subse- disease is characterised by diffuse deposition quent to tongue enlargement and can lead to of a hyaline-like substance in the dermis, respiratory obstruction. In addition to the oral submucosal connective tissue and various in- findings, evidence of amyloid deposits will be ternal organs. 67 Rough, yellowish-white papu- noted on the skin in such areas as the nose, lar deposits in the skin and oral mucosa usual- anus, and genitals. A typical sign of this disor- ly develop during childhood. 38 The oral muco- der is the presence of mucous folds by the sa of affected people later becomes nodular corners of the mouth that cause difficulties and thickened, with primary involvement of with chewing, swallowing or even talking. 61 the labial, buccal and palatal mucosa, posteri- Oral amyloidosis may be clinially difficult to or tongue, and lingual frenulum (Figure 1g). 67 distinguish from fibrous, neurogenous, or sali- With age the lesions become more yellowish vary gland pathosis. However histologic speci- and indurated. 69 The shows mens stained with Congo red have a charac- collection of hyaline-like material around the teristic ‘apple green’ appearance when exam- blood vessels a finding consistent with lipoid ined microscopically with polarised light. 62 proteinosis. This disease has a fluctuating When a dentist is requested to take a course. Although there have been many ther- specimen to detect amyloidosis, the specimen apeutic trials in hyalinosis cutis et mucosae, must include muscle tissue from the mucobuc- the treatment of this condition remains unsat- cal fold or tongue. isfactory. Generally, treatment include patient education and parents should be told about The management for localised forms the risk of having an affected offspring. Medi- such as involvement of tongue is typically with cal treatment includes oral steroids, dimethyl surgery. However in challenging cases, laser sulphoxide, intra-lesional heparin, and etreti- treatment may be employed. Alternatively, nate. 70 Surgical care involves resection of vo- the patient may simply be kept under obser- cal cord papules in improving vocal quality. vation. 63, 64 The prognosis for patients with the localised forms of amyloidosis is good, but Accessory lymphoid aggregates there is still a dearth of data. Accessory lymphoid aggregates or ectopic lymphoid tissue are tissue located away from Hyalinosis cutis et mucosae normal lymphoid tissue in the soft palate, floor Hyalinosis cutis et mucosae (lipoid proteinosis, of the mouth and tonsillar arches. The diagno- Urbach-Wiethe disease) is a rare syndrome sis is generally established relying on the clini- with autosomal recessive inheritance. There cal features. 71, 72 Oral manifestations include are fewer than 300 reported cases in the med- asymptomatic submucosal nodules which are ical literature. 65 It was first officially reported mobile yellow to white in colour and transpar- in 1929 by Erich Urbach and Camillo Wiethe. 66 ent. These lymphoid aggregates tends to be- MOHAMMED et al. Brunei Int Med J. 2013; 9 (5): 297 come prominent but most asymptomatic adenoma has a greater growth potential. 76 No where there is oral or poor oral hy- treatment is required for Fordyce granules, giene. Histopathological examination may except for cosmetic removal from the labial show aggregates of mature lymphocytes. The lesions. Inflamed glands can be treated topi- lymphocytes may show and pres- cally with clindamycin. When surgically ex- ence of germinal centres. 73 The benign lym- cised, recurrence does not occur. Neoplastic phoid aggregate requires no treatment, but in transformation is very rare. 77 cases where it is associated with poor oral hygiene or it can be treated with Yellow hairy tongue mild antibiotics and maintenance of oral hy- Hairy tongue (lingua villosa) is a commonly giene. Conversely, excisional biopsied may observed condition of defective desquamation been to be done in order to provide an appro- of the filiform papillae resulting from a variety priate diagnosis and to rule out lymphoma or of precipitating factors (Figure 1i). The preva- other malignancies. lence of hairy tongue varies widely, from 8.3% in children and young adults to 57% in Fordyce spots persons have drug addictions. 78 The condition Sebaceous glands are normal adnexal struc- is most frequently referred to as yellow hairy tures of the dermis but may also be found in tongue depending on the specific aetiology the oral cavity, where they are referred to as and secondary factors (e.g. use of coloured Fordyce spots/granules or ectopic sebaceous mouthwashes, breath mints, candies). 78 Pre- glands. 74 This variation of normal anatomy is cipitating factors for hairy tongue include poor seen in majority of adults, perhaps as much oral hygiene, the use of medications as 80%. However, they are seldom found in (especially broad-spectrum antibiotics) and large numbers. Fordyce granules appear as therapeutic radiation of the head and the rice-like, yellow-white, asymptomatic papules neck. All cases of hairy tongue are character- of 1-3 mm in greatest dimension (Figure 1h). ised by a and elongation of fili- There is no surrounding mucosal change and form papillae, with a lack of normal desqua- the granules remain constant throughout life. mation. Distinguishing between oral hairy leu- The most common sites of occurrence are the koplakia and hairy tongue is important espe- buccal mucosa (often bilateral), upper lip ver- cially in patients diagnosed or suspected to be milion, and mandibular retromolar pad and HIV positive. This can be accomplished by a tonsillar areas, but any oral surface may be simple mucosal punch biopsy and appropriate involved. Patients will have hundreds of gran- immunostaining of the specimen for the pres- ules while most have only one or two. Occa- ence of Epstein-Barr , the causative sionally, several adjacent glands will coalesce agent of oral hairy . 79 Culture of into a larger cauliflower-like cluster similar to the tongue's dorsal surface may be taken if a of the skin. In such an superimposed or other specific instance, it may be difficult to distinguish it oral infection is suspected. The treatment of from sebaceous hyperplasia or sebaceous ade- hairy tongue is variable. In many cases, simp- . 75 The distinction may be moot as both ly brushing the tongue with a toothbrush or entities have the same treatment, although using a commercially available tongue scraper MOHAMMED et al. Brunei Int Med J. 2013; 9 (5): 298 is sufficient to remove elongated filiform pa- gation to diagnose the disease at the earliest pillae and retard the growth of additional phase, which will reduce the morbidity and ones. Surgical removal of the papillae by us- mortality of the patients. The path to be taken ing electrodessication, carbon dioxide laser, or or the strategy to be adopted depends on the even scissors is the treatment of last resort judgment making capacity of the dental diag- when less complicated therapies prove inef- nostician . “Clinical diagnostic skills and good fective. 80 judgment forms the key to successful man- agement of yellow lesions of the oral cavity”. Conclusion The process of clinical diagnosis and treat- Acknowledgement ment planning of yellow lesions of oral cavity The authors gratefully acknowledge the assistance is of great concern to the patient as it deter- and support provided by the following research scholars in the form of constructive comments and mines the nature of future follow up care. clinical pictures of lesions: Professor Dr Giliyar With regard to clinical features, many issues Subraya Bhat, Professor, Department of Periodon- relevant to the management and understand- tics, College of Dentistry, University of Dammam, KSA; Dr Gaetano Magro, Department G.F. In- ing of disease behaviour are still unclear. grassia, University of Catania, Catania, Italy; Dr Spontaneous regression is observed in the Marika Dubin Russell, Assistant Professor, Depart- majority of patients with mild disease, where- ment of Otolaryngology, Columbia University – New York, USA; Dr Marceli Moço Silva, Department of as involvement of vital organs is burdened by Pathology and Clinical Propaedeutic, Ara atuba significant transience. School of Dentistry, Unesp - Univ Estadual Paulista, Brazil; Dr Rajiv Joshi, Consultant Dermatologist and Dermatopathologist, PD Hinduja Hospital, In spite of a large number of studies Mahim, Mumbai, India; Dr Marcello M. S. Nico, De- identified by this review, the presence of qual- partment of Dermatology, Medical School, Universi- ity follow-up data for these lesions remains ty of Sao Paulo, Sao Paulo, Brazil; Dr G. Saaveh, Assistant Professor, School of Dentistry, Ahvaz Uni- limited. Of the few studies where follow-up versity of Medical Sciences, Ahvaz, Iran; Assoc. data are reported, issues of definition of cases Prof. Asena Cigdem Dogramaci, School of Medicine, Mustafa Kemal University, Hatay, Turkey. (clinical vs. pathological) reduce the ability to draw stronger conclusions. REFERENCES

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