British Journal of Dermatology (1975) 93) 399.
Median rhomboid glossitis CANDIDIASIS AND NOT A DEVELOPMENTAL ANOMALY* BRIAN E.D.COOKE Department of Oral Medicine and Oral Pathology, Welsh National School of Medicine, Dental School, Heath, Cardiff Accepted for publication 23 December 1974
SUMMARY
Infection by Candida Albicans was found in all biopsies taken from ten patients presenting with midline lesions of the tongue which clinically presented as median rhomboid glossitis.
The subject of this paper concerns a relatively uncommon anomaly of the tongue to which the rather grandiloquent title of 'Glossite losangique mediane de la face dorsale de la langue' was first given by Brocq & Pautrier (1914). In common with many other conditions where the diagnosis can be made confidently on the clinical features alone, biopsies are rarely performed and there is often only speculation regarding the underlying pathology. On a priori evidence Martin & Howe (1938) propounded the very plausible theory that median rhomboid glossitis results from the persistence of the tuberculum impar on the surface of the tongue. It was the striking appearance of the lesion that not unnaturally provoked cancerophobia in the mind of the patient and resulted in advice being sought. Just occasionally there was discomfort, generally in the form of a burning sensation, and if it persisted the lesion was excised. The diagnosis of median rhomboid glossitis has not been a comfortable one and the purpose of this study is to provide support for those who believe that the condition is infiammatory and not developmental and is in fact a pattern of chronic hyperplastic candidiasis. Sammett in 1939, in the most unlikely of all journals for this subject, namely the Radiology, set out six diagnostic clinical features: (a) (b) (c) (d) (e) (f)
The characteristic location in the median raphe anterior to the circumvallate papillae. Its roughly rhomboidal pattern. Its rosy colour. Its smooth common nodular or fissured surface devoid of papillae. Its slight induration. Its asymptomatic character and chance discovery.
* Part of the Presidential Address to the Section of Odontology of the Royal Society of Medicine, 28 October 1974-
399
400
B.E.D.Cooke
For some time, I have been puzzled by the many variations that can occur within these six diagnostic features for certainly, though all the lesions are generally in the median raphe, they are not always just anterior to the circumvallate papillae and may appear well forward in the midline of the dorsum of the tongue. The lesions are not always asymptomatic and increasingly they are the cause of discomfort and particularly a burning sensation. Certainly they are always without papillae but may vary in colour from the bright red, to red flecked with white, to frankly white patches within the well demarcated zone (Eigs i and 2). To the best of my knowledge the first serious suggestion that median rhomboid glossitis was not a developmental anomaly appeared in a paper by R.A.Baughman
FIGURE r. Male aged 60 years. Chronic candidiasis with midline fissure of dorsum of tongue.
in 1971 and he favoured the lesion to be either infiammatory, infective or degenerative in nature. In his paper he reviewed the literature and noted that no case had ever been reported in a child, and he himself examined the tongues of 10,010 children from kindergarten to the 12th grade at school and did not find a single example. He also could not find any evidence of median rhomboid glossitis in the tongues of 184 cadavers. He then examined the histology of seven cases labelled as median rhomboid glossitis in the files. All were Caucasians, six were female, one male and all aged between 39 and 62 years. His study of the histological features revealed similar findings to three cases described by the writer in 1962. There is parakeratosis of the stratified squamous epithelium devoid of papillae. There are deep branching epithelial ridges described as test-tube like and the subepithelial foci of lymphocytes are the most striking feature. There is no atypia of the epithelium and the supramuscular sclerosis is probably normal to the median raphe of the tongue. I think that the first reference
Median rhomboid glossitis
401
FIGURE 2. Female aged 35 years with acute pseudo-membranous candidiasis superimposed on chronic candidiasis of midline of dorsum of tongue.
to the possibility that this is a candidal lesion is made by J.J.Pindborg in the Textbook of Dermatology (2nd edn) edited by Rook et al. (1972). Patients Ten patients presented with chronic median glossitis. Six were female, four male, and their ages ranged from 30 to 63 years, with four in the fourth decade, three in the fifth, and three in the seventh. All the patients complained of discomfort, mainly a burning sensation and these symptoms were present for up to a year before examination. The lesions ranged from a position just anterior to the circumvallate papillae to the junction of the anterior and the middle third of the tongue, but all
B.E.D.Cooke
402
were in the midline. They varied from being smooth to grossly nodular and fissured. There was loss of suppleness within them all and with a degree of induration in some. There were varying degrees of a superimposed acute candidiasis so that the surface would sometimes appear thrushlike in part or in whole. One of the patients gave a history of having had warts removed from the dorsum of the tongue 3 years previously and presented with four or five papules about 3 mm in diameter in the middle of the dorsum of the tongue. As the condition was recognized to be probably a form of chronic candidiasis, antifungal therapy was prescribed for these patients but with very limited success. Histopathology
Candidal hyphae were found within the superficial layers of the parakeratotic epithelium in all cases (Fig. 3). Where there was a superimposed acute candidiasis there was a plaque of epithelial cells and chronic infiammatory cells invaded by the Candida albicans superimposed upon the parakeratotic epithelium (Fig, 4). Test-tube like deep epithelial ridges, some of which branched, were also very striking features. Furthermore, there were foci of lymphocytes closely related to the
FIGURE 3. Candidal hyphae in parakeratotic epithelium from lesion illustrated in Fig. i (PAS x 40).
Median rhomboid glossitis
403
FIGURE 4. Candidal hyphae within the superficial plaque of epithelial cells and within the parakeratotic epithelium of lesion illustrated in Fig. 2 (PAS x 40).
epithelial ridges, particularly near the tips and there were many dilated capillaries within the corium between the epithelial ridges (Fig. 5). The degree of oedema between the superficial prickle cells varied from case to case, but in one there were micro-abscesses filled with polymorphonuclear leukocytes. The intercellular oedema and the infiltrate of lymphocytes and occasional polymorphonuclear leukocytes disturbed the arrangement of the prickle cells, but no atypia was observed. DISCUSSION
In 1966 Cawson described the clinical and histological features of chronic oral candidiasis and made three important points; first, the presence of hyphae invading the epithelium was a positive finding that cannot easily be ignored; secondly, the infection was associated with a fairly consistent set of histological changes; and thirdly, Candida was not seen to be superimposed on some other recognizable pathological process. As in his cases, so in those under discussion, the tendency of the superficial epithelial layers to separate once candidal infection was established meant that the number of hyphae observed in the PAS sections varied considerably from just a few to a heavy invasion.
404
B.E.D.Cooke
FIGURE 5. Parakeratotic and acamhocic epithelium wich 'a-sl-lube like' deep epithelial ridges and foci of lymphocytes within the corium from the lesion illustrated in Figs i and 3.
The site of election suggests that the foramen caecum and perhaps the depression of the midline raphe of the tongue favour the localization of candidal infection. From these sites it then spreads laterally and is characterized by its chronicity. There is little doubt that the loss of the papillae is permanent and that the histoiogical changes induced by the candidal hyphae are irreversible and permanent. Otherwise, how could the histological appearances be described in such a uniform manner by so many different authors over the years ? The slides illustrated in Figs 3 and 4 of the author's paper (1962) have been stained with PAS and candidal hyphae have been identified in the parakeratotic epithelium. Of the greatest relevance have been the observations made by Jones & Russell (1974) in a recent paper entitled 'The histology of chronic candidal infection of the rat's tongue'. The changes they observed in the lingual epithelium were very similar to the findings just described, with the addition that some dysplasia of the epithelium was noted, together with muscle changes as seen in the regenerative phases following damage to muscle. This, the authors presumed, might eventually lead to extensive sub-epithelial fibrosis and alter the ability of the oral mucosa to respond to any potentially harmful stimulus. No treatment is either necessary or efiective in those asymptomatic cases with a smooth, nodular or fissured surface devoid of papillae. However, in those lesions with a fissured surface associated with a persistent painful glossitis, excision is indicated. Anti-fungal therapy would appear only to correct any superimposed pseudo-membranous candidiasis, although it may well be efficacious in the treatment of the early lesions, so preventing the condition becoming permanent. In conclusion, therefore, it is submitted that the condition that has been described as median rhomboid glossitis is chronic hyperplastic candidiasis and not a developmental anomaly.
Median rhomboid glossitis
405
ACKNOWLEDGMENTS I am grateful to Mr John Wolfie and Mr Russel Hopkins for undertaking the surgical treatment of many of the patients referred to in this paper; Mr R.T.Allison for technical assistance in the preparation of the sections and photomicrographs; and to Mr B.A.Jones of the Dental Audio-visual Aids Unit for the clinical photographs.
REFERENCES BAUGHMAN, R.A. (1971) Median rhomboid glossitis: a developmental anomaly? Oral Surgery, 31, 56. BBOCQ, L . & PAUTRIER, L.M. (1914) Glossite losangique mediane de la face dorsale dc la langue. Atmali di DermaTologia e Sifilologia (Paris), 5, i. CAWSON, R.A. (1966) Chronic oral candidiasis and leucoplakia. Oral Surgery, 22, 582. CooKE, B.E.D. (1962) Median rhomboid glossitis and benign glossitis migrans (Geographical tongue). British Denial Journal, I12, 389. JONES, J.H. & RUSSELL, C. (1974) The histology of chronic candidal infection of the rat's tongue. Journal of Pathology, 113, 97. MARTIN, H.E. & HOWE, M . E . (193S) Glossitis rhombica mediana. Annals of Surgery, 107, 39. PiNDBORG, J.J. (1972) Disorders of the oral cavity and lips. In: Textbook of Dermatology (Ed. by A.J.Rook, D.S.Wilkinson and F.J.Ebling) 2nd edn. Vol. 2, p. 1712. Blackwell Scientific Publications, Oxford. SAMMETT, J.F. (1939) Median rhomboid glossitis. Radiology, 32, 215.
Median rhomboid glossitis CANDIDIASIS AND NOT A DEVELOPMENTAL ANOMALY* BRIAN E.D.COOKE Department of Oral Medicine and Oral Pathology, Welsh National School of Medicine, Dental School, Heath, Cardiff Accepted for publication 23 December 1974
SUMMARY
Infection by Candida Albicans was found in all biopsies taken from ten patients presenting with midline lesions of the tongue which clinically presented as median rhomboid glossitis.
The subject of this paper concerns a relatively uncommon anomaly of the tongue to which the rather grandiloquent title of 'Glossite losangique mediane de la face dorsale de la langue' was first given by Brocq & Pautrier (1914). In common with many other conditions where the diagnosis can be made confidently on the clinical features alone, biopsies are rarely performed and there is often only speculation regarding the underlying pathology. On a priori evidence Martin & Howe (1938) propounded the very plausible theory that median rhomboid glossitis results from the persistence of the tuberculum impar on the surface of the tongue. It was the striking appearance of the lesion that not unnaturally provoked cancerophobia in the mind of the patient and resulted in advice being sought. Just occasionally there was discomfort, generally in the form of a burning sensation, and if it persisted the lesion was excised. The diagnosis of median rhomboid glossitis has not been a comfortable one and the purpose of this study is to provide support for those who believe that the condition is infiammatory and not developmental and is in fact a pattern of chronic hyperplastic candidiasis. Sammett in 1939, in the most unlikely of all journals for this subject, namely the Radiology, set out six diagnostic clinical features: (a) (b) (c) (d) (e) (f)
The characteristic location in the median raphe anterior to the circumvallate papillae. Its roughly rhomboidal pattern. Its rosy colour. Its smooth common nodular or fissured surface devoid of papillae. Its slight induration. Its asymptomatic character and chance discovery.
* Part of the Presidential Address to the Section of Odontology of the Royal Society of Medicine, 28 October 1974-
399
400
B.E.D.Cooke
For some time, I have been puzzled by the many variations that can occur within these six diagnostic features for certainly, though all the lesions are generally in the median raphe, they are not always just anterior to the circumvallate papillae and may appear well forward in the midline of the dorsum of the tongue. The lesions are not always asymptomatic and increasingly they are the cause of discomfort and particularly a burning sensation. Certainly they are always without papillae but may vary in colour from the bright red, to red flecked with white, to frankly white patches within the well demarcated zone (Eigs i and 2). To the best of my knowledge the first serious suggestion that median rhomboid glossitis was not a developmental anomaly appeared in a paper by R.A.Baughman
FIGURE r. Male aged 60 years. Chronic candidiasis with midline fissure of dorsum of tongue.
in 1971 and he favoured the lesion to be either infiammatory, infective or degenerative in nature. In his paper he reviewed the literature and noted that no case had ever been reported in a child, and he himself examined the tongues of 10,010 children from kindergarten to the 12th grade at school and did not find a single example. He also could not find any evidence of median rhomboid glossitis in the tongues of 184 cadavers. He then examined the histology of seven cases labelled as median rhomboid glossitis in the files. All were Caucasians, six were female, one male and all aged between 39 and 62 years. His study of the histological features revealed similar findings to three cases described by the writer in 1962. There is parakeratosis of the stratified squamous epithelium devoid of papillae. There are deep branching epithelial ridges described as test-tube like and the subepithelial foci of lymphocytes are the most striking feature. There is no atypia of the epithelium and the supramuscular sclerosis is probably normal to the median raphe of the tongue. I think that the first reference
Median rhomboid glossitis
401
FIGURE 2. Female aged 35 years with acute pseudo-membranous candidiasis superimposed on chronic candidiasis of midline of dorsum of tongue.
to the possibility that this is a candidal lesion is made by J.J.Pindborg in the Textbook of Dermatology (2nd edn) edited by Rook et al. (1972). Patients Ten patients presented with chronic median glossitis. Six were female, four male, and their ages ranged from 30 to 63 years, with four in the fourth decade, three in the fifth, and three in the seventh. All the patients complained of discomfort, mainly a burning sensation and these symptoms were present for up to a year before examination. The lesions ranged from a position just anterior to the circumvallate papillae to the junction of the anterior and the middle third of the tongue, but all
B.E.D.Cooke
402
were in the midline. They varied from being smooth to grossly nodular and fissured. There was loss of suppleness within them all and with a degree of induration in some. There were varying degrees of a superimposed acute candidiasis so that the surface would sometimes appear thrushlike in part or in whole. One of the patients gave a history of having had warts removed from the dorsum of the tongue 3 years previously and presented with four or five papules about 3 mm in diameter in the middle of the dorsum of the tongue. As the condition was recognized to be probably a form of chronic candidiasis, antifungal therapy was prescribed for these patients but with very limited success. Histopathology
Candidal hyphae were found within the superficial layers of the parakeratotic epithelium in all cases (Fig. 3). Where there was a superimposed acute candidiasis there was a plaque of epithelial cells and chronic infiammatory cells invaded by the Candida albicans superimposed upon the parakeratotic epithelium (Fig, 4). Test-tube like deep epithelial ridges, some of which branched, were also very striking features. Furthermore, there were foci of lymphocytes closely related to the
FIGURE 3. Candidal hyphae in parakeratotic epithelium from lesion illustrated in Fig. i (PAS x 40).
Median rhomboid glossitis
403
FIGURE 4. Candidal hyphae within the superficial plaque of epithelial cells and within the parakeratotic epithelium of lesion illustrated in Fig. 2 (PAS x 40).
epithelial ridges, particularly near the tips and there were many dilated capillaries within the corium between the epithelial ridges (Fig. 5). The degree of oedema between the superficial prickle cells varied from case to case, but in one there were micro-abscesses filled with polymorphonuclear leukocytes. The intercellular oedema and the infiltrate of lymphocytes and occasional polymorphonuclear leukocytes disturbed the arrangement of the prickle cells, but no atypia was observed. DISCUSSION
In 1966 Cawson described the clinical and histological features of chronic oral candidiasis and made three important points; first, the presence of hyphae invading the epithelium was a positive finding that cannot easily be ignored; secondly, the infection was associated with a fairly consistent set of histological changes; and thirdly, Candida was not seen to be superimposed on some other recognizable pathological process. As in his cases, so in those under discussion, the tendency of the superficial epithelial layers to separate once candidal infection was established meant that the number of hyphae observed in the PAS sections varied considerably from just a few to a heavy invasion.
404
B.E.D.Cooke
FIGURE 5. Parakeratotic and acamhocic epithelium wich 'a-sl-lube like' deep epithelial ridges and foci of lymphocytes within the corium from the lesion illustrated in Figs i and 3.
The site of election suggests that the foramen caecum and perhaps the depression of the midline raphe of the tongue favour the localization of candidal infection. From these sites it then spreads laterally and is characterized by its chronicity. There is little doubt that the loss of the papillae is permanent and that the histoiogical changes induced by the candidal hyphae are irreversible and permanent. Otherwise, how could the histological appearances be described in such a uniform manner by so many different authors over the years ? The slides illustrated in Figs 3 and 4 of the author's paper (1962) have been stained with PAS and candidal hyphae have been identified in the parakeratotic epithelium. Of the greatest relevance have been the observations made by Jones & Russell (1974) in a recent paper entitled 'The histology of chronic candidal infection of the rat's tongue'. The changes they observed in the lingual epithelium were very similar to the findings just described, with the addition that some dysplasia of the epithelium was noted, together with muscle changes as seen in the regenerative phases following damage to muscle. This, the authors presumed, might eventually lead to extensive sub-epithelial fibrosis and alter the ability of the oral mucosa to respond to any potentially harmful stimulus. No treatment is either necessary or efiective in those asymptomatic cases with a smooth, nodular or fissured surface devoid of papillae. However, in those lesions with a fissured surface associated with a persistent painful glossitis, excision is indicated. Anti-fungal therapy would appear only to correct any superimposed pseudo-membranous candidiasis, although it may well be efficacious in the treatment of the early lesions, so preventing the condition becoming permanent. In conclusion, therefore, it is submitted that the condition that has been described as median rhomboid glossitis is chronic hyperplastic candidiasis and not a developmental anomaly.
Median rhomboid glossitis
405
ACKNOWLEDGMENTS I am grateful to Mr John Wolfie and Mr Russel Hopkins for undertaking the surgical treatment of many of the patients referred to in this paper; Mr R.T.Allison for technical assistance in the preparation of the sections and photomicrographs; and to Mr B.A.Jones of the Dental Audio-visual Aids Unit for the clinical photographs.
REFERENCES BAUGHMAN, R.A. (1971) Median rhomboid glossitis: a developmental anomaly? Oral Surgery, 31, 56. BBOCQ, L . & PAUTRIER, L.M. (1914) Glossite losangique mediane de la face dorsale dc la langue. Atmali di DermaTologia e Sifilologia (Paris), 5, i. CAWSON, R.A. (1966) Chronic oral candidiasis and leucoplakia. Oral Surgery, 22, 582. CooKE, B.E.D. (1962) Median rhomboid glossitis and benign glossitis migrans (Geographical tongue). British Denial Journal, I12, 389. JONES, J.H. & RUSSELL, C. (1974) The histology of chronic candidal infection of the rat's tongue. Journal of Pathology, 113, 97. MARTIN, H.E. & HOWE, M . E . (193S) Glossitis rhombica mediana. Annals of Surgery, 107, 39. PiNDBORG, J.J. (1972) Disorders of the oral cavity and lips. In: Textbook of Dermatology (Ed. by A.J.Rook, D.S.Wilkinson and F.J.Ebling) 2nd edn. Vol. 2, p. 1712. Blackwell Scientific Publications, Oxford. SAMMETT, J.F. (1939) Median rhomboid glossitis. Radiology, 32, 215.
