An unusual case report showing combination of melanotic oral pigmentation and nonspecific ulcer in human immunodeficiency virus positive patient

Mayur Chaudhary, Shweta Dixit Chaudhary1, Anuraag Choudhary2 Departments of Oral Pathology and Microbiology and 1Pedodontics and Preventive Dentistry, Bharati Vidyapeeth Deemed University

CASE REPORT CASE Dental College and Hospital, Pune, 2Department of Oral Diagnosis, Medicine and Radiology, Vidyashikshan Prasarak Mandals (VSPM) Dental College and Hospital, Digdoh Hills, Nagpur, Maharashtra, India

ABSTRACT Human immunodeiciency virus (HIV) infection has caused a severe degree of morbidity and mortality amongst the individuals worldwide. Those affected usually belong to low socioeconomic status especially from developing countries like India. There are numerous diagnostic criteria and thousands of tests to detect HIV positivity. One such criterion is European Commission Clearinghouse, wherein HIV positivity may be detected by thorough clinical observation of the oral cavity. This criterion is being followed worldwide and has been proven to be effective for dental professionals and general physicians, as it is stated that these oral lesions precede the other lesions of HIV positivity. We report one such case of unusual combination of melanotic hyperpigmentation and nonspeciic ulcer in an adult patient, where HIV positivity was conirmed later by Western blot. To our knowledge, none such case has been reported in literature previously.

Key words: HIV, non-speciic ulcer, oral pigmentation

Introduction oral lesions (melanotic hyperpigmentation, nonspecific ulcers) that lead us to the suspicion of HIV positivity Human immunodeficiency virus (HIV) infection which was later confirmed by Western blot. among the individuals is of greater concern in the developing countries, especially India. The dearth Case Report of knowledge in past few decades regarding the oral manifestation of HIV infections has lead to a A 35-year-old female patient reported to a private formation of clinical diagnostic criteria [European institution with a chief complaint of ulceration on Commission Clearinghouse (ECC) criteria] for prompt lateral border of tongue since 2 months and black diagnosis of specific oral lesions that might reveal the pigmentation on the dorsum as well as lateral border underlying stage and prognosis of HIV. The occurrence of tongue and bilaterally on buccal mucosa. There was no history of any traumatic bite, stress, use of oral lesions, for example, acute pseudomembranous of chemotherapeutic drug, or any other relevant candidiasis, melanotic hyperpigmentation, nonspecific underlying systemic cause for pigmentation and ulcer. ulcers, and so on may incur one to be suspicious of On thorough clinical examination, a suspicion of HIV being HIV positive. We report here an unusual case positivity was felt. Patient was advised for routine of an adult female showing combination of two such hemogram and confirmatory HIV positivity test Western blot. Patient was found to be positive for HIV. Access this article online Patient later on revealed a history of sexual contact with Quick Response Code: Website: multiple sex partners. The patient’s family history was www.atmph.org noncontributory. A detailed oral examination revealed poor oral hygiene status with stains, calculus, carious

DOI: teeth, missing upper right first molar tooth, generalized 10.4103/1755-6783.140250 , and localized periodontitis in lower anterior region. Soft tissue examination of the oral cavity revealed a single, well-circumscribed, yellowish-white

Correspondence: Dr. Mayur Chaudhary, Department of Oral Pathology and Microbiology, Bharati Vidyapeeth Deemed University Dental College and Hospital, Pune - 411 043, Maharashtra, India. E-mail: [email protected]

664 Annals of Tropical Medicine and Public Health | Nov-Dec 2013 | Vol 6 | Issue 6 Chaudhary, et al.: Melanotic oral pigmentation and non-specific ulcer in HIV ulcer measuring 0.4-0.5 cm on lateral border of tongue, biopsy under local anesthesia, also the follow-up was melanotic hyperpigmentation bilaterally on buccal not possible because of lack of cooperation on the part mucosa, dorsum of tongue, and lateral border of tongue of the patient. To our knowledge, none of such case [Figures 1-4]. Informed consent was obtained from the report has been reported wherein a combined lesion patient for laboratory investigations and various dental of melanotic hyperpigmentation and nonspecific ulcer treatment procedures and also for publication of a case is seen that may aid in diagnosis of HIV positivity for report without revealing the identity. The laboratory general practitioners. investigations for underlying bacterial, viral, and fungal infection were performed by swabbing the ulcerated Discussion lesion and sending it to laboratory. For viral culture, swab was inserted into 4 mL of Hank’s balanced salt A set of definitions and diagnostic criteria for more solution and sent to the laboratory where it was further commonly seen oral manifestations of HIV infection cultured on primary human embryonic kidney cells was proposed initially by Greenspan and Sciubba in and primary rabbit kidney cells. All the three cultures 1992.[1] Later in 1993, a classification and diagnostic were found to be negative. All the above findings led criteria for oral lesions in HIV infection was introduced to our conclusion of a non specific ulcer observed by ECC and accepted worldwide.[2] Since then, there were on lateral border of the tongue. Patient was advised numerous studies that have used oral manifestations application of triamcenolone TESS buccal paste three as a diagnostic criterion for detection of HIV positivity times a day for a week and then recalled. The ulcer as these lesions have been considered to be useful in did not subside after application of triamcinolone epidemiologic surveys.[3-8] Oral ulcers associated with TESS buccal paste three times a day for a week. Patient HIV infection had been already reported in past.[1] They refused to undergo systemic steroid therapy and also

Figure 2: Melanotic hyperpigmentation on right buccal mucosa Figure 1: Melanotic hyperpigmentation on the dorsum of tongue

Figure 4: Melanotic hyperpigmemtation and nonspecifi c ulcer on Figure 3: Melanotic hyperpigmentation on left buccal mucosa lateral border of tongue

Annals of Tropical Medicine and Public Health | Nov-Dec 2013 | Vol 6 | Issue 6 665 Chaudhary, et al.: Melanotic oral pigmentation and non-specific ulcer in HIV have been classified as Group II lesions as per European The peculiarity of this case lies on the fact that till date, comission (EC)-Clearinghouse classification (1993).[2] there is none such case reported in literature where The numerous etiological factors for formation of oral nonspecific ulcer and melanotic hyperpigmentation ulcers include viral infections (e.g., virus, occur simultaneously when compared with the other cytomegalovirus, varicella zoster virus, human papilloma studies [Tables 1 and 2]. There are reports in literature virus), bacterial infections (e.g., acute necrotizing to compare CD4 counts with oral manifestations ulcerative , enterobacteriaceae infection), of HIV infection.[8] But since, patient desisted from fungal infections (e.g., candidiasis, cryptococcosis, any invasive procedure; the comparison could not be histoplasmosis), drug induced (foscarnet, interferon, established. clofazamine, and ketokonazole).[9] The ulcers seen in HIV infection may be single or multiple, yellowish in Conclusion color with or without erythematous halo, occurring on keratinized or nonkeratinized mucosa and ranging Oral manifestations of HIV have been considered as from 0.2 to 0.5cm and above.[1] In our current case, the the prognostic factors in monitoring the progress of ulcer was single, well-circumscribed, yellowish-white, the infection to full blown acquired immunodeficiency measuring 0.4-0.5 cm on lateral border of tongue. All syndrome. The most common and strongly associated other possible etiological factors were sorted out and being candidiasis, oral hairy , necrotizing we concluded the ulcer to be a nonspecific one. A gingivitis, and so on. When it comes to the diagnosis systematic diagnostic process and its interrelationship of HIV by clinically observing oral manifestations, two with treatment modalities for recurrent aphthous such lesions viz; melanotic hyperpigmentation and ulcers in HIV had been described earlier,[10] based upon nonspecific ulcers should not be neglected as these simple which, we advised application of triamcenolone (TESS occurring lesions may be mistaken for pigmentation and ulcers due to intake of certain drugs or nutritional buccal paste) three times a day for a week and then deficiency. The lack of knowledge about such lesions recalled. The ulcer did not subside after application of amongst dental professionals and general practitioners triamcenolone (TESS buccal paste) three times a day may lead to spread of infections among them if proper for a week. Patient refused to undergo systemic steroid infection control protocol is not followed. We report therapy and also biopsy under local anesthesia.

Melanotic hyperpigmentation is another oral lesion Table 1: Comparison of the present case with other cases that is less commonly associated with HIV. The Name Year Melanotic oral Melanotic oral causes related to this hyperpigmentation are systemic pigmentation pigmentation and nonspecifi c ulcer medication with clofazamine and ketokonazole,[11] Zhang et al.,[11] 1989 06 None use of certain antiretroviral and antifungal drugs, Ranganathan et al.,[5] 2000 68 None Addison’s disease, increased release of -melanocyte α Ranganathan et al.,[13] 2004 208 None stimulating hormone, and so on. There are reports Sharma et al.,[14] 2006 35 None that say even melanotic hyperpigmentation is also Taiwo and Hassan[15] 2010 14 None seen in HIV patients not on any medication. Due to Bodhade et al.,[16] 2011 78 None immunosuppression, the adrenocortical gland may Sontakke et al.,[8] 2011 38 None get infected by numerous parasites and results in Naidu et al.,[17] 2013 17 None melanotic hyperpigmentation. Pigmentation in HIV Present case 2013 01 01 may be seen on skin, nails, and mucous membranes. The actual cause of melanotic hyperpigmentation in Table 2: Comparison of the present case with other cases HIV is still remains undetermined.[12] In one study, melanin pigment was found in basal epithelial cells and Name Year Nonspecifi c Melanotic oral ulcer pigmentation and in subepithelial connective tissue under microscopic nonspecifi c ulcer examination. The same study reported presence of Silverman et al.,[18] 1986 30 None premature melanosomes in subepithelial keratinocytes Palmer et al.,[19] 1996 27 None [11] when the cells were observed under ultramicroscope. Ranganathan et al.,[5] 2000 01 None In our case, melanotic hyperpigmentation was seen on Ranganathan et al.,[5] 2004 22 None buccal mucosa, dorsum, and lateral border of tongue. Delgado et al.,[20] 2009 25 None After ruling out all other causes and the case history Taiwo and Hassan[15] 2010 01 None given by the patient, the cause could not be established Bodhade et al.,[16] 2011 47 None might be because patient refrained from performing Sontakke et al.,[8] 2011 04 None any invasive procedure. Present case 2013 01 01

666 Annals of Tropical Medicine and Public Health | Nov-Dec 2013 | Vol 6 | Issue 6 Chaudhary, et al.: Melanotic oral pigmentation and non-specific ulcer in HIV an unusual case of melanotic hyperpigmentation and 10. MacPhail LA, Greenspan D, Greenspan JS. Recurrent aphthous ulcers nonspecific ulcer in HIV positive patient to create in association with HIV infection. Oral Surg Oral Med Oral Pathol 1992;73:283-8. awareness among the dental professionals and general 11. Zhang X, Langford A, Gelderblom H, Reichart P. Ultrastructural practitioners for prompt diagnosis and proper infection fi ndings in oral hyperpigmentation of HIV-infected patients. J Oral control practices. The main etiology behind occurrence Pathol Med 1989;18:471-4. of such lesions is still irresolute and further research is 12. Burkit’s Oral Medicine Diagnosis and Treatment, Infectious Diseases. 10th ed. New Delhi: BC Decker Inc, Elsevier; 2003. p. 542. required pertaining to this field. 13. Ranganathan K, Umadevi M, Saraswathi TR, Kumaraswamy N, Solomon S, Johnson N. Oral lesions and conditions associated with References human immunodefi ciency virus infection in 1000 south Indian patients. Ann Acad Med Singapore 2004;33:37-42. 14. Sharma G, Pai KM, Suhas S, Ramapuram JT, Doshi D, 1. Greenspan JS, Barr CE, Sciubba JJ, Winkler JR. Oral manifestations Anup N. Oral manifestations in HIV/AIDS infected patients from of HIV infection. Defi nitions, diagnostic criteria, and principles of India. Oral Dis 2006;12:537-42. therapy. The U.S.A. Oral AIDS Collaborative Group. Oral Surg Oral 15. Taiwo OO, Hassan Z. HIV-related oral lesions as markers of Med Oral Pathol 1992;73:142-4. immunosuppression in HIV sero-positive Nigerian patients. 2. Classifi cation and diagnostic criteria for oral lesions in HIV infection. J Med Sci 2010;1:166-70. EC-Clearinghouse on Oral Problems Related to HIV infection and WHO 16. Bodhade AS, Ganvir SM, Hazarey VK. Oral manifestations of HIV Collaborating Centre on Oral Manifestations of Immunodefi ciency infection and their correlation with CD4 count. J Oral Sci 2011;53:203-11. Virus. J Oral Pathol Med 1993;22:289-91. 17. Naidu SG, Thakur R, Singh AK, Rajbhandary S, Mishra RK, Sagtani 3. Anil S, Challacombe SJ. Oral lesions of HIV and AIDS in Asia: An A. Oral lesions and immune status of HIV infected adults from eastern overview. Oral Dis 1997;3 Suppl 1:S36-40. Nepal. Exp Dent 2013;5:e1-7. 4. Arendorf TM, Bredekamp B, Cloete CA, Sauer G. Oral manifestations 18. Silverman S Jr, Migliorati CA, Lozada-Nur F, Greenspan D, Conant of HIV infection in 600 South African patients. J Oral Pathol Med MA. Oral fi ndings in people with or at high risk of AIDS: A study of 1998;27:176-9. 375 homosexual males. J Am Dent Assoc 1986;112:187-92. 5. Ranganathan K, Reddy BV, Kumarasamy N, Solomon S, Viswanathan 19. Palmer GD, Robinson PG, Challacombe SJ, Birnbaum W, Croser D, R, Johnson NW. Oral lesions and conditions associated with human Erridge PL, et al. Aetiological factors for oral manifestations of HIV. immunodefi ciency virus infection in 300 south Indian patients. Oral Dis Oral Dis 1996;2:193-7. 2000;6:152-7. 20. Delgado WA, Almeida OP, Vargas PA, Leon JE. Oral ulcers in 6. Reznik DA. Oral manifestations of HIV disease. Top HIV Med HIV-positive Peruvian patients: An immunohistochemical and in situ 2005;13:143-8. hybridization study. J Oral Pathol Med 2009;38:120-5. 7. Fabian FM, Kahabuka FK, Petersen PE, Shubi FM, Jürgensen N. Oral manifestations among people living with HIV/AIDS in Tanzania. Int Dent J 2009;59:187-91. Cite this article as: Chaudhary M, Chaudhary SD, Choudhary A. An 8. Sontakke SA, Umarji HR, Karjodkar F. Comparison of oral unusual case report showing combination of melanotic oral pigmentation manifestations with CD4 count in HIV-infected patients. Indian and nonspecifi c ulcer in human immunodefi ciency virus positive patient. J Dent Res 2011;22:732. Ann Trop Med Public Health 2013;6:664-7. 9. Gilquin J, Weiss L, Kazatchkine MD. Genital and oral erosions induced Source of Support: Nil, Confl ict of Interest: None declared. by foscarnet. Lancet 1990;335:287.

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