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 Table of Contents  
Year : 2014  |  Volume : 26  |  Issue : 3  |  Page : 347-350

Diagnosis of a case of relapse of acute lymphoblastic leukemia based on oral manifestation of leukemic gingival enlargement and acute necrotizing gingivitis: A case report

1 Department of Oral Medicine and Radiology, Darshan Dental College and Hospital, Udaipur, Rajasthan, India
2 Department of Periodontics, Pacific Dental College and Hospital, Udaipur, Rajasthan, India
3 Department of Oral Medicine and Radiology, Institute of Dental Studies and Technologies, Modinagar, Uttar Pradesh, India
4 Department of Orthodontics, College of Dental Sciences, Davangere, Karnataka, India

Date of Submission04-Jul-2014
Date of Acceptance05-Nov-2014
Date of Web Publication19-Nov-2014

Correspondence Address:
Gopikrishna Kolli
#3696, 9th Cross, 13th D main, HAL 2nd Stage, Bangalore - 560 038, Karnataka
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0972-1363.145025

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Acute leukemias are the most common malignancy in childhood. They represent approximately 30% of malignant diseases in patients under the age of 15 years. Acute lymphoblastic leukemia (ALL) is the most frequent type of leukemia in children. Despite high cure rates, approximately 20% of patients with ALL have disease relapse. Oral manifestations are common in leukemia, particularly in acute leukemias. One of the oral manifestations of leukemia is diffuse gingival enlargement thought to be, at least partly, the result of gross infiltration of the gingiva by blast cells. The occurrence of acute necrotizing gingivitis, although a rare occurrence, is seen in such immunocompromised individuals. This is a case report of a 19-year-old patient who was under remission after treatment for ALL in whom a recurrence of leukemia was detected based on the oral findings and highlights the importance of its early detection by the dentist in preventing further complications and for instituting therapy swiftly.

Keywords: Acute lymphoblastic leukemia, acute necrotizing gingivitis, leukemic enlargement, oral manifestations of leukemia

How to cite this article:
Kolli G, Chaitra N, Ranjan V, Nateshkumar DK. Diagnosis of a case of relapse of acute lymphoblastic leukemia based on oral manifestation of leukemic gingival enlargement and acute necrotizing gingivitis: A case report . J Indian Acad Oral Med Radiol 2014;26:347-50

How to cite this URL:
Kolli G, Chaitra N, Ranjan V, Nateshkumar DK. Diagnosis of a case of relapse of acute lymphoblastic leukemia based on oral manifestation of leukemic gingival enlargement and acute necrotizing gingivitis: A case report . J Indian Acad Oral Med Radiol [serial online] 2014 [cited 2022 May 22];26:347-50. Available from: https://www.jiaomr.in/text.asp?2014/26/3/347/145025

   Introduction Top

Leukemia is a malignant neoplasm characterized by a proliferation of abnormal leukocytes within the bone marrow. Leukemias are classified based on the type of white blood cells involved as lymphoid or myeloid. All types of leukemias result from the abnormal development of leukocytes in the bone marrow. Maturational arrest occurs, and a proliferative, clonal population of cells results. Acute lymphoblastic leukemia (ALL) is the most common type of cancer diagnosed in children, accounting for 75% of childhood leukemias. The 5-year survival rate in patients with ALL has increased from 60% to approximately 90% for children younger than 15 years and from 28% to approximately 70% for adolescents aged 15-19 years. [1],[2],[3] Despite the high cure rates, approximately 20% of patients with ALL have disease relapse.

Acute necrotizing gingivitis (ANG), which is an opportunistic gingival infection, is also known to occur in such immunodeficient individuals. [4] With the advent of antibiotics and with improved nutritional status, the incidence of ANG has decreased in developed countries; however, due to increasing incidence of acquired immunodeficiency syndrome (AIDS), the lesion has once again become a well-recognized clinical entity. [5]

This report describes a case of one such immunocompromised patient who was under remission after treatment for ALL, in whom a relapse of the disease was suspected, based on the oral findings, and was promptly referred to the oncologist for treatment of relapse of the ALL.

   Case Report Top

A 19-year-old female patient presented with diffuse gingival enlargement since 1 month. The patient was diagnosed as suffering from ALL at the age of 14 years. She had undergone radiotherapy and chemotherapy, as well as maintenance therapy for 5 years after which she had discontinued all medication as the bone marrow aspiration did not reveal any abnormality.

The intraoral examination revealed a generalized gingival enlargement which was dark pink in color. The normal scalloped contour was disturbed. The enlargement was diffuse, mild to firm in consistency, and the surface appeared smooth and shiny. Stippling was absent and there was bleeding on gentle probing. There was recession in the region of 41. There were generalized pseudopockets [Figure 1]a-d. There was moderate amount of stains and calculus on the teeth [Figure 2]. Due to the enlargement, the patient was unable to maintain oral hygiene and complained of food lodgement.
Figure 1: (a-d) Generalized leukemic enlargement seen when the patient reported to us for the first time

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Figure 2: Intraoral view showing moderate amount of stains and calculus on the teeth

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Routine hematological investigation revealed pancytopenia [Table 1]. A peripheral blood smear was carried out which further confirmed pancytopenia [Figure 3]. The blood picture and oral findings led us to suspect a relapse of ALL. The patient was referred back to her oncologist for further examination, who advised a bone marrow aspiration for further investigation and also confirmed the patient's fitness for undergoing oral prophylaxis.
Table 1: Hematological values observed in the patient (abnormal values are in bold)

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Figure 3: Peripheral blood smear showing pancytopenia

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Supragingival scaling was carried out taking care not to induce much bleeding. The patient was given instructions for maintenance of her oral hygiene and 0.12% chlorhexidine rinses were prescribed. She was asked to report after 7 days for follow-up examination. However, the patient reported back to us after 25 days complaining of pain in the interdental papilla of 11 and 21. She also complained of mild fever since 3 days. On examination, necrotic and punched-out ulceration of the interdental papilla in the region of 11 and 21 was observed with a pseudomembrane covering the necrotic papilla [Figure 4]. Bleeding on slight provocation was also noticed. A smear was prepared from the pseudomembrane and stained for Fontana stain. Microscopic examination revealed a large number of spirochetes [Figure 5]. Based on the symptoms, clinical signs, and histological examination, a diagnosis of ANG was made and treatment was initiated immediately.
Figure 4: Pseudomembrane covering the necrotic interdental papilla, which was seen when the patient reported back to us

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Figure 5: Photomicrograph showing the spirochetes after the pseudomembranous slough was stained by Fontana stain

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After application of topical anesthetic for 2-3 min, the pseudomembrane was removed gently with cotton swabs. Patient was instructed to rinse her mouth with 3% hydrogen peroxide in 1:1 dilution with warm water once in every 2 h, and was advised to continue 0.12% chlorhexidine mouth rinses twice daily. The patient was also put under 400 mg of metronidazole for 7 days, and a mild analgesic-antipyretic was also administered for 5 days. [6] She was recalled for the next 2 days. The pseudomembranous slough was removed, instructions on plaque control measures were given, and the patient was asked to come after 5 days. After 5 days, on intraoral examination, the necrotic tissue was eliminated and the patient's general health was found to have improved considerably [Figure 6].
Figure 6: Intraoral view 10 days after treatment was initiated

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   Discussion Top

Leukemias are the most common neoplastic diseases of the white blood cells, with an incidence of 9 per 1,00,000 of the population, while the risk of developing acute leukemia in children under the age of 10 years is estimated to be 1 in 2500. [7] They can arise either from B- or T-cell progenitors that arrest at an early stage of maturation and then proliferate.

Oral manifestations are more common in acute leukemias, but are also seen in subacute leukemias and are rarely seen in chronic leukemia. [8],[9] Lesions in the orofacial region, such as abrupt generalized or localized gingival enlargement, chloroma, alveolar bone destruction, bone invasion with pain and tooth displacement, tooth ache due to the leukemic infiltration of the pulp, cervical lymphadenopathy, and leukemia cutis, may occur due to the extramedullary neoplastic infiltration of oral mucosal and maxillofacial tissues. [10],[11],[12],[13]

Gross infiltration of the gingiva by the blast cells causes gingival enlargement with partial or total coverage of the crowns of the teeth, which is a common feature, especially in acute monocytic leukemia. The impairment of oral hygiene and accumulation of microbial plaque is increased due to the increased tendency to oral bleeding. The impaired oral hygiene and pre-existing local factors act as an inflammatory stimulus for an exaggerated response to plaque with subsequent reactive connective tissue hyperplasia of the gingiva and accelerated periodontal destruction.

Acute necrotizing gingivitis, a distinct and specific disease, is known to occur in such immunocompromised individuals. In the present case, the diagnosis of ANG was made as the patient presented with the classical triad of pain, bleeding, and ulceration. She also had secondary signs such as low-grade fever and pseudomembrane covering the punched-out papilla.

In this case, we used a specific stain (Fontana stain) to observe the spirochetes. The spirochetes are stained brownish black on a brownish yellow background when stained by the Fontana stain. Apart from spirochetes, the other organisms seen specifically correlating with ANG are Borrelia species, gram-positive cocci, beta hemolytic group B streptococci, and Candida albicans. Bacteroides and Selenomonas are also associated with ANG.

This case report highlights the role of the dentist in diagnosing systemic conditions at the earliest that would help in instituting prompt treatment and preventing complications. Also, the dental clinicians must keep in mind the clinical entity of ANG as a differential diagnosis while treating patients with any systemic or immune disorder and presenting with necrotic or ulcerative oral lesions. Instituting an early and prompt treatment can prevent the development of a more aggressive lesion (necrotizing periodontitis) and its sequelae (necrotizing stomatitis).

   Conclusion Top

Acute lymphoblastic leukemias are the most common leukemias in children. In comparison to other leukemias, ALL responds well to treatment. With appropriate therapy, the survival rate is highest of all leukemias; hence, early diagnosis and swift management are very crucial. The oral findings are the presenting signs of leukemias in many cases; hence, it is highly imperative that a dentist identifies such oral findings and refers the patient to the oncologist for further management.

   References Top

1.Smith MA, Seibel NL, Altekruse SF, Ries LA, Melbert DL, O'Leary M, et al. Outcomes for children and adolescents with cancer: Challenges for the twenty-first century. J Clin Oncol 2010;28:2625-34.  Back to cited text no. 1
2.Howlader N, Noone AM, Krapcho M, Garshell J, Neyman N, Altekruse SF, et al., editors. SEER Cancer Statistics Review, 1975-2010 [Internet]. Bethesda, MD: National Cancer Institute; 2013. Available from: http://seer.cancer.gov/archive/csr/1975_2010/#revision [Last accessed 2014 Apr 04].  Back to cited text no. 2
3.Hunger SP, Lu X, Devidas M, Camitta BM, Gaynon PS, Winick NJ, et al. Improved survival for children and adolescents with acute lymphoblastic leukemia between 1990 and 2005: A report from the children's oncology group. J Clin Oncol 2012;30:1663-9.  Back to cited text no. 3
4.Barrett AP. Leukemic cell infiltration of the gingivae. J Periodontol 1986;57:579-81.  Back to cited text no. 4
5.Bermejo-Fenoll A, Sánchez-Pérez A. Necrotising periodontal diseases. Med Oral Patol Oral Cir Bucal 2004;9(Suppl):104-19.  Back to cited text no. 5
6.Johnson BD, Engel D. Acute necrotizing ulcerative gingivitis. A review of diagnosis, etiology and treatment. J Periodontol 1986;57:141-50.  Back to cited text no. 6
7.Folayan MO. The epidemiology, etiology, and pathophysiology of acute necrotizing ulcerative gingivitis associated with malnutrition. J Contemp Dent Pract 2004;5:28-41.  Back to cited text no. 7
8.Alexandra Sklavounou-Andricopoulou, Evangelia Piperi, Spyros Paikos. Oral and maxillofacial manifestations of malignant haemopoietic and lymphoreticular disorders ñ Part IIA. Haema 2002;5:305-19.  Back to cited text no. 8
9.Anil S, Smaranayake LP, Nair RG, Beena VT. Gingival enlargement as a diagnostic indicator in leukaemia. Case report. Aust Dent J 1996;41:235-7.  Back to cited text no. 9
10.Lynch MA, Ship II. Initial oral manifestations of leukaemia. J Am Dent Assoc 1967;75:932-40.  Back to cited text no. 10
11.Stafford R, Sonis S, Lockhart P, Sonis A. Oral pathoses as diagnostic indicators in leukemia. Oral Surg Oral Med Oral Pathol 1980;50:134-9.  Back to cited text no. 11
12.Bressman E, Decter JA, Chasens AI, Sackler RS. Acute myeloblastic leukemia with oral manifestations. Report of a case. Oral Surg Oral Med Oral Pathol 1982;54:401-3.  Back to cited text no. 12
13.Takagi M, Sakota Y, Ishikawa G, Kamiyama R, Nakajima T, Nomura T. Oral manifestations of acute promyelocytic leukemia. J Oral Surg 1978;36:589-93.  Back to cited text no. 13


  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]

  [Table 1]


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