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Table of Contents
CASE REPORT
Year : 2015  |  Volume : 5  |  Issue : 2  |  Page : 83-86

Generalized gingival enlargement in non-Hodgkins lymphoma during pregnancy: A rare case report


Department of Periodontics, KLE's V. K. Institute of Dental Sciences, KLE University, Belgaum, Karnataka, India

Date of Web Publication5-Jan-2016

Correspondence Address:
Shaila Kothiwale
Department of Periodontics, KLE's V. K. Institute of Dental Sciences, KLE University, Belgaum, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2229-5194.173223

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   Abstract 

Lymphoma is the second most common neoplasm of the head and neck region. Non-Hodgkin's lymphoma (NHL) affects not only lymphoid organs but other organs and tissues that do not contain lymphoid cells. NHL cases occur extranodally and in 3% of these cases the initial presentation may be in the oral cavity. This case report presents an unusual occurrence of NHL in a young pregnant female presenting as generalized gingival enlargement which was the first and only clinical manifestation of the malignancy.
CLINICAL RELEVANCE TO INTERDISCIPLINARY DENTISTRY

  • The present case emphasizes the importance of the diagnosis of gingival enlargement, as this can be the manifestation
  • Early diagnosis may allow treatment of early stage disease, resulting in a better prognosis for the patient
  • Thus, an interdisciplinary approach among periodontist, gynecologist, pathologist, and oncologist will help in precise and timely diagnosis and treatment of such malignancies.

Keywords: Gingival enlargement, non-Hodgkins lymphoma, pregnancy


How to cite this article:
Kothiwale S, Kore S, Rathore A. Generalized gingival enlargement in non-Hodgkins lymphoma during pregnancy: A rare case report. J Interdiscip Dentistry 2015;5:83-6

How to cite this URL:
Kothiwale S, Kore S, Rathore A. Generalized gingival enlargement in non-Hodgkins lymphoma during pregnancy: A rare case report. J Interdiscip Dentistry [serial online] 2015 [cited 2019 Jun 19];5:83-6. Available from: http://www.jidonline.com/text.asp?2015/5/2/83/173223


   Introduction Top


Lymphoma is a general term for a complex group of malignancies of the lymphoreticular system. It is the second most common neoplasm of the head and neck after squamous cell carcinoma. Areas affected in the head and neck region include Waldeyer's ring, the orbit, the paranasal sinuses, the salivary glands, and the thyroid.[1] Lymphoma is categorized broadly into Hodgkin lymphoma and non-Hodgkin lymphoma (NHL). Among all the malignant lymphomas, 90% comprises of NHL and the remaining 10% of Hodgkins lymphoma.[2] These malignancies initially arise within the lymphatic tissues and may progress to an extranodular mass (NHL) or to a nontender mass or masses in a lymph node region (Hodgkin's lymphoma) that later may spread to other lymph node groups and involve the bone.[1]

NHL is a nonspecific term that includes several lymphoproliferative malignant diseases with different clinical and histological appearances. Extranodal lymphomas represent 20-30% of NHL. Oral manifestations are seen in 3-5% of cases. An oral lesion as an initial manifestation is very rare.[3] Oral lesions of NHL may develop in the soft tissues or centrally within the jaws appearing as nontender swellings commonly affecting the vestibule, gingiva, or posterior hard palate and develop slowly.

The second most common cause of mortality during the reproductive year is cancer.[4] The most common malignant tumor associated with pregnancy include cervical and breast carcinoma, malignant melanoma, ovarian cancers, and lymphoma. The occurrence of lymphoma during pregnancy is a rare condition with inherent poor prognosis.[5] Management of cancer during pregnancy is a challenge, and hence the early diagnosis of lymphoma during pregnancy becomes important. Its staging and therapeutic interventions must be performed carefully, bearing in mind the risk factors for both pregnant mother and the unborn child.[5]

The present case reports generalized gingival enlargement as the first clinical manifestation of NHL in a pregnant female.


   Case Report Top


A 24 years old patient with a history of pregnancy for 4 months reported to the Department of Periodontics, KLE's VK Institute of Dental Sciences, Belgaum with a complaint of generalized enlarged gingiva in the maxillary and mandibular jaw since 1 month. The patient was referred by a physician, Department of General Medicine, KLE's Dr. Prabhakar Kore Charitable Hospital whom she had consulted for weakness, fever, and gingival enlargement.

Medical history

The patient was in her second trimester of pregnancy (4th month). The blood investigations showed normocytic hypochromic anemia with the hemoglobin level 7.5 g/dL along with thrombocytopenia with the platelet count of 1,40,000 cells/mm 3. Patient also gave a history of intermittent fever since 15 days. Obstetric history revealed the patient had two children with no complications.

Clinical dental examination

Generalized gingival overgrowth was noted in maxillary and mandibular jaw both on the buccal and lingual/palatal surfaces. According to the patient, the growth was present since 1 month and was insidious in onset and progressed slowly. She noticed a sudden increase in the size of the growth of gingival tissue 15 days prior to the referral. Furthermore, there was a generalized pain, throbbing in nature along with spontaneous bleeding of gingiva since 10 days. She had difficulty in speech, chewing and was unable to maintain proper oral hygiene due to the interference caused by the increased gingival tissue. The overgrown gingival tissue facilitated further plaque accumulation which added an inflammatory component to the overgrowth.

On extra-oral examination, tender and enlarged submandibular lymph nodes on left and right side were noticed. Intraoral examination revealed generalized gingival overgrowth in maxillary and mandibular region on the buccal and lingual/palatal and interdental papilla areas [Figure 1]. The growth extended to two-third of the crown in the anterior region whereas it covered almost the entire crown structure in the posterior region. The gingival enlargement was related to Grade 3 according to Bokenkamp and Bonhorst. The growth was fibrotic and firm in consistency with lobulated appearance. The palatal gingival enlargement of the maxillary anterior area impeded the occlusion. Grade 1 mobility was seen in relation to maxillary central incisors, mandibular anteriors and molars. A generalized probing pocket depth of 5-6 mm was recorded. Halitosis was present.
Figure 1: Clinical intraoral pictures. (a) Right Iateral view, (b) left Iateral view, (c) Frontal view, (d) Maxillary Palatal view, (e) mandibular lingual view

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Dental radiographic examination

A panoramic radiograph was taken which revealed generalized bone loss in both maxillary and mandibular arches. The generalized horizontal bone loss was seen in the interdental areas with 70-80% of bone remaining. No other pathologic changes were observed [Figure 2].
Figure 2: Orthopantomogram of the patient

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Periodontal therapy

An informed consent was obtained from the Department of Medicine and Gynecology to perform the periodontal therapy. Oral prophylaxis was done; the patient was recalled after a week for the re-evaluation. Further gingivectomy procedure was carried out in the fourth quadrant, and a periodontal pack was placed. The follow-up was planned after a week to evaluate the healing. Postoperative instructions were given along with prescription in consultation with the gynecologist. The excised tissue was sent for histopathologic examination. After 7 days, the surgical site showed uneventful healing with bluish-red discoloration and also there was regrowth of the tissue to approximately its original size [Figure 3]. The third quadrant also showed similar healing and gingival regrowth postoperatively.
Figure 3: Clinical picture of healing of gingiva after surgery

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Histopathologic findings

The biopsy specimens obtained were sent to Department of General Pathology JN Medical College. Microscopic examination revealed oral mucosa lined by stratified squamous epithelium. The subepithelial tissue showed monotonous sheets of lymphoid cells arranged in solid sheet and lobules. The intervening delicate septa showed congested blood vessels along with mixed inflammatory infiltrate comprising of neutrophils, plasma cells, and mature lymphocytes. These findings were suggestive of NHL [Figure 4].
Figure 4: Histopathology picture of biopsy of gingival tissue (a) Low Magnification, (b) High Magnification

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   Discussion Top


NHLs comprises of a group of highly diverse malignancies, their treatment and prognosis are varied.[6] The risk factors frequently associated with NHL include primary immunodeficiency disorder, HIV infection, genetic predisposition and organ transplantation; however, the underlying factor behind most diagnosed NHL patients remain unknown.[2],[7] Most NHL arises in lymph nodes, and initial oral manifestation of extranodal involvement of NHL is very uncommon accounting for only 0.1-5% of the cases reported.[8]

Many a times, lymphoma presents in the oral cavity as the first identifiable evidence of the disease.[8] Patients often present with nonspecific clinical signs and symptoms such as local swelling, pain or discomfort, and ulcer. The clinical definition of gingival NHL varies, but it is usually described as an asymptomatic gingival enlargement or a mass of tissue that resembles a pyogenic granuloma.[9] For a lymphoma patient who presents with gingival involvement, it is important to determine the origin of lesion whether from the bone or soft tissue. Because lymphoid tissue is normally found in the bone marrow, the skeleton is thus commonly involved.[6] The gingiva is one of the rarest intraoral sites where isolated cases have been reported.[10] This is partially accounted for by the fact that lymphoid tissue is not normally found in the gingiva.[10] Even in the largest case series of intraoral lymphoma by Eisenbud and Seiabba, no single case with gingival involvement has been reported.[11] In the present case, clinical and radiographic examination, the possible differential diagnosis for this lesion were pregnancy induced gingival enlargement, squamous cell carcinoma, or any other malignancy. The histopathologic report was received after 15 days confirmed with the diagnosis of NHL.To the best of our knowledge, this is the first reported case presenting with generalized gingival enlargement due to NHL in a pregnant patient.

Once a diagnosis of lymphoma was made on an oral biopsy, several clinical considerations were addressed prior to the institution of the therapy. Hence, gingivectomy procedure was discontinued, and the patient was referred to the Department of Oncology for the further treatment. The pregnancy was terminated, and the patient was given chemotherapy. The standard modality chemotherapy included for the patient was cyclophosphamide, doxorubicin, oncovin and prednisolone therapy. However, treatment was incomplete after two cycles as the patient did not report back for next cycle. The use of chemotherapy in pregnancy complicated with NHL is a subject of great concern because of its teratogenic effect.[12]

Several theories have been suggested to explain the aggressive nature of NHL in pregnant women. These theories focus either on the hormone-dependent growth or on the immunosuppressive effect of pregnancy, which facilitate tumor dissemination.[4]

There is considerable evidence that lymphomas at specific sites show a local inflammatory process in the beginning, followed by increased rate of cell division of lymphocytes and thereby increasing the chance of a malignant clone developing.[8] Primary NHL of the gingiva can mimic other benign or reactive lesions and hence it is necessary to perform a biopsy and microscopic examination.[8]

An early biopsy would provide an appropriate diagnosis and may help in avoiding the disease progression, thus improving the prognosis.


   Conclusion Top


Although NHL during pregnancy is uncommon, it is a grave problem due to unsuitable maternal and fetal outcomes. Early diagnosis may allow treatment of early stage disease, resulting in a better prognosis for the patient. The present case emphasizes the importance of the diagnosis of gingival enlargement as this can be the only manifestation for detection of such rare extranodal presentation of malignancies. Thus, an interdisciplinary approach between a periodontist, gynecologist, pathologist and oncologist will help in precise and timely diagnosis and treatment of such malignancies.


   Acknowledgment Top


Dr. Sunita Patil, M.D., Professor, Department of General Pathology, Jawaharlal Nehru Medical College, KLE University.

 
   References Top

1.
Epstein JB, Epstein JD, Le ND, Gorsky M. Characteristics of oral and paraoral malignant lymphoma: A population-based review of 361 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;92:519-25.  Back to cited text no. 1
    
2.
Ekström-Smedby K. Epidemiology and etiology of non-Hodgkin lymphoma: A review. Acta Oncol 2006;45:258-71.  Back to cited text no. 2
    
3.
Vaswani B, Shah M, Shah PM, Parikh BJ, Anand AS, Sharma G. Non Hodgkin's lymphoma of tongue – A case report. Indian J Med Paediatr Oncol 2008;29:59.  Back to cited text no. 3
  Medknow Journal  
4.
Weisz B, Schiff E, Lishner M. Cancer in pregnancy: Maternal and fetal implications. Hum Reprod Update 2001;7:384-93.  Back to cited text no. 4
    
5.
Karimi-Zarchi M, Ezabadi MG, Hekmatimoghaddam S, Mortazavizade M, Taghipour S, Vahidfar M, et al. Cancer in pregnancy: A 10-year experience in Shahid Sadoughi Hospital, Yazd, Iran. Int J Biomed Sci 2013;9:168-73.  Back to cited text no. 5
    
6.
Wilson TG, Wright JM. Non-Hodgkin's lymphoma of the gingiva: Review of the literature. Report of a case. J Periodontol 1986;57:155-8.  Back to cited text no. 6
[PUBMED]    
7.
Nagalaxmi V, Bhavana SM, Deshpande P, Rajalakshmi C. Non Hodgkin's lymphoma – A Grostesque presentation. Pak Oral Dent J 2013;33:50-4.  Back to cited text no. 7
    
8.
Manjunatha BS, Gowramma R, Nagarajappa D, Tanveer A. Extranodal non-Hodgkin's lymphoma presenting as gingival mass. J Indian Soc Periodontol 2011;15:418-20.  Back to cited text no. 8
[PUBMED]  Medknow Journal  
9.
Kwon JH, Song JC, Lee SH, Lee SY, Yang CW, Kim YS, et al. Non-Hodgkin's lymphoma manifest as gingival hyperplasia in a renal transplant recipient. Korean J Intern Med 2005;20:330-4.  Back to cited text no. 9
    
10.
Basavaraj KF, Ramalingam K, Sarkar A, Muddaiah S. Primary non-Hodgkin's lymphoma of gingiva in a 28-year-old HIV-positive patient. J Nat Sci Biol Med 2012;3:189-91.  Back to cited text no. 10
    
11.
Eisenbud L, Seiabba J. Oral presentations in non-Hodgkin's lymphoma: Review of thirty-one cases. Part 1 data analysis. Oral Surg 1983;56:151.  Back to cited text no. 11
    
12.
Silva PT, de Almeida HM, Príncipe F, Pereira-Leite L. Non-Hodgkin lymphoma during pregnancy. Eur J Obstet Gynecol Reprod Biol 1998;77:249-51.  Back to cited text no. 12
    


    Figures

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



 

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   Introduction
   Case Report
   Discussion
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