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Table of Contents   
CASE REPORT  
Year : 2014  |  Volume : 25  |  Issue : 3  |  Page : 406-409
Atypical histiocytic granuloma: A differential to nonhealing ulcers of the oral cavity


1 Department of Oral and Maxillofacial Pathology, Buddha Institute of Dental Sciences and Hospital, Patna, Bihar, India
2 Department of Conservative and Endodontics, Buddha Institute of Dental Sciences and Hospital, Patna, Bihar, India
3 Department of Dentistry, All India Institue of Medical Sciences, Patna, Bihar, India

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Date of Submission19-Apr-2013
Date of Decision27-Sep-2013
Date of Acceptance21-May-2014
Date of Web Publication7-Aug-2014
 

   Abstract 

This is a case report of a rare clinical entity known as atypical histiocytic granuloma (AHG), which was previously grouped under a broad category known as pseudolymphoma or traumatic eosinophilic granuloma. Less than 15 cases of AHG have been reported until date. AHG poses diagnostic dilemma due to its clinical as well as histopathological appearance where it stimulates malignancy. A proper clinicopathological evaluation is necessary to establish the diagnosis and to avoid overtreatment. In this report, we review previously reported cases in literature and try to establish proper clinicopathological correlation, differential diagnosis and management. These will familiarize clinicians to include AHG in their differential diagnosis as well as for the pathologist to segregate pseudolymphomatous lesion in their proper categories. The role of immunohistochemistry (IHC) has been given prime importance to establish the exact diagnosis. Further in this report, we review different status on lymphoproliferative disorders and advocate the use of IHC in categorizing these lesions upon cell lineage and to establish proper nomenclature for these lesions.

Keywords: Atypical histiocytic granuloma, lymphoproliferative disorders, pseudolymphoma, traumatic eosinophilic granuloma, traumatic granuloma, traumatic ulcerative granuloma with stromal eosinophilia

How to cite this article:
Ranjan S, Mukul SK, Issar R. Atypical histiocytic granuloma: A differential to nonhealing ulcers of the oral cavity. Indian J Dent Res 2014;25:406-9

How to cite this URL:
Ranjan S, Mukul SK, Issar R. Atypical histiocytic granuloma: A differential to nonhealing ulcers of the oral cavity. Indian J Dent Res [serial online] 2014 [cited 2021 Oct 17];25:406-9. Available from: https://www.ijdr.in/text.asp?2014/25/3/406/138359
Oral cavity is frequently subjected to frictional and traumatic events. These events either result from mastication, any oral habits or even physical trauma from other sources in the form of hard food particles, tobacco chewing or ill-fitting dentures, etc. The eventual outcome is a clinical presentation of the traumatic site in the form of hyperkeratosis, growth or even ulcerations. Such lesions are categorized as reactive processes, which spontaneously heal after removal of stimuli. Hence, most of these lesions are self-limiting requiring no additional treatment. However, if stimuli persist then many such lesions are prone to malignant transformation. Hence, clinical evaluation of such lesions are necessary and if any doubt a biopsy should be done to establish an exact diagnosis. From the point of view of pathologist, who comes across many such lesions which mimic malignancy, the differential diagnosis becomes imperative, a simple example may be pseudoepithelomatous hyperplasia or necrotizing sialometaplasia.

Oral cavity constitutes of diverse types of tissues so different cell types can be seen in different types of lesion. Among these tissues reticuloendothelial cell lesions poses maximum diversity in its cellular presentation. Many of such lesions mimic lymphoma and are previously categorized under a roof known as pseudolymphomas, which includes lymphoid hyperplasia and a broad category known as traumatic eosinophilic granuloma (TEG). The later includes eosinophil rich CD30+ LPDs, atypical histiocytic granuloma (AHG), angiolymphoid hyperplasia with eosinophilia (ALHE), etc. [1]

Atypical histiocytic granuloma despite having a benign clinical course shows cytological atypia simulating malignancy posing a diagnostic dilemma for the pathologist. A clinicopathological distinction is a must to such pseudotumors not only for establishing diagnosis but also preventing unnecessary treatment and expenses. Role of immunohistochemistry (IHC) has shown tremendous promise in establishing exact cell lineage in these pseudolymphomatous lesions and to categorize these lesions according to a proper nomenclature.

The following is a case of AHG of buccal mucosa clinically suspected for oral squamous cell carcinoma. In this case report, we also try to place AHG as definite entity in the differential diagnosis of nonhealing ulcers to prevent a radical treatment.


   Case report Top


A 62-year-old male patient reported to an oral and maxillofacial surgeon with pain in left back region of cheek since 3 weeks. On clinical examination, a large ulcerative lesion of approximately 1.5 cm diameter was noted on posterior region of left buccal mucosa with rolled out borders [Figure 1]. It was erythematous in the center and whitish in the peripheral area merging with normal mucosa. No bleeding was observed, but was slight tender on palpation. No other mucosal lesion was noted. Adjacent second maxillary molar was extracted about 6 months back due to carious involvement of teeth by his local dentist. Existing teeth were evaluated for any sharp edges. General physical examination revealed nothing relevant. Cervical lymph nodes were nonpalpable.

A detailed history revealed trauma to the site 1½ years back due to sugarcane chewing leading to ulceration. This ulceration recurred 2-3 times with pain in the same area.
Figure 1: Preoperative picture showing ulcerative lesion with thick peripheral margins

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Due to the persistent nonhealing ulcer with a history of previous recurrence an incisional biopsy was taken to rule out malignancy and sent for histopathological analysis. The entire tissue was processed and haemotoxylin and eosin stained section showed ulcerated epithelium replaced by fibrinopruluent membrane covering underlying stroma showing dense infiltrate of mononuclear cell subepithelially as well as in submucosa almost infiltrating the muscle tissue. Mononuclear cell showed pleomorphism ranging from ovoid to polyhedral to spindled appearance with hyperchromatic basophilic nuclei along with foamy to clear cytoplasm in many cells. Many mononuclear cells showed mitotic figures and blast appearance. Furthermore, mixed inflammatory infiltrate containing eosinophils was seen subjacent to epithelium [Figure 2] and [Figure 3].
Figure 2: Ulcerative epithelium replaced by fibrinopruluent membrane covering underlying stroma showing diffuse infiltration of inflammatory cells till deeper muscle layers (HP ×10 H and E, section)

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Figure 3: Monocytoid appearing atypical, hyperchromatic cells with clear cytoplasm (HP ×40 H and E, section)

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To rule out lymphoma and establish histiocytic lineage IHC staining for CD68 was done. It showed diffuse intense staining of infiltrates confirming histiocytic origin [Figure 4] and [Figure 5].
Figure 4: Diffuse cytoplasmic positivity for CD68 immunohistochemistry marker (×10)

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Figure 5: Diffuse cytoplasmic positivity for CD68 immunohistochemistry marker (×40)

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A final diagnosis of AHG was given and was advised for no further surgical intervention and regular follow-up. After 2½ months of follow-up, the lesion totally healed [Figure 6]. The patient had no recurrence during the follow-up period of 9 months.
Figure 6: Postoperative picture after 2½ months shows uneventful healing

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   Discussion Top


Atypical histiocytic granuloma is a pseudolymphomatous lesion which clinically appears as aggressive, destructive noduloulcerative lesion and mimics malignancy. Biopsy is mandatory for such recurrent nonhealing ulcers to know the exact pathology. Interestingly again in histopathology it mimics lymphoma by showing cytological atypia, mitotic figures and pleomorphic lymphoid lineage cells. Hence, AHG mimics malignancy both clinically as well as histopathologically. In order to establish an exact diagnosis clinical and histopathological evaluation along with IHC is mandatory.

Very few cases of AHG have been reported. The first case been reported by Ruckley et al. in 1984 on the dorsum of the tongue. [2] Eversole et al. in 1984 presented additional 4 cases of AHG and he only suggested a histiocytic origin. [3]

No known etiology has been established although in one case tobacco placement on the lower labial vestibule was reported by Throndson et al. as probable etiology. Viral and candidal pathogenesis was suggested in past, but not established. Low grade trauma as etiology has also been reported in an isolated case. [4]

Atypical histiocytic granuloma has been reported mostly over tongue and lip mucosa, gingival and buccal mucosa, lower and upper lip, vestibular mucosa, and palatal mucosa. [4] It has been shown to affect both the younger and the older age groups with almost equal gender predilection [4] [Table 1]. Clinically, the lesion mostly appears to be noduloulcerative of variable durations. Kabani et al. presented a case of AHG as submucosal mass in maxillary mucobuccal fold. [5]
Table 1: Summary of cases of atypical histiocytic granuloma in literature[4]


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The treatment protocol followed in the past was no further intervention after incisional biopsy to complete excision of the lesion. Throndson et al. discussed the fact that a mere incisional biopsy stimulates local repair and release of growth factors, which favors healing. [6] Although a few cases have recurred within 5-16 weeks of follow-up and in such circumstances complete excision should be done.

Histopathologically, the lesion shows ulcerated epithelium and stroma showing mixed inflammatory infiltrate in lamina propria and submucosal infiltration by polymorphous population of cells having variable shape and size with nuclear pleomorphism, hyperchromatism, few mitosis and atypical cells. AHG is confirmed by IHC by showing lesional cells to be positive for CD68. As histiocytic lymphomas are exceedingly rare so a distinction is must to avoid a misdiagnosis. [6]

Histopathologically, it should be differentiated with eosinophilic granuloma, TEG, wegener's granulomatosis, ALHE, benign follicular lymphoid hyperplasia. A detailed clinicopathological study and absence of atypical cells is helpful in differentiating from these lesions.

Eversole et al. established a histiocytic origin of AHG by using ultrastructural studies, esterase reactivity, peanut lectin binding, and lack of immunoglobulin reactivity. [3] Later other authors confirmed histiocytic origin by using IHC. We also confirmed our case by showing lesional cells to be positive for CD68 immunohistochemical marker.

Traumatic eosinophilic granuloma can be considered as an umbrella term covering the spectrum of lesions with diverse cells of origin. Alobeid et al. has differentiated these subset of lesions according to their cell of origin. [1] He concluded that TEG with or without eosinophilia, which has shown to express CD30 positivity represents traumatic ulcerative granuloma with stromal eosinophilia, the oral counterpart of primary cutaneous CD30+ lymphoproliferative disorder. el-Mofty et al. have shown a second subset of lesions to be of myofibroblastic origin showing to be positive for only vimentin and absence of expression of all lymphoid and histiocytic markers. [7] Third set of lesions shown to be positive for histiocytic marker CD68 are grouped under term AHG. Other lesions ALHE and Kimura disease which rarely involve oral mucosa can be differentiated on clinicopathological assessment and absence of atypical cells.


   Conclusion Top


Hence, we conclude by suggesting a reactive pathogenesis for AHG as these cases mostly occur in sites prone to trauma although mostly due to long standing cases patient gives vague history. Source of irritation should be established and must be treated. Further IHC should be performed to establish exact cell lineage and to rule out malignancy. Although most of the cases have shown healing after incisional biopsy but such cases should be followed up periodically and if recurrence occur total excision should be done. This case is reported further to establish AHG as a distinct clinicopathological entity and treatment protocol to familiarize oral surgeons and pathologist.

 
   References Top

1.Alobeid B, Pan LX, Milligan L, Budel L, Frizzera G. Eosinophil-rich CD30+lymphoproliferative disorder of the oral mucosa. A form of "traumatic eosinophilic granuloma". Am J Clin Pathol 2004;121:43-50.  Back to cited text no. 1
    
2.Ruckley RW, Gordon D, Conroy B. Pseudolymphoma of the tongue mimicking Hodgkin's disease. (A case report). J Laryngol Otol 1984;98:737-41.  Back to cited text no. 2
[PUBMED]    
3.Eversole LR, Leider AS, Jacobsen PL, Kidd PM. Atypical histiocytic granuloma. Light microscopic, ultrastructural, and histochemical findings in an unusual pseudomalignant reactive lesion of the oral cavity. Cancer 1985;55:1722-9.  Back to cited text no. 3
[PUBMED]    
4.Romero-Ruiz MM, Torres-Lagares D, Alonso-Alejandro E, Serrano-González A, Gutiérrez-Pérez JL. Palatal atypical histiocytic granuloma. Med Oral Patol Oral Cir Bucal 2009;14:e480-4.  Back to cited text no. 4
    
5.Kabani S, Cataldo E, Folkerth R, Delellis RA, Bhan I, Farren P, et al. Atypical lymphohistiocytic infiltrate (pseudolymphoma) of the oral cavity. Oral Surg Oral Med Oral Pathol 1988;66:587-92.  Back to cited text no. 5
    
6.Throndson RR, Wright JM, Watkins D. Atypical histiocytic granuloma of the oral mucosa: An unusual clinicopathologic entity simulating malignancy. J Oral Maxillofac Surg 2001;59:822-6.  Back to cited text no. 6
    
7.el-Mofty SK, Swanson PE, Wick MR, Miller AS. Eosinophilic ulcer of the oral mucosa. Report of 38 new cases with immunohistochemical observations. Oral Surg Oral Med Oral Pathol 1993;75:716-22.  Back to cited text no. 7
    

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Correspondence Address:
Shashi Ranjan
Department of Oral and Maxillofacial Pathology, Buddha Institute of Dental Sciences and Hospital, Patna, Bihar
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.138359

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