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CASE REPORT Table of Contents   
Year : 2009  |  Volume : 20  |  Issue : 4  |  Page : 503-505
A rare case of sarcoid-like reaction of lymph nodes associated with squamous cell carcinoma of alveolar mucosa


1 Department of Oral and Maxillofacial Pathology, Meenakshi Ammal Dental College and Hospital, Alapakkam Main Road, Maduravoyal, Chennai - 600 095, Tamil Nadu, India
2 Department of Pathology, Meenakshi Ammal Dental College and Hospital, Alapakkam Main Road, Maduravoyal, Chennai - 600 095, Tamil Nadu, India

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Date of Submission30-Apr-2009
Date of Decision16-Jul-2009
Date of Acceptance08-Sep-2009
Date of Web Publication29-Jan-2010
 

   Abstract 

Non-necrotizing granulomas are occasionally seen in patients with certain malignant disorders and are termed as "sarcoid-like reaction," which have many similarities with sarcoidosis. Sarcoidosis is a multisystem granulomatous disease of unknown etiology characterized by organ involvement and interference of organ function by granuloma or fibrosis. Sarcoidosis is occasionally found in a variety of malignant diseases with an overall incidence of 4.4% in carcinoma patients. We present here a rare case of moderately differentiated squamous cell carcinoma of alveolar mucosa with regard to mandible associated with sarcoid-like reaction of cervical lymph nodes in a female patient in the absence of clinical evidence of systemic sarcoidosis. The relevant literature including pathogenesis is also discussed.

Keywords: Sarcoidosis, sarcoid-like reaction, granuloma, squamous cell carcinoma

How to cite this article:
Nag S, Saraswathi T R, Sekhar G, Einstein A, Sivapathasundharam B. A rare case of sarcoid-like reaction of lymph nodes associated with squamous cell carcinoma of alveolar mucosa. Indian J Dent Res 2009;20:503-5

How to cite this URL:
Nag S, Saraswathi T R, Sekhar G, Einstein A, Sivapathasundharam B. A rare case of sarcoid-like reaction of lymph nodes associated with squamous cell carcinoma of alveolar mucosa. Indian J Dent Res [serial online] 2009 [cited 2014 Jul 29];20:503-5. Available from: http://www.ijdr.in/text.asp?2009/20/4/503/59446
Non-necrotizing epithelioid granulomas are occasionally found in lymph nodes draining a region containing a malignant tumor. [1],[2],[3] Although such tumor-associated histological changes have been termed as "sarcoid reaction," the term "sarcoid-like reaction" is a more accurate descriptor. These granulomas have also been reported to exist within the primary tumor [4] and in patients following radiation therapy and chemotherapy. [5]

Sarcoidosis is an immune system disorder of unknown etiology, characterized by non-necrotizing granulomas. Distinguishing sarcoid-like reaction from sarcoidosis is based on the location and distribution of granulomas as well as correlation of microscopic findings with clinical, radiologic and laboratory features. This article presents the report of a 55-year-old woman with a moderately differentiated squamous cell carcinoma of alveolar mucosa along with sarcoid-like reaction involving the level IV and V group of cervical lymph nodes.


   Case Report Top


A 55-year-old woman reported with the complaint of a painful ulcer in her right lower back tooth region from past two weeks. The patient noticed the ulcer 15 days back, which was initially of a small size and had gradually increased to present size. This was followed by pain, which was continuous in nature. The patient also gave a history of chewing tobacco three to four times in a day, for the past five years. On extraoral examination, right submandibular lymph nodes were enlarged, palpable and mobile. Intraorally, an ulceroproliferative growth was present on the lingual aspect of right alveolar mucosa of mandible, extending from molar region anteriorly to the anterior faucial pillars posteriorly. Superiorly, it involved the free gingiva and inferiorly extended upto the floor of the mouth. The lesion was erythematous, with everted and irregular margins and measured 5 × 3 cm 2 in size. Bleeding was observed during palpation of the lesion. Teeth 47 and 48 were mobile (Grade 3). The lesion was provisionally diagnosed as carcinoma of the right alveolar mucosa, on the basis of clinical picture. Papanicolau (PAP) and hematoxylin-eosin stained cytologic smears from the lesional area revealed features that were suggestive of malignancy.Preoperative incisional biopsy of the lesion was performed. Histological sections revealed stratified squamous epithelial surface, exhibiting hyperplasia and in situ changes such as cellular pleomorphism, nuclear hyperchromatism, altered nuclear cytoplasmic ratio, and abnormal mitotic figures. The underlying connective tissue showed infiltration with malignant epithelial islands, associated with a dense diffuse chronic inflammatory cell infiltrate. Keratin formation was minimal. On the basis of these histological findings, the lesion was diagnosed as moderately differentiated invasive squamous cell carcinoma [Figure 1].The lesion was excised with adequate clearance, including the lateral border of the mandible and radical neck dissection of lymph nodes from levels I to V was performed. The excised specimens were submitted for histopathological examination. The section of mandible showed margins free of tumor. Nodes LI, LII and LIII showed features of reactive hyperplasia. Nodes LIV and LV revealed the presence of non-necrotizing granulomas composed of macrophages, lymphocytes, plasma cells, numerous multinucleate giant cells and few Langhans-type giant cells [Figure 2] and [Figure 3]. Many laminated basophilic structures suggestive of Schaumann bodies [Figure 4] and few stellate-like inclusions suggestive of asteroid bodies [Figure 5] located within the multinucleate giant cells and Langhans-type giant cells were also seen. The above histological features suggested the diagnosis of sarcoid-like reaction. The chest x0 -ray was normal and no history of sarcoidosis or any other pulmonary disease was noted.


   Discussion Top


Sarcoidosis is an immune system disorder of unknown etiology characterized by non-necrotizing granulomas in the lungs or lymph nodes or virtually any organ, with resultant interference of function of the organ involved. In our case, non-necrotizing granulomas were observed in the sections from the lymph nodes. The association of oral malignancies with sarcoidosis has been reported in the literature. [6],[7] Hanibuchi et al. reported sarcoidosis accompanied with squamous cell carcinoma of mandibular gingiva. [7]

Three possibilities have been proposed to explain the association of malignancy in sarcoidosis, namely, the occurrence of these two disorders together being a coincidence; sarcoidosis initiating the carcinogenesis and/or promoting the growth of malignancy via suppression of cellular immunity; and sarcoid-like reaction occurring as a result of host immunological response against malignancy. [7] However, in our case, in the absence of systemic involvement, the diagnosis was consistent with a sarcoid-like reaction associated with a malignancy, rather than sarcoidosis.Besides sarcoid granulomas of excised lymph nodes, squamous cell carcinoma of alveolar mucosa was also observed in our case. Evidence of reactive hyperplasia in LI-LIII and formation of sarcoid granulomas in lymph nodes LIV and LV could be explained on the basis of specific cell-mediated immune response against the soluble antigenic factors derived from the tumor cells by both activated macrophages and T lymphocytes. Thus, the granulomatous reaction and release of cytokines such as TNF-á, IL-2 and IL-8 might be a marker of an immunologically mediated antitumor response. There was no evidence of metastasis in the surgically resected specimens of lymph nodes indicating the possibility of an activated host-immunological response.Asteroid bodies were observed within the Langhans giant cells in our case. Asteroid bodies, first described by Freidman in 1944 are eosinophilic inclusion bodies having radiating lines in the vacuolated area of cytoplasm of multinucleate giant cells and are common in sarcoidosis and sarcoid granulomas. They have peripheral pointed arms converging and appearing to criss-cross at the center of asteroid. The asteroid arms are made of parallel collagen fibers, which under electron microscope show a typical 640-700 A° periodicity, probably undergoing phagocytic digestion. Regarding the source of collagen in asteroid bodies, it is assumed that it is either formed in situ by the epithelioid cells that have the potential to synthesize collagen or it is trapped in between epithelioid cells during giant cell formation. This latter view suggests that collagen precursors were synthesized by distantly located mesenchymal cells trapped and polymerized within giant cells at a later period.Three possible mechanisms are offered for the mode of formation of asteroid bodies, namely, phagocytosis and partly fused cell membranes; trapping and polymerization of collagen within incompletely fused epithelioid cells; and transformation of epithelioid cells into fibroblasts during the healing of granulomas. [8] The latter possibility is favored but requires more evidence in support of fibroblastic potentiality of epithelioid cells and macrophages.Schaumann bodies are laminated basophilic calcifications present within the multinucleate giant cells. They were first reported by Schaumann in 1917 as being found in tonsils and lymph nodes. Various possibilities have been proposed for the formation of Schaumann bodies. Most of the authors who have previously examined these structures have considered them to be products of degeneration of elastic fibrils impregnated with calcium and iron salts. Schaumann advanced the opinion that tubercle bacilli can be transformed into such bodies. [9] It is also believed that these bodies may result from deposition around elastic fibrils, connective tissue fibrils, or hair resulting from disintegrated tubercle bacilli and proposed that they are products of intracellular globulin precipitate. [10]

Both asteroid bodies and Schumann bodies were seen within the Langhans giant cells in our case.Thus, coexistence of malignant tumor of head and neck region and sarcoid-like reactions should be carefully evaluated and systemic sarcoidosis should be ruled out as it has some serious complications such as pulmonary fibrosis leading to dyspnea and impairment of pulmonary function.


   Conclusion Top


We have reported a rare case of sarcoid-like reaction of the cervical lymph nodes associated with squamous cell carcinoma of the alveolar mucosa. Considering all the data reviewed, the correlation between various types of malignancies and sarcoid-like reaction as well as sarcoidosis is still controversial and requires further evidence to explain. As it is not possible to distinguish ­sarcoid- like reaction from sarcoidosis histologically, a thorough investigation of the patient is mandatory to rule out systemic sarcoidosis or other granulomatous diseases.

 
   References Top

1.Gorton G, Linell F. Malignant tumours and sarcoid reactions in regional lymph nodes. Acta radiol 1957;47:381-92.  Back to cited text no. 1  [PUBMED]  [FULLTEXT]  
2.Llombart A Jr, Escudero JM. The incidence and significance of epithelioid and sarcoid-like cellular reaction in the stromata of malignant tumours. A morphological and experimental study. Eur J Cancer 1970;6:545-51.  Back to cited text no. 2      
3.Brincker H. Sarcoid reactions in malignant tumours. Cancer Treat Rev 1986;13:147-56.  Back to cited text no. 3  [PUBMED]  [FULLTEXT]  
4.Kashiwabara K, Toyonaga M, Yamaguchi Y, Nakamura H. Sarcoid reaction in primary tumor of bronchogenic large cell carcinoma accompanied with massive necrosis. Internal Medicine 2001;40:127-30.  Back to cited text no. 4      
5.Bogaerts Y, van Der Straeten M, Tasson J, Pauwels R. Sarcoidosis or malignancy: A diagnostic dilemma. Eur J Respir Dis 1983;64:541-50.  Back to cited text no. 5  [PUBMED]  [FULLTEXT]  
6.Pollock JL, Catalano E. Metastatic ductal carcinoma of the parotid gland in a patient with sarcoidosis. Arch Dermatol 1979;115:1098-9.  Back to cited text no. 6  [PUBMED]  [FULLTEXT]  
7.Hanibuchi M, Matsumori Y, Nishioka Y, Sone S. A case of sarcoidosis accompanying squamous cell carcinoma in the mandibular gingiva. J Med Invest 2005;52:118-21.   Back to cited text no. 7  [PUBMED]  [FULLTEXT]  
8.Azar HA, Lunardelli C. Collagen nature of asteroid bodies of giant cells in sarcoidosis. Am J Pathol 1969;57:81-9.  Back to cited text no. 8  [PUBMED]  [FULLTEXT]  
9.Schaumann J. On the nature of certain peculiar corpuscles present in tissue of lymphogranulomatosis benigna. Acta med Scandinav 1941;106:239-53.  Back to cited text no. 9      
10.Teilum G. Allergic hyperglobulinosis and hyalinosis (paramyloidosis) in the reticulo-endothelial system in Boeck's sarcoid and other conditions; A morphologic immunity reaction. Am J Pathol 1948;24:389-407.  Back to cited text no. 10  [PUBMED]  [FULLTEXT]  

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Correspondence Address:
Shweta Nag
Department of Oral and Maxillofacial Pathology, Meenakshi Ammal Dental College and Hospital, Alapakkam Main Road, Maduravoyal, Chennai - 600 095, Tamil Nadu
India
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DOI: 10.4103/0970-9290.59446

PMID: 20139581

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    Figures

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

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