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Solitary Langerhans cell histiocytosis of the hard palate: a diagnostic pitfall

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ABSTRACT

Langerhans cell histiocytosis (LCH) is a relatively rare and unique disease characterized by an abnormal proliferation of immature dendritic cells. It is predominantly seen in children with adults showing less than ten times the incidence compared to childhood. The clinical presentation and organ involvement is highly variable. Oral manifestations generally consist of mucosal ulceration associated with lesions of the underlying bone. Lesions limited to the oral mucosa are rare. We present a case of a 45-year-old male who presented with an ulcer on the hard palate showing histopathologic features of LCH. The present case is a reminder of the possibility of occurrence of this unusual entity in the oral cavity. Appropriate use of immunohistochemistry is advocated to avoid diagnostic pitfalls.

No MeSH data available.


Immunohistochemical staining showing (a) LCA reactivity, (b) S100 reactivity, (c) CD1a reactivity in the large cells. (d) IHC staining for EMA is negative in these cells.
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Figure 2: Immunohistochemical staining showing (a) LCA reactivity, (b) S100 reactivity, (c) CD1a reactivity in the large cells. (d) IHC staining for EMA is negative in these cells.

Mentions: A 45-year-old male presented with an ulcer on the hard palate. Physical examination revealed an area of ulceration measuring 3x2 cm with ill-defined margins. X-ray did not show any bony lesion. On CT scan, no obvious mass lesion was observed in palate. Trucut biopsy from the site was reported elsewhere as poorly differentiated carcinoma, which was followed by right inferior maxillectomy. Gross examination of the specimen received showed an indurated area measuring 2.5x1.2x0.8 cm on the hard palate. Extensive sampling of this area revealed an ill-defined nodular lesion beneath the epithelium composed of sheets of large, polygonal cells with moderate eosinophilic cytoplasm and reniform nuclei exhibiting grooves (Figure 1 (Fig. 1)). These were admixed with lymphoid aggregates, plasma cells, eosinophils and neutrophils. On immunohistochemistry the large cells were diffusely positive for CD1a, S100, CD68 and negative for LCA, EMA, cytokeratin (Figure 2 (Fig. 2)). Sections from the underlying bone were unremarkable. Subsequent to the diagnosis, involvement of other systems was ruled out. The final diagnosis of Langerhans cell histiocytosis of the oral mucosa was rendered. Currently the patient is disease-free after a two year follow-up.


Solitary Langerhans cell histiocytosis of the hard palate: a diagnostic pitfall
Immunohistochemical staining showing (a) LCA reactivity, (b) S100 reactivity, (c) CD1a reactivity in the large cells. (d) IHC staining for EMA is negative in these cells.
© Copyright Policy - open-access
Related In: Results  -  Collection

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getmorefigures.php?uid=PMC5036401&req=5

Figure 2: Immunohistochemical staining showing (a) LCA reactivity, (b) S100 reactivity, (c) CD1a reactivity in the large cells. (d) IHC staining for EMA is negative in these cells.
Mentions: A 45-year-old male presented with an ulcer on the hard palate. Physical examination revealed an area of ulceration measuring 3x2 cm with ill-defined margins. X-ray did not show any bony lesion. On CT scan, no obvious mass lesion was observed in palate. Trucut biopsy from the site was reported elsewhere as poorly differentiated carcinoma, which was followed by right inferior maxillectomy. Gross examination of the specimen received showed an indurated area measuring 2.5x1.2x0.8 cm on the hard palate. Extensive sampling of this area revealed an ill-defined nodular lesion beneath the epithelium composed of sheets of large, polygonal cells with moderate eosinophilic cytoplasm and reniform nuclei exhibiting grooves (Figure 1 (Fig. 1)). These were admixed with lymphoid aggregates, plasma cells, eosinophils and neutrophils. On immunohistochemistry the large cells were diffusely positive for CD1a, S100, CD68 and negative for LCA, EMA, cytokeratin (Figure 2 (Fig. 2)). Sections from the underlying bone were unremarkable. Subsequent to the diagnosis, involvement of other systems was ruled out. The final diagnosis of Langerhans cell histiocytosis of the oral mucosa was rendered. Currently the patient is disease-free after a two year follow-up.

View Article: PubMed Central - HTML - PubMed

ABSTRACT

Langerhans cell histiocytosis (LCH) is a relatively rare and unique disease characterized by an abnormal proliferation of immature dendritic cells. It is predominantly seen in children with adults showing less than ten times the incidence compared to childhood. The clinical presentation and organ involvement is highly variable. Oral manifestations generally consist of mucosal ulceration associated with lesions of the underlying bone. Lesions limited to the oral mucosa are rare. We present a case of a 45-year-old male who presented with an ulcer on the hard palate showing histopathologic features of LCH. The present case is a reminder of the possibility of occurrence of this unusual entity in the oral cavity. Appropriate use of immunohistochemistry is advocated to avoid diagnostic pitfalls.

No MeSH data available.