Limits...
A case of mantle cell lymphoma presenting as IgG4-related dacryoadenitis and sialoadenitis, so-called Mikulicz's disease.

Hayashi Y, Moriyama M, Maehara T, Goto Y, Kawano S, Ohta M, Tanaka A, Furukawa S, Hayashida JN, Kiyoshima T, Shimizu M, Chikui T, Nakamura S - World J Surg Oncol (2015)

Bottom Line: Lip biopsy and fine needle biopsy of submandibular gland were performed, and subsequently, MCL was diagnosed through the histopathological findings.MCL most commonly occurs in the Waldeyer ring, but rarely in the stomach, spleen, skin, LG, and SG.We report an unusual case of MCL involving LGs and SGs mimicking IgG4-DS, which suggests that IgG4 testing may be useful in the differentiation of IgG4-DS in the presence of bilateral swelling of LGs or SGs.

View Article: PubMed Central - PubMed

Affiliation: Section of Oral and Maxillofacial Oncology, Division of Maxillofacial Diagnostic and Surgical Sciences, Faculty of Dental Science, Kyushu University, 3-1-1 Maidashi, Higashi-ku, Fukuoka, 812-8582, Japan. hayashi@dent.kyushu-u.ac.jp.

ABSTRACT

Background: Mantle cell lymphoma (MCL) is a relatively uncommon type of non-Hodgkin lymphoma. It develops in the outer edge of a lymph node called the mantle zone. In contrast, IgG4-related dacryoadenitis and sialoadenitis (IgG4-DS) is characterized by elevated serum IgG4 and persistent bilateral enlargement of lacrimal glands (LGs) and salivary glands (SGs), with infiltration of IgG4-positive plasma cells. Recent studies indicated the importance of differentiation between IgG4-DS and malignant lymphoma.

Case presentation: An 82-year-old man was suspected of IgG4-DS because of a high serum IgG level (2174 mg/dL) and bilateral swelling of LGs and SGs. Lip biopsy and fine needle biopsy of submandibular gland were performed, and subsequently, MCL was diagnosed through the histopathological findings.

Conclusions: MCL most commonly occurs in the Waldeyer ring, but rarely in the stomach, spleen, skin, LG, and SG. We report an unusual case of MCL involving LGs and SGs mimicking IgG4-DS, which suggests that IgG4 testing may be useful in the differentiation of IgG4-DS in the presence of bilateral swelling of LGs or SGs.

No MeSH data available.


Related in: MedlinePlus

Histological findings in LSG. Marked lymphoplasmacytic infiltration with hyperplastic lymphoid follicles. The infiltrating lymphocytes were stained with hematoxylin and eosin (HE) (a, b), anti-CD3 (c), anti-CD5 (d), anti-CD10 (e), anti-CD79a (f), anti-bcl-2 (g), anti-cyclin D1 (h), IgG (i), and IgG4 (j) monoclonal antibodies
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
getmorefigures.php?uid=PMC4513633&req=5

Fig3: Histological findings in LSG. Marked lymphoplasmacytic infiltration with hyperplastic lymphoid follicles. The infiltrating lymphocytes were stained with hematoxylin and eosin (HE) (a, b), anti-CD3 (c), anti-CD5 (d), anti-CD10 (e), anti-CD79a (f), anti-bcl-2 (g), anti-cyclin D1 (h), IgG (i), and IgG4 (j) monoclonal antibodies

Mentions: IgG4-DS was suspected because of persistent symmetrical swelling of at least two pairs of LGs and major SGs for at least 3 months. We thus additionally examined serum IgG4 and it was in the normal range (43.7 mg/dL). We performed LSG biopsy and fine needle biopsy (FNB) of the left SMG in addition to LG biospy to check if LSG and SMG were the same disease. Histologically, all sections showed severe uniform infiltration of lymphoplasmacytes, without lymphoid follicular formation. The plasmacytoid cells showed nuclear pleomorphism. Immunohistochemical staining showed monotypic predominance of kappa-light chain and no infiltration of IgG4-positive plasma cells. The infiltrating lymphocytes were positive for B-cell markers (CD20 and CD79a), CD5, bcl-2, and cyclin D1, but negative for T-cell markers (CD3) and CD10 (Fig. 3).Fig. 3


A case of mantle cell lymphoma presenting as IgG4-related dacryoadenitis and sialoadenitis, so-called Mikulicz's disease.

Hayashi Y, Moriyama M, Maehara T, Goto Y, Kawano S, Ohta M, Tanaka A, Furukawa S, Hayashida JN, Kiyoshima T, Shimizu M, Chikui T, Nakamura S - World J Surg Oncol (2015)

Histological findings in LSG. Marked lymphoplasmacytic infiltration with hyperplastic lymphoid follicles. The infiltrating lymphocytes were stained with hematoxylin and eosin (HE) (a, b), anti-CD3 (c), anti-CD5 (d), anti-CD10 (e), anti-CD79a (f), anti-bcl-2 (g), anti-cyclin D1 (h), IgG (i), and IgG4 (j) monoclonal antibodies
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4513633&req=5

Fig3: Histological findings in LSG. Marked lymphoplasmacytic infiltration with hyperplastic lymphoid follicles. The infiltrating lymphocytes were stained with hematoxylin and eosin (HE) (a, b), anti-CD3 (c), anti-CD5 (d), anti-CD10 (e), anti-CD79a (f), anti-bcl-2 (g), anti-cyclin D1 (h), IgG (i), and IgG4 (j) monoclonal antibodies
Mentions: IgG4-DS was suspected because of persistent symmetrical swelling of at least two pairs of LGs and major SGs for at least 3 months. We thus additionally examined serum IgG4 and it was in the normal range (43.7 mg/dL). We performed LSG biopsy and fine needle biopsy (FNB) of the left SMG in addition to LG biospy to check if LSG and SMG were the same disease. Histologically, all sections showed severe uniform infiltration of lymphoplasmacytes, without lymphoid follicular formation. The plasmacytoid cells showed nuclear pleomorphism. Immunohistochemical staining showed monotypic predominance of kappa-light chain and no infiltration of IgG4-positive plasma cells. The infiltrating lymphocytes were positive for B-cell markers (CD20 and CD79a), CD5, bcl-2, and cyclin D1, but negative for T-cell markers (CD3) and CD10 (Fig. 3).Fig. 3

Bottom Line: Lip biopsy and fine needle biopsy of submandibular gland were performed, and subsequently, MCL was diagnosed through the histopathological findings.MCL most commonly occurs in the Waldeyer ring, but rarely in the stomach, spleen, skin, LG, and SG.We report an unusual case of MCL involving LGs and SGs mimicking IgG4-DS, which suggests that IgG4 testing may be useful in the differentiation of IgG4-DS in the presence of bilateral swelling of LGs or SGs.

View Article: PubMed Central - PubMed

Affiliation: Section of Oral and Maxillofacial Oncology, Division of Maxillofacial Diagnostic and Surgical Sciences, Faculty of Dental Science, Kyushu University, 3-1-1 Maidashi, Higashi-ku, Fukuoka, 812-8582, Japan. hayashi@dent.kyushu-u.ac.jp.

ABSTRACT

Background: Mantle cell lymphoma (MCL) is a relatively uncommon type of non-Hodgkin lymphoma. It develops in the outer edge of a lymph node called the mantle zone. In contrast, IgG4-related dacryoadenitis and sialoadenitis (IgG4-DS) is characterized by elevated serum IgG4 and persistent bilateral enlargement of lacrimal glands (LGs) and salivary glands (SGs), with infiltration of IgG4-positive plasma cells. Recent studies indicated the importance of differentiation between IgG4-DS and malignant lymphoma.

Case presentation: An 82-year-old man was suspected of IgG4-DS because of a high serum IgG level (2174 mg/dL) and bilateral swelling of LGs and SGs. Lip biopsy and fine needle biopsy of submandibular gland were performed, and subsequently, MCL was diagnosed through the histopathological findings.

Conclusions: MCL most commonly occurs in the Waldeyer ring, but rarely in the stomach, spleen, skin, LG, and SG. We report an unusual case of MCL involving LGs and SGs mimicking IgG4-DS, which suggests that IgG4 testing may be useful in the differentiation of IgG4-DS in the presence of bilateral swelling of LGs or SGs.

No MeSH data available.


Related in: MedlinePlus