Synovitis

* Dutcher bodies are periodic acid-Schiff-positive intranuclear pseudoinclusions found in plasma cells in mucosa-- associated lymphoid tissue lymphoma, Waldenstrom macroglobulinemia, and myeloma. We describe the case of a 68-year-old woman with chronic synovitis containing multiple butcher bodies. The patient's workup for lymphoma was negative. Hematoxylin-eosin- and periodic acid-- Schiff-stained sections of 7 cases of marked chronic synovitis selected from the files spanning a 10-year period at Memorial Hospital of Rhode Island (Pawtucket, RI) were examined, and none showed butcher bodies. This case illustrates that butcher bodies may rarely occur in benign reactive conditions, such as chronic synovitis.

Dutcher bodies are periodic acid-Schiff (PAS)-positive eosinophilic intranuclear pseudoinclusions, a histologic feature strongly associated with low-grade malignant lymphomas, particularly lymphoplasmacytic lymphoma, mucosa-associated lymphoid tissue (MALT)-type lymphoma, or myeloma. butcher bodies are not typically associated with benign reactive conditions. We present the case of a 68-year-old woman with chronic synovitis showing multiple plasma cells containing PAS-positive butcher bodies and no evidence of a lymphoproliferative disorder.

REPORT OF A CASE

A 68-year-old woman with no significant past medical history presented with a soft tissue mass of her left foot. The mass was excised and consisted of a few irregularly shaped fragments of yellow-pink fibrofatty tissue measuring 5.5 X 4.0 x 1.0 cm. The patient's laboratory workup revealed a normal complete blood cell count.

MATERIALS AND METHODS

The entire specimen was routinely processed and stained with hematoxylin-eosin and PAS. Immunoperoxidase stains for (kappa) and (lambda) light chains were performed. Serum and urine electrophoretic studies were also performed. Patient nucleic acid was isolated from paraffin blocks and subjected to 35 cycles of polymerase chain reaction designed to detect immunoglobulin (Ig) H gene rearrangement, as previously described.1 Positive and negative controls were run in parallel to monitor for reaction quality and contamination.

Seven cases identified from a 10-year search of the files at Memorial Hospital of Rhode Island (Pawtucket, RI) and coded as marked chronic synovitis or rheumatoid synovitis were stained with PAS stain. Both hematoxylin-eosin- and PAS-stained sections were examined for butcher bodies.

RESULTS

Microscopically, the specimen showed fibrofatty tissue with chronic inflammation and focal hemosiderin deposits. Some areas were recognizable as synovium. These areas had marked chronic synovitis with sheets of plasma cells with numerous butcher bodies, that is, PAS-positive intranuclear inclusions (Figure 1). Immunoperoxidase stains for (kappa) and (lambda) light chains showed a polyclonal pattern not consistent with lymphoma (Figure 2). Both serum and urine protein electrophoretic studies were negative for a monoclonal spike, also arguing against the diagnosis of lymphoma. Polymerase chain reaction was negative for IgH gene rearrangement. The marked chronic synovitis and hemosiderin deposits were thought to be consistent with trauma. No butcher bodies were identified in the 7 file cases of marked chronic synovitis.

COMMENT

butcher bodies are PAS-positive, diastase-resistant nuclear pseudoinclusions of eosinophilic cytoplasm found in plasma cells described by butcher and Fahey2 in Waldenstrom macroglobulinemia. Ultrastructurally, the nuclear pseudoinclusions are formed by a cytoplasmic invagination into the nucleus. They are smooth, membrane-bound, and surrounded by clumped chromatin. The pseudoinclusions are thought to result from the accumulation of immunoglobulin in the perinuclear cisterna.3

Distinguishing malignant and benign B-cell proliferations in extranodal sites can be challenging because of overlapping morphologic features.4,5 Features favoring lymphoma include architectural effacement, formation of masses, infiltrative growth pattern, cellular monomorphism, and atypia, as well as the presence of proliferation centers.5 butcher bodies are a feature of clinically indolent MALT lymphomas.4,5 In a study of endoscopic biopsy specimens of lymphoid infiltrates of the stomach, butcher bodies were associated with gastric lymphomas only (3/ 25) and were not found in any cases of gastritis (0/58).4 Other features identified in the same study that were associated only with lymphoma were prominent lymphoepithelial lesions and moderate cytologic atypia.4

Medeiros and Harris6 considered butcher bodies to be presumptive evidence of malignant lymphoma, but cautioned that a strict definition of butcher bodies must be used. In most of the cases they studied, butcher bodies, when present, were not difficult to identify and were present in large numbers. However, they also found rare PAS-- negative amphophilic intranuclear structures in some of the monotypic and polytypic infiltrates. These structures appeared to be related to cell degeneration and were not interpreted as butcher bodies.6

In the present case, the intranuclear inclusions were PAS positive, and therefore they qualify as butcher bodies. This specimen contained no other morphologic features of lymphoma, additional workup for lymphoma was negative, and there was no clinical evidence of lymphoma. Thus, this is a case of benign chronic synovitis, most likely related to trauma, with an unusual finding of butcher bodies and no other features of lymphoma. Briton and coworkers7 found plasma cells with intranuclear inclusion bodies in 6 patients with multiple myeloma, in 3 patients with macroglobulinemia, and in "a single plasma cell of a patient with an apparently reactive plasmacytosis." In a major textbook of hematopathology, it is also mentioned that butcher bodies are "rarely seen in reactive proliferations" and are "only rarely, if ever, identified in lymphoid hyperplasias."8,9 Thus, this case illustrates an underappreciated observation that butcher bodies may rarely occur in a benign reactive condition, such as synovitis. While butcher bodies may be a clue to the presence of low-grade lymphoma, they are not a definitive feature, particularly in unusual contexts.

References

1. Elenitoba-johnson KSJ, Khorsand j, KingTC. Splenic marginal zone cell lymphoma associated with clonal B-cell populations showing different immunoglobulin heavy chain sequences. Mod Pathol. 1998;11:905-913.

2. butcher TF, Fahey JL. The histopathology of the macroglobulinemia of Waldenstrom. J Natl Cancer Inst. 1959;22:887-917.

3. Brunning RD, Parkin J. Intranuclear inclusions in plasma cells and lymphocytes from patients with monoclonal gammopathies. Am / Clin PathoL 1976;66: 10-21.

4. Zukerberg LR, Ferry JA, Southern IF, Harris NL. Lymphoid infiltrates of the stomach: evaluation of histologic criteria for the diagnosis of low-grade gastric lymphoma on endoscopic biopsy specimens. Am I Surg Pathol. 1990;14:10871099.

5. Kurtin PJ. How do you distinguish benign from malignant extranodal small B-cell proliferations? Am I Clin PathoL 1999;11 11(suppl 1):51 19-5126.

6. Medeiros LJ, Harris NL. Lymphoid infiltrates of the orbit and conjunctiva: a morphologic and immunophenotypic study of 99 cases. Am J Surg Pathol. 1989; 13:459-471.

7. Brittin GM, Tanaka Y, Brecher G. Intranuclear inclusions in multiple myeloma and macroglobulinemia. Blood. 1963;14:335-351.

8. Flieder DB, Yousem SA. Pulmonary lymphomas and lymphoid hyperplasias. In: Knowles DM, ed. Neoplastic Hematopathology. 2nd ed. Philadelphia, Pa: Lippincott Williams & Wilkins; 2001:1263-1301.

9. Knowles DM. Malignant lymphomas and lymphoid hyperplasias that occur in the ocular adnexa (orbit, conjunctiva, and eyelids). In: Knowles DM, ed. Neoplastic Hematopathology. 2nd ed. Philadelphia, Pa: Lippincott Williams & Wilkins; 2001:1303-1349.

Yulia Gray, MD; Stanley Schwartz, MD

Accepted for publication July 23, 2001.

From Roger Williams Medical Center, Providence, RI (Dr Gray); Memorial Hospital of Rhode Island, Pawtucket, RI (Dr Schwartz).

Reprints: Stanley Schwartz, MD, Department of Pathology, Memorial Hospital of Rhode Island, 111 Brewster St, Pawtucket, RI 02860 (e-mail: stanley_schwartz@MHRI.org).

Copyright College of American Pathologists Feb 2002
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