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Anti-CD20 monoclonal antibody (rituximab) for refractory erosive stomatitis secondary to CD20(+) follicular lymphoma-associated paraneoplastic pemphigus.
| Content Provider | Semantic Scholar |
|---|---|
| Author | Borradori, Luca Lombardi, Tommaso Samson, J. Jason Girardet, Christophe Saurat, Jean Hilaire Hügli, Anne |
| Copyright Year | 2001 |
| Abstract | A 61-year-old white woman was referred in April 1999 with a 6-week history of extremely painful and severe erosive stomatitis accompanied by asthenia, difficulty in eating, and weight loss. Her medical history was unremarkable except for taking atenolol and chlorthalidone for arterial hypertension and hormone replacement therapy. Results of clinical examination revealed fibrin-covered shallow coalescing ulcers on the buccal mucosa, lower labial mucosa, the anterior region of floor of the mouth, and the dorsum and undersurface of the tongue (Figure 1). The initial lesions appeared as red macules followed by erosions. The vermilion of the lips showed extensive erosions and crusting. She also had some isolated 2to 5-mm erythematous blue maculopapular and slight hyperkeratotic lesions on the palms. With no improvement after discontinuation of the therapy with atenolol and chlorthalidone and administration of prednisone (60 mg/d for 1 week), a biopsy of a lip lesion was performed. Results of histological examination showed a large inflammatory ulceration covered by a thick layer of fibrin exudate. The underlying fibrous corium contained an inflammatory infiltrate of predominantly lymphocytes located around blood vessels without true vasculitis. The adjacent epithelium was massively spongiotic and penetrated by numerous lymphocytes. Necrosis of individual keratinocytes also occurred (Figure 2). The rest of the findings from physical examination disclosed a slight enlargement of axillary lymph nodes and the spleen. Results of light microscopy studies of a biopsy specimen obtained from the palm disclosed lichenoid interface changes with some colloid bodies and necrotic keratinocytes scattered through the epidermis. Results of direct microscopy immunofluorescence studies of perilesional skin obtained from the palm and buttock revealed faint and focal deposits of C3 in the epidermal intercellular space, while results of indirect immunofluorescence microscopy studies using normal human skin, monkey normal esophagus, and rat bladder remained normal. The patient's serum immunoprecipitated a 170kDa protein band from biosynthetically radiolabeled keratinocyte extracts. Findings from laboratory investigations revealed an erythrocyte sedimentation rate of 11 mm in the first hour, a hemoglobin level of 12.6 g/L, a white cell count of 8.2310/L, and a platelet count of 255310/L. Liver enzyme values, serum creatinine level, and results of urinalysis were normal. Findings from protein immunoelectrophoresis revealed a slight polyclonal hypogammaglobulinemia. A search for antinuclear, anti-DNA, and anticytoplasmic antibodies was negative as were the results of extensive serologic tests, including those for human immunodeficiency virus, human T-lymphotropic virus 1, cytomegalovirus, and herpes simplex viruses. Viral and fungal cultures remained negative for organisms, while a throat swab yielded Lancefield group A b-hemolytic streptococci. Findings from a chest radiograph disclosed a significant pleural effusion of the right hemithorax. A diagnosis of follicular nonHodgkin lymphoma grade 1 (according to Revised European-American Lymphoma classification) was established through the results of an axillary lymph node biopsy that demonstrated a nodular lymphocytic infiltration containing CD20CD79a, CD5CD23, CD43, and CD10 cells. The Bcl-2 protein was expressed (confirming the diagnosis), while findings for Bcl-1 protein were negative. Finally, the proliferation rate as tested with MiB-1 was 5% to 10%. Findings from a total-body computed tomographic scan revealed involvement of all deep lymph node areas with bulky lesions in the mediastinum and in the retroperitoneum. Left pleural effusion findings were positive for tumor cells, and bone marrow infiltration was present. The stage of the disease was IVA. The patient received 6 courses of standard CHOP (cyclophosphamide, doxorubicin, vincristine, prednisone) chemotherapy. Tumor response was evaluated after 3 cycles of CHOP with partial response noted. After 6 cycles, mediastinal lesions resolved, but pleural effusion and a residual retroperitoneal mass remained. The stomatitis showed only slight improvement. THE CUTTING EDGE |
| File Format | PDF HTM / HTML |
| PubMed reference number | 11255323 |
| Journal | Medline |
| Volume Number | 137 |
| Issue Number | 3 |
| Alternate Webpage(s) | https://archderm.jamanetwork.com/journals/DERM/articlepdf/478211/dce00005.pdf |
| Journal | Archives of dermatology |
| Language | English |
| Access Restriction | Open |
| Content Type | Text |
| Resource Type | Article |
