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According to the fourth revised edition of the world health organization classification of hematopoietic tumors, methotrexate (MTX)-associated lymphoproliferative disorder (MTX-LPD), an atypical pathological condition, is categorized under other iatrogenic immunodeficiency-associated lymphoproliferative disorders (OIIA-LPDs). An 80-year-old woman presented with OIIA-LPDs that simultaneously occurred in the gingiva and intestinal mucosa, causing intestinal perforation requiring emergency surgery. She was referred to our hospital because of poor wound healing after maxillary right first molar extraction. She had been prescribed MTX and prednisolone for rheumatoid arthritis and bisphosphonate for osteoporosis. We diagnosed her with medication-related osteonecrosis of the jaw, as the lesion showed bone exposure with mild redness and swelling of the surrounding gingiva and a cytological result of Class II. However, 9 months later, following the exfoliation of a partial sequestrum of the maxillary right first molar, a rapidly growing mass with gingival necrosis appeared, necessitating a biopsy. Simultaneously, abdominal pain worsened, and computed tomography revealed intestinal perforation; an emergency laparotomy was performed to remove the perforation and the surrounding ulcer-prone area. Histopathological examination of both lesions revealed diffuse large B-cell lymphomas. The final diagnosis was of MTX-LPD, as the mass, gingival necrosis, and residual small intestinal ulcer disappeared 2 months after MTX discontinuation. The exposed bone completely disappeared after 24 months, and no recurrence was noted after 60 months following MTX discontinuation. Although intestinal perforation due to MTX-LPD is extremely rare, patients with RA are immunosuppressed. Therefore, MTX-LPD must be recognized as a dangerous condition that requires careful general monitoring in such patients. [ABSTRACT FROM AUTHOR]

Other iatrogenic immunodeficiency-associated lymphoproliferative disorders (OIIA-LPDs), which are uncontrolled lymphoid proliferation or lymphoma induced by immunosuppressive medication, arise mainly in patients with rheumatoid arthritis (RA). The vast majority of OIIA-LPDs are caused by methotrexate (MTX), and the reported incidence rates in the oral and maxillofacial regions are similar to those in other organs. In addition, tofacitinib (TFC), which is a Janus kinase inhibitor, has been clinically applied for the immunosuppressive treatment of RA in recent years. Herein, we present a case of OIIA-LPD of the oral floor in a patient with RA who had been treated with TFC and MTX. Clinical and pathological findings suggested that inflammatory stomatitis and/or infective response by oral bacteria promoted lymphoid activation in the oral mucosa, which could potentially become malignant. This case report indicates that OIIA-LPD might result from the combined usage of MTX and TFC, and a synergistic influence between MTX and TFC on immunosuppression may lead to diversification of OIIA-LPDs. The findings suggest the difficulty in controlling complications of RA and the importance of understanding the mechanisms of immunosuppressive treatments. [ABSTRACT FROM AUTHOR]

Objective: The aim was to investigate the diagnostic imaging characteristics of Mikulicz disease (MD), especially sonographic ones, and to clarify the differences between them and those in Sjögren syndrome (SS), based on new criteria of MD. Study design: The sonographic and sialographic images, as well as clinical, histopathologic, and serologic findings of 9 patients satisfying the new criteria of MD were analyzed and compared with those in SS. Results: All swollen submandibular glands showed bilateral nodal hypoechoic areas with high vascularization on sonograms and a parenchymal defect on sialograms, whereas parotid glands showed normal or slight change on both images. Nodal areas in submandibular gland sonograms were unclear on computerized tomography and on magnetic resonance imaging, but showed accumulation on gallium scintigraphy. Conclusion: Mikulicz disease showed a high rate of bilateral nodal change in submandibular glands, which was completely different from SS. For detection and follow-up of these changes, sonography may be the best imaging modality. [Copyright &y& Elsevier]

Objective: To evaluate the usefulness of the vascularity in parotid glands in sonographic diagnosis for Sjögren syndrome. Study design: Sonographic images of 72 cases of previously suspected Sjögren syndrome (including 43 actual cases) were analyzed retrospectively for the abnormal vascularity in the parotid gland parenchyma. The relationships between the vascularity and the results of sialographic, serologic, and histopathologic examinations were analyzed. We also compared the diagnostic accuracy of B-mode only with that of B-mode plus Doppler-mode. Results: Sjögren-positive cases showed significantly higher vascularity. As the grade of vascularity became higher, the rate of the Sjögren-negative cases became lower. The highest mean vascular score could be observed both in the initial stage and in the cavitary-destructive stage in the sialographic grades. Sensitivity and accuracy were markedly improved with vascular information. Conclusion: By using vascular information, sonographic diagnosis for Sjögren syndrome can be improved. [Copyright &y& Elsevier]

Objective: The objective of this study is to establish readily applied sonographic diagnostic criteria for Sjögren’s syndrome. Study design: Sonographic images of 79 cases of previously suspected Sjögren’s syndrome (including 43 actual cases) were analyzed retrospectively for the following characteristic features: (1) multiple hypoechoic areas, (2) multiple hyperechoic lines or spots, (3) multiple hypoechoic areas surrounded with hyperechoic lines or spots, and (4) obscuration of the gland configuration. Logistic regression analysis was used to extract valuable sonographic findings. Sonographic images of 80 prospective patients (of whom 48 proved to have Sjögren’s syndrome) were scored prospectively using selected features to verify the usefulness of the established criteria. Results: Three sonographic findings in parotid and submandibular glands were selected by logistic regression analysis and retrospective and prospective patients compared. Experienced observers could differentiate positive cases of Sjögren’s syndrome from negative controls to a highly significant degree. Findings correlated very well with sialographic grading. Conclusion: Sonography can be substituted for sialography when applying the selected criteria in screening for Sjögren’s syndrome. [Copyright &y& Elsevier]