Kikuchi-Fujimoto disease (KFD), also known as necrotizing histiocytic lymphadenopathy, is a rare and benign lymph node disorder that mainly occurs in young women. It is clinically characterized by fever with tender and painful cervical lymphadenopathy mostly; however, all areas of lymph nodes can be involved. This disorder is often mistaken for malignant lymphoma or infection. The precise pathophysiology of KFD remains unknown, but it is theorized that it may be post-viral or associated with an autoimmune disease. The diagnosis is based on the histological analysis of the excised involved lymph node. The treatment is mainly supportive with favorable outcomes within a few weeks or months. In this case, we present a 24-year-old woman without a past medical history, who consulted for painful bilateral cervical lymphadenopathy associated with fever that has been evolving for one month following the coronavirus disease 2019 (COVID-19) vaccination. The initial diagnostic workup was performed and the diagnosis of KFD was confirmed based on the histopathological findings of the excised lymphadenopathy. Therapeutic management was based on oral corticosteroid treatment with clinical and radiological improvement after a few days without recurrence during follow-up. This article aims to report a rare case of KFD in a patient after receiving the messenger ribonucleic acid (mRNA)-based COVID-19 vaccine. Therefore, this case highlights the possible association between COVID-19 vaccination and KFD and this should be considered in the differential diagnosis.
A primary hemangiopericytoma (HP) of the bone is rare, because the vast majority of these tumors arise in soft tissue. This report presents a case of a hemangiopericytoma in the sacrum (S1-S2) with extension to the retrorectal space. Only a few cases of osseous hemangiopericytomas in the sacrum and involving the retrorectal space have so far been reported. The difficult diagnosis of HP and the surgical strategy was chosen according to the location of the lesion in the sacrum and retrorectal space. A local excision was indicated. A sacral resection should be considered for tumors below S4. This report demonstrated the safety of this strategy. Adjuvant radiotherapy is useful in HP. The value of chemotherapy is still doubtful, although patients with high-grade tumors or metastatic spread seem to gain substantial benefit. Due to the often unpredictable behavior of this neoplasm, extended follow-up is strongly recommended.
Gastrointestinal duplication is a rare congenital anomaly of the gastrointestinal tract. Gastric duplication cysts (GDCs) are uncommon in adults, and most cases are discovered incidentally. Here, we report a fortuitous discovery of a rare case of an asymptomatic noncommunicating GDC in an adult revealed after an endoscopic ultrasound-guided fine-needle aspiration of a suspected mucinous cystadenoma of the pancreas. A 34-year-old female presented with renal colic. Her abdominal examination was normal. She presented a cystic image at the left lumbar discovered fortuitously during ultrasonography. On uro-computed tomography, there was a suspicion of a pancreatic cystadenoma. Magnetic resonance imaging of the pancreas suggested a mucinous cystadenoma of the pancreatic tail. The endoscopic ultrasound showed a cystic thick-walled formation in the tail of the pancreas. After guided fine-needle aspiration, a split aspect of the gastric wall appeared evoking a GDC. The cytology showed epithelial cells without mucin. Three years later, the patient does not have any gastrointestinal symptoms. GDCs are a rare anomaly, and accurate diagnosis of these cysts is difficult. Surgical resection can offer a definitive diagnosis. The mainstay of treatment is surgery to avoid the risk of malignancy.
Primary plasma cell leukemia (PCL) is a rare and aggressive hematological malignancy exhibiting a circulating plasma cell count exceeding 20% of peripheral blood leukocytes or an absolute plasma cell count >2000/mm3. We report a case of a 37-year-old woman presented to the Department of Hematology with a two-month history of growth inside the oral cavity in the upper jaw and weakness. The physical examination revealed a voluminous mass involving the left side of the maxillary gingiva. The maxillofacial computerized tomography (CT) scan confirmed the presence of a solid tissue mass at the left upper maxilla. A biopsy sample obtained from the lesion showed a plasma cell infiltration. The laboratory findings revealed anemia, renal impairment with high levels of creatinine and calcium. Serum protein electrophoresis found a monoclonal peak at IgG lambda, a high level of lambda free light. The diagnosis was subsequently confirmed by a peripheral-blood smear revealed 25% of plasma cells and bone marrow aspiration with 50% of plasma cell infiltration. Primary plasma-cell leukemia (pPCL) was confirmed. The patient received VTD chemotherapy (bortezomib, thalidomide, and dexamethasone) followed by autologous stem cell transplant (ASCT), which resulted in complete remission. At the six-month follow-up, the patient relapsed with extramedullary multiple lesions under ineffective rescue therapy. Response to frontline treatments may be significant initially but short-lived with a dismal median overall survival below one year. This case report aims to highlight the need for awareness among clinicians of the relevance of examining other associated clinical features of pPCL, given its aggressive course and rapid progress without the therapy.
Neutrophilic dermatoses (ND) refer to a group of skin diseases characterized histologically by a cutaneous infiltrate of mature polymorphonuclear cells without an identifiable cause. Previously described as autonomous, these clinically distinct entities are included in the spectrum of neutrophilic disease due to the existence of overlapping forms, as described in our observation. Erythema elevatum diutinum (EED) is a rare dermatosis characterized by reddish-violaceous to browning papulonodular and plaques and belongs to the spectrum of cutaneous leukocytoclastic vasculitis. Chronic recurrent annular neutrophilic dermatosis (CRAND) is an exceptional neutrophilic dermatosis characterized by chronic annular lesions and the absence of generalized signs or hematological abnormalities. Histological features are similar to those seen in Sweet's syndrome.A 55-year-old woman with a history of pyoderma gangrenosum (PG) presented successively with two rare forms of ND, namely, EED and CRAND. There were no clinical or paraclinical arguments for any underlying systemic disease. Treatment with azathioprine 100 mg/day and topical steroids led to a total regression of lesions after a nine-month follow-up.Our observation is important because it reports two rare entities, CRAND and EED. Their occurrence in a single patient with a history of PG illustrates the concept of "neutrophilic disease" reported in the 1990s.
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