Case Reports in Rheumatology最新文献

Catastrophic antiphospholipid syndrome (CAPS) is a rare, life-threatening variant of antiphospholipid syndrome (APS) characterized by rapid, widespread thrombosis leading to multiorgan failure. Affecting less than 1% of APS patients, CAPS is associated with a high mortality rate of 30%-50%, necessitating prompt diagnosis and aggressive treatment. The mainstay of management includes anticoagulation, high-dose glucocorticoids, and plasma exchange or intravenous immunoglobulins, with biologic therapies such as rituximab and eculizumab reserved for refractory cases. We report a case of a 60-year-old male with a history of triple-antibody-positive APS complicated by recurrent diffuse alveolar hemorrhage (DAH), adrenal hemorrhage, chronic kidney disease, and superficial vein thrombosis. His condition progressed to CAPS approximately 5 years prior with a course complicated by heparin-induced thrombocytopenia. His condition stabilized with high-dose corticosteroids and rituximab therapy with sustained symptomatic improvement after 10 months of rituximab. This case highlights the complexity of CAPS diagnosis and management, in the context of DAH, emphasizing the importance of early recognition, multidisciplinary care, and individualized treatment strategies. Our patient's prolonged disease stabilization with rituximab underscores its potential role in long-term CAPS management. Further research is needed to refine treatment protocols and improve outcomes for this rare but life-threatening condition.

Objective: Crowned dens syndrome (CDS), caused by calcium pyrophosphate or hydroxyapatite deposition around the odontoid process, is an under-recognized cause of acute severe neck pain and headache. It can closely mimic septic arthritis, osteomyelitis, giant cell arteritis (GCA), polymyalgia rheumatica (PMR), or cervical spondylitis. We report a case of CDS in a patient with diffuse systemic sclerosis initially managed for presumed infection.

Methods: A 55-year-old man with diffuse cutaneous systemic sclerosis on mycophenolate mofetil and methotrexate presented with severe throbbing headache, facial tenderness, and diffuse pain. CT/CTA of the head and neck were negative for vascular events; MRI revealed inflammatory changes at the atlantoaxial and atlanto-occipital joints concerning for septic arthritis/osteomyelitis with abscess formation. Broad-spectrum antibiotics were initiated.

Results: The patient's symptoms persisted despite antibiotics, prompting repeat imaging. CT demonstrated calcifications surrounding the odontoid process, raising suspicion for CDS. Colchicine and low-dose prednisone were introduced while antibiotics were continued, given ongoing concern for occult infection in an immunosuppressed host. Within 1 week, the patient experienced near-complete resolution of pain. At follow-up, he remained symptom-free, allowing reintroduction of methotrexate while mycophenolate was held.

Conclusion: CDS should be considered in the differential diagnosis of severe headache and neck pain in rheumatology patients, particularly when imaging shows craniocervical inflammation and calcifications. CT of the odontoid is diagnostic, but MRI findings may mimic infection. Awareness of CDS is critical to avoid misdiagnosis, unnecessary procedures, or prolonged inappropriate therapy. Prompt recognition and anti-inflammatory treatment can be rapidly effective and permit safe reintroduction of disease-modifying therapy.

Lupus erythematosus panniculitis or lupus erythematosus profundus (LEP) is a rare manifestation of cutaneous lupus erythematosus, with an estimated prevalence of 2%-3% in those with either systemic lupus erythematosus (SLE) or discoid lupus erythematosus (DLE). LEP with involvement of the breasts is termed lupus mastitis (LM). Its presentation is heterogenous, with epidermal changes, erythema, violaceous skin changes, lipoatrophy, and ulceration with or without breast masses. LM may resemble breast malignancy; however, the clinical course, laboratory investigations, and imaging may often differentiate these. Should uncertainty still exist, LM may be confirmed on histopathology. LM is a chronic disease, and its natural course may include exacerbations and remissions. Antimalarial agents are the mainstay of treatment, whilst corticosteroids and cyclophosphamide have demonstrated utility. There is no standardized treatment protocol. We here present the case of a 50-year-old woman who was diagnosed with SLE and LM after several indeterminate breast biopsies with the intention of furthering awareness of this presentation.

Pseudogout or calcium pyrophosphate dihydrate deposition disease rarely occurs in the young. Known risk factors for pseudogout include age, previous surgery, trauma, metabolic conditions, and medications. Isotretinoin, a retinoid frequently used to control acne vulgaris, is known to cause arthralgia, arthritis, and myalgia. We describe a case of an adolescent using isotretinoin who presented with acute left upper extremity pain and weakness. Birefringent calcium pyrophosphate dihydrate crystals were seen on synovial fluid analysis. The patient's symptoms resolved after discontinuing isotretinoin. This is the first reported case of pseudogout in an adolescent on isotretinoin.

[This corrects the article DOI: 10.1155/2023/5780733.].

Giant cell arteritis is the most common primary systemic vasculitis among individuals over 50 years of age. It primarily affects large- and medium-size arteries and is not mediated by antibodies. One of the most recognizable and important symptoms of the disease is headache. The presence of headaches, along with other common cranial manifestations such as vision loss, jaw claudication, and scalp tenderness in the temporal arteries, can assist in diagnosing the condition. We present a complex case involving a 76-year-old male with prolonged headaches, a pituitary macroadenoma, and vestibular schwannoma. Initially, his headaches were attributed to his existing intracranial lesions; however, his symptoms continued to evolve. He continued to have headaches of varying intensity over 2 years, and subsequently developed diffuse scalp tenderness, visual disturbances, and tongue claudication. Input from various medical specialties expanded the differential diagnosis and raised the possibility of giant cell arteritis (GCA). Although the temporal artery biopsy did not reveal the classic giant cells typically associated with the condition, it supported the clinical diagnosis of GCA. Appropriate treatment with high-dose corticosteroids and anti-Interleukin 6 therapy resulted in the rapid resolution of his symptoms. This case emphasizes the importance of recognizing different types of headaches, maintaining a broad differential diagnosis, and thoroughly evaluating all clinical symptoms for timely diagnosis and treatment. It also highlights the significance of a multidisciplinary approach to ensure prompt diagnosis and to prevent irreversible complications, such as permanent vision loss.

Background: Nontuberculous Mycobacterium (NTM) infections affecting musculoskeletal structures are rare, particularly in patients with well-controlled rheumatoid arthritis (RA). This case is reported to highlight the potential risk of focal tenosynovitis due to Mycobacterium intracellulare following intra-articular glucocorticoid injection. Case presentation: A 79-year-old man with well-controlled RA developed tenosynovitis with bone destruction in the right index finger metacarpophalangeal joint following a single intra-articular injection of triamcinolone acetonide. Despite antibiotic treatment, the condition progressively worsened. Synovectomy revealed Mycobacterium intracellulare infection involving both flexor tendons, joint space, and bone marrow. The patient regularly engaged in gardening activities without protective gloves. Conclusion: This case highlights the importance of considering NTM infection in the differential diagnosis of persistent monoarthritis that worsens after intra-articular glucocorticoid injection, especially in patients with exposure risk factors such as gardening.

Giant cell arteritis (GCA), also known as temporal arteritis, is the most common systemic vasculitis in individuals over 50 and presents diagnostic challenges due to its nonspecific symptoms such as fever, headache, and fatigue. This case report describes the details of a male patient in his 70s who presented with recurrent intermittent fevers of unknown origin and was ultimately diagnosed with GCA after an extensive workup. His initial CT scans and lab tests were unremarkable. However, after a rheumatological workup displayed elevated erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP) levels, along with new symptoms of ataxia and headaches, a temporal artery biopsy (TAB) was performed and confirmed the patient had GCA. This case underscores the difficulty in diagnosing GCA primarily due to physician anchoring bias, particularly when typical symptoms are not present. The case also showcases the need for increased awareness and prompt evaluation of potential GCA symptoms to prevent severe complications. Public education as well as improved hospital protocols can lead to earlier detection and treatment of GCA, reducing the risk of morbidity.

[This corrects the article DOI: 10.1155/2024/3558853.].

Genital tuberculosis (GT) is a rare but significant extrapulmonary tuberculosis form, often mimicking ovarian malignancy. We report a case of a 58-year-old woman with Sjögren's syndrome and rheumatoid arthritis, previously treated with infliximab, who presented with abdominal distension, weight loss, night sweats, and intermittent abdominal pain. Initial imaging and elevated CA-125 levels suggested ovarian cancer. However, intraoperative findings revealed a frozen pelvis with granulomatous inflammation, caseating granulomas, and Langhans' giant cells. Histopathological analysis and RT-PCR confirmed GT coexisting with a serous cystadenoma. GT should be considered in the differential diagnosis of pelvic masses, especially in immunocompromised patients. This case emphasizes the importance of thorough diagnostic evaluation using molecular, serological, and imaging techniques to avoid misdiagnosis and unnecessary surgical interventions. Prompt initiation of antituberculosis treatment led to significant clinical improvement. Early and accurate diagnosis of GT is crucial to prevent morbidity associated with misdiagnosis and to provide effective treatment. This case underscores the need for heightened clinical awareness and multidisciplinary approaches in managing complex cases where GT mimics malignancy, ensuring optimal patient outcomes.