Large vessel vasculitis with rare presentation of acute rhabdomyolysis:A case report and review of literature

BACKGROUND Musculoskeletal involvement in primary large vessel vasculitis(LVV),including giant cell arteritis and Takayasu's arteritis(TAK),tends to be subacute.With the progression of arterial disease,patients may develop polyarthralgia and myalgias,mainly involving muscle stiffness,limb/jaw claudication,cold/swelling extremities,etc.Acute development of rhabdomyolysis in addition to aortic aneurysm is uncommon in LVV.Herein,we report a rare case of LVV with the first presentation of acute rhabdomyolysis.CASE SUMMARY A 70-year-old Asian woman suffering from long-term low back pain was hospitalized due to limb claudication,dark urine and an elevated creatine kinase(CK)level.After treatment with fluid resuscitation and antibiotics,the patient remained febrile.Her workup showed persistent elevated levels of inflammatory markers,and imaging studies revealed an aortic aneurysm.A decreasing CK was evidently combined with elevated inflammatory markers and negativity for antineutrophilic cytoplasmic antibodies.LVV was suspected and confirmed by magnetic resonance angiography and positron emission tomography with 18Ffluorodeoxyglucose/computed tomography.With a favourable response to immunosuppressive treatment,her symptoms resolved,and clinical remission was achieved one month later.However,after failing to follow the tapering schedule,the patient was readministered 25 mg/d prednisolone due to disease relapse.Follow-up examinations showed decreased inflammatory markers and substantial improvement in artery lesions after 6 mo of treatment.At the twelvemonth follow-up,she was clinically stable and maintained on corticosteroid therapy.CONCLUSION An exceptional presentation of LVV with acute rhabdomyolysis is described in this case,which exhibited a good response to immunosuppressive therapy,suggesting consideration for a differential diagnosis when evaluating febrile patients with myalgia and elevated CK.Timely use of high-dose steroids until a diagnosis is established may yield a favourable outcome.

Relationship between intralobar pulmonary sequestration and type A aortic dissection:A case report

BACKGROUND Pulmonary sequestrations often lead to serious complications such as infections,tuberculosis,fatal hemoptysis,cardiovascular problems,and even malignant degeneration,but it is rarely documented with medium and large vessel vasculitis,which is likely to result in acute aortic syndromes.CASE SUMMARY A 44-year-old man with a history of acute Stanford type A aortic dissection status post-reconstructive surgery five years ago.The contrast-enhanced computed tomography of the chest at that time had also revealed an intralobar pulmonary sequestration in the left lower lung region,and the angiography also presented perivascular changes with mild mural thickening and wall enhancement,which indicated mild vasculitis.The intralobar pulmonary sequestration in the left lower lung region was long-term unprocessed,which was probably associated with his intermittent chest tightness since no specific medical findings were detected but only positive sputum culture with mycobacterium avium-intracellular complex and Aspergillus.We performed uniportal video-assisted thoracoscopic surgery with wedge resection of the left lower lung.Hypervascularity over the parietal pleura,engorgement of the bronchus due to a moderate amount of mucus,and firm adhesion of the lesion to the thoracic aorta were histopathologically noticed.CONCLUSION We hypothesized that a long-term pulmonary sequestration-related bacterial or fungal infection can result in focal infectious aortitis gradually,which may threateningly aggravate the formation of aortic dissection.