BackgroundOsteogenesis imperfecta (OI) is a heterogeneous group of inherited disorders that occur owing to the abnormalities in type 1 collagen, and is characterized by increased bone fragility and other extraskeletal manifestations. We report the case of a patient who was diagnosed with OI following subarachnoid hemorrhage (SAH) secondary to a ruptured saccular intracranial aneurysm (IA).Case PresentationA 37-year-old woman was referred to our hospital because of sudden headache and vomiting. She was diagnosed with SAH (World Federation of Neurosurgical Society grade 2) owing to an aneurysm of the middle cerebral artery. She then underwent surgical clipping of the aneurysm successfully. She had blue sclerae, a history of several fractures of the extremities, and a family history of bone fragility and blue sclerae in her son. According to these findings, she was diagnosed with OI type 1. We performed genetic analysis for a single nucleotide G/C polymorphism (SNP) of exon 28 of the gene encoding for alpha-2 polypeptide of collagen 1, which is a potential risk factor for IA. However, this SNP was not detected in this patient or in five normal control subjects. Other genetic analyses did not reveal any mutations of the COL1A1 or COL1A2 gene. The cerebrovascular system is less frequently involved in OI. OI is associated with increased vascular weakness owing to collagen deficiency in and around the blood vessels. SAH secondary to a ruptured IA with OI has been reported in only six cases.ConclusionThe patient followed a good clinical course after surgery. It remains controversial whether IAs are caused by OI or IAs are coincidentally complicated with OI.
Lemierre syndrome (LS) is a rare life-threatening disease that is often caused by an acute oropharyngeal infection with a secondary thrombophlebitis of the internal jugular vein. LS rarely manifests as cranial nerve palsy. To the best of our knowledge, this is the second case report of LS associated with recurrent laryngeal nerve palsy. A 66-year-old female presented to a dental clinic with gingivitis and sore throat. Due to moderate periodontitis, her left first upper molar was extracted. A few days later, she subsequently developed a coarse voice and occipital headaches, and was referred to an otolaryngologist. She was diagnosed with left recurrent laryngeal nerve palsy and subsequent left-sided otitis media, and was referred to us for persistent headaches. She intermittently presented with high-grade fever and complained of salty taste disturbance. Her head magnetic resonance imaging (MRI) revealed left mastoiditis, thrombosis in the left transverse and sigmoid sinus, and left internal jugular vein. Her laboratory tests revealed an elevated white blood cell count, levels of C-reactive protein, and D-dimer. No endogenous coagulopathy was confirmed. Although, blood and cerebrospinal fluid culture grew no microorganisms, respectively, the empirically determined antibiotic therapy was initiated. In a week, the patient defervesced and had no headaches despite persistent thrombosis. Early diagnosis and an immediate antibiotic treatment are crucial for LS patients. Anticoagulation therapy was not needed for our patient and is still controversial for LS.
Objective: Coil migration is a complication related to endovascular coil embolization. In this study, we report a patient in whom an intraoperatively migrated coil was successfully retrieved using a stent retriever.Case Presentation: A 57-year-old male. To treat an unruptured aneurysm of the right internal carotid artery, endovascular coil embolization was performed. During embolization, one end of a framing coil migrated into the parent artery, and it was retrieved using a microsnare. However, this resulted in the extra-aneurysmal deviation of a filling coil. We tried to retrieve it using a microsnare, but the formation of a coil mass made retrieval difficult. Eventually, retrieval with a stent retriever was successful. After operation, mild subarachnoid hemorrhage and slight muscle weakness of the left upper/ lower limbs were observed, but recovery was achieved during the course, and the patient was discharged. Conclusion:To our knowledge, no study has reported retrieval with a stent retriever for the management of coil migration in Japan. However, this method was considered to be effective. We report this patient, and review the literature.
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