Adolescent Sjogren's syndrome presenting as psychosis: a case series
Adolescent Sjogren's syndrome presenting as psychosis: a case series
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DOI：
10.1186/s12969-020-0412-8
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发表时间：
2020-02-11
影响因子：
2.5
通讯作者：
Radhakrishna, Suhas M.
中科院分区：
文献类型：
--
作者：
Hammett, Erin K.;Fernandez-Carbonell, Cristina;Radhakrishna, Suhas M.
背景：据报道，高达80%的原发性干燥综合征（pSS）成人患者伴有精神异常，包括焦虑、抑郁和认知功能障碍。成人患者中有因pSS引起的精神病的报道，但在青少年/儿科文献中从未有过报道。在这里，我们首次描述了四例青少年干燥综合征的精神病症状。利妥昔单抗治疗后，所有患者的精神症状均有改善。病例表现：16岁女性，无明显既往病史，因行为异常、震颤、失眠、多食、多尿、有自杀意念4天就诊于急诊科。2: 16岁女性，重度焦虑、强迫症和抽动障碍病史4年，氟西汀治疗部分有效，出现焦虑、强迫症突然严重恶化和新的幻听。19岁女性，无明显既往病史，表现为3天进行性行为改变、语无伦次、失眠、头痛和心不在焉。17岁女性，无明显既往病史，表现为新发自杀意念，偏执，精神错乱，情绪不稳定。结论精神病在自身免疫性疾病中比以往认识的更为常见。据我们所知，上述四名青少年女性是首次报道的青少年pSS表现为精神病的患者。在新发精神疾病的年轻患者的鉴别诊断中，即使没有sicca症状，也应考虑pSS。我们所有4例患者的精神症状均在输注利妥昔单抗后得到改善，这表明利妥昔单抗可能是一种有效的治疗选择，应在诊断pss相关精神障碍后尽早考虑。
Background Neurological involvement has been reported in up to 80% of adults with Primary Sjogren's syndrome (pSS) with psychiatric abnormalities including anxiety, depression, and cognitive dysfunction being common. Psychosis due to pSS has been reported in adult patients but has never been previously reported in the adolescent/pediatric literature. Here we describe for the first time four cases of adolescent Sjogren's syndrome that presented with psychotic symptoms. Rituximab treatment was followed by improvement of psychiatric symptoms in all patients. Case presentation 1: 16 year old female without significant past medical history presented to the emergency department with 4 days of abnormal behavior, tremors, insomnia, polyphagia, polyuria, and suicidal ideation. 2: 16 year old female with a 4 year history of severe anxiety, OCD, and tic disorder treated with fluoxetine with partial benefit presented with an abrupt and severe worsening of anxiety, OCD and new auditory hallucinations. 3: 19 year old female without significant past medical history presented with a 3 day history of progressively altered behavior, incoherent speech, insomnia, headache, and tangential thoughts. 4: 17 year old female without significant past medical history presented with new onset suicidal ideation, paranoia, confusion, and emotional lability. Conclusion Psychosis is more common in autoimmune disease than previously known. To our knowledge, the four teenage women described above are the first reported patients with adolescent pSS manifesting as psychosis. pSS should be considered in the differential diagnosis of young patients with new psychiatric disorders, even in the absence of sicca symptoms. Psychiatric symptoms improved with rituximab infusions in all 4 of our patients, which suggests rituximab may be an effective treatment option that should be considered early after the diagnosis of pSS-associated psychiatric disturbance.