Lymphocytic panhypophysitis and anti-rabphilin-3A antibody with pulmonary sarcoidosis
Lymphocytic panhypophysitis and anti-rabphilin-3A antibody with pulmonary sarcoidosis
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淋巴细胞性全垂体炎和抗 rabphilin-3A 抗体伴肺结节病
DOI：
10.1007/s11102-021-01200-0
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发表时间：
2022
期刊：
影响因子：
3.8
通讯作者：
Sugim
中科院分区：
文献类型：
--
作者：
Takahashi Yuka;Kameda Hiraku;Miya Aika;Nomoto Hiroshi;Cho Kyu Yong;Nakamura Akinobu;Nishimura Hiroki;Kimura Hirokazu;Suzuki Masaru;Konno Satoshi;Shimizu Ai;Matsuno Yoshihiro;Okamoto Michinari;Motegi Hiroaki;Iwata Naoko;Fujisawa Haruki;Suzuki Atsushi;Sugim
目的探讨抗拉苯西林-3A抗体在淋巴细胞性全垂体炎鉴别诊断中的临床意义。方法与结果1例58岁日本男性患者于2017年发生不明原因葡萄膜炎。2019年，他意识到了多尿症。2020年8月，他注意到一过性复视，并被诊断为右侧外展神经麻痹。与此同时，他抱怨疲劳和食欲不振。头部磁共振成像显示垂体柄和垂体腺增大，对应于垂体炎。激素刺激试验显示对所有脑下垂体前叶激素反应迟钝。中枢性尿崩症的诊断依据是高渗盐水负荷试验。综合这些发现，诊断为全垂体功能减退症。CT显示肺门淋巴结肿大。肺门淋巴结活检发现非干酪性上皮样细胞肉芽肿，符合结节病。垂体前叶活检发现轻度淋巴细胞浸润，无IgG4阳性细胞、非干酪性肉芽肿或肿瘤。Western blotting显示抗兔血球蛋白-3A抗体的存在，支持淋巴细胞性全垂体炎的诊断。由于患者无视力损害或严重葡萄膜炎，我们继续对葡萄膜炎进行生理激素替代治疗和局部类固醇治疗。结论据我们所知，这是首例抗兔血球蛋白3A抗体阳性的淋巴细胞性全垂体炎合并结节病，经垂体腺和肺门淋巴结活检确诊。抗RabPhilin-3A抗体在此例垂体炎的鉴别诊断中的作用应通过进一步的病例研究来阐明。
PurposeTo explore the clinical significance of anti-rabphillin-3A antibody for the differential diagnosis of lymphocytic panhypophysitis.Methods and resultsA 58-year-old Japanese man developed uveitis of unknown cause in 2017. In 2019, he became aware of polyuria. In August 2020, he noticed transient diplopia and was diagnosed with right abducens nerve palsy. At the same time, he complained of fatigue and loss of appetite. Head magnetic resonance imaging demonstrated enlargement of the pituitary stalk and pituitary gland, corresponding to hypophysitis. Hormone stimulation tests showed blunted responses with respect to all anterior pituitary hormones. Central diabetes insipidus was diagnosed on the basis of a hypertonic saline loading test. Taking these findings together, a diagnosis of panhypopituitarism was made. Computed tomography showed enlargement of hilar lymph nodes. Biopsies of the hilar lymph nodes revealed non-caseating epithelioid cell granulomas that were consistent with sarcoidosis. Biopsy of the anterior pituitary revealed mild lymphocyte infiltration in the absence of IgG4-positive cells, non-caseating granulomas, or neoplasia. Western blotting revealed the presence of anti-rabphilin-3A antibody, supporting a diagnosis of lymphocytic panhypophysitis. Because the patient had no visual impairment or severe uveitis, we continued physiological hormone replacement therapy and topical steroid therapy for the uveitis.ConclusionTo the best of our knowledge, this is the first case of anti-rabphilin 3A antibody positive lymphocytic panhypophysitis comorbid with sarcoidosis, diagnosed by both pituitary and hilar lymph node biopsy. The utility of anti-rabphilin-3A antibody for the differential diagnosis of hypophysitis like this case should be clarified with further case studies.
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