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矮小身材作为儿童单中心Castleman病的首发表现:一例长期随访病例报告及文献综述

Short stature as an initial presenting presentation of unicentric Castleman disease in a child: A case report with long-term follow-up and a literature review.

作者信息

Sonoda Kaori, Kaneko Utako, Hiura Makoto, Kinoshita Yoshiaki, Umezu Hajime, Ito Shuichi, Saitoh Akihiko, Imai Chihaya

机构信息

Department of Pediatrics, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan.

Department of Pediatrics, Niigata Medical-Care Cooperative, Kido Hospital, Niigata, Japan.

出版信息

Mod Rheumatol Case Rep. 2023 Jan 3;7(1):261-266. doi: 10.1093/mrcr/rxac034.

Abstract

Short stature is a common clinical condition in paediatric outpatient clinics and is associated with various clinical conditions, ranging from normal variants to severe diseases. Short stature is known to be caused by chronic inflammatory conditions, in which over-produced inflammatory cytokines are reported to be involved in growth suppression. Castleman disease is a rare lymphoproliferative disorder known as a chronic inflammatory disease with overproduction of interleukin 6, which often causes systemic symptoms such as fever, fatigue, weight loss, and night sweats. Here, we report the case of a 10-year-old female diagnosed with unicentric Castleman disease, who presented with short stature as the sole clinical sign but lacked typical systemic symptoms of Castleman disease. An elevated serum C-reactive protein level led us to suspect a chronic inflammatory condition, and we found an intra-abdominal tumour that was histopathologically confirmed as Castleman disease. The tumour removal resulted in a steady catch-up in her height in the six years following the surgery. We also present a brief review of relevant literature on paediatric cases of Castleman disease associated with growth impairment. Clinicians should be aware that chronic inflammatory conditions can cause growth impairment, which may be a key clinical manifestation of such conditions.

摘要

身材矮小是儿科门诊常见的临床病症,与多种临床情况相关,从正常变异到严重疾病不等。已知身材矮小由慢性炎症性疾病引起,据报道,过量产生的炎性细胞因子参与了生长抑制过程。Castleman病是一种罕见的淋巴增生性疾病,是一种伴有白细胞介素6过量产生的慢性炎症性疾病,常引起发热、疲劳、体重减轻和盗汗等全身症状。在此,我们报告一例10岁女性诊断为单中心Castleman病的病例,该患者仅以身材矮小作为唯一临床体征,但缺乏Castleman病典型的全身症状。血清C反应蛋白水平升高使我们怀疑存在慢性炎症性疾病,我们发现了一个腹腔内肿瘤,经组织病理学证实为Castleman病。肿瘤切除术后六年,她的身高稳步追赶上来。我们还简要回顾了与生长发育受损相关的Castleman病儿科病例的相关文献。临床医生应意识到慢性炎症性疾病可导致生长发育受损,这可能是此类疾病的关键临床表现。

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