Nargund Ashwini R, Madhumathi Davanam S, Premalatha Chennagiri S, Rao Clementina Rama, Appaji Lingappa, Lakshmidevi Vishweshvaraih
Department of Hematopathology, Kidwai Memorial Institute of Oncology, Bangalore, Karnataka, India.
J Pediatr Hematol Oncol. 2010 Aug;32(6):e223-6. doi: 10.1097/MPH.0b013e3181e62663.
Chediak Higashi Syndrome (CHS) is a rare autosomal recessive disease, characterized by partial oculocutaneous albinism, frequent pyogenic infections, and the presence of abnormal large granules in leukocytes and other granule containing cells. The abnormal granules are readily seen in blood and marrow granulocytes. About 50% to 85% of patients eventually enter an accelerated phase, manifested by fever, lymphadenopathy, anemia, jaundice, neutropenia, thrombocytopenia, and widespread lymphohistiocytic organ infiltrates. The first accelerated phase of CHS may occur shortly after birth or several years later. Most patients undergo a variable period of recurrent infections before going into the accelerated phase. Therefore, primary presentation in the accelerated phase is unusual. This case was referred to our institution that is a tertiary care cancer centre, with a clinical diagnosis of lymphoma/leukemia. Hence this interesting case of CHS in accelerated phase at presentation is described. The child had 1-month history of fever, bilateral neck swellings, and loss of appetite. On the basis of the clinical presentation, hematologic, and histopathologic findings, a diagnosis of accelerated phase of CHS was made. The child was treated with antipyretics, antibiotics, and stem cell transplantation was suggested to him. When the child presents to a hospital with oculocutaneous albinism and recurrent infections, careful examination of the peripheral blood smear by an experienced morphologist cannot be overemphasized. A high degree of awareness and early recognition of the syndrome, could lead to the institution of the only possible curative treatment, bone marrow transplant, before the accelerated phase supervenes.
切东综合征(CHS)是一种罕见的常染色体隐性疾病,其特征为部分眼皮肤白化病、频繁的化脓性感染,以及白细胞和其他含颗粒细胞中存在异常大颗粒。异常颗粒在血液和骨髓粒细胞中很容易看到。约50%至85%的患者最终进入加速期,表现为发热、淋巴结病、贫血、黄疸、中性粒细胞减少、血小板减少以及广泛的淋巴组织细胞器官浸润。CHS的首个加速期可能在出生后不久或数年后出现。大多数患者在进入加速期之前会经历一段反复感染的时期。因此,以加速期作为主要表现并不常见。该病例被转诊至我们作为三级医疗癌症中心的机构,临床诊断为淋巴瘤/白血病。因此,本文描述了这例初诊时处于加速期的CHS的有趣病例。该患儿有1个月的发热、双侧颈部肿胀及食欲不振病史。根据临床表现、血液学和组织病理学检查结果,诊断为CHS加速期。给予患儿退烧药和抗生素治疗,并建议进行干细胞移植。当患儿因眼皮肤白化病和反复感染就诊时,经验丰富的形态学家对其外周血涂片进行仔细检查至关重要。对该综合征有高度的认识并早期识别,可能会在加速期出现之前开展唯一可能的治愈性治疗,即骨髓移植。