Wang Lin, Ye Yang, Li Delian, Li Kexing, Yang Peng, Hu Huanrui, Liu Xiaoliang, Zhao Yongmei
Department of Pediatrics, Longquanyi District of Chengdu Maternity & Child Health Care Hospital, Chengdu, Sichuan, China.
Division of Vascular Surgery, Department of General Surgery, West China Hospital, Sichuan University, Chengdu, China.
Front Pediatr. 2025 May 13;13:1563277. doi: 10.3389/fped.2025.1563277. eCollection 2025.
Cyanosis is a common clinical finding in infants and children. Particularly, central cyanosis can be associated with significant and potentially life-threatening diseases. Acquired methemoglobinemia is a rare but severe or even fatal cause of cyanosis in infants. Due to its rarity, timely diagnosis and appropriate management, particularly in infants, can be challenging in a clinical setting.
We report the case of a previously healthy 49-day-old female infant who presented with central cyanosis. Four hours prior to presentation, her guardian had inappropriately prepared her milk formula using spinach juice. This critical clue hinted to us that this infant might suffer from acquired methemoglobinemia. Upon blood sampling, her blood appeared chocolate brown in color. Furthermore, arterial blood gas analysis revealed abnormal findings, with a significantly elevated percentage of methemoglobinemia at 44.7%. Regarding the history of inappropriate formula preparation using vegetable juice and the abnormal finding of methemoglobinemia, a diagnosis of acquired methemoglobinemia was proposed. Other causes of methemoglobinemia were further excluded. Treatment with methylene blue and vitamin C was immediately initiated. Encouragingly, the cyanosis of this infant resolved 1 h later, with normal results of the repeated blood gas analysis. This infant was discharged home 2 days later and had no abnormal findings during the follow-up.
In this study, we reported a rare case of acquired methemoglobinemia in a 49-day-old infant. Inappropriate preparation of the infant milk formula with spinach juice was the potential cause of methemoglobinemia in this case, which presented with central cyanosis. Our findings also suggested that pediatricians should be aware of acquired methemoglobinemia as a potential cause of cyanosis in infants.
发绀是婴幼儿常见的临床体征。尤其是中枢性发绀可能与严重且潜在危及生命的疾病相关。获得性高铁血红蛋白血症是婴幼儿发绀的一种罕见但严重甚至致命的病因。由于其罕见性，在临床环境中及时诊断和恰当处理，尤其是对于婴幼儿，可能具有挑战性。
我们报告一例先前健康的49日龄女婴出现中枢性发绀的病例。在就诊前4小时，其监护人用菠菜汁不当配制了她的配方奶。这一关键线索提示我们该婴儿可能患有获得性高铁血红蛋白血症。采血时，她的血液呈巧克力棕色。此外，动脉血气分析显示异常结果，高铁血红蛋白血症百分比显著升高至44.7%。鉴于使用蔬菜汁不当配制配方奶的病史以及高铁血红蛋白血症的异常发现，提出了获得性高铁血红蛋白血症的诊断。进一步排除了其他高铁血红蛋白血症的病因。立即开始用亚甲蓝和维生素C进行治疗。令人鼓舞的是，该婴儿的发绀在1小时后消退，重复血气分析结果正常。该婴儿在2天后出院，随访期间无异常发现。
在本研究中，我们报告了一例49日龄婴儿罕见的获得性高铁血红蛋白血症病例。用菠菜汁不当配制婴儿配方奶是该病例中高铁血红蛋白血症的潜在病因，表现为中枢性发绀。我们的研究结果还提示儿科医生应意识到获得性高铁血红蛋白血症是婴幼儿发绀的潜在病因。
