Guo Rui, Dong Chunhua, Zhai Yunpeng, Zhao Huashan, Lv Longfei, Zhang Shisong
Department of Thoracic and Tumor Surgery, Children's Hospital Affiliated to Shandong University, Jinan, Shandong, China.
Department of Thoracic and Tumor Surgery, Jinan Children's Hospital, Jinan, Shandong, China.
Front Pediatr. 2024 Aug 14;12:1442347. doi: 10.3389/fped.2024.1442347. eCollection 2024.
Congenital pulmonary sequestration is a rare lung anomaly that can be classified as intralobar pulmonary sequestration or extralobar lung sequestration (ELS). Infradiaphragmatic pulmonary sequestration is a rare type of ELS. Furthermore, intrathoracic kidney (ITK) is a rare disease that can be associated with a congenital diaphragmatic hernia (CHD) in 0.25% of cases. We report the first case of infradiaphragmatic pulmonary sequestration and ITK associated with CDH in a child.
The patient, male, aged 6 months, visited our hospital 2 months prior due to shortness of breath. Based on chest ultrasonography and enhanced computed tomography (CT) examination, infradiaphragmatic pulmonary sequestration and ITK were considered to be associated with CDH. The patient was admitted to our hospital for treatment. After admission, his blood pressure was 85/61 mmHg, there was no hematuria or proteinuria, creatinine was 14 µmol/L, and urea nitrogen was 2.96 mmol/L, all of which showed no abnormalities. A complete preoperative examination was performed prior to surgical treatment. Thoracoscopy revealed that the right kidney had herniated into the chest cavity on the posterolateral side of the diaphragm. The right kidney was returned to the abdominal cavity, the hernia sac was opened, and a bright red lesion tissue with clear boundaries and an abnormal blood vessel supply was observed. After cutting off the abnormal blood vessels, LigaSure TM was used to remove the diseased tissue, and the renal fat sacs and renal tissue were visible. Intermittent suturing of the hernia ring was performed to seal the diaphragmatic hernia. Postoperative pathological examination revealed infradiaphragmatic pulmonary sequestration. The postoperative recovery of the patient was smooth, and a chest CT scan at 2 months showed that the right kidney had returned to the abdominal cavity and the right diaphragm was in the normal position.
Infradiaphragmatic pulmonary sequestration and ITK associated with CDH is extremely rare. A diagnosis and appropriate surgical planning can be developed using enhanced CT. For infradiaphragmatic pulmonary sequestration located at the top of the hernia sac in CHD, thoracoscopic resection of the infradiaphragmatic pulmonary sequestration and repair of the diaphragmatic hernia is feasible and effective.
先天性肺隔离症是一种罕见的肺部异常，可分为叶内型肺隔离症或叶外型肺隔离症（ELS）。膈下肺隔离症是ELS的一种罕见类型。此外，胸腔内肾（ITK）是一种罕见疾病，在0.25%的病例中可与先天性膈疝（CHD）相关。我们报告了首例儿童膈下肺隔离症和ITK合并CHD的病例。
该患者为6个月大男性，2个月前因呼吸急促前来我院就诊。根据胸部超声和增强计算机断层扫描（CT）检查，考虑膈下肺隔离症和ITK与CHD相关。患者入院接受治疗。入院后，其血压为85/61 mmHg，无血尿或蛋白尿，肌酐为14 µmol/L，尿素氮为2.96 mmol/L，均无异常。在手术治疗前进行了全面的术前检查。胸腔镜检查发现右肾疝入膈肌后外侧的胸腔内。将右肾回纳至腹腔，打开疝囊，观察到一个边界清晰、血管供应异常的鲜红色病变组织。切断异常血管后，使用LigaSure TM切除病变组织，可见肾脂肪囊和肾组织。间断缝合疝环以封闭膈疝。术后病理检查显示为膈下肺隔离症。患者术后恢复顺利，术后2个月的胸部CT扫描显示右肾已回纳至腹腔，右膈肌位置正常。
膈下肺隔离症和ITK合并CHD极为罕见。使用增强CT可进行诊断并制定适当的手术方案。对于CHD中位于疝囊顶部的膈下肺隔离症，胸腔镜下切除膈下肺隔离症并修复膈疝是可行且有效的。
