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Successful treatment of polyarteritis nodosa with intestinal necrosis in a Chinese boy: a case report

Polyarteritis nodosa (PAN) is a systemic necrotizing inflammatory disease of the medium and small arteries which has variable clinical manifestations, course, and organ involvement. Intestinal necrosis resulting from PAN is rare, and successful treatment of such cases is even more uncommon. Here, we...

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Autores principales: Guo, Rui, Liu, Hongzhen, Liu, Xiumei, Liu, Wei, Zhang, Shisong
Formato: Online Artículo Texto
Lenguaje:English
Publicado: AME Publishing Company 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8825934/
https://www.ncbi.nlm.nih.gov/pubmed/35242662
http://dx.doi.org/10.21037/tp-21-378
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author Guo, Rui
Liu, Hongzhen
Liu, Xiumei
Liu, Wei
Zhang, Shisong
author_facet Guo, Rui
Liu, Hongzhen
Liu, Xiumei
Liu, Wei
Zhang, Shisong
author_sort Guo, Rui
collection PubMed
description Polyarteritis nodosa (PAN) is a systemic necrotizing inflammatory disease of the medium and small arteries which has variable clinical manifestations, course, and organ involvement. Intestinal necrosis resulting from PAN is rare, and successful treatment of such cases is even more uncommon. Here, we report the first successful treatment of PAN with intestinal necrosis in a young Chinese child. A 5-year-old boy was admitted to our hospital with a 5-day history of abdominal pain. The patient underwent an emergency exploratory laparotomy, which showed skipping necrosis of the intestinal wall. Intestinal resection and anastomosis failed to improve his abdominal pain. The patient’s blood pressure remained high (140/120 mmHg), despite captopril treatment. The erythrocyte sedimentation rate (ESR) and D-dimer level were also elevated at (106.00 mm/h and 11.16 mg/L, respectively), as was the 24-hour urine protein (197.6 mg/24 h). Echocardiography revealed that the diameters of the left and right coronary arteries were increased (0.45 and 0.49 cm, respectively). Ultrasound showed polyarteritis in the anterior tibial, radial, iliac, and renal arteries. Histopathologic examination revealed elastic fiber rupture and partial mesenteric arteriolar stenosis with occlusion. After 2 months of treatment with systemic methylprednisolone, cyclophosphamide, and prednisone, the patient’s abdominal pain was relieved. Furthermore, the patient’s ESR and D-dimer levels had reduced to 5 mm/h and 0.63 mg/L, respectively; his 24-hour urine protein was normal (60.0 mg/24 h); and his blood pressure had dropped to 101/46 mmHg. The left and right coronary arteries had reduced to 0.35 and 0.38 cm in diameter, respectively; however, no significant improvement was observed in the other vessels involved. Unfortunately, the child’s parents did not continue to seek medical attention; therefore, his long-term outcome is unknown. In this case, the patient was operated on immediately after symptom onset. The postoperative infection was also quickly controlled, avoiding the occurrence of septic shock. Vascular B-ultrasound and pathology aided in establishing a clear and timely diagnosis, which allowed systematic medical treatment to be delivered, achieving good short-term results.
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spelling pubmed-88259342022-03-02 Successful treatment of polyarteritis nodosa with intestinal necrosis in a Chinese boy: a case report Guo, Rui Liu, Hongzhen Liu, Xiumei Liu, Wei Zhang, Shisong Transl Pediatr Case Report Polyarteritis nodosa (PAN) is a systemic necrotizing inflammatory disease of the medium and small arteries which has variable clinical manifestations, course, and organ involvement. Intestinal necrosis resulting from PAN is rare, and successful treatment of such cases is even more uncommon. Here, we report the first successful treatment of PAN with intestinal necrosis in a young Chinese child. A 5-year-old boy was admitted to our hospital with a 5-day history of abdominal pain. The patient underwent an emergency exploratory laparotomy, which showed skipping necrosis of the intestinal wall. Intestinal resection and anastomosis failed to improve his abdominal pain. The patient’s blood pressure remained high (140/120 mmHg), despite captopril treatment. The erythrocyte sedimentation rate (ESR) and D-dimer level were also elevated at (106.00 mm/h and 11.16 mg/L, respectively), as was the 24-hour urine protein (197.6 mg/24 h). Echocardiography revealed that the diameters of the left and right coronary arteries were increased (0.45 and 0.49 cm, respectively). Ultrasound showed polyarteritis in the anterior tibial, radial, iliac, and renal arteries. Histopathologic examination revealed elastic fiber rupture and partial mesenteric arteriolar stenosis with occlusion. After 2 months of treatment with systemic methylprednisolone, cyclophosphamide, and prednisone, the patient’s abdominal pain was relieved. Furthermore, the patient’s ESR and D-dimer levels had reduced to 5 mm/h and 0.63 mg/L, respectively; his 24-hour urine protein was normal (60.0 mg/24 h); and his blood pressure had dropped to 101/46 mmHg. The left and right coronary arteries had reduced to 0.35 and 0.38 cm in diameter, respectively; however, no significant improvement was observed in the other vessels involved. Unfortunately, the child’s parents did not continue to seek medical attention; therefore, his long-term outcome is unknown. In this case, the patient was operated on immediately after symptom onset. The postoperative infection was also quickly controlled, avoiding the occurrence of septic shock. Vascular B-ultrasound and pathology aided in establishing a clear and timely diagnosis, which allowed systematic medical treatment to be delivered, achieving good short-term results. AME Publishing Company 2022-01 /pmc/articles/PMC8825934/ /pubmed/35242662 http://dx.doi.org/10.21037/tp-21-378 Text en 2022 Translational Pediatrics. All rights reserved. https://creativecommons.org/licenses/by-nc-nd/4.0/Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0 (https://creativecommons.org/licenses/by-nc-nd/4.0/) .
spellingShingle Case Report
Guo, Rui
Liu, Hongzhen
Liu, Xiumei
Liu, Wei
Zhang, Shisong
Successful treatment of polyarteritis nodosa with intestinal necrosis in a Chinese boy: a case report
title Successful treatment of polyarteritis nodosa with intestinal necrosis in a Chinese boy: a case report
title_full Successful treatment of polyarteritis nodosa with intestinal necrosis in a Chinese boy: a case report
title_fullStr Successful treatment of polyarteritis nodosa with intestinal necrosis in a Chinese boy: a case report
title_full_unstemmed Successful treatment of polyarteritis nodosa with intestinal necrosis in a Chinese boy: a case report
title_short Successful treatment of polyarteritis nodosa with intestinal necrosis in a Chinese boy: a case report
title_sort successful treatment of polyarteritis nodosa with intestinal necrosis in a chinese boy: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8825934/
https://www.ncbi.nlm.nih.gov/pubmed/35242662
http://dx.doi.org/10.21037/tp-21-378
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