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Anaesthetic considerations in Shrinking Man syndrome: two case reports

BACKGROUND: Shrinking Man syndrome (SMS) is a rare but often serious complication of dialysis-dependent end-stage renal disease, characterized by significant loss of height, bone pain, bone deformity, and skin itching. Patients with SMS always have abnormal facial changes and cardiovascular system d...

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Autores principales: Xu, Yan, Hong, Ying, Liu, Xin, Zhou, Li, Jiang, Chunling
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9835252/
https://www.ncbi.nlm.nih.gov/pubmed/36631742
http://dx.doi.org/10.1186/s12871-023-01978-5
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author Xu, Yan
Hong, Ying
Liu, Xin
Zhou, Li
Jiang, Chunling
author_facet Xu, Yan
Hong, Ying
Liu, Xin
Zhou, Li
Jiang, Chunling
author_sort Xu, Yan
collection PubMed
description BACKGROUND: Shrinking Man syndrome (SMS) is a rare but often serious complication of dialysis-dependent end-stage renal disease, characterized by significant loss of height, bone pain, bone deformity, and skin itching. Patients with SMS always have abnormal facial changes and cardiovascular system damage (manifested by hypertension, hypotension, cardiovascular calcification, and valvular heart disease), which pose a great challenge to anaesthesiologists. The purpose of this report is to describe our anaesthetic experience regarding two patients with SMS combined with alterations of the airway and cardiovascular system. CASE PRESENTATION: We describe two cases of SMS treated at West China Hospital, a tertiary care centre in Chengdu, China. All cases met the diagnostic criteria, which comprised 1) dialysis-dependent end-stage renal disease, 2) loss of height, and 3) bone pain and bone deformity. One patient had an anticipated difficult airway and moderate-to-severe mitral stenosis. The other patient presented with significant hypotension. Anaesthetic considerations included awake fibreoptic bronchoscopy-assisted tracheal intubation, real-time transoesophageal echocardiogram monitoring and individualized blood pressure management strategies. CONCLUSION: This case series highlights the importance of adequate preoperative assessment and preparation, as well as individualized anaesthetic management, in patients with SMS.
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spelling pubmed-98352522023-01-13 Anaesthetic considerations in Shrinking Man syndrome: two case reports Xu, Yan Hong, Ying Liu, Xin Zhou, Li Jiang, Chunling BMC Anesthesiol Case Report BACKGROUND: Shrinking Man syndrome (SMS) is a rare but often serious complication of dialysis-dependent end-stage renal disease, characterized by significant loss of height, bone pain, bone deformity, and skin itching. Patients with SMS always have abnormal facial changes and cardiovascular system damage (manifested by hypertension, hypotension, cardiovascular calcification, and valvular heart disease), which pose a great challenge to anaesthesiologists. The purpose of this report is to describe our anaesthetic experience regarding two patients with SMS combined with alterations of the airway and cardiovascular system. CASE PRESENTATION: We describe two cases of SMS treated at West China Hospital, a tertiary care centre in Chengdu, China. All cases met the diagnostic criteria, which comprised 1) dialysis-dependent end-stage renal disease, 2) loss of height, and 3) bone pain and bone deformity. One patient had an anticipated difficult airway and moderate-to-severe mitral stenosis. The other patient presented with significant hypotension. Anaesthetic considerations included awake fibreoptic bronchoscopy-assisted tracheal intubation, real-time transoesophageal echocardiogram monitoring and individualized blood pressure management strategies. CONCLUSION: This case series highlights the importance of adequate preoperative assessment and preparation, as well as individualized anaesthetic management, in patients with SMS. BioMed Central 2023-01-12 /pmc/articles/PMC9835252/ /pubmed/36631742 http://dx.doi.org/10.1186/s12871-023-01978-5 Text en © The Author(s) 2023 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Xu, Yan
Hong, Ying
Liu, Xin
Zhou, Li
Jiang, Chunling
Anaesthetic considerations in Shrinking Man syndrome: two case reports
title Anaesthetic considerations in Shrinking Man syndrome: two case reports
title_full Anaesthetic considerations in Shrinking Man syndrome: two case reports
title_fullStr Anaesthetic considerations in Shrinking Man syndrome: two case reports
title_full_unstemmed Anaesthetic considerations in Shrinking Man syndrome: two case reports
title_short Anaesthetic considerations in Shrinking Man syndrome: two case reports
title_sort anaesthetic considerations in shrinking man syndrome: two case reports
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9835252/
https://www.ncbi.nlm.nih.gov/pubmed/36631742
http://dx.doi.org/10.1186/s12871-023-01978-5
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