Brazilian Journal of Pain
https://brjp.org.br/article/doi/10.63231/2595-0118.20250025-en
Brazilian Journal of Pain
Case Report

Treatment of refractory bone pain with intravenous lidocaine, ketamine and magnesium sulfate in a patient with CamuratiEngelmann Disease. Case report

Tratamento de dor óssea refratária com lidocaína, cetamina e sulfato de magnésio por via venosa em paciente portadora da Doença de Camurati-Engelmann. Relato de caso

Emica Shimozono; Vanessa Henriques Carvalho

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Abstract

BACKGROUND AND OBJECTIVES: Camurati-Engelmann Disease (CED) or Progressive Diaphyseal Dysplasia is a rare genetic syndrome, characterized by progressive periosteal and endostal hyperostosis mainly in bone diaphysis. CED can presents intense pain that is difficult to control with conventional treatment using corticosteroids and non-steroidal anti-inflammatory drugs (NSAIDs), which may be secondary to diaphyseal hyperostosis and narrowed medullary canal, and in most severe cases, there is a need surgical decompression. The aim of this study was to report the case of a patient diagnosed with CED, in whom sympathetic venous blockade (SVB) was performed by intravenous infusion of lidocaine, ketamine and magnesium sulfate associated with opioids to treat severe chronic pain refractory to several usual treatment methods, bringing to light new therapeutic proposals for pain in this rare disease.

CASE REPORT: A female patient, 38 years old, diagnosed with CED at 16 years old, with intense chronic widespread pain, functional limitation and previous unsuccessful treatments. She underwent a serial intravenous infusion of lidocaine, ketamine, and magnesium sulfate associated with opioid use, resulting in a significant reduction in pain intensity, improvement in physical limitation and increasing the interval between pain attacks. This report is unprecedented, with no previous description of this therapeutic approach to control pain in this disease.

CONCLUSION: Treatment with intravenous infusion of lidocaine, ketamine and magnesium sulfate associated with opioid can be a safe option for treating refractory pain related to CED.

Keywords

Carmurati-Engelmann syndrome; Chronic pain; Lidocaine; Opioid analgesics; Rare diseases

Resumo

JUSTIFICATIVA E OBJETIVOS: A Doença de Camurati-Engelmann (DCE), ou Displasia Diafisária Progressiva, é uma síndrome genética rara, caracterizada por hiperostose progressiva periostal e endostal, principalmente nas diáfises dos ossos. A doença pode cursar com quadro álgico de difícil controle, mesmo em vigência de tratamento convencional com corticosteroides e anti-inflamatórios não esteroides (AINES), secundariamente à hiperostose diafisária e redução do canal medular, sendo necessária, nos casos mais graves, a descompressão cirúrgica. O objetivo deste estudo foi descrever o caso de uma paciente com diagnóstico de DCE, tendo sido realizado o bloqueio simpático venoso (BSV) através de infusão por via venosa de lidocaína, cetamina e sulfato de magnésio associado ao uso de opioide para tratar o quadro de dor crônica intensa e refratária aos diversos tratamentos experimentados, trazendo à tona novas propostas terapêuticas álgicas para essa doença rara.

RELATO DO CASO: Trata-se de uma paciente do sexo feminino, 38 anos, com diagnóstico de DCE aos 16 anos de idade, portadora de quadro álgico crônico intenso generalizado, limitação funcional e tratamentos anteriores sem sucesso. Foi submetida a BSV seriado semanal, associado ao uso de opioide, resultando em importante redução na intensidade da dor, melhora da limitação física e aumento do intervalo entre as crises. Este relato é inédito, não havendo descrição anterior dessa associação terapêutica para o controle da dor.

CONCLUSÃO: A associação do bloqueio simpático venoso ao uso de opioide pode ser uma opção segura de tratamento da dor refratária causada pela DCE.

Palavras-chave

Analgésicos opioides; Doenças raras; Dor crônica; Lidocaína; Síndrome de Camurati-Engelmann

References

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Submitted date:
05/01/2024

Accepted date:
03/29/2025

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