RESUMO

Abstract The current case report describes two cases of alkaptonuric ochronosis for anesthetic management. Alkaptonuria is a rare genetic orphan disease of tyrosine metabolism characterized by an accumulation of homogentisic acid in cartilage and connective tissues. Patients present most commonly for orthopedic joint surgery due to progressive arthropathy that can be misdiagnosed many a times. However respiratory, airway, cardiovascular and genitourinary systems complications can occur with age progressing. Restricted range of motion of cervical spine may lead to difficulty with airway management. In addition, degenerative changes and stiffness of lumbar spine due to ochronosis would make neuraxial blockade challenging. Although this inherited condition is extremely rare, anesthesiologists should be aware of its existence and prepare for management of potential challenging problems. This report highlights special care and precautions that need to be taken during anesthetic management.

Resumo Este relato descreve o manejo anestésico em dois casos de ocronose alcaptonúrica. Alcaptonúria é uma doença genética rara do metabolismo de tirosina caracterizada por acúmulo de ácido homogentísico em cartilagem e tecidos conjuntivos. Os pacientes geralmente recorrem à cirurgia ortopédica devido à artropatia progressiva, que, muitas vezes, pode ser diagnosticada incorretamente. No entanto, complicações das vias respiratórias, cardiovasculares e geniturinárias podem ocorrer com o avanço da idade. A restrição de mobilidade da coluna cervical pode levar ao manejo difícil das vias aéreas. Além disso, as alterações degenerativas e a rigidez da coluna lombar devido à ocronose podem tornar o bloqueio neuroaxial um desafio. Embora essa condição hereditária seja extremamente rara, os anestesiologistas devem estar cientes de sua existência e se preparar para o manejo de potenciais problemas desafiadores. Este relato destaca os cuidados e as precauções especiais que devem ser tomadas durante o manejo anestésico.

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Abstract Background and objectives: Fluctuations of female sex hormones during menstrual cycle influence pain perception. Endogenous pain inhibition is impaired in follicular phase of menstrual cycle. We tested the primary hypothesis that the women having surgery during their follicular phase have more acute pain and require higher opioids than those in the luteal phase, and secondarily we tested that women who have surgery during their follicular phase have more incisional pain at 3 month postoperatively. Methods: 127 adult females having laparoscopic cholecystectomy were randomized to have surgery during the luteal or follicular phase of their menstrual cycle. Standardized anesthesia and pain management regimen was given to all patients. Pain and analgesic consumption were evaluated in post-anesthesia care unit and every 4 h in the first 24 h. Adverse effects were questioned every 4 h. Time to oral intake and ambulation were recorded. Post-surgical pain, hospital anxiety, depression scale, SF-12 questionnaire were evaluated at 1 and 3 month visits. Results: There was no difference in acute pain scores and analgesic consumption through the 24 h period, Visual Analog Scale at 24 h was 1.5 ± 1.5 cm for follicular group 1.4 ± 1.7 cm for luteal group (p = 0.57). Persistent postoperative pain was significantly more common one and at three month, with an incidence was 33% and 32% in the patients at follicular phase versus 16% and 12% at luteal phase, respectively. The Visual Analog Scale at one and at three month was 1.6 ± 0.7 cm and 1.8 ± 0.8 cm for follicular group and 2.7 ± 1.3 cm and 2.9 ± 1.7 cm in the luteal group (p = 0.02), respectively. There were no significant differences between the groups with respect to anxiety and depression, SF-12 scores at either time. Nausea was more common in follicular-phase group (p = 0.01) and oral feeding time was shorter in follicular phase (5.9 ± 0.9 h) than in luteal phase (6.8 ± 1.9 h, p = 0.02). Conclusions: Although persistent postoperative pain was significantly more common one and three months after surgery the magnitude of the pain was low. Our results do not support scheduling operations to target particular phases of the menstrual cycle.

Resumo Justificativa e objetivos: As flutuações dos hormônios sexuais femininos durante o ciclo menstrual influenciam a percepção da dor. A inibição endógena da dor é prejudicada na fase folicular do ciclo menstrual. Testamos a hipótese primária de que cirurgias em mulheres durante a fase folicular têm mais dor aguda e precisam de mais opioide do que aquelas na fase lútea e a hipótese secundária testada foi que as cirurgias em mulheres durante a fase folicular têm mais dor incisional aos três meses de pós-operatório. Métodos: No total, 127 mulheres adultas submetidas à colecistectomia laparoscópica foram randomizadas para serem operadas durante a fase lútea ou folicular de seus ciclos menstruais. Um regime padronizado para anestesia e tratamento da dor foi administrado a todas as pacientes. A dor e o consumo de analgésico foram avaliados na sala de recuperação pós-anestésica e a cada quatro horas nas primeiras 24 horas. Efeitos adversos foram avaliados a cada quatro horas. Os tempo para ingestão oral e deambulação foram registrados. Dor pós-cirúrgica, ansiedade hospitalar, escala de depressão e questionário SF-12 foram avaliados em visitas feitas no primeiro e terceiro meses. Resultados: Não houve diferença nos escores de dor aguda e no consumo de analgésicos durante o período de 24 horas, Escala Visual Analógica em 24 horas foi de 1,5 ± 1,5 cm para o grupo folicular e 1,4 ± 1,7 cm para o grupo lúteo (p = 0,57). A dor persistente no pós-operatório foi significativamente mais prevalente no primeiro e terceiro mês, com incidência de 33% e 32% nas pacientes em fase folicular versus 16% e 12% na fase lútea, respectivamente. A Escala Visual Analógica no primeiro e terceiro mês foi 1,6 ± 0,7 cm e 1,8 ± 0,8 cm no grupo folicular e 2,7 ± 1,3 cm e 2,9 ± 1,7 cm no grupo lúteo (p = 0,02), respectivamente. Não houve diferença significativa entre os grupos em relação à ansiedade e à depressão, escore SF-12 em ambos os tempos. Náusea foi mais comum no grupo na fase folicular (p = 0,01) e o tempo para alimentação oral foi menor na fase folicular (5,9 ± 0,9 horas) do que na fase lútea (6,8 ± 1,9 horas, p = 0,02). Conclusões: Embora a dor persistente no pós-operatório tenha sido significativamente mais prevalente no primeiro e no terceiro mês após a cirurgia, a magnitude da dor foi baixa. Nossos resultados não apoiam o agendamento de cirurgias tendo como alvo fases específicas do ciclo menstrual.

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BACKGROUND AND OBJECTIVES: Fluctuations of female sex hormones during menstrual cycle influence pain perception. Endogenous pain inhibition is impaired in follicular phase of menstrual cycle. We tested the primary hypothesis that the women having surgery during their follicular phase have more acute pain and require higher opioids than those in the luteal phase, and secondarily we tested that women who have surgery during their follicular phase have more incisional pain at 3 month postoperatively. METHODS: 127 adult females having laparoscopic cholecystectomy were randomized to have surgery during the luteal or follicular phase of their menstrual cycle. Standardized anesthesia and pain management regimen was given to all patients. Pain and analgesic consumption were evaluated in post-anesthesia care unit and every 4h in the first 24h. Adverse effects were questioned every 4h. Time to oral intake and ambulation were recorded. Post-surgical pain, hospital anxiety, depression scale, SF-12 questionnaire were evaluated at 1 and 3 month visits. RESULTS: There was no difference in acute pain scores and analgesic consumption through the 24h period, Visual Analog Scale at 24h was 1.5±1.5cm for follicular group 1.4±1.7cm for luteal group (p=0.57). Persistent postoperative pain was significantly more common one and at three month, with an incidence was 33% and 32% in the patients at follicular phase versus 16% and 12% at luteal phase, respectively. The Visual Analog Scale at one and at three month was 1.6±0.7cm and 1.8±0.8cm for follicular group and 2.7±1.3cm and 2.9±1.7cm in the luteal group (p=0.02), respectively. There were no significant differences between the groups with respect to anxiety and depression, SF-12 scores at either time. Nausea was more common in follicular-phase group (p=0.01) and oral feeding time was shorter in follicular phase (5.9±0.9h) than in luteal phase (6.8±1.9h, p=0.02). CONCLUSIONS: Although persistent postoperative pain was significantly more common one and three months after surgery the magnitude of the pain was low. Our results do not support scheduling operations to target particular phases of the menstrual cycle.

RESUMO

The current case report describes two cases of alkaptonuric ochronosis for anesthetic management. Alkaptonuria is a rare genetic orphan disease of tyrosine metabolism characterized by an accumulation of homogentisic acid in cartilage and connective tissues. Patients present most commonly for orthopedic joint surgery due to progressive arthropathy that can be misdiagnosed many a times. However respiratory, airway, cardiovascular and genitourinary systems complications can occur with age progressing. Restricted range of motion of cervical spine may lead to difficulty with airway management. In addition, degenerative changes and stiffness of lumbar spine due to ochronosis would make neuraxial blockade challenging. Although this inherited condition is extremely rare, anesthesiologists should be aware of its existence and prepare for management of potential challenging problems. This report highlights special care and precautions that need to be taken during anesthetic management.

RESUMO

Abstract We report a case of venous air embolism during abdominal myomectomy. Although true incidence of venous air embolism is not known, in literature most of reported cases are belongs to sitting position craniotomies. Many of those are subclinical, and diagnostic methods have varying degrees of sensitivity and specificity. At time of suspicion, prevention of any subsequent air emboli is the cornerstone of treatment.

Resumo Relatamos um caso de embolia gasosa durante miomectomia abdominal. Embora a incidência exata de embolia gasosa não seja conhecida, a maioria dos casos relatados na literatura se refere à posição sentada em craniotomias. Muitos casos são subclínicos e os métodos diagnósticos têm diferentes graus de sensibilidade e especificidade. No momento da suspeita, a prevenção de qualquer êmbolo de ar subsequente é a chave fundamental do tratamento.

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We report a case of venous air embolism during abdominal myomectomy. Although true incidence of venous air embolism is not known, in literature most of reported cases are belongs to sitting position craniotomies. Many of those are subclinical, and diagnostic methods have varying degrees of sensitivity and specificity. At time of suspicion, prevention of any subsequent air emboli is the cornerstone of treatment.

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