A Very Uncommon Case of Pulmonary Arterial Hypertension

Azar, Jehad; Varasteh, Ali; Iltchev, Daniel; Soliman, Mona; Báez, Víctor H.; Altaqi, Basel

doi:10.12691/ajmcr-7-5-2

Cited by 4 publications

(12 citation statements)

References 15 publications

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“…Azar et al 17 reported a 35-year-old woman presenting to the hospital with painful lower limb oedema, dyspnoea, gingival bleeding and ecchymosis that had progressively worsened over 6 months. Physical examination showed poor dental hygiene, corkscrew hair and crackles on the respiratory exam.…”

Section: Discussionmentioning

confidence: 99%

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Scurvy-induced pulmonary arterial hypertension

et al. 2023

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Pulmonary arterial hypertension (PAH) is a progressive disease of the pulmonary vasculature that results in precapillary pulmonary hypertension. PAH is caused by a group of clinical conditions involving multiple organ systems. Several cases have been reported in the literature demonstrating an association between vitamin C deficiency and PAH. Low endothelial nitric oxide levels in the pulmonary vasculature, combined with the inappropriate activation of hypoxia-inducible transcription factors, seen in patients with ascorbic acid deficiency, are believed to be the main contributors to the pathogenesis of pulmonary vasculopathy and the exaggerated pulmonary vasoconstrictive response seen in patients with scurvy-induced PAH. Vitamin C supplementation is considered the definitive treatment.

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Section: Discussionmentioning

confidence: 99%

“…Finally, Azar et al ,17 described a patient who presented with progressive exertional dyspnoea and signs of advanced right heart failure. The initial diagnostic workup did not reveal any significant findings.…”

Section: Discussionmentioning

confidence: 99%

Scurvy-induced pulmonary arterial hypertension

et al. 2023

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“…This means that scurvy should be considered in the differential diagnosis of patients with such symptoms, in particular when plasma vitamin C levels are very low. Two of the patients in the case reports died [ 40 , 43 ]. Two case reports diagnosed scurvy in patients with sepsis indicating that they can coexist [ 49 , 50 ].…”

Section: Discussionmentioning

confidence: 99%

“…Experimental vitamin C deprivation in healthy volunteers has led to dyspnea, chest pain, edema, fatigue and reduced autonomic reflexes [30,[34][35][36][37][38]. Case reports of scurvy have reported hypotension, tachypnea, and tachycardia [39][40][41][42][43][44][45][46], which are important symptoms of sepsis [3,6,7]. In the case reports, vitamin C deficiency also caused dyspnea [42][43][44][45][46][47][48], chest pain [42], edema [40-43, 46, 47], petecchia and ecchymoses [39][40][41][42][43][44][45][46][47][48][49][50], fatigue [39,40,45,46] and musculoskeletal pains [40,42,47] which are also symptoms of sepsis [7].…”

Section: Ethical Concerns With Not Treating Overt Vitamin C Deficiencymentioning

confidence: 99%

“…Case reports of scurvy have reported hypotension, tachypnea, and tachycardia [39][40][41][42][43][44][45][46], which are important symptoms of sepsis [3,6,7]. In the case reports, vitamin C deficiency also caused dyspnea [42][43][44][45][46][47][48], chest pain [42], edema [40-43, 46, 47], petecchia and ecchymoses [39][40][41][42][43][44][45][46][47][48][49][50], fatigue [39,40,45,46] and musculoskeletal pains [40,42,47] which are also symptoms of sepsis [7]. This means that scurvy should be considered in the differential diagnosis of patients with such symptoms, in particular when plasma vitamin C levels are very low.…”

Section: Ethical Concerns With Not Treating Overt Vitamin C Deficiencymentioning

confidence: 99%

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Abrupt termination of vitamin C from ICU patients may increase mortality: secondary analysis of the LOVIT trial

Hemilä

Chalker²

2022

Eur J Clin Nutr

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Background The LOVIT trial examined the effect of vitamin C on sepsis patients, and concluded that in adults with sepsis receiving vasopressor therapy in the ICU, those who received 4-day intravenous vitamin C had a higher risk of death or persistent organ dysfunction at 28 days than those who received placebo. The aim of this study was to determine whether the abrupt termination of vitamin C administration could explain the increased mortality in the vitamin C group. Methods We used Cox regression with two time periods to model the distribution of deaths over the first 11 days in the LOVIT trial. Results Compared with a uniform difference between vitamin C and placebo groups over the 11-day follow-up period, addition of a separate vitamin C effect starting from day 5 improved the fit of the Cox model (p = 0.026). There was no difference in mortality between the groups during the 4-day vitamin C administration with RR = 0.97 (95% CI: 0.65–1.44). During the week after the sudden termination of vitamin C, there were 57 deaths in the vitamin C group, but only 32 deaths in the placebo group, with RR = 1.9 (95% CI: 1.2–2.9; p = 0.004). Conclusion The increased mortality in the vitamin C group in the LOVIT trial is not explained by ongoing vitamin C administration, but by the abrupt termination of vitamin C. The LOVIT trial findings should not be interpreted as evidence against vitamin C therapy for critically ill patients.

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Vitamin C deficiency can lead to pulmonary hypertension: a systematic review of case reports

Hemilä,

de Man

2024

BMC Pulm Med

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Background In the early literature, unintentional vitamin C deficiency in humans was associated with heart failure. Experimental vitamin C deficiency in guinea pigs caused enlargement of the heart. The purpose of this study was to collect and analyze case reports on vitamin C and pulmonary hypertension. Methods We searched Pubmed and Scopus for case studies in which vitamin C deficiency was considered to be the cause of pulmonary hypertension. We selected reports in which pulmonary hypertension was diagnosed by echocardiography or catheterization, for any age, sex, or dosage of vitamin C. We extracted quantitative data for our analysis. We used the mean pulmonary artery pressure (mPAP) as the outcome of primary interest. Results We identified 32 case reports, 21 of which were published in the last 5 years. Dyspnea was reported in 69%, edema in 53% and fatigue in 28% of the patients. Vitamin C plasma levels, measured in 27 cases, were undetectable in 24 and very low in 3 cases. Diet was poor in 30 cases and 17 cases had neuropsychiatric disorders. Right ventricular enlargement was reported in 24 cases. During periods of vitamin C deficiency, the median mPAP was 48 mmHg (range 29–77 mmHg; N = 28). After the start of vitamin C administration, the median mPAP was 20 mmHg (range 12–33 mmHg; N = 18). For the latter 18 cases, mPAP was 2.4-fold (median) higher during vitamin C deficiency. Pulmonary vascular resistance (PVR) during vitamin C deficiency was reported for 9 cases, ranging from 4.1 to 41 Wood units. PVR was 9-fold (median; N = 5) higher during vitamin C deficiency than during vitamin C administration. In 8 cases, there was direct evidence that the cases were pulmonary artery hypertension (PAH). Probably the majority of the remaining cases were also PAH. Conclusions The cases analyzed in our study indicate that pulmonary hypertension can be one explanation for the reported heart failure of scurvy patients in the early literature. It would seem sensible to measure plasma vitamin C levels of patients with PH and examine the effects of vitamin C administration.

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A Very Uncommon Case of Pulmonary Arterial Hypertension

Cited by 4 publications

References 15 publications

Scurvy-induced pulmonary arterial hypertension

Scurvy-induced pulmonary arterial hypertension

Abrupt termination of vitamin C from ICU patients may increase mortality: secondary analysis of the LOVIT trial

Vitamin C deficiency can lead to pulmonary hypertension: a systematic review of case reports

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