Reversible severe pulmonary hypertension related to scurvy in children.

BACKGROUND: Severe pulmonary hypertension (PH) in childhood is rare and can manifest as a life-threatening episode. We present 2 children with restrictive dietary habits with severe pulmonary hypertension secondary to scurvy and iron deficiency anemia with treatment and outcome. CASE PRESENTATION: The first case is a 2-year-old boy who presented with vomiting, diarrhea, and fever. After rehydration, he had recurrent episodes of hypotension with intermittent abdominal pain. Fluid resuscitation and inotropic medication were given. Then he suddenly collapsed. After 4-min cardiopulmonary resuscitation, his hemodynamic was stabilized. Most of the medical workup was unremarkable except for PH from the echocardiogram with estimated systolic pulmonary artery pressure (PAP) at 67 mmHg. Transient PH was diagnosed, and milrinone was prescribed. Since he had restrictive dietary habits and sclerotic rim at epiphysis in chest films, his vitamin C level was tested and reported low-level result. The second case is a 6-year-old boy with acute dyspnea, a month of low-grade fever, mild cyanosis, and a swollen left knee. Echocardiogram indicated moderate TR with estimated systolic PAP at 56 mmHg (systolic blood pressure 90 mmHg). Milrinone was given. Right cardiac catheterization showed PAP 66/38 (mean 50) mmHg and PVRi 5.7 WU.m2. Other medical conditions causing PH were excluded. With a history of improper dietary intake and clinical suspicion of scurvy, vitamin C was tested and reported undetectable level. Administration of vitamin C in both cases rapidly reversed pulmonary hypertension. CONCLUSION: Pediatric PH related to vitamin C deficiency can manifest with a wide range of symptoms, varying from mild and nonspecific to severe life-threatening episodes characterized by pulmonary hypertensive crises. PH associated with scurvy is entirely reversible with appropriate investigation, diagnosis, and treatment. Our report highlights the importance of considering nutritional deficiencies as potential confounding factors in pediatric PH, emphasizing the need for comprehensive evaluation and management of these patients.

Pediatric scurvy case report: a novel presentation with deep vein thrombosis secondary to large bilateral spontaneous iliac subperiosteal hematomas.

BACKGROUND: Scurvy is an uncommon disease in developed countries caused by deficiency of vitamin C. We present a case of scurvy in a 14-year-old male with autism with both novel presentation and imaging findings. This case had the novel presentation of lower limb deep vein thrombosis (DVT) secondary to compression of the external iliac vein from large bilateral iliac wing subperiosteal hematomas. Subperiosteal hematoma is a well-recognised feature of scurvy but large and bilateral pelvic subperiosteal hematoma causing DVT has not previously been described. CASE PRESENTATION: A 14 year old Caucasian male with background of autism and severe dietary restriction presented with lower limb swelling and immobility. He was diagnosed with lower limb DVT. Further investigation revealed an iron deficiency anaemia, and he was found on MRI to have large bilateral subperiosteal iliac hematomata causing compression of the iliac vessels. He improved following treatment with vitamin C replacement and follow-up imaging demonstrated resolution of the DVT and hematoma. CONCLUSION: DVT is rare in children and when diagnosed should prompt investigation as to the underlying cause. This case demonstrates an unusual cause of DVT and as an unusual presentation of paediatric scurvy.

A 3-Year-Old With Gingival Hemorrhage and Musculoskeletal Pain.

A 3-year-old had spontaneous gingival hemorrhage and bilateral limb weakness with inability to bear weight. He had no preceding oral trauma or recent infection, took no regular medications, and had no recent use of aspirin or nonsteroidal anti-inflammatory drugs; his diet was limited to primarily chicken nuggets and milk. What is the diagnosis and what would you do next?

What Do We C in Children With Scurvy? A Case Series Focused on Musculoskeletal Symptoms.

OBJECTIVES: Vitamin C deficiency in children commonly presents with musculoskeletal symptoms such as gait disturbance, refusal to bear weight, and bone or joint pain. We aimed to identify features that could facilitate early diagnosis of scurvy and estimate the cost of care for patients with musculoskeletal symptoms related to scurvy. METHODS: We conducted a retrospective chart review of patients at a single site with diagnostic codes for vitamin C deficiency, ascorbic acid deficiency, or scurvy. Medical records were reviewed to identify characteristics including presenting symptoms, medical history, and diagnostic workup. The Pediatric Health Information System was used to estimate diagnostic and hospitalization costs for each patient. RESULTS: We identified 47 patients with a diagnosis of scurvy, 49% of whom had a neurodevelopmental disorder. Sixteen of the 47 had musculoskeletal symptoms and were the focus of the cost analysis. Three of the 16 had moderate or severe malnutrition, and 3 had overweight or obesity. Six patients presented to an emergency department for care, 11 were managed inpatient, and 3 required critical care. Diagnostic workups included MRI, computed tomography, echocardiogram, endoscopy, lumbar puncture, and/or EEG. Across all patients evaluated, the cost of emergency department utilization, imaging studies, diagnostic procedures, and hospitalization totaled $470 144 (median $14 137 per patient). CONCLUSIONS: Children across the BMI spectrum, particularly those with neurodevelopmental disorders, can develop vitamin C deficiency. Increased awareness of scurvy and its signs and symptoms, particularly musculoskeletal manifestations, may reduce severe disease, limit adverse effects related to unnecessary tests/treatments, and facilitate high-value care.

Scurvy. A forgotten pseudovasculitis.

Scurvy is a nutritional disease caused by ascorbic acid (vitamin C) deficiency. Althought currently it is a rare disease, we should considerer it in the differential diagnosis of purpura and arthritis in patients with restrictive diets. We present the case of a 49-year-old man with a history of a nutritional disorder presented to our hospital with generalized purpura and hemarthros. Following the anamnesis and laboratory findings, rheumatological, infectious and hematological etiologies were excluded. Finally, the diagnosis of scurvy was made upon demostration poor levels of vitamin C and a spectacular response to nutritional supplements. We compare this case with 19 similar cases reported in the medical literature.

A 7-year-old boy with scurvy owing to coeliac disease.

ABBREVIATIONS: ESR: erythrocyte sedimentation rate; Hb: haemoglobin; HSP: Henoch-Schönlein purpura; WCC: white-cell count.

Increase in the frequency of scurvy in children with food selectivity: A case series. / Aumento de la frecuencia de escorbuto en niños con selectividad alimentaria: serie de casos.

Scurvy is a disease caused by vitamin C deficiency. Although rare, in recent years, the number of scurvy cases in children with eating disorders has increased. Its manifestations are varied because vitamin C is a cofactor in numerous processes, such as collagen synthesis. The typical skin manifestations include petechiae, bruising, and hyperkeratosis. Mucosal involvement manifests as gingivitis with hypertrophy, bleeding, and loss of teeth. The diagnosis is based on clinical findings and may be confirmed by measuring plasma vitamin C levels. The objective of this study was to describe a cohort of patients diagnosed with scurvy in recent years, its clinical manifestations, and findings in relation to their eating behavior and neurodevelopmental disorders.

El escorbuto es una enfermedad producida por déficit de vitamina C. Aunque es poco frecuente, en los últimos años observamos un incremento de casos en niños con trastornos de la conducta alimentaria. Sus manifestaciones son variadas, ya que esta vitamina actúa como cofactor en numerosos procesos, como la síntesis de colágeno. Las manifestaciones cutáneas características son las petequias, equimosis e hiperqueratosis. El compromiso mucoso se manifiesta como gingivitis con hipertrofia, hemorragias y pérdida de piezas dentarias. El diagnóstico es clínico y puede confirmarse mediante la determinación de la vitamina C plasmática. El objetivo de este trabajo es describir una cohorte de pacientes diagnosticados en los últimos años, manifestaciones clínicas y hallazgos en relación con su conducta alimentaria y trastornos del neurodesarrollo.

Scurvy presenting as difficulty in walking in a child.

Scurvy, a disease caused by ascorbic acid (vitamin C) deficiency, is a rare disease in the modern world. We report a case of a boy in middle childhood, with a background of autism, presenting bed-bound due to progressive bilateral lower limb pain, with concomitant rashes, bleeding gums and worsening lethargy. Detailed dietary history revealed a severely restricted diet. Physical examination showed bilateral lower limb ecchymoses, perifollicular hemorrhages, perifollicular hyperkeratosis and cockscrew hairs which are pathognomonic features of scurvy. A low serum ascorbic acid level confirmed the diagnosis. Therapy with oral vitamin C supplement and rehabilitation with multidisciplinary care was successful, with complete resolution of symptoms. This case emphasises the importance of thorough dietary evaluation in children with autism and food selectivity presenting with non-specific symptoms. Physician awareness of nutritional deficiencies avoids unnecessary extensive investigations and sub-specialty referrals and translates to savings in medical expenses.

Gait Disturbance of Red Herrings, Oranges, and Lemons - A Case of Missed Vitamin C Deficiency and Lessons Learnt.

Vitamin C deficiency resulting in scurvy, is considered to be a rare nutritional disorder in developed countries, thus leading to underdiagnosis with exposure to unnecessary investigations and delay in appropriate treatment. The wide myriad of clinical signs and symptoms with which vitamin C deficiency can present (including haematological, musculoskeletal and vague constitutional symptoms that overlap with other common medical conditions), also contributes to this diagnostic challenge. Despite scurvy being habitually thought to be present in children with neurodevelopmental conditions such as autism spectrum disorder, other important at-risk groups that frequently tend to be forgotten include children with persistent fussy eating behaviour, and children with abnormal vitamin C metabolism. We hereunder present a case of a 10-year-old boy who presented to an acute general hospital for further investigation with gait disturbance. The lack of detailed nutritional assessment on presentation in the first instance led to a missed diagnosis of vitamin C deficiency, thus exposing the child to a wide array of unnecessary investigations and treatments. The added perplexity to the case resulting from false positive results of investigations performed as part of this child's workup, is also discussed.

Hemartrosis por escorbuto / Hemarthrosis and scurvy

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Dieta restrictiva en frutas como causa de escorbuto en niño de 7 años / Restricted diet in fruits causes scurvy in a child of 7 years old

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Lesiones purpúricas en manos / Purpura in hands

Presentamos el caso de un varón institucionalizado de 57 años, afectado por síndrome de Down, que presentó lesiones purpúricas en manos y extremidades inferiores tras administración de una dosis de amoxicilina-ácido clavulánico. Se sospechó una reacción de hipersensibilidad por fármacos, pero la reacción cutánea persistió tras la suspensión del antibiótico. Tras descartar otras etiologías, fue diagnosticado de escorbuto. El paciente fue tratado con suplementos de vitamina C, mostrando clara mejoría de las lesiones. El escorbuto no es un diagnóstico frecuente en la actualidad, sin embargo esta patología de otras épocas no ha desaparecido del todo. Se trata de una enfermedad que resulta fácil olvidar al plantearnos un diagnóstico diferencial y cuyo tratamiento con la suplementación vitamínica es simple y efectivo (AU)

We report the case of an institutionalized 57-year-old man affected by Down syndrome, with purpura in hands and lower extremities after the administration of a dose of amoxicillin-clavulanic acid. We suspected a drug hypersensitivity reaction but the skin reaction persisted after discontinuation of the antibiotic. After ruling out any other etiology, scurvy was diagnosed. The patient was treated with vitamin C supplements, showing a clear improvement. Scurvy is not a common diagnosis at the present time, but this disease from the past has not disappeared entirely. It can be easily overlooked in differential diagnosis, but vitamin supplementation is a simple and effective treatment (AU)

Scurvy in a patient with AIDS: case report / Escorbuto em paciente com AIDS: relato de caso

We report the case of a 35-year-old homeless alcoholic and illicit drug user, with AIDS, who was admitted to the emergency unit complaining of asthenia and a weight loss of 30kg over the preceding three months. Clinical and laboratory data confirmed a diagnosis of marasmus, bacterial pneumonia, chorioretinitis caused by Toxoplasma gondii and oral Candida infection. The patient also presented loss of tongue papillae, gingival hypertrophy, perifollicular hyperkeratosis and hemorrhage, coiled, corkscrew-like hair, anemia, hypoalbuminemia, increased C-reactive protein levels and low serum vitamin C levels. The patient developed severe gastric hemorrhage, with hemodynamic instability and terminal disseminated intravascular coagulopathy.

Relatamos o caso de um paciente alcoólatra e usuário de drogas ilícitas com 35 anos de idade, morador de rua com AIDS, admitido na Unidade de Emergência referindo astenia e perda ponderal de trinta quilos nos últimos três meses. Dados clínicos e laboratoriais confirmaram o diagnóstico de marasmo, pneumonia, corioretinite por Toxoplasma gondii e candidíase oral. Apresentava ainda: língua despapilada com hipertrofia gengival, hiperqueratose e hemorragia folicular associada a pêlos tipo saca-rolhas, anemia, hipoalbuminemia, aumento dos níveis de proteína C reativa com baixos níveis séricos de vitamina C. O paciente desenvolveu hemorragia gástrica grave, com instabilidade hemodinâmica e coagulação intravascular disseminada terminal.

Un caso de escorbuto en un joven del siglo XXI / A scurvy case in a XXI century young men

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Púrpura perifolicular: signo clásico de escorbuto / Perifollicular purpura: Classical sign of scurvy

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Pathoecology and paleodiet in Postclassic: Historic Maya from northern coastal Belize

This paper examines the synergism among diet, disease, and ecology at two related coastal Maya sites in Belize (Marco Gonzalez and San Pedro) for the Postclassic and Historic periods (1350-1650 AD), which immediately follow the Classic period collapse. Stable carbon- and nitrogen-isotope ratios in collagen and stable carbon-isotope ratios in structural carbonate were analysed for bones from 65 humans and a wide variety of faunal species. There are no apparent differences in whole diets or degree of carnivory between individuals with lesions indicative of anemia and those without, but those with lesions appear to have consumed significantly more C4 foods and protein from lower trophic levels. Non-specific infection (periostitis) and vitamin C deficiency (scurvy) are also present in high frequencies and appear to co-occur with lesions indicative of anemia, particularly in childhood. Individuals with scurvy also appear to have consumed significantly more C4 foods than normal individuals. Spondyloarthropathy is common in adults. These findings are discussed in light of: (1) the debate on how anemia versus scurvy are manifest and diagnosed, (2) Spanish ethnohistoric descriptions of the poor state of Maya health at the time of contact, and (3) the Osteological Paradox. We suggest that although this coastal environment exacerbated morbidity because of possible parasitic infection, the inhabitants were probably able to survive physiological stresses better than either their inland contemporaries or their modern counterparts.