Twin Premature Infants With Riboflavin and Biotin Deficiency Presenting With Refractory Lactic Acidosis, Rash, and Multiorgan Failure During Prolonged Parenteral Nutrition
We are reporting monochorionic, diamniotic twin premature infants born at 25 weeks and 6 days gestation with riboflavin (vitamin B2) and biotin (vitamin B7) deficiency, while on prolonged total parenteral nutrition (TPN) during vitamin shortage. They presented initially with skin rash, lactic acidos...
| Main Authors: | , , , , , , , |
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| Format: | Article |
| Language: | English |
| Published: |
SAGE Publishing
2023-04-01
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| Series: | Journal of Investigative Medicine High Impact Case Reports |
| Online Access: | https://doi.org/10.1177/23247096231168111 |
| _version_ | 1797842413420544000 |
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| author | Mohammad Amin Adie MD Maria Martes Gomez DO Jessica Yom MD Manuel Durand MD Fiona Wertheimer DO Rachel McGowan MS, RD Shoji Yano MD, PhD Rangasamy Ramanathan MD |
| author_facet | Mohammad Amin Adie MD Maria Martes Gomez DO Jessica Yom MD Manuel Durand MD Fiona Wertheimer DO Rachel McGowan MS, RD Shoji Yano MD, PhD Rangasamy Ramanathan MD |
| author_sort | Mohammad Amin Adie MD |
| collection | DOAJ |
| description | We are reporting monochorionic, diamniotic twin premature infants born at 25 weeks and 6 days gestation with riboflavin (vitamin B2) and biotin (vitamin B7) deficiency, while on prolonged total parenteral nutrition (TPN) during vitamin shortage. They presented initially with skin rash, lactic acidosis, and thrombocytopenia. Both twins progressed to severe respiratory failure, severe lactic acidosis, with refractory vasodilatory shock, pancytopenia, ischemic bowel injury, acute kidney injury, liver injury, and capillary leak syndrome leading to death of twin A. The surviving twin B was diagnosed with riboflavin and biotin deficiency that presented with abnormal metabolic work up suggestive of maple syrup urine disease, glutaric acidemia type 2, and X-linked adrenoleukodystrophy. Twin B was started on riboflavin and biotin supplementation at 41 days of life, with rapid improvement in clinical findings and laboratory abnormalities within days of starting biotin and riboflavin supplementation. He was discharged home in stable condition at 49 weeks of postmenstrual age. |
| first_indexed | 2024-04-09T16:47:30Z |
| format | Article |
| id | doaj.art-fb20042455894f3596a94c73ad8b1022 |
| institution | Directory Open Access Journal |
| issn | 2324-7096 |
| language | English |
| last_indexed | 2024-04-09T16:47:30Z |
| publishDate | 2023-04-01 |
| publisher | SAGE Publishing |
| record_format | Article |
| series | Journal of Investigative Medicine High Impact Case Reports |
| spelling | doaj.art-fb20042455894f3596a94c73ad8b10222023-04-22T11:33:23ZengSAGE PublishingJournal of Investigative Medicine High Impact Case Reports2324-70962023-04-011110.1177/23247096231168111Twin Premature Infants With Riboflavin and Biotin Deficiency Presenting With Refractory Lactic Acidosis, Rash, and Multiorgan Failure During Prolonged Parenteral NutritionMohammad Amin Adie MD0Maria Martes Gomez DO1Jessica Yom MD2Manuel Durand MD3Fiona Wertheimer DO4Rachel McGowan MS, RD5Shoji Yano MD, PhD6Rangasamy Ramanathan MD7Keck School of Medicine of University of Southern California, LAC+USC Medical Center, Los Angeles, CA, USAKeck School of Medicine of University of Southern California, LAC+USC Medical Center, Los Angeles, CA, USAKeck School of Medicine of University of Southern California, LAC+USC Medical Center, Los Angeles, CA, USAKeck School of Medicine of University of Southern California, LAC+USC Medical Center, Los Angeles, CA, USAKeck School of Medicine of University of Southern California, LAC+USC Medical Center, Los Angeles, CA, USAKeck School of Medicine of University of Southern California, LAC+USC Medical Center, Los Angeles, CA, USAKeck School of Medicine of University of Southern California, LAC+USC Medical Center, Los Angeles, CA, USAKeck School of Medicine of University of Southern California, LAC+USC Medical Center, Los Angeles, CA, USAWe are reporting monochorionic, diamniotic twin premature infants born at 25 weeks and 6 days gestation with riboflavin (vitamin B2) and biotin (vitamin B7) deficiency, while on prolonged total parenteral nutrition (TPN) during vitamin shortage. They presented initially with skin rash, lactic acidosis, and thrombocytopenia. Both twins progressed to severe respiratory failure, severe lactic acidosis, with refractory vasodilatory shock, pancytopenia, ischemic bowel injury, acute kidney injury, liver injury, and capillary leak syndrome leading to death of twin A. The surviving twin B was diagnosed with riboflavin and biotin deficiency that presented with abnormal metabolic work up suggestive of maple syrup urine disease, glutaric acidemia type 2, and X-linked adrenoleukodystrophy. Twin B was started on riboflavin and biotin supplementation at 41 days of life, with rapid improvement in clinical findings and laboratory abnormalities within days of starting biotin and riboflavin supplementation. He was discharged home in stable condition at 49 weeks of postmenstrual age.https://doi.org/10.1177/23247096231168111 |
| spellingShingle | Mohammad Amin Adie MD Maria Martes Gomez DO Jessica Yom MD Manuel Durand MD Fiona Wertheimer DO Rachel McGowan MS, RD Shoji Yano MD, PhD Rangasamy Ramanathan MD Twin Premature Infants With Riboflavin and Biotin Deficiency Presenting With Refractory Lactic Acidosis, Rash, and Multiorgan Failure During Prolonged Parenteral Nutrition Journal of Investigative Medicine High Impact Case Reports |
| title | Twin Premature Infants With Riboflavin and Biotin Deficiency Presenting With Refractory Lactic Acidosis, Rash, and Multiorgan Failure During Prolonged Parenteral Nutrition |
| title_full | Twin Premature Infants With Riboflavin and Biotin Deficiency Presenting With Refractory Lactic Acidosis, Rash, and Multiorgan Failure During Prolonged Parenteral Nutrition |
| title_fullStr | Twin Premature Infants With Riboflavin and Biotin Deficiency Presenting With Refractory Lactic Acidosis, Rash, and Multiorgan Failure During Prolonged Parenteral Nutrition |
| title_full_unstemmed | Twin Premature Infants With Riboflavin and Biotin Deficiency Presenting With Refractory Lactic Acidosis, Rash, and Multiorgan Failure During Prolonged Parenteral Nutrition |
| title_short | Twin Premature Infants With Riboflavin and Biotin Deficiency Presenting With Refractory Lactic Acidosis, Rash, and Multiorgan Failure During Prolonged Parenteral Nutrition |
| title_sort | twin premature infants with riboflavin and biotin deficiency presenting with refractory lactic acidosis rash and multiorgan failure during prolonged parenteral nutrition |
| url | https://doi.org/10.1177/23247096231168111 |
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