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Lomitapide treatment in a female with homozygous familial hypercholesterolaemia: a case report
BACKGROUND: Homozygous familial hypercholesterolaemia (FH) is an autosomal-dominant inherited disease presenting with highly elevated low-density lipoprotein cholesterol (LDL-C) levels. Untreated, the patient can develop atherosclerosis and cardiovascular disease already in adolescence. Treatment wi...
Autores principales: | , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Oxford University Press
2020
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7047050/ https://www.ncbi.nlm.nih.gov/pubmed/32128483 http://dx.doi.org/10.1093/ehjcr/ytaa020 |
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author | Littmann, Karin Szummer, Karolina Hagström, Hannes Dolapcsiev, Karoly Brinck, Jonas Eriksson, Mats |
author_facet | Littmann, Karin Szummer, Karolina Hagström, Hannes Dolapcsiev, Karoly Brinck, Jonas Eriksson, Mats |
author_sort | Littmann, Karin |
collection | PubMed |
description | BACKGROUND: Homozygous familial hypercholesterolaemia (FH) is an autosomal-dominant inherited disease presenting with highly elevated low-density lipoprotein cholesterol (LDL-C) levels. Untreated, the patient can develop atherosclerosis and cardiovascular disease already in adolescence. Treatment with statins and ezetimibe is usually not sufficient and LDL apheresis is often required. Lomitapide, an inhibitor of the microsomal triglyceride transfer protein, reduces LDL-C and triglyceride levels and can be used alone or in combination with other therapies in homozygous FH. However, experience with this agent is still limited. CASE SUMMARY: We present a young female who was diagnosed with homozygous FH at 6 years of age. She shows a complete lack of normal LDL receptor activity and no cholesterol-lowering effect from statins. The patient was treated with LDL apheresis from 7 years of age. When LDL apheresis treatment extended to twice a week, she began to experience adverse effects, including catheter-related complications, infections, and hospital admissions. When lomitapide treatment was initiated, the frequency of apheresis reduced, the LDL-C levels improved and she has not had any further hospital admissions since. Initially, she suffered from gastrointestinal disturbances. However, after 3 years of treatment with lomitapide 20 mg/day, the patient has not experienced any adverse effects. DISCUSSION: In this female with homozygous FH adding lomitapide treatment to LDL apheresis has contributed to improved LDL-C levels, a reduction in LDL apheresis sessions and enhanced quality of life. No adverse effects have been reported. These findings suggest that lomitapide can be a drug of choice in patients with homozygous FH. |
format | Online Article Text |
id | pubmed-7047050 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2020 |
publisher | Oxford University Press |
record_format | MEDLINE/PubMed |
spelling | pubmed-70470502020-03-03 Lomitapide treatment in a female with homozygous familial hypercholesterolaemia: a case report Littmann, Karin Szummer, Karolina Hagström, Hannes Dolapcsiev, Karoly Brinck, Jonas Eriksson, Mats Eur Heart J Case Rep Grand Rounds BACKGROUND: Homozygous familial hypercholesterolaemia (FH) is an autosomal-dominant inherited disease presenting with highly elevated low-density lipoprotein cholesterol (LDL-C) levels. Untreated, the patient can develop atherosclerosis and cardiovascular disease already in adolescence. Treatment with statins and ezetimibe is usually not sufficient and LDL apheresis is often required. Lomitapide, an inhibitor of the microsomal triglyceride transfer protein, reduces LDL-C and triglyceride levels and can be used alone or in combination with other therapies in homozygous FH. However, experience with this agent is still limited. CASE SUMMARY: We present a young female who was diagnosed with homozygous FH at 6 years of age. She shows a complete lack of normal LDL receptor activity and no cholesterol-lowering effect from statins. The patient was treated with LDL apheresis from 7 years of age. When LDL apheresis treatment extended to twice a week, she began to experience adverse effects, including catheter-related complications, infections, and hospital admissions. When lomitapide treatment was initiated, the frequency of apheresis reduced, the LDL-C levels improved and she has not had any further hospital admissions since. Initially, she suffered from gastrointestinal disturbances. However, after 3 years of treatment with lomitapide 20 mg/day, the patient has not experienced any adverse effects. DISCUSSION: In this female with homozygous FH adding lomitapide treatment to LDL apheresis has contributed to improved LDL-C levels, a reduction in LDL apheresis sessions and enhanced quality of life. No adverse effects have been reported. These findings suggest that lomitapide can be a drug of choice in patients with homozygous FH. Oxford University Press 2020-02-13 /pmc/articles/PMC7047050/ /pubmed/32128483 http://dx.doi.org/10.1093/ehjcr/ytaa020 Text en © The Author(s) 2020. Published by Oxford University Press on behalf of the European Society of Cardiology. http://creativecommons.org/licenses/by-nc/4.0/ This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com |
spellingShingle | Grand Rounds Littmann, Karin Szummer, Karolina Hagström, Hannes Dolapcsiev, Karoly Brinck, Jonas Eriksson, Mats Lomitapide treatment in a female with homozygous familial hypercholesterolaemia: a case report |
title | Lomitapide treatment in a female with homozygous familial hypercholesterolaemia: a case report |
title_full | Lomitapide treatment in a female with homozygous familial hypercholesterolaemia: a case report |
title_fullStr | Lomitapide treatment in a female with homozygous familial hypercholesterolaemia: a case report |
title_full_unstemmed | Lomitapide treatment in a female with homozygous familial hypercholesterolaemia: a case report |
title_short | Lomitapide treatment in a female with homozygous familial hypercholesterolaemia: a case report |
title_sort | lomitapide treatment in a female with homozygous familial hypercholesterolaemia: a case report |
topic | Grand Rounds |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7047050/ https://www.ncbi.nlm.nih.gov/pubmed/32128483 http://dx.doi.org/10.1093/ehjcr/ytaa020 |
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