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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...

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Autores principales: Littmann, Karin, Szummer, Karolina, Hagström, Hannes, Dolapcsiev, Karoly, Brinck, Jonas, Eriksson, Mats
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2020
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.
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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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