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Case report: primary osteonecrosis associated with thrombophilia-hypofibrinolysis and worsened by testosterone therapy
BACKGROUND: Familial and acquired thrombophilia are often etiologic for idiopathic hip and jaw osteonecrosis (ON), and testosterone therapy (TT) can interact with thrombophilia, worsening ON. CASE PRESENTATION: Case 1: A 62-year-old Caucasian male (previous deep venous thrombosis), on warfarin 1 yea...
Autores principales: | , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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BioMed Central
2017
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5368894/ https://www.ncbi.nlm.nih.gov/pubmed/28361003 http://dx.doi.org/10.1186/s12878-017-0076-x |
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author | Jarman, Michael Ian Lee, Kevin Kanevsky, Ariel Min, Sarah Schlam, Ilana Mahida, Chris Huda, Ali Milgrom, Alexander Goldenberg, Naila Glueck, Charles J. Wang, Ping |
author_facet | Jarman, Michael Ian Lee, Kevin Kanevsky, Ariel Min, Sarah Schlam, Ilana Mahida, Chris Huda, Ali Milgrom, Alexander Goldenberg, Naila Glueck, Charles J. Wang, Ping |
author_sort | Jarman, Michael Ian |
collection | PubMed |
description | BACKGROUND: Familial and acquired thrombophilia are often etiologic for idiopathic hip and jaw osteonecrosis (ON), and testosterone therapy (TT) can interact with thrombophilia, worsening ON. CASE PRESENTATION: Case 1: A 62-year-old Caucasian male (previous deep venous thrombosis), on warfarin 1 year for atrial fibrillation (AF), had non-specific right hip-abdominal pain for 2 years. CT scan revealed bilateral femoral head ON without collapse. Coagulation studies revealed Factor V Leiden (FVL) heterozygosity, 4G/4G plasminogen activator inhibitor (PAI) homozygosity, high anti-cardiolipin (ACLA) IgM antibodies, and endothelial nitric oxide (NO) synthase (eNOS) T786C homozygosity (reduced conversion of L-arginine to NO, required for bone health). Apixaban 5 mg twice daily was substituted for warfarin; and L-arginine 9 g/day was started to increase NO. On Apixaban for 8 months, he became asymptomatic. Case 2: A 32-year-old hypogonadal Caucasian male had 10 years of unexplained tooth loss, progressing to primary jaw ON with cavitation 8 months after starting TT gel 50 mg/day. Coagulation studies revealed FVL heterozygosity, PAI 4G/4G homozygosity, and the lupus anticoagulant. TT was discontinued. Jaw pain was sharply reduced within 2 months. CONCLUSIONS: Idiopathic ON, often caused by thrombophilia-hypofibrinolysis, is worsened by TT, and its progression may be slowed or stopped by discontinuation of TT and, thereafter, anticoagulation. Recognition of thrombophilia-hypofibrinolysis before joint collapse facilitates anticoagulation which may stop ON, preserving joints. |
format | Online Article Text |
id | pubmed-5368894 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2017 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
spelling | pubmed-53688942017-03-30 Case report: primary osteonecrosis associated with thrombophilia-hypofibrinolysis and worsened by testosterone therapy Jarman, Michael Ian Lee, Kevin Kanevsky, Ariel Min, Sarah Schlam, Ilana Mahida, Chris Huda, Ali Milgrom, Alexander Goldenberg, Naila Glueck, Charles J. Wang, Ping BMC Hematol Case Report BACKGROUND: Familial and acquired thrombophilia are often etiologic for idiopathic hip and jaw osteonecrosis (ON), and testosterone therapy (TT) can interact with thrombophilia, worsening ON. CASE PRESENTATION: Case 1: A 62-year-old Caucasian male (previous deep venous thrombosis), on warfarin 1 year for atrial fibrillation (AF), had non-specific right hip-abdominal pain for 2 years. CT scan revealed bilateral femoral head ON without collapse. Coagulation studies revealed Factor V Leiden (FVL) heterozygosity, 4G/4G plasminogen activator inhibitor (PAI) homozygosity, high anti-cardiolipin (ACLA) IgM antibodies, and endothelial nitric oxide (NO) synthase (eNOS) T786C homozygosity (reduced conversion of L-arginine to NO, required for bone health). Apixaban 5 mg twice daily was substituted for warfarin; and L-arginine 9 g/day was started to increase NO. On Apixaban for 8 months, he became asymptomatic. Case 2: A 32-year-old hypogonadal Caucasian male had 10 years of unexplained tooth loss, progressing to primary jaw ON with cavitation 8 months after starting TT gel 50 mg/day. Coagulation studies revealed FVL heterozygosity, PAI 4G/4G homozygosity, and the lupus anticoagulant. TT was discontinued. Jaw pain was sharply reduced within 2 months. CONCLUSIONS: Idiopathic ON, often caused by thrombophilia-hypofibrinolysis, is worsened by TT, and its progression may be slowed or stopped by discontinuation of TT and, thereafter, anticoagulation. Recognition of thrombophilia-hypofibrinolysis before joint collapse facilitates anticoagulation which may stop ON, preserving joints. BioMed Central 2017-03-27 /pmc/articles/PMC5368894/ /pubmed/28361003 http://dx.doi.org/10.1186/s12878-017-0076-x Text en © The Author(s). 2017 Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. |
spellingShingle | Case Report Jarman, Michael Ian Lee, Kevin Kanevsky, Ariel Min, Sarah Schlam, Ilana Mahida, Chris Huda, Ali Milgrom, Alexander Goldenberg, Naila Glueck, Charles J. Wang, Ping Case report: primary osteonecrosis associated with thrombophilia-hypofibrinolysis and worsened by testosterone therapy |
title | Case report: primary osteonecrosis associated with thrombophilia-hypofibrinolysis and worsened by testosterone therapy |
title_full | Case report: primary osteonecrosis associated with thrombophilia-hypofibrinolysis and worsened by testosterone therapy |
title_fullStr | Case report: primary osteonecrosis associated with thrombophilia-hypofibrinolysis and worsened by testosterone therapy |
title_full_unstemmed | Case report: primary osteonecrosis associated with thrombophilia-hypofibrinolysis and worsened by testosterone therapy |
title_short | Case report: primary osteonecrosis associated with thrombophilia-hypofibrinolysis and worsened by testosterone therapy |
title_sort | case report: primary osteonecrosis associated with thrombophilia-hypofibrinolysis and worsened by testosterone therapy |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5368894/ https://www.ncbi.nlm.nih.gov/pubmed/28361003 http://dx.doi.org/10.1186/s12878-017-0076-x |
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