Cargando…

Life-Threatening Tongue and Retropharyngeal Hemorrhage in a Patient with Hemophilia A with Inhibitors

Patient: Male, 71 Final Diagnosis: Hemophilia A Symptoms: Dysarthria • dysphagia • dyspnea Medication: — Clinical Procedure: Cricothyrotomy • tracheotomy Specialty: Hematology OBJECTIVE: Unusual clinical course BACKGROUND: Massive tongue hemorrhage in patients with hemophilia is a medical emergency...

Descripción completa

Detalles Bibliográficos
Autores principales: Kageyama, Yuki, Matsumoto, Takeshi, Tawara, Isao, Wada, Hideo, Katayama, Naoyuki
Formato: Online Artículo Texto
Lenguaje:English
Publicado: International Scientific Literature, Inc. 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6647615/
https://www.ncbi.nlm.nih.gov/pubmed/31303639
http://dx.doi.org/10.12659/AJCR.916151
_version_ 1783437749587542016
author Kageyama, Yuki
Matsumoto, Takeshi
Tawara, Isao
Wada, Hideo
Katayama, Naoyuki
author_facet Kageyama, Yuki
Matsumoto, Takeshi
Tawara, Isao
Wada, Hideo
Katayama, Naoyuki
author_sort Kageyama, Yuki
collection PubMed
description Patient: Male, 71 Final Diagnosis: Hemophilia A Symptoms: Dysarthria • dysphagia • dyspnea Medication: — Clinical Procedure: Cricothyrotomy • tracheotomy Specialty: Hematology OBJECTIVE: Unusual clinical course BACKGROUND: Massive tongue hemorrhage in patients with hemophilia is a medical emergency because it can lead to airway obstruction. However, managing bleeding in patients with inhibitors is more difficult than in patients without inhibitors. We report a case of life-threatening massive tongue and retropharyngeal hematoma in a patient with hemophilia A who had inhibitors. CASE REPORT: The patient was a 71-year-old man with severe hemophilia A with high-responding inhibitors. He was admitted to our hospital with dysarthria and dysphagia secondary to a massive tongue hematoma. Although bypassing therapy was started immediately after admission, he rapidly developed an airway obstruction and cardiopulmonary arrest secondary to suffocation. Cardiopulmonary resuscitation and surgical cricothyrotomy were performed, which restored his pulse and breathing. On day 5 of hospitalization, he underwent tracheotomy under inhibitor-neutralizing therapy, and we began emicizumab on day 19 of hospitalization to prevent further bleeding events. He recovered and was transferred to another hospital for rehabilitation on day 64 of hospitalization. CONCLUSIONS: Because tongue hematomas progress dramatically within a few days, prompt airway maintenance by tracheotomy under appropriate hemostatic therapy must be considered. Furthermore, emicizumab induction after primary hemostasis prevents further bleeding. We suggest that initiating emicizumab therapy is a good choice to prevent further bleeding after critical bleeding events if the patient has not received the drug previously.
format Online
Article
Text
id pubmed-6647615
institution National Center for Biotechnology Information
language English
publishDate 2019
publisher International Scientific Literature, Inc.
record_format MEDLINE/PubMed
spelling pubmed-66476152019-08-09 Life-Threatening Tongue and Retropharyngeal Hemorrhage in a Patient with Hemophilia A with Inhibitors Kageyama, Yuki Matsumoto, Takeshi Tawara, Isao Wada, Hideo Katayama, Naoyuki Am J Case Rep Articles Patient: Male, 71 Final Diagnosis: Hemophilia A Symptoms: Dysarthria • dysphagia • dyspnea Medication: — Clinical Procedure: Cricothyrotomy • tracheotomy Specialty: Hematology OBJECTIVE: Unusual clinical course BACKGROUND: Massive tongue hemorrhage in patients with hemophilia is a medical emergency because it can lead to airway obstruction. However, managing bleeding in patients with inhibitors is more difficult than in patients without inhibitors. We report a case of life-threatening massive tongue and retropharyngeal hematoma in a patient with hemophilia A who had inhibitors. CASE REPORT: The patient was a 71-year-old man with severe hemophilia A with high-responding inhibitors. He was admitted to our hospital with dysarthria and dysphagia secondary to a massive tongue hematoma. Although bypassing therapy was started immediately after admission, he rapidly developed an airway obstruction and cardiopulmonary arrest secondary to suffocation. Cardiopulmonary resuscitation and surgical cricothyrotomy were performed, which restored his pulse and breathing. On day 5 of hospitalization, he underwent tracheotomy under inhibitor-neutralizing therapy, and we began emicizumab on day 19 of hospitalization to prevent further bleeding events. He recovered and was transferred to another hospital for rehabilitation on day 64 of hospitalization. CONCLUSIONS: Because tongue hematomas progress dramatically within a few days, prompt airway maintenance by tracheotomy under appropriate hemostatic therapy must be considered. Furthermore, emicizumab induction after primary hemostasis prevents further bleeding. We suggest that initiating emicizumab therapy is a good choice to prevent further bleeding after critical bleeding events if the patient has not received the drug previously. International Scientific Literature, Inc. 2019-07-15 /pmc/articles/PMC6647615/ /pubmed/31303639 http://dx.doi.org/10.12659/AJCR.916151 Text en © Am J Case Rep, 2019 This work is licensed under Creative Common Attribution-NonCommercial-NoDerivatives 4.0 International (CC BY-NC-ND 4.0 (https://creativecommons.org/licenses/by-nc-nd/4.0/) )
spellingShingle Articles
Kageyama, Yuki
Matsumoto, Takeshi
Tawara, Isao
Wada, Hideo
Katayama, Naoyuki
Life-Threatening Tongue and Retropharyngeal Hemorrhage in a Patient with Hemophilia A with Inhibitors
title Life-Threatening Tongue and Retropharyngeal Hemorrhage in a Patient with Hemophilia A with Inhibitors
title_full Life-Threatening Tongue and Retropharyngeal Hemorrhage in a Patient with Hemophilia A with Inhibitors
title_fullStr Life-Threatening Tongue and Retropharyngeal Hemorrhage in a Patient with Hemophilia A with Inhibitors
title_full_unstemmed Life-Threatening Tongue and Retropharyngeal Hemorrhage in a Patient with Hemophilia A with Inhibitors
title_short Life-Threatening Tongue and Retropharyngeal Hemorrhage in a Patient with Hemophilia A with Inhibitors
title_sort life-threatening tongue and retropharyngeal hemorrhage in a patient with hemophilia a with inhibitors
topic Articles
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6647615/
https://www.ncbi.nlm.nih.gov/pubmed/31303639
http://dx.doi.org/10.12659/AJCR.916151
work_keys_str_mv AT kageyamayuki lifethreateningtongueandretropharyngealhemorrhageinapatientwithhemophiliaawithinhibitors
AT matsumototakeshi lifethreateningtongueandretropharyngealhemorrhageinapatientwithhemophiliaawithinhibitors
AT tawaraisao lifethreateningtongueandretropharyngealhemorrhageinapatientwithhemophiliaawithinhibitors
AT wadahideo lifethreateningtongueandretropharyngealhemorrhageinapatientwithhemophiliaawithinhibitors
AT katayamanaoyuki lifethreateningtongueandretropharyngealhemorrhageinapatientwithhemophiliaawithinhibitors