Initial Manifestation of Acquired Hemophilia A After a Routine Tooth Extraction. A Case Report and Literature Review

Bennetts, Nicholas A.; Mergelmeyer, James E.; Reimer, Eric J.; Melville, James C.

doi:10.1016/j.joms.2017.08.005

Cited by 5 publications

(5 citation statements)

References 11 publications

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“…One of the most important points in hemophilia management is maintaining the airway under appropriate hemostatic therapy. Ten case reports in the last 20 years discuss airway obstruction secondary to tongue and/or retropharyngeal hemorrhage in patients with hemophilia who required airway maintenance by intubation or tracheotomy [4–13]. Five of the 10 patients required tracheotomy, 4 of the 10 patients suffered failed intubation and required emergency cricothyrotomy, and 1 of the 10 patients died.…”

Section: Discussionmentioning

confidence: 99%

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

et al. 2019

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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.

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Section: Discussionmentioning

confidence: 99%

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

et al. 2019

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“…We range 5 papers of AHA cases in the region of dentistry over references in NCBI Reference Sequences [4–8]. Only 3 papers concerning about tooth extraction of AHA patients have been reported [4–6].…”

Section: Discussionmentioning

confidence: 99%

A Case of a Patient Who Is Diagnosed with Mild Acquired Hemophilia A after Tooth Extraction Died of Acute Subdural Hematoma due to Head Injury

Kitamura

Sato

Ikami

et al. 2018

Case Reports in Dentistry

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Background Acquired hemophilia A (AHA) is a rare disorder which results from the presence of autoantibodies against blood coagulation factor VIII. The initial diagnosis is based on the detection of an isolated prolongation of the activated partial thromboplastin time (aPTT) with negative personal and family history of bleeding disorder. Definitive diagnosis is the identification of reduced FVIII levels with evidence of FVIII neutralizing activity. Case report We report a case of a 93-year-old female who was diagnosed as AHA after tooth extraction at her home clinic. Prolongation of aPTT and a reduction in factor VIII activity levels were observed with the presence of factor VIII inhibitor. AHA condition is mild. However, acute subdural hematoma of this patient occurred due to an unexpected accident in our hospital. Hematoma was gradually increased and the patient died 13 days after admission. Discussion Although AHA is mild, intracranial bleeding is a life-threatening condition. We also should pay attention to the presence of AHA patients when we extract teeth.

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“…Twelve cases appeared to be associated with a delayed diagnosis (Figure 2). *Three studies [16,17,26]. †Four studies [13,14,18,23].…”

Section: Numbermentioning

confidence: 99%

“…n: the number of studies; AHA: acquired hemophilia A Nine of these 12 patients either did not discuss all treatment options or did not mention consultation with a hemophilia expert (or both). Bleeding appeared to be controlled in 20 cases, although multiple hemostatic agents were required in 11 of 20 cases [12,16,18,21,[23][24][25][27][28][29][30]34] and considerable time was required to achieve bleeding control (hospitalization of ≥7 days) in nine of 20 cases [16,18,21,[23][24][25][26]29,34].…”

Section: Numbermentioning

confidence: 99%

Review of Potential Barriers to Effective Hemostatic Management of Acquired Hemophilia A by Non-Hemophilia Experts in the United States

Sharathkumar¹,

Mokdad²

2023

Cureus

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Acquired hemophilia A (AHA) is an ultra-rare autoimmune disorder caused by autoantibodies against factor VIII. It often presents with life-threatening bleeding to non-hemophilia experts, who have limited awareness of this condition. This review evaluated hemostatic management and identified barriers to optimal management of AHA by non-hemophilia experts in the United States through a literature review. AHA case reports published by non-hemophilia experts from January 2016 through November 2021 in nonhematology journals were critically reviewed for a chronology of clinical course and management, consultation with a hemophilia expert, referencing of available AHA recommendations, discussion of all hemostatic options, and bleed control outcomes; 24 case reports representing 24 patients were identified. Twelve patients had an apparent delay in diagnosis, 17 cases did not seek expert consultation, and 15 did not reference the 2009 International AHA Recommendations, including six in whom hemostatic treatment was not consistent with the recommendations. Of the 17 articles published after the 2017 AHA Guidance, eight did not reference them. Of the five articles published after the 2020 International Recommendations for AHA, three did not reference them. Overall, 14 articles did not discuss all available hemostatic treatment options. Four patients died. Our findings reveal variability in hemostatic management of AHA by nonhemophilia experts in the United States. Lack of AHA awareness remains a primary barrier for optimal management of AHA among non-hemophilia experts. Increasing education about existing AHA guidelines, including available therapies and access to expert care at hemophilia treatment centers, may help improve the outcomes of patients with AHA.

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Initial Manifestation of Acquired Hemophilia A After a Routine Tooth Extraction. A Case Report and Literature Review

Cited by 5 publications

References 11 publications

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

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

A Case of a Patient Who Is Diagnosed with Mild Acquired Hemophilia A after Tooth Extraction Died of Acute Subdural Hematoma due to Head Injury

Review of Potential Barriers to Effective Hemostatic Management of Acquired Hemophilia A by Non-Hemophilia Experts in the United States

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