Tamoxifen Therapy for Recurrent Mucosal Bleeding in Hereditary Hemorrhagic Telangiectasia

Yung, Stephanie; Bell, Julie-Anne; Brooker, Jim C.

doi:10.14740/jh794

Cited by 3 publications

(2 citation statements)

References 10 publications

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“…Of note, the use of tamoxifen has been supported by a small number of cases. One such case discussed how tamoxifen therapy in HHT patients with mucosal bleeding may decrease the need for blood transfusion (Yung et al, 2021). Another case demonstrated long-term cessation of epistaxis in a postmenopausal woman with the use of tamoxifen therapy (Jameson et al, 2004).…”

Section: Discussionmentioning

confidence: 99%

Hereditary Hemorrhagic Telangiectasia in a Geriatric Patient: A Case of Treatment Resistance

Demirjian¹,

Tou²,

Sugerik³

et al. 2022

FMCR

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Hereditary hemorrhagic telangiectasia (HHT) is a condition known for frequent and severe epistaxis, telangiectasias that form along various surfaces of the body, and potentially life-threatening arteriovenous malformations (AVMs) that may develop in major organ systems. Management of this condition begins with prompt diagnosis, followed by careful screening for systemic complications, and care to minimize morbidity associated with blood loss. We hereby describe a case of treatment-resistant HHT in a geriatric patient who suffers from recurrent and severe epistaxis warranting frequent hospitalizations and blood transfusions. The case underscores the need to recognize HHT early, begin treatment promptly, and closely follow these patients to minimize the risk of hospital readmission.

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Section: Discussionmentioning

confidence: 99%

Hereditary Hemorrhagic Telangiectasia in a Geriatric Patient: A Case of Treatment Resistance

Demirjian¹,

Tou²,

Sugerik³

et al. 2022

FMCR

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“…There is increasing interest in recent literature regarding potential novel therapies for HHT-related bleeding non-responsive to currently recommended treatments. One such example is tamoxifen, a selective estrogen receptor modulator, which has been administered in a transfusion-dependent patient with HHT-related epistaxis and gastrointestinal hemorrhage with favorable outcome and no significant adverse effects [31]. In addition to that, the potential use of the somatostatin analogue octreotide has been investigated in case reports and small series, particularly involving patients suffering from severe gastrointestinal bleeding.…”

mentioning

confidence: 99%

Hereditary Hemorrhagic Telangiectasia and Arterio-Venous Malformations—From Diagnosis to Therapeutic Challenges

Floria

Năfureanu

Iov

et al. 2022

JCM

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Hereditary hemorrhagic telangiectasia is a rare autosomal dominant vascular disease defined by the presence of mucosal and cutaneous telangiectasia and visceral arterio-venous malformations. The latter are abnormal capillary-free direct communications between the pulmonary and systemic circulations with the following consequences: arterial hypoxemia caused by right-to-left shunts; paradoxical embolism with transient ischemic attack or stroke and brain abscess caused by the absence of the normally filtering capillary bed; and hemoptysis or hemothorax due to the rupture of the thin-walled arterio-venous malformations (particularly during pregnancy). It is frequently underdiagnosed, commonly presenting as complications from shunting through arterio-venous malformations: dyspnea, chronic bleeding, or embolism. Arterio-venous malformations are present not only in the lungs, but can also be found in the liver, central nervous system (mainly in the brain), nasal mucosa, or the gastrointestinal tract. The first choice of therapy is embolization of the afferent arteries of the arterio-venous malformations, a minimally invasive procedure with a high efficacy, a low morbidity, and low mortality. Other therapeutic modalities are surgery (resection) or stereotactic radiosurgery (using radiation). Routine screening for arterio-venous malformations is indicated in patients diagnosed with this condition and can prevent severe complications such as acute hemorrhages, brain abscesses, or strokes. Clinicians should provide a long-term follow-up for patients with arterio-venous malformations, in an effort to detect their growth or reperfusion in case of previously treated malformations. In spite of two experts’ consensuses, it still possesses multiple therapeutic challenges for physicians, as several aspects regarding the screening and management of arterio-venous malformations still remain controversial. Multidisciplinary teams are especially useful in complex cases.

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Hereditary hemorrhagic telangiectasia (Osler‑Weber‑Rendu syndrome) - Part II. Pharmacological therapy and international guidelines for the therapy 2020

Adam¹,

Brančíková²,

Romanová³

et al. 2021

Vnitr Lek

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Tamoxifen Therapy for Recurrent Mucosal Bleeding in Hereditary Hemorrhagic Telangiectasia

Cited by 3 publications

References 10 publications

Hereditary Hemorrhagic Telangiectasia in a Geriatric Patient: A Case of Treatment Resistance

Hereditary Hemorrhagic Telangiectasia in a Geriatric Patient: A Case of Treatment Resistance

Hereditary Hemorrhagic Telangiectasia and Arterio-Venous Malformations—From Diagnosis to Therapeutic Challenges

Hereditary hemorrhagic telangiectasia (Osler‑Weber‑Rendu syndrome) - Part II. Pharmacological therapy and international guidelines for the therapy 2020

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