Successful management of life-threatening spontaneous inferior thyroid artery rupture in neurofibromatosis type 1: a rare case report
Dublin Core
Title
Successful management of life-threatening spontaneous inferior thyroid artery rupture in neurofibromatosis type 1: a rare case report
Subject
Airway, Emergency, Spontaneous artery rupture, Neurofibromatosis type 1, Case report
Description
Abstract
Background Neurofibromatosis type 1 (NF1) is an autosomal dominant disorder associated with cutaneous
and vascular complications. Arterial rupture, including the rare involvement of the subclavian artery, can be life-
threatening. We present a case of a ruptured inferior thyroid artery in a patient with NF1, presenting with a rapidly
expanding neck hematoma.
Case presentation A 35-year-old male with a history of NF1 presented to the emergency department with sudden-
onset right-sided neck swelling and pain. The swelling rapidly expanded, leading to severe dyspnea, irritability, and
hoarseness, necessitating emergent intubation. After failed attempts of intubation, a surgical tracheostomy was
performed. Imaging revealed active arterial extravasation at the inferior thyroid artery near the thyrocervical trunk,
with a large neck hematoma extending into the mediastinum. Coil embolization was performed, resulting in a
favorable outcome. Postoperative follow-up confirmed successful embolization and resolution of symptoms.
Discussion and conclusion NF1 is commonly associated with cutaneous manifestations but can also lead to
vascular complications, including arterial stenosis and aneurysms, due to impaired vascular endothelial and smooth
muscle cell function. Rupture of the inferior thyroid artery in NF1 is extremely rare and can present with symptoms
such as hoarseness, dysphagia, and swelling, complicating initial diagnosis. Management of vascular complications in
NF1 can be challenging due to the fragility of affected vessels. Endovascular interventions, such as coil embolization,
offer a less invasive treatment option with promising outcomes. In this case, rapid airway management followed by
angiographic embolization led to a successful resolution.
Keywords Airway, Emergency, Spontaneous artery rupture, Neurofibromatosis type 1, Case report
Background Neurofibromatosis type 1 (NF1) is an autosomal dominant disorder associated with cutaneous
and vascular complications. Arterial rupture, including the rare involvement of the subclavian artery, can be life-
threatening. We present a case of a ruptured inferior thyroid artery in a patient with NF1, presenting with a rapidly
expanding neck hematoma.
Case presentation A 35-year-old male with a history of NF1 presented to the emergency department with sudden-
onset right-sided neck swelling and pain. The swelling rapidly expanded, leading to severe dyspnea, irritability, and
hoarseness, necessitating emergent intubation. After failed attempts of intubation, a surgical tracheostomy was
performed. Imaging revealed active arterial extravasation at the inferior thyroid artery near the thyrocervical trunk,
with a large neck hematoma extending into the mediastinum. Coil embolization was performed, resulting in a
favorable outcome. Postoperative follow-up confirmed successful embolization and resolution of symptoms.
Discussion and conclusion NF1 is commonly associated with cutaneous manifestations but can also lead to
vascular complications, including arterial stenosis and aneurysms, due to impaired vascular endothelial and smooth
muscle cell function. Rupture of the inferior thyroid artery in NF1 is extremely rare and can present with symptoms
such as hoarseness, dysphagia, and swelling, complicating initial diagnosis. Management of vascular complications in
NF1 can be challenging due to the fragility of affected vessels. Endovascular interventions, such as coil embolization,
offer a less invasive treatment option with promising outcomes. In this case, rapid airway management followed by
angiographic embolization led to a successful resolution.
Keywords Airway, Emergency, Spontaneous artery rupture, Neurofibromatosis type 1, Case report
Creator
Hamza A. Abdul-Hafez1*, Yazan Abudeyak1
, Mahmoud S. Mansour2
, Mohammed A. Barakat3
and
Mohanad A. Abuzahra4
, Mahmoud S. Mansour2
, Mohammed A. Barakat3
and
Mohanad A. Abuzahra4
Source
https://doi.org/10.1186/s12245-025-00854-y
Date
2025
Contributor
Peri Irawan
Format
pdf
Language
english
Type
text
Files
Collection
Citation
Hamza A. Abdul-Hafez1*, Yazan Abudeyak1
, Mahmoud S. Mansour2
, Mohammed A. Barakat3
and
Mohanad A. Abuzahra4, “Successful management of life-threatening spontaneous inferior thyroid artery rupture in neurofibromatosis type 1: a rare case report,” Repository Horizon University Indonesia, accessed April 11, 2026, https://repository.horizon.ac.id/items/show/12724.