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Rupture of inferior thyroid artery aneurysm
Rupture of inferior thyroid artery ancurysm S a n d r a L. Beal, M . D . , A r t h u r B. D u b l i n , M . D . , * and W i l l i a m K. Stone, M . D . ,
Sacramento, Calif. We report a case in which rupture of an aneurysm o f the inferior thyroid artery caused respiratory arrest. The aneurysm was embolized successfully by an angiographic technique. Published reports o f rupture o f thyroid artery aneurysms are reviewed. (J V a s e SURG 1987;6:194-6.) Spontaneous rupture o f an inferior thyroid artery aneurysm is a rare and potentially lethal event. Only six cases o f thyroid artery aneurysms have been reported: five in the inferior thyroid artery and one in the superior thyroid artery. 1-6All patients initially had neck swelling and pain; in some cases respiratory arrest and death occurred. W e describe the seventh case o f spontaneous rupture o f a thyroid artery aneurysm, which is the sixth case o f inferior thyroid artery aneurysm rupture. CASE REPORT
A 78-year-old white man with chronic obstructive pulmonary disease and atherosclerosis was seen at the Kaiser Permanente Medical Center in Sacramento, California, for an asymptomatic left carotid bruit and a 10 cm abdominal aortic aneurysm. Selective carotid and abdominal angiography showed 90% stenosis of the left internal carotid artery and an infrarenal aortic aneurysm. Left carotid endarterectomy was performed with no complications; 21 days later, the patient returned for aneurysmorrhaphy. The aneurysm was replaced with an aortoiliac Dacron graft. Recovery was uneventful initially; however, on the second postoperative day, ~i severe "tearing" pain suddenly developed low in the left side of the neck, and progressive shortness of breath necessitated intubation and ventilatory support. Physical examination showed a left-sided, nonpulsatile neck swelling slightly above the sternoclavicular joint and spreading anteriorly over the chest. Tracheal deviation to the right was also noted. The hematocrit decreased by 9 vol%. The patient was taken to the angiography suite, where an 8 mm aneurysm of the left inferior thyroid artery was seen in a mass effect compressing the trachea and mediastinal structures (Figs. 1 and 2). Because the vital
From the Departments of Surge~ and Radiology, Universi~ of California Davis Medical Center, Sacramento; and the Department of Surgery, Kaiser Permanente Medical Center, Sacramento. Reprint requests: William Kenneth Stone, M.D., Department of Surgery, Kaiser Permanente Medical Center, 2025 Morse Ave., Sacramento, CA 95825. *Current address: Diagnostic Radiological Imaging Medical Group, 79 Scripps Dr., Sacramento, CA 95825. 194
Fig. 1. Arch angiogram shows aneurysm (arrow) in mass effect. I = innominate artery; 2 = left common carotid artery; 3 = left subclavian artery; 4 = aortic arch.
signs were stable and it was believed that a direct operative approach would be difficult, the patient was transferred to the radiology department at the University of California Davis Medical Center for embolization under fluoroscopic control. The procedure was performed according to the Institutional Review Board-approved protocol, according to Food and Drug Administration (FDA) Title 21, §50.20, and under FDA guidelines for experimental devices (§G820155/$2). During the embolization procedure, digital subtraction angiography (Fig. 3) was used to confirm the position of the catheter after its placement in the artelT. A rapidly
Volume 6 Number 2 August 1987
Aneurysm rupture of inferior thyroid artery 195
Fig. 2. Selective left subclavian view on angiogram shows aneurysm of inferior thyroid artery (arrow). Note tracheal deviation to right. I = intravenous line placed in right jugular vein; 2 = endotracheal tube; 3 = left subclavian artery; 4 = left vertebral artery.
polymerizing glue, isobu~Tl 2-cyanoacrylate (Bucrylate, Ethicon, Inc., Somerville, N.J.), was mixed with 0.3 ml of iophendylate (Pantopaque, Alcon (Puerto Rico) Inc., Humacao, Puerto Rico, used here under a special FDA license), and injected into the aneurysm. Fluoroscopy with manual dye injections confirmed that the aneurysm was thrombosed. The patient returned to Kaiser Permanente Medical Center, Sacramento. During the next three days, the swelling progressively decreased. Follow-up chest x-ray films confirmed the presence of the dye-filled glue in the aneurysm and in the left inferior thyroid artery with resolution of the tracheal deLtion and mass effect. Angiography was not repeated. The patient was extubated and continued to do well at 2-year follow-up.
METHODS Because we had never seen this entity before, a literature search was done by the University o f California Davis Medical Library staff with the Medical Literature Analysis and Retrieval System (MEDLARS) database. The search extended back to 1966 and included all languages. In addition, the bibliographies o f the respective articles were searched for earlier references.
DISCUSSION Our case o f rupture o f a thyroid artery aneurysm is consistent with previously published reports (Table I). Only one patient had a long-standing mass1;
Fig. 3. Intra-arterial digital subtraction angiogram (taken before glue injection) shows aneurysm.
the others h a d a n acutc prescntation. 2-6 Two o f thc six patients had dissecting ancurysms3,5; the others had athcrosclcrosis 1,2,4,6 mad included one patient in whom rupture was related to trauma. 6 Iatrogenic trauma may be possible in some such cases. The type o f aneurysm in our patient is unknown. Rupture was commonly associated with respiratory distress, 3-s
~'ournalof VASCULAR SURGERY
Beal, Dublin, and Stone
196
Table I. Reported cases of rupture of inferior thyroid artery aneurysm Reference
Age (yr)
Sex
Thyroid artery affected
Biglioli et al.1
74
F
Right
Dottmanian et al?
60
M
Right
Golby & Kaya
65
M
Left
Habib4
67
F
Right
Lin~
80
F
Right
Martin et al2
43
F
Left
Present case
67
M
Left
Symptoms
Etiology
Treatment
Aneurysm rupture; respiratory arrest Neck mass; vocal cord paralysis Aneu~sm rupture; resDratory arrest Aneurysm rupture; vocal cord paralysis Aneu~sm rupture; respiratory arrest Aneurysm rupture after trauma Aneu~sm rupture; resptratory arrest
Probably atherosclerosis Atherosclerosis
Aneurysmectomy
Death
Aneurysmectomy
Survival
Dissection
Aneurysmectomy
Survival
Atherosderosis
Tracheostomy
Death
Dissection
None
Death
Atherosclerosis
Aneurysmectomy
Survival
Unknown
Embolization
Survival
which led to dcath in two of the three patients with this condition. Iatrogenic injury was improbable in our patient: because of a high bifurcation, the carotid endarterectomy was done high in the left side of the neck well above the level of the thyroid gland. Preoperative selective carotid angiography was done with the femoral approach; arch or subclavian injections were not performed. Monitoring of central venous pressure was done without difficulty via the right internal jugular approach, and no evidence of pre- or postoperative hypertension existed to suggest dissection. Venipuncture was not attempted on the left side of the neck because of the paticnt's recent carotid operation. Direct surgical approach to the aneurysm was considered; however, because of the age of the patient, moderate chronic obstructive pulmonary disease, risk to the phrenic nerves with operation, and clinical and angiographic evidence that the ancurysm was no longer leaking, we decided to embolize the aneurysm to prevent further bleeding. In retrospect,
Outcome
after review of the literaturc mad of the prcvious r~ suits of surgical intervention, this seems to have been a reasonable choice, although the patient probably would have done well with a direct surgical approach. Embolization is a technique that will have wider application as more experience is gained. REFERENCES
1. Biglioli P, Arena V, Malan E. A case of aneurysm of superior thyroid artery. J Cardiovasc Surg (Torino) 1977;18:539-41. 2. Doumanian AV, Soule EH, Ellis FH Jr. Ruptured aneurysm of the inferior thyroid artery associated with paralysis of the vocal cord: report of case. Ptoc Staff Meetings Mayo Clin 1959;34:303-9. 3. Golby MGS, Kay JM. Primary dissecting aneurysm of the inferior thyroid artery. Br J Surg 1965;52:389-91. 4. Habib MA. Fatal hemorrhage due to ruptured inferior thyroid artery aneurysm. J Laryngol Otot 1977;91:437-40. 5. Lin C-S. Spontaneous cervical hemorrhage due to ruptured dissecting aneurysm of thyroid artery: a case report and review of literature. Mt Sinai J Med (NY) 1978;45:179-83. 6. Martin H, Rebatm JP, Quincy R, Boulud B. Apropos d'un cas d'hdmatome cervical du ~ la rupture d'an&rysmes de la thyro/dienne inf&ieure gauche associ& ~ une arteria-lusoria. J Fr Otorhinolaryngol 1974;23:259-62.
Sacramento, Calif. We report a case in which rupture of an aneurysm o f the inferior thyroid artery caused respiratory arrest. The aneurysm was embolized successfully by an angiographic technique. Published reports o f rupture o f thyroid artery aneurysms are reviewed. (J V a s e SURG 1987;6:194-6.) Spontaneous rupture o f an inferior thyroid artery aneurysm is a rare and potentially lethal event. Only six cases o f thyroid artery aneurysms have been reported: five in the inferior thyroid artery and one in the superior thyroid artery. 1-6All patients initially had neck swelling and pain; in some cases respiratory arrest and death occurred. W e describe the seventh case o f spontaneous rupture o f a thyroid artery aneurysm, which is the sixth case o f inferior thyroid artery aneurysm rupture. CASE REPORT
A 78-year-old white man with chronic obstructive pulmonary disease and atherosclerosis was seen at the Kaiser Permanente Medical Center in Sacramento, California, for an asymptomatic left carotid bruit and a 10 cm abdominal aortic aneurysm. Selective carotid and abdominal angiography showed 90% stenosis of the left internal carotid artery and an infrarenal aortic aneurysm. Left carotid endarterectomy was performed with no complications; 21 days later, the patient returned for aneurysmorrhaphy. The aneurysm was replaced with an aortoiliac Dacron graft. Recovery was uneventful initially; however, on the second postoperative day, ~i severe "tearing" pain suddenly developed low in the left side of the neck, and progressive shortness of breath necessitated intubation and ventilatory support. Physical examination showed a left-sided, nonpulsatile neck swelling slightly above the sternoclavicular joint and spreading anteriorly over the chest. Tracheal deviation to the right was also noted. The hematocrit decreased by 9 vol%. The patient was taken to the angiography suite, where an 8 mm aneurysm of the left inferior thyroid artery was seen in a mass effect compressing the trachea and mediastinal structures (Figs. 1 and 2). Because the vital
From the Departments of Surge~ and Radiology, Universi~ of California Davis Medical Center, Sacramento; and the Department of Surgery, Kaiser Permanente Medical Center, Sacramento. Reprint requests: William Kenneth Stone, M.D., Department of Surgery, Kaiser Permanente Medical Center, 2025 Morse Ave., Sacramento, CA 95825. *Current address: Diagnostic Radiological Imaging Medical Group, 79 Scripps Dr., Sacramento, CA 95825. 194
Fig. 1. Arch angiogram shows aneurysm (arrow) in mass effect. I = innominate artery; 2 = left common carotid artery; 3 = left subclavian artery; 4 = aortic arch.
signs were stable and it was believed that a direct operative approach would be difficult, the patient was transferred to the radiology department at the University of California Davis Medical Center for embolization under fluoroscopic control. The procedure was performed according to the Institutional Review Board-approved protocol, according to Food and Drug Administration (FDA) Title 21, §50.20, and under FDA guidelines for experimental devices (§G820155/$2). During the embolization procedure, digital subtraction angiography (Fig. 3) was used to confirm the position of the catheter after its placement in the artelT. A rapidly
Volume 6 Number 2 August 1987
Aneurysm rupture of inferior thyroid artery 195
Fig. 2. Selective left subclavian view on angiogram shows aneurysm of inferior thyroid artery (arrow). Note tracheal deviation to right. I = intravenous line placed in right jugular vein; 2 = endotracheal tube; 3 = left subclavian artery; 4 = left vertebral artery.
polymerizing glue, isobu~Tl 2-cyanoacrylate (Bucrylate, Ethicon, Inc., Somerville, N.J.), was mixed with 0.3 ml of iophendylate (Pantopaque, Alcon (Puerto Rico) Inc., Humacao, Puerto Rico, used here under a special FDA license), and injected into the aneurysm. Fluoroscopy with manual dye injections confirmed that the aneurysm was thrombosed. The patient returned to Kaiser Permanente Medical Center, Sacramento. During the next three days, the swelling progressively decreased. Follow-up chest x-ray films confirmed the presence of the dye-filled glue in the aneurysm and in the left inferior thyroid artery with resolution of the tracheal deLtion and mass effect. Angiography was not repeated. The patient was extubated and continued to do well at 2-year follow-up.
METHODS Because we had never seen this entity before, a literature search was done by the University o f California Davis Medical Library staff with the Medical Literature Analysis and Retrieval System (MEDLARS) database. The search extended back to 1966 and included all languages. In addition, the bibliographies o f the respective articles were searched for earlier references.
DISCUSSION Our case o f rupture o f a thyroid artery aneurysm is consistent with previously published reports (Table I). Only one patient had a long-standing mass1;
Fig. 3. Intra-arterial digital subtraction angiogram (taken before glue injection) shows aneurysm.
the others h a d a n acutc prescntation. 2-6 Two o f thc six patients had dissecting ancurysms3,5; the others had athcrosclcrosis 1,2,4,6 mad included one patient in whom rupture was related to trauma. 6 Iatrogenic trauma may be possible in some such cases. The type o f aneurysm in our patient is unknown. Rupture was commonly associated with respiratory distress, 3-s
~'ournalof VASCULAR SURGERY
Beal, Dublin, and Stone
196
Table I. Reported cases of rupture of inferior thyroid artery aneurysm Reference
Age (yr)
Sex
Thyroid artery affected
Biglioli et al.1
74
F
Right
Dottmanian et al?
60
M
Right
Golby & Kaya
65
M
Left
Habib4
67
F
Right
Lin~
80
F
Right
Martin et al2
43
F
Left
Present case
67
M
Left
Symptoms
Etiology
Treatment
Aneurysm rupture; respiratory arrest Neck mass; vocal cord paralysis Aneu~sm rupture; resDratory arrest Aneurysm rupture; vocal cord paralysis Aneu~sm rupture; respiratory arrest Aneurysm rupture after trauma Aneu~sm rupture; resptratory arrest
Probably atherosclerosis Atherosclerosis
Aneurysmectomy
Death
Aneurysmectomy
Survival
Dissection
Aneurysmectomy
Survival
Atherosderosis
Tracheostomy
Death
Dissection
None
Death
Atherosclerosis
Aneurysmectomy
Survival
Unknown
Embolization
Survival
which led to dcath in two of the three patients with this condition. Iatrogenic injury was improbable in our patient: because of a high bifurcation, the carotid endarterectomy was done high in the left side of the neck well above the level of the thyroid gland. Preoperative selective carotid angiography was done with the femoral approach; arch or subclavian injections were not performed. Monitoring of central venous pressure was done without difficulty via the right internal jugular approach, and no evidence of pre- or postoperative hypertension existed to suggest dissection. Venipuncture was not attempted on the left side of the neck because of the paticnt's recent carotid operation. Direct surgical approach to the aneurysm was considered; however, because of the age of the patient, moderate chronic obstructive pulmonary disease, risk to the phrenic nerves with operation, and clinical and angiographic evidence that the ancurysm was no longer leaking, we decided to embolize the aneurysm to prevent further bleeding. In retrospect,
Outcome
after review of the literaturc mad of the prcvious r~ suits of surgical intervention, this seems to have been a reasonable choice, although the patient probably would have done well with a direct surgical approach. Embolization is a technique that will have wider application as more experience is gained. REFERENCES
1. Biglioli P, Arena V, Malan E. A case of aneurysm of superior thyroid artery. J Cardiovasc Surg (Torino) 1977;18:539-41. 2. Doumanian AV, Soule EH, Ellis FH Jr. Ruptured aneurysm of the inferior thyroid artery associated with paralysis of the vocal cord: report of case. Ptoc Staff Meetings Mayo Clin 1959;34:303-9. 3. Golby MGS, Kay JM. Primary dissecting aneurysm of the inferior thyroid artery. Br J Surg 1965;52:389-91. 4. Habib MA. Fatal hemorrhage due to ruptured inferior thyroid artery aneurysm. J Laryngol Otot 1977;91:437-40. 5. Lin C-S. Spontaneous cervical hemorrhage due to ruptured dissecting aneurysm of thyroid artery: a case report and review of literature. Mt Sinai J Med (NY) 1978;45:179-83. 6. Martin H, Rebatm JP, Quincy R, Boulud B. Apropos d'un cas d'hdmatome cervical du ~ la rupture d'an&rysmes de la thyro/dienne inf&ieure gauche associ& ~ une arteria-lusoria. J Fr Otorhinolaryngol 1974;23:259-62.