AMER IC AN JOURNAL OF OT OLA RYNGOLOGY– H E A D A N D NE CK M E D ICI N E AN D S U RGE RY X X (2 0 1 5) XXX – XXX
Available online at www.sciencedirect.com
ScienceDirect www.elsevier.com/locate/amjoto
Case Report
Percutaneous ultrasound-guided alcohol ablation of solitary parathyroid adenoma in a patient with primary hyperparathyroidism☆ Ameen Z. Alherabi, FACS, FRCSC a,⁎, Osama A. Marglani, FRCSC a , Mohamed G. Alfiky, MBBS, MRCS (ENT) b , Mohamed M. Raslan, MSc, MD, FRCR c , Bandar Al-Shehri, FRCPC d a
Department of Otolaryngology–Head & Neck Surgery, Umm Al-Qura University, Makkah, Saudi Arabia Department of Otolaryngology–Head & Neck Surgery, King Abdullah Medical City, Makkah, Saudi Arabia c Department of Interventional Radiology, King Abdullah Medical City, Makkah, Saudi Arabia d Department of Endocrinology, International Medical Center, Jeddah, Saudi Arabia b
ARTI CLE I NFO
A BS TRACT
Article history:
Parathyroidectomy is considered the definitive cure for primary hyperparathyroidism due to a
Received 6 March 2015
single parathyroid adenoma, which represents the most common cause of chronic hypercalcemia. However, in few cases, surgery may be technically difficult or risky. We report the use of percutaneous ultrasound-guided alcohol ablation of a parathyroid adenoma as an alternative to surgery in an 88-year-old male patient with significant medical comorbidities. © 2015 Elsevier Inc. All rights reserved.
1.
Introduction
Surgical parathyroidectomy is considered the best curative treatment option for patients with symptomatic primary hyperparathyroidism (PHPT) caused by parathyroid adenoma [1]. Medical therapy is approved by the Food and Drug Administration (FDA) as a treatment option only in secondary hyperparathyroidism, while in PHPT, it offers little hope for long-term eucalcemia. Percutaneous alcohol ablation (PAA) of the parathyroid adenoma may represent an alternative therapeutic approach in a small number of cases of PHPT in which, due to other comorbidities, surgery carries a high risk associated with anesthesia and/or technical difficulties [2].
A few studies have reported the use of PAA for the management of PHPT. We present here the first case of parathyroid adenoma treated by PAA in our institution, with a follow-up period of one year after the ablation.
2.
Case report
An 88-year-old male patient with hypercalcemia was referred to our institute after failure of medical therapy (calcimimetics and vitamin D) to achieve long-term normocalcemia. His hypercalcemia was due to PHPT caused by a single parathyroid adenoma. The patient’s chief complaint was severe musculoskeletal weakness. He had a medical history of myocardial infarction, chronic
☆
Disclosure of Benefits: The authors declare no conflicts of interest and the work was not supported or funded by any pharmaceutical company. ⁎ Corresponding author at: Department of Otolaryngology–Head & Neck Surgery, Umm Al-Qura University, Makkah, Saudi Arabia, and P. O. Box 41405, Jeddah 21521, Saudi Arabia. Tel.: + 966 50 3832 472. E-mail address: [email protected] (A.Z. Alherabi). http://dx.doi.org/10.1016/j.amjoto.2015.04.006 0196-0709/© 2015 Elsevier Inc. All rights reserved.
Please cite this article as: Alherabi AZ, et al, Percutaneous ultrasound-guided alcohol ablation of solitary parathyroid adenoma in a patient with..., Am J Otolaryngol–Head and Neck Med and Surg (2015), http://dx.doi.org/10.1016/j.amjoto.2015.04.006
2
AMER ICA N JOURNAL OF OT OLA RYNGOLOGY– H E A D A N D N E CK M EDI CI N E AN D S U RGE RY X X (2 0 1 5) XXX – XXX
obstructive pulmonary disease, renal impairment, and severe kyphoscoliosis. Laboratory investigations showed very high levels of parathyroid hormone (PTH), up to 1900 pg/mL (normal range 11.1–79.5 pg/mL), and serum calcium was elevated to 11.3 mg/dL (normal range 8.6–10.1 mg/dL) in spite of the use of calcimimetic therapy. Ultrasound (US) examination of the neck revealed a welldefined hypoechoic mass measuring 34 × 21 mm in diameter. The mass involved the posterolateral aspect of the left thyroid lobe, suggesting the presence of a parathyroid adenoma. Parathyroid scintigraphy (sestamibi scan) confirmed the diagnosis of a left inferior parathyroid gland adenoma (Fig. 1). The patient was considered a high-risk patient for anesthesia due to his multiple comorbidities. We suggested that the patient could undergo PAA as an alternative to surgical parathyroidectomy, and he agreed by signing a written informed consent form. An experienced interventional radiologist performed the procedure. After sterilization of the skin, a small amount of local anesthetic was injected into the skin and subcutaneous tissue. A 25-gauge needle attached to a syringe containing 96% ethyl alcohol was carefully inserted into the parathyroid adenoma under real-time US guidance; a total of 6 mL of ethanol was injected into the 4 quadrants of the adenoma while monitoring the diffusion of the alcohol by US (Fig. 2). Ablation was confirmed by a marked decrease in the vascularity of the adenoma on Doppler US (Fig. 3). Another injection was administered after 4 months due to slowly elevated levels of both calcium and PTH. The patient is now normocalcemic, and his PTH level has been stable at around 400 pg/mL for a period of one year of follow-up.
3.
Discussion
A single autonomous parathyroid adenoma is the cause of PHPT in 85%–90% of cases [3], representing the most common cause of chronic hypercalcemia [4]. Currently, surgical parathyroidectomy is the gold standard for the treatment of PHPT
Fig. 1 – Parathyroid scintigraphy (sestamibi) showing intense focally increased uptake inferior to left thyroid lobe keeping with the diagnosis of left inferior parathyroid adenoma.
Fig. 2 – Ultrasonography showing needle (arrow) injection of ethanol into the left inferior parathyroid adenoma.
[4]. However, in some selected cases in which surgery cannot be performed or is contraindicated, other options could be offered to patients to improve their calcium levels. Medical therapy is indicated and approved by the FDA in the management of secondary hyperparathyroidism. However, in a recent study, calcimimetic-based medical therapy was used in the treatment of PHPT, and normalization of serum calcium has been reported to have been achieved in 70.6% of patients [3]. Unfortunately, our patient did not tolerate and was not compliant with long-term calcimimetic therapy, which was considered a failure of medical therapy. Therefore, in selected cases, such as the one presented here, PAA could represent an alternative therapeutic option. PAA was first introduced for the management of parathyroid hyperplasia in the early 1980s, with subsequent reports describing its use only in secondary or tertiary hyperparathyroidism [4]. The Japanese Society for Parathyroid Intervention has published guidelines for PAA therapy of the parathyroid glands in chronic dialysis patients [5]. On the other hand, the role of PAA in the treatment of PHPT has not been well established. In examining the indications for this procedure in patients with PHPT, Herman et al. [6] had highlighted 4 indications: (1) medical comorbidities, (2) patient choice, (3) technically difficult surgery, and (4) partial ablation of the only remaining gland after previous subtotal parathyroidectomy for multiple gland disease [6]. PAA is less expensive, less invasive, requires less extensive preparation, and avoids the usual risks of surgery [7]. Furthermore, it was found that PAA increases neither the risk nor the difficulty of subsequent parathyroidectomy in cases in which the procedure fails to achieve normocalcemia and surgical intervention becomes possible [8]. Few complications have been reported with the procedure, including incomplete necrosis of the adenoma, temporary or rarely permanent recurrent laryngeal nerve injury [6], temporary hypocalcemia, and periglandular fibrosis [7–9]. Recurrence of
Please cite this article as: Alherabi AZ, et al, Percutaneous ultrasound-guided alcohol ablation of solitary parathyroid adenoma in a patient with..., Am J Otolaryngol–Head and Neck Med and Surg (2015), http://dx.doi.org/10.1016/j.amjoto.2015.04.006
AMER IC AN JOURNAL OF OT OLA RYNGOLOGY– H E A D A N D NE CK M E D ICI N E AN D S U RGE RY X X (2 0 1 5) XXX – XXX
3
Fig. 3 – Doppler ultrasonography showing left inferior parathyroid adenoma (arrow). (A) Internal vascularity of the adenoma before percutaneous alcohol ablation (PAA). (B) Marked decrease of vascularity of the adenoma after PAA.
hypercalcemia is common several months to years after ablation with alcohol, which is an indication for regular monitoring of serum calcium levels after the procedure. Subsequent PAA can be safely performed several times to successfully treat recurrence [9].
4.
Conclusions
In conclusion, PAA can represent a useful alternative to surgery in a few selected cases of PHPT caused by a single parathyroid adenoma, when parathyroidectomy cannot be performed. The ability to safely repeat the procedure several times in case of hypercalcemia recurrence overcomes the limitations of PAA in long-term efficacy.
REFERENCES
[1] Bilezikian JP, Khan AA, Potts JT. Guidelines for the management of asymptomatic primary hyperparathyroidism: summary statement from the third international workshop. J Clin Endocrinol Metab 2009;94:335–9.
[2] Farford B, Presutti RJ, Moraghan TJ. Nonsurgical management of primary hyperparathyroidism. Mayo Clin Proc 2007;82:351–5. [3] Keutgen XM, Buitrago D, Filicori F, et al. Calcimimetics versus parathyroidectomy for treatment of primary hyperparathyroidism: retrospective chart analysis of a prospective database. Ann Surg 2012;255:981–5. [4] Cappelli C, Pelizzari G, Pirola I, et al. Modified percutaneous ethanol injection of parathyroid adenoma in primary hyperparathyroidism. Q J Med 2008;101:657–62. [5] Fukagawa M, Kitaoka M, Tominaga Y, et al. Guidelines for percutaneous ethanol injection therapy of the parathyroid glands in chronic dialysis patients. Nephrol Dial Transplant 2003(18 Suppl 3):iii31–3. [6] Harman CR, Grant CS, Hay ID, et al. Indications, technique, and efficacy of alcohol injection of enlarged parathyroid glands in patients with primary hyperparathyroidism. Surgery 1998;124:1011–9. [7] Stratigis S, Stylianou K, Mamalaki E, et al. Percutaneous ethanol injection therapy: a surgery-sparing treatment for primary hyperparathyroidism. Clin Endocrinol (Oxf) 2008;69:542–8. [8] Chen HH, Hsu MT, Wu CJ, et al. Effects of percutaneous ethanol injection therapy on subsequent parathyroidectomy. Am J Surg 2008;196:155–9. [9] Veldman MW, Reading CC, Farrell MA, et al. Percutaneous parathyroid ethanol ablation in patients with multiple endocrine neoplasia type 1. Am J Roentgenol 2008;191:1740–4.
Please cite this article as: Alherabi AZ, et al, Percutaneous ultrasound-guided alcohol ablation of solitary parathyroid adenoma in a patient with..., Am J Otolaryngol–Head and Neck Med and Surg (2015), http://dx.doi.org/10.1016/j.amjoto.2015.04.006
Available online at www.sciencedirect.com
ScienceDirect www.elsevier.com/locate/amjoto
Case Report
Percutaneous ultrasound-guided alcohol ablation of solitary parathyroid adenoma in a patient with primary hyperparathyroidism☆ Ameen Z. Alherabi, FACS, FRCSC a,⁎, Osama A. Marglani, FRCSC a , Mohamed G. Alfiky, MBBS, MRCS (ENT) b , Mohamed M. Raslan, MSc, MD, FRCR c , Bandar Al-Shehri, FRCPC d a
Department of Otolaryngology–Head & Neck Surgery, Umm Al-Qura University, Makkah, Saudi Arabia Department of Otolaryngology–Head & Neck Surgery, King Abdullah Medical City, Makkah, Saudi Arabia c Department of Interventional Radiology, King Abdullah Medical City, Makkah, Saudi Arabia d Department of Endocrinology, International Medical Center, Jeddah, Saudi Arabia b
ARTI CLE I NFO
A BS TRACT
Article history:
Parathyroidectomy is considered the definitive cure for primary hyperparathyroidism due to a
Received 6 March 2015
single parathyroid adenoma, which represents the most common cause of chronic hypercalcemia. However, in few cases, surgery may be technically difficult or risky. We report the use of percutaneous ultrasound-guided alcohol ablation of a parathyroid adenoma as an alternative to surgery in an 88-year-old male patient with significant medical comorbidities. © 2015 Elsevier Inc. All rights reserved.
1.
Introduction
Surgical parathyroidectomy is considered the best curative treatment option for patients with symptomatic primary hyperparathyroidism (PHPT) caused by parathyroid adenoma [1]. Medical therapy is approved by the Food and Drug Administration (FDA) as a treatment option only in secondary hyperparathyroidism, while in PHPT, it offers little hope for long-term eucalcemia. Percutaneous alcohol ablation (PAA) of the parathyroid adenoma may represent an alternative therapeutic approach in a small number of cases of PHPT in which, due to other comorbidities, surgery carries a high risk associated with anesthesia and/or technical difficulties [2].
A few studies have reported the use of PAA for the management of PHPT. We present here the first case of parathyroid adenoma treated by PAA in our institution, with a follow-up period of one year after the ablation.
2.
Case report
An 88-year-old male patient with hypercalcemia was referred to our institute after failure of medical therapy (calcimimetics and vitamin D) to achieve long-term normocalcemia. His hypercalcemia was due to PHPT caused by a single parathyroid adenoma. The patient’s chief complaint was severe musculoskeletal weakness. He had a medical history of myocardial infarction, chronic
☆
Disclosure of Benefits: The authors declare no conflicts of interest and the work was not supported or funded by any pharmaceutical company. ⁎ Corresponding author at: Department of Otolaryngology–Head & Neck Surgery, Umm Al-Qura University, Makkah, Saudi Arabia, and P. O. Box 41405, Jeddah 21521, Saudi Arabia. Tel.: + 966 50 3832 472. E-mail address: [email protected] (A.Z. Alherabi). http://dx.doi.org/10.1016/j.amjoto.2015.04.006 0196-0709/© 2015 Elsevier Inc. All rights reserved.
Please cite this article as: Alherabi AZ, et al, Percutaneous ultrasound-guided alcohol ablation of solitary parathyroid adenoma in a patient with..., Am J Otolaryngol–Head and Neck Med and Surg (2015), http://dx.doi.org/10.1016/j.amjoto.2015.04.006
2
AMER ICA N JOURNAL OF OT OLA RYNGOLOGY– H E A D A N D N E CK M EDI CI N E AN D S U RGE RY X X (2 0 1 5) XXX – XXX
obstructive pulmonary disease, renal impairment, and severe kyphoscoliosis. Laboratory investigations showed very high levels of parathyroid hormone (PTH), up to 1900 pg/mL (normal range 11.1–79.5 pg/mL), and serum calcium was elevated to 11.3 mg/dL (normal range 8.6–10.1 mg/dL) in spite of the use of calcimimetic therapy. Ultrasound (US) examination of the neck revealed a welldefined hypoechoic mass measuring 34 × 21 mm in diameter. The mass involved the posterolateral aspect of the left thyroid lobe, suggesting the presence of a parathyroid adenoma. Parathyroid scintigraphy (sestamibi scan) confirmed the diagnosis of a left inferior parathyroid gland adenoma (Fig. 1). The patient was considered a high-risk patient for anesthesia due to his multiple comorbidities. We suggested that the patient could undergo PAA as an alternative to surgical parathyroidectomy, and he agreed by signing a written informed consent form. An experienced interventional radiologist performed the procedure. After sterilization of the skin, a small amount of local anesthetic was injected into the skin and subcutaneous tissue. A 25-gauge needle attached to a syringe containing 96% ethyl alcohol was carefully inserted into the parathyroid adenoma under real-time US guidance; a total of 6 mL of ethanol was injected into the 4 quadrants of the adenoma while monitoring the diffusion of the alcohol by US (Fig. 2). Ablation was confirmed by a marked decrease in the vascularity of the adenoma on Doppler US (Fig. 3). Another injection was administered after 4 months due to slowly elevated levels of both calcium and PTH. The patient is now normocalcemic, and his PTH level has been stable at around 400 pg/mL for a period of one year of follow-up.
3.
Discussion
A single autonomous parathyroid adenoma is the cause of PHPT in 85%–90% of cases [3], representing the most common cause of chronic hypercalcemia [4]. Currently, surgical parathyroidectomy is the gold standard for the treatment of PHPT
Fig. 1 – Parathyroid scintigraphy (sestamibi) showing intense focally increased uptake inferior to left thyroid lobe keeping with the diagnosis of left inferior parathyroid adenoma.
Fig. 2 – Ultrasonography showing needle (arrow) injection of ethanol into the left inferior parathyroid adenoma.
[4]. However, in some selected cases in which surgery cannot be performed or is contraindicated, other options could be offered to patients to improve their calcium levels. Medical therapy is indicated and approved by the FDA in the management of secondary hyperparathyroidism. However, in a recent study, calcimimetic-based medical therapy was used in the treatment of PHPT, and normalization of serum calcium has been reported to have been achieved in 70.6% of patients [3]. Unfortunately, our patient did not tolerate and was not compliant with long-term calcimimetic therapy, which was considered a failure of medical therapy. Therefore, in selected cases, such as the one presented here, PAA could represent an alternative therapeutic option. PAA was first introduced for the management of parathyroid hyperplasia in the early 1980s, with subsequent reports describing its use only in secondary or tertiary hyperparathyroidism [4]. The Japanese Society for Parathyroid Intervention has published guidelines for PAA therapy of the parathyroid glands in chronic dialysis patients [5]. On the other hand, the role of PAA in the treatment of PHPT has not been well established. In examining the indications for this procedure in patients with PHPT, Herman et al. [6] had highlighted 4 indications: (1) medical comorbidities, (2) patient choice, (3) technically difficult surgery, and (4) partial ablation of the only remaining gland after previous subtotal parathyroidectomy for multiple gland disease [6]. PAA is less expensive, less invasive, requires less extensive preparation, and avoids the usual risks of surgery [7]. Furthermore, it was found that PAA increases neither the risk nor the difficulty of subsequent parathyroidectomy in cases in which the procedure fails to achieve normocalcemia and surgical intervention becomes possible [8]. Few complications have been reported with the procedure, including incomplete necrosis of the adenoma, temporary or rarely permanent recurrent laryngeal nerve injury [6], temporary hypocalcemia, and periglandular fibrosis [7–9]. Recurrence of
Please cite this article as: Alherabi AZ, et al, Percutaneous ultrasound-guided alcohol ablation of solitary parathyroid adenoma in a patient with..., Am J Otolaryngol–Head and Neck Med and Surg (2015), http://dx.doi.org/10.1016/j.amjoto.2015.04.006
AMER IC AN JOURNAL OF OT OLA RYNGOLOGY– H E A D A N D NE CK M E D ICI N E AN D S U RGE RY X X (2 0 1 5) XXX – XXX
3
Fig. 3 – Doppler ultrasonography showing left inferior parathyroid adenoma (arrow). (A) Internal vascularity of the adenoma before percutaneous alcohol ablation (PAA). (B) Marked decrease of vascularity of the adenoma after PAA.
hypercalcemia is common several months to years after ablation with alcohol, which is an indication for regular monitoring of serum calcium levels after the procedure. Subsequent PAA can be safely performed several times to successfully treat recurrence [9].
4.
Conclusions
In conclusion, PAA can represent a useful alternative to surgery in a few selected cases of PHPT caused by a single parathyroid adenoma, when parathyroidectomy cannot be performed. The ability to safely repeat the procedure several times in case of hypercalcemia recurrence overcomes the limitations of PAA in long-term efficacy.
REFERENCES
[1] Bilezikian JP, Khan AA, Potts JT. Guidelines for the management of asymptomatic primary hyperparathyroidism: summary statement from the third international workshop. J Clin Endocrinol Metab 2009;94:335–9.
[2] Farford B, Presutti RJ, Moraghan TJ. Nonsurgical management of primary hyperparathyroidism. Mayo Clin Proc 2007;82:351–5. [3] Keutgen XM, Buitrago D, Filicori F, et al. Calcimimetics versus parathyroidectomy for treatment of primary hyperparathyroidism: retrospective chart analysis of a prospective database. Ann Surg 2012;255:981–5. [4] Cappelli C, Pelizzari G, Pirola I, et al. Modified percutaneous ethanol injection of parathyroid adenoma in primary hyperparathyroidism. Q J Med 2008;101:657–62. [5] Fukagawa M, Kitaoka M, Tominaga Y, et al. Guidelines for percutaneous ethanol injection therapy of the parathyroid glands in chronic dialysis patients. Nephrol Dial Transplant 2003(18 Suppl 3):iii31–3. [6] Harman CR, Grant CS, Hay ID, et al. Indications, technique, and efficacy of alcohol injection of enlarged parathyroid glands in patients with primary hyperparathyroidism. Surgery 1998;124:1011–9. [7] Stratigis S, Stylianou K, Mamalaki E, et al. Percutaneous ethanol injection therapy: a surgery-sparing treatment for primary hyperparathyroidism. Clin Endocrinol (Oxf) 2008;69:542–8. [8] Chen HH, Hsu MT, Wu CJ, et al. Effects of percutaneous ethanol injection therapy on subsequent parathyroidectomy. Am J Surg 2008;196:155–9. [9] Veldman MW, Reading CC, Farrell MA, et al. Percutaneous parathyroid ethanol ablation in patients with multiple endocrine neoplasia type 1. Am J Roentgenol 2008;191:1740–4.
Please cite this article as: Alherabi AZ, et al, Percutaneous ultrasound-guided alcohol ablation of solitary parathyroid adenoma in a patient with..., Am J Otolaryngol–Head and Neck Med and Surg (2015), http://dx.doi.org/10.1016/j.amjoto.2015.04.006
