Hashimoto's Thyroiditis Presenting with Severe Pressure Symptoms --A Case Report-Tsukasa TSUNODA,Nobuo MOCHINAGA,Toshifumi ETO, Masazumi TERADAand Ryoichi TSUCHIYA ABSTRACT: A extremely rare case of Hashimoto's thyroiditis presenting with pressure symptoms is described herein. A 50 year old Japanese woman was referred to our department with swelling of the anterior neck, facial edema and recent heavy snoring. Oto-rhinolaryngological examinations revealed no movement of the bilateral vocal cords, severe laryngeal edema and diffuse edema of the tongue and pharynx. These findings had apparently been induced by compression of the bilateral recurrent nerves and internal jugular veins by an enlarged thyroid gland. The results of thyroid function and autoimmune tests were compatible with a diagnosis of Hashimoto's disease and thus, total thyroidectomy with a tracheostomy was performed uneventfully. The resected specimen weighed 168 grams and was confirmed histologically to be Hashimoto's disease. Following her operation, all the above symptoms disappeared and 4 months later, the patient is well and asymptomatic. KEY WORDS: Hashimoto's thyroiditis, pressure symptoms, recurrent nerve palsy, laryngeal edema
INTRODUCTION
H a s h i m o t o ' s disease, or lymphocytic thyroiditis, has become common and the number of cases may even be increasing since antithyroid antibody examinations are now widely used for its diagnosis. 1,2 Usually, longterm suppressive therapy with the thyroid hormone is recommended for patients with symptomatic goiter or hypothyroidism with surgical intervention being justified only when the goiter shows pressure symptoms, cosmetic problems a n d / o r a suspicion of
The Second Department of Surgery, Nagasaki University School of Medicine, Nagasaki, Japan Reprint requests to: Tsukasa Tsunoda, MD, The Second Department of Surgery, Nagasaki University School of Medicine, 7-1, Sakamoto, Nagasaki 852, Japan
associated malignant lesions. 1-3 There are very few reports of a huge goiter of Hashimoto's thyroiditis being treated surgically because of pressure symptoms. In this paper, we report a case of Hashim0to's d i s e a s e with pressure symptoms, namely, bilateral r e c u r r e n t nerve palsy, severe laryngeal edema and facial edema, which was treated successfully by a total thyroidectomy. A CASE REPORT
A 50 year old Japanese woman was admitted to the Second Department of Surgery, Nagasaki University Hospital on December 5, 1989, complaining of facial swelling, hoarseness and recent heavy snoring, and swelling of t h e anterior neck. The swelling of the anterior neck had first been pointed out by a
JAPANESEJOURNALOF SURGERY,VOL.21, No. 4 pp. 450-453, 1991
Volume 21 Number 4
Hashimoto's thyroiditis
n e i g h b o r 4 years prior to the admission, however, there were no other symptoms at this timel In September, 1989, her voice b e c a m e husky a n d by N o v e m b e r , 1989, general fatigue, hoarseness, facial swelling and heavy snoring during sleep had developed, which prompted h e r to visit a physician, The physician referred her to Nagasaki University Hospital.
Fig. 1. Front view of the neck and face showing a huge goiter and facial edema. Note the edema around the eyes. Table la.
T3 : T~ : TSH : TBG : TGHA:
Physical examination revealed the patient to be in a good nutritional state and have a good appetite. H e r height was 155 cm and weight 76 kg. T h e r e Was no edema o f the trunk or extremities. A hard cartilage-like mass was palpable at the site o f the thyroid gland, the size o f which was 8.5 X 4.0 cm in the right lobe and 7.5 X 3.5 cm in the left lobe. No bruit was audible over the thyroid gland. H e r face was edematous, especially a r o u n d the eyes ~(Fig. 1) and her voice showed a severe husky change with heavy snoring that disturbed the sleep o f other inpatients even o n the opposite side o f the ward. She had inspiratory and expiratory stridor but no dyspnea or a sense of choking. T h e p r e o p e r a t i v e l a b o r a t o r y data are shown in Tables l a and lb. Thyroid function tests revealed low levels o f T 3 and T~, and a high level of TSH, indicating hypothyroidism. The thyroid autoantibodies, namely, antithyroglobulin antibodies and antimicrosomal, antibodies, were elevated and blood chemistry showed abnormally high levels of SGOT, LDH, CPK, aldolase, T I T , ZTT, total
Thyroid Function and Autoimmune Tests
24 ng/dl (80-180) 0.5 ug/dl (6-13) 68.4 uU/ml (0.3-3.2) 22.5 ug/ml (15-28) 5120~
free T3: 0.6 pg/ml (2.3-5.8) free T,: 0.0 ng/dl (0.8-2.2) T~ uptake: 22.8%(25-35) TSH receptor antibody: (+) MCHA: 5120~
( ): normal value, TBG: thyroxine-binding globulin, TGHA: thyroglobuline hemagglutination antibodies, MCHA: microsomal hemagglutination antibodies. Table lb.
SGOT: 98 IU/1 (11-39) SGPT: 31 IU/1 (4-33) Ch-E; 0.59 ApH/H (0.6 1.3) Al-p: 123 IU/1 (88-270) LAP: 60 IU/1 (40-100) y-GTP: 20 IU/1 (0-50) TIT: 20.9 Kunkel (0• ZTT: 29.9 Kunkel (4.0-12.0)
451
Blood Chemistry CPK: 1418 IU/1 (0-52) LDH: 1308IU/1 (202-435) Albumin: 11.0 IU/1 (2.0-7.0) T Chol: 256 mg/dl (111-247) Total protein: 9.3 g/dl (6.2-8.0) Albumin: 51.5%,al-globulin: 2.2% a2-globulin: 5.0%,t-globulin: 6.9% y-globulin: 34.4%
Ch-E: cholinesterase, Al-p: alkaline phosphatase, LAP: leuicine aminopeptidase, y-GTP: y-glutamyltranspeptidase, CPK~ creatine phosphokinase, LDH: lactate dehydrogenase, T Chol: total cholesterol.
452
Tsunoda et al.
Jpn. J.
Fig. 3. ACT scan of the neck demonstrating the enlarged thyroid, deformity of the trachea and dilated right internal jugular vein.
Fig. 2. Lateral X-ray of the neck Showing the expanded retrotracheal space.
cholesterol and y-globulin, all b e i n g compatible with a diagnosis of H a s h i m o t o ' s disease. An analysis of arterial blood gases was done in r o o m air, the result o f which was withifi normal limits. A lateral X-ray o f the neck showed that the retrotracheal space between the posterior wall o f the trachea a n d anterior rim of the cervical vertebrae h a d expanded to 2.8 cm (Fig. 2). A C T scan of the neck revealed the enlarged h o m o g e n e o u s thyroid mass to compress the trachea, a n d that the right internal jugular vein was unusually dilated (Fig. 3). Ukrasonography revealed the bilateral internal jugular veins to be c o m p r e s s e d by a goiter at the caudal level o f the neck and dilated at the cephalad part o f the neck. 6~Ga scintigraphy showed diffuse and scanty accumulations in the area of the thyroid gland. T h e oto-rhinolaryngological findings inc l u d e d severe l a r y n g e a l e d e m a , diffuse e d e m a o f the tongue a n d p h a r y n x a n d no m o v e m e n t of the bilateral vocal cords. U n d e r the diagnosis of H a s h i m o t o ' s disease associated with c o m p r e s s i o n o f the
bilateral recurrent nerves a n d internal jugular veins, a total thyroidectomy was carried out on D e c e m b e r 18, 1989. T h e operation was m o r e difficult t h a n a typical total thyroidectomy because of the e n o r m o u s size a n d hardness of the goiter, and a tracheostorey was also p e r f o r m e d to prevent postoperative respiratory distress. T h e resected thyroid gland weighed 168 grams. T h e administration o f thyroxine (100/2g/day) was started o n the 5th postoperative day. A histological examination of the resected specim e n confirmed the diagnosis o f Hashimoto's disease, which was c o n s i d e r e d to be in an advanced stage because the connective tissues were evenly distributed. Oto-rhinolaryngological examinations on the 8th postoperative day revealed improved m o v e m e n t o f the bilateral vocal cords and the absence of e d e m a except for the arytenoid. T h e tracheostomy tube was removed o n the 17th postoperative day and h e r voice returned to normal. Serum calcium levels were normal, and a m a r k e d i m p r o v e m e n t was seen in all the blood tests. T h e patient was discharged on the 21st postoperative day and has b e e n well since.
Volume 21 Number 4
Hashimoto's thyroiditis
Discussion Hashimoto's thyroiditis is an autoimmune disease which may be associated with varying degrees of thyroid enlargement. It has occasionally been reported that the goiter o f Hashimoto's disease causes mild pressure symptoms such as a sensation of fullness in the neck or o f chokifig ordysphagia, but in general, these symptoms can be alleviated successfully by medical treatment. O u r patient, however, developed symptoms o f pressure on the bilateral recurrent nerves and internal jugular veins: T h e facial and laryngeal edema in this patient was also regarded as a pressure symptom because there was no edema on the trunk or extremities as is usually observed in myxedema. LucarottP reported 3 cases of vocal cord paralysis as a very u n c o m m o n symptom in Hashimoto's disease in which the right recurrent nerve alone was paralyzed by pressure from a diffuse goiter. Shaw ~ reported a patient with venous compression, presenting with superior/vena cava obstruction while L i n d e m ~ r e p o r t e d 41 surgically t r e a t e d Hashimoto's patients, a m o n g whom 21 h a d p r e s s u r e symptoms involving e i t h e r the esophagus, trachea, recurrent nerve or carotid artery. However, we were unable to find a case similar to ours in the literature and therefore consider it to be extremely rare. What type o f thyroidectomy should be employed to relieve the pressure symptoms o f Hashimoto's goiter? In a discussion o f the report by Thomas, Letton r e c o m m e n d e d cutting o f the isthmus and resection o f the anterior portion o f the gland for' patients with mild pressure symptoms? His method may be appropriate for patients complaining o f a pressure sensation without any evidence o f compressio n to the adjacent structures after failure o f medical treatment. Lindem ~ r e c o m m e n d e d near total thyroidectomy by the "off the.trachea" technique, emphasizing that the r e m a i n i n g portion o f the goiter
453
could cause pressure symptoms again. He u s e d the "off the trachea" technique for complete removal of the goiter in the retrotracheal area. This method may be effective for patients with marked compression to the esophagus, trachea a n d / o r internal jugular vein, however, near total thyroidectomy may fail to relieve the compression to the recurrent nerve because the posterior part o f the thyroid adjacent to the nerves is left compressed. Shaw ~ performed total thyroidectomy on a patient with venous compression symptoms without any complications. In our case, total thyroidectomy was employed because a huge goiter occupied even the retrotracheal space and compressed both the recurrent nerves and internal jugular veins. Near total tyroidectomy was not performed due to the possibility that recurrent nerve palsy would persist. Thus, total thyroidectomy seems the most reliable a n d efficient method when pressure symptoms are very severe. Moreover, it can be performed by a trained surgeon without damage to the recurrent nerve or parathyroid glands. 7 (Received for publication on Apr. 20, 1990) REFERENCES 1. Ingbar SH. Hashimoto's disease (Lymphocyticthyroiditis, Struma lymphomatosa) In: Wilson JD, Foster DW, eds. Textbook of Endocrinology.W.B. Saunders Co. 1985; 806-808. 2. Volpe R. Autoimmune thyroiditis, In: Ingbar SH, Bravennan LE, eds. The Thyroid, Philadelphia: J.B. LippincottCo. 1986; 1266-1286. 3. Thomas CGJr, Rutledge RG. Surgical intervention in chronic (Hashimoto's) thyroiditis. Ann Surg 1981; 193: 769-776. 4. Lucarotti ME, Holl-Allen RTJ. Recurrent laryngeal nerve palsy associated with thyroiditis. BrJ Surg 1988; 75: 1041-1042. 5. ShawJFL, Tayler MJ. Superior vena cava obstruction dueto Hashimoto'sthyroiditis. BrJ Clin Pract 1983; 37: 73-74. 6. Lindem MC, Clark JH. Indication for surgery in thyroiditis. AmJ Surg 1969; 118:829-831. 7. Attie JN, Khafif RA. Preservation of parathyroid glands during total thyroidectomy.AmJ Surg 1075; t30: 399-404.
INTRODUCTION
H a s h i m o t o ' s disease, or lymphocytic thyroiditis, has become common and the number of cases may even be increasing since antithyroid antibody examinations are now widely used for its diagnosis. 1,2 Usually, longterm suppressive therapy with the thyroid hormone is recommended for patients with symptomatic goiter or hypothyroidism with surgical intervention being justified only when the goiter shows pressure symptoms, cosmetic problems a n d / o r a suspicion of
The Second Department of Surgery, Nagasaki University School of Medicine, Nagasaki, Japan Reprint requests to: Tsukasa Tsunoda, MD, The Second Department of Surgery, Nagasaki University School of Medicine, 7-1, Sakamoto, Nagasaki 852, Japan
associated malignant lesions. 1-3 There are very few reports of a huge goiter of Hashimoto's thyroiditis being treated surgically because of pressure symptoms. In this paper, we report a case of Hashim0to's d i s e a s e with pressure symptoms, namely, bilateral r e c u r r e n t nerve palsy, severe laryngeal edema and facial edema, which was treated successfully by a total thyroidectomy. A CASE REPORT
A 50 year old Japanese woman was admitted to the Second Department of Surgery, Nagasaki University Hospital on December 5, 1989, complaining of facial swelling, hoarseness and recent heavy snoring, and swelling of t h e anterior neck. The swelling of the anterior neck had first been pointed out by a
JAPANESEJOURNALOF SURGERY,VOL.21, No. 4 pp. 450-453, 1991
Volume 21 Number 4
Hashimoto's thyroiditis
n e i g h b o r 4 years prior to the admission, however, there were no other symptoms at this timel In September, 1989, her voice b e c a m e husky a n d by N o v e m b e r , 1989, general fatigue, hoarseness, facial swelling and heavy snoring during sleep had developed, which prompted h e r to visit a physician, The physician referred her to Nagasaki University Hospital.
Fig. 1. Front view of the neck and face showing a huge goiter and facial edema. Note the edema around the eyes. Table la.
T3 : T~ : TSH : TBG : TGHA:
Physical examination revealed the patient to be in a good nutritional state and have a good appetite. H e r height was 155 cm and weight 76 kg. T h e r e Was no edema o f the trunk or extremities. A hard cartilage-like mass was palpable at the site o f the thyroid gland, the size o f which was 8.5 X 4.0 cm in the right lobe and 7.5 X 3.5 cm in the left lobe. No bruit was audible over the thyroid gland. H e r face was edematous, especially a r o u n d the eyes ~(Fig. 1) and her voice showed a severe husky change with heavy snoring that disturbed the sleep o f other inpatients even o n the opposite side o f the ward. She had inspiratory and expiratory stridor but no dyspnea or a sense of choking. T h e p r e o p e r a t i v e l a b o r a t o r y data are shown in Tables l a and lb. Thyroid function tests revealed low levels o f T 3 and T~, and a high level of TSH, indicating hypothyroidism. The thyroid autoantibodies, namely, antithyroglobulin antibodies and antimicrosomal, antibodies, were elevated and blood chemistry showed abnormally high levels of SGOT, LDH, CPK, aldolase, T I T , ZTT, total
Thyroid Function and Autoimmune Tests
24 ng/dl (80-180) 0.5 ug/dl (6-13) 68.4 uU/ml (0.3-3.2) 22.5 ug/ml (15-28) 5120~
free T3: 0.6 pg/ml (2.3-5.8) free T,: 0.0 ng/dl (0.8-2.2) T~ uptake: 22.8%(25-35) TSH receptor antibody: (+) MCHA: 5120~
( ): normal value, TBG: thyroxine-binding globulin, TGHA: thyroglobuline hemagglutination antibodies, MCHA: microsomal hemagglutination antibodies. Table lb.
SGOT: 98 IU/1 (11-39) SGPT: 31 IU/1 (4-33) Ch-E; 0.59 ApH/H (0.6 1.3) Al-p: 123 IU/1 (88-270) LAP: 60 IU/1 (40-100) y-GTP: 20 IU/1 (0-50) TIT: 20.9 Kunkel (0• ZTT: 29.9 Kunkel (4.0-12.0)
451
Blood Chemistry CPK: 1418 IU/1 (0-52) LDH: 1308IU/1 (202-435) Albumin: 11.0 IU/1 (2.0-7.0) T Chol: 256 mg/dl (111-247) Total protein: 9.3 g/dl (6.2-8.0) Albumin: 51.5%,al-globulin: 2.2% a2-globulin: 5.0%,t-globulin: 6.9% y-globulin: 34.4%
Ch-E: cholinesterase, Al-p: alkaline phosphatase, LAP: leuicine aminopeptidase, y-GTP: y-glutamyltranspeptidase, CPK~ creatine phosphokinase, LDH: lactate dehydrogenase, T Chol: total cholesterol.
452
Tsunoda et al.
Jpn. J.
Fig. 3. ACT scan of the neck demonstrating the enlarged thyroid, deformity of the trachea and dilated right internal jugular vein.
Fig. 2. Lateral X-ray of the neck Showing the expanded retrotracheal space.
cholesterol and y-globulin, all b e i n g compatible with a diagnosis of H a s h i m o t o ' s disease. An analysis of arterial blood gases was done in r o o m air, the result o f which was withifi normal limits. A lateral X-ray o f the neck showed that the retrotracheal space between the posterior wall o f the trachea a n d anterior rim of the cervical vertebrae h a d expanded to 2.8 cm (Fig. 2). A C T scan of the neck revealed the enlarged h o m o g e n e o u s thyroid mass to compress the trachea, a n d that the right internal jugular vein was unusually dilated (Fig. 3). Ukrasonography revealed the bilateral internal jugular veins to be c o m p r e s s e d by a goiter at the caudal level o f the neck and dilated at the cephalad part o f the neck. 6~Ga scintigraphy showed diffuse and scanty accumulations in the area of the thyroid gland. T h e oto-rhinolaryngological findings inc l u d e d severe l a r y n g e a l e d e m a , diffuse e d e m a o f the tongue a n d p h a r y n x a n d no m o v e m e n t of the bilateral vocal cords. U n d e r the diagnosis of H a s h i m o t o ' s disease associated with c o m p r e s s i o n o f the
bilateral recurrent nerves a n d internal jugular veins, a total thyroidectomy was carried out on D e c e m b e r 18, 1989. T h e operation was m o r e difficult t h a n a typical total thyroidectomy because of the e n o r m o u s size a n d hardness of the goiter, and a tracheostorey was also p e r f o r m e d to prevent postoperative respiratory distress. T h e resected thyroid gland weighed 168 grams. T h e administration o f thyroxine (100/2g/day) was started o n the 5th postoperative day. A histological examination of the resected specim e n confirmed the diagnosis o f Hashimoto's disease, which was c o n s i d e r e d to be in an advanced stage because the connective tissues were evenly distributed. Oto-rhinolaryngological examinations on the 8th postoperative day revealed improved m o v e m e n t o f the bilateral vocal cords and the absence of e d e m a except for the arytenoid. T h e tracheostomy tube was removed o n the 17th postoperative day and h e r voice returned to normal. Serum calcium levels were normal, and a m a r k e d i m p r o v e m e n t was seen in all the blood tests. T h e patient was discharged on the 21st postoperative day and has b e e n well since.
Volume 21 Number 4
Hashimoto's thyroiditis
Discussion Hashimoto's thyroiditis is an autoimmune disease which may be associated with varying degrees of thyroid enlargement. It has occasionally been reported that the goiter o f Hashimoto's disease causes mild pressure symptoms such as a sensation of fullness in the neck or o f chokifig ordysphagia, but in general, these symptoms can be alleviated successfully by medical treatment. O u r patient, however, developed symptoms o f pressure on the bilateral recurrent nerves and internal jugular veins: T h e facial and laryngeal edema in this patient was also regarded as a pressure symptom because there was no edema on the trunk or extremities as is usually observed in myxedema. LucarottP reported 3 cases of vocal cord paralysis as a very u n c o m m o n symptom in Hashimoto's disease in which the right recurrent nerve alone was paralyzed by pressure from a diffuse goiter. Shaw ~ reported a patient with venous compression, presenting with superior/vena cava obstruction while L i n d e m ~ r e p o r t e d 41 surgically t r e a t e d Hashimoto's patients, a m o n g whom 21 h a d p r e s s u r e symptoms involving e i t h e r the esophagus, trachea, recurrent nerve or carotid artery. However, we were unable to find a case similar to ours in the literature and therefore consider it to be extremely rare. What type o f thyroidectomy should be employed to relieve the pressure symptoms o f Hashimoto's goiter? In a discussion o f the report by Thomas, Letton r e c o m m e n d e d cutting o f the isthmus and resection o f the anterior portion o f the gland for' patients with mild pressure symptoms? His method may be appropriate for patients complaining o f a pressure sensation without any evidence o f compressio n to the adjacent structures after failure o f medical treatment. Lindem ~ r e c o m m e n d e d near total thyroidectomy by the "off the.trachea" technique, emphasizing that the r e m a i n i n g portion o f the goiter
453
could cause pressure symptoms again. He u s e d the "off the trachea" technique for complete removal of the goiter in the retrotracheal area. This method may be effective for patients with marked compression to the esophagus, trachea a n d / o r internal jugular vein, however, near total thyroidectomy may fail to relieve the compression to the recurrent nerve because the posterior part o f the thyroid adjacent to the nerves is left compressed. Shaw ~ performed total thyroidectomy on a patient with venous compression symptoms without any complications. In our case, total thyroidectomy was employed because a huge goiter occupied even the retrotracheal space and compressed both the recurrent nerves and internal jugular veins. Near total tyroidectomy was not performed due to the possibility that recurrent nerve palsy would persist. Thus, total thyroidectomy seems the most reliable a n d efficient method when pressure symptoms are very severe. Moreover, it can be performed by a trained surgeon without damage to the recurrent nerve or parathyroid glands. 7 (Received for publication on Apr. 20, 1990) REFERENCES 1. Ingbar SH. Hashimoto's disease (Lymphocyticthyroiditis, Struma lymphomatosa) In: Wilson JD, Foster DW, eds. Textbook of Endocrinology.W.B. Saunders Co. 1985; 806-808. 2. Volpe R. Autoimmune thyroiditis, In: Ingbar SH, Bravennan LE, eds. The Thyroid, Philadelphia: J.B. LippincottCo. 1986; 1266-1286. 3. Thomas CGJr, Rutledge RG. Surgical intervention in chronic (Hashimoto's) thyroiditis. Ann Surg 1981; 193: 769-776. 4. Lucarotti ME, Holl-Allen RTJ. Recurrent laryngeal nerve palsy associated with thyroiditis. BrJ Surg 1988; 75: 1041-1042. 5. ShawJFL, Tayler MJ. Superior vena cava obstruction dueto Hashimoto'sthyroiditis. BrJ Clin Pract 1983; 37: 73-74. 6. Lindem MC, Clark JH. Indication for surgery in thyroiditis. AmJ Surg 1969; 118:829-831. 7. Attie JN, Khafif RA. Preservation of parathyroid glands during total thyroidectomy.AmJ Surg 1075; t30: 399-404.
