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Frozen shoulder in hyperthyroidism.

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936
BRIEF REPORT
FROZEN SHOULDER IN HYPERTHYROIDISM
JEFFREY R. WOHLGETHAN
A patient who presented with bilateral frozen
shoulders and unrecognized hyperthyroidism is described. Both frozen shoulder and the related shoulderhand syndrome may occur in this setting. These poorly
understood rheumatic conditions often are complications of stroke, spinal cord injury, or diabetes. Dysfunction of the autonomic nervous system is thought to be of
pathogenic importance. It is postulated that the close
resemblance of hyperthyroidism to activation of the
sympathetic nervous system may underlie its association
with frozen shoulder and shoulder-hand syndrome.
Thyroid disease may have rheumatic complications (1). A patient with bilateral frozen shoulders who
had unrecognized hyperthyroidism prompted a review
of this infrequently encountered association.
Case report. The patient, a 62-year-old man,
was referred for evaluation of restricted motion of the
shoulders. Six months earlier he had begun losing
weight, in spite of a good appetite. While moving into
a new house, he noted unusual difficulty lifting boxes
and furniture, and found he was having trouble getting
out of a car. He discovered that he could not raise his
arms beyond 90" and had discomfort over the anterior
shoulder region when reaching backward. His weight,
From the Medical Service, Boston Veterans Administration
Medical Center, and the Arthritis Center of Boston University,
Boston, Massachusetts.
Supported by the Veterans Administration.
Jeffrey R. Wohlgethan, MD: Assistant Professor of Medicine, Boston University School of Medicine, and Chief, Rheumatology Section, Boston Veterans Administration Medical Center.
Address reprint requests to Jeffrey R. Wohlgethan, MD,
Rheumatology, A 10-90, Boston VA Medical Center, 150 S.
Huntington Avenue, Boston, MA 02130.
Submitted for publication August 20, 1986; accepted in
revised form November 18, 1986.
Arthritis and Rheumatism, Vol. 30, No. 8 (August 1987)
which had been 160 pounds, reached a low of 129, and
then rose to 136 pounds. At the time of the visit, he
was feeling well; his only complaints concerned the
failure to regain all of his lost weight, and the restriction of shoulder motion. There was no history of
shoulder problems, and no personal or family history
of diabetes.
An extensive diagnostic evaluation had been
fruitless. On 2 occasions, serum levels of muscle
enzymes were normal. A random serum glucose level
was 101 mgldl and a fasting level was 96 mgldl.
Radiographs of the shoulders, chest, and gastrointestinal tract gave normal results. Nerve conduction
studies and electromyographic study of both upper
extremities disclosed no evidence of neuropathy,
myopathy, or radiculopathy. Muscle biopsy was not
believed to be indicated. Thyroid tests were not done.
At physical examination, he was a calm man
who appeared exceedingly thin, but not ill. The blood
pressure was 120/70 mm Hg, with an irregularly irregular pulse rate of 120 beats per minute. The hands
were steady, and his skin was of normal texture. There
was no exophthalmos, and no lid lag. His thyroid was
not palpable. Both shoulders were limited to 45" of
passive glenohumoral abduction, with virtually no
internal or external rotation, and there was atrophy of
the shoulder girdle musculature (Figure 1). Proximal
strength was diminished in the upper, as well as the
lower, extremities, but was still at or above 4/5in all
muscle groups. The deep tendon reflexes were normal.
The remainder of the examination results were
unremarkable.
An electrocardiogram (EKG), obtained a few
minutes later, showed sinus rhythm at a rate of 90;
937
BRIEF REPORTS
Figure 1. Demonstration of marked limitation of passive glenohumeral abduction (active shoulder abduction was 90”)and
atrophy of the shoulder girdle musculature.
results of a study 3 months earlier had been normal
except for occasional premature atrial contractions.
Thyroid function studies revealed a T4 of 14
pgldl (normal 5-12), a T3 uptake of 44% (normal
35-45), and a T3 of 314 ng/dl (normal 100-200). The
thyroid stimulating hormone (TSH) level was 0.1
pU/ml (normal 0-4).A stimulation test with TSHreleasing hormone (TRH) was performed, and there
was no increase in TSH level 30 minutes following
injection of 500 pg of TRH (normal 2 2 pU/rnl; no
response in hyperthyroidism). During the procedure
there was an episode of atrial fibrillation, documented
by EKG. The 24-hour uptake of I3lI was 60% (normal
5-40); the scan showed that the thyroid gland was of
normal size. Serum antithyroid antibodies were not
detected.
In the interval between the first clinic visit and
the institution of treatment, the patient had several
episodes of palpitations, each lasting about 2 hours.
Two months later he was euthyroid; he had regained
his lost weight, palpitations had abated, and the range
of motion of the shoulders had returned to almost
normal. He had been taught shoulder range of motion
exercises to practice at home, but had received no
other therapy for frozen shoulder, and no medications
except propylthiouracil.
Discussion. In retrospect, several aspects of the
case pointed to hyperthyroidism: alarming weight loss
despite a good appetite, proximal muscle weakness,
and, almost certainly, intermittent atrial fibrillation.
However, the patient’s calm and relaxed manner, the
absence of thyroid enlargement and classic signs of
hyperthyroidism, and the (usually) normal findings on
cardiac examination, all acted to obscure the diagnosis. His shoulder condition was an important diagnostic clue.
In 1928, Duncan described 50 patients who
underwent surgery for hyperthyroidism and who had
“toxic periarthritis” (2). The syndrome was characterized by joint pain and limitation of motion, with no
evidence of effusions, erythema, or warmth. In 80% of
the patients, the condition first involved 1 or both
shoulders, but the knees, metacarpophalangeal joints,
interphalangeal joints, feet, lumbosacral spine, and
hips were sometimes affected. Two patients were
described in detail. Both had marked limitation of
938
shoulder motion, along with involvement of other
joints. There was dramatic relief of pain in almost all
cases within 48-72 hours of thyroid surgery.
In a review of 200 cases of “periarthritis of the
shoulder,” Dickson and Crosby found 18 patients with
hyperthyroidism and 7 with hypothyroidism (3). Few
clinical details were given, but it was noted that relief
of pain followed thyroid surgery within 48-72 hours.
Skillern reported that 20 patients from a series
of 300 patients with Graves’ disease had “periarthritis
of the shoulder”; 4 also had swelling and stiffness of
the h g e r s on the same side and were said to have the
shoulder-hand syndrome (4). Of 300 patients with
toxic adenomas, 10 had a similar shoulder condition,
and 1 had hand involvement as well. With a single
exception, only older patients were afflicted. There
was no response to any form of treatment except that
of control of the hyperthyroidism. Nine patients had
surgery, and all had relief of pain within days. When
drug or radioactive iodine therapies were used, the
pain resolved more gradually. It was noted that in a
control group of subjects with nontoxic goiters, 5
patients with periarthritis of the shoulder had no
response to thyroidectomy.
Oldham described 5 patients with “periarthrosis of the shoulder” and hyperthyroidism, including 1 who had bilateral shoulder-hand syndrome
( 5 ) . In 2 patients, thyroid surgery produced rapid relief
of the shoulder pain, while the response was more
gradual in the 3 patients treated with drugs.
Weiss and colleagues described 3 patients with
acute shoulder pain due to hyperthyroidism, and they
emphasized the lack of response to traditional therapy,
including injections of corticosteroids (6). They speculated that the shoulder pain caused by hyperthyroidism arises from excessive stretching of connective
tissue secondary to the action of thyroid hormone on
the osseous, ligamentous, and muscular components
of the joint.
Chapman and Maloof described 1 patient who
had calcific tendinitis of the shoulder which was
thought to respond to the treatment of hyperthyroidism with radioactive iodine (7).
Care must be taken in interpreting the findings
reported in the earlier literature. A paper on periarthritis scapulohumeralis (8), for example, which lists
hyperthyroidism as an underlying condition in some
c a s e s , includes patients who probably had
polyrnyalgia rheumatica or other illnesses. Dickson
and Crosby’s 7 cases of “periarthritis” associated
with hypothyroidism may represent instances of the
BRIEF REPORTS
arthropathy of hypothyroidism. It is clear, nonetheless, that a disorder closely resembling the idiopathic
frozen shoulder, or sometimes the shoulder-hand syndrome, occurs in hyperthyroidism. Since it appears to
arise primarily after a period of established hyperthyroidism, earlier diagnosis and treatment of thyroid
disease probably accounts for the fact that this complication is now apparently seldom seen.
The temporal association of weakness, weight
loss, and limitation of shoulder motion; the absence of
any other condition predisposing to frozen shoulder;
and the recovery of shoulder motion coincident with
treatment with propylthiouracil strongly suggest that
our patient’s shoulder problem was a complication of
hyperthyroidism. The absence of moderate or severe
pain is unusual, and would be so in frozen shoulder of
any etiology. The relation of pain to loss of motion in
frozen shoulder is not well understood, although studies of the natural history have shown that pain is most
prominent early in the disease course (9,lO).
Thyroid myopathy (1 1) could underlie the limitation of shoulder movement in this case. In spite of a
normal result on electromyography, the patient was
weak, though not to a degree that would interfere with
his usual daily activities, and did have shoulder girdle
muscle atrophy, though this could have been a result,
rather than a cause, of frozen shoulder (9). Yet, while
frozen shoulder is often seen in the hemiplegia that
follows a stroke, there is no documentation that it
occurs as a complication of weakness in primary
muscle disease. Moreover, the many reports of
prompt relief of shoulder pain following thyroid surgery suggest that there is a direct and rapidly reversible effect of thyroid hormone on the tissues at the
shoulder.
The possible relation of frozen shoulder to 2
other musculoskeletal complications of hyperthyroidism, accelerated osteoporosis and thyroid acropachy
( l ) , is intriguing. All 3 are characterized by connective
tissue proliferation (l), involving the articular capsule,
bone, and periosteum, respectively. The clinical picture of thyroid acropachy shares with frozen shoulder
the finding of stiff joints; it is possible that some of
Duncan’s patients with widespread toxic periarthritis
(2) had thyroid acropachy. It is interesting to speculate
that these diverse conditions result from a common
effect of thyroid hormone on connective tissues.
The very earliest literature on the painful stiff
shoulder includes some patients who had pain, tingling, and chilling of the hand on the same side (12).
The frozen shoulder may in fact be a forme fruste of
BRIEF REPORTS
the shoulder-hand syndrome (9). The association of
hyperthyroidism with either lends further support to
the notion that they are related.
Stroke, spinal cord injury, a n d diabetes are
prominent among t h e conditions that predispose to
frozen shoulder (9) a n d t h e shoulder-hand syndrome
(10). Dysfunction of the autonomic nervous system is
thought to be of pathogenic importance. Indeed, a n
accepted term for t h e shoulder-hand syndrome is
reflex sympathetic dystrophy. Successful management
of this condition with drugs and procedures that block
the sympathetic nerves has been reported (10).
It is noteworthy in this regard that hyperthyroidism is similar to activation of t h e sympathetic
nervous system. The basis for the resemblance is not
known, but it is possible that thyroid hormones themselves have actions similar to those of the catecholamines, or that they alter the number o r function of
adrenergic receptors on cells (13). The sympathetic
effects of hyperthyroidism may contribute t o the development of frozen shoulder and reflex sympathetic
dystrophy.
REFERENCES
1 . Bland JH, Frymoyer JW, Newberg AH, Revers R,
Norman RJ: Rheumatic syndromes in endocrine disease. Semin Arthritis Rheum 9:23-65, 1979
2. Duncan WS: The relationship of hyperthyroidism to
joint conditions. JAMA 91: 1779-1782, 1928
939
3. Dickson JA, Crosby EH: Periarthritis of the shoulder:
an analysis of 200 cases. JAMA 99:2252-2257, 1932
4. Skillern PG: The association of periarthritis of the shoulder with hyperthyroidism, Annual Meeting of the American Goiter Association. St. Louis, MO, May 1-3, 1952
5. Oldham BE: Periarthrosis of the shoulder associated
with thyrotoxicosis. NZ Med J 58:76&770, 1959
6. Weiss JJ, Thompson GR, Woodbury D: Hyperthyroidism presenting as acute shoulder pain. Mich Med
72:77 1-774, 1973
7. Chapman EM, Maloof F: Bizarre clinical manifestations
of hyperthyroidism. N Engl J Med 254:l-5, 1956
8. Meulengracht E, Schwartz M: Course and prognosis of
periarthritis humeroscapularis with special regard to
cases with general symptoms. Acta Med Scand 143:
350-360, 1952
9. Rizk TE, Pinals RS: Frozen shoulder. Semin Arthritis
Rheum 11:440452, 1982
10. Kozin F: Painful shoulder and the reflex sympathetic
dystrophy syndrome, Arthritis and Allied Conditions.
Tenth edition. Edited by DJ McCarty. Philadelphia, Lea
& Febiger, 1985, pp 1322-1355
1 1 . Swanson JW, Kelly JJ, McConahey WM: Neurologic
aspects of thyroid dysfunction. Mayo Clin Proc 56:
504512, 1981
12. Duplay S: De la peri-arthrite scapulo-humerale et des
raideurs de l’epaule qui en sont la consequence. Arch
Gen Med 20513-542, 1872
13. Ingbar SH: The thyroid gland, Williams Textbook of
Endocrinology. Seventh edition. Edited by JD Wilson,
DW Foster. Philadelphia, WB Saunders, 1985, pp
682-8 15
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