Graves Disease With Only Unilateral Invo 2024 International Journal of Surg

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International Journal of Surgery Case Reports 114 (2024) 109138

Contents lists available at ScienceDirect

International Journal of Surgery Case Reports


journal homepage: www.elsevier.com/locate/ijscr

Case report

Graves' disease with only unilateral involvement; a case report


Mohammed Saad Bu Bshait *
Department of Surgery, College of Medicine, King Faisal University, Saudi Arabia

A R T I C L E I N F O A B S T R A C T

Keywords: Introduction: Graves' disease characteristically presents with a diffuse goiter secondary to the autoantibodies that
Hyperthyroidism target the thyrotropin receptors of the thyroid gland. Few cases have been reported of only one of the two lobes
Unilateral Graves' disease being affected. The cause of this phenomenon is still uncertain. Here we report on another case of unilateral
Case report
Graves' disease.
Case presentation: A 43-year-old female patient presented with a history of weight loss, palpitations and right
sided neck swelling for 4 months. Clinical examination showed an enlarged right thyroid lobe. Laboratory in­
vestigations yielded evidence of thyrotoxicosis with suppressed thyroid stimulating hormone. In addition, anti-
TSH receptor and anti-thyroperoxidase antibodies were positive. Neck Ultrasound showed an enlarged right
thyroid lobe with increased vascularization. The isthmus and left lobe were both normal in size. A Tc99m per­
technetate thyroid scan demonstrated enlargement of the right thyroid lobe with diffuse intense uptake, whereas
the left lobe was suppressed. A diagnosis of unilateral Graves' disease was made. The thyrotoxicosis was treated
and maintained with methimazole.
Discussion: Unilateral Graves' disease is a rare manifestation of Graves' disease, sharing the same autoimmune
background and the symptoms of thyrotoxicosis. Enlargement of only one lobe was evident on clinical exami­
nation. The distinctive feature was unilateral uptake during thyroid scintigraphy. The exact pathophysiology of
this condition has yet to be elucidated. Management options and responses are similar to those of classical
Graves' disease.
Conclusion: Unilateral uptake during thyroid scintigraphy and/or unilateral lobar goiter in the setting of hy­
perthyroidism can be the presentation of unilateral Graves' disease.

1. Introduction 2. Case presentation

Graves' disease is an autoimmune condition in which the thyrotropin


receptor is stimulated by circulating autoantibodies, leading to excessive
release of thyroid hormones. The homogeneous exposure of the gland to
these circulating antibodies characteristically affects it in a diffuse 2.1.1. Patient information
manner. This broad involvement is evident as diffuse goiter, with A 43-year-old female patient presented with a history of weight loss
sonographic hypervascularity, and universally intense uptake on scin­ despite an increase in appetite, palpitations and a right sided neck
tigraphy [1]. In contrast, Graves' disease with unilateral involvement in swelling of four months' duration. The patient denied compression
a bilobed gland has been reported only occasionally. Here we describe symptoms or a family history of thyroid cancer.
another rare case of unilateral Graves' disease. This work has been re­
ported in accordance with the SCARE criteria [2]. 2.1.2. Clinical findings
There was enlargement of the right thyroid that moved with deglu­
tition. There was no retrosternal extension nor lymph node enlargement.
The patient had no exophthalmos or pretibial myxedema.

* King Faisal University, College of Medicine, Alahsa 36377, Saudi Arabia.


E-mail address: mbubshait@kfu.edu.sa.

https://doi.org/10.1016/j.ijscr.2023.109138
Received 24 October 2023; Received in revised form 21 November 2023; Accepted 1 December 2023
Available online 9 December 2023
2210-2612/© 2023 The Author. Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd. This is an open access article under the CC BY license
(http://creativecommons.org/licenses/by/4.0/).
M.S. Bu Bshait International Journal of Surgery Case Reports 114 (2024) 109138

3. Diagnostic assessment 3.2. Therapeutic intervention

The patient was started on antithyroid medication (Methimazole) to


control thyrotoxicosis.

3.1.1. # Thyroid function test 3.3. Follow-up and outcome


T3 and T4 were elevated with suppressed TSH. T3 was 17.1 pmol/L
(Reference range: 3–6.9), T4 was 37.5 pmol/L (Reference range: 12–22), Thyrotoxicosis regressed and patient stayed in normal status for the
and TSH was <0.005 uIU/ml (Reference range: 0.35–4.9). next four months. Based on the patient preference, she was referred to
Anti-TSH receptor (TRAb) and anti-thyroperoxidase (TPO-Ab) anti­ surgery clinic for considering the option of surgical management.
bodies were positive. However, surgical intervention has not yet been carried out due to pa­
tient hesitancy.
3.1.2. # Neck ultrasonography
This showed an enlarged right thyroid lobe with a normal isthmus 4. Discussion
and left lobe. Both lobes were hypervascular but this was more evident
on the right. (Fig. 1a–b). Unilateral involvement of thyroid lobes is a rare presentation of
Graves' disease. Skata and colleagues reported the first two cases of
3.1.3. # Thyroid scintigraphy unilateral Graves' disease [3]. The first case presented with right nodular
A Tc99m pertechnetate thyroid scan showed diffuse hyperemia and goiter and euthyroid status but with positive antimicrosomal antibodies.
enlargement of the right thyroid lobe. Uptake was homogeneously dense Thyroid ultrasound revealed an enlarged right thyroid lobe with a
with a smooth outline. In contrast, the left thyroid lobe was normal in hypoechoic nodule. Thyroid scintigraphy with Tc99m showed intensive
size with homogeneous radiotracer suppression. Total thyroid uptake uptake in the right lobe. The patient underwent right hemi­
was 8.4 %. Right and left lobe uptake was 7.1 % and 1.3 % respectively. thyroidectomy on suspicion of malignancy. Surprisingly, histopatho­
(Fig. 2). logical evaluation was concordant with Graves' disease. Subsequently,
the patient presented after 27 months with symptoms of thyrotoxicosis,
an enlarged contralateral lobe, elevated thyroid function tests, low TSH
and positive TRAb which had been negative preoperatively. A thyroid

Fig. 1. a. Neck ultrasound showing enlarged right thyroid lobe.


b. Color doppler neck ultrasound demonstrating the greater hypervascularity of right thyroid lobe.

2
M.S. Bu Bshait International Journal of Surgery Case Reports 114 (2024) 109138

Fig. 2. Tc99m pertechnetate thyroid scan demonstrated enlarged right thyroid lobe with diffuse homogeneous dense uptake. Left thyroid expressed radiotracer
suppression. Total thyroid uptake was 8.4 %. Right and left lobe uptake were 7.1 % and 1.3 % respectively.

scintigram with 123I-uptake showed an increase in uptake in the bilobed isolated lymphatic drainage) or by acquired conditions (such as
remaining left thyroid lobe. The second case was a 61-year-old female previous bacterial or viral thyroid inflammation) [11]. Impaired
patient who presented with weight loss, palpitations and elevated thy­ thyroidal radioiodine uptake as a result of local suppression of sodium
roid function tests but negative TRAb. Ultrasound excluded the presence iodide symporter gene expression is another speculation [12]. Lobar
of nodules, with a normal gland size. Thyroid 123I-uptake showed diffuse differences in sensitivity to TSH stimulation is another possibility.
unilateral uptake in the right lobe. Given the possibility of a diffuse hot Muller-Gartner and colleagues evaluated the resistance of TSH receptors
nodule, the patient underwent right hemithyroidectomy. Histopatholo­ to TSH autoantibodies in 256 patients with Graves' disease. They found
gy was compatible with Graves' disease. Eight months following surgery, single or several foci of resistance to TSH-receptor autoantibodies in
thyrotoxic symptoms developed, with laboratory findings and increased about 5 % [13]. The relationship of the aforementioned ideas to the
thyroid scintigraphy uptake in the left lobe comparable with the first development of unilateral Graves' disease is uncertain and further
case. investigation is needed.
Since then, few other cases have been reported [4–8]. In these, In spite unilateral lobar involvement, management should be guided
clinical symptoms of thyrotoxicosis are similar to those in ordinary in the same way as with ordinary Graves' disease. As in the current
Graves' disease, including ophthalmopathy. It might be different in se­ patient, most of the cases reported in the literature were treated and
lective lobe enlargement which may mimic toxic adenoma or Plummer's responded well to antithyroid medication [4–7]. Bolognesi and Rossi [6]
disease. This distinctive presentation was seen in the current case as well reported the findings of a Tc99m scan carried out after two years of
as others [3–8]. On the other hand, one reported case showed no dif­ medical treatment. This showed almost identical scanning pattern of the
ference in the size of both lobes [3]. All reported cases demonstrated affected right lobe but a trace of weak uptake in the left lobe which had
high thyroid function tests with low TSH, except for one case of Saka initially been suppressed. Chen et al. [8] had managed their patient with
et al. in whom thyroid function tests and TSH were initially normal. radioiodine ablation. Following ablation, 131I uptake revealed a homo­
Thyroid autoantibodies displayed disparate results. This is consistent geneous normal uptake in the initially suppressed right thyroid lobe
with what is found in classical Graves' disease. In the current patient, with small amount seen in the affected left lobe. Hemithyroidectomy
both TPO-Ab and TRAb were positive in a manner comparable to several was performed by Saka et al. for reasons other than Graves' disease. The
other cases of unilateral Graves' disease [4–6]. Cytological evaluation remaining lobe was diseased, as shown by clinical, laboratory and thy­
was carried out in two of the reported cases, showing lymphocytic roid scintigraphy findings. Nevertheless, disease regression was suc­
infiltration of the affected right lobe but not the left [4,5]. Unilateral cessfully achieved with antithyroid medication. Thus, when surgical
uptake on scintigraphy was the distinctive feature of unilateral Graves' management is selected for unilateral Graves' disease, the optimal sur­
disease. All except one [7] involved the right lobe in a manner identical gical intervention and possibility of recurrence should be taken into
to our patient. A larger right thyroid lobe in a normal gland, and a consideration.
preponderance of left lobe involvement in thyroid hemiagenesis, sug­
gests the possibility of predominant right lobe organogenesis and func­ 5. Conclusion
tional advantages over the left [5]. Furthermore, a study evaluating
lobar activity in Graves' disease with diffuse intense TC99m uptake Unilateral uptake on thyroid scintigraphy and/or selective lobar
showed that the right thyroid lobe expressed a higher uptake than the goiter in the context of hyperthyroidism could be the presentation of
left [9]. unilateral Graves' disease. Collection of clinical, laboratory and imaging
The cause of unilateral Graves' disease is not yet known. Various results are needed to distinguish this rare manifestation of the disease.
possibilities have been suggested. Since TSH stimulation affects thyroid Management procedures are identical to those adopted in the classical
gland function and growth, a variable lobar reaction may be present in form of Graves' disease including the extent of surgery.
unilateral Graves' disease [10]. This functional and/or structural vari­
ation might be caused by a pre-existing congenital condition (such as

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M.S. Bu Bshait International Journal of Surgery Case Reports 114 (2024) 109138

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Conflict of interest is denied.

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