Cystic thyroid lesions in children

Cystic Thyroid By Adi Yoskovitch,

Jean-Martin

Lesions in Children

Laberge, Montreal,

/3a&ground/furpose; pass a wide and children, ranging nant tumors. The presentation and pediatric population.

Cystic lesions of the thyroid encomheterogeneous group of disease states in from benign purely cystic entities to maligpurpose of this study was to study both the management of cystic thyroid lesions in the

Methocfs:A retrospective review of all thyroid masses presenting between 1978 and 1996 and found to be purely or partially cystic on ultrasound examination was conducted, looking at presentation, family history, laboratory values, ultrasound scan and radionuclide imaging, and pathological and cytological evaluation. /?esu/ts; Twenty-four patients (19 girls, 5 boys) aged years received the diagnosis of cystic lesions of the Of these, 23 presented with painless neck masses, clinically euthyroid, only one had a single abnormal function test, only two had mildly positive antithyroid body test results, and nearly 30% had a positive history of thyroid disease. Ultrasonography showed cysts in five patients and mixed solid cystic lesions

6 to 18 thyroid. 21 were thyroid antifamily pure in 19

R

ELATIVELY COMMON in the adult population. thyroid disease is uncommon in the pediatric population, the prevalence of nodular thyroid disease being less than 1% in chi1dren.l However, the percentage of malignancy in thyroid nodules in the pediatric setting has been reported to be as high as 50%.z-4 Cystic lesions of the thyroid are considered by many investigators to represent benign disease in children,5.6 echoing the adult literaturey-9 with the cysts thought to be the result of necrosis and degeneration of thyroid nodules.s The following represents a review of cases, seen over an l&year period at our center, of children with cystic or mixed solid-cystic thyroid lesions. Their presentation, evaluation, management, pathology, and follow-up are addressed.

From the Depurtments of Otoluyngology, Pediatric Szzrguy, Ezzdocrznoiogy, Radiology, apzd Puthofogy, Montreal Children k Hospital, McGill University, Montreal, Quebec, Canada. Presented at tlze 29th Annual Meetzng of the Canadian Assoczation of Paediutric Surgeons, Bar& Alberta, Cunada, October 3-6, 1997. Address reprint requests to Jean-Martin L.aberge, MD, The Montreal Clzildrenk Hospital, Department of Pediatric Szzrgery, 2300 Tupper St, Montreal, Quebec, Canada H3H lP3. Copyright 0 1998 by WB. Saunders Company 0022-3468/98/3306-0015$03.00/0

Celia Rodd, Anna Sinsky, Quebec

and David Gaskin

patients. On scintiscan, six lesions were hot, 13 were cold, three showed normal uptake, and two were mixed. Treatment included either observation, aspiration, cyst sclerosis, surgery, or combinations thereof. Pathological and cytological results included follicular adenoma (n = 9), cystic degeneration (t-r = 6), multinodular goiter (n = 4), carcinoma (n = 2), branchial cleft cyst (n = I), and undetermined (n = 2). Conc/usiorrs: Thyroid cysts are often thought to represent benign degenerative disease. Our study, which is the first in the literature to specifically address thyroid cysts in children, shows that ultrasound scan is useful in evaluating thyroid masses, whereas laboratory and radionuclide are of less value, and that single lesions of mixed echogeneity are likely to represent neoplasms, a significant percentage of which are malignant. J Pediatr Surg 33:866-870. Copyright @ 1998 by W.B. Saunders Company. INDEX sound,

WORDS: aspiration,

Thyroid, cyst, surgery.

MATERIALS

malignant,

AND

radionuclide,

ultra-

METHODS

A retrospective study was undertaken that reviewed all patients at the Montreal Children’s Hospital as seen in the departments of surgery. otolaryngology. and endocrinology, between 1978 and 1996, who had cystrc lesions seen on ultrasound examinations for neck masses. All patients underwent radionuclide thyroid imaging studies to ascertain the degree of function of cysts, as well as thyrotd function testing and antithyroid and antimicrosomal antibody testing. The patients were subsequently treated in a variety of methods, including pharmacological treatment, cyst aspiration, surgery. or combinations thereof. Those undergoing cyst aspiration or surgery had tissue and fluid samples sent for cytological and pathological evaluation.

RESULTS

Between 1978 and 1996,24 children, five boys and 19 girls, ranging in age from 6 through 18 years (mean, 13 years), were found to have cystic lesions seen on ultrasound examination. Twenty-three of the patients were being evaluated for neck masses, and one patient was found to have a cystic neck mass as an incidental finding on magnetic resonance imaging (MRI) (Fig 1) for evaluation of an unrelated condition, later reconfirmed as cystic on ultrasound scan. The results are summarized in Table 1. Twenty-three of 24 patients (95.8%) presented with painless neck masses, with 1 of 24 presenting with a tender mass and 1 of 24 being discovered incidentally. Journa/

ofPediatric

Surgery,

Vol33,

No 6 (June),

1998: pp 866-870

THYROID

867

CYSTS

Fig 1. MRI of a 12~year-old boy investigated for leg spasticity. (A) MRI shows an incidental cystic mass Tz-weighted imaging. (Bl On ultrasound scan the nodule was described as solid with a small cystic component. discovered to be an intrathyroid branchial cleft cyst, ie, fluid filled with debris.

Twenty-one of 24 (87.5%) patients were euthyroid, with the remaining three patients having symptoms of either hypo- or hyperthyroidism. Only one patient had abnormal thyroid function test results (decreased TSH), whereas two patients had detectable microsomal antibodies, albeit with low titers, Seven of the 24 patients (29.2%) had some form of family history of thyroid disease. Of the 24 patients, five had purely cystic lesions on ultrasound, whereas 19 (79.2%) had mixed solid and cystic components. Furthermore, 12 of 24 were single cystic lesions, and 12 of 24 were multiple lesions. Radionuclide uptake studies showed 6 of 24 (25%) to exhibit increased uptake (“hot”), 13 of 24 (54.2%) to have decreased uptake (“cold”), 2 of 24 (8.3%) to exhibit both hot and cold areas, and 3 of 24 (12.5%) to have normal uptake. In terms of initial treatment, 23 of 24 patients were Table Fmal Diagnow (n = 24)

1. Cystic

Thyroid

Cyst Quahty

Lesions

Data Summary

Multicentricrty

RadmnwAde

lmagmg

~ Pure

Partial

Smgle

MultIpIe

Hot

0

9

6

3

42

12

3

3

2

4

13

2

0

1

3

0

4

04

0

0

0

1

1

0

010

0

0

1

1

0

010

0

0

1

1

0

010

0

1

1

I

1

II

Cold

Normal

Both

Folkular adenoma In = 91 Cystic

degen-

eratlon (n = 6) Multinodular goiter (n = 4) Papillary carcinomatn = I) FollicularcarcinomaIn=l) Branchial cleft cyst (fl = I) No pathology or W~b7Y h = 2)

0

0

in the left lobe of the thyroid on At the time of operation, it was

treated with a trial of suppression therapy, with 2 of 24 (8.3%) responding to treatment. Subsequently. 4 of 24 (16.7%) had cyst aspiration with continued suppression therapy, 17 of 24 (66.7%) underwent surgery, 2 following an initial aspiration, 1 of 24 had the cyst injected with tetracycline and subsequently aspirated, and 2 of 24 (8.3%) were observed. Of the four patients undergoing cyst aspiration alone, three (75%) required repeat aspiration. As such, of the six patients treated initially with aspiration, five (83.3%) required either additional aspirations or surgery. Of the patients treated with surgery, 2 of 17 underwent total or subtotal thyroidectomy, whereas 15 of 17 underwent hemithyroidectomy. Of the twenty-four patients, twenty-three (including both patients treated with observation) received suppression or replacement therapy. The pathological and cytological results include cystic degeneration (n = 6), follicular adenoma (n = 9) multinodular goiter (n = 4), branchial cleft cyst (n = l), papillary carcinoma (n = l), follicular carcinoma (n = l), and undetermined (n = 2). For those patients in which tissue or fluid samples were available, 2 of 24 (8.3%) were found to be malignant. In looking at specific pathological diagnoses, of the two cases diagnosed as malignant, both were single lesions, and both malignant cases were mixed solidcystic (Fig 2). In considering radionuclide imaging both were cold. Of the four cases diagnosed as multinodular goiter, all appeared as multiple cystic lesions and all were cold on radionuclide imaging. In examining those lesions diagnosed as follicular adenoma, all nine were single lesions with seven of nine (77.7%) being mixed solid-cystic (Figs 3 and 4).

YOSKOVITCH

Fig 2. Seven-year-old with a cystic component tion showed a follicuiar sion.

boy with a thyroid nodule described as solid on ultrasound scan. Pathological examinacarcinoma with capsular and vascular inva-

For those patients with a diagnosis of cystic degeneration, multinodular goiter, or cyst not pathologically diagnosed, a follow-up was undertaken. Of the eleven patients, three were lost to follow-up after age 18. However, until age 18, on follow-up examination (ranging between 4 and 9 years) these three patients had no recurrence of disease. In the remaining eight patients, at follow-up (ranging between 2 and 10 years) all eight were without recurrence or progression of disease.

A c3

Fig 3. (AI Sixteen-year-old areas on ultrasonography.

ET AL

,~

girl with Pathological

Fig 4. Sixteen-year-old boy with a 4.4- x 3-cm thyroid cyst with a small mural nodule. Pathological findings showed a follicular adenema.

..!

a hot nodule on technetium findings showed a follicular

scan. (61 The nodule adenoma.

is largely

cystic

with

multiple

thick

septations

and solid

THYROID

CYSTS

869

DISCUSSION

Although uncommon in clinical practice, cystic lesions of the thyroid in children and adolescents command an important degree of attention. The differential diagnosis, as seen in our and other studiesl”,ll is summarized in Table 2. Clearly, the initial point of investigation of the thyroid cyst centers around a comprehensive history and physical examination. A positive history of previous head and neck irradiation is extremely important to note because of the well-known link between such radiation and the development of thyroid malignancy.lz As well, a family history of thyroid cancer or thyroid disease is also important to investigate for possible hereditary entities such as multiple endocrine neoplasia. During the history, the presentation of the thyroid mass needs to be evaluated with respect to growth pattern, laterality, and associated symptoms of hoarseness, dysphagia, and hyper or hypothyroidism, all of which have different implications with respect to management of the cystic lesion. In the course of the physical examination the presence of single versus multiple nodules should be carefully noted. The presence of lymphadenopathy in tandem with a thyroid nodule is often an indicator of malignancy and warrants swift attenti0n.l Of key importance in evaluating thyroid nodules is the use of ultrasound scan. Ultrasound scan is useful to distinguish between cystic and solid massesI and to evaluate the presence of smaller, nonpalpable lesions and the overall echotexture of the gland. Considered to be benign by many investigators,G,scystic lesions are thought to represent thyroid nodule hemorrhage, degeneration, and necrosis.s Our study showed that of the 24 cystic thyroid lesions reviewed, seven were consistent with cystic degeneration, and, in total, 22 were considered as benign. However, 8.3% of the cystic lesions were malignant with an additional 37.5% representing benign neoplasia. As such, malignancy is an important part of the differential diagnosis of cystic thyroid lesions, and therefore cystic lesions should not be merely dismissed as a benign condition. In further evaluating cystic thyroid lesions in children, thyroid function testing does not appea to be of great Table 2. Differential Lesions

Diagnosis

in the Pediatric

of Cystic Population

Bentgn cystic degeneration Thyroglossal duct cyst Parathyrold cyst Branchial cleft cyst Follicular adenoma Chronic lymphocytic MultInodular Carcinoma

thyroiditis

goiter of the thyroid

Thyroid

usetilness. In the present study, only one patient had abnormal values. This is a point previously noted by Hung et al,s in which 70 of 71 patients had normal laboratory values. Furthermore, testing for the presence of antithyroglobulin and thyroid antimicrosomal antibodies again seems of little value because a positive test for antimicrosomal antibodies occurred in only two of the patients in the current study. These values were only mildly positive and seemingly not specific for any particular disease. Therefore, it can be concluded that serum thyroid function tests and antibody testing is of little diagnostic value in evaluating cystic thyroid lesions in those instances in which the patient is clinically euthyroid. The next step in evaluating pediatric patients with cystic lesions on ultrasound scan is the use of radionuelide imaging. Thyroid scintiscanning is controversial. Several investigators have described its limited usefulness.14J5It is our contention that radionuclide imaging does provide useful adjunct information but rarely does it change the management of cystic lesions. With respect to treatment, several avenues need to be investigated. Trial suppression with thyroid hormone therapy did not appear to have much influence on long-term treatment, as echoed by other studies;.16 although it may have a role postoperatively in preventing recurrence. Cyst aspiration also remains a controversial area. Although effective in aiding in the assignment of a pathological diagnosis as well as decreasing cyst size,‘j.‘O our study, along with others, has shown the necessity for multiple aspirations in many instances, with a reported recurrence rate of 30%.l” Although not studied in the pediatric setting, the adult literature yielded several reports documenting a significant false-negative rate of fine-needle aspiration ranging from 0.5% to ll.5%,17-zo with the false-negative results attributed to sampling error and interpretive mistakes.21 As such, we recommend cyst aspirations be performed only in those patients with multiple cysts and that solitary cystic lesions, either purely cystic or mixed solid-cystic, be surgically excised rather than aspirated. It should be noted that, although unlikely, needle aspiration does have a potential risk of spread of malignancy along a needle biopsy tract and a single case of such an occurrence has been reported previouslySZz In this current study, one cyst was injected with tetracycline. The cyst recurred, as seen in other studies,23 and subsequent aspiration was required. However, several investigators24-Z6have concluded that either tetracycline or ethanol instillation into thyroid cysts is a safe and effective management tool. Our limited use of cyst sclerosis techniques precludes us from making formal recommendations.

870

YOSKOVITCH

By far the most definitive management of thyroid cysts is surgical excision, with the resultant specimen available in toto for conclusive pathological examination. The decision of when to undertake surgery can provide certain challenges. Clearly, all solitary cystic lesions that show decreased radionuclide uptake should be excised because of the incidence of malignancy as documented in the literature27 and seen in the current study. Furthermore, cystic or partially cystic lesions found to contain malignant cells on fine needle aspiration should also be excised. The development of a thyroid cyst in the setting of previous irradiation warrants surgery in light of the well-documented association of thyroid malignancy and radiation.12.zs The presence of cervical lymphadenopathy in conjunction with a thyroid cyst should be extremely suspicious for malignancy1~i2J4J9 and warrants excision. As for solitary cystic lesions showing increased radionuelide uptake, although not seen in the current study, other investigatorGr6 have recommended that all hot solitary

ET AL

nodules should be removed because of a higher incidence of carcinoma in children. The only lesions that need not be removed unless symptomatic are multicystic lesions, which are most often multinodular goiters. These may be followed up closely and treated pharmacologically. In these lesions aspiration and cytological evaluation may be useful, and cyst sclerosis may be considered for large conspicuous cysts that recur despite aspiration and suppression. Cystic lesions of the pediatric and adolescent thyroid are uncommon but clinically important entities. Ultrasound examination is particularly useful in defining these lesions, with radionuchde imaging useful as an adjunct tool. Thyroid function and serum antibody testing are of little apparent usefulness. Treatment should center around surgical excision of these cysts, with the exception of multicystic lesions. Preoperative suppression and cyst aspiration have only a limited role in the management of these cystic lesions.

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