Parathyroid Adenoma: Diverse Modalities of Clinical Presentation. Series of Cases and Literature Review. January 2010-October 2020
Introduction
The surgical treatment of primary hyperparathyroidism (pHPT) changed in the past two decades [1, 2, 3]. The prevalence of this condition is very variable. The anatomic knowledge and imaging methods allow a surgical planning with success rates of minimally invasive parathyroidectomy similar to those established for bilateral neck explorations [4, 5, 6]. The choice of imaging modality should be a consideration for efficacy, expertise, and availability of such techniques in clinical practice [7, 8]. The reported success rate of this traditional approach, for skilled surgeons, is 90% [9]. There is controversy in surgical criteria, extent of surgery and minimally invasive approach, etc. There is universal agreement to surgical treatment. However, some protocols in highly specialized centers are not possible to perform in ours center due to their high costs. The main of this study is to present the clinical characteristic, the diagnosis method biochemical, imaging and surgical approached and follow up of patients with PA.
Methods: Descriptive study, Cohort, Series of cases. We reviewed the record of 450 patients diagnosed with a tumor in head and neck during the period January 2010 to October 2020. We selected the record of patients with diagnosed of PA. The clinical characteristics of the patients according demographic aspect, symptoms, biochemical and imaging methods, complementary diagnosis test, surgical approach, histological classification according WHO Tumor of Endocrine Organs 4th edition 2017 [10]. The frozen section during the surgery indicted with the freezing microtome and stained with hematoxylin and eosin. The univariable review realized with frequency and percentage using Excel 2016. Results: We selected 6 (1.3%) patients with PA, male 5(83%) female 1(17%) The age range was 28-72 years old, the median 49.6 in the neck located 4(66%), 1(17%) in upper mediastinum and 1(17%) in cervical and thoracic location.1 patient with recurrence after of five years. 1 patient with three PA and 1 with two. The parathyroid gland affected according to Table 1.
| Parathyroid Adenoma Localization | Case 1 | Case 2 | Case 3 | Case 4 | Case 5 | Case 6 | |
|---|---|---|---|---|---|---|---|
| Upper right gland | |||||||
| Lower right gland | (+) | (+) | (+) | ||||
| Upper left gland | (+) | ||||||
| Lower left gland | (+) | Recurrence (+) | (+) | (+) | (+) | ||
| Ectopic ( Upper Mediastinum) | (+) | ||||||
| Symptoms | Case 1 | Case 2 | Case 3 | Case 4 | Case 5 | Case 6 | |
| Bone loss( Spine/Hip) | (-) | (+) | (-) | (-) | (+) | (-) | |
| Renal function | chronic failure | chronic failure | chronic failure | (-) | Chronic failure | (-) | |
| Cardiovascular diseases | (-) | (-) | (-) | Hypertension | (-) | (-) | |
| Neck swelling | (-) | (-) | (-) | (-) | (-) | (+) | |
| Disphagia | (-) | (-) | (-) | (-) | (-) | (+) |
Table 1: Symptoms more frequent in patients with PA.
Table1: Anatomic Localization of PA.
Symptoms show in Table 2.
Imaging methods used to locate PA according show Table 3.
| Case 1 | Case 2 | Case 3 | Case 4 | Case 5 | Case 6 | |
|---|---|---|---|---|---|---|
| Ultrasonography | (+) | (+) | (+) | (+) | (+) | (+) |
| Technetium-99 Sestamibi Scanning | (+) | (+) | (+) | (+) | (+) | (-) |
| Magnetic Resonance Imagen | (-) | (+) | (+) | (+) | (+) | (-) |
| CT Scan | (-) | (-) | (-) | (-) | (-) | (+) |
Table 2: Imaging methods to locate parathyroid tissue.
and 1 unknown. The surgical approach with biochemical control of PTH and Calcium prior and post-surgery according to Table 4.
| Surgical approaches | PTH pg/Ml prior surgery | PTH pg/Ml post-surgery 1 | PTH pg/Ml post-surgery 2 | Calcium mg/dl prior surgery | Calcium mg/ dl post- surgery 1 | Calcium mg/ dl post- surgery 2 | |
|---|---|---|---|---|---|---|---|
| Case 1 | Bilateral cervical exploration | 1806 | 934 | 6.5 | 10.1 | 9.5 | 8.5 |
| Case 2 | Cervical exploration | 2142 | 1428 | 1059 | 11.53 | 9.8 | 8.9 |
| Case 3 | Cervical exploration | 3000 | 1000 | 25 | 9.8 | 8.8 | 8.2 |
| Case 4 | Minimally invasive- Thorascopic | 384 | 250 | 56.1 | 14.9 | 11.3 | 9.9 |
| Case 5 | Bilateral cervical exploration | 1319 | 263 | 21.8 | 11.2 | 10.7 | 7.9 |
| Case 6 | Doble cervical and thoracic open | unknown | 9.72 | 9.65 | 8.46 |
Table 3: Surgical approach and biochemical control PTH and Calcium prio and post-surgery.
Pathology register reveled PA, 5(83%), and 1(17%) a giant nonfunctioning Parathyroid cyst. During the follow only 1 patient presented recurrence five years later in the contralateral parathyroid gland and required reintervention. None with post-surgery complication.
Discussion
The behavior of the parathyroid disease is very variable, depend of the region where was realized the research. The prevalence of this condition in the United States between women vs. men is similar to the reported by Eufrasino with a major incidence of menopause women [11, 12]. The european studies reported relatively low incidence, however, Scotland reported higher incidence [13]. Our series diagnose a small group of patients with parathyroid diseases concomitant with CKF. This explains that our results are different from other studies. The majority of our patients were men, however, De Lucia reported the effected of gender and geographic location on the expression of pHPT [14]. Others, reported that PD depend of race and was highest among blacks, followed by whites, but hispanic and other races were lower than that for whites [15, 16]. The age range and median age are similar to studied by Eufrasino. Other papers reported age ranged from 20 to 99 years, the incidence is higher, such that in groups 70-79 the risk is grater [17]. The majority of our patients presented adenoma in a normal arrangement of the parathyroid glands, this could be explained becouse location of the parathyroid glands are constant in 75% of the population. However, the presence of one in ectopic position (mediastinum) demonstrates the complex embryonic development of the inferior parathyroid [18, 19]. One of our cases was a giant cystic adenoma, occupying the neck and the mediastinum. The review of 14 cases show that large cysts were nonfunctioning as our study [20]. Large cyst formation is always associated with an adenoma with hemorrhage. Physiological pressure changes over the thoracic inlet may well explain the upward movement of any mass into the neck on forced expiration [21]. A cyst mass on the left side of the neck with dysphagia is a rare clinical manifestation [22, 23, 24].
The majority of patients had a single PA, similar to the reported by Udelsman, et al. [25, 26]. A Review of the pathological findings in 140 Cases of pHPT by Lewis, the single adenoma represented 80% and parathyroid cancer is uncommon with less than 1.5% [27] there was no patient with tumor in other endocrine organ. Cushing and Davidoff reported a case of gigantism with an eosinophilic pituitary adenoma, an adrenal adenoma, an adenomatous goiter and parathyroid hyperplasia [28]. Coincidental adenomas of the islet cells of the pancreas, parathyroid, and pituitary has been reported by Shelburne, et al. [29] the majority of the our patients had nephrolithiasis as described Rejnmark [30] other trial reported prevalence ranged from 7 to 20% [31]. Sorensen reported the lowest range 3% in adults [32]. Nephrocalcinosis or silent nephrolithiasis are indications for parathyroidectomy in patients with suspected pHPT [3]. The association between parathyroid hyperplasia and CKF has been known since early 1930 [33]. Many observations suggest that secondary hyperparathyroidism (HPT) is often an early event in renal disease and parathyroid tissue involvement generally increases proportionately with the increasing severity and duration of functional renal impairment.
These results suggested that in renal HPT, the parathy- roid glands initially grow diffusely and polyclonally, and that the cells in the nodules are later transformed to monoclonal neoplasia and proliferate aggressively [34]. A little more than a third of our patients with bone mineral density were be- low at the spine or hip with higher risk to fracture. Similar to the reported by Misiorowski suggested that the prevalence of HPT is higher among patients with low bone mineral den- sity [35]. The risk of fracture is frequent in patients with HPT compared with persons who did not have the condition. This group of patients makes them a candidate for surgical treat- ment as our two cases [36, 37]. The accurate preoperative localization has become more important to enable a success- ful surgical outcome. Parathyroid ultrasound requires skilled radiologist and interpreters with knowledge of parathyroid embryology and anatomy to access parathyroid glands in eu- topic and ectopic positions [38]. The sensitivity and speci- ficity of sonography for identifying enlarged parathyroid glands were 74% and 96%; similar reported by Siperstein and Solorzano. Sonography correctly predicted a solitary ad- enoma or multiglandular disease [39, 40, 41]. All patients used the ultrasound as diagnosis tool of low cost but of great util- ity in low income countries, according The American Associ- ation of Endocrine Surgeons Guidelines. The CT and MRI are complementary imaging studies [42, 43, 44]. Nuclear medicine today has a rol in the detection of the number of sick glands or ectopic gland [3, 45, 46]. Singh reported 63 % of sensibility and predicted positive value of 90% but decreased between 53%-74% in localization in second surgery [47, 48]. On the other hand, a metanalysis reported that the sensibility de- creased according the number of gland disease [49]. 99mTc- MIBI y SPECT/CT have demonstrated greater precision in the preoperative localization, with great superiority than ultrasound, CT and MRI but it is a very expensive technique [50]. Our patients presented indication for parathyroidec- tomy, according to the guidelines of The American Associa- tion of Endocrine Surgeons [13, 51, 52, 53]. The success rate for surgeons in centers with expertise is a cure rate of 95% [54]. A case presented as a recurrence in the contralateral gland five years after the first surgery, similar to the reported by Karakas and Weber [55, 56]. One of our cases approached by Thorascopic, demonstrate the importance of surgical skills with minimal invasive techniques with similar percentage of cure [57, 58, 59]. We had a multiple parathyroid adenoma case, as reported for several series. This patient classified as group 2 according to criteria of Lewis and Reymond: multiple para- thyroid adenoma, three parathyroid glands large and renal insufficiency present [60, 61]. The majority of our patients presented with intraoperative specimen biopsy, with the advent of other complementary intraoperative techniques, some authors have argued for the superiority of intraopera- tive hormonal analyzes over frozen cutting. PTH fast is a fast and reliable test to ensure adequate resection of parathyroid tissue, however it is expensive for our country. Aygun con- cluded that the routine use of frozen section examination is not recommended [62, 63, 64]. While experts condemn the util- ity of the frozen tissue examination of the surgical specimen in parathyroid disease, as it is deemed difficult and ineffec- tive in the event of multiglandular disease [65]. Secondly, the frozen tissue examination diagnosis is thus feasible in a well- selected population. The pathologist’s expertise is, there- fore, the main factor determining the reliability of this ex- amination [66, 67].
Conclusions
The multidisciplinary approach permitted the success obtained and surgery planning for each patient with PA. The results show like other papers, that using the technological development of imaging studies, the support of the frozen biopsy for the lack of PTH fast, allowed to corroborate the surgical remotion of the diseases gland with high percentage of cure.
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