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Painful osteitis fibrosa cystica associated with parathyroid adenoma

Santivañez Juan José1, González- Devia Deyanira2, Vergara Catalina3, Salamanca Omar3, Buriticá Catalina4 Escallón Alberto1

¹Department of Head and Neck Surgery, Fundación Santa Fe de Bogotá University Hospital, Bogotá, Colombia.
²Department of Internal Medicine, endocrinology section, Fundación Santa Fe de Bogotá University Hospital, Bogotá, Colombia.
³Department of Internal Medicine, Fundación Santa Fe de Bogotá University Hospital, Bogotá, Colombia.
⁴Department of Pathology, Fundación Santa Fe de Bogotá University Hospital, Bogotá, Colombia.

*Corresponding author

*Juan Jose Santivañez, MD, General surgeon, Department of Head and Neck Surgery, Fundación Santa Fe de Bogotá, University Hospital, Carrera 7 No. 117-15, Bogotá, 110111, Colombia.

ABSTRACT

Primary hyperparathyroidism (PHPT) is a common endocrine disorder characterized by hypercalcemia and elevated or abnormally normal parathyroid hormone (PTH) levels, resulting from the excessive secretion of PTH by one or more parathyroid glands. In this report, we emphasize the significance of a proper approach when dealing with bone lesions. Our patient experienced a 4-month history of incapacitating pain in her right hip, leading to serious difficulty in walking. An MRI revealed multiple bone lesions, initially raising suspicion of plasma cell neoplasia. A comprehensive evaluation of the phosphocalcic panel is always essential to rule out conditions like primary hyperparathyroidism as the underlying cause of the lesions, as seen in this case. Once hyperparathyroidism is diagnosed, imaging becomes crucial for identifying the side of the lesion. Additionally, a thorough ultrasound evaluation of the thyroid tissue is important. This is underscored by the incidental finding of multifocal thyroid carcinoma in this case, emphasizing the need for resolution during the same surgical intervention. The improvement following surgical management was immediate, with a return to usual mobility within a few days after the surgery.

Introduction

Primary hyperparathyroidism (PHPT) is a common endocrine disorder characterized by hypercalcemia and elevated or abnormally normalparathyroid hormone (PTH) levels due to excessive secretion of PTH by one or more parathyroid glands (1). In most cases, the cause of PHPT is a benign adenoma (80-85%), while hyperplasia and carcinoma are rare causes (10-15% and <1%, respectively) (1,2).

In Western countries, the symptomatology of primary hyperparathyroidism is relatively mild, and its progression is slow. The main effects manifest in the renal and skeletal systems. Nevertheless, patients may encounter non-specific symptoms such as fatigue, weakness, paresthesias, digestive disorders, and mental alterations (3).

Currently, prompt diagnosis has led to a reduced association between primary hyperparathyroidism (PHPT) and severe skeletal features, however, when the disease advances, characteristic bone lesions may be encountered. Brown tumor of the bone is a characteristic, uncommon non-neoplastic lesion arising from abnormal bone metabolism in hyperparathyroidism, primarily impacts facial bones, clavicle, ribs, pelvis, and femur (4).

This report highlights a case involving a patient experiencing incapacitating pain in her right hip for four months, along with multiple lesions detected on MRI. The suspicion of plasma cell neoplasia was prompted by hypercalcemia and the presence of multiple lytic bone lesions. The report delves into the diagnostic and therapeutic challenges posed by this atypical presentation.

Case Report

A 45-year-old previously healthy woman presented to the emergency department with a 4-month history of incapacitating pain in her right hip, leading to serious difficulty in walking. She had sought consultation with orthopedics and underwent a pelvic magnetic resonance imaging (MRI), which revealed a lytic lesion in the right hip.

On physical examination at admission, her vital signs were normal, and there was no limitation in hip range of motion. Further investigations showed hypercalcemia (ionized calcium 1.76 mmol/L) and a chest computed tomography (CT) scan revealed polyostotic involvement in the iliac bones, pubic rami, and left scapula.

The suspicion of neoplasia of plasma cells was raised due to hypercalcemia and multiple lytic bone lesions. Bone marrow biopsy, PET scan, and expanded phosphocalcic profile studies were ordered. The investigations revealed Vitamin D deficiency (11.3 ng/ml) and elevated PTH (1385 pg/ml). PET scan with FDG showed multiple hypermetabolic lytic lesions located in the axial and appendicular skeleton with a tumoral appearance (Figure 1), along with a hypercaptant isthmic thyroid nodule.

Thyroid ultrasound revealed a 1 cm isthmic nodule with irregular borders and a 3 cm anterior thyroid gland image suggestive of a parathyroid adenoma (confirmed with parathyroid SPECT).

Figure 1: CT scan and PET-CT study revealing multiple lytic lesions located in the axial and appendicular skeleton with tumoral appearance, predominantly observed in both iliac bones and ileopubic branches

Figure 2: A) Thyroid gland with four papillary thyroid carcinomas between 0,1 to 9 mm, inflammatory mediastinal lymph nodes, and a 3cm left parathyroid adenoma. B) Parathyroid gland adenoma, consisting of well circumscribed benign cellular proliferation of parathyroid cells, with absent adipocytes. C) Thyroid gland with isthmic papillary carcinoma.

Figure 3: Marked improvement of the osteoclastic component and remineralization of the lesions located in the iliac wings in the superior aspect of both acetabulums.

Fine-needle aspiration biopsy from isthmic nodule identified papillary thyroid carcinoma. Bone marrow biopsy ruled out neoplasia of plasma cells and showed findings suggestive of brown tumor.The patient underwent parathyroidectomy and total thyroidectomy. Prior to the start of surgery, a parathyroid hormone (PTH) sample was taken, resulting in a measurement of 1048 pg/mL. During the surgery, upon opening the infrahyoid muscles, a roughly 3 cm adenoma on the left side, situated anterior to the gland, was observed. This adenoma was excised, and the total thyroidectomy was performed without complications (figure 2). New PTH samples were taken at 10- and 20-minutes post-surgery, yielding values of 139 pg/dL and 65 pg/dL, respectively.

The patient exhibited a satisfactory postoperative recovery, experiencing a remarkable improvement in both bone and muscle pain almost immediately. She was discharged within 2 days. Pathological examination confirmed a left-sided parathyroid adenoma measuring 3 cm, along with a 6 mm thyroid microcarcinoma. Follow-up imaging examinations at 5 months revealed significant improvement in all lytic lesions as well as no pain at all. Particularly noteworthy was the marked improvement in the osteoclastic component, with remineralization of lesions located in the iliac wings at the superior aspect of both acetabulae (figure 3).

Discussion

Brown tumors are giant cell lesions arising from abnormal bone metabolism in hyperparathyroidism, result from the heightened circulating parathyroid hormone (PTH) levels, intensifying osteoclastic bone resorption. This process contributes to diffuse osteopenia, fractures, or the formation of multiple circumscribed lytic lesions (5).

Currently, the occurrence of bone findings associated with hyperparathyroidism is less frequent. This trend is generally attributed to improved access to healthcare, early comprehensive bone laboratory profiles, and increased availability of previously more intricate tests, including measuring parathyroid hormone (PTH), ionized calcium, or Vitamin D.

As illustrated in this case, a meticulous approach is imperative, particularly when addressing bone lesions. A thorough evaluation of the phosphocalcic panel remains essential to exclude conditions such as primary hyperparathyroidism as the underlying cause for the lesions. Furthermore, upon diagnosing the parathyroid adenoma, a detailed ultrasound assessment of thyroid tissue becomes crucial. This is underscored by the incidental discovery of multifocal thyroid carcinoma in this case, emphasizing the necessity for resolution during the same surgical intervention.

The post-surgical management in this case aligns with the existing literature, emphasizing the effectiveness of prompt surgical intervention. The progression of the patient's recovery at the 5-month follow-up, showcasing substantial improvement in lytic lesions, offers insights into the positive impact of timely surgical intervention. Continued monitoring of such cases and the development of standardized follow-up protocols may enhance our understanding and contribute to improved patient outcomes in the future.

Ethical Component: This case report was carried out within the ethical principlesfor medical research in humans according to the Declaration of Helsinki—59th General Assembly, Seoul, Korea, October2008. The national regulations of the Ministry of Health andSocial Protection of Colombia Resolution 8430 of 1993regarding Chapter I “Of the ethical aspects of research inhuman beings” were taken into account. This research isclassified within the research category without risk, andwith the informed consent of the patient.

Data Availability: The case information was obtained in an authorized manner by the patient and the treating surgical group.

Funding Statement: There is no funding to report for this paper.

Declaration of Conflicting Interests and grants: The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article. No grants where received.

References

  1. Khan AA, Hanley DA, Rizzoli R, et al. Primary hyperparathyroidism: review and recommendations on evaluation, diagnosis, and management. A Canadian and international consensus. Osteoporos Int. 2017;28(1):1-19.
  2. Bilezikian JP, Bandeira L, Khan A, Cusano NE. Lancet. 2018;391(10116):168-178.
  3. Multifocal Brown Tumor as A Manifestation Of Primary Parathyroid Carcinoma. A Case Report. Reyes et al. Rev. Colomb. Radiol. 2020; 31(2): 5254-9
  4. Agnihotri M, Kothari K, Naik L. Brown tumor of hyperparathyroidism. Diagn Cytopathol. 2017;45:43–4.
  5. Selvi F, Cakarer S, Tanakol R, Guler SD, Keskin C. Brown tumour of the maxilla and mandible: a rare complication of tertiary. DentomaxillofacRadiol. 2009;38:53–8.

Journal of Clinical Case Reports, Medical Images and Health Sciences (ISSN 2832-1286) is a peer-reviewed journal dedicated to publishing clinical images from all sectors of medicine. The goal of this magazine is to disseminate information about new discoveries and treatments in science and medicine.

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