Adenocarcinoma with squamous metaplasia in the parathyroid gland of a spur-tighed tortoise (Testudo graeca)
Case report



GÁL János1*, SZABÓ Gyula1, JAKAB Csaba1, GÉCZY Csaba1, SÁTORHELYI Tamás2


1 Szent István University, Faculty of Veterinary Medicine, Department of Pathology and Forensic Veterinary Medicine. István u. 2. H-1078 Budapest. *E-mail: gal.janos@vipmail.hu
2 Ófalu Pets’ Clinic

Summary
The authors diagnosed primary adenocarcinoma while dissecting a spur-tighed tortoise carcass. In the tumour tissue secondary squamous metaplasia was also observed, as the first reported case in this species. Due to the adenocarcinoma of the parathyroid gland, the concentration of parathyroid hormone in the blood increased, which resulted in the extraction of inorganic matrix substances from the bony tissue of the carapace and plastron. To identify the tumour type and to characterise its behaviour immunohistochemical assays routinely used in human diagnostics – such as Ki-67 and PAN-cytokeratine – were applied by the authors.

Amongst the endocrine organs of reptiles, parathyroid gland plays an important role in calcium and phosphorous metabolism. In general, the parathyroid gland is located near to thyroid gland. In turtles and tortoises the gland is located between the lobes of the thymus, which makes it difficult to find it during dissection (4). The gland parenchyma consists only of main cells with spherical nucleus and smooth granulation in the cytoplasm. The main cells form bands with one or two cell layers and in some cases follicular structure can also be observed (1).
The level of the parathyroid hormone – produced by the parathyroid gland – in the blood increases by the decrease of calcium level. This causes an increase in the activity of the osteoclasts in the bony tissue, which results in the disassembly of the inorganic matrix in the bones. Meanwhile, the excretion of phosphorous in the kidneys intensifies, and the depletion of calcium decreases. Parathyroid hormone significantly increases the intensity of 1,25-dihidrocolecalcipherol synthesis, which stimulates calcium uptake in the guts (3, 4, 5).
Up to our knowledge, only adenoma was reported as a parathyroid tumour in reptiles until now. In lizards, it was detected in green iguanas (Iguana iguana). In tortoises adenoma was reported in redfoot tortoise (Geochelone carbonaria), spur-tighed tortoise (Testudo graeca) and desert tortoises [Xerobates(Gopherus) agassizi]. In each case the parathyroid gland was swollen in these reptiles. In tortoises the bony tissue of the shell became soft and could be pressed easily with a finger. In macroscopic investigations of other organs no pathological malformations were detected. The pathohistological investigations detected multiple layers of main cells forming bands. Occasionally, these bands had a spindle shape and a remarkable eosinophil granulation was observed in the cytoplasm. In some cases the vacuolization of the cells was also observed (1, 2, 4). In redfoot tortoises the increased activity of osteoclasts and the prolification of (collagene fibre) connective tissue were reported. In the kidneys of tortoises deposition of calcium salts was also observed (2).

Case report
On September 5th, 2004, a carcass of a female spur-tighed tortoise (Testudo graeca) originating from a private collection was investigated in the Department of Pathology and Forensic Veterinary Medicine, Faculty of Veterinary Sciences, Szent István University. The animal was approximately 8 years old, with a carapace length of 15.5 cm.
The tortoise was previously kept in a home terrarium with a floor space of 100X90 cm. The bedding of the terrarium was garden soil. The fodder was fresh vegetables and fruits supplemented with vitamins and mineral salts which were provided to the animals daily.
Following the external investigation of the body, the carcass was dissected. Samples were fixed in formaldehyde solution of 8% from the organs showing malformations. After embedding the samples in paraffin, sections were cut and stained with haematoxylin eosin stain.
To study the calcification status of bones von Kossa silver staining was used. Besides, for allocating the replicative status of the tumour found, Ki-67 assay and for detecting the ephitelial marker PAN-cytokeratin immunohistochemical analysis was also performed. These immunohistochemical reactions were also performed on the parathyroid gland of a healthy spur-tighed tortoise, as a control. Amongst the antibodies used, Ki-67 primary antibody was a DAKO product (Dako, Denmark, Ki-67-MIB 1) while the rest were mouse monoclonal antibodies produced by Novocastra NCL, Great-Britain. To detect cytoceratin, the formalin-fixed, paraffin-embedded tissue samples were reacted with Multi-Cytokeratin antibody (NCL-AE1/AE3) diluted to 1:50. Besides the blocking of the endogen peroxidase, protease digestion was performed for 12 minute durations as a special treatment. For practical use, Ki-67 antibody was diluted to 1:20. Samples were incubated in citrate buffer for antigen retrieval at 99 oC, in a microwave oven for 30 minutes, except in the case of cytokeratin. In each sample, the duration of the antibody reaction was 30 minutes which was performed at 37 oC in vacuum. As a second step, it was followed by the avidin-biotin reaction which was performed at 37 oC in vacuum for 20 and 30 minutes respectively. For the Ki-67 assay amplification was also performed. For detecting, aminoethyl carbazol (AEC) was used as a chromogen and slides were mounted with glycerin-gelatin.

Results
During the dissection of the tortoise carcass, carapace and plastron were found to be extremely soft and flexible. The shell was easy to be pressed-in by fingers or to be slit by a lance; however, no signs of morphological malformations alluding to previous growth disorders were detected.
After removing the plastron no pathological abnormality was detected in the inner organs. The parathyroid glands located in front of the basis of the heart were enlarged, smoothly contorted. The surface of the glands was scraggly, the colour was brownish red.
The slit surfaces were moderately moist and showed a macroscopically homogenous structure. After staining with haematoxylin eosin, in the sections prepared from the parathyroid glands, it was clearly visible that the populations of the polymorphic tumour cells that has a similar appearance as the gland epithelial cells formed irregular, solid, sometimes slurring bands unlike the normal nest-like structure. The cells had a large spheric or oval shaped nucleus with a smoothly granuled chromatine and perichromasia (Figure 1) and (Figure 2). The neoplastic cells had prominent nucleoli. Dividing cells were rarely observed. The irregularly located proliferating gland epithelial cells had infiltrated the hedging connective tissue system of the parathyroid gland at several places. Besides, as a pathological phenomenon islands of squamous metaplasia were also observed (Figure 3) in the tumour tissue. The PAN-cytokeratin immunohistochemical analysis showed a positive result at the metaplasic areas (Figure 4). The PAN-cytokeratin assay was also performed on the parathyroid gland of a healthy tortoise, which resulted in a negative reaction (Figure 5). Dividing ability of tumour cells was found negative by the performed Ki-67 reaction.
The soft, easily sliceable bones of the tortoise shell have suggested the decrease, or at some point the extinction of the inorganic matrix of the bony tissue. Therefore, for the preparation of sections from the shell, decalcination of the bony tissue was not necessary. In the haematoxylin eosin stained embedded samples, the bone trabeculae were almost completely replaced by collagen fibres
(Figure 6). The increased activity of the osteoclasts was also apparent. After von Kossa silver staining, only a few small islands of calcium salts were observed in the bony tissue sections of the test (Figure 7). Virtually the whole inorganic matrix was completely missing.
In other inner organs no pathological malformations were detected during macroscopic and microscopic investigations.

Discussion
Adenocarcinoma, a rare type of parathyroid tumour with squamous metaplasia was identified causing softening of the shell of the studied spur-tighed tortoise. After transforming to tumour cells, the gland epithelial cells typically had large nuclei and prominent nucleoli. However, these cells did not show any pronounced aptitude for division. This finding was supported by the negative outcome of Ki-67 immunohistochemical probe. Due to the high activity of parathyroid hormone production in the tumorous gland epithelial cells, the bony tissue of the shell became demineralised within a short time (primary hyperparathyroidism). The flexible, rubber-like touch of the dorsal and ventral parts of the shell was caused by remarkable mobilisation of the mineral elements. The loss of the inorganic matrix from the shell was already suspected during the dissection of the animal. This supposition was confirmed by the results of von Kossa staining of the shell sections, which showed severe decrease of calcium salts.
The present study is the first report on squamous metaplasia of the parathyroid gland in reptiles. The appearance of squamous epithelial cells in the gland tissue always indicates a pathological condition. These squamous epithelial cells contain keratohyalin granules in their cytoplasm, which can be detected by PAN-cytokeratin assay. In the present study PAN-cytokeratin reaction had a positive result, proving the presence of squamous epithelial cells in the parathyroid gland. Keratohyalin granules ought not to be mistaken for the Hassel’s corpuscles of the thymus.
The tumour tissue detected in the present study showed no tendency for division, despite the enlarged nuclei in the cells and the explicit perichromasia. The tumour formed no metastases. The parathyroid adenocarcinoma consists of tumour cells with a significant ability to produce hormones. The elevated parathyroid hormone level caused by such cells activated osteoclast cells in a relatively short time. The demineralization of the shell is incidental to the increased activity of the osteoclasts.
The different assays on the parathyroid gland that are routinely used in human oncology such as Ki-67 and PAN-cytokeratin were utilizable to characterize certain features (such as dividing ability, keratohyalin accumulation) of the cells even in spur-tighed tortoise. Therefore we assume that these methods are usable to study the evolution of such tumours when vertebrate species of different evolutionary levels suffer from the same type of tumour. It is important to note, that the positive reaction of PAN-cytokeratin assay in a spur-tighed tortoise was very similar to that of in humans, despite of the phylogenetic distance between the two species. Our results raise the question, how much the same types of tumours that develop both in reptiles and in mammals differ genetically, morphologically and regarding their behaviour, and show similarities.



Figure 1.
Parathyroid gland with normal structure (control) of a healthy spur-tighed tortoise (T. graeca)

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Figure 2.
Irregularly located gland epithelium cells with large nucleoli in the parathyroid gland of a spur-tighed tortoise (T. graeca)

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Figure 3.
Squamous metaplasia island in the parathyroid gland

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Figure 4.
PAN-cytokeratin reaction in the cytoplasm of the cells in squamous metaplasia islands

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Figure 5.
PAN-cytokeratin reaction in the parathyroid gland of a healthy spur-tighed tortoise (T. graeca) (negative control)

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Figure 6.
Transformation in the tissue of the dorsal part of the shell: connective tissue formation in the place of bony tissue (osteodystrophia fibrosa). The inorganic matrix is demineralised

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Figure 7.
Von Kossa’s staining of a section from the carapace of a spur-tighed tortoise suffering from parathyroid adenocarcinom. There is calcium salt only in traces in the shell

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