Thyrotropin-secreting pituitary adenomas (TSHomas) are a rare cause of hyperthyroidism and account for less than 2% of pituitary adenomas. Medical therapy with somatostatin analogues (SSAs) effectively reduces TSH secretion in approximately 80% of patients and induces shrinkage in about 45% of tumors. According with previous data, resistance to SSA treatment might be due to heterogeneity in somatostatin receptors (SSTRs) expression. We report the case of TSHoma in a 41-year-old man treated with octreotide LAR that caused a dramatic decrease of TSH and thyroid hormones and tumor shrinkage already after 3 months of pre-surgical therapy. In search of potential molecular determinants of octreotide effectiveness, we measured, in primary cultures from this tumor, SSTR and dopamine D2 receptor (D2R) expression, and octreotide and/or cabergoline effects on TSH secretion and cell proliferation. SSTR5 and D2R expression was higher than SSTR2. Octreotide significantly inhibited TSH secretion more effectively than cabergoline (P<0.001), whereas the combined treatment was comparable with cabergoline alone. Similarly, octreotide resulted more effective than cabergoline on cell proliferation, while the combination did not show any additive or synergistic effects. In conclusion, the significant antisecretive and antiproliferative effect of octreotide in this patient might be related to the high expression of SSTR5, in the presence of SSTR2. After reviewing the literature, indeed, in line with previous observations, we hypothesize that SSTR5/SSTR2 ratio in TSHomas may represent a useful marker in predicting the outcome of therapy with SSAs. The role of D2R should be further explored considering that the presence of D2R can influence SSTRs functionality.
Pituicytomas are often difficult to distinguish from other hypothalamic or pituitary lesions. However, their identification would be preferable in a pre-operative setting in order to optimize the work-up and to initiate a proactive management of the expected complications.
Intravascular lymphoma (IVL) is a rare subtype of extranodal lymphomas that is characterized by the selective growth of neoplastic cells within the lumen of small vessels. Authors document the case of an unexpected death caused by an undiagnosed intravascular large B‐cell lymphoma with multi‐organ involvement, which had initially manifested as an infection and then as an unclarified central nervous system pathology. Histological examination showed a diffuse intravascular large B‐cell brain lymphoma with prominent cerebral involvement. The relevance of the case report reveals the importance of an autopsy of an extremely rare and threatening pathology that in most cases is diagnosed only postmortem. As a result, the role of the forensic pathologist becomes particularly important. When specifically performing an in‐depth autopsy evaluation with a specific histologic analysis, it is possible to identify the intravascular lymphoma and declare a more accurate cause of death.
IntroductionMyositis ossificans traumatica is a pathological condition characterized by the extraskeletal formation of bony tissue, induced by violent or repeated trauma.Case presentationA 30-year-old Italian man, after surgical treatment for multiple facial fractures, presented with a progressive limitation of mouth opening. A computed tomography scan showed a significant calcification of the fibers of the left lateral pterygoid muscle. The working diagnosis was myositis ossificans traumatica of the left lateral pterygoid muscle. Surgical excision was suggested but not performed. Our patient underwent physiotherapy treatment resulting not in a complete restoration of mandibular movements but in an acceptable recovery of mouth opening.ConclusionsMyositis ossificans is a rare complication that can be caused by muscle trauma. Therefore, special attention should be paid to surgical trauma. In the present case, surgical excision was considered, in accordance with the literature, and suggested to our patient, but he declined due to the absence of any pain or any significant limitation to his daily life activities. He therefore underwent physiotherapy treatment, in line with our unit’s guidelines, resulting not in a complete restoration of mandibular movements but in an acceptable recovery of mouth opening.
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