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Alveolar Rhabdomyosarcoma in a 69-Year-Old Woman Receiving Glucagon-Like Peptide-2 Therapy.

Zyczynski LE, McHugh JB, Gribbin TE, Schuetze SM - Case Rep Oncol Med (2015)

Bottom Line: Though a causative relationship is unlikely between the peptide and ARMS due to the brief time course between teduglutide therapy and sarcoma diagnosis, neoplastic growth may have been accelerated by the GLP-2 analog, causing release of IGF-1.The transmembrane receptor for IGF-1 is frequently overexpressed in ARMS and is implicated in cell proliferation and metastatic behavior.This case describes a rare incidence of metastatic alveolar rhabdomyosarcoma in a sexagenarian and possibly the first case reported associated with the use of teduglutide.

View Article: PubMed Central - PubMed

Affiliation: Department of Internal Medicine, University of Michigan Health System, 1500 E. Medical Center Drive, Ann Arbor, MI 48109, USA.

ABSTRACT
A 69-year-old woman was diagnosed with alveolar rhabdomyosarcoma (ARMS) of the nasopharynx. She has a history of catastrophic thromboembolic event in the abdomen that caused short-gut syndrome and dependence on total parenteral nutrition (TPN) twelve hours per day. She was treated for short-gut syndrome with teduglutide, a glucagon-like peptide-2 (GLP-2) analog, which led to reduction of TPN requirements. However, a few months later, she developed metastatic alveolar rhabdomyosarcoma. Though a causative relationship is unlikely between the peptide and ARMS due to the brief time course between teduglutide therapy and sarcoma diagnosis, neoplastic growth may have been accelerated by the GLP-2 analog, causing release of IGF-1. The transmembrane receptor for IGF-1 is frequently overexpressed in ARMS and is implicated in cell proliferation and metastatic behavior. This case describes a rare incidence of metastatic alveolar rhabdomyosarcoma in a sexagenarian and possibly the first case reported associated with the use of teduglutide. Teduglutide was discontinued due to a potential theoretical risk of acceleration of sarcoma growth, and the patient's rhabdomyosarcoma is in remission following sarcoma chemotherapy.

No MeSH data available.


Related in: MedlinePlus

Photomicrographs of diagnostic biopsy demonstrating rhabdomyosarcoma. (a) 40x magnification of hematoxylin- and eosin-stained section demonstrating diffuse small round cells with a high nuclear to cytoplasmic ratio. (b) 40x magnification of horseradish peroxidase immunohistochemical stain for IGF1-R demonstrating uniform expression. (c) 40x magnification of horseradish peroxidase immunohistochemical stain for desmin demonstrating cytoplasmic expression. (d) 40x magnification of horseradish peroxidase immunohistochemical stain for myogenin demonstrating nuclear localization.
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fig1: Photomicrographs of diagnostic biopsy demonstrating rhabdomyosarcoma. (a) 40x magnification of hematoxylin- and eosin-stained section demonstrating diffuse small round cells with a high nuclear to cytoplasmic ratio. (b) 40x magnification of horseradish peroxidase immunohistochemical stain for IGF1-R demonstrating uniform expression. (c) 40x magnification of horseradish peroxidase immunohistochemical stain for desmin demonstrating cytoplasmic expression. (d) 40x magnification of horseradish peroxidase immunohistochemical stain for myogenin demonstrating nuclear localization.

Mentions: An excisional biopsy of the nasopharyngeal mass was performed. Pathology was consistent with ARMS. Immunohistochemical staining was notable for diffuse and strong positivity for CD56, vimentin, desmin, and myogenin (see Figure 1). There was patchy positivity for S-100. Tumor was negative for melan-A, HMB45, and cytokeratin cocktail. An antibody to the insulin-like growth factor-1 receptor reacted with tumor cells. The patient had a fine needle aspiration biopsy performed on the left neck and left flank masses, both of which were positive for metastatic ARMS. A portion of the nasopharyngeal mass biopsy was sent for reverse transcriptase-polymerase chain reaction (RT-PCR) for PAX3/FOX01 translocation chimeric transcript, and this was present, confirming the diagnosis.


Alveolar Rhabdomyosarcoma in a 69-Year-Old Woman Receiving Glucagon-Like Peptide-2 Therapy.

Zyczynski LE, McHugh JB, Gribbin TE, Schuetze SM - Case Rep Oncol Med (2015)

Photomicrographs of diagnostic biopsy demonstrating rhabdomyosarcoma. (a) 40x magnification of hematoxylin- and eosin-stained section demonstrating diffuse small round cells with a high nuclear to cytoplasmic ratio. (b) 40x magnification of horseradish peroxidase immunohistochemical stain for IGF1-R demonstrating uniform expression. (c) 40x magnification of horseradish peroxidase immunohistochemical stain for desmin demonstrating cytoplasmic expression. (d) 40x magnification of horseradish peroxidase immunohistochemical stain for myogenin demonstrating nuclear localization.
© Copyright Policy - open-access
Related In: Results  -  Collection

Show All Figures
getmorefigures.php?uid=PMC4525456&req=5

fig1: Photomicrographs of diagnostic biopsy demonstrating rhabdomyosarcoma. (a) 40x magnification of hematoxylin- and eosin-stained section demonstrating diffuse small round cells with a high nuclear to cytoplasmic ratio. (b) 40x magnification of horseradish peroxidase immunohistochemical stain for IGF1-R demonstrating uniform expression. (c) 40x magnification of horseradish peroxidase immunohistochemical stain for desmin demonstrating cytoplasmic expression. (d) 40x magnification of horseradish peroxidase immunohistochemical stain for myogenin demonstrating nuclear localization.
Mentions: An excisional biopsy of the nasopharyngeal mass was performed. Pathology was consistent with ARMS. Immunohistochemical staining was notable for diffuse and strong positivity for CD56, vimentin, desmin, and myogenin (see Figure 1). There was patchy positivity for S-100. Tumor was negative for melan-A, HMB45, and cytokeratin cocktail. An antibody to the insulin-like growth factor-1 receptor reacted with tumor cells. The patient had a fine needle aspiration biopsy performed on the left neck and left flank masses, both of which were positive for metastatic ARMS. A portion of the nasopharyngeal mass biopsy was sent for reverse transcriptase-polymerase chain reaction (RT-PCR) for PAX3/FOX01 translocation chimeric transcript, and this was present, confirming the diagnosis.

Bottom Line: Though a causative relationship is unlikely between the peptide and ARMS due to the brief time course between teduglutide therapy and sarcoma diagnosis, neoplastic growth may have been accelerated by the GLP-2 analog, causing release of IGF-1.The transmembrane receptor for IGF-1 is frequently overexpressed in ARMS and is implicated in cell proliferation and metastatic behavior.This case describes a rare incidence of metastatic alveolar rhabdomyosarcoma in a sexagenarian and possibly the first case reported associated with the use of teduglutide.

View Article: PubMed Central - PubMed

Affiliation: Department of Internal Medicine, University of Michigan Health System, 1500 E. Medical Center Drive, Ann Arbor, MI 48109, USA.

ABSTRACT
A 69-year-old woman was diagnosed with alveolar rhabdomyosarcoma (ARMS) of the nasopharynx. She has a history of catastrophic thromboembolic event in the abdomen that caused short-gut syndrome and dependence on total parenteral nutrition (TPN) twelve hours per day. She was treated for short-gut syndrome with teduglutide, a glucagon-like peptide-2 (GLP-2) analog, which led to reduction of TPN requirements. However, a few months later, she developed metastatic alveolar rhabdomyosarcoma. Though a causative relationship is unlikely between the peptide and ARMS due to the brief time course between teduglutide therapy and sarcoma diagnosis, neoplastic growth may have been accelerated by the GLP-2 analog, causing release of IGF-1. The transmembrane receptor for IGF-1 is frequently overexpressed in ARMS and is implicated in cell proliferation and metastatic behavior. This case describes a rare incidence of metastatic alveolar rhabdomyosarcoma in a sexagenarian and possibly the first case reported associated with the use of teduglutide. Teduglutide was discontinued due to a potential theoretical risk of acceleration of sarcoma growth, and the patient's rhabdomyosarcoma is in remission following sarcoma chemotherapy.

No MeSH data available.


Related in: MedlinePlus