Hypokalemic Rhabdomyolysis due to WDHA Syndrome Caused by VIP-producing Composite Pheochromocytoma: A Case in Neurofibromatosis Type 1
A 47-year-old woman with neurofibromatosis type 1 suffered from general muscle weakness and watery diarrhea. Laboratory findings showed elevated muscular enzymes, severe hypokalemia and excessive production of catecholamines and vasoactive intestinal polypeptide (VIP). A computed tomography scan sho...
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| Veröffentlicht in: | Japanese journal of clinical oncology 2005-09, Vol.35 (9), p.559-563 |
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| container_title | Japanese journal of clinical oncology |
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| creator | Onozawa, Masahiro Fukuhara, Takashi Minoguchi, Madoka Takahata, Mutsumi Yamamoto, Yasushi Miyake, Takayoshi Kanagawa, Koichi Kanda, Makoto Maekawa, Isao |
| description | A 47-year-old woman with neurofibromatosis type 1 suffered from general muscle weakness and watery diarrhea. Laboratory findings showed elevated muscular enzymes, severe hypokalemia and excessive production of catecholamines and vasoactive intestinal polypeptide (VIP). A computed tomography scan showed a 10 cm left adrenal mass, in which [131I]-metaiodobenzylguanidine scintigraphy showed high uptake. After she underwent surgical removal of the tumor, all the symptoms and signs subsided. A histological study revealed that the mass consisted of pheochromocytoma and ganglioneuroma, respectively producing catecholamines and VIP. In immunohistochemical staining of neurofibromin, pheochromocytoma and ganglion cells showed positive staining, whereas the staining was negative for nerve bundles and Schwann cells. We concluded that the patient had hypokalemic rhabdomyolysis due to watery diarrhea, hypokalemia and achlorhydria (WDHA) syndrome, which was induced by a VIP-producing composite pheochromocytoma. Composite pheochromocytoma is a neuroendocrine tumor that is composed of pheochromocytoma and ganglioneuroma, both derived from the neural crest. Deficiency of neurofibromin in Schwann cells might have played an important role in the development and the growth of the composite pheochromocytoma in this patient. |
| doi_str_mv | 10.1093/jjco/hyi139 |
| format | Article |
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Laboratory findings showed elevated muscular enzymes, severe hypokalemia and excessive production of catecholamines and vasoactive intestinal polypeptide (VIP). A computed tomography scan showed a 10 cm left adrenal mass, in which [131I]-metaiodobenzylguanidine scintigraphy showed high uptake. After she underwent surgical removal of the tumor, all the symptoms and signs subsided. A histological study revealed that the mass consisted of pheochromocytoma and ganglioneuroma, respectively producing catecholamines and VIP. In immunohistochemical staining of neurofibromin, pheochromocytoma and ganglion cells showed positive staining, whereas the staining was negative for nerve bundles and Schwann cells. We concluded that the patient had hypokalemic rhabdomyolysis due to watery diarrhea, hypokalemia and achlorhydria (WDHA) syndrome, which was induced by a VIP-producing composite pheochromocytoma. Composite pheochromocytoma is a neuroendocrine tumor that is composed of pheochromocytoma and ganglioneuroma, both derived from the neural crest. Deficiency of neurofibromin in Schwann cells might have played an important role in the development and the growth of the composite pheochromocytoma in this patient.</description><identifier>ISSN: 0368-2811</identifier><identifier>EISSN: 1465-3621</identifier><identifier>DOI: 10.1093/jjco/hyi139</identifier><identifier>PMID: 16027147</identifier><language>eng</language><publisher>England: Oxford University Press</publisher><subject>3-Iodobenzylguanidine ; Achlorhydria - etiology ; Adrenal Gland Neoplasms - complications ; Adrenal Gland Neoplasms - diagnostic imaging ; Adrenal Gland Neoplasms - metabolism ; Adrenal Gland Neoplasms - surgery ; composite pheochromocytoma ; Diarrhea - etiology ; Female ; Humans ; Hypokalemia - etiology ; hypokalemia and achlorhydria syndrome ; hypokalemic rhabdomyolysis ; Middle Aged ; Neurofibromatosis 1 - complications ; neurofibromin ; Pheochromocytoma - complications ; Pheochromocytoma - diagnostic imaging ; Pheochromocytoma - metabolism ; Pheochromocytoma - surgery ; Radionuclide Imaging ; Radiopharmaceuticals ; Rhabdomyolysis - etiology ; Syndrome ; Tomography, X-Ray Computed ; Vasoactive Intestinal Peptide - metabolism ; vasoactive intestinal polypeptide ; watery diarrhea</subject><ispartof>Japanese journal of clinical oncology, 2005-09, Vol.35 (9), p.559-563</ispartof><rights>Copyright Oxford University Press(England) Sep 15, 2005</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c488t-581e8a384d351a4ccf7488ab6082e98a21dd41de9ee00a1ccba6e4c84450269a3</citedby><cites>FETCH-LOGICAL-c488t-581e8a384d351a4ccf7488ab6082e98a21dd41de9ee00a1ccba6e4c84450269a3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,780,784,27924,27925</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/16027147$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Onozawa, Masahiro</creatorcontrib><creatorcontrib>Fukuhara, Takashi</creatorcontrib><creatorcontrib>Minoguchi, Madoka</creatorcontrib><creatorcontrib>Takahata, Mutsumi</creatorcontrib><creatorcontrib>Yamamoto, Yasushi</creatorcontrib><creatorcontrib>Miyake, Takayoshi</creatorcontrib><creatorcontrib>Kanagawa, Koichi</creatorcontrib><creatorcontrib>Kanda, Makoto</creatorcontrib><creatorcontrib>Maekawa, Isao</creatorcontrib><title>Hypokalemic Rhabdomyolysis due to WDHA Syndrome Caused by VIP-producing Composite Pheochromocytoma: A Case in Neurofibromatosis Type 1</title><title>Japanese journal of clinical oncology</title><addtitle>JJCO</addtitle><description>A 47-year-old woman with neurofibromatosis type 1 suffered from general muscle weakness and watery diarrhea. Laboratory findings showed elevated muscular enzymes, severe hypokalemia and excessive production of catecholamines and vasoactive intestinal polypeptide (VIP). A computed tomography scan showed a 10 cm left adrenal mass, in which [131I]-metaiodobenzylguanidine scintigraphy showed high uptake. After she underwent surgical removal of the tumor, all the symptoms and signs subsided. A histological study revealed that the mass consisted of pheochromocytoma and ganglioneuroma, respectively producing catecholamines and VIP. In immunohistochemical staining of neurofibromin, pheochromocytoma and ganglion cells showed positive staining, whereas the staining was negative for nerve bundles and Schwann cells. We concluded that the patient had hypokalemic rhabdomyolysis due to watery diarrhea, hypokalemia and achlorhydria (WDHA) syndrome, which was induced by a VIP-producing composite pheochromocytoma. Composite pheochromocytoma is a neuroendocrine tumor that is composed of pheochromocytoma and ganglioneuroma, both derived from the neural crest. Deficiency of neurofibromin in Schwann cells might have played an important role in the development and the growth of the composite pheochromocytoma in this patient.</description><subject>3-Iodobenzylguanidine</subject><subject>Achlorhydria - etiology</subject><subject>Adrenal Gland Neoplasms - complications</subject><subject>Adrenal Gland Neoplasms - diagnostic imaging</subject><subject>Adrenal Gland Neoplasms - metabolism</subject><subject>Adrenal Gland Neoplasms - surgery</subject><subject>composite pheochromocytoma</subject><subject>Diarrhea - etiology</subject><subject>Female</subject><subject>Humans</subject><subject>Hypokalemia - etiology</subject><subject>hypokalemia and achlorhydria syndrome</subject><subject>hypokalemic rhabdomyolysis</subject><subject>Middle Aged</subject><subject>Neurofibromatosis 1 - complications</subject><subject>neurofibromin</subject><subject>Pheochromocytoma - complications</subject><subject>Pheochromocytoma - diagnostic imaging</subject><subject>Pheochromocytoma - metabolism</subject><subject>Pheochromocytoma - surgery</subject><subject>Radionuclide Imaging</subject><subject>Radiopharmaceuticals</subject><subject>Rhabdomyolysis - etiology</subject><subject>Syndrome</subject><subject>Tomography, X-Ray Computed</subject><subject>Vasoactive Intestinal Peptide - metabolism</subject><subject>vasoactive intestinal polypeptide</subject><subject>watery diarrhea</subject><issn>0368-2811</issn><issn>1465-3621</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2005</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqF0c-L1DAUB_AgijuunrxL8LAXqZs0aZJ6G-qPWRjWQddd8RLS5I2T2bapTQv2H_DvNsMMCl48Bd77vJeEL0LPKXlNScku93sbLnezp6x8gBaUiyJjIqcP0YIwobJcUXqGnsS4J4QUisvH6IwKkkvK5QL9Ws19uDcNtN7iTztTu9DOoZmjj9hNgMeA796ulvjz3LkhtIArM0VwuJ7x7dUm64fgJuu777gKbR-iHwFvdhDsLuFg5zG05g1epqkI2Hf4GqYhbH2dumYMh0tu5h4wfYoebU0T4dnpPEdf3r-7qVbZ-uOHq2q5zixXaswKRUEZprhjBTXc2q1MdVMLonIolcmpc5w6KAEIMdTa2gjgVnFekFyUhp2ji-Pe9PAfE8RRtz5aaBrTQZiiFkpwISX5L6Qlp0SQIsGX_8B9mIYufULnVFLCy5wl9OqI7BBiHGCr-8G3Zpg1JfoQoj6EqI8hJv3itHKqW3B_7Sm1BLIj8HGEn3_6ZrjXQjJZ6NXXb3pzK_O760rqNfsNC4GpCg</recordid><startdate>200509</startdate><enddate>200509</enddate><creator>Onozawa, Masahiro</creator><creator>Fukuhara, Takashi</creator><creator>Minoguchi, Madoka</creator><creator>Takahata, Mutsumi</creator><creator>Yamamoto, Yasushi</creator><creator>Miyake, Takayoshi</creator><creator>Kanagawa, Koichi</creator><creator>Kanda, Makoto</creator><creator>Maekawa, Isao</creator><general>Oxford University Press</general><general>Oxford Publishing Limited (England)</general><scope>BSCLL</scope><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7T5</scope><scope>7T7</scope><scope>7TM</scope><scope>7TO</scope><scope>7U9</scope><scope>8FD</scope><scope>C1K</scope><scope>FR3</scope><scope>H94</scope><scope>K9.</scope><scope>P64</scope><scope>7TK</scope><scope>7X8</scope></search><sort><creationdate>200509</creationdate><title>Hypokalemic Rhabdomyolysis due to WDHA Syndrome Caused by VIP-producing Composite Pheochromocytoma: A Case in Neurofibromatosis Type 1</title><author>Onozawa, Masahiro ; 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Laboratory findings showed elevated muscular enzymes, severe hypokalemia and excessive production of catecholamines and vasoactive intestinal polypeptide (VIP). A computed tomography scan showed a 10 cm left adrenal mass, in which [131I]-metaiodobenzylguanidine scintigraphy showed high uptake. After she underwent surgical removal of the tumor, all the symptoms and signs subsided. A histological study revealed that the mass consisted of pheochromocytoma and ganglioneuroma, respectively producing catecholamines and VIP. In immunohistochemical staining of neurofibromin, pheochromocytoma and ganglion cells showed positive staining, whereas the staining was negative for nerve bundles and Schwann cells. We concluded that the patient had hypokalemic rhabdomyolysis due to watery diarrhea, hypokalemia and achlorhydria (WDHA) syndrome, which was induced by a VIP-producing composite pheochromocytoma. Composite pheochromocytoma is a neuroendocrine tumor that is composed of pheochromocytoma and ganglioneuroma, both derived from the neural crest. Deficiency of neurofibromin in Schwann cells might have played an important role in the development and the growth of the composite pheochromocytoma in this patient.</abstract><cop>England</cop><pub>Oxford University Press</pub><pmid>16027147</pmid><doi>10.1093/jjco/hyi139</doi><tpages>5</tpages><oa>free_for_read</oa></addata></record> |
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| ispartof | Japanese journal of clinical oncology, 2005-09, Vol.35 (9), p.559-563 |
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| source | MEDLINE; Elektronische Zeitschriftenbibliothek - Frei zugängliche E-Journals; Oxford University Press Journals All Titles (1996-Current); Freely Accessible Japanese Titles |
| subjects | 3-Iodobenzylguanidine Achlorhydria - etiology Adrenal Gland Neoplasms - complications Adrenal Gland Neoplasms - diagnostic imaging Adrenal Gland Neoplasms - metabolism Adrenal Gland Neoplasms - surgery composite pheochromocytoma Diarrhea - etiology Female Humans Hypokalemia - etiology hypokalemia and achlorhydria syndrome hypokalemic rhabdomyolysis Middle Aged Neurofibromatosis 1 - complications neurofibromin Pheochromocytoma - complications Pheochromocytoma - diagnostic imaging Pheochromocytoma - metabolism Pheochromocytoma - surgery Radionuclide Imaging Radiopharmaceuticals Rhabdomyolysis - etiology Syndrome Tomography, X-Ray Computed Vasoactive Intestinal Peptide - metabolism vasoactive intestinal polypeptide watery diarrhea |
| title | Hypokalemic Rhabdomyolysis due to WDHA Syndrome Caused by VIP-producing Composite Pheochromocytoma: A Case in Neurofibromatosis Type 1 |
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