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[A Case of WDHA Water Diarrhea Hypokalemia Achlorhydria Syndrome that Developed after Multimodal Therapy for Retroperitoneal Paraganglioma].
Hinyokika Kiyo 2019; 65(7):277-282HK

Abstract

A 45-year-old woman visited a local clinic with left-flank abdominal pain. Abdominal computed tomography (CT) revealed a tumor 20 cm in diameter in the left adrenal gland. She was referred to our hospital for further treatment. No endocrinological abnormality was detected on either serum or urine examination. CT and haematology findings led to a preoperative diagnosis of primary adrenal carcinoma, and we performed a left adrenalectomy. Histopathological examination revealed a paraganglioma with intact adrenal gland. Therefore we diagnosed this case as primary retroperitoneal paraganglioma. Six months after the surgery, she developed peritoneal dissemination including bilateral ovarian metastases. After cytoreductive metastasectomy, she received 131I-meta-iodobenzylguanidine (MIBG) radiotherapy. During the following five-year follow-up, MIBG radiotherapy in conjunction with cytoreductive metastasectomy (3 surgeries and 6 sessions of 131I-MIBG radiotherapy) was performed, aiming at disease control. Five years after the initial surgery, liver, lung, and intra-peritoneal dissemination progressed. Thereafter, she developed severe diarrhea, hypokalemia, and metabolic acidosis with an elevated level of vasoactive intestional peptide, which was consistent with water diarrhea, hypokalemia, achlorhydria (WDHA) syndrome. Despite intensive treatments such as with a somatostatin analogue, she died two months after the onset of this syndrome.

Authors+Show Affiliations

The Department of Urology, Hokkaido University Hospital.The Department of Urology, Hokkaido University Hospital.The Department of Urology, Hokkaido University Hospital.The Department of Urology, Hokkaido University Hospital.The Diagnostic and Therapeutic Nuclear Medicine, National Institute of Radiological Sciences.The Department of Nuclear Medicine, Hokkaido University Hospital.The Department of Surgical Pathology, Hokkaido University Hospital.The Department of Surgical Pathology, Hokkaido University Hospital.The Department of Urology, Hokkaido University Hospital.

Pub Type(s)

English Abstract
Journal Article

Language

jpn

PubMed ID

31501391

Citation

Murahashi, Norihiro, et al. "[A Case of WDHA Water Diarrhea Hypokalemia Achlorhydria Syndrome That Developed After Multimodal Therapy for Retroperitoneal Paraganglioma]." Hinyokika Kiyo. Acta Urologica Japonica, vol. 65, no. 7, 2019, pp. 277-282.
Murahashi N, Abe T, Matsumoto R, et al. [A Case of WDHA Water Diarrhea Hypokalemia Achlorhydria Syndrome that Developed after Multimodal Therapy for Retroperitoneal Paraganglioma]. Hinyokika Kiyo. 2019;65(7):277-282.
Murahashi, N., Abe, T., Matsumoto, R., Oosawa, T., Yoshinaga, K., Shiga, T., ... Shinohara, N. (2019). [A Case of WDHA Water Diarrhea Hypokalemia Achlorhydria Syndrome that Developed after Multimodal Therapy for Retroperitoneal Paraganglioma]. Hinyokika Kiyo. Acta Urologica Japonica, 65(7), pp. 277-282. doi:10.14989/ActaUrolJap_65_6_277.
Murahashi N, et al. [A Case of WDHA Water Diarrhea Hypokalemia Achlorhydria Syndrome That Developed After Multimodal Therapy for Retroperitoneal Paraganglioma]. Hinyokika Kiyo. 2019;65(7):277-282. PubMed PMID: 31501391.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - [A Case of WDHA Water Diarrhea Hypokalemia Achlorhydria Syndrome that Developed after Multimodal Therapy for Retroperitoneal Paraganglioma]. AU - Murahashi,Norihiro, AU - Abe,Takashige, AU - Matsumoto,Ryuji, AU - Oosawa,Takahiro, AU - Yoshinaga,Keiichiro, AU - Shiga,Tohru, AU - Hatanaka,KanakoC, AU - Matsuno,Yoshihiro, AU - Shinohara,Nobuo, PY - 2019/9/11/entrez SP - 277 EP - 282 JF - Hinyokika kiyo. Acta urologica Japonica JO - Hinyokika Kiyo VL - 65 IS - 7 N2 - A 45-year-old woman visited a local clinic with left-flank abdominal pain. Abdominal computed tomography (CT) revealed a tumor 20 cm in diameter in the left adrenal gland. She was referred to our hospital for further treatment. No endocrinological abnormality was detected on either serum or urine examination. CT and haematology findings led to a preoperative diagnosis of primary adrenal carcinoma, and we performed a left adrenalectomy. Histopathological examination revealed a paraganglioma with intact adrenal gland. Therefore we diagnosed this case as primary retroperitoneal paraganglioma. Six months after the surgery, she developed peritoneal dissemination including bilateral ovarian metastases. After cytoreductive metastasectomy, she received 131I-meta-iodobenzylguanidine (MIBG) radiotherapy. During the following five-year follow-up, MIBG radiotherapy in conjunction with cytoreductive metastasectomy (3 surgeries and 6 sessions of 131I-MIBG radiotherapy) was performed, aiming at disease control. Five years after the initial surgery, liver, lung, and intra-peritoneal dissemination progressed. Thereafter, she developed severe diarrhea, hypokalemia, and metabolic acidosis with an elevated level of vasoactive intestional peptide, which was consistent with water diarrhea, hypokalemia, achlorhydria (WDHA) syndrome. Despite intensive treatments such as with a somatostatin analogue, she died two months after the onset of this syndrome. SN - 0018-1994 UR - https://www.unboundmedicine.com/medline/citation/31501391/[A_Case_of_WDHA_Water_Diarrhea_Hypokalemia_Achlorhydria_Syndrome_that_Developed_after_Multimodal_Therapy_for_Retroperitoneal_Paraganglioma] DB - PRIME DP - Unbound Medicine ER -