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Nakanoh H, Tsuji K, Morimoto S, Fukushima K, Iwamuro M, Uchida HA, Wada J. A case of membranous nephropathy complicated by Cronkhite-Canada syndrome successfully treated with mizoribine. CEN Case Rep 2025; 14:72-80. [PMID: 38954395 PMCID: PMC11785862 DOI: 10.1007/s13730-024-00908-9] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/29/2024] [Accepted: 06/21/2024] [Indexed: 07/04/2024] Open
Abstract
Cronkhite-Canada syndrome (CCS) is a non-hereditary disorder characterized by non-neoplastic hamartomatous gastrointestinal polyposis, hair loss, nail atrophy, hyperpigmentation, and diarrhea. While the relationship between CCS and nephritis remains unclear, seven cases of nephritis complicated by CCS have been reported to date, all of which were membranous nephropathy (MN). A 57-year-old man presented with taste disturbance, hair loss, nail plate atrophy, skin pigmentation, and frequent diarrhea. Endoscopic findings showed multiple polyposis of the stomach and large intestine. Given the above, he was diagnosed with CCS. The symptoms gradually improved with prednisolone treatment, although urinary protein and hypoproteinemia appeared during the tapering of prednisolone. He was diagnosed with MN using a renal biopsy, and immunofluorescence microscopy with IgG subclass staining showed predominantly diffuse granular capillary wall staining of IgG4. The cause of secondary MN was not found, including malignant tumors. Nephrotic-range proteinuria persisted despite treatment with prednisolone and cyclosporine. Additional treatment with mizoribine resulted in incomplete remission type 1 of nephrotic syndrome, suggesting that mizoribine may be a treatment option for patients with CCS with steroid-resistant MN. Considering a high prevalence of hypoproteinemia due to chronic diarrhea and protein-losing enteropathy in patients with CCS, proteinuria might be overlooked; thus, follow-up urinalysis would be recommended in patients with CCS.
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Affiliation(s)
- Hiroyuki Nakanoh
- Department of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, 2-5-1 Shikata-Cho, Okayama, 700-8558, Japan
| | - Kenji Tsuji
- Department of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, 2-5-1 Shikata-Cho, Okayama, 700-8558, Japan.
| | - Shiho Morimoto
- Department of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, 2-5-1 Shikata-Cho, Okayama, 700-8558, Japan
| | - Kazuhiko Fukushima
- Department of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, 2-5-1 Shikata-Cho, Okayama, 700-8558, Japan
| | - Masaya Iwamuro
- Department of Gastroenterology and Hepatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama, Japan
| | - Haruhito A Uchida
- Department of Chronic Kidney Disease and Cardiovascular Disease, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama, Japan
| | - Jun Wada
- Department of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, 2-5-1 Shikata-Cho, Okayama, 700-8558, Japan
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Shimoyama M, Iwagami H, Minaga K, Akamatsu T, Uenoyama Y, Yamashita Y. Cronkhite-Canada syndrome tends to be accompanied by colorectal cancer: Report of seven cases. JGH Open 2024; 8:e70032. [PMID: 39364060 PMCID: PMC11446959 DOI: 10.1002/jgh3.70032] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/04/2024] [Revised: 09/10/2024] [Accepted: 09/16/2024] [Indexed: 10/05/2024]
Abstract
Cronkhite-Canada syndrome (CCS) can be difficult to diagnose. To diagnose CCS, it is important to perform endoscopic examination for patients with chronic diarrhea, check for the presence or absence of polyposis, and evaluate inflammation in the mucosa between the polyps. This study reported seven cases of CCS. The age of the patients, which included four men and three women, ranged 48-72 years, and all patients were Asian. The most common symptom among these patients was chronic diarrhea. Three of the patients had rectal cancer. In two patients, the lesions were detected at an early stage and resected via endoscopic treatment. CCS is associated with a high risk of malignant gastrointestinal lesions, especially rectal cancers, and periodic surveillance endoscopy and careful observation are required.
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Affiliation(s)
- Masayuki Shimoyama
- Department of Gastroenterology and Hepatology Japanese Red Cross Wakayama Medical Center Wakayama Japan
| | - Hiroyoshi Iwagami
- Department of Gastroenterology and Hepatology Japanese Red Cross Wakayama Medical Center Wakayama Japan
| | - Kosuke Minaga
- Department of Gastroenterology and Hepatology Japanese Red Cross Wakayama Medical Center Wakayama Japan
| | - Takuji Akamatsu
- Department of Gastroenterology and Hepatology Japanese Red Cross Wakayama Medical Center Wakayama Japan
| | - Yoshito Uenoyama
- Department of Gastroenterology and Hepatology Japanese Red Cross Wakayama Medical Center Wakayama Japan
| | - Yukitaka Yamashita
- Department of Gastroenterology and Hepatology Japanese Red Cross Wakayama Medical Center Wakayama Japan
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He ML, Zheng Y, Tian SX. Cronkhite-Canada syndrome complicated with pulmonary embolism: A case report. World J Clin Cases 2024; 12:4820-4826. [PMID: 39070830 PMCID: PMC11235495 DOI: 10.12998/wjcc.v12.i21.4820] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/17/2024] [Revised: 05/23/2024] [Accepted: 06/11/2024] [Indexed: 06/30/2024] Open
Abstract
BACKGROUND Cronkhite-Canada syndrome (CCS) is a rare disease, that causes gastrointestinal polyps, ectodermal abnormalities, and gastrointestinal symptoms. CCS is prone to thromboembolism, but clinical workers have not yet established a clinical consciousness of preventing thromboembolism. The present case illustrates pulmonary embolism (PE) complicated by CCS. CASE SUMMARY A 46-year-old male patient presented with mucus, purulent, and bloody stool. Ectodermal changes included skin pigmentation, alopecia, and nail dystrophy. Colonoscopy revealed the presence of multiple polyps. After a comprehensive evaluation, the patient was diagnosed with CCS. During the disease, he was also diagnosed with pulmonary embolism, Riehl's melanosis, and intestinal flora imbalance. After symptomatic treatment with omeprazole, mesalazine, rivaroxaban, nutritional support, and regulation of intestinal flora, the patient's symptoms were significantly relieved. CONCLUSION CCS complicated with PE was reported for the first time in China in this study. Despite the fact that CCS is extremely rare, patients with CCS should be classified as a high-risk venous thromboembolism (VTE) population, and emphasis should be placed on venous thromboembolism risk assessment and stratification, deep venous thromboembolism screening, prevention of VTE, and careful long-term follow-up.
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Affiliation(s)
- Mao-Lang He
- College of Medicine, Shihezi University, Shihezi 832099, Xinjiang Uygur Autonomous Region, China
| | - Yong Zheng
- Department of Gastroenterology, The First Affiliated Hospital of Shihezi University, Shihezi 832008, Xinjiang Uygur Autonomous Region, China
| | - Shu-Xin Tian
- Department of Gastroenterology, The First Affiliated Hospital of Shihezi University, Shihezi 832008, Xinjiang Uygur Autonomous Region, China
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Lv YQ, Wang ML, Tang TY, Li YQ. Comprehensive treatment and a rare presentation of Cronkhite-Canada syndrome: Two case reports and review of literature. World J Gastrointest Surg 2023; 15:2646-2656. [PMID: 38111781 PMCID: PMC10725548 DOI: 10.4240/wjgs.v15.i11.2646] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/15/2023] [Revised: 09/15/2023] [Accepted: 10/17/2023] [Indexed: 11/26/2023] Open
Abstract
BACKGROUND Cronkhite-Canada syndrome (CCS) is a rare sporadic polyposis syndrome that presents with gastrointestinal and ectodermal symptoms in addition to nutritional deficiencies. CCS combined with hypothyroidism is an even rarer condition, with no standard treatment guidelines. CASE SUMMARY The present study described 2 patients with CCS: A 67-year-old woman with concomitant hypothyroidism and 68-year-old man treated with endoscopic mucosal resection (EMR). Both patients had multiple gastrointestinal symptoms and ectodermal changes, along with multiple gastrointestinal polyps. Microscopic examination showed that the mucosa in both patients was hyperemic and edematous, with pathologic examination showing distorted, atrophic, and dilated glands. Patient 1 had concomitant hypothyroidism and was treated with levothyroxine. Due to her self-reduction of hormone dose, her disease relapsed. Patient 2 underwent EMR, but refused further hormonal or biological treatments. Subsequently, he was treated with an oral Chinese medical preparation. CONCLUSION Pharmacotherapy can induce and maintain remission in CCS patients, with adjuvant EMR, long-term follow-up, and endoscopic surveillance being necessary.
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Affiliation(s)
- Yan-Qing Lv
- Department of Hepatobiliary and Pancreatic Medicine, The First Hospital of Jilin University, Changchun 130021, Jilin Province, China
| | - Mei-Lan Wang
- Department of Gastroenterology, Jilin Provincial People's Hospital, Changchun 130021, Jilin Province, China
| | - Tong-Yu Tang
- Department of Gastroenterology, The First Hospital of Jilin University, Changchun 130021, Jilin Province, China
| | - Yu-Qin Li
- Department of Gastroenterology, The First Hospital of Jilin University, Changchun 130021, Jilin Province, China
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Matsumoto Y, Koyama F, Morita K, Kuge H, Obara S, Iwasa Y, Takei T, Sadamitsu T, Ohbayashi C, Sho M. A case of Cronkhite–Canada syndrome with repeated linked color imaging observation of the subepithelial capillary network in the colon. Clin J Gastroenterol 2022; 15:934-940. [DOI: 10.1007/s12328-022-01678-6] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/15/2022] [Accepted: 07/12/2022] [Indexed: 02/07/2023]
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Wu ZY, Sang LX, Chang B. Cronkhite-Canada syndrome: from clinical features to treatment. Gastroenterol Rep (Oxf) 2020; 8:333-342. [PMID: 33163187 PMCID: PMC7603875 DOI: 10.1093/gastro/goaa058] [Citation(s) in RCA: 17] [Impact Index Per Article: 3.4] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/30/2019] [Revised: 03/04/2020] [Accepted: 04/16/2020] [Indexed: 02/07/2023] Open
Abstract
Cronkhite–Canada syndrome (CCS) is a rare acquired polyposis with unknown etiology. To date, >500 cases have been reported worldwide. CCS is typically characterized by gastrointestinal symptoms, such as diarrhea and skin changes (e.g. alopecia, pigmentation, and nail atrophy). Endoscopic features include diffuse polyps throughout the entire gastrointestinal tract, except for the esophagus. Pathological types of polyps in CCS mainly include inflammatory, hyperplastic, hamartomatous, and adenomatous polyps. CCS can be complicated by many diseases and has a canceration tendency with a high mortality rate. Moreover, there is no uniform standard treatment for CCS. A review of the reported cases of CCS is presented herein, with the goal of improving our understanding of this disease.
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Affiliation(s)
- Ze-Yu Wu
- Department of Gastroenterology, First Hospital of China Medical University, Shenyang, Liaoning, P. R. China
| | - Li-Xuan Sang
- Department of Geriatrics, First Hospital of China Medical University, Shenyang, Liaoning, P. R. China
| | - Bing Chang
- Corresponding author. Department of Gastroenterology, First Affiliated Hospital of China Medical University, No. 155, Nanjing North Street, Shenyang, Liaoning 110001, P. R. China. Tel: +86-24-83283765; Fax: +86-24-83283764;
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Successful surgical treatment of Cronkhite-Canada Syndrome with bilateral flail chest: a case report. BMC Surg 2020; 20:103. [PMID: 32404137 PMCID: PMC7222535 DOI: 10.1186/s12893-020-00766-z] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/13/2019] [Accepted: 05/04/2020] [Indexed: 11/10/2022] Open
Abstract
Background Development of multiple rib fractures leading to bilateral flail chest in Cronkhite–Canada Syndrome (CCS) has not been reported. Case presentation A 59-year-old man presented with complaints of fatigue, chest pain, respiratory distress and orthopnea requiring ventilatory support to maintain oxygenation. CCS with bilateral anterior and posterior flail chest due to multiple rib fractures (2nd-10th on the right side and 2nd-11th on the left side). He underwent open reduction and anterior and posterior internal fixation using a titanium alloy fixator and a nickel-titanium memory alloy embracing fixator for chest wall reconstruction. He recovered gradually from the ventilator and showed improvement in his symptoms. He gained about 20 kg of weight in the follow up period (6 months after discharge from the hospital). Conclusion CCS is a rare, complex disease that increases the risk of developing multiple rib fractures, which can be successfully treated with open reduction and internal fixation.
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Morino K, Honma Y, Kumei S, Watanabe T, Kume K, Yoshikawa I, Harada M. Recombinant Thrombomodulin Used to Successfully Treat Cronkhite-Canada Syndrome with Disseminated Intravascular Coagulation due to Sepsis in a Compromised Patient. Intern Med 2018; 57:3079-3085. [PMID: 29877275 PMCID: PMC6262703 DOI: 10.2169/internalmedicine.0633-17] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/06/2022] Open
Abstract
Cronkhite-Canada syndrome (CCS) is a rare non-inherited disease characterized by gastrointestinal polyposis, chronic diarrhea, ectodermal dysplasia, skin hyperpigmentation, hair loss and nail atrophy. Although the efficacy of corticosteroid and immunomodulatory agents has been demonstrated, no standard therapy regimen has been established, and the prognosis of CCS is still poor due to various complications. We here in report a CCS patient complicated with severe sepsis and disseminated intravascular coagulation who was successfully treated by combined modality therapies, including recombinant human soluble thrombomodulin.
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Affiliation(s)
- Kahori Morino
- Third Department of Internal Medicine, University of Occupational and Environmental Health, School of Medicine, Japan
| | - Yuichi Honma
- Third Department of Internal Medicine, University of Occupational and Environmental Health, School of Medicine, Japan
| | - Shinsuke Kumei
- Third Department of Internal Medicine, University of Occupational and Environmental Health, School of Medicine, Japan
| | - Tatsuyuki Watanabe
- Third Department of Internal Medicine, University of Occupational and Environmental Health, School of Medicine, Japan
| | - Keiichiro Kume
- Third Department of Internal Medicine, University of Occupational and Environmental Health, School of Medicine, Japan
| | - Ichiro Yoshikawa
- Third Department of Internal Medicine, University of Occupational and Environmental Health, School of Medicine, Japan
| | - Masaru Harada
- Third Department of Internal Medicine, University of Occupational and Environmental Health, School of Medicine, Japan
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Cronkhite-Canada Syndrome: Sustained Clinical Response with Anti-TNF Therapy. Case Rep Med 2018; 2018:9409732. [PMID: 30057620 PMCID: PMC6051112 DOI: 10.1155/2018/9409732] [Citation(s) in RCA: 18] [Impact Index Per Article: 2.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/21/2017] [Revised: 02/01/2018] [Accepted: 04/15/2018] [Indexed: 12/18/2022] Open
Abstract
Cronkhite-Canada syndrome (CCS) is a rare, nonfamilial syndrome that occurs in the sixth to seventh decades of life. It is characterized by acquired gastrointestinal polyposis with an associated ectodermal triad, including alopecia, onchodystrophy, and hyperpigmentation. CCS is characteristically a progressive disease, with a high mortality rate despite medical interventions. Disease complications are typically secondary to severe malnutrition, malignancy, GI bleeding, and infection. CCS is believed secondary to immune dysregulation; however, the underlying etiology remains to be determined. Treatment for CCS is largely anecdotal, and randomized controlled therapeutic trials are lacking due to the rarity of the disease. Aggressive nutritional support in conjunction with immunosuppression has been used previously with inconsistent results. In this report, we describe the presentation and diagnosis of a case of CCS and report encouraging treatment response with anti-TNF therapy.
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Haghighi S, Noorali S, Mohammad Alizadeh AH. Cronkhite-Canada Syndrome Associated with Metastatic Colon Cancer. Case Rep Gastroenterol 2018; 12:109-115. [PMID: 29805353 PMCID: PMC5968282 DOI: 10.1159/000479220] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/15/2017] [Accepted: 07/05/2017] [Indexed: 12/20/2022] Open
Abstract
Cronkhite-Canada syndrome is characterized by gastrointestinal and ectodermal manifestations. In this paper, we describe a 64-year-old Iranian male, presenting with Cronkhite-Canada syndrome with metastatic colon cancer. The patient was suffering from hair loss, which occurred on the scalp at first and then, during 5 months, extended to the whole body. After that, his sense of taste was impaired, and 2 months later, gastrointestinal symptoms gradually started, with weight loss of 20 kg over 2 months with an initial weight of 100 kg. Finally, he was admitted to our center 10 months after the onset of symptoms. On skin examination, generalized hair loss and hyperpigmentation and dysmorphic nail changes were observed. Multiple polyps within the colon and sigmoid were observed on colonoscopy. According to biopsies, a serrated adenoma and an invasive adenocarcinoma were reported in the ascending colon and sigmoid, respectively. Other polyps were pseudopolyps, and their characteristics were not significant. Computed tomography of the lungs and abdomen showed multiple adenopathies. On biopsy, metastatic adenocarcinoma was reported. The patient underwent chemotherapy with FOLFIRI and ERBITUX. Finally, after 5 courses of chemotherapy, his regimen was changed to FOLFOX and Avastin because of evidence of progression on computed tomography. The etiology of Cronkhite-Canada syndrome is currently unknown, and the optimal therapy has not been reported so far. This syndrome has many complications; the major of them is malignancy, and the prognosis is poor with a mortality rate of 50%. Therefore, annual monitoring is necessary in these patients.
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Affiliation(s)
- Shirin Haghighi
- Shahid Beheshti University of Medical Sciences, Taleghani Hospital, Tehran, Iran
| | - Sima Noorali
- Shahid Beheshti University of Medical Sciences, Taleghani Hospital, Tehran, Iran
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Yuan W, Tian L, Ai FY, Liu SJ, Shen SR, Wang XY, Liu F. Cronkhite-Canada syndrome: A case report. Oncol Lett 2018; 15:8447-8453. [PMID: 29805581 PMCID: PMC5950510 DOI: 10.3892/ol.2018.8409] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/23/2016] [Accepted: 12/12/2017] [Indexed: 12/20/2022] Open
Abstract
Cronkhite-Canada syndrome (CCS) is a rare non-inherited condition characterized by gastrointestinal (GI) hamartomatous polyposis, alopecia, onychodystrophy, hyperpigmentation, weight loss and diarrhea. The etiology is most likely autoimmune and diagnosis is based on patient history, physical examination, endoscopic findings of GI polyposis and histology. The disease is very rare; thus far more than 500 cases of CCS have been reported globally. A 58-years-old male with CCS was reported in the present case study. The patient experienced a history of diarrhea and hematochezia for 4 months, with abdominal pain for 1 month and additional nail and toenail loss for half a month. The clinical, endoscopic and histological data confirmed the diagnosis.
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Affiliation(s)
- Wei Yuan
- Department of Gastroenterology, Hunan Key Laboratory of Non-Resolving Inflammation and Cancer, The Third Xiangya Hospital of Central South University, Changsha, Hunan 410013, P.R. China.,Department of Hepatology, The First Affiliated Hosipital of Hu-Nan University of Chinese Medicine, Changsha, Hunan 410007, P.R. China
| | - Li Tian
- Department of Gastroenterology, Hunan Key Laboratory of Non-Resolving Inflammation and Cancer, The Third Xiangya Hospital of Central South University, Changsha, Hunan 410013, P.R. China
| | - Fei-Yan Ai
- Department of Gastroenterology, Hunan Key Laboratory of Non-Resolving Inflammation and Cancer, The Third Xiangya Hospital of Central South University, Changsha, Hunan 410013, P.R. China
| | - Shao-Jun Liu
- Department of Gastroenterology, Hunan Key Laboratory of Non-Resolving Inflammation and Cancer, The Third Xiangya Hospital of Central South University, Changsha, Hunan 410013, P.R. China
| | - Shou-Rong Shen
- Department of Gastroenterology, Hunan Key Laboratory of Non-Resolving Inflammation and Cancer, The Third Xiangya Hospital of Central South University, Changsha, Hunan 410013, P.R. China
| | - Xiao-Yan Wang
- Department of Gastroenterology, Hunan Key Laboratory of Non-Resolving Inflammation and Cancer, The Third Xiangya Hospital of Central South University, Changsha, Hunan 410013, P.R. China
| | - Fen Liu
- Department of Gastroenterology, Hunan Key Laboratory of Non-Resolving Inflammation and Cancer, The Third Xiangya Hospital of Central South University, Changsha, Hunan 410013, P.R. China
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Abstract
RATIONALE Cronkhite-Canada syndrome (CCS) is a rare disease, the etiology of CCS is currently unknown. Although CCS is widely accepted as a benign disorder, the malignant potential of the polyps in CCS patients is yet controversial. PATIENT CONCERNS A 55-year-old Chinese male was first admitted to Beijing Friendship Hospital with a 3-month history of frequent watery diarrhea (10-15 times/day), loss of taste, and a weight loss of 10 kg in August 2010. The left heel bone fracture in the patient occurred about 2 weeks prior to his diarrhea. DIAGNOSES He was diagnosed as Cronkhite-Canada syndrome. INTERVENTIONS Oral administration of prednisone was initiated at a dosage of 20 mg/day. OUTCOMES After 3 months of treatment, the clinical manifestations disappeared, and colonoscopy showed sparsely distributed small polyps in the colon. Consequently, the dose of prednisone was reduced to 10mg. However, after 4 months, his fingernails were again found atrophic along with mild abdominal discomfort without diarrhea. Colonoscopy revealed a recurrence of the polyps in March 2011. The treatment was repeated with prednisone at a dosage of 20 mg/day resulting in subsided symptoms. In September 2011, he underwent colonoscopy although no significant clinical manifestations were observed. In addition, the polyp in the sigmoid colon was cancerated. LESSONS The present case indicated that the physical stress was related to CCS and malignant transformation occurred in Cronkhite-Canada syndrome polyp. After the diffused inflammatory polyps have responded to steroid therapy, other existing adenomas require endoscopic treatments, which can decrease the possibility of neoplastic transformation.
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Watanabe C, Komoto S, Tomita K, Hokari R, Tanaka M, Hirata I, Hibi T, Kaunitz JD, Miura S. Endoscopic and clinical evaluation of treatment and prognosis of Cronkhite-Canada syndrome: a Japanese nationwide survey. J Gastroenterol 2016; 51. [PMID: 26216651 PMCID: PMC4805704 DOI: 10.1007/s00535-015-1107-7] [Citation(s) in RCA: 70] [Impact Index Per Article: 7.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/04/2023]
Abstract
BACKGROUND First reported in 1955, Cronkhite-Canada syndrome (CCS), a rare syndrome characterized by ectodermal abnormalities and inflammatory changes of the gastrointestinal tract mucosa, has been associated with a poor prognosis and life-threatening malignant complications. In a large population survey, we endeavored to characterize the course and treatment outcome of CCS through clinical and endoscopic assessment, and to explore its optimal treatment and surveillance strategy. METHODS A retrospective analysis of 210 patients with CCS was conducted via a questionnaire-based nationwide survey of 983 teaching hospitals located throughout Japan. We assessed clinical features, endoscopic findings, treatments used, and short- and long-term outcomes. RESULTS The average age at diagnosis was 63.5 years. In all cases, upper or lower gastrointestinal tract polyposis was confirmed, accompanied by characteristic ectodermal abnormalities. Of the treatments used, oral corticosteroids (30-49 mg/day) were the most effective treatment for active disease, with adjunctive nutritional support considered beneficial. With corticosteroid treatment, abdominal symptoms were relieved within a few months, whereas polyp regression often required more than 6 months. Maintenance of endoscopic remission with or without steroids for 3 years significantly lowered the development of CCS-related cancer, compared with relapsers or nonresponders, underscoring the importance of sustained endoscopic remission for cancer prevention. CONCLUSIONS The prognosis of CCS has greatly improved through the use of improved medical treatment. Although CCS continues to be relentlessly progressive, carrying a high cancer risk, a sufficient dose and duration of corticosteroid therapy accompanied by nutritional support and periodic endoscopic surveillance appears to improve its natural history.
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Affiliation(s)
- Chikako Watanabe
- />Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama 359-8513 Japan
| | - Shunsuke Komoto
- />Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama 359-8513 Japan
| | - Kengo Tomita
- />Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama 359-8513 Japan
| | - Ryota Hokari
- />Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama 359-8513 Japan
| | - Masanori Tanaka
- />Department of Pathology and Laboratory Medicine, Hirosaki City Hospital, Aomori, Japan
| | - Ichiro Hirata
- />Department of Gastroenterology, Fujita Health University, Aichi, Japan
| | - Toshifumi Hibi
- />Center for Advanced IBD Research and Treatment, Kitasato Institute Hospital, Tokyo, Japan
| | - Jonathan D. Kaunitz
- />Greater Los Angeles VA Medical Center and Department of Medicine and Department of Surgery, David Geffen School of Medicine, University of California, Los Angeles, Los Angeles, CA USA
| | - Soichiro Miura
- />Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama 359-8513 Japan
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Ota S, Kasahara A, Tada S, Tanaka T, Umena S, Fukatsu H, Noguchi T, Matsumura T. Cronkhite-Canada syndrome showing elevated levels of antinuclear and anticentromere antibody. Clin J Gastroenterol 2014; 8:29-34. [PMID: 25518819 DOI: 10.1007/s12328-014-0547-6] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/07/2014] [Accepted: 12/01/2014] [Indexed: 11/26/2022]
Abstract
A 56-year-old female initially visited an otorhinolaryngologist because of an impaired sense of taste in September, 2010 and was referred to our facility in October, 2010. She was diagnosed with Basedow's disease for which she underwent subtotal thyroidectomy in 1984 and arthritis involving multiple joints, primarily affecting her hands. In addition, the anticentromere antibody (ACA) level was markedly high. On physical examination, alopecia as well as hyperpigmentation of the dorsum of the hands and back was observed. Dystrophic changes of the fingernails and a bilateral thumb abduction deformity were observed. Antinuclear antibodies were elevated. Gastrointestinal endoscopy and colonoscopy revealed the mucosa carpeted with strawberry-like polypoid lesions. Histopathological examination of the biopsied specimen of the stomach revealed a corkscrew-like appearance. Thus, the patient was diagnosed with Cronkhite-Canada syndrome (CCS). She admitted to our hospital in November, 2010. Oral prednisolone was administered with success. In July, 2012, her antimitochondrial M2 antibody level was elevated. To the best of our knowledge, the present case is the first patient with CCS, a history of Basedow's disease, and elevated levels of ACA and antimitochondrial M2 antibody. We consider the present case suggests CCS could be caused by immunological abnormality.
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Affiliation(s)
- Seisuke Ota
- Department of Internal Medicine, Himeji St. Mary Hospital, Himeji, Japan,
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Cronkhite-Canada syndrome associated with esophageal and gastric cancers: report of a case. Surg Today 2014; 45:777-82. [PMID: 25008327 DOI: 10.1007/s00595-014-0977-2] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/17/2013] [Accepted: 04/01/2014] [Indexed: 10/25/2022]
Abstract
Cronkhite-Canada Syndrome (CCS) is a rare non-inherited gastrointestinal polyposis syndrome with characteristic ectodermal changes. We report an extremely unusual case of CCS associated with primary esophageal and gastric cancers. A 74-year-old Japanese man with symptoms of anorexia and diarrhea was found to have primary esophageal and gastric cancers, as well as multiple gastric and colonic polyps. Based on the physical findings of onychodystrophy and alopecia, we diagnosed CCS. Because of his age and nutritional status, we decided to perform total gastrectomy for gastric cancer and chemoradiotherapy for esophageal cancer, upon completion of which the patient was started on steroid therapy for the CCS. After 1 week of steroid therapy, the patient's watery diarrhea improved. We recommend that for patients with CCS, the therapeutic strategy be carefully considered based on the patient's nutritional status, the severity of the CCS, and the extent of gastrointestinal cancer.
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16
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Wen XH, Wang L, Wang YX, Qian JM. Cronkhite-Canada syndrome: Report of six cases and review of literature. World J Gastroenterol 2014; 20:7518-7522. [PMID: 24966624 PMCID: PMC4064099 DOI: 10.3748/wjg.v20.i23.7518] [Citation(s) in RCA: 23] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/14/2014] [Revised: 03/28/2014] [Accepted: 04/09/2014] [Indexed: 02/06/2023] Open
Abstract
Cronkhite-Canada syndrome (CCS) is a rare nonfamilial polyposis syndrome characterized by epithelial disturbances in the gastrointestinal tract and skin. The aim of this study was to investigate the clinical features and potential therapies for CCS. Six patients with CCS admitted from December 1992 to July 2008 to Peking Union Medical College Hospital were evaluated. All patients had clinical manifestation of nonhereditary gastrointestinal polyposis with diarrhea, skin hyperpigmentation, alopecia, and nail dystrophy. Fecal occult blood was positive in all six cases. Serum hemoglobin, potassium, calcium and protein were below the normal range in two cases. Anti-Saccharomyces cerevisiae and antinuclear antibodies were present in three cases. Multiple polyps were found in all patients by gastroscopy and colonoscopy, with only one in the esophagus. Histologically, there were hyperplastic polyps in five cases, tubular adenoma in three, and juvenile polyp in one with chronic inflammation and mucosal edema. Comprehensive treatment led by corticosteroids can result in partial remission of clinical symptoms, and long-term follow-up is necessary.
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17
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Zhang P, Zhang D, Li YK. Cronkhite-Canada syndrome: a report of one case. Shijie Huaren Xiaohua Zazhi 2011; 19:429-433. [DOI: 10.11569/wcjd.v19.i4.429] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/06/2023] Open
Abstract
Cronkhite-Canada syndrome (CCS) is a rare clinic entity of unknown etiology and has poor prognosis, characterized by gastrointestinal polyposis with ectodermal changes. Main clinical manifestations include chronic diarrhea, abdominal pain, alopecia, skin pigmentation, and nail changes. Here, we report a case of CCS in a 50-year-old male and review the relevant literature.
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18
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Ishikawa E, Kudo M, Minami Y, Ueshima K, Kitai S, Ueda K. Cecal intussusception in an adult with Cronkhite-Canada syndrome relieved by colonoscopy. Intern Med 2010; 49:1123-6. [PMID: 20558928 DOI: 10.2169/internalmedicine.49.2813] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/06/2022] Open
Abstract
Cronkhite-Canada syndrome (CCS) is a rare, noninherited gastrointestinal polyposis syndrome associated with characteristic ectodermal abnormalities. Here, we report a case of Cronkhite-Canada syndrome with cecal intussusception relieved by colonoscopy. A 52-year-old man who was diagnosed as CCS pathologically two years previously presented abdominal pain and sub fever-up. Physical examination revealed the palpable mass sized approximate 10 cm in diameter in the upper abdominal site, in addition to the symptoms of alopecia, absent fingernails and toenails. However, abdominal wall rigidity and rebound tenderness were never expressed. Abdominal plain CT showed concentric circles from the ascending to the middle of the transverse colon, and a tumor in the lumen at the middle of the transverse colon. Colonoscopic reduction was performed first because we diagnosed it as intussusception due to CCS polyps without peritoneal irritation, and his symptoms were improved dramatically after careful reduction. Therefore, he was able to undergo the laparoscopic ascending colectomy as scheduled.
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Affiliation(s)
- Emi Ishikawa
- Division of Gastroenterology and Hepatology, Department of Internal Medicine, Kinki University School of Medicine, Kinki University School of Medicine, Osaka-Sayama, Japan
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19
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Karasawa H, Miura K, Ishida K, Sase T, Kobayashi T, Kinouchi M, Okabe M, Ando T, Kaneko N, Yamamura A, Shibata C, Sasaki I. Cronkhite-Canada syndrome complicated with huge intramucosal gastric cancer. Gastric Cancer 2009; 12:113-7. [PMID: 19562466 DOI: 10.1007/s10120-009-0506-y] [Citation(s) in RCA: 15] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/01/2008] [Accepted: 03/19/2009] [Indexed: 02/07/2023]
Abstract
Cronkhite-Canada syndrome (CCS) is a rare nonhereditary disorder with gastrointestinal polyposis and associated ectodermal changes. This report documents a 59-year-old Japanese man with CCS who underwent a total gastrectomy for gastric tumors. The resected specimen showed a huge gastric adenocarcinoma with numerous polyps throughout the stomach. The cancer was pathologically limited to within the mucosa and its histological structure resembled that of hyperplasia in CCS polyps, which led us to suppose that the carcinoma had arisen from hyperplastic CCS polyps. These results urged us to study the phenotypic expression of mucins, which revealed MUC2(-) and MUC5AC(+) and supported the diagnosis of the tumor as a gastric-type well-differentiate adenocarcinoma. A literature search revealed that 32 gastric carcinomas which developed in patients with CCS were mostly limited to within the submucosa in spite of their huge sizes, and such cancer development in CCS polyposis is therefore not considered to be unusual.
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Affiliation(s)
- Hideaki Karasawa
- Department of Surgery, Tohoku University Graduate School of Medicine, 1-1 Seiryo-machi, Aoba-ku, Sendai, 980-8574, Japan
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20
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Cronkhite-Canada Syndrome: Gastric Involvement Diagnosed by MDCT. Case Rep Med 2009; 2009:148795. [PMID: 19721708 PMCID: PMC2729239 DOI: 10.1155/2009/148795] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/28/2009] [Accepted: 06/22/2009] [Indexed: 12/03/2022] Open
Abstract
Chronkhite-Canada is a rare nonfamilial polyposis syndrome that usually presents as chronic malabsorption in adults. We present a case of a-73-year old woman with chronic gastrointestinal bleeding and malnutrition. On CT imaging she was found to have massive gastric polyps, which on biopsy was most consistent with Cronkhite-Canada syndrome.
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21
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Abstract
Since the histologic description of the hamartomatous polyp in 1957 by Horrilleno and colleagues, descriptions have appeared of several different syndromes with the propensity to develop these polyps in the upper and lower gastrointestinal tracts. These syndromes include juvenile polyposis, Peutz-Jeghers syndrome, hereditary mixed polyposis syndrome, and the phosphatase and tensin homolog gene (PTEN) hamartoma tumor syndromes (Cowden and Bannayan-Riley-Ruvalcaba syndromes), which are autosomal-dominantly inherited, and Cronkhite-Canada syndrome, which is acquired. This article reviews the clinical aspects, the molecular pathogenesis, the affected organ systems, the risks of cancer, and the management of these hamartomatous polyposis syndromes. Although the incidence of these syndromes is low, it is important for clinicians to recognize these disorders to prevent morbidity and mortality in these patients, and to perform presymptomatic testing in patients at risk.
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Affiliation(s)
- Daniel Calva
- Resident in Surgery, Roy J. and Lucille A. Carver University of Iowa College of Medicine, Iowa City, IA
| | - James R. Howe
- Professor of Surgery, Chief, Division of Surgical Oncology and Endocrine Surgery, Roy J. and Lucille A. Carver University of Iowa College of Medicine; Iowa City, IA
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22
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Sampson JE, Harmon ML, Cushman M, Krawitt EL. Corticosteroid-responsive Cronkhite-Canada syndrome complicated by thrombosis. Dig Dis Sci 2007; 52:1137-40. [PMID: 17342394 DOI: 10.1007/s10620-006-9375-y] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/08/2006] [Accepted: 04/01/2006] [Indexed: 01/05/2023]
Affiliation(s)
- Joanna E Sampson
- Departments of Medicine and Pathology, University of Vermont, Given C-246, Burlington, VT 05405-0068, USA
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23
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Marín Serrano E, Guillén Mariscal P, Pérez-Requena J, Rendón Unceta P. Síndrome de Cronkhite-Canada: una poliposis adquirida y potencialmente reversible. GASTROENTEROLOGIA Y HEPATOLOGIA 2006; 29:619-21. [PMID: 17198639 DOI: 10.1157/13095196] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/21/2022]
Abstract
Cronkhite-Canada syndrome is an infrequent, nonadenomatous, acquired polyposis that is associated with ectodermic alterations. The etiopathogenesis and optimal therapeutic management are unknown. We present a case of Cronkhite-Canada syndrome in a 58-year-old man who, after combined treatment with corticosteroids, disodium cromoglycate, loratadine, ciprofloxacin, and zinc, showed complete clinical and partial endoscopic remission. The polypoid lesions of Cronkhite-Canada syndrome can be reversed with medical treatment. Consequently, the natural history of the disease can be modified and its prognosis improved.
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Affiliation(s)
- Eva Marín Serrano
- Servicio de Aparato Digestivo, Hospital Universitario Puerta del Mar, Cádiz, España
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24
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Anderson RD, Patel R, Hamilton JK, Boland CR. Cronkhite-Canada syndrome presenting as eosinophilic gastroenteritis. Proc AMIA Symp 2006; 19:209-12. [PMID: 17252034 PMCID: PMC1484525 DOI: 10.1080/08998280.2006.11928163] [Citation(s) in RCA: 16] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/18/2022] Open
Abstract
Cronkhite-Canada syndrome (CCS), first described in 1955, is a rare clinical syndrome of unknown etiology. CCS is diagnosed clinically, and the presenting symptoms include alopecia, cutaneous hyperpigmentation, gastrointestinal polyposis, and onychodystrophy, often accompanied by diarrhea, weight loss, and abdominal pain. We describe a unique case of CCS that presented with eosinophilic infiltrate on gastric and duodenal biopsies and review the literature pertaining to this rare syndrome.
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Affiliation(s)
- Robert D Anderson
- Department of Internal Medicine, Baylor University Medical Center, Dallas, Texas, USA
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25
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Vernia P, Marcheggiano A, Marinaro V, Morabito S, Guzzo I, Pierucci A. Is Cronkhite-Canada Syndrome necessarily a late-onset disease? Eur J Gastroenterol Hepatol 2005; 17:1139-41. [PMID: 16148564 DOI: 10.1097/00042737-200510000-00022] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/10/2022]
Abstract
Cronkhite-Canada Syndrome is a non-inherited, non-congenital disease characterized by juvenile hamartomatous gastrointestinal polyps with a typically late onset. In the case described herein the disease was diagnosed in a 17-year-old male with type I diabetes and thalassaemia minor, in coincidence with severe symptomatic intestinal candidiasis. Following the disappearance of the mycosis and correction of the protein and electrolyte imbalance, the ectodermal abnormalities returned to normal and the patient remained asymptomatic during a 7-year follow-up period, despite proteinuria resulting from membranous glomerulopathy. The concept that Cronkhite-Canada Syndrome is a late-onset disease should probably be reconsidered as it may remain asymptomatic, and thus not diagnosed, for a long a time.
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Affiliation(s)
- Piero Vernia
- Gastroenterology Unit, Department of Clinical Science, University of Rome La Sapienza, Rome, Italy.
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26
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Takakura M, Adachi H, Tsuchihashi N, Miyazaki E, Yoshioka Y, Yoshida K, Oryo F, Sawada T. A Case of Cronkhite-Canada Syndrome markedly improved with mesalazine therapy. Dig Endosc 2004. [DOI: 10.1111/j.1443-1661.2004.00306.x] [Citation(s) in RCA: 17] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/23/2023]
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27
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Chadalavada R, Brown DK, Walker AN, Sedghi S. Cronkhite-Canada syndrome: sustained remission after corticosteroid treatment. Am J Gastroenterol 2003; 98:1444-6. [PMID: 12818298 DOI: 10.1111/j.1572-0241.2003.07509.x] [Citation(s) in RCA: 34] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
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28
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Abstract
Cronkhite-Canada syndrome (CCS) is a rare, non-inherited gastrointestinal polyposis syndrome associated with characteristic ectodermal abnormalities. A number of potentially life-threatening complications including malnutrition, gastrointestinal bleeding and infection may occur in affected patients and CCS is fatal in many cases. The optimal therapy for CCS is not known but several treatment options have been described. Nutritional support, antibiotics, corticosteroids, anabolic steroids, histamine-receptor antagonists and surgical treatment have all been used with varying degrees of success. Unfortunately, controlled therapeutic trials have not been possible because of the rarity of the disease. Most recently, a combination regimen using histamine-receptor antagonists, cromolyn sodium, prednisone and suppressive antibiotics has been described. The reported treatment options and rates of success are reviewed.
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Affiliation(s)
- Eric M Ward
- Division of Gastroenterology and Hepatology, Mayo Clinic, 4500 San Pablo Road, Jacksonville, FL 32224, USA
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29
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Abstract
The non-inherited gastrointestinal polyposis syndromes represent a group of rare disorders characterized by the presence of multiple, non-adenomatous polyps on the gastrointestinal mucosa occurring in unrelated patients. We present here a review of the clinical and histo- pathological aspects of the syndromes to include the Cronkhite-Canada syndrome, hyperplastic polyposis and lipomatous polyposis. While infrequently encountered, these diseases can have devastating clinical effects that may be aggravated by delays in diagnosis and treatment. Prompt accurate diagnosis and treatment of these uncommon disorders depend on a sound working knowledge of the distinct clinical and pathological features described herein.
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Affiliation(s)
- E M Ward
- Department of Internal Medicine and Division of Gastroenterology and Hepatology, Mayo Clinic, Jacksonville, FL 32224, USA
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30
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Fossati M, Lombardi F, Cattaneo C, Borsa C, Colombo F. Cronkhite-Canada syndrome. A case of sustained partial-remission. Dig Liver Dis 2001; 33:785-7. [PMID: 11838614 DOI: 10.1016/s1590-8658(01)80696-4] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/08/2023]
Abstract
A case of Cronkhite-Canada syndrome is described. Few cases have been published and in most patients the prognosis is poor. A variety of medical measures have been attempted in those in whom remission has been reported. In the patient presented here, a sustained partial-remission has been achieved with steroids.
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Affiliation(s)
- M Fossati
- Department of Internal Medicine, San Leopoldo Mandic Hospital, Merate, Lecco, Italy.
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