Lymphangiomyomatosis associated with chylous ascites and high serum CA-125 levels. A case report.

Kanbe, Akira; Hajiro, Kiyoshi; Adachi, Yukihiko; Honda, Kiyoshi; Suzuki, Tsuneyuki; Yamamoto, Tetsuro

doi:10.2169/internalmedicine1962.26.237

Cited by 13 publications

(7 citation statements)

References 6 publications

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“…There are several case reports available indeed in the literature, describing the LAM patients with chylous ascites responded successfully to fat-restricted diet and was asymptomatic and free of ascites for 2 to 3 years (6,7). However, the failure of medical management including fatrestricted diet to control chylous ascites, like our case, was also reported in LAM (8)(9)(10)(11). In these circumstances, the instillation of peritoneovenous shunt appears to be of choice (12) because it returns chylous ascites to blood circulation without discarding and is expected to improve the hemodynamic, nutritional, and immunological status.…”

Section: F I G U R E 1 Ra D I O L O G I C F I N D I N G S O F T H Ementioning

confidence: 62%

Peritoneovenous Shunting for Intractable Chylous Ascites Complicated with Lymphangioleiomyomatosis

et al. 2008

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Section: F I G U R E 1 Ra D I O L O G I C F I N D I N G S O F T H Ementioning

confidence: 62%

Peritoneovenous Shunting for Intractable Chylous Ascites Complicated with Lymphangioleiomyomatosis

et al. 2008

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“…Hormone therapy: other anti-oestrogen interventions Despite a number of case reports describing the use of oophorectomy [74][75][76][77][78], tamoxifen [79][80][81][82] and GnRH agonists [83][84][85] in LAM no confident data exist on the efficacy of any anti-oestrogen strategy for LAM. One recent open label study that examined 11 patients treated with the GnRH agonist triptorelin found that no patients improved and the drug was associated with a reduction in bone mineral density [86].…”

Section: Inhaled Bronchodilatorsmentioning

confidence: 99%

European Respiratory Society guidelines for the diagnosis and management of lymphangioleiomyomatosis

Johnson¹,

Cordier²,

Lazor³

et al. 2009

Eur Respir J

483

561

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“…Surgical intervention is considered the ultima ratio if the previous options fail. This approach is comprised of reexploration and ligation of the leakage, which can include i) use of glue, but showing relatively poor outcome (Naber and Isringhaus 1988), and ii) insertion of a peritoneovenous shunt (Hoffmann 1902;Kanbe et al 1987;Schumpelick and Riesner 1993;Gupta et al 2004). In our case, chylous ascites was diagnosed in an appropriate manner by the detection of chylomicrons and an elevated content of triglycerides -though lymphoscintigraphy is also suggested (Pui and Yueh 1998;Seo et al 1999), but not always suitable (Cope 1998) -during the patient's reconvalescence period after long-term artifi cial respiration.…”

Section: Discussionmentioning

confidence: 99%

Successful conservative treatment of chylous ascites as rare intermediate complication after resection of an aboral esophageal carcinoma

Meyer¹

2008

OAS

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Background: Chylous ascites is an uncommon symptom that needs specifi c expertise with regard to the diagnostic and therapeutic management. Methods: We are reporting on an uncommon and exceptional case of a 65-year-old man in whom postoperative chylous ascites was diagnosed using both daily ascites inspection and laboratory investigation. Treatment was initiated according to a previously established protocol, which summarized our own experiences and recommendations from the literature. Results: The patient showed a prolonged postoperative course after partial esophagectomy of the aboral segment (2-cavity intervention) because of Barrett carcinoma (pT3N1M0G3R0), including lymphadenectomy and esophagogastric anastomosis, which was initially complicated by i) hemorrhage (d2), leading to relaparotomy, ii) prolonged weaning maneuver, and iii) suspicion of chylous ascites characterized by beige and milky effusion out of the abdominal drainage after appendicostoma placement for colonic decompression (d28). Diagnosis was confi rmed by laboratory detection of chylomicrons and triglycerides of 4.8 mmol/L. After an observation period of two weeks, total parenteral nutrition (TPN) was initiated for 5 weeks. This resulted in the clearance of ascites (decrease of its amount partially after 5-10 d, completely after 20 d) and fi nal removal of the abdominal drain. There were no further abnormal symptoms or signs. Conclusions: The established therapeutic concept favoring conservative long-term TPN and avoiding reintervention, which was repeatedly used in rare cases of chylous ascites, is suitable to achieve spontaneous healing of lymphatic leakage by diminishing lymphatic fl ow due to decreased i) enteral fat absorption and ii) transport along the lymphatic vessels as shown in this exceptional case. To our best knowledge, this is only the sixth case with chylous ascites after esophagectomy reported in the literature.

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Lymphangiomyomatosis associated with chylous ascites and high serum CA-125 levels. A case report.

Cited by 13 publications

References 6 publications

Peritoneovenous Shunting for Intractable Chylous Ascites Complicated with Lymphangioleiomyomatosis

Peritoneovenous Shunting for Intractable Chylous Ascites Complicated with Lymphangioleiomyomatosis

European Respiratory Society guidelines for the diagnosis and management of lymphangioleiomyomatosis

Successful conservative treatment of chylous ascites as rare intermediate complication after resection of an aboral esophageal carcinoma

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