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LETTER TO THE EDITOR
Year : 2013  |  Volume : 17  |  Issue : 5  |  Page : 947-949

Raised CA19.9 and hepatic space occupying lesion after teriparatide therapy in a case of polyostotic fibrous dysplasia


Department of Endocrinology, Army Hospital (Research and Referral), Delhi Cantonment, India

Date of Web Publication29-Aug-2013

Correspondence Address:
M K Garg
Department of Endocrinology, Army Hospital (Research and Referral), Delhi Cantonment - 110 010
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2230-8210.117209

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How to cite this article:
Pandit A, Kharb S, Garg M K. Raised CA19.9 and hepatic space occupying lesion after teriparatide therapy in a case of polyostotic fibrous dysplasia. Indian J Endocr Metab 2013;17:947-9

How to cite this URL:
Pandit A, Kharb S, Garg M K. Raised CA19.9 and hepatic space occupying lesion after teriparatide therapy in a case of polyostotic fibrous dysplasia. Indian J Endocr Metab [serial online] 2013 [cited 2019 Nov 20];17:947-9. Available from: http://www.ijem.in/text.asp?2013/17/5/947/117209

Sir,

We have previously submitted case report in our journal titled "Spinal polyostotic fibrous dysplasia in two adults: Does only biopsy unravel the mystery?" [1] Case-2 was treated with bisphosphonates and narcotic analgesics without any relief and became bed bound. PFD is a disease of admixture of mutated cells with abnormal cAMP levels and normal cells. Over the time bony lesions in PFD get normalized. [2] It is hypothesized that mutated cells undergo apoptosis following period of rapid turnover like puberty and proliferation of remaining normal cells leads to normalization. Parathyroid hormone (PTH) is stimulant for osteoblastic cells, and may lead to early apoptosis of mutated cells. Our patient had low bone mineral density at lumbar spine with compression fractures. Hence after taking ethical clearance and written consent from patient, we started patient on teriparatide therapy 20 μg subcutaneously daily. After two months, patient reported for review. His clinical condition had deteriorated. Re-evaluation revealed deterioration with fresh lesion appearing on PET-CT [Figure 1] and [Figure 2]. Space occupying hypoechoic lesions appeared in liver on ultrasonography, CT scan and FDG PET (SUV - 6.3). New lesions were also seen in bones with maximum SUV at anterior superior iliac spine (5.7). Oncological markers showed elevated CA19.9 (671 U/ml normal < 37). In view of above malignancy was suspected. FNAC of liver showed normal liver cells. Open biopsy from anterior superior iliac spine demonstrated classical Chinese letter pattern [Figure 3] again confirming the diagnosis of PFD.
Figure 1: FDG-PET showing avid uptake in spine, humerus, liver, and pelvis

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Figure 2: Multiple FDG avid space occupying lesions in liver

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Figure 3: H and E, ×10 staining showing classical Chinese letter pattern in bone biopsy

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To best of our knowledge, this is the first report of raised CA19.9 levels in PFD. CA 19.9 (carbohydrate antigen 19.9), also known as sialylated Lewis a-antigen (a blood protein in red blood cells), is an antigen defined by the monoclonal antibody 1116NS 19.9. It was first mentioned by Koprowski et al. [3] CA19.9 is synthesized by varied cells in the body like normal human pancreatic and biliary ducts and by gastric, colonic, endometrial, salivary and bronchial epithelium. False positive values are reported pancreatobiliary disorders, inflammatory bowel disease, hypothyroidism, kidney problems in up to 31%. [4] After thorough search we could only find one case report where an intramuscular myxoma was associated with elevated CA 19.9 levels. [5] Intramuscular myxomas are associated with GNAS1 mutation and their association with PFD is known as Mazabraud's syndrome. [6] Our patient also developed hepatic space occupying lesion and FNAC revealed normal hepatic morphology suggestive of hepatic nodular hyperplasia. Elevation of CA19.9 has also been reported with liver disorder. Our patient had persistently elevated liver enzyme for last one year without evidence of viral infection or autoimmunity. Deterioration in our patient can be either due to teriparatide therapy or some unknown environmental agent. If it was due to PTH associated stimulation of Gsá receptor, it can be speculated that other drugs or chemicals stimulating Gsá receptor can also cause deterioration and may require to be avoided.

 
   References Top

1.Gundgurthi A, Garg MK, Bhardwaj R, Kharb S, Pandit A, Brar KS, et al. Spinal polyostotic fibrous dysplasia in two adults: Does only biopsy unravel the mystery? Indian J Endocrinol Metab 2013. [In Press]  Back to cited text no. 1
    
2.Collins MT. Spectrum and natural history of fibrous dysplasia of bone. J Bone Miner Res 2006;21:P99-P104.  Back to cited text no. 2
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3.Ballehaninna UK, Chamberlain RS. Serum CA 19-9 as a Biomarker for Pancreatic Cancer-A Comprehensive Review. Indian J Surg Oncol 2011;2:88-100.  Back to cited text no. 3
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4.Ventrucci M, Pozzato P, Cipolla A, Uomo G. Persistent elevation of serum CA 19-9 with no evidence of malignant disease. Dig Liver Dis 2009;41:357-63.  Back to cited text no. 4
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5.Theodorou D, Kleidi ES, Doulami GI, Drimousis PG, Larentzakis A, Toutouzas K, et al. Intramuscular myxoma associated with an increased carbohydrate antigen 19.9 level in a woman: A case report. J Med Case Rep 2011;5:184.  Back to cited text no. 5
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6.Delaney D, Diss TC, Presneau N, Hing S, Berisha F, Idowu BD, et al. GNAS1 mutations occur more commonly than previously thought in intramuscular myxoma. Mod Pathol 2009;22:718-24.  Back to cited text no. 6
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  [Figure 1], [Figure 2], [Figure 3]



 

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