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Incapacitating pain from Tenofovir Induced Hypophosphatemic Osteomalacia in a Hemophilia Patient - A Case Report.

ABSTRACT: Background: Pain in patients with hemophilia is common and usually a result of arthropathy. Clinicians should, however, consider a wide range of etiologies for pain in patients with hemophilia including infection, osteoporotic fractures, arthritis, and osteomalacia. Aims: This case demonstrates an instance of poorly localized back and hip pain, severe enough to prevent ambulation, caused by hypophosphatemic osteomalacia due to tenofovir treatment for blood transfusion acquired Human Immunodeficiency Virus (HIV) in a patient with hemophilia A. Methods: Case Report. Results: With termination of tenofovir treatment, this patient returned to baseline function. Conclusion: This report serves to emphasize the need for accurate diagnosis of pain in hemophilia patients, especially among the aging demographic of people with hemophilia in which there is a significant likelihood of an HIV infection and among patients who may be on Pre-exposure Prophylaxis (PrEP) or clinical trials involving tenofovir.

SUBMITTER: Woo E

PROVIDER: S-EPMC7951154 | biostudies-literature | 2020 Dec

REPOSITORIES: biostudies-literature

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Incapacitating pain from Tenofovir Induced Hypophosphatemic Osteomalacia in a Hemophilia Patient - A Case Report.

Woo Emma E Kumbhare Dinesh D Winston Paul P

Canadian journal of pain = Revue canadienne de la douleur 20201214 1

<b>Background:</b> Pain in patients with hemophilia is common and usually a result of arthropathy. Clinicians should, however, consider a wide range of etiologies for pain in patients with hemophilia including infection, osteoporotic fractures, arthritis, and osteomalacia. <b>Aims:</b> This case demonstrates an instance of poorly localized back and hip pain, severe enough to prevent ambulation, caused by hypophosphatemic osteomalacia due to tenofovir treatment for blood transfusion acquired Huma ...[more]

PMID: 33987506

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Project description:Iron deficiency anemia is treated by iron supplementation. Increasing evidence has shown that the carbohydrate components in iron infusions can cause hypophosphatemia and subsequent osteomalacia due to excess intact fibroblast growth factor 23 (iFGF23). We here undertook an in-depth characterization of 13 patients with iron infusion-induced osteomalacia (IIIO). Patients were characterized (monocentric institutional practice) by means of laboratory, bone density, HR-pQCT, and virtual osteoid volume estimation. We additionally report a patient who was treated with burosumab because iron infusions had to be continued despite osteomalacia. All 13 patients received ferric carboxymaltose (FCM) infusions and presented with low phosphate levels. Stopping the FCM infusions and supportive treatment by substitution of phosphate, calcium, native, and/or active Vitamin D was the chosen therapeutic approach. Pain, mobility, and biochemical data, such as serum phosphate levels, BMD, bone microstructure, and virtual osteoid volume, were the main outcome measures. Our results indicate biochemical normalization (eg, phosphate levels pre: 0.50 mmol/L ± 0.23 mmol/L, post: 0.93 mmol/L ± 0.32 mmol/L, p<.001) after stopping the FCM infusion and establishing supportive treatment. Additionally, pain levels on the visual analog scale (VAS) decreased (VASpre 7.31 ± 1.22, VASpost 2.73 ± 1.27, p<.0001) and areal BMD (expressed by T-score) improved significantly (T-scorepre: -1.85 ± 1.84, T-scorepost: -0.91 ± 2.13, p<.05). One patient requiring ongoing FCM infusions experienced significant additional benefits from burosumab treatment. In conclusion, our results highlight the importance of monitoring phosphate in patients treated with FCM infusions. Stopping FCM infusions is effective in addressing the excess of iFGF23 and thereby phosphate wasting. Supportive therapy enables quick recovery of the musculoskeletal system and improves pain levels in these patients.

Project description:BackgroundIn this paper, we present a rare case of tumor-induced osteomalacia (TIO) and a literature review of this rare disease.MethodsA case of TIO of the isolated sphenoid sinus was reported. Furthermore, the clinical features of TIO in the sphenoid sinus and other sinonasal sinuses were also reviewed and summarized.ResultsA 35-year-old man with muscle weakness and lower back pain came to the Department of Neurology. No obvious neurological disease was found; however, magnetic resonance imaging of the extremities accidentally showed a tumor in the axilla. Bone scintigraphy showed suspicious bone metastasis. Hypophosphatemia was neglected. Interestingly, 2-deoxy-2-[fluorine-18]fluoro-d-glucose positron emission tomography/computed tomography (18F-FDG PET/CT) detected a tumor in the axilla and another in the sphenoid sinus, but only the tumor in the sphenoid sinus had somatostatin receptor (SSTR) expression in 68-gallium 1,4,7,10-tetraazacyclododecane-1,4,7,10-tetraacetic acid octreotate (Ga-68 DOTATATE) PET/CT. The sphenoid sinus tumor was proven to be a phosphaturic mesenchymal tumor (PMT), and the phosphate levels returned to normal after surgery. The literature review showed only 17 cases of TIOs that occurred in the sphenoid sinus, with an average age of 43.3 ± 13.7 years. Only three cases of TIOs in the sphenoid sinus did not invade the nasal cavity or other paranasal sinuses, which could be identified as isolated sphenoid sinus diseases. We compared the clinical features of sphenoid TIOs with those of non-sphenoid sinonasal TIOs, and it was found that the concentration of 1,25-dihydroxy vitamin D in the group with sphenoid TIOs was much higher than that in the group with non-sphenoid sinonasal TIOs. A total of 153 cases of TIOs in the sinonasal sinus were reviewed. The ethmoid sinus was found to be the major site (64.7%), followed by the nasal cavity (50.3%), maxillary sinus (19.0%), frontal sinus (16.4%), and sphenoid sinus (11.8%). There were 66 patients (43.1%) who showed tumors invading more than one sinus. Most of the tumors (69.3%) were diagnosed as PMTs by pathology, followed by hemangiopericytoma (14.3%). Immunostaining was beneficial in the differential diagnosis of these tumors; however, larger sample sizes are needed for better accuracy.ConclusionTIO in the sinonasal sinus, especially in the sphenoid sinus, is rare. Moreover, isolated sphenoid sinus disease can be easily misdiagnosed. When the clinical manifestation of osteomalacia is atypical, associating it with sphenoid sinus disease is even more difficult. Thus, TIO in the sphenoid sinus needs further exploration.

Dataset Information

Incapacitating pain from Tenofovir Induced Hypophosphatemic Osteomalacia in a Hemophilia Patient - A Case Report.

Publications

Incapacitating pain from Tenofovir Induced Hypophosphatemic Osteomalacia in a Hemophilia Patient - A Case Report.

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