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PULMONARY ALVEOLAR MICROLITHIASIS

Year 2016, Volume: 30 Issue: 1, 53 - 57, 01.05.2016

Abstract

Pulmonary alveolar microlithiasis (PAM) is a rare lung disease characterized by the deposition of calcium in the alveolar spaces and bilateral diffuse micronodular ‘‘sandstorm'' radiographic pattern. The current report presents the case of a 19-yearold woman with pulmonary alveolar microlithiasis. The patient presented with exertional dyspnea and cough for two months. The physical examination was within normal limits and respiratory sounds were normal. The chest x-ray revealed a bilateral diffuse micronodular pattern in the middle and the lower areas of both lungs. On the parenchymal window of thorax computed tomography, bilateral diffuse widespread millimetric calcified nodules that were more prominent in the lower lobes were observed. Whole body bone scintigraphy with technetium-99m revealed bilateral, diffuse, heterogeneous, increased uptake in the pulmonary parenchyma. Transbronchial biopsy forceps example obtained from left lung lower lobe by fiberoptic bronchoscopy intra-alveolar PAS (+) deposits displayed. The patient was diagnosed with PAM and continues to be followed-up on without treatment.

References

  • 1. Friedrich N. Corpora amylacea in den Lungen. Arch Pathol Anat 1856; 9:613–8.
  • 2. Harbitz F. Extensive calcifications of the lungs as distinct disease. Arch Intern Med 1918; 21:139–46.
  • 3. Puhr L. Microlithiasis alveolarus pulmonum. Virchows Arch 1933; 290:156-60.
  • 4. Ucan ES, Keyf AI, Aydilek R, Yalcin Z, Sebit S, Kudu M, et al. Pulmonary alveolar microlithiasis: review of Turkish reports. Thorax 1993; 48:171-3.
  • 5. Ricci A, Papale M, De Clementi F, Sposato B, Guidi L, et al. Pulmonary alveolar microlithiasis: report on 576 cases published in the literature. Sarcoidosis Vasc Diffuse Lung Dis 2004; 21(3):173-81
  • 6. Castellana G, Lamorgese V. Pulmonary alveolar microlithiasis: World cases and review of the literature. Respiration 2003; 70:549-55.
  • 7. Deniz O. Pulmonary alveolar microlithiasis. Tuberk Thorax 2005; 53:293-8.
  • 8. Millqvist E, Bende M. Capsaicin cough sensitivity is decreased in smokers. Respir Med 2001; 95(1):19-21.
  • 9. Wang BM, Stern EJ, Schmidt RA, Pierson DJ. Diagnosing pulmonary alveolar proteinosis. A review and an update. Chest 1997; 111:460-6.
  • 10. Deniz O, Ors F, Tozkoparan E, Ozcan A, Gumus S, Boz-lar U, et al. High resolution computed tomographic features of pulmonary alveolar microlithiasis. Eur J Radiol 2005; 55:452-60.
  • 11. Shah TC, Talwar A, Shah RD, Margouleff D. Pulmonary alveolar microlithiasis: radiographic and scintigraphic correlation. Clin Nucl Med 2007;32 3:249–51.
  • 12. Elboğa U, Kalender E, Dikensoy Ö, Demir HD, Çelen Z, Yılmaz M. The importance of the bone scintigraphy in the diagnosis of pulmonary alveolar microlithiasis. Respir Case Rep 2014; 3(2):90-2.
  • 13. Sahin U, Yildiz M, Bircan HA, Akkaya A, Candir O. Absence of pulmonary uptake of Tc-99m methylenediphosphonate in alveolar microlithiasis. Ann Nucl Med 2004;18(8):695-8.
  • 14. Weinstein DS. Pulmonary sarcoidosis: calcified micronodular pattern simulating pulmonary alveolar microlithiasis. J Thorac Imaging 1999;14(3):218-20.
  • 15. Saputo V, Zocchi M, Mancosu M, Bonaldi U, Croce P. Pulmonary alveolar microlithiasis: a case report with a discussion of differential diagnosis. Helv Paediatr Acta 1979;34(3):245- 55.
  • 16. Moran CA, Hochholzer L, Hasleton PS, Johnson FB, Koss MN. Pulmonary alveolar microlithiasis. A clinicopathologic and chemical analysis of seven cases. Arch Pathol Lab Med 1997;121:607–11.
  • 17. Pant K, Shah A, Mathur RK, Chhabra SK, Jain SK. Pulmonary alveolar microlithiasis with pleural calcification and nephrolithiasis.Chest 1990; 98(1):245-6.
  • 18. Coetzee T. Pulmonary alveolar microlithiasis with involvement of the sympathetic nervous system and gonads. Thorax 1970; 25(5):637- 42.
  • 19. Arslan A, Yalin T, Akan H, Belet U. Pulmonary alveolar microlithiasis associated with calcifications in the seminal vesicles. JBelge Radiol 1996;79(3):118-9
  • 20. Gunay E, Ozcan A, Gu¨nay S, Tatci E, Keyf AI, Simsek C. Pulmonary alveolar microlithiasis with low fluorodeoxyglucose accumulation in PET/computed tomography. Ann Thorac Med 2011;6(4):237-40.
  • 21. Jankovic S, Pavlov N, Ivkosic A, Erceg I, Glavina-Durdov M, Tocilj J, et al. Pulmonary alveolar microlithiasis in childhood: clinical and radiological follow-up. Pediatric Pulmonol 2002; 34:384-7.
  • 22. Göcmen A, Toppare MF, Kiper N, Büyükpamukcu N. Treatment of pulmonary alveolar microlithiasis with a di-phosphonatepreliminary results of a case. Respiration 1992; 59:250-2.
  • 23. Edelman JD, Bavaria J, Kaiser LR, Litzky LA, Palevsky HI, Kotloff RM. Bilateral sequential lung transplantation for pulmonary alveolar microlithiasis. Chest 1997; 112:1140-4.
  • 24. Shigemura N, Bermudez C, Hattler BG, Johnson B, Cre-spo M, Pilewski J, et al. Lung transplantation for pulmo-nary alveolar microlithiasis. J Thorac Cardiovasc Surg 2010; 139:50-2.

PULMONER ALVEOLAR MİKROLİTİYAZİS

Year 2016, Volume: 30 Issue: 1, 53 - 57, 01.05.2016

Abstract

Pulmoner alveoler mikrolitiyazis (PAM) alveollerde kalsiyum birikmesi ile karakterize nadir görülen bir akciğer hastalığıdır. Her iki akciğerde yaygın simetrik mikronodüler radyolojik patern mevcuttur. Bu yazıda 19 yaşında PAM tanısı alan bayan hasta sunulmuştur. Hasta 2 aydır egzersizle nefes darlığı ve öksürük yakınmasıyla başvurdu. Fizik muayene ve solunum sesleri normaldi. Akciğer grafisinde orta ve alt alanlarda bilateral yaygın mikronodüler patern saptandı. Bilgisayarlı toraks tomografisinde parankim penceresinde bilateral alt loblarda daha belirgin olmak üzere yaygın milimetrik kalsifiye nodüller izlenmekteydi. Teknesyum-99m ile yapılan tüm vücut kemik sintigrafisinde her iki hemitoraksta yumuşak doku alanlarında heterojen tarzda diffüz artmış aktivite tutulumu saptandı. Sol akciğer alt lobtan fiberoptik bronkoskopi ile elde edilen tranbronşial biyopsi örneğinde intra-alveoler PAS (+) depozitler gösterildi. Hasta PAM tanısı ile tedavisiz takibe alındı.

References

  • 1. Friedrich N. Corpora amylacea in den Lungen. Arch Pathol Anat 1856; 9:613–8.
  • 2. Harbitz F. Extensive calcifications of the lungs as distinct disease. Arch Intern Med 1918; 21:139–46.
  • 3. Puhr L. Microlithiasis alveolarus pulmonum. Virchows Arch 1933; 290:156-60.
  • 4. Ucan ES, Keyf AI, Aydilek R, Yalcin Z, Sebit S, Kudu M, et al. Pulmonary alveolar microlithiasis: review of Turkish reports. Thorax 1993; 48:171-3.
  • 5. Ricci A, Papale M, De Clementi F, Sposato B, Guidi L, et al. Pulmonary alveolar microlithiasis: report on 576 cases published in the literature. Sarcoidosis Vasc Diffuse Lung Dis 2004; 21(3):173-81
  • 6. Castellana G, Lamorgese V. Pulmonary alveolar microlithiasis: World cases and review of the literature. Respiration 2003; 70:549-55.
  • 7. Deniz O. Pulmonary alveolar microlithiasis. Tuberk Thorax 2005; 53:293-8.
  • 8. Millqvist E, Bende M. Capsaicin cough sensitivity is decreased in smokers. Respir Med 2001; 95(1):19-21.
  • 9. Wang BM, Stern EJ, Schmidt RA, Pierson DJ. Diagnosing pulmonary alveolar proteinosis. A review and an update. Chest 1997; 111:460-6.
  • 10. Deniz O, Ors F, Tozkoparan E, Ozcan A, Gumus S, Boz-lar U, et al. High resolution computed tomographic features of pulmonary alveolar microlithiasis. Eur J Radiol 2005; 55:452-60.
  • 11. Shah TC, Talwar A, Shah RD, Margouleff D. Pulmonary alveolar microlithiasis: radiographic and scintigraphic correlation. Clin Nucl Med 2007;32 3:249–51.
  • 12. Elboğa U, Kalender E, Dikensoy Ö, Demir HD, Çelen Z, Yılmaz M. The importance of the bone scintigraphy in the diagnosis of pulmonary alveolar microlithiasis. Respir Case Rep 2014; 3(2):90-2.
  • 13. Sahin U, Yildiz M, Bircan HA, Akkaya A, Candir O. Absence of pulmonary uptake of Tc-99m methylenediphosphonate in alveolar microlithiasis. Ann Nucl Med 2004;18(8):695-8.
  • 14. Weinstein DS. Pulmonary sarcoidosis: calcified micronodular pattern simulating pulmonary alveolar microlithiasis. J Thorac Imaging 1999;14(3):218-20.
  • 15. Saputo V, Zocchi M, Mancosu M, Bonaldi U, Croce P. Pulmonary alveolar microlithiasis: a case report with a discussion of differential diagnosis. Helv Paediatr Acta 1979;34(3):245- 55.
  • 16. Moran CA, Hochholzer L, Hasleton PS, Johnson FB, Koss MN. Pulmonary alveolar microlithiasis. A clinicopathologic and chemical analysis of seven cases. Arch Pathol Lab Med 1997;121:607–11.
  • 17. Pant K, Shah A, Mathur RK, Chhabra SK, Jain SK. Pulmonary alveolar microlithiasis with pleural calcification and nephrolithiasis.Chest 1990; 98(1):245-6.
  • 18. Coetzee T. Pulmonary alveolar microlithiasis with involvement of the sympathetic nervous system and gonads. Thorax 1970; 25(5):637- 42.
  • 19. Arslan A, Yalin T, Akan H, Belet U. Pulmonary alveolar microlithiasis associated with calcifications in the seminal vesicles. JBelge Radiol 1996;79(3):118-9
  • 20. Gunay E, Ozcan A, Gu¨nay S, Tatci E, Keyf AI, Simsek C. Pulmonary alveolar microlithiasis with low fluorodeoxyglucose accumulation in PET/computed tomography. Ann Thorac Med 2011;6(4):237-40.
  • 21. Jankovic S, Pavlov N, Ivkosic A, Erceg I, Glavina-Durdov M, Tocilj J, et al. Pulmonary alveolar microlithiasis in childhood: clinical and radiological follow-up. Pediatric Pulmonol 2002; 34:384-7.
  • 22. Göcmen A, Toppare MF, Kiper N, Büyükpamukcu N. Treatment of pulmonary alveolar microlithiasis with a di-phosphonatepreliminary results of a case. Respiration 1992; 59:250-2.
  • 23. Edelman JD, Bavaria J, Kaiser LR, Litzky LA, Palevsky HI, Kotloff RM. Bilateral sequential lung transplantation for pulmonary alveolar microlithiasis. Chest 1997; 112:1140-4.
  • 24. Shigemura N, Bermudez C, Hattler BG, Johnson B, Cre-spo M, Pilewski J, et al. Lung transplantation for pulmo-nary alveolar microlithiasis. J Thorac Cardiovasc Surg 2010; 139:50-2.
There are 24 citations in total.

Details

Other ID JA65UU58TN
Journal Section Case Report
Authors

Ayşe Coşkun Beyan This is me

Ali Kadri Çırak This is me

Publication Date May 1, 2016
Published in Issue Year 2016 Volume: 30 Issue: 1

Cite

APA Beyan, A. C., & Çırak, A. K. (2016). PULMONER ALVEOLAR MİKROLİTİYAZİS. İzmir Göğüs Hastanesi Dergisi, 30(1), 53-57.