RENAL AUTOTRANSPLANTATION FOR LOIN PAIN-HEMATURIA SYNDROME 1235 5. Burden, R. P., Dathan, J . R., Etherington, M. D., Guyer, P. B. EDITORIAL COMMENT

and MacIver, A. G.: The loin painhaematuria syndrome. Lan- cet, 1: 897, 1979.

6. Blacklock, A. R. E.: Renal denervation with releasing renal cap- sule incision in the loin painhaematuria syndrome. Brit. J. Urol., 6 4 203, 1989.

7. Guyer, P. B.: Radiology of the loin pain-haematuria syndrome. Clin. Rad., 29 561, 1978.

8. Sheil, A. G. R., Ibels, L. S., Thomas, M. A. and Graham, J . C.: Renal autotransplantation for severe loin painhaematuria syndrome. Lancet, 2 1216, 1985.

9. Sheil, A. G. R., Ibels, L. S., Pollock, C., Graham, J . C. and Short, J.: Treatment of loin painhaematuria syndrome by renal au- totransplantation (Letter to the Editor). Lancet, 2: 907, 1987.

10. Chin, J . L.: Loin pain-hematuria syndrome: role for renal auto- transplantation. J . Urol., 147: 987, 1992.

11. Jones, K., Naish, P. F. and Aber, G. M.: Oestrogen-associated disease of the renal microcirculation. Clin. Sci. Mol. Med., 52 33, 1977.

12. Parbtani, A. and Cameron, J . S.: Platelet involvement in loin painhaematuria syndrome (Letter to the Editor). Lancet, 1: 1413, 1979.

13. Bloom, P. B., Viner, E. D., Mazala, M., Janetta, P. J., Stieber, A. C. and Simmons, R. L.: Treatment of loin pain hematuria syndrome by renal autotransplantation. Amer. J. Med., 87: 228, 1989.

14. Karvelas, J . P. and Ramsey, E. W.: Renal autotransplantation in patients with loin pain-hematuria syndrome. Canad. J. Surg., 39 121, 1996.

15. Turini, D., Barbanti, G., Beneforti, P. and Lazzeri, M.: Auto- transplantation for intractable loin pain: report of a case with long-term followup. J . Urol., 153 389, 1995.

16. Spitz, A,, Huffman, J . L. and Mendez, R.: Autotransplantation as an effective therapy for the loin pain-hematuria syndrome: case reports and a review of the literature. J . Urol., 157: 1554, 1997.

17. Harney, J., Rodgers, E., Campbell, E. and Hickey, D. P.: Loin pain-hematuria syndrome: how effective is renal autotrans- plantation in its treatment? Urology, 44 493, 1994.

18. Parnham, A. P., Low, A,, Finch, P., Perlman, D. and Thomas, M. A.: Recurrent graR pain following renal autotransplanta- tion for loin pain haematuria syndrome. Brit. J. Urol., 78: 25, 1996.

19. Hutchison, S. M. W., Doig, A. and Jenkins, A. M.: Recurrence of loin painhaematuria syndrome after renal autotransplanta- tion. Lancet, 2 1501, 1987.

20. Weisberg, L. S., Bloom, P. B., Simmons, R. L. and Viner, E. D.: Loin pain hematuria syndrome (editorial). h e r . J. Nephrol., 1 3 229, 1993.

21. Dimski, D. S., Hebert, L. A., Sedmak, D., Ogrodowski, J . L., Elkhammas, E. A,, Tesi, R. J., Gold, M. and Courville, C. S.: Renal autotransplantation in the loin pain-hematuria syn- drome: a cautionary note. Amer. J. Kidney Dis., 20 180, 1992.

22. Couch, N. P., McBride, R. A,, Dammin, G. J . and Murray, J. E.: Observations on the nature of the enlargement and regenera- tion of the nerves and the function of the canine renal au- tograft. Brit. J . Exp. Path., 42 106, 1961.

23. Norvell, J . E., Weisten, H. A. and Dwyer, J . J.: Degeneration and regeneration of adrenergic nerves in the autotransplanted kid- ney. Transplantation, 7: 218, 1969.

24. Gazdar, A. F. and Dammin, G. J.: Neuronal degeneration and regeneration in human renal transplants. New Engl. J. Med., 283 222, 1970.

25. Lucas, P. A,, Leaker, B. R. and Neild, G. H.: Psychiatric aspects of loin painhaematuria syndrome (Letter to the Editor). Lan- cet, 3 4 0 1038, 1992.

26. Kelly, B.: Psychoiogical aspects of loin-painhaematuria syn- drome (Letter to the Editor). Lancet, 340 1294, 1992.

27. Kelly, B.: Psychiatric issues in the "loin pain and haematuria syndrome". Aust. New Zeal. J. Psychiat., 128 302, 1994.

28. Chin, J. L., Kloth, D. and Stenn, P.: Predictive factors for positive outcome with renal autotransplants in loin pain-hematuria syndrome. J . Urol., part 2, 155 365A, abstract 548, 1995.

29. Chin, J. L.: The loin pain-hematuria syndrome. (Letter to the Editor). Urology, 46 1085, 1995.

In this small descriptive study the authors present long-term followup of patients with the loin pain-hematuria syndrome treated with autotransplantation. With median followup of 78.5 months they report that 70% of the autotransplant procedures were successful in alleviating pain. Many smaller studies have shown a high early success rate with this procedure but the few that have reported longer term results have been disappointing (reference 17 in article). The diagnosis and treatment of the loin pain-hematuria syndrome remain controversial, as is typically for chronic pain syndromes and diagnoses of exclusion. Radiographic and histological features usu- ally are lacking, although the loin pain-hematuria syndrome has been associated with glomerular basement membrane disease' and evidence of complement activation2 (as in 1 nephrectomy specimen). While we assume that autotransplantation works by surgical renal denervation and that symptomatic failures arise from reinnervation, it should be emphasized that we have no evidence of the real mech- anism and other neurovascular pathways may be important in the pathophysiology.

As with any major surgical intervention for pain control, careful patient screening is essential to rule out other physical or psycho- logical pathological conditions. Of the 64 patients referred 40 were deemed unsuitable for the operation, the majority because they did not have severe enough symptoms to warrant surgery or the psychi- atric examination revealed an underlying psychopathological condi- tion. It would be interesting to know the outcome of the patients who were refused the surgery to determine whether natural history was progressive and whether surgery for this mild form of the disease would have had a higher success rate. Unfortunately, since the exclusion criteria have such a large subjective component, it may be difficult for other investigators attempting to duplicate these excel- lent long-term results to know whether they are dealing with the same patient population.

As this surgical series indicates, vascular complications can occur in any form of renal transplantation even in the most experienced hands and, therefore, the small but definite risk of renal failure needs to be explained in patients with bilateral disease.3 While these impressive long-term results should encourage more referrals for autotransplantation, those referrals should be to experienced trans- plant surgeons to minimize the risks involved.

Daniel Shoskes Renal Transplant Service Harbor-UCLA Medical Center Torrance, California

1. Hebert, L. A., Betts, J . A., Sedmak, D. D., Cosio, F. G., Bay, W. H. and Carlton, S.: Loin pain-hematuria syndrome associated with thin glomerular basement membrane disease and hem- orrhage into renal tubules. Kidney Int., 49 168, 1996.

2. Miller, F., Lane, B. P., Kirsch, M., Ilamathi, E., Moore, B. and Finger, M.: Loin pain-hematuria syndrome with a distinctive vascular lesion and alternative pathway complement activa- tion. Arch. Path. Lab. Med., 118 1016, 1994.

3. Talic, R. F., Parr, N. and Hargreave, T. B.: Anephric state after graft nephrectomy in a patient treated with renal autotrans- plantation for bilateral metachronous loin painhematuria syndrome. J . Urol., 152 1194, 1994.

Reply by Authors. We agree that renal denervation and reinner- vation are simply theories based on limited clinical experience and even less experimental evidence. We should point out that reference 17 in the article, which reported disappointing results, was in fact based on fairly short followup. Followup for our series extends to 138 months (mean 78.7). Sheil et al report slightly longer followup with remarkably similar long-term results as in our series.'

The outcome of patients who are refused surgery was not well documented, and it would have been interesting to know the outcome in the natural disease course of these patients. All patients who were refused surgery at the initial consultation were advised that if symp- toms worsened to the point when narcotic requirements increased or disruption of daily activities became problematic, they would be reassessed and reconsidered for renal autotransplantation. Since 1 of us (J. L. C.) has the most clinical experience in our geographic region with this rare entity, it would be reasonable to assume that the symptoms of these patients have either regressed or remained sta- ble, or else they would have sought a repeat consultation. The ques-

1236 RENAL AUTOTRANSPLANTATION FOR LOIN PAIN-HEMATURIA SYNDROME

tion as to whether surgery for this mild form of the disease would have had a higher success rate is worth considering. However, our main message is that renal autotransplantation is a major surgical undertaking (with inherent vascular complications) and we would not recommend it for patients with a milder form of the disease when less invasive therapeutic options are still available.

We also wish to point out that, although it appears that the exclusion criteria have a significant subjective component, a key message is that the inclusion criteria for surgery as outlined should

be stringent. We agree that this article should serve only to increase the awareness of the condition and to encourage referral to experi- enced surgeons to consider autotransplantation as a viable option in severe cases.

1. Sheil, A. G. R. et a1 Evaluation of the loin paidhematuria syndrome treated by renal autotransplantation or radical re- nal neurectomy. Amer. J. Kidney Dis., 1998. In press.