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Ranula and HIV: An Explanation of their Relationship
1670
LETTERS TO THE EDITOR
provide a secure protective barrier against tampering. This is a simple and efficient technique, and we can recommend it as a potential solution for a difficult problem when patients cannot comply with treatment and are at risk of interfering with their wounds on the scalp, whether these are skin grafts, flaps, or open wounds. W.Y CHAN, MSC, MRCS A.R. PATTERSON, FDSRCS, MRCS York, United Kingdom
Reference 1. Di Benedetto G, Pierangelli M, Scalise A, et al. An improved tie-over dressing technique for skin grafts using a hydrocellular dressing. Plast Reconstr Surg 106:507, 2000 doi:10.1016/j.joms.2007.04.005
RANULA AND HIV: AN EXPLANATION OF THEIR RELATIONSHIP To the Editor:—In the article, “Ranula: Experience with 83 cases in Zimbabwe,” in the January 2007 issue of Journal of Oral and Maxillofacial Surgery, the authors reported on a retrospective review of patients with ranulas from 1981 to 2003.1 On the basis of the high incidence of HIV of these patients, they concluded that “HIV salivary gland disease could be an etiologic factor.” It is the purpose of this Letter to the Editor to hopefully provide a rational explanation dispelling a portion of their conclusion.
The authors noted that from 1981 to 1999 (18 years) there were 45 ranula patients treated with an average of 2.5 patients per year while from 1999 to 2003 (5 years) there were 38 such patients with an average of 7.6 per year. They also noted that within that group, 73.5% were young (⬍10 years of age), whereas Zhao et al2, noted in their study of 580 ranulas that they were most prevalent in the second decade of life. Excluding the plunging ranula, my own experience was that about half the ranulas occurred under the age of 12. During the latter period from 1999 to 2003, HIV testing was performed on all of the 38 and it was found that overall, 88.5% were HIV positive while 95% of the young were HIV positive. The only HIV patient I treated was a 7-year-old boy with a large midline superficial dissecting ranula (Fig 1A). At age 4, he had been diagnosed as positive for the virus and was receiving treatment with AZT. The ranula was successfully managed by marsupialization along with packing (Figs 1B,C). With this 3-fold increase in the incidence of ranula development, it was only natural for the authors to arrive at the question, “Could this high prevalence explain the occurrence of ranula in our population or is it a phenomenon of double pathology?” They reported that there was no history of trauma or surgery related to their development and the size of the swelling was in the range of 2 to 3 centimeters. This clearly describes what is considered the true ranula, having its origin from the body of the sublingual gland and developing as a consequence of obstruction in Bartholin’s duct or more likely, the aberrant ducts of the sublingual gland. Harrison and Garrett,4 in their study of mucocele formation in cats, may provide the answer to the author’s question. They studied the effects of ligating the sublingual duct in cats with
FIGURE 1. A, Large midline ranula in a 7-year-old boy with a positive diagnosis of HIV. The lesion had enlarged over a period of 20 months. B, After unroofing the ranula, the cavity was packed with gauze (arrow). C, Complete healing shown at 6 months follow-up.
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LETTERS TO THE EDITOR the chorda tympani intact. In all of the cases, there was initial extravasation for the first 20 days but subsequently only half persisted as mucoceles (ranulas). They suggested that duct obstruction led to the extravasation of mucus from ruptured acini rather than from duct leakage and the mucus then passed through the imperfect gland capsule into the tissues. In another study on the histologic effects of ductal ligation,5 they were able to explain why those that initially extravasated mucus failed to develop into ranula. In those cases, they noted the occurrence of a severe inflammatory reaction to the foreign body (mucus) with macrophages absorbing the material along with an extensive connective tissue response which apparently sealed the leak, leading to atrophy of the effected acini and the cessation of ranula development. Regarding the apparent greater incidence of ranula with HIV patients in Zimbabwe,1 there are 2 possibilities. The infection by HIV, resulting in lymphocytic malfunction, might decrease the effectiveness of the macrophages, lowering the body’s ability to remove the mucus in the tissue, so that fewer mucoceles would resolve. However, this is questionable, because there were no suggestions in the literature to support such an effect by the retrovirus on macrophagic or fibroblastic activity. A more likely answer is that with the greater incidence of HIV in the population from 1999 to 2003, there was a far greater number of patients seeking care at Zimbabwe’s major medical facility. With this surge of patients, one could expect to find a greater number of patients with ranulas during this period compared with the earlier group. I believe that the increase was phantom rather than real. After all, much greater numbers were reported by Takeda and Yamamoto6 in Japan (24.3 per year) and Zhao et al7 in China (14.3 per year) without any association with HIV. Even if we were to assume that not all of them had their origin from the body of the sublingual gland, the numbers would still be significant. Based upon the study by McArthur et al8 regarding salivary gland disease in HIV/AIDS and primary Sjögren’s syndrome where extensive fibrosis was found in African mucus salivary glands, Chidzonga and Rusakaniko,9 in another article, suggested that “. . . this may result in blockage of the sublingual gland and hence, ranula.” John Harrison stated (personal communication, December 2006) that he was “. . . excited to realize that McArthur had found an increase of inflammatory cells in the glands of Africans with untreated HIV and my guess is that this could be the important factor . . . Thus, the children with HIV have more inflammation in the glands, and inflammatory obstruction could lead to acinar rupture and extravasation in the resistant sublingual glands.” Whether it be fibrous tissue, inflammatory tissue, or both causing obstruction, this could account for the significant increase in the percentage of children with ranula observed by Chinzonga’s group. Their statement that “HIV salivary gland disease could be an etiologic factor” is correct. However, the question, “Could this high prevalence explain the occurrence in our population?” is not justified. I do not believe that the incidence of ranula is greater in the HIV patient but certainly it has some effect regarding the greater incidence of this lesion in the young patient. The authors are to be commended for their observation and report to the profession on the association of HIV and ranula.
Acknowledgment I express my deep appreciation to Dr John Harrison for his help in reviewing this Letter to the Editor.
HAROLD D. BAURMASH, DDS Lake Worth, FL
References 1. Chidzonga MM, Mahomva L: Ranula: Experience with 83 cases in Zimbabwe. J Oral Maxillofac Surg 65:79, 2007 2. Zhao YF, Chen XM, Zhang WF: Clinical review of 580 ranulas. Oral Surg Oral Med Oral Path Oral Radiol Endod 98:281, 1998 3. Baurmash HD: Marsupialization for treatment of oral ranula: A second look at the procedure. J Oral Maxillofac Surg 50:1274, 1992 4. Harrison JD, Garrett JR: Histologic effects of ductal ligation of salivary glands of the cat. J Pathol 118:245, 1975 5. Harrison JD, Garrett JR: Histologic effects of ductal ligation of salivary glands of the cat. J Pathol 118:245, 1975 6. Takeda Y, Yamamoto H: Salivary duct cyst: Its frequency in a certain Japanese population group (Tohoku districts) with special reference to adenomatous proliferation of the epithelial lining. J Oral Sci 43:9, 2001 7. Zhao VF, Jia J, Jia Y: Complications associated with surgical management of ranulas. J Oral Maxillofac Surg 63:51, 2005 8. McArthur CP, Africa CW, Castellani WJ, et al: Salivary gland disease in HIV/AIDS and primary Sjögren’s syndrome: Analysis of collagen I distribution and histopathology in American and African patients. J Oral Pathol Med 32:544, 2003 9. Chidzonga MM, Rusakaniko S: Ranula: Another HIV/AIDS associated oral lesion in Zimbabwe? Oral Dis 10:229, 2004
doi:10.1016/j.joms.2007.02.007
RANULA AND HIV: AN EXPLANATION OF THEIR RELATIONSHIP To the Editor:—Several papers describing the wide spectrum of oral manifestations of human immunodeficiency virus (HIV) and acquired immune deficiency syndrome (AIDS) have been published. There are, however, unique differences in these oral manifestations in the various regions with unusual lesions appearing in HIV-infected individuals in developing countries.1 Ranula, as one of the manifestations of HIV-associated salivary gland disease (HIVSGD), could be another unusual lesion appearing in HIVinfected individuals in developing countries as documented in Zimbabwe.2 It is known that HIV-SGD is most common in children with a high prevalence in the pediatric population in Africa and Latin America.3-6 HIV-SGD also includes lymphoepithelial lesions, cysts, and intraglandular lymph nodes along with diffuse infiltrative lymphocytosis syndrome (DILS).7,8 In a study on HIVpositive black Africans in Cameroon more severe HIV-SGD was noted compared with HIV-positive American patients.8 There was also marked fibrosis in the minor salivary glands of the African patients. In the series of 35 patients with DILS reported by Kazi et al, an African-American predominance was observed.8 Thus this fibrosis coupled with DILS in the African minor salivary glands could also be present in the sublingual gland, resulting in blockage of the ducts with consequent rupture of the acini and subsequent extravasation of the saliva, forming the ranula. DILS has been noted by McArthur et al to be high among West Africans.9 In the same study, they also demonstrated that severe salivary gland atypia (96%) was the feature most strongly associated with HIV and AIDS. This leads one to believe that these changes could lead to blockage of the salivary gland ducts with the attendant disruption of the gland acini followed by extravasation of saliva to form the ranula. To date HIV and AIDS-associated ranula has been reported among Africans.2
LETTERS TO THE EDITOR
provide a secure protective barrier against tampering. This is a simple and efficient technique, and we can recommend it as a potential solution for a difficult problem when patients cannot comply with treatment and are at risk of interfering with their wounds on the scalp, whether these are skin grafts, flaps, or open wounds. W.Y CHAN, MSC, MRCS A.R. PATTERSON, FDSRCS, MRCS York, United Kingdom
Reference 1. Di Benedetto G, Pierangelli M, Scalise A, et al. An improved tie-over dressing technique for skin grafts using a hydrocellular dressing. Plast Reconstr Surg 106:507, 2000 doi:10.1016/j.joms.2007.04.005
RANULA AND HIV: AN EXPLANATION OF THEIR RELATIONSHIP To the Editor:—In the article, “Ranula: Experience with 83 cases in Zimbabwe,” in the January 2007 issue of Journal of Oral and Maxillofacial Surgery, the authors reported on a retrospective review of patients with ranulas from 1981 to 2003.1 On the basis of the high incidence of HIV of these patients, they concluded that “HIV salivary gland disease could be an etiologic factor.” It is the purpose of this Letter to the Editor to hopefully provide a rational explanation dispelling a portion of their conclusion.
The authors noted that from 1981 to 1999 (18 years) there were 45 ranula patients treated with an average of 2.5 patients per year while from 1999 to 2003 (5 years) there were 38 such patients with an average of 7.6 per year. They also noted that within that group, 73.5% were young (⬍10 years of age), whereas Zhao et al2, noted in their study of 580 ranulas that they were most prevalent in the second decade of life. Excluding the plunging ranula, my own experience was that about half the ranulas occurred under the age of 12. During the latter period from 1999 to 2003, HIV testing was performed on all of the 38 and it was found that overall, 88.5% were HIV positive while 95% of the young were HIV positive. The only HIV patient I treated was a 7-year-old boy with a large midline superficial dissecting ranula (Fig 1A). At age 4, he had been diagnosed as positive for the virus and was receiving treatment with AZT. The ranula was successfully managed by marsupialization along with packing (Figs 1B,C). With this 3-fold increase in the incidence of ranula development, it was only natural for the authors to arrive at the question, “Could this high prevalence explain the occurrence of ranula in our population or is it a phenomenon of double pathology?” They reported that there was no history of trauma or surgery related to their development and the size of the swelling was in the range of 2 to 3 centimeters. This clearly describes what is considered the true ranula, having its origin from the body of the sublingual gland and developing as a consequence of obstruction in Bartholin’s duct or more likely, the aberrant ducts of the sublingual gland. Harrison and Garrett,4 in their study of mucocele formation in cats, may provide the answer to the author’s question. They studied the effects of ligating the sublingual duct in cats with
FIGURE 1. A, Large midline ranula in a 7-year-old boy with a positive diagnosis of HIV. The lesion had enlarged over a period of 20 months. B, After unroofing the ranula, the cavity was packed with gauze (arrow). C, Complete healing shown at 6 months follow-up.
1671
LETTERS TO THE EDITOR the chorda tympani intact. In all of the cases, there was initial extravasation for the first 20 days but subsequently only half persisted as mucoceles (ranulas). They suggested that duct obstruction led to the extravasation of mucus from ruptured acini rather than from duct leakage and the mucus then passed through the imperfect gland capsule into the tissues. In another study on the histologic effects of ductal ligation,5 they were able to explain why those that initially extravasated mucus failed to develop into ranula. In those cases, they noted the occurrence of a severe inflammatory reaction to the foreign body (mucus) with macrophages absorbing the material along with an extensive connective tissue response which apparently sealed the leak, leading to atrophy of the effected acini and the cessation of ranula development. Regarding the apparent greater incidence of ranula with HIV patients in Zimbabwe,1 there are 2 possibilities. The infection by HIV, resulting in lymphocytic malfunction, might decrease the effectiveness of the macrophages, lowering the body’s ability to remove the mucus in the tissue, so that fewer mucoceles would resolve. However, this is questionable, because there were no suggestions in the literature to support such an effect by the retrovirus on macrophagic or fibroblastic activity. A more likely answer is that with the greater incidence of HIV in the population from 1999 to 2003, there was a far greater number of patients seeking care at Zimbabwe’s major medical facility. With this surge of patients, one could expect to find a greater number of patients with ranulas during this period compared with the earlier group. I believe that the increase was phantom rather than real. After all, much greater numbers were reported by Takeda and Yamamoto6 in Japan (24.3 per year) and Zhao et al7 in China (14.3 per year) without any association with HIV. Even if we were to assume that not all of them had their origin from the body of the sublingual gland, the numbers would still be significant. Based upon the study by McArthur et al8 regarding salivary gland disease in HIV/AIDS and primary Sjögren’s syndrome where extensive fibrosis was found in African mucus salivary glands, Chidzonga and Rusakaniko,9 in another article, suggested that “. . . this may result in blockage of the sublingual gland and hence, ranula.” John Harrison stated (personal communication, December 2006) that he was “. . . excited to realize that McArthur had found an increase of inflammatory cells in the glands of Africans with untreated HIV and my guess is that this could be the important factor . . . Thus, the children with HIV have more inflammation in the glands, and inflammatory obstruction could lead to acinar rupture and extravasation in the resistant sublingual glands.” Whether it be fibrous tissue, inflammatory tissue, or both causing obstruction, this could account for the significant increase in the percentage of children with ranula observed by Chinzonga’s group. Their statement that “HIV salivary gland disease could be an etiologic factor” is correct. However, the question, “Could this high prevalence explain the occurrence in our population?” is not justified. I do not believe that the incidence of ranula is greater in the HIV patient but certainly it has some effect regarding the greater incidence of this lesion in the young patient. The authors are to be commended for their observation and report to the profession on the association of HIV and ranula.
Acknowledgment I express my deep appreciation to Dr John Harrison for his help in reviewing this Letter to the Editor.
HAROLD D. BAURMASH, DDS Lake Worth, FL
References 1. Chidzonga MM, Mahomva L: Ranula: Experience with 83 cases in Zimbabwe. J Oral Maxillofac Surg 65:79, 2007 2. Zhao YF, Chen XM, Zhang WF: Clinical review of 580 ranulas. Oral Surg Oral Med Oral Path Oral Radiol Endod 98:281, 1998 3. Baurmash HD: Marsupialization for treatment of oral ranula: A second look at the procedure. J Oral Maxillofac Surg 50:1274, 1992 4. Harrison JD, Garrett JR: Histologic effects of ductal ligation of salivary glands of the cat. J Pathol 118:245, 1975 5. Harrison JD, Garrett JR: Histologic effects of ductal ligation of salivary glands of the cat. J Pathol 118:245, 1975 6. Takeda Y, Yamamoto H: Salivary duct cyst: Its frequency in a certain Japanese population group (Tohoku districts) with special reference to adenomatous proliferation of the epithelial lining. J Oral Sci 43:9, 2001 7. Zhao VF, Jia J, Jia Y: Complications associated with surgical management of ranulas. J Oral Maxillofac Surg 63:51, 2005 8. McArthur CP, Africa CW, Castellani WJ, et al: Salivary gland disease in HIV/AIDS and primary Sjögren’s syndrome: Analysis of collagen I distribution and histopathology in American and African patients. J Oral Pathol Med 32:544, 2003 9. Chidzonga MM, Rusakaniko S: Ranula: Another HIV/AIDS associated oral lesion in Zimbabwe? Oral Dis 10:229, 2004
doi:10.1016/j.joms.2007.02.007
RANULA AND HIV: AN EXPLANATION OF THEIR RELATIONSHIP To the Editor:—Several papers describing the wide spectrum of oral manifestations of human immunodeficiency virus (HIV) and acquired immune deficiency syndrome (AIDS) have been published. There are, however, unique differences in these oral manifestations in the various regions with unusual lesions appearing in HIV-infected individuals in developing countries.1 Ranula, as one of the manifestations of HIV-associated salivary gland disease (HIVSGD), could be another unusual lesion appearing in HIVinfected individuals in developing countries as documented in Zimbabwe.2 It is known that HIV-SGD is most common in children with a high prevalence in the pediatric population in Africa and Latin America.3-6 HIV-SGD also includes lymphoepithelial lesions, cysts, and intraglandular lymph nodes along with diffuse infiltrative lymphocytosis syndrome (DILS).7,8 In a study on HIVpositive black Africans in Cameroon more severe HIV-SGD was noted compared with HIV-positive American patients.8 There was also marked fibrosis in the minor salivary glands of the African patients. In the series of 35 patients with DILS reported by Kazi et al, an African-American predominance was observed.8 Thus this fibrosis coupled with DILS in the African minor salivary glands could also be present in the sublingual gland, resulting in blockage of the ducts with consequent rupture of the acini and subsequent extravasation of the saliva, forming the ranula. DILS has been noted by McArthur et al to be high among West Africans.9 In the same study, they also demonstrated that severe salivary gland atypia (96%) was the feature most strongly associated with HIV and AIDS. This leads one to believe that these changes could lead to blockage of the salivary gland ducts with the attendant disruption of the gland acini followed by extravasation of saliva to form the ranula. To date HIV and AIDS-associated ranula has been reported among Africans.2