272
Alveolar Bone Loss in Two Children With Short-Bowel Syndrome Receiving Total Parenteral Nutrition Kevin B.
Wright, * Gideon Holan,f Paul S. Casamassimo* and Dennis R. Kings
Two preschool children who were receiving total parenteral nutrition (TPN) for short-bowel syndrome (SBS) were noted to have radiographie evidence of alveolar bone loss in their primary dentition. Tooth mobility, gingival recession, and premature tooth loss were clinical findings in these children. Both had a 2-year history of recurrent infections and fluctuating serum electrolytes prior to identification of their dental problems. / Periodontol 1991; 62:272-275.
Key Words:
Bone
loss; alveolar process; parenteral nutrition.
Short-bowel syndrome (SBS), also termed short small intestine syndrome and short gut syndrome, has two major etiologies: congenital shortness of the small intestine including intestinal artresias and abdominal wall defects, and acute illness such as necrotizing enterocolitis necessitating massive intestinal resection.1,2 The primary signs and symptoms such as hyponatremia and diarrhea are the result of poor fluid and nutrient balance.1 The severity of symptoms following resection of large segments of the small intestine is related to the amount of absorptive surface remaining and specific location of the resection.2,3 The numerous metabolic consequences of massive small bowel loss are, in part, the result of substrate, vitamin, and mineral malabsorption.2 Patients with prolonged calcium, magnesium, vitamin D, and protein malabsorption may develop osteomalacia, osteoporosis, and even spontaneous bone fractures.3 The dental literature generally supports malnutrition as a passive factor aggravating, but not initiating, periodontal disease.4,5 Low calcium/high phosphorus ratios in animal diets may produce nutritional secondary hyperparathyroidism with subsequent alveolar bone résorption as a result of the body's attempt to achieve an equilibrium.6,7 In the primary dentition, conditions affecting calcium/phosphorus balance such as renal disease and hypophosphatasia have been associated with changes in alveolar bone.8,9 The literature on hypophosphatasia describes premature loss of primary teeth10 in association with elevated serum levels of phosphoethànolamine and decreased alkaline Phosphatase. Another condition, familial hypophospha*Private practice, pediatrie dentistry, Fort Wayne, IN. tFaculty of Dental Medicine, Hadassah University, Jerusalem, Israel. ^College of Dentistry, The Ohio State University and Columbus Chil-
Hospital, Columbus, OH. §Columbus Children's Hospital, Department of Pediatrie Surgery, Columbus, OH. dren's
temic vitamin D-resistant rickets, has also been associated with alveolar osseous abnormalities11 of the primary dentition. Both of these case reports,10,11 totaling 5 children, described normal serum calcium levels. The advent of total parenteral nutrition (TPN) has allowed for nutritional support of SBS patients to maintain proper levels of fluids, protein, fat, electrolytes, carbohydrates, trace elements, and vitamins. During TPN intravenous or gastric tube feeding, chewing is absent, as is the functional stimulation essential to maintain a healthy periodontal ligament (PDL) and preserve the structure of the alveolar bone.4,12 Its absence has been suggested to result in narrowing, loss of cellularity, and atrophy of the PDL,12 and subsequently, osteoporosis of the alveolar bone, reduction of bone height, and loose teeth. This paper reports two cases of clinically detectable alveolar bone loss in the primary dentition in patients with SBS.
CASE REPORT 1 B.B. is a 2-1/2 year-old well-developed, well-nourished white female. The dental department at Columbus Children's Hospital was consulted to evaluate the patient's poor oral hygiene. The patient's past medical history consisted of megacystitis, microcolon, and pancreatitis. The patient had a gastric tube and a Broviac catheter in place. She had had numerous admissions to Columbus Children's Hospital due to complications of SBS. Most of these admissions had been for pancreatitis and Broviac catheter sepsis. B.B. was on a total TPN diet with feedings through her broviac catheter every 8 hours. Her medications at the time consisted of Isupride, Nystatin, Bactrim, and Tylenol. Her electrolytes were within normal limits, but she had an extremely high serum lipase and amylase. Serial semm calcium levels over a prior 2-year span were normal, while serum phosphorus and alkaline Phosphatase
Volume 62 Number 4
WRIGHT, HOLAN, CASAMASSIMO, KING
1A. Maxillary primary dentition in a 2-112-year-old white female with evidence of gingival recession and alveolar bone loss.
Figure
Figure IB. Mandibular primary less pronounced findings.
dentition
of same child with similar but
fluctuated between low and normal levels during the same period. White cell counts were within normal limits and neutrophil function tests were not performed. The oral examination revealed a complete primary dentition with fair oral hygiene and no dental caries. Mild marginal gingivitis was noted throughout the entire dentition. Significant gingival recession was noted in the maxillary and mandibular anterior regions. Approximately 3 to 4 mm of exposed root surface was present on the primary maxillary central incisors and approximately 2 to 3 mm of root surface exposed on the primary maxillary lateral incisors (Figs. 1A and IB). These teeth were asymptomatic and without abnormal mobility. A bilateral posterior crossbite was present with mild mandibular anterior crowding. Radiographic examination revealed a decreased bone level in both maxillary and mandibular anterior areas (Figs. 2A and 2B). CASE REPORT 2 J.M. was a 2-year-old well-nourished, male when the Columbus Children's
well-developed black Hospital Dental De-
273
Figure 2A. Maxillary primary dentition of same child depicted in IA with obvious alveolar bone loss around anterior teeth.
Figure 2B. Mandibular primary dentition
with bone loss
(same child).
consulted to evaluate his loose teeth. A review of his medical history revealed SBS secondary to necrotizing enterocolitis. He also had a history of hydrocephalus from superior vena cava syndrome, chronic hyponatremia, gastrostomy with gastric tube, febrile seizures, chronic iron deficiency anemia, and frequent hospital admissions for Broviac catheter sepsis with a variety of bacterial and fungal agents. A review of J.M.'s laboratory tests over multiple admissions spanning the 2 previous years showed consistently low serum sodium and phosphorus, consistently normal serum calcium, and serum alkaline Phosphatase fluctuating between normal and elevated levels. The patient's entire nutrition came from TPN feeding through his Broviac catheter. White cell counts were within normal limits, but neutrophil function tests were not performed. J.M.'s oral exam revealed a complete primary dentition with no visible dental caries. His dentition was generally decalcified with scattered calculus deposits and mild marginal gingivitis with some bleeding of these tissues upon stimulation. Primary maxillary central incisors exhibited + 2 mobility. A maxillary occlusal radiograph showed moderate
partment
was
274
J Periodontol April 1991
ALVEOLAR BONE LOSS IN SHORT BOWEL SYNDROME
Figure 3: Radiograph of maxillary dentition of a 2-year-old indicating missing maxillary left primary central incisor.
black male
Figure 4B: Maxillary occlusa! radiograph (same child) showing missing maxillary left central incisor and root abnormality of maxillary right cen4A: Maxillary dentition indicating missing central incisor (same child as in Figure 3).
Figure
maxillary left primary
tral incisor.
calcium as the only consistent measure. The other interesting characteristic common to both children is recurrent infection. Both had recurrent catheter sepsis episodes and the young boy was plagued with recurrent otitis media. Page et al.,14 in a case series of 5 children with Periodontitis affecting primary teeth, found 3 of the 5 with a significant history of infection in addition to their periodontal disease. All 3 had white cell abnormalities. Unfortunately, at the a serum
bone loss in the maxillary anterior region (Fig. 3). During his hospital course, J.M. lost his left primary maxillary central incisor (Figs. 4A and 4B). The nursing staff reported that his tooth "just fell out while eating."
DISCUSSION Alveolar bone loss in the primary dentition is an uncommon occurrence. It is often accompanied by some form of systemic illness such as leukemia, hypophosphatasia, neutropenia, or histiocytosis X.13 Watanabe's excellent review of the literature13 describes in some detail reports of prepubertal Periodontitis in conjunction with systemic conditions and as isolated events. The dental literature does not address the possible connection between SBS and alveolar bone loss. Since malabsorption of minerals and vitamins can lead to bone disorders,3 SBS and its related electrolyte imbalances may contribute to the alveolar bone loss observed in these two cases. Both children exhibited long-term lability of electrolytes prior to the identification of alveolar bone loss, with
time of this report, neither of the 2 children had received neutrophil function tests. The mechanism of alveolar bone loss may be multifactorial, with lack of mastication and poor oral hygiene as minor contributing factors in addition to the suspected calcium/phosphorus imbalances. Local etiologies cannot be ruled out either, with both toothbrush abrasion and trauma as possible culprits. Both children had root résorption evident upon radiographie examination, which might be caused by a traumatic injury, although neither child had a reported history of injury and oral hygiene was intermittent at best. The lack of mastication may also be a factor, although mastication is only a small part of periodontal stimulation.
Volume 62 Number 4
A
complicating
factor in arriving at an etiology for the oral clinical observation is the complex medical profile of these children. Both had an average of 10 hospital admissions over a 2-year period prior to identification of alveolar bone destruction. Both children provide a variable electrolyte profile which at some admissions changed daily, based on therapeutic intervention. It remains a possibility that the periodontal disease seen in these 2 children is coincidental to their systemic disease. Further systematic study is needed, including testing of white cell function, to identify a possible etiology of the phenomenon observed.
Acknowledgment The authors wish to thank Margaret Ginn-Pease for her help in preparation of this report. REFERENCES 1. Behrman RE, Vaughan VC, Nelson WE. Textbook of Pediatrics. 13th ed. Philadelphia: W.B. Saunders Company; 1987:803-804. 2. Schwartz MZ, Maeda K. Short bowel syndrome in infants and children. Ped Clin Amer 1985; 32:1625-1679. 3. Sleisenger , Fordtran JS. Gastrointestinal Disease. Philadelphia: W.B. Saunders Company; 1989:1106-1112. 4. Nizel AE. Nutrition in preventive dentistry. In: Science and Practice. Philadelphia: WB Saunders Company; 1972:403^107. 5. Stewart RE, Barber TK, Troutman KC, Wei SHY. Pediatrìe Den-
WRIGHT, HOLAN, CASAMASSIMO,
KING
275
tistry. Scientific Foundation and Clinical Practice. St. Louis: The CV Mosby Company; 1982:633-634. 6. Ferguson HW, Hartles RL. The effect of diets deficient of calcium
phosphorus in the presence and absence of supplements of vitamin D on the secondary cementum and alveolar bone of young rats. Arch Oral Biol 1964; 9:647. Henrikson PA. Periodontal disease and calcium deficiency. Acta Odontol Scand 1968; 26(Suppl.):50. Jedrychowski JR, Duperon D. Childhood hypophosphatasia with oral manifestations. J Oral Med 1979; 35:18. Eastwood JB, Bordier PHJ, DeWander HE. Some biochemical histological, radiological and clinical features of renal osteodystrophy. Kidney Int 1973; 4:128. Cheung WS. A mild form of hypophosphatasia as a cause of premature exfoliation of primary teeth: Report of two cases. PediatrDent 1987; 9:49. Ozkan S, Ucok Z, Alagol F. Dental manifestations of familial hypophosphatemic vitamin D-resistant Rickets: Report of case. / Dent Child 1984; 51:448. Grant DA, Stern IB, Listgarten MA. Periodontics. St. Louis: The CV Mosby Company; 1988:71. Watanabe K. Prepubertal Periodontitis: A review of diagnostic criteria, pathogenesis, and differential diagnosis. J Periodont Res 1990; 25:31. Page RC, Bowen T, Altaian L, et al. Prepubertal Periodontitis I. Definition of a clinical disease entity. J Periodontal 1983; 54:257. or
7.
8. 9.
10.
11.
12. 13.
14.
Send reprint requests to: Dr. Paul S. Casamassimo, Professor and Chief of Dentistry, Columbus Children's Hospital, 700 Children's Dr., Columbus, OH 43205. Accepted for publication October 11, 1990.
Alveolar Bone Loss in Two Children With Short-Bowel Syndrome Receiving Total Parenteral Nutrition Kevin B.
Wright, * Gideon Holan,f Paul S. Casamassimo* and Dennis R. Kings
Two preschool children who were receiving total parenteral nutrition (TPN) for short-bowel syndrome (SBS) were noted to have radiographie evidence of alveolar bone loss in their primary dentition. Tooth mobility, gingival recession, and premature tooth loss were clinical findings in these children. Both had a 2-year history of recurrent infections and fluctuating serum electrolytes prior to identification of their dental problems. / Periodontol 1991; 62:272-275.
Key Words:
Bone
loss; alveolar process; parenteral nutrition.
Short-bowel syndrome (SBS), also termed short small intestine syndrome and short gut syndrome, has two major etiologies: congenital shortness of the small intestine including intestinal artresias and abdominal wall defects, and acute illness such as necrotizing enterocolitis necessitating massive intestinal resection.1,2 The primary signs and symptoms such as hyponatremia and diarrhea are the result of poor fluid and nutrient balance.1 The severity of symptoms following resection of large segments of the small intestine is related to the amount of absorptive surface remaining and specific location of the resection.2,3 The numerous metabolic consequences of massive small bowel loss are, in part, the result of substrate, vitamin, and mineral malabsorption.2 Patients with prolonged calcium, magnesium, vitamin D, and protein malabsorption may develop osteomalacia, osteoporosis, and even spontaneous bone fractures.3 The dental literature generally supports malnutrition as a passive factor aggravating, but not initiating, periodontal disease.4,5 Low calcium/high phosphorus ratios in animal diets may produce nutritional secondary hyperparathyroidism with subsequent alveolar bone résorption as a result of the body's attempt to achieve an equilibrium.6,7 In the primary dentition, conditions affecting calcium/phosphorus balance such as renal disease and hypophosphatasia have been associated with changes in alveolar bone.8,9 The literature on hypophosphatasia describes premature loss of primary teeth10 in association with elevated serum levels of phosphoethànolamine and decreased alkaline Phosphatase. Another condition, familial hypophospha*Private practice, pediatrie dentistry, Fort Wayne, IN. tFaculty of Dental Medicine, Hadassah University, Jerusalem, Israel. ^College of Dentistry, The Ohio State University and Columbus Chil-
Hospital, Columbus, OH. §Columbus Children's Hospital, Department of Pediatrie Surgery, Columbus, OH. dren's
temic vitamin D-resistant rickets, has also been associated with alveolar osseous abnormalities11 of the primary dentition. Both of these case reports,10,11 totaling 5 children, described normal serum calcium levels. The advent of total parenteral nutrition (TPN) has allowed for nutritional support of SBS patients to maintain proper levels of fluids, protein, fat, electrolytes, carbohydrates, trace elements, and vitamins. During TPN intravenous or gastric tube feeding, chewing is absent, as is the functional stimulation essential to maintain a healthy periodontal ligament (PDL) and preserve the structure of the alveolar bone.4,12 Its absence has been suggested to result in narrowing, loss of cellularity, and atrophy of the PDL,12 and subsequently, osteoporosis of the alveolar bone, reduction of bone height, and loose teeth. This paper reports two cases of clinically detectable alveolar bone loss in the primary dentition in patients with SBS.
CASE REPORT 1 B.B. is a 2-1/2 year-old well-developed, well-nourished white female. The dental department at Columbus Children's Hospital was consulted to evaluate the patient's poor oral hygiene. The patient's past medical history consisted of megacystitis, microcolon, and pancreatitis. The patient had a gastric tube and a Broviac catheter in place. She had had numerous admissions to Columbus Children's Hospital due to complications of SBS. Most of these admissions had been for pancreatitis and Broviac catheter sepsis. B.B. was on a total TPN diet with feedings through her broviac catheter every 8 hours. Her medications at the time consisted of Isupride, Nystatin, Bactrim, and Tylenol. Her electrolytes were within normal limits, but she had an extremely high serum lipase and amylase. Serial semm calcium levels over a prior 2-year span were normal, while serum phosphorus and alkaline Phosphatase
Volume 62 Number 4
WRIGHT, HOLAN, CASAMASSIMO, KING
1A. Maxillary primary dentition in a 2-112-year-old white female with evidence of gingival recession and alveolar bone loss.
Figure
Figure IB. Mandibular primary less pronounced findings.
dentition
of same child with similar but
fluctuated between low and normal levels during the same period. White cell counts were within normal limits and neutrophil function tests were not performed. The oral examination revealed a complete primary dentition with fair oral hygiene and no dental caries. Mild marginal gingivitis was noted throughout the entire dentition. Significant gingival recession was noted in the maxillary and mandibular anterior regions. Approximately 3 to 4 mm of exposed root surface was present on the primary maxillary central incisors and approximately 2 to 3 mm of root surface exposed on the primary maxillary lateral incisors (Figs. 1A and IB). These teeth were asymptomatic and without abnormal mobility. A bilateral posterior crossbite was present with mild mandibular anterior crowding. Radiographic examination revealed a decreased bone level in both maxillary and mandibular anterior areas (Figs. 2A and 2B). CASE REPORT 2 J.M. was a 2-year-old well-nourished, male when the Columbus Children's
well-developed black Hospital Dental De-
273
Figure 2A. Maxillary primary dentition of same child depicted in IA with obvious alveolar bone loss around anterior teeth.
Figure 2B. Mandibular primary dentition
with bone loss
(same child).
consulted to evaluate his loose teeth. A review of his medical history revealed SBS secondary to necrotizing enterocolitis. He also had a history of hydrocephalus from superior vena cava syndrome, chronic hyponatremia, gastrostomy with gastric tube, febrile seizures, chronic iron deficiency anemia, and frequent hospital admissions for Broviac catheter sepsis with a variety of bacterial and fungal agents. A review of J.M.'s laboratory tests over multiple admissions spanning the 2 previous years showed consistently low serum sodium and phosphorus, consistently normal serum calcium, and serum alkaline Phosphatase fluctuating between normal and elevated levels. The patient's entire nutrition came from TPN feeding through his Broviac catheter. White cell counts were within normal limits, but neutrophil function tests were not performed. J.M.'s oral exam revealed a complete primary dentition with no visible dental caries. His dentition was generally decalcified with scattered calculus deposits and mild marginal gingivitis with some bleeding of these tissues upon stimulation. Primary maxillary central incisors exhibited + 2 mobility. A maxillary occlusal radiograph showed moderate
partment
was
274
J Periodontol April 1991
ALVEOLAR BONE LOSS IN SHORT BOWEL SYNDROME
Figure 3: Radiograph of maxillary dentition of a 2-year-old indicating missing maxillary left primary central incisor.
black male
Figure 4B: Maxillary occlusa! radiograph (same child) showing missing maxillary left central incisor and root abnormality of maxillary right cen4A: Maxillary dentition indicating missing central incisor (same child as in Figure 3).
Figure
maxillary left primary
tral incisor.
calcium as the only consistent measure. The other interesting characteristic common to both children is recurrent infection. Both had recurrent catheter sepsis episodes and the young boy was plagued with recurrent otitis media. Page et al.,14 in a case series of 5 children with Periodontitis affecting primary teeth, found 3 of the 5 with a significant history of infection in addition to their periodontal disease. All 3 had white cell abnormalities. Unfortunately, at the a serum
bone loss in the maxillary anterior region (Fig. 3). During his hospital course, J.M. lost his left primary maxillary central incisor (Figs. 4A and 4B). The nursing staff reported that his tooth "just fell out while eating."
DISCUSSION Alveolar bone loss in the primary dentition is an uncommon occurrence. It is often accompanied by some form of systemic illness such as leukemia, hypophosphatasia, neutropenia, or histiocytosis X.13 Watanabe's excellent review of the literature13 describes in some detail reports of prepubertal Periodontitis in conjunction with systemic conditions and as isolated events. The dental literature does not address the possible connection between SBS and alveolar bone loss. Since malabsorption of minerals and vitamins can lead to bone disorders,3 SBS and its related electrolyte imbalances may contribute to the alveolar bone loss observed in these two cases. Both children exhibited long-term lability of electrolytes prior to the identification of alveolar bone loss, with
time of this report, neither of the 2 children had received neutrophil function tests. The mechanism of alveolar bone loss may be multifactorial, with lack of mastication and poor oral hygiene as minor contributing factors in addition to the suspected calcium/phosphorus imbalances. Local etiologies cannot be ruled out either, with both toothbrush abrasion and trauma as possible culprits. Both children had root résorption evident upon radiographie examination, which might be caused by a traumatic injury, although neither child had a reported history of injury and oral hygiene was intermittent at best. The lack of mastication may also be a factor, although mastication is only a small part of periodontal stimulation.
Volume 62 Number 4
A
complicating
factor in arriving at an etiology for the oral clinical observation is the complex medical profile of these children. Both had an average of 10 hospital admissions over a 2-year period prior to identification of alveolar bone destruction. Both children provide a variable electrolyte profile which at some admissions changed daily, based on therapeutic intervention. It remains a possibility that the periodontal disease seen in these 2 children is coincidental to their systemic disease. Further systematic study is needed, including testing of white cell function, to identify a possible etiology of the phenomenon observed.
Acknowledgment The authors wish to thank Margaret Ginn-Pease for her help in preparation of this report. REFERENCES 1. Behrman RE, Vaughan VC, Nelson WE. Textbook of Pediatrics. 13th ed. Philadelphia: W.B. Saunders Company; 1987:803-804. 2. Schwartz MZ, Maeda K. Short bowel syndrome in infants and children. Ped Clin Amer 1985; 32:1625-1679. 3. Sleisenger , Fordtran JS. Gastrointestinal Disease. Philadelphia: W.B. Saunders Company; 1989:1106-1112. 4. Nizel AE. Nutrition in preventive dentistry. In: Science and Practice. Philadelphia: WB Saunders Company; 1972:403^107. 5. Stewart RE, Barber TK, Troutman KC, Wei SHY. Pediatrìe Den-
WRIGHT, HOLAN, CASAMASSIMO,
KING
275
tistry. Scientific Foundation and Clinical Practice. St. Louis: The CV Mosby Company; 1982:633-634. 6. Ferguson HW, Hartles RL. The effect of diets deficient of calcium
phosphorus in the presence and absence of supplements of vitamin D on the secondary cementum and alveolar bone of young rats. Arch Oral Biol 1964; 9:647. Henrikson PA. Periodontal disease and calcium deficiency. Acta Odontol Scand 1968; 26(Suppl.):50. Jedrychowski JR, Duperon D. Childhood hypophosphatasia with oral manifestations. J Oral Med 1979; 35:18. Eastwood JB, Bordier PHJ, DeWander HE. Some biochemical histological, radiological and clinical features of renal osteodystrophy. Kidney Int 1973; 4:128. Cheung WS. A mild form of hypophosphatasia as a cause of premature exfoliation of primary teeth: Report of two cases. PediatrDent 1987; 9:49. Ozkan S, Ucok Z, Alagol F. Dental manifestations of familial hypophosphatemic vitamin D-resistant Rickets: Report of case. / Dent Child 1984; 51:448. Grant DA, Stern IB, Listgarten MA. Periodontics. St. Louis: The CV Mosby Company; 1988:71. Watanabe K. Prepubertal Periodontitis: A review of diagnostic criteria, pathogenesis, and differential diagnosis. J Periodont Res 1990; 25:31. Page RC, Bowen T, Altaian L, et al. Prepubertal Periodontitis I. Definition of a clinical disease entity. J Periodontal 1983; 54:257. or
7.
8. 9.
10.
11.
12. 13.
14.
Send reprint requests to: Dr. Paul S. Casamassimo, Professor and Chief of Dentistry, Columbus Children's Hospital, 700 Children's Dr., Columbus, OH 43205. Accepted for publication October 11, 1990.
